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However&#44; in a minority of cases&#44; approximately 5&#37; of patients with infantile spasms&#44; the outcomes are favourable&#44; with full resolution of the spasms and normal psychomotor development&#44; which has led some authors to consider it a separate aetiological group labelled <span class="elsevierStyleItalic">idiopathic</span>&#44;<a class="elsevierStyleCrossRef" href="#bib0175"><span class="elsevierStyleSup">5</span></a> although this category has yet to be adopted by the ILAE&#46; This aetiological category is based on disease outcomes&#44; so it can only be applied through the followup of patients&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Since developmental delays present at diagnosis could be explained by both the underlying disease and the epileptic encephalopathy itself &#40;spasms that were not detected previously or presence of hypsarrhythmia prior to onset&#41;&#44; some authors propose abandoning the distinction between symptomatic and cryptogenic WS in favour of a classification based on the development preceding onset&#46; 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such as genetic and metabolic investigations&#46;<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">6&#44;8&#44;9</span></a> They conclude that in children without an aetiological diagnosis after the initial investigation&#44; a cost-efficient strategy would be performance of microarray-based comparative genomic hybridisation &#40;aCGH&#41; followed by an epilepsy gene panel and a general metabolic study &#40;lactate&#44; pyruvate&#44; amino acids and organic acids in urine&#41; in those patients in which the results of aCGH are inconclusive&#46; It is estimated that this approach achieves aetiological diagnosis in an additional 10&#37;&#8211;15&#37; of cases&#46; The study conducted by the National Infantile Spasms Consortium of the United States found that performance of aCGH resulted in an aetiological diagnosis in 11&#37; of the patients that did not previously have one&#44; the epilepsy gene panel in 31&#37; of those patients&#44; and the metabolic study in 4&#46;5&#37;&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Advances in diagnostic techniques and the interest in the underlying cause on account of its prognostic value have led to a decrease in the frequency of cryptogenic cases with an associated increase in symptomatic cases&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Infantile spasms are refractory to most anticonvulsant drugs&#44; and only 2 drugs have clearly proven efficacious in WS&#44; vigabatrin &#40;VGB&#41; and adrenocorticotropic hormone &#40;ACTH&#41;&#46; At present&#44; as the most recent Cochrane review emphasises&#44; the existing evidence is insufficient to determine the superiority of either drug for use as first-line treatment&#46; Some studies have shown that ACTH therapy is more effective in controlling the spasms in the short-term&#44; although its superiority in the long term has yet to be demonstrated&#46;<a class="elsevierStyleCrossRefs" href="#bib0200"><span class="elsevierStyleSup">10&#8211;12</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">There are numerous studies and reviews that focus on WS overall&#44; with particular emphasis on the symptomatic form on account of its higher prevalence&#46; However&#44; few studies have specifically analysed the cryptogenic form&#44; and consequently it is less well understood and its prognosis unclear&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">The aim of this study was to define the profile of cryptogenic WS in our region&#44; exploring the clinical characteristics of the cases&#44; the diagnostic tests performed and the subsequent outcomes&#46; Based on these data&#44; we identified potential prognostic factors for the purpose of improving outcomes and counselling families&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Patients and methods</span><p id="par0055" class="elsevierStylePara elsevierViewall">We conducted a retrospective observational study by reviewing the electronic health records of 16 patients&#46; The statistical analysis was solely descriptive&#44; and we did not apply any inferential methods&#46; The inclusion criteria were&#58; cryptogenic aetiology &#40;understood as normal psychomotor development prior to diagnosis&#41;&#44; no focal signs in the neurologic evaluation at diagnosis&#44; no history of seizures prior to onset of spasms &#40;excluding neonatal and febrile seizures&#41;&#44; normal neuroimaging findings and lack of an attributable aetiology&#59; diagnosis made in the 2000&#8211;2015 period&#59; and treatment and followup in our hospital&#46; The mean duration of followup was 6&#46;6 years&#44; with a minimum of 2 years and a maximum 15 years&#46; There were no losses to followup&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">We created a data collection form that included the following variables&#58; sex&#44; age at onset&#44; history &#40;obstetric&#44; personal and family&#41;&#44; psychomotor development at onset&#44; epileptic seizures prior to onset of spasms&#44; type of spasms&#44; EEG abnormalities&#44; delay in treatment initiation&#44; initial treatment&#44; response to initial treatment&#44; persistence of spasms&#47;hypsarrhythmia&#44; received treatments&#44; adverse events&#44; relapse&#44; psychomotor development during followup&#44; development of other forms of epilepsy&#44; seizure-free time and development of neurologic disease&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">In adherence with the treatment protocol of our hospital&#44; all patients received escalating doses of VGB &#40;up to 200<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41; as first-line treatment&#46; If VGB monotherapy proved ineffective after 2 weeks &#40;persistence of spasms and&#47;or hypsarrhythmia&#41;&#44; we added a daily dose of ACTH for 14 days that was tapered off over an additional 5 weeks&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">We defined unfavourable outcome as the subsequent development of other forms of epilepsy and&#47;or the development of moderate to severe cognitive impairment based on a neuropsychiatric evaluation&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0085">Results</span><p id="par0075" class="elsevierStylePara elsevierViewall">We studied a total of 16 patients&#44; and there were no losses during the followup period&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">When it came to sex&#44; we found a predominance of the male sex&#44; as 11 out of the 16 patients &#40;69&#37;&#41; were male&#46; The age at onset ranged between 1&#46;5 and 10 months&#44; with a predominance of patients in the 5&#8211;7 months group &#40;50&#37;&#41;&#46; The mean age at presentation of spasms was 6 months&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">As for the medical history&#44; 6 of the 16 patients had a family history of epilepsy of some form&#44; all of them benign idiopathic forms in first- or second-degree relatives&#44; and 2 had a personal history of seizures prior to onset of spasms &#40;neonatal seizures in one and febrile seizures in the other&#41;&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">The most frequent type of spasms were extensor spasms &#40;50&#37;&#41;&#44; followed by flexor spasms &#40;43&#37;&#41; and mixed spasms &#40;7&#37;&#41;&#46; Only 4 of the patients had mild developmental delay at onset of spasms&#46;</p><p id="par0095" class="elsevierStylePara elsevierViewall">The time elapsed between onset of spasms and initiation of treatment was less than 1 week in 50&#37; of patients&#46; Per the treatment protocol of the department&#44; all patients received VGB as initial therapy &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0100" class="elsevierStylePara elsevierViewall">The response to VGB was favourable in 5 of the 16 patients&#44; with resolution of the spasms in the first week of treatment and normalisation of EEG features in 2 weeks&#46; Patients that had not improved after 15 days of VGB received ACTH as second-line treatment&#46; All of the 11 patients treated with ACTH had a favourable response&#44; achieving control of hypsarrhythmia&#46;</p><p id="par0105" class="elsevierStylePara elsevierViewall">There were adverse events in 32&#37; of patients&#44; most of them associated with use of ACTH&#46; Of the 16 patients treated with VGB&#44; only 1 suffered an adverse reaction&#44; an acute encephalopathy that improved a few hours after discontinuing the drug&#46; Of the 11 patients treated with ACTH&#44; 4 developed high blood pressure requiring antihypertensive therapy&#59; 2 of them also had echocardiographic abnormalities indicative of septal hypertrophy&#44; although this state was transient and resolved after completion of ACTH therapy&#46;</p><p id="par0110" class="elsevierStylePara elsevierViewall">The outcome was unfavourable in 7 of the 16 patients &#40;43&#46;7&#37;&#41;&#44; all of who developed other forms of epilepsy&#46; Five of them also had moderate or severe cognitive impairment&#44; which was associated with a pervasive developmental disorder in 2&#46; The mean time they remained seizure-free until the development of another form of epilepsy was 10 months&#44; and secondary epilepsy developed within 2 years of diagnosis in all&#46; Other patients exhibited milder impairment in the long term&#58; language disorder&#44; attention-deficit hyperactivity disorder and mild cognitive impairment&#44; and while we have included these data in the analysis of outcomes&#44; we did not include these patients in the unfavourable outcome group because these problems did not have a significant impact on their everyday functioning&#46; None of the patients experienced a relapse &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0115" class="elsevierStylePara elsevierViewall">An aetiological diagnosis was eventually achieved in 2 of