Acrodermatitis enteropathica during parenteral nutrition: a pediatric case report
Keywords:
Acrodermatitis enteropathica, pediatric parenteral nutrition, Zinc deficiencyAbstract
Background and aim Acrodermatitis enteropathica is a rare disorder characterized by the triad composed by dermatitis, alopecia and diarrhoea. Its acquired form can be caused by inadequate zinc intake, malabsorptive processes, excessive renal or intestinal loss. A rare cause of acquired zinc deficiency is iatrogenic nutritional deficiency due to parenteral nutrition. The diagnosis can be really difficult because the early clinical signs are non-specific and patient’s eventual comorbidities can often mask symptoms.
Methods: A 5-years-old child affected by several comorbidities, consequent to C. Koseri meningo-encephalitis occurred in the neonatal period, was admitted to Pediatric ward for acute pancreatitis and had been fed via total parenteral nutrition for one month. Symptoms started approximately 15 days after the start of a standardized parenteral nutrition mixture. The child presented with diarrhoea, alopecia and erythematous bullous skin lesions, distributed predominantly in acral and periorificial sites and not responsive to topical treatments. Zinc serum dosage were very low (10 µg/dL, with normal values 68-107 µg/dL). Clinical improvement was very fast after oral zinc supplementation (5mg/daily), with a rapid regularisation in the intestinal habits and re‐epithelialization of the skin lesions.
Results and Conclusions: Trace elements are an essential component of parenteral nutrition. The supplementation of trace elements is an important part of the parenteral nutrition prescription. Even few days of zinc shortage, especially in frail patients, may cause a severe dermatitis that can be easily prevented. Despite its rarity, acrodermatitis enteropathica should be strongly considered in the differential diagnosis of skin lesions for these patients.
References
Jagadeesan S, Kaliyadan F. Acrodermatitis Enteropathica 2022 Jul 17. In: StatPearls (Internet). Treasure Island (FL): StatPearls Publishing; 2022 Jan–..
Ciampo IRLD, Sawamura R, Ciampo LAD, Fernandes MIM. Acrodermatitis enteropathica: clinical manifestations and pediatric diagnosis Rev Paul Pediatr 2018 Apr-Jun;36(2):238-241. doi: 10.1590/1984-0462/;2018;36;2;00010.
Schmitt S, Küry S, Giraud M, Dréno B, Kharfi M, Bézieau S. An update on mutations of the SLC39A4 gene in acrodermatitis enteropathica. Hum Mutat 2009 Jun;30(6):926-33. doi: 10.1002/humu.20988.
Wells BT, winkelmann RK. Acrodermatitis enteropathica. Report of 6 cases. Arch Dermatol 1961 Jul;84:40-52. doi: 10.1001/archderm.1961.01580130046007.
Jensen SL, McCuaig C, Zembowicz A, Hurt MA. Bullous lesions in acrodermatitis enteropathica delaying diagnosis of zinc deficiency: a report of two cases and review of the literature. J Cutan Pathol 2008 Oct;35 Suppl 1:1-13. doi: 10.1111/j.1600-0560.2008.00981.x. Epub 2008 Jun 4.
Simmer K, Rakshasbhuvankar A, Deshpande G. Standardised parenteral nutrition. Nutrients 2013 Mar 28;5(4):1058-70. doi: 10.3390/nu5041058.
Riskin A, Picaud JC, Shamir R; ESPGHAN/ESPEN/ESPR/CSPEN working group on pediatric parenteral nutrition. ESPGHAN/ESPEN/ESPR/CSPEN guidelines on pediatric parenteral nutrition: Standard versus individualized parenteral nutrition. Clin Nutr 2018 Dec;37(6 Pt B):2409-2417. doi: 10.1016/j.clnu.2018.06.955. Epub 2018 Jun 18.
