Scientific Letter
d-Lactic acidosis in a ten months old infant with short bowel syndrome: Early suspicion equals early treatment
Acidosis d-láctica en un niño de diez meses afecto de un síndrome de intestino corto: la rápida sospecha equivale a un rápido tratamiento
P. Obando Pachecoa,
, V.M. Navas Lópezb, R. Yahyaoui Macíasc, C. Sierra Salinasb
Corresponding author
a Unidad de Gestión Clínica de Pediatría, Hospital Materno Infantil, Hospital Regional Universitario de Málaga, Spain
b Unidad de Gastroenterología y Nutrición Infantil, Unidad de Gestión Clínica de Pediatría, IBIMA, Hospital Materno Infantil, Hospital Regional Universitario de Málaga, Spain
c Laboratorio de Metabolopatías y Centro de Cribado Neonatal de Andalucía Oriental, Unidad de Gestión Clínica de Laboratorios, Hospital Materno Infantil, Hospital Regional Universitario de Málaga, Spain
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Stratosphere sign: linear pattern at both sides of the pleural line (*), indicating the absence of lung sliding. (b) M-mode. Lung point: transition from the normal lung (right side, linear granular pattern) to the pneumothorax (left side, linear pattern). (c) M-mode after resolution of pneumothorax. Seashore sign: combination of a linear patter and a granular pattern at either side of the pleural line (*), indicating normal lung sliding. <span class="elsevierStyleItalic">Case 2</span>: (a) right-sided pleural effusion (quad sign) with consolidated underlying lung parenchyma (tissue-like sign) and bronchograms (hyperechoic foci [^]). (b) Resolution of effusion after chest tube placement. Lung consolidation above the diaphragm (*), difficult to differentiate from the liver (arrow). (c) Left hemithorax: basal pleural effusion and consolidated underlying lung (tissue-like sign and bronchograms [^]). (d) Left hemithorax: lung parenchyma consolidation, with detection of tissue-like sign and shred sign (pattern of consolidation with an irregular lower border indicating where the consolidated parenchyma makes contact with the normal lung tissue [arrows]). <span class="elsevierStyleItalic">Case 3</span>: coalescing B lines in (a) anterior and superior (AS) regions of right hemithorax, (b) anterior and inferior (AI) right hemithorax, (c) AS left hemithorax, (d) AI left hemithorax. Diagnosis of bilateral diffuse interstitial syndrome. (e) Ideal areas for ultrasound examination for the diagnosis of diffuse interstitial lung syndrome. AS, AI, lateral superior (LS), lateral inferior (LI), right (D), left (I). <span class="elsevierStyleItalic">Case 4</span>: M-mode (a) Right diaphragm: maximal diaphragmatic excursion between inspiration and expiration of 0.88<span class="elsevierStyleHsp" style=""></span>cm (50% compared to contralateral excursion). 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Obando Pacheco, V.M. Navas López, R. Yahyaoui Macías, C. Sierra Salinas" "autores" => array:4 [ 0 => array:4 [ "nombre" => "P." "apellidos" => "Obando Pacheco" "email" => array:1 [ 0 => "pabobapac@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "V.M." "apellidos" => "Navas López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "R." "apellidos" => "Yahyaoui Macías" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "C." "apellidos" => "Sierra Salinas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Unidad de Gestión Clínica de Pediatría, Hospital Materno Infantil, Hospital Regional Universitario de Málaga, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Unidad de Gastroenterología y Nutrición Infantil, Unidad de Gestión Clínica de Pediatría, IBIMA, Hospital Materno Infantil, Hospital Regional Universitario de Málaga, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Laboratorio de Metabolopatías y Centro de Cribado Neonatal de Andalucía Oriental, Unidad de Gestión Clínica de Laboratorios, Hospital Materno Infantil, Hospital Regional Universitario de Málaga, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Acidosis d-láctica en un niño de diez meses afecto de un síndrome de intestino corto: la rápida sospecha equivale a un rápido tratamiento" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Metabolic acidosis is a disorder of the acid–base balance which may be due to bicarbonate losses, deficient acid elimination by the kidney, exogenous intake of acids or an endogenous increased acid production. Within this last group, lactic acidosis should be noted, because of its frequency, morbidity and potential mortality. Lactic acidosis, which is generally caused by accumulation of the <span class="elsevierStyleSmallCaps">l</span>-lactic isomer, can be congenital or secondary, the latter is in relation to tissue hypoxia (type A) or to hepatic, renal or oncologic diseases, intense exercise, seizures or toxics (type B). Less common are the situations where the cumulative isomer is the <span class="elsevierStyleSmallCaps">d</span>-lactic,<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–3</span></a> which have occasionally been reported in patients suffering from short bowel syndrome (SBS), presenting with a metabolic acidosis, increased anion GAP and neurological symptoms.