the 16 patients during the followup&#46; In one&#44; it was biotinidase deficiency&#44; identified when the metabolic evaluation was expanded in this patient due to the presence of persistent dermatitis&#44; and the other a focal cortical dysplasia identified 10 years after diagnosis of spasms&#59; both patients belonged to the unfavourable outcome group&#46; Therefore&#44; the overall outcome of our cryptogenic patients would improve if we were to exclude these 2 patients &#40;who eventually became part of the symptomatic group&#41; from the results&#44; with unfavourable outcomes in only 5 of a total of 14 patients &#40;35&#46;7&#37;&#41;&#46;</p><p id="par0120" class="elsevierStylePara elsevierViewall">We made a descriptive analysis of several variables&#44; searching for potential prognostic factors with the purpose of developing working hypotheses for future inferential research&#46; We found a greater probability of developing secondary epilepsy in patients of the male sex &#40;55&#37;&#41; or onset before age 6 months &#40;71&#37;&#41;&#44; while early treatment &#40;initiated in the first 2 weeks from onset of spasms&#41; and early control of hypsarrhythmia &#40;in the first 3 weeks&#41; were associated with a decreased probability of developing secondary epilepsy &#40;36&#37; and 33&#37; respectively&#41; &#40;<a class="elsevierStyleCrossRef" href="#tbl0010">Table 2</a>&#41;&#46;</p><elsevierMultimedia ident="tbl0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0090">Discussion</span><p id="par0125" class="elsevierStylePara elsevierViewall">Our study was consistent with the previous literature as regards the age of onset of spasms&#44; the predominance of the male sex and the distribution of the type of spasms&#46;</p><p id="par0130" class="elsevierStylePara elsevierViewall">When we analysed the cryptogenic subgroup in particular&#44; our study did find a higher proportion of patients with a family history of epilepsy&#46; In previous studies on WS&#44; the overall proportion of patients with a family history of epilepsy ranged between 15&#37; and 25&#37;&#44;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">1&#44;3</span></a> while in our series of cryptogenic cases it was as high as 37&#37;&#46; This finding is congruent with the importance of genetic factors in this group of patients&#44; something that has already been noted in previous reviews that found an association between WS of unknown aetiology with other idiopathic forms of epilepsy&#44; such as childhood absence epilepsy&#46;<a class="elsevierStyleCrossRef" href="#bib0175"><span class="elsevierStyleSup">5</span></a></p><p id="par0135" class="elsevierStylePara elsevierViewall">Several studies have found a clear relationship between early initiation of treatment and favourable outcomes&#44; mainly in cryptogenic cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">9&#44;13&#8211;17</span></a> In our study&#44; 50&#37; of patients were referred to our hospital early&#44; and treatment was initiated in the first week since onset of spasms&#46; In 5 patients&#44; treatment was initiated more than 2 weeks after onset&#44; and 3 of them &#40;60&#37;&#41; had unfavourable outcomes with subsequent development of other forms of epilepsy&#44; which also represented a worse outcome relative to the overall outcomes of WS of any type&#46; However&#44; of the 11 patients that started treatment within 2 weeks of onset&#44; only 4 &#40;36&#37;&#41; developed secondary epilepsy&#46;</p><p id="par0140" class="elsevierStylePara elsevierViewall">Since Chiron et al&#46;<a class="elsevierStyleCrossRef" href="#bib0240"><span class="elsevierStyleSup">18</span></a> demonstrated the efficacy of VGB for control of spasms&#44; numerous authors have compared VGB with ACTH and observed a similar response in the long term&#44; although with a lower rate of adverse events&#46;<a class="elsevierStyleCrossRefs" href="#bib0205"><span class="elsevierStyleSup">11&#44;12&#44;19</span></a> As we already mentioned&#44; the treatment protocol of our department&#44; based on the European survey study conducted by Aicardi&#44;<a class="elsevierStyleCrossRefs" href="#bib0250"><span class="elsevierStyleSup">20&#8211;22</span></a> uses VGB monotherapy as first-line treatment and reserves ACTH therapy for patients that did not respond to the initial treatment&#46;</p><p id="par0145" class="elsevierStylePara elsevierViewall">In our study&#44; we defined a favourable response to initial therapy as resolution of spasms and normalisation of the EEG in the first 2 weeks of treatment as long as the improvement persisted for at least one month&#46; Initial therapy with VGB achieved a favourable response in 30&#37; of the patients with cryptogenic WS in our study&#44; a proportion that was lower than the 50&#37;&#8211;70&#37; reported in other case series&#46;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">1&#44;19&#44;23</span></a> Of the patients that responded well to initial treatment with VGB monotherapy&#44; 60&#37; remained free of seizures during the subsequent followup&#44; a percentage that was similar to the percentage we found in the long-term followup of patients treated with VGB and ACTH&#46; Therefore&#44; while the response to initial treatment with VGB was less favourable than expected&#44; long-term outcomes were favourable in the 56&#37; of patients regardless of treatment&#44; which was consistent with the findings of other studies<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">9&#44;10&#44;14</span></a> &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0150" class="elsevierStylePara elsevierViewall">GABAergic retinopathy&#44; which causes irreversible peripheral visual field constriction in adults&#44; continues to be the most important adverse effect of treatment with VGB&#44; although it is difficult to identify in the paediatric age group&#46; Different studies have found a direct association with the dose and duration of treatment&#44; and indicated that treatment can be safe if it lasts less than 6 months and at doses not exceeding 200<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#46;<a class="elsevierStyleCrossRefs" href="#bib0225"><span class="elsevierStyleSup">15&#44;24&#44;25</span></a> Of the 16 patients we followed up&#44; none experienced this complication&#44; even though most of them received treatment for much longer&#44; for a maximum of 3 years&#46; On the other hand&#44; treatment with ACTH produced moderate adverse effects that required treatment in 36&#37; of cases&#46;</p><p id="par0155" class="elsevierStylePara elsevierViewall">Monitoring of retinal toxicity proved challenging due to the young age and the intellectual disability of some of the patients&#44; although their families did not report any visual problems in their everyday lives during the followup visits&#46; All patients underwent periodic checkups in the ophthalmology department with gross assessment of the visual field &#40;menace response&#44; kinetic perimetry and confrontation visual field testing&#41; that revealed no signs of impairment&#46; Automated perimetry was only performed in 2 patients&#44; both without intellectual disability and once they were of prepubertal age&#44; with results within the normal range in both&#44; which leads us to believe that they did not suffer retinal toxicity or else the latter was reversible&#46;</p><p id="par0160" class="elsevierStylePara elsevierViewall">Therefore&#44; contrary to the suggestion of other authors that advocate for first-line treatment with ACTH&#44;<a class="elsevierStyleCrossRef" href="#bib0280"><span class="elsevierStyleSup">26</span></a> our study did not find poorer long-term outcomes in patients treated solely with VGB&#44; and the safety profile of this drug was adequate&#46;</p><p id="par0165" class="elsevierStylePara elsevierViewall">The prognosis of cryptogenic WS continues to be bleak&#46; In our study&#44; 44&#37; percent of patients developed another form of epilepsy &#40;in 71&#37; of cases associated with moderate-to-severe cognitive impairment&#41;&#44; findings that were pretty congruent with those of other authors&#46;<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">9&#44;10&#44;14</span></a> The incidence of Lennox&#8211;Gastaut syndrome was low &#40;1&#47;16&#41;&#44; and focal epilepsy was the most frequent outcome&#46; The patient that developed Lennox&#8211;Gastaut syndrome did not respond to monotherapy with VGB and required the addition of ACTH&#44; in spite of which the time elapsed from initiation of treatment to resolution of hypsarrhythmia was 3 months and a half&#46; Thus&#44; although treatment was initiated in the first week since onset of the spasms&#44; the persistence of hypsarrhythmia in an immature brain may have contributed to the less favourable outcome&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">14&#44;27&#44;28</span></a></p><p id="par0170" class="elsevierStylePara elsevierViewall">Since our study only included patients with a cryptogenic aetiology&#44; it was reasonable to expect better long-term outcomes&#46; However&#44; the proportion of favourable outcomes barely reached 55&#37;&#44; which was probably due to the presence in some cases of an unidentified underlying organic cause that worsened the prognosis independently of the control of hypsarrhythmia&#46; Advances in diagnostic techniques and the increased use of genetic testing will lead to the identification of additional underlying conditions that could account for this epileptic encephalopathy&#44; which a more accurate identification of cryptogenic patients that would be associated with a greater proportion of favourable long-term outcomes&#46;<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">6&#44;29&#44;30</span></a> In our case&#44; due to lack of resources in our hospital during the period under study&#44; genetic testing was limited to karyotype analysis and testing for microdeletion syndromes&#44; without access