New Findings of Immunodysregulation, Polyendocrinopathy, and Enteropathy X-linked Syndrome (IPEX); Granulomas in Lung and Duodenum.
Colitis
/ genetics
Diabetes Mellitus, Type 1
/ congenital
Diarrhea
/ genetics
Duodenum
/ pathology
Forkhead Transcription Factors
/ genetics
Genetic Diseases, X-Linked
/ genetics
Granuloma
/ genetics
Granuloma, Respiratory Tract
/ genetics
Humans
Immune System Diseases
/ congenital
Infant, Newborn
Male
Mutation
Crohn disease
IPEX syndrome
VEOIBD
colitis
granuloma
ulcerative colitis
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