Case and Research Letter
Acrodermatitis Enteropathica in a Breast-Fed Infant
Acrodermatitis enteropática en bebé alimentado con lactancia materna
E. Gutiérrez-González
, A. Álvarez-Pérez, M. Loureiro, D. Sánchez-Aguilar, J. Toribio
Corresponding author
Departamento de Dermatología, Complejo Hospitalario Universitario, Facultad de Medicina, Santiago de Compostela, Spain
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Gutiérrez-González, A. Álvarez-Pérez, M. Loureiro, D. Sánchez-Aguilar, J. Toribio" "autores" => array:5 [ 0 => array:4 [ "nombre" => "E." "apellidos" => "Gutiérrez-González" "email" => array:1 [ 0 => "egutgon@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "A." "apellidos" => "Álvarez-Pérez" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Loureiro" ] 3 => array:2 [ "nombre" => "D." "apellidos" => "Sánchez-Aguilar" ] 4 => array:2 [ "nombre" => "J." "apellidos" => "Toribio" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Departamento de Dermatología, Complejo Hospitalario Universitario, Facultad de Medicina, Santiago de Compostela, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Acrodermatitis enteropática en bebé alimentado con lactancia materna" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2055 "Ancho" => 2737 "Tamanyo" => 442775 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Scaly, erythematous plaques on the face (in a periorificial distribution) (A), on the scalp (B), and in the diaper area (C).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Acrodermatitis enteropathica (AE) is a rare autosomal recessively inherited disease caused by alterations in zinc absorption in the digestive tract.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> Although it was first described in 1942 by Danbolt and Closs,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> it was not until 1973 that Moynahan and Barnes<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> identified its association with low plasma zinc levels. It is estimated to affect 1 in 500<span class="elsevierStyleHsp" style=""></span>000 children.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Clinically, it is characterized by acral and periorificial dermatitis, alopecia, and diarrhea, although this triad of symptoms only occurs in 20% of cases.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> AE usually appears days or weeks after birth in bottle-fed infants, or shortly after discontinuing breastfeeding in exclusively breastfed infants.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Here we describe the case of a full-term male infant who was diagnosed with AE. The only remarkable finding in his history was neonatal indirect hyperbilirubinemia. The patient, who had been exclusively breastfed since birth, developed cutaneous lesions on the scalp, around the mouth, and in the diaper area at 3 months of age. Additionally, his parents reported that he was more irritable at night. He had received various treatments for the cutaneous lesions, including topical and systemic antibiotics, topical antifungal drugs, and topical corticosteroids, but showed no improvement.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Skin examination revealed scaly, erythematous plaques with well-demarcated edges and psoriasiform morphology, and some honey-colored crusts covering zones of erosion. The plaques were located on the face, predominantly in the perioral, perinasal, and frontal areas (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A), as well as on the scalp (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B) and on the neck, buttocks, and intergluteal cleft (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). Additionally, several isolated plaques with similar characteristics were observed on the thighs and on the dorsal surfaces of fingers and toes.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A serum zinc test showed clearly deficient levels of zinc (9<span class="elsevierStyleHsp" style=""></span>μg/dL; normal range, 70–120<span class="elsevierStyleHsp" style=""></span>μg/dL), supporting the suspected diagnosis of AE. Normal zinc levels in maternal milk ruled out an AE-like eruption. After confirming the diagnosis of AE, zinc sulfite treatment was started at a dosage of 3<span class="elsevierStyleHsp" style=""></span>mg/kg/d; this led to rapid resolution of the cutaneous lesions and an improvement in irritability. Currently, after 2 years of treatment, the child shows normal growth parameters for his age, has had no recurrence of the symptoms, and has normal serum zinc levels.