Literature DB >> 6891556

X-linked congenital adrenal hypoplasia. A study of five generations of a Greenlandic Family.

K E Petersen, T Bille, B B Jacobsen, T Iversen.   

Abstract

In a Greenlandic family comprising 171 descendants in 5 generations, 11 boys had died with a clinical picture of adrenocortical insufficiency within three weeks of birth. Three treated male patients, who survived, were closely studied. The adrenal glands could not be identified by computed tomography. The clinical picture and biochemical studies were consistent with congenital adrenal hypoplasia. The pedigree indicates X-linked recessive inheritance. The pubertal development was delayed in two patients aged 14 years. LHRH stimulation yielded a significant rise in LH and FSH in at least one of the patients, adrenal androgen values were very low. A family instruction programme has been set up.

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Year:  1982        PMID: 6891556     DOI: 10.1111/j.1651-2227.1982.tb09554.x

Source DB:  PubMed          Journal:  Acta Paediatr Scand        ISSN: 0001-656X


  5 in total

Review 1.  Hypogonadotropic hypogonadism in subjects with DAX1 mutations.

Authors:  Unmesh Jadhav; Rebecca M Harris; J Larry Jameson
Journal:  Mol Cell Endocrinol       Date:  2011-06-13       Impact factor: 4.102

2.  Adrenal hypoplasia congenita with hypogonadotropic hypogonadism: evidence that DAX-1 mutations lead to combined hypothalmic and pituitary defects in gonadotropin production.

Authors:  R L Habiby; P Boepple; L Nachtigall; P M Sluss; W F Crowley; J L Jameson
Journal:  J Clin Invest       Date:  1996-08-15       Impact factor: 14.808

3.  Progressive high frequency hearing loss: an additional feature in the syndrome of congenital adrenal hypoplasia and gonadotrophin deficiency.

Authors:  M Zachmann; E Fuchs; A Prader
Journal:  Eur J Pediatr       Date:  1992-03       Impact factor: 3.183

4.  NR0B1 (DAX1) mutations in patients affected by congenital adrenal hypoplasia with growth hormone deficiency as a new finding.

Authors:  Aleksandra Rojek; Monika Obara-Moszynska; Elzbieta Malecka; Malgorzata Slomko-Jozwiak; Marek Niedziela
Journal:  J Appl Genet       Date:  2013-02-02       Impact factor: 3.240

5.  Congenital adrenal hypoplasia, myopathy, and glycerol kinase deficiency: molecular genetic evidence for deletions.

Authors:  U Francke; J F Harper; B T Darras; J M Cowan; E R McCabe; A Kohlschütter; W K Seltzer; F Saito; J Goto; J P Harpey
Journal:  Am J Hum Genet       Date:  1987-03       Impact factor: 11.025

  5 in total

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