to gene panels&#46; Nevertheless&#44; metabolic studies and neuroimaging allowed the identification of an underlying cause in 2 of the 16 patients&#44; and excluding these patients from the analysis would increase the proportion of patients with a favourable outcome to 64&#37;&#44; which is closer to our original expectations&#46;</p><p id="par0175" class="elsevierStylePara elsevierViewall">We are aware of the limitations of our study&#44; which is the reason we urge for the performance of additional research on this subject&#46; Some of the methodological aspects that could be improved are the small sample size&#44; the restriction of the analysis to descriptive methods&#44; the limited access to diagnostic techniques that could help discriminate cryptogenic cases more accurately&#44; and the subjective assessment of retinal toxicity&#46;</p><p id="par0180" class="elsevierStylePara elsevierViewall">In conclusion&#44; although our series was limited to cases originally classified as cryptogenic&#44; we found that a considerable percentage had unfavourable outcomes in the long term&#44; which underscores the severity of this disease&#46; An exhaustive aetiological investigation continues to be essential in patients with WS in order to accurately identify patients in the cryptogenic group and be able to make an accurate prognosis&#46; In our study&#44; the factors we identified that may be associated with a favourable prognosis were female sex&#44; onset of spasms after age 6 months&#44; early initiation of treatment and early control of hypsarrhythmia&#44; with the last 2 being particularly relevant&#44; as they are the only factors that are actually modifiable&#46; However&#44; we did not find an association between first-line treatment with VGB monotherapy and a greater probability of developing secondary epilepsy&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0095">Conflicts of interest</span><p id="par0185" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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            0 => "S&#237;ndrome de West"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Introduction</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">West syndrome &#40;WS&#41; is an age-dependent epileptic encephalopathy in which the prognosis varies according to the&#44; not always identified&#44; underlying origin&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Objectives</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">To define the profile of cryptogenic &#40;a least studied isolated sub-group&#41; WS&#44; in Spain&#46; To study its outcome&#44; response to different treatments&#44; and to establish prognostic factors&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Patients and methods</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The study included a review of the medical records of 16 patients diagnosed with cryptogenic WS during the period&#44; 2000&#8211;2015&#46; The mean follow-up time was 6&#46;6 years&#44; with a minimum of 2 years&#46;</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Results</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">The large majority &#40;11&#47;16&#41; were male&#46; The mean age at onset was 6 months&#44; and 6&#47;16 had a family history of idiopathic epilepsy&#46; The first line treatment with vigabatrin had an electrical&#8211;clinical response in 5&#47;16 patients&#44; with the remaining cases responding to adrenocorticotropic hormone &#40;ACTH&#41;&#46; Almost half &#40;44&#37;&#41; of the patients progressed to other types of epilepsy&#44; with no difference between those treated with vigabatrin or ACTH&#46; A greater number of adverse effects were obtained with ACTH&#44; with no retinal involvement being observed with vigabatrin&#46; The aetiological cause was found in 2&#47;16&#46; Being female&#44; late onset&#44; and early control of the hypsarrhythmia&#44; were factors of a good prognosis&#46;</p></span> <span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Conclusions</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">The overall prognosis of cryptogenic WS was more serious than expected&#46; Although the incidence of Lennox&#8211;Gastaut syndrome was low&#44; the progression to focal epilepsy was the most common&#44; with it appearing within the first 2 years of the diagnosis&#46; The initial response to vigabatrin was lower than expected&#44; but the long-term result was comparable to ACTH&#46;</p></span>"
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        "resumen" => "<span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Introducci&#243;n</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">El s&#237;ndrome de West &#40;SW&#41; es una encefalopat&#237;a epil&#233;ptica dependiente de la edad con pron&#243;stico variable seg&#250;n la etiolog&#237;a subyacente&#44; no siempre identificada&#46;</p></span> <span id="abst0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Objetivos</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Definir el perfil del SW criptog&#233;nico en nuestro medio&#44; subgrupo menos estudiado de forma aislada&#46; Estudiar su evoluci&#243;n&#44; respuesta a los distintos tratamientos y establecer factores pron&#243;sticos&#46;</p></span> <span id="abst0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Pacientes y m&#233;todos</span><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Revisi&#243;n de historias cl&#237;nicas de 16 pacientes diagnosticados de SW criptog&#233;nico durante el per&#237;odo 2000-2015&#46; El tiempo de seguimiento medio fue 6&#44;6 a&#241;os y m&#237;nimo de 2 a&#241;os&#46;</p></span> <span id="abst0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Resultados</span><p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">11 de 16 fueron varones&#44; la edad media de inicio fue de 6 meses y 6&#47;16 presentaban antecedente familiar de epilepsia idiop&#225;tica&#46; El tratamiento de primera l&#237;nea con vigabatrina tuvo respuesta electrocl&#237;nica en 5&#47;16 pacientes&#44; respondiendo los casos restantes a hormona adrenocorticotropa &#40;ACTH&#41;&#46; El 44&#37; de los pacientes evolucionaron a otras epilepsias&#44; sin diferencia entre los tratados con vigabatrina o ACTH&#46; Se obtuvo un mayor n&#250;mero de efectos adversos con la ACTH&#44; no se evidenci&#243; afectaci&#243;n retiniana con la vigabatrina&#46; Durante el seguimiento se lleg&#243; a la causa etiol&#243;gica en 2&#47;16&#46; El sexo femenino&#44; el comienzo tard&#237;o y el control precoz de la hipsarritmia resultaron factores de buen pron&#243;stico&#46;</p></span> <span id="abst0050" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conclusiones</span><p id="spar0050" class="elsevierStyleSimplePara elsevierViewall">El pron&#243;stico global del SW criptog&#233;nico result&#243; m&#225;s grave de los esperado&#46; Aunque la incidencia de s&#237;ndrome de Lennox-Gastaut fue baja&#44; la epilepsia focal result&#243; la evoluci&#243;n m&#225;s frecuente apareciendo en los 2 primeros a&#241;os del diagn&#243;stico&#46; La respuesta inicial a vigabatrina fue menor a la esperada&#44; pero el resultado a largo plazo result&#243; superponible a la ACTH&#46;</p></span>"
        "secciones" => array:5 [
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            "identificador" => "abst0030"
            "titulo" => "Introducci&#243;n"
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          1 => array:2 [
            "identificador" => "abst0035"
            "titulo" => "Objetivos"
          ]
          2 => array:2 [
            "identificador" => "abst0040"
            "titulo" => "Pacientes y m&#233;todos"
          ]
          3 => array:2 [
            "identificador" => "abst0045"
            "titulo" => "Resultados"
          ]
          4 => array:2 [
            "identificador" => "abst0050"
            "titulo" => "Conclusiones"
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      ]
    ]
    "NotaPie" => array:2 [
      0 => array:2 [
        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Calder&#243;n Romero M&#44; Arce Portillo E&#44; L&#243;pez Lobato M&#44; Mu&#241;oz Cabello B&#44; Blanco Mart&#237;nez B&#44; Madruga Garrido M&#44; et al&#46; S&#237;ndrome de West criptog&#233;nico&#58; perfil cl&#237;nico&#44; respuesta al tratamiento y factores pron&#243;sticos&#46; An Pediatr &#40;Barc&#41;&#46; 2018&#59;89&#58;176&#8211;182&#46;</p>"
      ]
      1 => array:2 [
        "etiqueta" => "&#9734;&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Previous presentation&#58; Partial results of this study were presented in poster format at the <span class="elsevierStyleSmallCaps">XXXIX</span> Meeting of the Sociedad Espanola de Neurolog&#237;a Pedi&#225;trica&#44; May19&#8211;21&#44; 2016&#59; Toledo&#44; Spain&#46;</p>"
      ]
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          "en" => "<p id="spar0055" class="elsevierStyleSimplePara elsevierViewall">Treatment protocol used in our unit and implemented in the series of patients with cryptogenic West syndrome&#46;</p>"
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          "en" => "<p id="spar0060" class="elsevierStyleSimplePara elsevierViewall">Comparison of the initial long-term response to the 2 treatment regimens&#46; Patients treated with vigabatrin monotherapy and patients treated with a combination of vigabatrin and ACTH&#46;</p>"
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          "leyenda" => "<p id="spar0070" class="elsevierStyleSimplePara elsevierViewall">ADHD&#44; attention-deficit hyperactivity disorder&#59; <span class="elsevierStyleItalic">n</span>&#44; absolute frequency&#46;</p>"
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                  <table border="0" frame="\n
                  \t\t\t\t\tvoid\n
