17p13.1 Microduplication Syndrome in a Child, Familial Short Stature, and Growth Hormone Deficiency: A Case Report and Review of the Literature.

Comparative genomic analysis Familial short stature Growth hormone deficiency Microduplication syndrome 17p13.1

Journal

Molecular syndromology
ISSN: 1661-8769
Titre abrégé: Mol Syndromol
Pays: Switzerland
ID NLM: 101525192

Informations de publication

Date de publication:
Jan 2019
Historique:
accepted: 02 09 2018
entrez: 26 2 2019
pubmed: 26 2 2019
medline: 26 2 2019
Statut: ppublish

Résumé

To date, 6 cases of 17p13.1 microduplications have been described in the literature. Intellectual disability is the core feature, together with minor facial dysmorphisms and obesity. We describe the first case of a young patient with a maternally inherited microduplication in 17p13.1 presenting with growth hormone deficiency. The boy was addressed to the endocrine division for growth retardation (weight and height <3rd percentile). Besides minor facial dysmorphisms, physical and neurological examinations were normal except for motor dyspraxia. Basic blood tests and endocrinological investigations were normal, but IGF1 levels were low for his age. Growth hormone deficiency was confirmed. Hypothalamic pituitary MRI was normal. His karyotype was 46XY. Array-CGH analysis detected a 422-kb copy number gain in the spanning region 17p13.1 inherited from his mother. Although familial short stature is considered a "normal" variation of growth retardation, hormonal and genetic investigation is essential in the etiological diagnosis.

Identifiants

pubmed: 30800046
doi: 10.1159/000494681
pii: msy-0009-0300
pmc: PMC6381889
doi:

Types de publication

Journal Article

Langues

eng

Pagination

300-305

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Auteurs

Sofia Leka-Emiri (S)

Department of Endocrinology, Growth and Development, Athens, Greece.

Vassilios Petrou (V)

Department of Endocrinology, Growth and Development, Athens, Greece.

Emmanouil Manolakos (E)

Department of Access to Genome (ATG P.C.), Clinical Laboratory Genetics, Athens, Greece.

Loretta Thomaidis (L)

Department of Developmental Assessment Unit, Department of Pediatrics, P&A Kyriakou Children's Hospital, School of Medicine, National and Kapodistrian University of Athens, Athens, Greece.

Aspasia Fotinou (A)

Department of Biochemistry and Hormonology, Athens, Greece.

Elpis Vlachopapadopoulou (E)

Department of Endocrinology, Growth and Development, Athens, Greece.

Stefanos Michalacos (S)

Department of Endocrinology, Growth and Development, Athens, Greece.

Classifications MeSH