Scientific Letter
Cow's milk protein intolerance imitating septic shock in a young infant
Intolerancia a la proteína de la leche de vaca simulando shock séptico en un lactante pequeño
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array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Departamento de Pediatría, Centro Hospitalar São João, Oporto, Portugal" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Departamento of de Inmunoalergología Pediátrica, Centro Hospitalar São João, Oporto, Portugal" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Intolerancia a la proteína de la leche de vaca simulando shock séptico en un lactante pequeño" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Food protein-induced enterocolitis syndrome (FPIES) is the most serious form of enteropathy. The pathophysiology of this syndrome is not completely understood, but it is believed that ingestion of the triggering food is followed by T-lymphocyte activation, resulting in a local inflammatory response, increased gastrointestinal (GI) permeability and subsequent passage of fluids to the GI lumen.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A boy aged 4 months visited the emergency department (ED) with profuse vomiting and signs of hypovolaemic shock. He had been breastfed until the age of three months, when supplementation with one feed of cow's milk formula a day was introduced. In the two days that followed, he vomited once a day right after ingesting the cow's milk. On day 3, he developed persistent vomiting and bloody diarrhoea and became increasingly lethargic.</p><p id="par0015" class="elsevierStylePara elsevierViewall">In the ED, he presented in shock, with a blood pressure of 60/25<span class="elsevierStyleHsp" style=""></span>mmHg, poor peripheral perfusion with cold extremities, and no other abnormalities in the physical examination.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The diagnoses considered at this point were sepsis and cow's milk protein allergy.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The blood tests showed absence of leucocytosis (10,990/μL) with relative neutrophilia (76.2% neutrophils), thrombocytosis (645,000/μL platelets), a negative result for C-reactive protein, normal levels of serum electrolytes and normal renal function. There were no abnormalities in the cerebrospinal fluid or the urine.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The boy was given a bolus of isotonic fluid (20<span class="elsevierStyleHsp" style=""></span>mL/kg) to reverse the shock, intramuscular adrenalin and methylprednisolone. Antibiotic therapy was initiated with ceftriaxone.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The patient had normal total IgE levels, and negative results for specific IgE against cow's milk protein. The triptase value measured at the emergency department was also normal.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Once admitted to the paediatrics ward, he was fed with breast milk or with an amino acid-based formula, with complete resolution of symptoms in less than 12<span class="elsevierStyleHsp" style=""></span>h. All the culture results were negative.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The compatible history and favourable clinical outcome allowed the conclusion that the patient had cow's milk protein-induced enterocolitis syndrome (MPIES).</p><p id="par0050" class="elsevierStylePara elsevierViewall">Two months after the acute event, the patient underwent an oral food challenge (OFC) with extensively hydrolysed milk formula (EHF), with recurrence of symptoms, but at the age of 12 months he tolerated this formula during a new OFC.</p><p id="par0055" class="elsevierStylePara elsevierViewall">At the age of 2, under medical supervision, the patient underwent oral tolerance induction. The starting dose was 1<span class="elsevierStyleHsp" style=""></span>mL, which was doubled weekly until reaching a 200<span class="elsevierStyleHsp" style=""></span>mL dose. The patient exhibited good tolerance.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The clinical manifestations of FPIES and its severity are dose-dependent. In this case, the identification of cow's milk proteins as the trigger was confirmed by the previous history of exposure to small amounts followed by reactions of increasing severity. The diagnosis of acute FPIES requires that a patient meets the major criterion<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">3</span></a>:<ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">•</span><p id="par0065" class="elsevierStylePara elsevierViewall">Vomiting in the 1–4<span class="elsevierStyleHsp" style=""></span>h period after ingestion of the suspect food in the absence of classic IgE-mediated allergic skin or respiratory symptoms;</p></li></ul></p><p id="par0070" class="elsevierStylePara elsevierViewall">and at least 3 minor criteria:<ul class="elsevierStyleList" id="lis0010"><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">•</span><p id="par0075" class="elsevierStylePara elsevierViewall">A second (or more) episode of repetitive vomiting after eating the same suspect food</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">•</span><p id="par0080" class="elsevierStylePara elsevierViewall">Episode of repetitive vomiting 1–4<span class="elsevierStyleHsp" style=""></span>h after eating a different food</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">•</span><p id="par0085" class="elsevierStylePara elsevierViewall">Extreme lethargy with any suspected reaction</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">•</span><p id="par0090" class="elsevierStylePara