                  \t\t\t\t" class=""><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleBold">Poor outcome</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top"><span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Secondary epilepsy</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">7&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Focal epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Generalised epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Lennox&#8211;Gastaut syndrome&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Refractory epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Moderate&#47;severe cognitive impairment</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Associated with epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Pervasive developmental disorder&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Aetiological diagnosis found</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleBold">Minor impairment</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top"><span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">ADHD</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Language disorder</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Mild cognitive impairment</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
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          "en" => "<p id="spar0065" class="elsevierStyleSimplePara elsevierViewall">Outcomes&#46;</p>"
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          "leyenda" => "<p id="spar0080" class="elsevierStyleSimplePara elsevierViewall">ACTH&#44; adrenocorticotropic hormone&#59; FHx&#44; family history&#59; <span class="elsevierStyleItalic">n</span>&#44; absolute frequency&#59; VGB&#44; vigabatrin&#46;</p>"
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                0 => """
                  <table border="0" frame="\n
                  \t\t\t\t\tvoid\n
                  \t\t\t\t" class=""><thead title="thead"><tr title="table-row"><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Factor&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " colspan="2" align="center" valign="top" scope="col" style="border-bottom: 2px solid black">Secondary epilepsy</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Male sex &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>11&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">55&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">6&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Female sex &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>5&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">20&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&#47;5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Onset before age 6 months &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>7&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">71&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Onset after age 6 months &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>9&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">22&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;9&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">With FHx of epilepsy &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>6&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">33&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;6&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Without FHx of epilepsy &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>10&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">50&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;10&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Treatment with VGB in monotherapy &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>5&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">40&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Treatment with VGB and ACTH &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>11&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">45&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Delay in treatment<span class="elsevierStyleHsp" style=""></span>&#62;<span class="elsevierStyleHsp" style=""></span>2 weeks &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>5&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">60&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">3&#47;5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Delay in treatment<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>2 weeks &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>11&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">36&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">4&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Persistence of hypsarrhythmia<span class="elsevierStyleHsp" style=""></span>&#62;<span class="elsevierStyleHsp" style=""></span>3 weeks &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>4&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">75&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">3&#47;4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Persistence of hypsarrhythmia<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>3 weeks&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">33&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">4&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
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Original Article
Cryptogenic West syndrome: Clinical profile, response to treatment and prognostic factors
Síndrome de West criptogénico: perfil clínico, respuesta al tratamiento y factores pronósticos
María Calderón Romero
Corresponding author
calderonromero@hotmail.com

Corresponding author.
, Elena Arce Portillo, Mercedes López Lobato, Beatriz Muñoz Cabello, Bárbara Blanco Martínez, Marcos Madruga Garrido, Olga Alonso Luego
Servicio de Neuropediatría, Hospitales Universitarios Virgen del Rocío, Sevilla, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">West syndrome &#40;WS&#41; is an age-dependent epileptic encephalopathy characterised by the triad of &#40;1&#41; infantile spasms&#44; usually clustered&#59; &#40;2&#41; evidence of hypsarrhythmia in the electroencephalogram &#40;EEG&#41;&#44; with tracings showing a chaotic&#44; multifocal and bilateral pattern of high-amplitude waves&#59; and &#40;3&#41; regression of psychomotor development&#44; although the latter may be absent&#46;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Its estimated incidence ranges from 2 to 4 in 10<span class="elsevierStyleHsp" style=""></span>000 individuals depending on the series&#44; and therefore it is the most frequent form of epilepsy in the first year of life outside of neonatal and febrile seizures&#46;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">1&#44;3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Due to the numerous underlying causes and the variability in neurologic development in these patients&#44; it is often categorised into different diagnostic groups&#46; The most common classification continues to be the one proposed by the International League Against Epilepsy &#40;ILAE&#41;&#44; which categorises infantile spasms as symptomatic or cryptogenic&#46;<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The symptomatic category corresponds to patients with an obvious underlying cause and&#47;or a history of developmental delay or epileptic seizures prior to the onset of spasms&#44; and accounts for approximately 80&#37; of all patients with WS&#46; Patients in whom an underlying condition is suspected but has not been identified after the aetiological investigation are categorised as having cryptogenic WS and account for the remaining 20&#37; of cases&#46; In the cryptogenic group&#44; patients have normal psychomotor development at onset that goes on to deteriorate progressively in most cases&#46; However&#44; in a minority of cases&#44; approximately 5&#37; of patients with infantile spasms&#44; the outcomes are favourable&#44; with full resolution of the spasms and normal psychomotor development&#44; which has led some authors to consider it a separate aetiological group labelled <span class="elsevierStyleItalic">idiopathic</span>&#44;<a class="elsevierStyleCrossRef" href="#bib0175"><span class="elsevierStyleSup">5</span></a> although this category has yet to be adopted by the ILAE&#46; This aetiological category is based on disease outcomes&#44; so it can only be applied through the followup of patients&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Since developmental delays present at diagnosis could be explained by both the underlying disease and the epileptic encephalopathy itself &#40;spasms that were not detected previously or presence of hypsarrhythmia prior to onset&#41;&#44; some authors propose abandoning the distinction between symptomatic and cryptogenic WS in favour of a classification based on the development preceding onset&#46; In the United Kingdom Infantile Spasms Study &#40;UKISS&#41;&#44; 61&#37; of patients had proven aetiology&#44; 33&#37; had no identified aetiology&#44; and 6&#37; had not been fully investigated&#46;<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">4</span></a> A recent study of the ILAE proposes replacing these terms with 3 new aetiologic groups&#58; genetic&#44; structural-metabolic and unknown&#46;<a class="elsevierStyleCrossRef" href="#bib0180"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">When it comes to the aetiological diagnosis of WS&#44; neuroimaging is the method that offers the highest yield&#46; The underlying cause is identified in 70&#37; of the patients after an adequate history-taking and physical examination and magnetic resonance imaging of the head&#46;<a class="elsevierStyleCrossRef" href="#bib0185"><span class="elsevierStyleSup">7</span></a> Several studies have evaluated the yield of other diagnostic tests&#44; such as genetic and metabolic investigations&#46;<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">6&#44;8&#44;9</span></a> They conclude that in children without an aetiological diagnosis after the initial