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The first symptom is usually acral and periorificial dermatitis and only more advanced cases present the classic triad of alopecia, diarrhea, and skin lesions.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The disease is characterized by low levels of serum zinc, caused by deficient absorption of this metal in the digestive tract.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is now known that this zinc deficiency is due to a defect in the AE gene (<span class="elsevierStyleItalic">SLC39A4)</span> located on the 8q24 chromosome, which encodes Zip4, a zinc transporter in the cells of the small intestine, and primarily in the jejunum.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Clinically, scaly erythematous plaques with psoriasiform morphology, distributed symmetrically in acral and periorificial areas, are frequently observed. Other symptoms include vesiculobullous lesions, alopecia, onychodystrophy, onycholysis, pachyonychia, stomatitis, cheilitis, blepharitis, photophobia, and conjunctivitis. When diagnosis is delayed, patients can develop advanced disease, characterized by increased morbidity and mortality; conditions include growth and mental retardation, irritability, slow wound healing, immunity alterations, hypogonadism, and anemia.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Zinc deficiency can have either a genetic or an acquired origin. The genetic form is classically known as AE and, as mentioned above, is caused by a genetically determined defect in zinc absorption. The acquired forms are clinically similar to classic AE. Three basic causes are known: (a) zinc deficiency in maternal milk,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> also known as lactogenic acrodermatitis; (b) a complete lack of zinc in the parenteral nutrition; and (c) the presence of other malabsorption syndromes, such as Crohn disease and celiac disease, in which absorption is compromised, or metabolic disorders associated with a deficit of other trace elements besides zinc.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Diagnosis is primarily clinical, but it is important to acquire data that support the diagnosis (e.g., low serum zinc levels) and to exclude other causes, such as malnutrition, zinc deficiency in maternal milk, and other metabolic alterations.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> While histopathologic findings are not specific, they can be useful for a preliminary diagnosis.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Characteristically, zinc replacement therapy at a dosage of 3<span class="elsevierStyleHsp" style=""></span>mg/kg/d<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> leads to rapid improvement and resolution of signs and symptoms, substantiating the diagnosis. It is advisable to begin treatment as soon as possible, since delaying it can have severe consequences, including death.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">AE usually presents after discontinuation of breastfeeding due to the fact that full-term newborns gain sufficient zinc stores during the last 10 weeks of pregnancy. Furthermore, maternal milk normally contains sufficient zinc for infant development. However, on rare occasions, AE can also occur in full-term infants who have been exclusively breastfed,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> as was the case for our patient. It is important to exclude zinc deficiency in maternal milk in such cases, since this condition requires a much shorter treatment than AE, which must be treated and monitored throughout life.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Gutiérrez-González E, et al. Acrodermatitis enteropática en bebé alimentado con lactancia materna. Actas Dermosifiliogr. 2012;103:170–2.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2055 "Ancho" => 2737 "Tamanyo" => 442775 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Scaly, erythematous plaques on the face (in a periorificial distribution) (A), on the scalp (B), and in the diaper area (C).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Acrodermatitis enteropathica and an overview of zinc metabolism" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "E. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 16 | 6 | 22 |
| 2024 October | 91 | 37 | 128 |
