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Intestinal epithelial dysplasia

MedGen UID:
897512
Concept ID:
C4275062
Disease or Syndrome
Synonyms: Congenital epithelial dysplasia of intestine; Congenital tufting enteropathy; Tufting enteropathy
SNOMED CT: Congenital tufting enteropathy (715669000); Tufting enteropathy (715669000); Congenital epithelial dysplasia of intestine (715669000); Intestinal epithelial dysplasia (715669000)

Definition

A congenital enteropathy presenting with early-onset severe intractable diarrhoea sometimes causing irreversible intestinal failure. Infants develop a watery diarrhoea within the first days after birth and the diarrhoea persists in spite of bowel rest and parenteral nutrition. Some infants are reported to have associated choanal, rectal or oesophageal atresia. Autosomal recessive transmission but the causative gene has not been yet identified. [from SNOMEDCT_US]

Professional guidelines

PubMed

Wu Z, Li Y, Zhang Y, Hu H, Wu T, Liu S, Chen W, Xie S, Lu Z
Technol Cancer Res Treat 2020 Jan-Dec;19:1533033820980426. doi: 10.1177/1533033820980426. PMID: 33353503Free PMC Article
Pimentel-Nunes P, Libânio D, Marcos-Pinto R, Areia M, Leja M, Esposito G, Garrido M, Kikuste I, Megraud F, Matysiak-Budnik T, Annibale B, Dumonceau JM, Barros R, Fléjou JF, Carneiro F, van Hooft JE, Kuipers EJ, Dinis-Ribeiro M
Endoscopy 2019 Apr;51(4):365-388. Epub 2019 Mar 6 doi: 10.1055/a-0859-1883. PMID: 30841008
Basturk O, Hong SM, Wood LD, Adsay NV, Albores-Saavedra J, Biankin AV, Brosens LA, Fukushima N, Goggins M, Hruban RH, Kato Y, Klimstra DS, Klöppel G, Krasinskas A, Longnecker DS, Matthaei H, Offerhaus GJ, Shimizu M, Takaori K, Terris B, Yachida S, Esposito I, Furukawa T; Baltimore Consensus Meeting
Am J Surg Pathol 2015 Dec;39(12):1730-41. doi: 10.1097/PAS.0000000000000533. PMID: 26559377Free PMC Article

Recent clinical studies

Etiology

Cuche C, Mastrogiovanni M, Juzans M, Laude H, Ungeheuer MN, Krentzel D, Gariboldi MI, Scott-Algara D, Madec M, Goyard S, Floch C, Chauveau-Le Friec G, Lafaye P, Renaudat C, Le Bidan M, Micallef C, Schmutz S, Mella S, Novault S, Hasan M, Duffy D, Di Bartolo V, Alcover A
Front Immunol 2023;14:1163466. Epub 2023 Jul 18 doi: 10.3389/fimmu.2023.1163466. PMID: 37533857Free PMC Article
Ashworth I, Wilson A, Aquilina S, Parascandalo R, Mercieca V, Gerada J, Macdonald S, Simchowitz V, Hill S
J Pediatr Gastroenterol Nutr 2018 Jun;66(6):967-971. doi: 10.1097/MPG.0000000000001894. PMID: 29334565
Martin BA, Kerner JA, Hazard FK, Longacre TA
Am J Surg Pathol 2014 Oct;38(10):1387-95. doi: 10.1097/PAS.0000000000000314. PMID: 25188866
Goulet O, Salomon J, Ruemmele F, de Serres NP, Brousse N
Orphanet J Rare Dis 2007 Apr 20;2:20. doi: 10.1186/1750-1172-2-20. PMID: 17448233Free PMC Article
Ruemmele FM
Nestle Nutr Workshop Ser Pediatr Program 2007;59:73-85; discussion 85-8. doi: 10.1159/000098514. PMID: 17245092

Diagnosis

Martin BA, Kerner JA, Hazard FK, Longacre TA
Am J Surg Pathol 2014 Oct;38(10):1387-95. doi: 10.1097/PAS.0000000000000314. PMID: 25188866
Goulet O, Salomon J, Ruemmele F, de Serres NP, Brousse N
Orphanet J Rare Dis 2007 Apr 20;2:20. doi: 10.1186/1750-1172-2-20. PMID: 17448233Free PMC Article
El-Matary W, Dalzell AM, Kokai G, Davidson JE
Eur J Pediatr 2007 Mar;166(3):265-8. Epub 2006 Aug 10 doi: 10.1007/s00431-006-0231-z. PMID: 16900309

Prognosis

Ashworth I, Wilson A, Aquilina S, Parascandalo R, Mercieca V, Gerada J, Macdonald S, Simchowitz V, Hill S
J Pediatr Gastroenterol Nutr 2018 Jun;66(6):967-971. doi: 10.1097/MPG.0000000000001894. PMID: 29334565
Martin BA, Kerner JA, Hazard FK, Longacre TA
Am J Surg Pathol 2014 Oct;38(10):1387-95. doi: 10.1097/PAS.0000000000000314. PMID: 25188866

Clinical prediction guides

Martin BA, Kerner JA, Hazard FK, Longacre TA
Am J Surg Pathol 2014 Oct;38(10):1387-95. doi: 10.1097/PAS.0000000000000314. PMID: 25188866

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