investigation&#44; a cost-efficient strategy would be performance of microarray-based comparative genomic hybridisation &#40;aCGH&#41; followed by an epilepsy gene panel and a general metabolic study &#40;lactate&#44; pyruvate&#44; amino acids and organic acids in urine&#41; in those patients in which the results of aCGH are inconclusive&#46; It is estimated that this approach achieves aetiological diagnosis in an additional 10&#37;&#8211;15&#37; of cases&#46; The study conducted by the National Infantile Spasms Consortium of the United States found that performance of aCGH resulted in an aetiological diagnosis in 11&#37; of the patients that did not previously have one&#44; the epilepsy gene panel in 31&#37; of those patients&#44; and the metabolic study in 4&#46;5&#37;&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Advances in diagnostic techniques and the interest in the underlying cause on account of its prognostic value have led to a decrease in the frequency of cryptogenic cases with an associated increase in symptomatic cases&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Infantile spasms are refractory to most anticonvulsant drugs&#44; and only 2 drugs have clearly proven efficacious in WS&#44; vigabatrin &#40;VGB&#41; and adrenocorticotropic hormone &#40;ACTH&#41;&#46; At present&#44; as the most recent Cochrane review emphasises&#44; the existing evidence is insufficient to determine the superiority of either drug for use as first-line treatment&#46; Some studies have shown that ACTH therapy is more effective in controlling the spasms in the short-term&#44; although its superiority in the long term has yet to be demonstrated&#46;<a class="elsevierStyleCrossRefs" href="#bib0200"><span class="elsevierStyleSup">10&#8211;12</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">There are numerous studies and reviews that focus on WS overall&#44; with particular emphasis on the symptomatic form on account of its higher prevalence&#46; However&#44; few studies have specifically analysed the cryptogenic form&#44; and consequently it is less well understood and its prognosis unclear&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">The aim of this study was to define the profile of cryptogenic WS in our region&#44; exploring the clinical characteristics of the cases&#44; the diagnostic tests performed and the subsequent outcomes&#46; Based on these data&#44; we identified potential prognostic factors for the purpose of improving outcomes and counselling families&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Patients and methods</span><p id="par0055" class="elsevierStylePara elsevierViewall">We conducted a retrospective observational study by reviewing the electronic health records of 16 patients&#46; The statistical analysis was solely descriptive&#44; and we did not apply any inferential methods&#46; The inclusion criteria were&#58; cryptogenic aetiology &#40;understood as normal psychomotor development prior to diagnosis&#41;&#44; no focal signs in the neurologic evaluation at diagnosis&#44; no history of seizures prior to onset of spasms &#40;excluding neonatal and febrile seizures&#41;&#44; normal neuroimaging findings and lack of an attributable aetiology&#59; diagnosis made in the 2000&#8211;2015 period&#59; and treatment and followup in our hospital&#46; The mean duration of followup was 6&#46;6 years&#44; with a minimum of 2 years and a maximum 15 years&#46; There were no losses to followup&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">We created a data collection form that included the following variables&#58; sex&#44; age at onset&#44; history &#40;obstetric&#44; personal and family&#41;&#44; psychomotor development at onset&#44; epileptic seizures prior to onset of spasms&#44; type of spasms&#44; EEG abnormalities&#44; delay in treatment initiation&#44; initial treatment&#44; response to initial treatment&#44; persistence of spasms&#47;hypsarrhythmia&#44; received treatments&#44; adverse events&#44; relapse&#44; psychomotor development during followup&#44; development of other forms of epilepsy&#44; seizure-free time and development of neurologic disease&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">In adherence with the treatment protocol of our hospital&#44; all patients received escalating doses of VGB &#40;up to 200<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41; as first-line treatment&#46; If VGB monotherapy proved ineffective after 2 weeks &#40;persistence of spasms and&#47;or hypsarrhythmia&#41;&#44; we added a daily dose of ACTH for 14 days that was tapered off over an additional 5 weeks&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">We defined unfavourable outcome as the subsequent development of other forms of epilepsy and&#47;or the development of moderate to severe cognitive impairment based on a neuropsychiatric evaluation&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0085">Results</span><p id="par0075" class="elsevierStylePara elsevierViewall">We studied a total of 16 patients&#44; and there were no losses during the followup period&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">When it came to sex&#44; we found a predominance of the male sex&#44; as 11 out of the 16 patients &#40;69&#37;&#41; were male&#46; The age at onset ranged between 1&#46;5 and 10 months&#44; with a predominance of patients in the 5&#8211;7 months group &#40;50&#37;&#41;&#46; The mean age at presentation of spasms was 6 months&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">As for the medical history&#44; 6 of the 16 patients had a family history of epilepsy of some form&#44; all of them benign idiopathic forms in first- or second-degree relatives&#44; and 2 had a personal history of seizures prior to onset of spasms &#40;neonatal seizures in one and febrile seizures in the other&#41;&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">The most frequent type of spasms were extensor spasms &#40;50&#37;&#41;&#44; followed by flexor spasms &#40;43&#37;&#41; and mixed spasms &#40;7&#37;&#41;&#46; Only 4 of the patients had mild developmental delay at onset of spasms&#46;</p><p id="par0095" class="elsevierStylePara elsevierViewall">The time elapsed between onset of spasms and initiation of treatment was less than 1 week in 50&#37; of patients&#46; Per the treatment protocol of the department&#44; all patients received VGB as initial therapy &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0100" class="elsevierStylePara elsevierViewall">The response to VGB was favourable in 5 of the 16 patients&#44; with resolution of the spasms in the first week of treatment and normalisation of EEG features in 2 weeks&#46; Patients that had not improved after 15 days of VGB received ACTH as second-line treatment&#46; All of the 11 patients treated with ACTH had a favourable response&#44; achieving control of hypsarrhythmia&#46;</p><p id="par0105" class="elsevierStylePara elsevierViewall">There were adverse events in 32&#37; of patients&#44; most of them associated with use of ACTH&#46; Of the 16 patients treated with VGB&#44; only 1 suffered an adverse reaction&#44; an acute encephalopathy that improved a few hours after discontinuing the drug&#46; Of the 11 patients treated with ACTH&#44; 4 developed high blood pressure requiring antihypertensive therapy&#59; 2 of them also had echocardiographic abnormalities indicative of septal hypertrophy&#44; although this state was transient and resolved after completion of ACTH therapy&#46;</p><p id="par0110" class="elsevierStylePara elsevierViewall">The outcome was unfavourable in 7 of the 16 patients &#40;43&#46;7&#37;&#41;&#44; all of who developed other forms of epilepsy&#46; Five of them also had moderate or severe cognitive impairment&#44; which was associated with a pervasive developmental disorder in 2&#46; The mean time they remained seizure-free until the development of another form of epilepsy was 10 months&#44; and secondary epilepsy developed within 2 years of diagnosis in all&#46; Other patients exhibited milder impairment in the long term&#58; language disorder&#44; attention-deficit hyperactivity disorder and mild cognitive impairment&#44; and while we have included these data in the analysis of outcomes&#44; we did not include these patients in the unfavourable outcome group because these problems did not have a significant impact on their everyday functioning&#46; None of the patients experienced a relapse &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0115" class="elsevierStylePara elsevierViewall">An aetiological diagnosis was eventually achieved in 2 of the 16 patients during the followup&#46; In one&#44; it was biotinidase deficiency&#44; identified when the metabolic evaluation was expanded in this patient due to the presence of persistent dermatitis&#44; and the other a focal cortical dysplasia identified 10 years after diagnosis of spasms&#59; both patients belonged to the unfavourable outcome group&#46; Therefore&#44; the overall outcome of our cryptogenic patients would improve if we were to exclude these 2 patients &#40;who eventually became part of the symptomatic group&#41; from the results&#44; with unfavourable outcomes in only 5 of a total of 14 patients &#40;35&#46;7&#37;&#41;&#46;</p><p id="par0120" class="elsevierStylePara elsevierViewall">We made a descriptive analysis of several variables&#44; searching for potential prognostic factors with the purpose of developing working hypotheses for future inferential research&#46; We found a greater probability of developing secondary epilepsy in patients of the male sex &#40;55&#37;&#41; or onset before age 6 months &#40;71&#37;&#41;&#44; while early treatment &#40;initiated in the first 2 weeks from onset of spasms&#41; and early control of hypsarrhythmia &#40;in the first 3 weeks&#41; were associated with a decreased probability of developing secondary epilepsy &#40;36&#37; and 33&#37; respectively&#41; &#40;<a class="elsevierStyleCrossRef" href="#tbl0010">Table 