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present a case of a ten month old infant with SBS secondary to gastroschisis and intestinal necrosis. He had undergone surgery three times during the neonatal period with a remaining small bowel of 30<span class="elsevierStyleHsp" style=""></span>cm preserving the ileocecal valve. The patient's diet was composed of extensively hydrolysed lactose-free formula, gluten-free cereal, vegetables and chicken. Feeding was done mostly through nasogastric tube, with some participation of oral feeding. Metronidazole 20<span class="elsevierStyleHsp" style=""></span>mg/kg/day and cefixime 8<span class="elsevierStyleHsp" style=""></span>mg/kg/day were administered alternatively the first week of each month as intestinal decontamination protocol. The month before admission, abundant stool production with decreased consistency associated to weight loss was present. Coincidentally, the decontamination protocol was not followed and 48<span class="elsevierStyleHsp" style=""></span>h prior to admission, he developed a respiratory syncytial virus respiratory tract infection. On admission, the anthropometric data showed severe malnutrition: weight 4.41<span class="elsevierStyleHsp" style=""></span>kg (<span class="elsevierStyleHsp" style=""></span>cm (<span class="elsevierStyleHsp" style=""></span>mmol/L) and the anion GAP was increased (27<span class="elsevierStyleHsp" style=""></span>mEq/L) with normal lactic levels (0.7<span class="elsevierStyleHsp" style=""></span>mmol/L). Ketonuria was absent. Correction therapy was started with intravenous fluids containing bicarbonate and oral metronidazole. Prior to this, a venous blood sample in a lithium heparin tube was immediately centrifuged. Plasma was frozen and sent for <span class="elsevierStyleSmallCaps">d</span>-lactic acid analysis to a reference laboratory (Birmingham Children's Hospital, Birmingham, through Reference Laboratory, Barcelona), confirming the suspected diagnosis (<span class="elsevierStyleSmallCaps">d</span>-lactic acid<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>6<span class="elsevierStyleHsp" style=""></span>mmol/L). The outcome was satisfactory with normalization of neurological symptoms and gradual improvement of the acid–base balance.</p><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleSmallCaps">d</span>-Lactic acidosis usually originates from an accumulation of <span class="elsevierStyleSmallCaps">d</span>-lactic secondary to an elevated synthesis by gastrointestinal tract bacteria.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> Production of <span class="elsevierStyleSmallCaps">d</span>-lactic is minimal under normal conditions and is easily metabolized by the mitochondrial <span class="elsevierStyleSmallCaps">d</span>-lactate dehydrogenase.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2,3</span></a> However, in the SBS, a high carbohydrate intake may generate an increased production and subsequent accumulation of <span class="elsevierStyleSmallCaps">d</span>-lactate because of the bacterial overgrowth.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–3</span></a> This entity should be suspected in patients associating neurological symptoms and metabolic acidosis with elevated anion GAP without increasing <span class="elsevierStyleSmallCaps">l</span>-lactic acid, as in our case.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4,5</span></a> We could not find in the literature any report involving such a young infant. We highlight that in our routine blood analysis, only <span class="elsevierStyleSmallCaps">l</span>-lactic acid is detected. When this condition is suspected, it should be confirmed by a special measurement of serum or plasma <span class="elsevierStyleSmallCaps">d</span>-lactic concentration. Normal values of <span class="elsevierStyleSmallCaps">d</span>-lactic acid in blood are undetectable, being those above 3<span class="elsevierStyleHsp" style=""></span>mmol/L pathological.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The treatment is based on pathogenic flora elimination (administration of oral nonabsorbable antibiotics as metronidazole, neomycin or vancomycin), correction of the acidosis and carbohydrate restriction diet.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2,3</span></a> Administration of probiotics is controversial. <span class="elsevierStyleItalic">Bifidobacterium breve</span> and <span class="elsevierStyleItalic">Lactobacillus casei</span> are theoretically non <span class="elsevierStyleSmallCaps">d</span>-lactate producing flora and have been occasionally used as adjuvant therapy with antibiotics, in order to replace the pathogenic flora.