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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Primary lymphoedema is a rare disease&#44; usually with onset in prepubertal girls&#44; and its prevalence ranges between 1&#47;6000 and 1&#47;100<span class="elsevierStyleHsp" style=""></span>000 in the population&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">It is caused by abnormal angiogenesis during embryonic development&#44; usually due to mutations in genes responsible for it&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In up to 80&#37; of cases it manifests in the lower extremities&#44; although it can involve the upper extremities&#44; face and genitals&#46; Focal lymphatic abnormalities lead to an accumulation of lymph that&#44; when sustained&#44; stimulates fibrosis and produces a localised&#44; chronic and progressive oedema&#46; In the early stages&#44; the oedema is not associated with pain&#44; warmth&#44; erythema or functional impairment&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">In 1934&#44; Allen classified primary lymphoedema based on the age of onset into congenital&#44; praecox and tarda&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Recently&#44; Connell et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> proposed a new classification that took into account the different clinical presentations and known mutations&#44; giving rise to a complex diagnostic algorithm that encompassed 26 possible diagnoses&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">We present the cases of six patients with primary lymphoedema diagnosed in our centre&#44; whose clinical and demographic characteristics are summarised in <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#46; Most of the patients were female&#44; and the age of onset ranged between 5 and 12 years &#40;median&#44; 10&#46;5&#41;&#46; None of the patients had associated malformations or a relevant personal history&#46; Fifty percent had a family history of lymphoedema&#46; The time elapsed from the onset of symptoms to diagnosis ranged between 3 months and 4&#46;3 years &#40;median&#44; 9 months&#41;&#46; Patients were referred to a specialist to rule out arthritis as the cause of the swelling of the hand or dorsum of the foot&#46; None of the patients reported arthralgia&#44; morning stiffness or functional impairment&#44; and&#44; as is characteristic of lymphoedema&#44; the swelling improved with night rest and worsened with standing&#46; Physical examination found soft tissue oedema in all patients&#44; with no abnormalities in the joints&#46; The results of the blood panel &#40;blood counts&#44; acute phase reactants&#44; TSH&#44; albumin&#44; total protein&#41; and the routine urinalysis were also normal&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Most of the patients had been assessed previously in other departments of the hospital &#40;traumatology&#44; plastic surgery&#44; dermatology&#44; emergency&#41;&#44; and underwent different imaging tests &#40;conventional ultrasound&#44; Doppler ultrasound&#44; MRI&#41; that revealed the presence of oedema in the subcutaneous tissue&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The gold standard for the diagnosis of lymphoedema is lymphoscintigraphy&#44; which assesses the velocity of the migration of a radioactive tracer through the lymphatic vessels&#46; The tracer is injected in the dermis of an interdigital web space and tracked by a scintillation camera&#46; This is a minimally invasive and reproducible test that can be performed in children&#46; In patients with primary lymphoedema&#44; this method detects absent or delayed tracer migration<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; In a study conducted in 107 patients with primary lymphoedema&#44; Kinmonth et al&#46; presented the structural abnormalities that could be detected by lymphoscintigraphy and classified them into three categories&#58; aplasia &#40;absence of lymphatic vessels&#41;&#44; hypoplasia &#40;small number or size of lymphatic vessels&#41; and hyperplasia &#40;tortuous lymphatic vessels&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> All our patients presented this type of abnormalities in the lymphatic system&#44; and were classified according to the scheme proposed by Connell et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Other imaging techniques that have been used in more recent studies but have not been widely introduced in clinical practice include indocyanine green lymphography&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> which exposes the patient to radiation&#44; and magnetic resonance lymphangiography using gadobenate dimeglumine as the contrast agent&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> which does not involve exposure to radiation and can obtain dynamic sequences&#44; both of which are as efficacious as lymphoscintigraphy in detecting abnormalities in lymph circulation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">The management of primary lymphoedema is mainly conservative&#44; with the first line treatment consisting of manual lymph drainage or compression therapy&#46; Avoidance of prolonged standing&#44; the use compression stockings&#44; and regular physical activity combined with adequate skin care usually suffice in most cases&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> These measures were implemented in all of our patients and succeeded in controlling the lymphoedema&#46; The prognosis of lymphoedema is good&#44; as the disease stabilises in half of the patients &#40;57&#37;&#41;&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> although surgical measures are available for the most severe cases&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Swelling of the extremities in the paediatric age group requires a broad differential diagnosis including traumatic injury&#44; cellulitis&#44; arthritis&#44; venous insufficiency&#44; deep vein thrombosis and primary lymphoedema&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Knowledge of this condition and its characteristics&#8212;painless swelling in the extremities that changes with posture and is not associated with articular pain or limited range of motion&#8212;can prevent diagnostic delays as well as unnecessary referrals and diagnostic tests&#46;</p></span>"
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        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Barral Mena E&#44; Soriano-Ramos M&#44; Pavo Garc&#237;a MR&#44; Llorente Otones L&#44; de Inocencio Arocena J&#46; Linfedema primario fuera del periodo neonatal&#46; An Pediatr &#40;Barc&#41;&#46; 2016&#59;85&#58;47&#8211;49&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Lymphoscintigraphy image &#40;anterior view&#41; showing delayed lymph drainage in the left lower extremity&#44; with persisting radioactive tracer activity at the level of the popliteal lymph nodes &#40;arrow&#41;&#44; compatible with primary lymphoedema&#46;</p>"