2</a>&#41;&#46;</p><elsevierMultimedia ident="tbl0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0090">Discussion</span><p id="par0125" class="elsevierStylePara elsevierViewall">Our study was consistent with the previous literature as regards the age of onset of spasms&#44; the predominance of the male sex and the distribution of the type of spasms&#46;</p><p id="par0130" class="elsevierStylePara elsevierViewall">When we analysed the cryptogenic subgroup in particular&#44; our study did find a higher proportion of patients with a family history of epilepsy&#46; In previous studies on WS&#44; the overall proportion of patients with a family history of epilepsy ranged between 15&#37; and 25&#37;&#44;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">1&#44;3</span></a> while in our series of cryptogenic cases it was as high as 37&#37;&#46; This finding is congruent with the importance of genetic factors in this group of patients&#44; something that has already been noted in previous reviews that found an association between WS of unknown aetiology with other idiopathic forms of epilepsy&#44; such as childhood absence epilepsy&#46;<a class="elsevierStyleCrossRef" href="#bib0175"><span class="elsevierStyleSup">5</span></a></p><p id="par0135" class="elsevierStylePara elsevierViewall">Several studies have found a clear relationship between early initiation of treatment and favourable outcomes&#44; mainly in cryptogenic cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">9&#44;13&#8211;17</span></a> In our study&#44; 50&#37; of patients were referred to our hospital early&#44; and treatment was initiated in the first week since onset of spasms&#46; In 5 patients&#44; treatment was initiated more than 2 weeks after onset&#44; and 3 of them &#40;60&#37;&#41; had unfavourable outcomes with subsequent development of other forms of epilepsy&#44; which also represented a worse outcome relative to the overall outcomes of WS of any type&#46; However&#44; of the 11 patients that started treatment within 2 weeks of onset&#44; only 4 &#40;36&#37;&#41; developed secondary epilepsy&#46;</p><p id="par0140" class="elsevierStylePara elsevierViewall">Since Chiron et al&#46;<a class="elsevierStyleCrossRef" href="#bib0240"><span class="elsevierStyleSup">18</span></a> demonstrated the efficacy of VGB for control of spasms&#44; numerous authors have compared VGB with ACTH and observed a similar response in the long term&#44; although with a lower rate of adverse events&#46;<a class="elsevierStyleCrossRefs" href="#bib0205"><span class="elsevierStyleSup">11&#44;12&#44;19</span></a> As we already mentioned&#44; the treatment protocol of our department&#44; based on the European survey study conducted by Aicardi&#44;<a class="elsevierStyleCrossRefs" href="#bib0250"><span class="elsevierStyleSup">20&#8211;22</span></a> uses VGB monotherapy as first-line treatment and reserves ACTH therapy for patients that did not respond to the initial treatment&#46;</p><p id="par0145" class="elsevierStylePara elsevierViewall">In our study&#44; we defined a favourable response to initial therapy as resolution of spasms and normalisation of the EEG in the first 2 weeks of treatment as long as the improvement persisted for at least one month&#46; Initial therapy with VGB achieved a favourable response in 30&#37; of the patients with cryptogenic WS in our study&#44; a proportion that was lower than the 50&#37;&#8211;70&#37; reported in other case series&#46;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">1&#44;19&#44;23</span></a> Of the patients that responded well to initial treatment with VGB monotherapy&#44; 60&#37; remained free of seizures during the subsequent followup&#44; a percentage that was similar to the percentage we found in the long-term followup of patients treated with VGB and ACTH&#46; Therefore&#44; while the response to initial treatment with VGB was less favourable than expected&#44; long-term outcomes were favourable in the 56&#37; of patients regardless of treatment&#44; which was consistent with the findings of other studies<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">9&#44;10&#44;14</span></a> &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0150" class="elsevierStylePara elsevierViewall">GABAergic retinopathy&#44; which causes irreversible peripheral visual field constriction in adults&#44; continues to be the most important adverse effect of treatment with VGB&#44; although it is difficult to identify in the paediatric age group&#46; Different studies have found a direct association with the dose and duration of treatment&#44; and indicated that treatment can be safe if it lasts less than 6 months and at doses not exceeding 200<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#46;<a class="elsevierStyleCrossRefs" href="#bib0225"><span class="elsevierStyleSup">15&#44;24&#44;25</span></a> Of the 16 patients we followed up&#44; none experienced this complication&#44; even though most of them received treatment for much longer&#44; for a maximum of 3 years&#46; On the other hand&#44; treatment with ACTH produced moderate adverse effects that required treatment in 36&#37; of cases&#46;</p><p id="par0155" class="elsevierStylePara elsevierViewall">Monitoring of retinal toxicity proved challenging due to the young age and the intellectual disability of some of the patients&#44; although their families did not report any visual problems in their everyday lives during the followup visits&#46; All patients underwent periodic checkups in the ophthalmology department with gross assessment of the visual field &#40;menace response&#44; kinetic perimetry and confrontation visual field testing&#41; that revealed no signs of impairment&#46; Automated perimetry was only performed in 2 patients&#44; both without intellectual disability and once they were of prepubertal age&#44; with results within the normal range in both&#44; which leads us to believe that they did not suffer retinal toxicity or else the latter was reversible&#46;</p><p id="par0160" class="elsevierStylePara elsevierViewall">Therefore&#44; contrary to the suggestion of other authors that advocate for first-line treatment with ACTH&#44;<a class="elsevierStyleCrossRef" href="#bib0280"><span class="elsevierStyleSup">26</span></a> our study did not find poorer long-term outcomes in patients treated solely with VGB&#44; and the safety profile of this drug was adequate&#46;</p><p id="par0165" class="elsevierStylePara elsevierViewall">The prognosis of cryptogenic WS continues to be bleak&#46; In our study&#44; 44&#37; percent of patients developed another form of epilepsy &#40;in 71&#37; of cases associated with moderate-to-severe cognitive impairment&#41;&#44; findings that were pretty congruent with those of other authors&#46;<a class="elsevierStyleCrossRefs" href="#bib0195"><span class="elsevierStyleSup">9&#44;10&#44;14</span></a> The incidence of Lennox&#8211;Gastaut syndrome was low &#40;1&#47;16&#41;&#44; and focal epilepsy was the most frequent outcome&#46; The patient that developed Lennox&#8211;Gastaut syndrome did not respond to monotherapy with VGB and required the addition of ACTH&#44; in spite of which the time elapsed from initiation of treatment to resolution of hypsarrhythmia was 3 months and a half&#46; Thus&#44; although treatment was initiated in the first week since onset of the spasms&#44; the persistence of hypsarrhythmia in an immature brain may have contributed to the less favourable outcome&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">14&#44;27&#44;28</span></a></p><p id="par0170" class="elsevierStylePara elsevierViewall">Since our study only included patients with a cryptogenic aetiology&#44; it was reasonable to expect better long-term outcomes&#46; However&#44; the proportion of favourable outcomes barely reached 55&#37;&#44; which was probably due to the presence in some cases of an unidentified underlying organic cause that worsened the prognosis independently of the control of hypsarrhythmia&#46; Advances in diagnostic techniques and the increased use of genetic testing will lead to the identification of additional underlying conditions that could account for this epileptic encephalopathy&#44; which a more accurate identification of cryptogenic patients that would be associated with a greater proportion of favourable long-term outcomes&#46;<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">6&#44;29&#44;30</span></a> In our case&#44; due to lack of resources in our hospital during the period under study&#44; genetic testing was limited to karyotype analysis and testing for microdeletion syndromes&#44; without access to gene panels&#46; Nevertheless&#44; metabolic studies and neuroimaging allowed the identification of an underlying cause in 2 of the 16 patients&#44; and excluding these patients from the analysis would increase the proportion of patients with a favourable outcome to 64&#37;&#44; which is closer to our original expectations&#46;</p><p id="par0175" class="elsevierStylePara elsevierViewall">We are aware of the limitations of our study&#44; which is the reason we urge for the performance of additional research on this subject&#46; Some of the methodological aspects that could be improved are the small sample size&#44; the restriction of the analysis to descriptive methods&#44; the limited access to diagnostic techniques that could help discriminate cryptogenic cases more accurately&#44; and the subjective assessment of retinal toxicity&#46;</p><p id="par0180" class="elsevierStylePara elsevierViewall">In conclusion&#44; although our series was limited to cases originally classified as cryptogenic&#44; we found that a considerable percentage had unfavourable outcomes in the long term&#44; which underscores the severity of this disease&#46; An exhaustive aetiological investigation continues to be essential in patients with WS in order to accurately identify patients in the cryptogenic group and be able to make an accurate prognosis&#46; In our study&#44; the factors we identified that may be associated with a favourable prognosis were female sex&#44; onset of spasms after age 6 months&#44; early initiation of treatment and early control of hypsarrhythmia&#44; with the last 2 being particularly relevant&#44; as they are the only factors that are actually modifiable&#46; However&#44; we did not find an association between first-line treatment with VGB monotherapy and a greater probability of developing secondary epilepsy&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0095">Conflicts of interest</span><p id="par0185" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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            0 => "West syndrome"