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> However, two cases of <span class="elsevierStyleSmallCaps">d</span>-lactic acidosis in patients with SBS attributed to the administration of probiotics containing <span class="elsevierStyleSmallCaps">d</span>-lactate producing strains (<span class="elsevierStyleItalic">Lactobacillus acidophilus</span>, <span class="elsevierStyleItalic">Lactobacillus bulgaricus</span> and <span class="elsevierStyleItalic">Bifidobacterium infantis</span>) have been reported.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> Probiotics must be carefully selected in children with SBS.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> There are few data available related to the ideal maintenance therapy due to the lack of controlled studies. Avoiding fast-acting carbohydrate diets, periodical decontamination or the use of non <span class="elsevierStyleSmallCaps">d</span>-lactate producing probiotics are different options which should be individualized in patients at risk of developing <span class="elsevierStyleSmallCaps">d</span>-lactic acidosis.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2,3,6</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">This case is a reminder of the importance of suspecting this rare entity in SBS affected children presenting with neurological symptoms and metabolic acidosis where routine blood tests do not detect the increased anion. In these situations an early treatment should be mandatory.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Pacheco PO, López VMN, Macías RY, Salinas CS. Acidosis d-láctica en un niño de diez meses afecto de un síndrome de intestino corto: la rápida sospecha equivale a un rápido tratamiento. An Pediatr (Barc). 2016;84:56–57.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Current management of short bowel syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "J.S. Thompson" 1 => "F.A. Rochling" 2 => "R.A. Weseman" 3 => "D.F. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 12 | 9 | 21 |
| 2024 October | 78 | 57 | 135 |
| 2024 September | 93 | 52 | 145 |
| 2024 August | 100 | 70 | 170 |
| 2024 July | 117 | 41 | 158 |
| 2024 June | 101 | 40 | 141 |
| 2024 May | 98 | 56 | 154 |
| 2024 April | 79 | 32 | 111 |
| 2024 March | 72 | 24 | 96 |
| 2024 February | 68 | 30 | 98 |
| 2024 January | 79 | 31 | 110 |
| 2023 December | 142 | 22 | 164 |
| 2023 November | 83 | 23 | 106 |
| 2023 October | 52 | 23 | 75 |
| 2023 September | 54 | 24 | 78 |
| 2023 August | 50 | 24 | 74 |
| 2023 July | 47 | 18 | 65 |
| 2023 June | 58 | 23 | 81 |
| 2023 May | 63 | 27 | 90 |
| 2023 April | 35 | 14 | 49 |
| 2023 March | 63 | 30 | 93 |
| 2023 February | 64 | 15 | 79 |
| 2023 January | 41 | 20 | 61 |
| 2022 December | 78 | 35 | 113 |
| 2022 November | 60 | 34 | 94 |
| 2022 October | 53 | 42 | 95 |
| 2022 September | 62 | 33 | 95 |
| 2022 August | 47 | 59 | 106 |
| 2022 July | 44 | 38 | 82 |
| 2022 June | 46 | 31 | 77 |
| 2022 May | 70 | 37 | 107 |
| 2022 April | 57 | 42 | 99 |
| 2022 March | 57 | 43 | 100 |
| 2022 February | 91 | 29 | 120 |
| 2022 January | 66 | 29 | 95 |
| 2021 December | 51 | 48 | 99 |
| 2021 November | 64 | 47 | 111 |
| 2021 October | 65 | 53 | 118 |
| 2021 September | 41 | 33 | 74 |
| 2021 August | 67 | 33 | 100 |
| 2021 July | 84 | 23 | 107 |
| 2021 June | 44 | 14 | 58 |
| 2021 May | 68 | 31 | 99 |
| 2021 April | 140 | 40 | 180 |
| 2021 March | 83 | 33 | 116 |
| 2021 February | 62 | 14 | 76 |
| 2021 January | 95 | 16 | 111 |
| 2020 December | 129 | 20 | 149 |
| 2020 November | 68 | 22 | 90 |
| 2020 October | 47 | 24 | 71 |
| 2020 September | 59 | 19 | 78 |
| 2020 August | 49 | 12 | 61 |
| 2020 July | 58 | 20 | 78 |
| 2020 June | 64 | 14 | 78 |
| 2020 May | 75 | 37 | 112 |
| 2020 April | 43 | 19 | 62 |
| 2020 March | 38 | 22 | 60 |
| 2020 February | 30 | 21 | 51 |
| 2020 January | 43 | 16 | 59 |
| 2019 December | 45 | 22 | 67 |
| 2019 November | 31 | 19 | 50 |
| 2019 October | 57 | 16 | 73 |
| 2019 September | 46 | 16 | 62 |
| 2019 August | 43 | 17 | 60 |
| 2019 July | 41 | 23 | 64 |
| 2019 June | 47 | 27 | 74 |
| 2019 May | 92 | 27 | 119 |
| 2019 April | 63 | 28 | 91 |
| 2019 March | 50 | 22 | 72 |
| 2019 February | 44 | 16 | 60 |
| 2019 January | 44 | 27 | 71 |
| 2018 December | 55 | 34 | 89 |
| 2018 November | 78 | 38 | 116 |
| 2018 October | 113 | 35 | 148 |
| 2018 September | 33 | 15 | 48 |
| 2018 August | 1 | 0 | 1 |
| 2018 June | 3 | 0 | 3 |
| 2018 May | 6 | 0 | 6 |
| 2018 April | 42 | 0 | 42 |
| 2018 March | 33 | 0 | 33 |
| 2018 February | 13 | 0 | 13 |
| 2018 January | 20 | 0 | 20 |
| 2017 December | 14 | 0 | 14 |
| 2017 November | 17 | 0 | 17 |
| 2017 October | 18 | 0 | 18 |
| 2017 September | 16 | 0 | 16 |
| 2017 August | 12 | 0 | 12 |
| 2017 July | 18 | 0 | 18 |
| 2017 June | 26 | 11 | 37 |
| 2017 May | 25 | 6 | 31 |
| 2017 April | 15 | 4 | 19 |
| 2017 March | 12 | 3 | 15 |
| 2017 February | 16 | 2 | 18 |
| 2017 January | 8 | 3 | 11 |
| 2016 December | 23 | 3 | 26 |
| 2016 November | 34 | 8 | 42 |
| 2016 October | 56 | 4 | 60 |
| 2016 September | 42 | 5 | 47 |
| 2016 August | 34 | 8 | 42 |
| 2016 July | 21 | 3 | 24 |
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