New Findings of Immunodysregulation, Polyendocrinopathy, and Enteropathy X-linked Syndrome (IPEX); Granulomas in Lung and Duodenum.


Journal

Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society
ISSN: 1615-5742
Titre abrégé: Pediatr Dev Pathol
Pays: United States
ID NLM: 9809673

Informations de publication

Date de publication:
Historique:
pubmed: 9 3 2021
medline: 23 11 2021
entrez: 8 3 2021
Statut: ppublish

Résumé

Immune dysregulation, polyendocrinopathy and enteropathy, X-linked (IPEX) syndrome is a rare disorder caused by loss-of-function mutations in the gene forkhead box protein 3 (FOXP3). IPEX patients frequently show chronic diarrhea (enteropathy) associated with villous atrophies in the small intestine. Our case is different from this classical information in the literature, since he presented with neonatal onset inflammatory bowel disease within the first months of life accompanied by deep ulcers throughout colonic mucosa. Moreover, he developed chronic lung disease during follow-up and histopathological examinations showed granulomas in both gastrointestinal tract and lung parenchyma. Genetic analysis revealed the diagnosis of IPEX syndrome with a germline mutation in FOXP3. Thus, our study provides an unusual presentation of IPEX syndrome with colitis and granulomas presence in histopathological examinations.

Identifiants

pubmed: 33683986
doi: 10.1177/1093526621998868
doi:

Substances chimiques

FOXP3 protein, human 0
Forkhead Transcription Factors 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

252-257

Auteurs

Demet Teker Duztas (DT)

Department of Pediatric Gastroenterology and Hepatology, Gazi University, Ankara, Turkey.

Lina Al-Shadfan (L)

Department of Pediatric Pulmonology, Bezmialem Vakif University, Istanbul, Turkey.

Hakan Ozturk (H)

Department of Pediatric Gastroenterology and Hepatology, Gazi University, Ankara, Turkey.

Hakan Yazan (H)

Department of Pediatric Pulmonology, Bezmialem Vakif University, Istanbul, Turkey.

Erkan Cakir (E)

Department of Pediatric Pulmonology, Bezmialem Vakif University, Istanbul, Turkey.

Nurcan Unver Ozgur Ekinci (NUO)

Department of Pathology, Gazi University, Ankara, Turkey.

Buket Dalgic (B)

Department of Pediatric Gastroenterology and Hepatology, Gazi University, Ankara, Turkey.

Meino Rohlfs (M)

Department of Pediatrics, Dr. von Hauner Children's Hospital, University Hospital, Ludwig-Maximilians-Universität (LMU), Munich, Germany.

Tim Jeske (T)

Department of Pediatrics, Dr. von Hauner Children's Hospital, University Hospital, Ludwig-Maximilians-Universität (LMU), Munich, Germany.

Christoph Klein (C)

Department of Pediatrics, Dr. von Hauner Children's Hospital, University Hospital, Ludwig-Maximilians-Universität (LMU), Munich, Germany.

Daniel Kotlarz (D)

Department of Pediatrics, Dr. von Hauner Children's Hospital, University Hospital, Ludwig-Maximilians-Universität (LMU), Munich, Germany.

Odul Egritas Gurkan (OE)

Department of Pediatric Gastroenterology and Hepatology, Gazi University, Ankara, Turkey.

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Classifications MeSH