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          "leyenda" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">FH&#58; family history&#59; F&#58; female&#59; &#40;R&#41;&#58; right&#59; &#40;L&#41;&#58; left&#59; LE&#58; lower extremity&#59; UE&#58; upper extremity&#59; APRs&#58; acute phase reactants&#59; MRI&#58; magnetic resonance imaging&#59; SCT&#58; subcutaneous tissue&#59; TSH&#58; thyroid-stimulating hormone&#59; M&#58; male&#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="" valign="top" scope="col" style="border-bottom: 2px solid black">&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Sex&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Family&#47;personal history&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Age of onset &#40;years&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Time to diagnosis &#40;months&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Lymphoedema location&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Blood tests &#40;WBC&#44; APRs&#44; TSH&#44; proteins&#41; and urinalysis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Lymphoscintigraphy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Other imaging tests&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Final diagnosis based on Connell et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> classification&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</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">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">No&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Left foot and ankle&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Delayed tracer elimination in &#40;L&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Ultrasound and MRI showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Unilateral late onset primary lymphoedema&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">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">M&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">No&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">52&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Left foot&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Delayed tracer elimination in &#40;L&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal X-ray&#44; MRI showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Unilateral late onset primary lymphoedema&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">3&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">FH of mother with lymphoedema of the LEs&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">12&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">13&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Feet and ankles&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Delayed tracer elimination in &#40;L&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal abdominal ultrasound&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Primary lymphoedema &#40;Meige disease&#41;&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">4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">No&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">16&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Left foot and ankle&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Absent visualisation of lymph drainage in &#40;L&#41;LE due to lymphatic system aplasia&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal X-ray&#44; normal Doppler ultrasound&#44; conventional ultrasound showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Unilateral late onset primary lymphoedema&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">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">FH of aunt and first-degree female cousin on the mother&#39;s side with oedema of the LEs&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">10&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">3&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Right foot and left hand&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Hypoplasia and lymphatic vascular insufficiency in &#40;L&#41;LE and &#40;R&#41;LE lymph node chains&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Ultrasound showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Late onset multisegmental lymphoedema&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">6&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">FH of mother and maternal first-degree female cousin with oedema of the LEs&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">10&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">13&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Right arm and forearm&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Increased diameter of lymphatic vessels in &#40;R&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">MRI showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Late onset multisegmental lymphoedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
                  """
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Demographic&#44; clinical and diagnostic characteristics of patients with a primary lymphoedema diagnosis&#46;</p>"
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Scientific Letter
Primary lymphoedema outside the neonatal period
Linfedema primario fuera del periodo neonatal
Estefanía Barral Menaa,b,
Corresponding author
, María Soriano-Ramosb, Maria Rosa Pavo Garcíaa, Lucía Llorente Otonesc, Jaime de Inocencio Arocenab
a Centro de Salud García Noblejas, Madrid, Spain
b Unidad de Reumatología Pediátrica, Hospital Universitario 12 de Octubre, Madrid, Spain
