A Turkish girl with H syndrome: stunted growth and development of autoimmune insulin dependent diabetes mellitus in the 6th year of diagnosis.


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
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

Pagination

89-93

Auteurs

Can Ozlu (C)

Koc University School of Medicine, Istanbul, Turkey.

Gul Yesiltepe Mutlu (G)

Koc University School of Medicine, Istanbul 34010, Turkey.
Department of Pediatric Endocrinology, Koc University Hospital, Davutpasa Street No 4, Zeytinburnu 34010, Turkey, Phone: +905057235725.

Sukru Hatun (S)

Koc University School of Medicine, Istanbul, Turkey.
Department of Pediatric Endocrinology, Koc University Hospital, Zeytinburnu, Turkey.

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Classifications MeSH