Literature DB >> 30517079

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

Can Ozlu1, Gul Yesiltepe Mutlu2,3, Sukru Hatun1,4.   

Abstract

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.

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Keywords:  H syndrome; diabetes mellitus; short stature

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Year:  2019        PMID: 30517079     DOI: 10.1515/jpem-2018-0380

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  1 in total

Review 1.  Atypical comorbidities in a child considered to have type 1 diabetes led to the diagnosis of SLC29A3 spectrum disorder.

Authors:  Özge Besci; Kashyap Amratlal Patel; Gizem Yıldız; Özlem Tüfekçi; Kübra Yüksek Acinikli; İbrahim Mert Erbaş; Ayhan Abacı; Ece Böber; Meral Torun Bayram; Şebnem Yılmaz; Korcan Demir
Journal:  Hormones (Athens)       Date:  2022-03-14       Impact factor: 3.419

  1 in total

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