            1 => "Infantile spasms"
            2 => "Cryptogenic"
            3 => "Aetiology"
            4 => "Vigabatrin"
            5 => "Prognostic factors"
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          "palabras" => array:6 [
            0 => "S&#237;ndrome de West"
            1 => "Espasmos infantiles"
            2 => "Criptog&#233;nico"
            3 => "Etiolog&#237;a"
            4 => "Vigabatrina"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Introduction</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">West syndrome &#40;WS&#41; is an age-dependent epileptic encephalopathy in which the prognosis varies according to the&#44; not always identified&#44; underlying origin&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Objectives</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">To define the profile of cryptogenic &#40;a least studied isolated sub-group&#41; WS&#44; in Spain&#46; To study its outcome&#44; response to different treatments&#44; and to establish prognostic factors&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Patients and methods</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The study included a review of the medical records of 16 patients diagnosed with cryptogenic WS during the period&#44; 2000&#8211;2015&#46; The mean follow-up time was 6&#46;6 years&#44; with a minimum of 2 years&#46;</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Results</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">The large majority &#40;11&#47;16&#41; were male&#46; The mean age at onset was 6 months&#44; and 6&#47;16 had a family history of idiopathic epilepsy&#46; The first line treatment with vigabatrin had an electrical&#8211;clinical response in 5&#47;16 patients&#44; with the remaining cases responding to adrenocorticotropic hormone &#40;ACTH&#41;&#46; Almost half &#40;44&#37;&#41; of the patients progressed to other types of epilepsy&#44; with no difference between those treated with vigabatrin or ACTH&#46; A greater number of adverse effects were obtained with ACTH&#44; with no retinal involvement being observed with vigabatrin&#46; The aetiological cause was found in 2&#47;16&#46; Being female&#44; late onset&#44; and early control of the hypsarrhythmia&#44; were factors of a good prognosis&#46;</p></span> <span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Conclusions</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">The overall prognosis of cryptogenic WS was more serious than expected&#46; Although the incidence of Lennox&#8211;Gastaut syndrome was low&#44; the progression to focal epilepsy was the most common&#44; with it appearing within the first 2 years of the diagnosis&#46; The initial response to vigabatrin was lower than expected&#44; but the long-term result was comparable to ACTH&#46;</p></span>"
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          0 => array:2 [
            "identificador" => "abst0005"
            "titulo" => "Introduction"
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            "titulo" => "Objectives"
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          2 => array:2 [
            "identificador" => "abst0015"
            "titulo" => "Patients and methods"
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        "resumen" => "<span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Introducci&#243;n</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">El s&#237;ndrome de West &#40;SW&#41; es una encefalopat&#237;a epil&#233;ptica dependiente de la edad con pron&#243;stico variable seg&#250;n la etiolog&#237;a subyacente&#44; no siempre identificada&#46;</p></span> <span id="abst0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Objetivos</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Definir el perfil del SW criptog&#233;nico en nuestro medio&#44; subgrupo menos estudiado de forma aislada&#46; Estudiar su evoluci&#243;n&#44; respuesta a los distintos tratamientos y establecer factores pron&#243;sticos&#46;</p></span> <span id="abst0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Pacientes y m&#233;todos</span><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Revisi&#243;n de historias cl&#237;nicas de 16 pacientes diagnosticados de SW criptog&#233;nico durante el per&#237;odo 2000-2015&#46; El tiempo de seguimiento medio fue 6&#44;6 a&#241;os y m&#237;nimo de 2 a&#241;os&#46;</p></span> <span id="abst0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Resultados</span><p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">11 de 16 fueron varones&#44; la edad media de inicio fue de 6 meses y 6&#47;16 presentaban antecedente familiar de epilepsia idiop&#225;tica&#46; El tratamiento de primera l&#237;nea con vigabatrina tuvo respuesta electrocl&#237;nica en 5&#47;16 pacientes&#44; respondiendo los casos restantes a hormona adrenocorticotropa &#40;ACTH&#41;&#46; El 44&#37; de los pacientes evolucionaron a otras epilepsias&#44; sin diferencia entre los tratados con vigabatrina o ACTH&#46; Se obtuvo un mayor n&#250;mero de efectos adversos con la ACTH&#44; no se evidenci&#243; afectaci&#243;n retiniana con la vigabatrina&#46; Durante el seguimiento se lleg&#243; a la causa etiol&#243;gica en 2&#47;16&#46; El sexo femenino&#44; el comienzo tard&#237;o y el control precoz de la hipsarritmia resultaron factores de buen pron&#243;stico&#46;</p></span> <span id="abst0050" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conclusiones</span><p id="spar0050" class="elsevierStyleSimplePara elsevierViewall">El pron&#243;stico global del SW criptog&#233;nico result&#243; m&#225;s grave de los esperado&#46; Aunque la incidencia de s&#237;ndrome de Lennox-Gastaut fue baja&#44; la epilepsia focal result&#243; la evoluci&#243;n m&#225;s frecuente apareciendo en los 2 primeros a&#241;os del diagn&#243;stico&#46; La respuesta inicial a vigabatrina fue menor a la esperada&#44; pero el resultado a largo plazo result&#243; superponible a la ACTH&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Calder&#243;n Romero M&#44; Arce Portillo E&#44; L&#243;pez Lobato M&#44; Mu&#241;oz Cabello B&#44; Blanco Mart&#237;nez B&#44; Madruga Garrido M&#44; et al&#46; S&#237;ndrome de West criptog&#233;nico&#58; perfil cl&#237;nico&#44; respuesta al tratamiento y factores pron&#243;sticos&#46; An Pediatr &#40;Barc&#41;&#46; 2018&#59;89&#58;176&#8211;182&#46;</p>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Previous presentation&#58; Partial results of this study were presented in poster format at the <span class="elsevierStyleSmallCaps">XXXIX</span> Meeting of the Sociedad Espanola de Neurolog&#237;a Pedi&#225;trica&#44; May19&#8211;21&#44; 2016&#59; Toledo&#44; Spain&#46;</p>"
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          "en" => "<p id="spar0055" class="elsevierStyleSimplePara elsevierViewall">Treatment protocol used in our unit and implemented in the series of patients with cryptogenic West syndrome&#46;</p>"
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          "en" => "<p id="spar0060" class="elsevierStyleSimplePara elsevierViewall">Comparison of the initial long-term response to the 2 treatment regimens&#46; Patients treated with vigabatrin monotherapy and patients treated with a combination of vigabatrin and ACTH&#46;</p>"
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          "leyenda" => "<p id="spar0070" class="elsevierStyleSimplePara elsevierViewall">ADHD&#44; attention-deficit hyperactivity disorder&#59; <span class="elsevierStyleItalic">n</span>&#44; absolute frequency&#46;</p>"
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                  \t\t\t\t\tvoid\n
                  \t\t\t\t" class=""><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleBold">Poor outcome</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top"><span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Secondary epilepsy</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">7&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Focal epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Generalised epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Lennox&#8211;Gastaut syndrome&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Refractory epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Moderate&#47;severe cognitive impairment</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Associated with epilepsy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleHsp" style=""></span>Pervasive developmental disorder&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Aetiological diagnosis found</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleBold">Minor impairment</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top"><span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">ADHD</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Language disorder</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Mild cognitive impairment</span>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
                  """
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          "en" => "<p id="spar0065" class="elsevierStyleSimplePara elsevierViewall">Outcomes&#46;</p>"