c Hospital de Fuenlabrada, Fuenlabrada, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Primary lymphoedema is a rare disease&#44; usually with onset in prepubertal girls&#44; and its prevalence ranges between 1&#47;6000 and 1&#47;100<span class="elsevierStyleHsp" style=""></span>000 in the population&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">It is caused by abnormal angiogenesis during embryonic development&#44; usually due to mutations in genes responsible for it&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In up to 80&#37; of cases it manifests in the lower extremities&#44; although it can involve the upper extremities&#44; face and genitals&#46; Focal lymphatic abnormalities lead to an accumulation of lymph that&#44; when sustained&#44; stimulates fibrosis and produces a localised&#44; chronic and progressive oedema&#46; In the early stages&#44; the oedema is not associated with pain&#44; warmth&#44; erythema or functional impairment&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">In 1934&#44; Allen classified primary lymphoedema based on the age of onset into congenital&#44; praecox and tarda&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Recently&#44; Connell et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> proposed a new classification that took into account the different clinical presentations and known mutations&#44; giving rise to a complex diagnostic algorithm that encompassed 26 possible diagnoses&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">We present the cases of six patients with primary lymphoedema diagnosed in our centre&#44; whose clinical and demographic characteristics are summarised in <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#46; Most of the patients were female&#44; and the age of onset ranged between 5 and 12 years &#40;median&#44; 10&#46;5&#41;&#46; None of the patients had associated malformations or a relevant personal history&#46; Fifty percent had a family history of lymphoedema&#46; The time elapsed from the onset of symptoms to diagnosis ranged between 3 months and 4&#46;3 years &#40;median&#44; 9 months&#41;&#46; Patients were referred to a specialist to rule out arthritis as the cause of the swelling of the hand or dorsum of the foot&#46; None of the patients reported arthralgia&#44; morning stiffness or functional impairment&#44; and&#44; as is characteristic of lymphoedema&#44; the swelling improved with night rest and worsened with standing&#46; Physical examination found soft tissue oedema in all patients&#44; with no abnormalities in the joints&#46; The results of the blood panel &#40;blood counts&#44; acute phase reactants&#44; TSH&#44; albumin&#44; total protein&#41; and the routine urinalysis were also normal&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Most of the patients had been assessed previously in other departments of the hospital &#40;traumatology&#44; plastic surgery&#44; dermatology&#44; emergency&#41;&#44; and underwent different imaging tests &#40;conventional ultrasound&#44; Doppler ultrasound&#44; MRI&#41; that revealed the presence of oedema in the subcutaneous tissue&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The gold standard for the diagnosis of lymphoedema is lymphoscintigraphy&#44; which assesses the velocity of the migration of a radioactive tracer through the lymphatic vessels&#46; The tracer is injected in the dermis of an interdigital web space and tracked by a scintillation camera&#46; This is a minimally invasive and reproducible test that can be performed in children&#46; In patients with primary lymphoedema&#44; this method detects absent or delayed tracer migration<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; In a study conducted in 107 patients with primary lymphoedema&#44; Kinmonth et al&#46; presented the structural abnormalities that could be detected by lymphoscintigraphy and classified them into three categories&#58; aplasia &#40;absence of lymphatic vessels&#41;&#44; hypoplasia &#40;small number or size of lymphatic vessels&#41; and hyperplasia &#40;tortuous lymphatic vessels&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> All our patients presented this type of abnormalities in the lymphatic system&#44; and were classified according to the scheme proposed by Connell et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Other imaging techniques that have been used in more recent studies but have not been widely introduced in clinical practice include indocyanine green lymphography&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> which exposes the patient to radiation&#44; and magnetic resonance lymphangiography using gadobenate dimeglumine as the contrast agent&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> which does not involve exposure to radiation and can obtain dynamic sequences&#44; both of which are as efficacious as lymphoscintigraphy in detecting abnormalities in lymph circulation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">The management of primary lymphoedema is mainly conservative&#44; with the first line treatment consisting of manual lymph drainage or compression therapy&#46; Avoidance of prolonged standing&#44; the use compression stockings&#44; and regular physical activity combined with adequate skin care usually suffice in most cases&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> These measures were implemented in all of our patients and succeeded in controlling the lymphoedema&#46; The prognosis of lymphoedema is good&#44; as the disease stabilises in half of the patients &#40;57&#37;&#41;&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> although surgical measures are available for the most severe cases&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Swelling of the extremities in the paediatric age group requires a broad differential diagnosis including traumatic injury&#44; cellulitis&#44; arthritis&#44; venous insufficiency&#44; deep vein thrombosis and primary lymphoedema&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Knowledge of this condition and its characteristics&#8212;painless swelling in the extremities that changes with posture and is not associated with articular pain or limited range of motion&#8212;can prevent diagnostic delays as well as unnecessary referrals and diagnostic tests&#46;</p></span>"
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        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Barral Mena E&#44; Soriano-Ramos M&#44; Pavo Garc&#237;a MR&#44; Llorente Otones L&#44; de Inocencio Arocena J&#46; Linfedema primario fuera del periodo neonatal&#46; An Pediatr &#40;Barc&#41;&#46; 2016&#59;85&#58;47&#8211;49&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Lymphoscintigraphy image &#40;anterior view&#41; showing delayed lymph drainage in the left lower extremity&#44; with persisting radioactive tracer activity at the level of the popliteal lymph nodes &#40;arrow&#41;&#44; compatible with primary lymphoedema&#46;</p>"
