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A novel CHD7 mutation in an adolescent presenting with growth and pubertal delay.
Antoniou, Maria-Christina; Bouthors, Thérèse; Xu, Cheng; Phan-Hug, Franziska; Elowe-Gruau, Eglantine; Stoppa-Vaucher, Sophie; Sloot, Almer van der; Acierno, James; Cassatella, Daniele; Richard, Celine; Dwyer, Andrew; Pitteloud, Nelly; Hauschild, Michael.
Afiliação
  • Antoniou MC; Department of Pediatric Endocrinology and Diabetology, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Bouthors T; Department of Pediatric Endocrinology and Diabetology, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Xu C; Service of Endocrinology, Diabetes and Metabolism, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Phan-Hug F; Department of Pediatric Endocrinology and Diabetology, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Elowe-Gruau E; Department of Pediatric Endocrinology and Diabetology, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Stoppa-Vaucher S; Department of Pediatric Endocrinology and Diabetology, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Sloot AV; Institute for Research in Immunology and Cancer (IRIC), University of Montreal, Montreal, Canada.
  • Acierno J; Service of Endocrinology, Diabetes and Metabolism, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Cassatella D; Service of Endocrinology, Diabetes and Metabolism, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Richard C; Otorhinolaryngology Service, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Dwyer A; Service of Endocrinology, Diabetes and Metabolism, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Pitteloud N; Department of Pediatric Endocrinology and Diabetology, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
  • Hauschild M; Service of Endocrinology, Diabetes and Metabolism, Centre Hospitalier Universitaire Vaudois (CHUV), Lausanne, Switzerland.
Ann Pediatr Endocrinol Metab ; 24(1): 49-54, 2019 Mar.
Article em En | MEDLINE | ID: mdl-30943680
Mutations in the CHD7 gene, encoding for the chromodomain helicase DNA-binding protein 7, are found in approximately 60% of individuals with CHARGE syndrome (coloboma, heart defects, choanal atresia, retarded growth and development, genital hypoplasia, ear abnormalities and/or hearing loss). Herein, we present a clinical case of a 14-year-old male presenting for evaluation of poor growth and pubertal delay highlighting the diagnostic challenges of CHARGE syndrome. The patient was born full term and underwent surgery at 5 days of life for bilateral choanal atresia. Developmental milestones were normally achieved. At age 14 his height and weight were -2.04 and -1.74 standard deviation score respectively. He had anosmia as well as prepubertal testes and micropenis (4 cm×1 cm). The biological profile showed low basal serum testosterone and gonadotropins (testosterone, 0.2 nmol/L; luteinizing hormone, 0.5 U/L; follicle-stimulating hormone, 1.3 U/L), and otherwise normal pituitary function and normal imaging of the hypothalamic-pituitary area. The constellation of choanal atresia, anosmia, mild dysmorphic features, micropenis and delayed puberty were suggestive of CHARGE syndrome. Targeted genetic testing of CHD7 was performed revealing a de novo heterozygous CHD7 mutation (c.4234T>G [p.Tyr1412Asp]). Further paraclinical investigations confirmed CHARGE syndrome. Despite the presence of suggestive features, CHARGE syndrome remained undiagnosed in this patient until adolescence. Genetic testing helps clarify the phenotypic and genotypic spectrum to facilitate diagnosis, thus promoting optimal follow-up, treatment, and appropriate genetic counselling.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Idioma: En Revista: Ann Pediatr Endocrinol Metab Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Suíça

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Idioma: En Revista: Ann Pediatr Endocrinol Metab Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Suíça