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          "leyenda" => "<p id="spar0080" class="elsevierStyleSimplePara elsevierViewall">ACTH&#44; adrenocorticotropic hormone&#59; FHx&#44; family history&#59; <span class="elsevierStyleItalic">n</span>&#44; absolute frequency&#59; VGB&#44; vigabatrin&#46;</p>"
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                  <table border="0" frame="\n
                  \t\t\t\t\tvoid\n
                  \t\t\t\t" class=""><thead title="thead"><tr title="table-row"><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Factor&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " colspan="2" align="center" valign="top" scope="col" style="border-bottom: 2px solid black">Secondary epilepsy</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Male sex &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>11&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">55&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">6&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Female sex &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>5&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">20&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">1&#47;5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Onset before age 6 months &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>7&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">71&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Onset after age 6 months &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>9&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">22&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;9&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">With FHx of epilepsy &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>6&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">33&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;6&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Without FHx of epilepsy &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>10&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">50&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;10&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Treatment with VGB in monotherapy &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>5&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">40&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">2&#47;5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Treatment with VGB and ACTH &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>11&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">45&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">5&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Delay in treatment<span class="elsevierStyleHsp" style=""></span>&#62;<span class="elsevierStyleHsp" style=""></span>2 weeks &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>5&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">60&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">3&#47;5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Delay in treatment<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>2 weeks &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>11&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">36&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">4&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Persistence of hypsarrhythmia<span class="elsevierStyleHsp" style=""></span>&#62;<span class="elsevierStyleHsp" style=""></span>3 weeks &#40;<span class="elsevierStyleItalic">n</span><span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>4&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">75&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">3&#47;4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Persistence of hypsarrhythmia<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>3 weeks&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">33&#37;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">4&#47;11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
                  """
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        "descripcion" => array:1 [
          "en" => "<p id="spar0075" class="elsevierStyleSimplePara elsevierViewall">Potential prognostic factors&#46;</p>"
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    ]
    "bibliografia" => array:2 [
      "titulo" => "References"
      "seccion" => array:1 [
        0 => array:2 [
          "identificador" => "bibs0015"
          "bibliografiaReferencia" => array:30 [
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              "identificador" => "bib0155"
              "etiqueta" => "1"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "S&#237;ndrome de West&#58; etiolog&#237;a&#44; opciones terap&#233;uticas&#44; evoluci&#243;n cl&#237;nica y factores pron&#243;sticos"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "E&#46; Arce"
                            1 => "M&#46; Rufo"
                            2 => "B&#46; Mu&#241;oz"
                            3 => "B&#46; Blanco"
                            4 => "M&#46; Madruga"
                            5 => "L&#46; Ruiz"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:6 [
                        "tituloSerie" => "Rev Neurol"
                        "fecha" => "2011"
                        "volumen" => "52"
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/21271547"
                            "web" => "Medline"
                          ]
                        ]
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                    ]
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                ]
              ]
            ]
            1 => array:3 [
              "identificador" => "bib0160"
              "etiqueta" => "2"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "West &#38; son&#58; the origins of West syndrome"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:1 [
                            0 => "A&#46;L&#46; Lux"
                          ]
                        ]
                      ]
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                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:7 [
                        "tituloSerie" => "Brain Dev"
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/11701237"
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                          "pii" => "S0140673615001531"
                          "estado" => "S300"
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            2 => array:3 [
              "identificador" => "bib0165"
              "etiqueta" => "3"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Long-term outcomes in patients with West syndrome&#58; an outpatient clinical study"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:6 [
                            0 => "B&#46;T&#46; G&#252; veli"
                            1 => "O&#46; &#199;okar"
                            2 => "N&#46; D&#246;rtcan"
                            3 => "G&#46; Benbir"
                            4 => "V&#46; Demirbilek"
                            5 => "A&#46; Dervent"
                          ]
                        ]
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                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1016/j.seizure.2015.01.001"
                      "Revista" => array:6 [
                        "tituloSerie" => "Seizure"
                        "fecha" => "2015"
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                    ]
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              ]
            ]
            3 => array:3 [
              "identificador" => "bib0170"
              "etiqueta" => "4"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "The underlying etiology of infantile spasms &#40;West syndrome&#41;&#58; information from the United Kingdom Infantile Spasms Study &#40;UKISS&#41; on contemporary causes and their classification"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "J&#46; Osborne"
                            1 => "A&#46; Lux"
                            2 => "S&#46; Edwards"
                            3 => "E&#46; Hancock"
                            4 => "A&#46; Johnson"
                            5 => "C&#46; Kennedy"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1111/j.1528-1167.2010.02695.x"
                      "Revista" => array:6 [
                        "tituloSerie" => "Epilepsia"
                        "fecha" => "2010"
                        "volumen" => "51"
                        "paginaInicial" => "2168"
                        "paginaFinal" => "2174"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20726878"
                            "web" => "Medline"
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            4 => array:3 [
              "identificador" => "bib0175"
              "etiqueta" => "5"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Idiopathic West syndrome followed by childhood absence epilepsy"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "N&#46; Specchio"
                            1 => "M&#46; Trivisano"
                            2 => "F&#46; Vigevano"
                            3 => "L&#46; Fusco"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1016/j.seizure.2010.07.016"
                      "Revista" => array:6 [
                        "tituloSerie" => "Seizure"
                        "fecha" => "2010"
                        "volumen" => "19"
                        "paginaInicial" => "597"
                        "paginaFinal" => "601"
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20729098"
                            "web" => "Medline"
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            5 => array:3 [
              "identificador" => "bib0180"
              "etiqueta" => "6"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
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Idiomas
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