A Turkish girl with H syndrome: stunted growth and development of autoimmune insulin dependent diabetes mellitus in the 6th year of diagnosis.
H syndrome
diabetes mellitus
short stature
Journal
Journal of pediatric endocrinology & metabolism : JPEM
ISSN: 2191-0251
Titre abrégé: J Pediatr Endocrinol Metab
Pays: Germany
ID NLM: 9508900
Informations de publication
Date de publication:
28 Jan 2019
28 Jan 2019
Historique:
received:
28
08
2018
accepted:
11
11
2018
pubmed:
6
12
2018
medline:
10
7
2019
entrez:
6
12
2018
Statut:
ppublish
Résumé
Background H syndrome ([OMIM] 602782) is an autosomal recessive disorder with systemic manifestations and characteristic skin lesions, caused by mutations of the SLC29A3 gene. Short stature and diabetes mellitus are the major endocrine problems related to H syndrome, however, clear data from clinical follow-up of H syndrome patients is lacking in the literature. Case presentation Here, we present follow-up of a Turkish girl diagnosed with H syndrome at the age of 10 with a homozygous 310(c.933T>A, p.C310X) early stop codon mutation on exon 6 of the SLC29A3 gene. She had severe short stature non-responsive to growth hormone (GH) treatment and gluten-free diet despite low GH levels and celiac antibody positivity. She developed insulin dependent diabetes mellitus (IDDM) symptoms 6 years after the initial diagnosis. Conclusions H syndrome patients can develop IDDM years after characteristic symptoms. Short stature in H syndrome patients may not respond to GH replacement or gluten-free diet alone.
Identifiants
pubmed: 30517079
doi: 10.1515/jpem-2018-0380
pii: /j/jpem.ahead-of-print/jpem-2018-0380/jpem-2018-0380.xml
doi:
pii:
Substances chimiques
Nucleoside Transport Proteins
0
SLC29A3 protein, human
0
Types de publication
Case Reports
Langues
eng
Sous-ensembles de citation
IM