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          "leyenda" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">FH&#58; family history&#59; F&#58; female&#59; &#40;R&#41;&#58; right&#59; &#40;L&#41;&#58; left&#59; LE&#58; lower extremity&#59; UE&#58; upper extremity&#59; APRs&#58; acute phase reactants&#59; MRI&#58; magnetic resonance imaging&#59; SCT&#58; subcutaneous tissue&#59; TSH&#58; thyroid-stimulating hormone&#59; M&#58; male&#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="" valign="top" scope="col" style="border-bottom: 2px solid black">&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Sex&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Family&#47;personal history&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Age of onset &#40;years&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Time to diagnosis &#40;months&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Lymphoedema location&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Blood tests &#40;WBC&#44; APRs&#44; TSH&#44; proteins&#41; and urinalysis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Lymphoscintigraphy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Other imaging tests&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Final diagnosis based on Connell et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> classification&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</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">1&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">No&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Left foot and ankle&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Delayed tracer elimination in &#40;L&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Ultrasound and MRI showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Unilateral late onset primary lymphoedema&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">2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">M&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">No&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">52&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Left foot&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Delayed tracer elimination in &#40;L&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal X-ray&#44; MRI showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Unilateral late onset primary lymphoedema&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">3&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">FH of mother with lymphoedema of the LEs&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">12&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">13&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Feet and ankles&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Delayed tracer elimination in &#40;L&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal abdominal ultrasound&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Primary lymphoedema &#40;Meige disease&#41;&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">4&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">No&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">11&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">16&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Left foot and ankle&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Absent visualisation of lymph drainage in &#40;L&#41;LE due to lymphatic system aplasia&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal X-ray&#44; normal Doppler ultrasound&#44; conventional ultrasound showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Unilateral late onset primary lymphoedema&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">5&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">FH of aunt and first-degree female cousin on the mother&#39;s side with oedema of the LEs&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">10&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">3&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Right foot and left hand&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Hypoplasia and lymphatic vascular insufficiency in &#40;L&#41;LE and &#40;R&#41;LE lymph node chains&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Ultrasound showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Late onset multisegmental lymphoedema&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">6&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">F&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">FH of mother and maternal first-degree female cousin with oedema of the LEs&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">10&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">13&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Right arm and forearm&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Normal&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Increased diameter of lymphatic vessels in &#40;R&#41;LE&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">MRI showing SCT oedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="left" valign="top">Late onset multisegmental lymphoedema&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
                  """
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Demographic&#44; clinical and diagnostic characteristics of patients with a primary lymphoedema diagnosis&#46;</p>"
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    "bibliografia" => array:2 [
      "titulo" => "References"
      "seccion" => array:1 [
        0 => array:2 [
          "identificador" => "bibs0005"
          "bibliografiaReferencia" => array:6 [
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              "identificador" => "bib0035"
              "etiqueta" => "1"
              "referencia" => array:1 [
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                          ]
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                  "host" => array:1 [
                    0 => array:2 [
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                        0 => array:2 [
                          "etal" => false
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                            0 => "F&#46;C&#46; Connell"
                            1 => "K&#46; Gordon"
                            2 => "G&#46; Brice"
                            3 => "V&#46; Keeley"
                            4 => "S&#46; Jeffery"
                            5 => "P&#46;S&#46; Mortimer"
                          ]
                        ]
                      ]
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                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1111/cge.12173"
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23621851"
                            "web" => "Medline"
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            2 => array:3 [
              "identificador" => "bib0045"
              "etiqueta" => "3"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
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Article information
ISSN: 23412879
Original language: English
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