Domellöf M, Szitanyi P, Simchowitz V, Franz A, Mimouni F; ESPGHAN/ESPEN/ESPR/CSPEN working group on pediatric parenteral nutrition. ESPGHAN/ESPEN/ESPR/CSPEN guidelines on pediatric parenteral nutrition: Iron and trace minerals. Clin Nutr 2018 Dec;37(6 Pt B):2354-2359. doi: 10.1016/j.clnu.2018.06.949. Epub 2018 Jun 18.
XChen X, Jiang Y, Wang Z, et al. Alteration in Gut Microbiota Associated with Zinc Deficiency in School-Age Children. Nutrients 2022 Jul 14;14(14):2895. doi: 10.3390/nu14142895.
Roohani N, Hurrell R, Kelishadi R, Schulin R. Zinc and its importance for human health: An integrative review. J Res Med Sci 2013 Feb;18(2):144-57.
Salgueiro MJ, Zubillaga MB, Lysionek AE, Caro RA, Weill R, Boccio JR. The role of zinc in the growth and development of children. Nutrition 2002 Jun;18(6):510-9. doi: 10.1016/s0899-9007(01)00812-7.
M. Jackson, Zinc in Human Biology - Chapter 1 Physiology of Zinc: General Aspect, Springer-Verlag Berlin Heidelberg GmbH, pp. 1-14, 1989.
Ogawa Y, Kinoshita M, Shimada S, Kawamura T. Zinc and Skin Disorders. Nutrients 2018 Feb 11;10(2):199. doi: 10.3390/nu10020199.
Cleminson K, Hull PR, Price E, Green PJ. Acute onset of blisters in an infant with acrodermatitis enteropathica: A case report. SAGE Open Med Case Rep 2021 Apr 8;9:2050313X20984119. doi: 10.1177/2050313X20984119.
Bin BH, Hojyo S, Seo J, et al. The Role of the Slc39a Family of Zinc Transporters in Zinc Homeostasis in Skin. Nutrients 2018 Feb 16;10(2):219. doi: 10.3390/nu10020219.
Wiznia LE, Bhansali S, Brinster N, Al-Qaqaa YM, Orlow SJ, Oza V. Acquired acrodermatitis enteropathica due to zinc-depleted parenteral nutrition. Pediatr Dermatol 2019 Jul;36(4):520-523. doi: 10.1111/pde.13865. Epub 2019 May 24.
T. Brandt, «Dermatitis in children with disturbances of the general conditions and the absorption of food elements» Acta Derm Venereol, vol. 17, pp. 513-46, 1936.
Danbolt N. Acrodermatitis enteropathica. Br J Dermatol 1979 Jan;100(1):37-40. doi: 10.1111/j.1365-2133.1979.tb03567.x.
Protheroe S. Long-Term Parenteral Nutrition. 2019 Paediatr Child Helath, Volume 29, Issue 9, Pages 369-37.
Lowe NM. The global challenge of hidden hunger: perspectives from the field. Proc Nutr Soc 2021 Aug;80(3):283-289. doi: 10.1017/S0029665121000902. Epub 2021 Apr 26.
Zemrani B, McCallum Z, Bines JE. Trace Element Provision in Parenteral Nutrition in Children: One Size Does Not Fit All. Nutrients 2018 Nov 21;10(11):1819. doi: 10.3390/nu10111819.
Downloads
Published
Issue
Section
License
Copyright (c) 2023 Francesco Proli, Gaia Margiotta, Serena Ferretti, Alessandra Drosi, Piero Valentini, Danilo Buonsenso, Roberta Onesimo, Valentina Giorgio, Cristina Guerriero, Giuseppe Zampino, Ilaria Lazzareschi
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Transfer of Copyright and Permission to Reproduce Parts of Published Papers.
Authors retain the copyright for their published work. No formal permission will be required to reproduce parts (tables or illustrations) of published papers, provided the source is quoted appropriately and reproduction has no commercial intent. Reproductions with commercial intent will require written permission and payment of royalties.