elsevierViewall">Marked pallor with any suspected reaction</p></li><li class="elsevierStyleListItem" id="lsti0030"><span class="elsevierStyleLabel">•</span><p id="par0095" class="elsevierStylePara elsevierViewall">Need for emergency room visit with any suspected reaction</p></li><li class="elsevierStyleListItem" id="lsti0035"><span class="elsevierStyleLabel">•</span><p id="par0100" class="elsevierStylePara elsevierViewall">Need for intravenous fluid support with any suspected reaction</p></li><li class="elsevierStyleListItem" id="lsti0040"><span class="elsevierStyleLabel">•</span><p id="par0105" class="elsevierStylePara elsevierViewall">Diarrhoea within 24<span class="elsevierStyleHsp" style=""></span>h</p></li><li class="elsevierStyleListItem" id="lsti0045"><span class="elsevierStyleLabel">•</span><p id="par0110" class="elsevierStylePara elsevierViewall">Hypotension</p></li><li class="elsevierStyleListItem" id="lsti0050"><span class="elsevierStyleLabel">•</span><p id="par0115" class="elsevierStylePara elsevierViewall">Hypothermia</p></li></ul></p><p id="par0120" class="elsevierStylePara elsevierViewall">This infant fulfilled the major criterion and all of the minor criteria with the exception of hypothermia. He also had thrombocytosis, which has been reported in 65% of patients with acute FPIES, and relative neutrophilia, a common laboratory finding in patients with positive oral food challenges (OFC).<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a></p><p id="par0125" class="elsevierStylePara elsevierViewall">In patients with a single documented episode, performance of an OFC can be considered.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a> In this case, we considered that the typical history made it unnecessary.</p><p id="par0130" class="elsevierStylePara elsevierViewall">As occurs in IgE-mediated cow's milk allergies, where reactions may be triggered by an EHF, about 10–40% of patients with MPIES do not tolerate these formulas and require formulas based on amino acids,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a> although their financial and nutritional impact must be taken into account.</p><p id="par0135" class="elsevierStylePara elsevierViewall">There are geographic and institutional variations in the timing of OFC in patients with FPIES. Most authors defend that it should not be tried before 12–24 months have elapsed since the last acute episode,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">2</span></a> but good results have been observed after 10 months in patients with MFPIES.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">3</span></a> It is important to emphasize that regardless of the timing, OFCs should always be performed in appropriate settings, where resuscitation equipment and staff trained in the identification and treatment of these potentially severe reactions are always available.</p><p id="par0140" class="elsevierStylePara elsevierViewall">In conclusion, knowledge of this syndrome and the severity of its presentation allows the early removal of the trigger and prevents unnecessary testing and pharmacological treatment. The follow-up and management of these patients can be challenging, but the disorder has a favourable prognosis and resolves fully in most cases by age of 3–5 years.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">1</span></a></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: Jacob S, Vitor AB. Intolerancia a la proteína de la leche de vaca simulando shock séptico en un lactante pequeño. An Pediatr (Barc). 2019;90:52–53.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:3 [ 0 => array:3 [ "identificador" => "bib0020" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Food protein-induced enterocolitis syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A. Nowak-Wegrzyn" 1 => "E. Jarocka-Cyrta" 2 => "A.P.B. 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| 2022 November | 101 | 30 | 131 |
| 2022 October | 143 | 42 | 185 |
| 2022 September | 89 | 26 | 115 |
| 2022 August | 139 | 46 | 185 |
| 2022 July | 94 | 40 | 134 |
| 2022 June | 99 | 28 | 127 |
| 2022 May | 92 | 37 | 129 |
| 2022 April | 61 | 32 | 93 |
| 2022 March | 92 | 42 | 134 |
| 2022 February | 70 | 25 | 95 |
| 2022 January | 84 | 32 | 116 |
| 2021 December | 70 | 31 | 101 |
| 2021 November | 82 | 51 | 133 |
| 2021 October | 252 | 76 | 328 |
| 2021 September | 60 | 36 | 96 |
| 2021 August | 65 | 38 | 103 |
| 2021 July | 50 | 35 | 85 |
| 2021 June | 75 | 46 | 121 |
| 2021 May | 86 | 67 | 153 |
| 2021 April | 128 | 74 | 202 |
| 2021 March | 99 | 42 | 141 |
| 2021 February | 72 | 17 | 89 |
| 2021 January | 96 | 19 | 115 |
| 2020 December | 92 | 29 | 121 |
| 2020 November | 63 | 32 | 95 |
| 2020 October | 56 | 34 | 90 |
| 2020 September | 66 | 21 | 87 |
| 2020 August | 78 | 10 | 88 |
| 2020 July | 73 | 17 | 90 |
| 2020 June | 81 | 15 | 96 |
| 2020 May | 56 | 13 | 69 |
| 2020 April | 36 | 18 | 54 |
| 2020 March | 46 | 10 | 56 |
| 2020 February | 39 | 11 | 50 |
| 2020 January | 30 | 18 | 48 |
| 2019 December | 51 | 16 | 67 |
| 2019 November | 37 | 14 | 51 |
| 2019 October | 35 | 693 | 728 |
| 2019 September | 27 | 22 | 49 |
| 2019 August | 41 | 20 | 61 |
| 2019 July | 38 | 29 | 67 |
| 2019 June | 44 | 21 | 65 |
| 2019 May | 93 | 17 | 110 |
| 2019 April | 53 | 25 | 78 |
| 2019 March | 37 | 26 | 63 |
| 2019 February | 55 | 10 | 65 |
| 2019 January | 198 | 27 | 225 |
| 2018 December | 35 | 20 | 55 |
| 2018 November | 73 | 19 | 92 |
| 2018 October | 75 | 26 | 101 |
| 2018 September | 28 | 18 | 46 |
| 2018 August | 0 | 0 | 0 |
| 2018 July | 0 | 3 | 3 |
| 2018 June | 0 | 3 | 3 |
| 2018 May | 0 | 8 | 8 |
| 2018 April | 0 | 5 | 5 |
Show all