| 2024 September | 112 | 29 | 141 |
| 2024 August | 106 | 63 | 169 |
| 2024 July | 118 | 47 | 165 |
| 2024 June | 134 | 38 | 172 |
| 2024 May | 92 | 38 | 130 |
| 2024 April | 108 | 18 | 126 |
| 2024 March | 99 | 30 | 129 |
| 2024 February | 88 | 36 | 124 |
| 2024 January | 76 | 46 | 122 |
| 2023 December | 81 | 38 | 119 |
| 2023 November | 97 | 38 | 135 |
| 2023 October | 77 | 29 | 106 |
| 2023 September | 99 | 41 | 140 |
| 2023 August | 101 | 24 | 125 |
| 2023 July | 114 | 28 | 142 |
| 2023 June | 136 | 20 | 156 |
| 2023 May | 158 | 36 | 194 |
| 2023 April | 180 | 18 | 198 |
| 2023 March | 107 | 22 | 129 |
| 2023 February | 157 | 23 | 180 |
| 2023 January | 93 | 25 | 118 |
| 2022 December | 97 | 28 | 125 |
| 2022 November | 63 | 21 | 84 |
| 2022 October | 62 | 18 | 80 |
| 2022 September | 40 | 34 | 74 |
| 2022 August | 31 | 35 | 66 |
| 2022 July | 47 | 37 | 84 |
| 2022 June | 36 | 30 | 66 |
| 2022 May | 122 | 34 | 156 |
| 2022 April | 120 | 44 | 164 |
| 2022 March | 125 | 63 | 188 |
| 2022 February | 147 | 37 | 184 |
| 2022 January | 102 | 36 | 138 |
| 2021 December | 68 | 46 | 114 |
| 2021 November | 100 | 46 | 146 |
| 2021 October | 91 | 52 | 143 |
| 2021 September | 69 | 41 | 110 |
| 2021 August | 70 | 35 | 105 |
| 2021 July | 93 | 34 | 127 |
| 2021 June | 76 | 30 | 106 |
| 2021 May | 100 | 44 | 144 |
| 2021 April | 148 | 67 | 215 |
| 2021 March | 91 | 27 | 118 |
| 2021 February | 75 | 25 | 100 |
| 2021 January | 63 | 19 | 82 |
| 2020 December | 46 | 19 | 65 |
| 2020 November | 55 | 21 | 76 |
| 2020 October | 40 | 13 | 53 |
| 2020 September | 36 | 16 | 52 |
| 2020 August | 30 | 22 | 52 |
| 2020 July | 26 | 19 | 45 |
| 2020 June | 39 | 26 | 65 |
| 2020 May | 21 | 27 | 48 |
| 2020 April | 26 | 24 | 50 |
| 2020 March | 45 | 30 | 75 |
| 2020 February | 6 | 9 | 15 |
| 2020 January | 0 | 1 | 1 |
| 2019 December | 0 | 5 | 5 |
| 2019 November | 0 | 6 | 6 |
| 2019 October | 0 | 4 | 4 |
| 2019 September | 0 | 6 | 6 |
| 2019 August | 0 | 6 | 6 |
| 2019 July | 0 | 15 | 15 |
| 2019 June | 0 | 20 | 20 |
| 2019 May | 1 | 30 | 31 |
| 2019 April | 1 | 33 | 34 |
| 2019 March | 0 | 16 | 16 |
| 2019 February | 0 | 19 | 19 |
| 2019 January | 0 | 8 | 8 |
| 2018 December | 0 | 9 | 9 |
| 2018 November | 0 | 3 | 3 |
| 2018 October | 1 | 0 | 1 |
| 2018 September | 2 | 1 | 3 |
| 2018 August | 0 | 6 | 6 |
| 2018 July | 0 | 2 | 2 |
| 2018 May | 0 | 6 | 6 |
| 2018 March | 17 | 4 | 21 |
| 2018 February | 127 | 10 | 137 |
| 2018 January | 117 | 17 | 134 |
| 2017 December | 123 | 8 | 131 |
| 2017 November | 77 | 15 | 92 |
| 2017 October | 81 | 7 | 88 |
| 2017 September | 77 | 23 | 100 |
| 2017 August | 92 | 17 | 109 |
| 2017 July | 83 | 9 | 92 |
| 2017 June | 99 | 35 | 134 |
| 2017 May | 76 | 27 | 103 |
| 2017 April | 66 | 22 | 88 |
| 2017 March | 62 | 40 | 102 |
| 2017 February | 50 | 22 | 72 |
| 2017 January | 72 | 21 | 93 |
| 2016 December | 145 | 20 | 165 |
| 2016 November | 222 | 18 | 240 |
| 2016 October | 221 | 27 | 248 |
| 2016 September | 277 | 19 | 296 |
| 2016 August | 248 | 9 | 257 |
| 2016 July | 86 | 21 | 107 |
| 2016 June | 13 | 18 | 31 |
| 2016 May | 9 | 21 | 30 |
| 2016 April | 7 | 3 | 10 |
| 2016 March | 13 | 1 | 14 |
| 2016 February | 9 | 5 | 14 |
| 2016 January | 14 | 3 | 17 |
| 2015 December | 7 | 1 | 8 |
| 2015 November | 34 | 11 | 45 |
| 2015 October | 65 | 11 | 76 |
| 2015 September | 75 | 1 | 76 |
| 2015 August | 18 | 10 | 28 |
| 2015 July | 160 | 32 | 192 |
| 2015 June | 82 | 29 | 111 |
| 2015 May | 103 | 37 | 140 |
| 2015 April | 82 | 18 | 100 |
| 2015 March | 51 | 26 | 77 |
| 2015 February | 32 | 18 | 50 |
| 2015 January | 38 | 19 | 57 |
| 2014 December | 47 | 18 | 65 |
| 2014 November | 44 | 17 | 61 |
| 2014 October | 49 | 25 | 74 |
| 2014 September | 51 | 14 | 65 |
| 2014 August | 61 | 20 | 81 |
| 2014 July | 61 | 17 | 78 |
| 2014 June | 77 | 16 | 93 |
| 2014 May | 59 | 18 | 77 |
| 2014 April | 76 | 10 | 86 |
| 2014 March | 76 | 29 | 105 |
| 2014 February | 17 | 13 | 30 |
| 2014 January | 22 | 7 | 29 |
| 2013 December | 24 | 9 | 33 |
| 2013 November | 20 | 10 | 30 |
| 2013 October | 18 | 8 | 26 |
| 2013 September | 12 | 12 | 24 |
| 2013 August | 13 | 16 | 29 |
| 2013 July | 12 | 20 | 32 |
| 2013 June | 10 | 17 | 27 |
| 2013 May | 17 | 17 | 34 |
| 2013 April | 13 | 20 | 33 |
| 2013 March | 19 | 14 | 33 |
| 2013 February | 44 | 14 | 58 |
| 2013 January | 62 | 15 | 77 |
| 2012 December | 25 | 13 | 38 |
| 2012 November | 1 | 1 | 2 |
| 2012 October | 1 | 1 | 2 |
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