Literature DB >> 1251844

A new familial syndrome characterized by pigmentary retinopathy, hypogonadism, mental retardation, nerve deafness and glucose intolerance.

J A Edwards, P K Sethi, A J Scoma, R M Bannerman, L A Frohman.   

Abstract

Three siblings with retinitis pigmentosa, deafness and mental retardation were studied. Physical abnormalities included nystagmus, acanthosis nigricans and multiple keloids. The two male siblings had gynecomastia, small testes and mild subvirilization whereas the only indication of hypogonadism in the female sibling was oligomenorrhea. Testosterone levels in the males, which were in the low to low normal range, were increased by the administration of large doses of chorionic gonadotropin. The two affected males had elevated plasma luteinizing hormone (LH) and follicle-stimulating hormone (FSH) levels which were decreased by the administration of testosterone and increased by the administration of clomiphene. One sibling had mild obesity and diabetes mellitus, one had moderate obesity, normal glucose tolerance and hyperinsulinism and the third had abnormal glucose tolerance and hyperinsulinism. This familial syndrome is distinct from either the Laurence-Moon, Bardet-Biedl or Alström disorders and provides further evidence of genetic heterogeneity in this group of autosomal recessive traits.

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Year:  1976        PMID: 1251844     DOI: 10.1016/0002-9343(76)90529-5

Source DB:  PubMed          Journal:  Am J Med        ISSN: 0002-9343            Impact factor:   4.965


  7 in total

1.  A patient with features of both Bardet-Biedl and Alström syndromes.

Authors:  C Hauser; C Rojas; A Roth; E Schmied; J H Saurat
Journal:  Eur J Pediatr       Date:  1990-08       Impact factor: 3.183

2.  [Alström syndrome--a rare disease of diabetic association].

Authors:  B Weichenhain; J Stemplinger; A G Ziegler; W Rabl; E Standl; H Stiegler
Journal:  Med Klin (Munich)       Date:  1997-03-15

3.  Intrafamilial variation of the phenotype in Bardet-Biedl syndrome.

Authors:  R Riise; S Andréasson; M K Borgaström; A F Wright; N Tommerup; T Rosenberg; K Tornqvist
Journal:  Br J Ophthalmol       Date:  1997-05       Impact factor: 4.638

Review 4.  Hypogonadism and neurological diseases.

Authors:  Abdulaziz Alsemari
Journal:  Neurol Sci       Date:  2013-01-05       Impact factor: 3.307

5.  Waardenburg-like features with cataracts, small head size, joint abnormalities, hypogonadism, and osteosarcoma.

Authors:  D M Parry; A W Safyer; J J Mulvihill
Journal:  J Med Genet       Date:  1978-02       Impact factor: 6.318

6.  The Alström syndrome: ophthalmic histopathology and retinal ultrastructure.

Authors:  J Sebag; D M Albert; J L Craft
Journal:  Br J Ophthalmol       Date:  1984-07       Impact factor: 4.638

7.  Leprechaunism: an inherited defect in a high-affinity insulin receptor.

Authors:  L J Elsas; F Endo; E Strumlauf; J Elders; J H Priest
Journal:  Am J Hum Genet       Date:  1985-01       Impact factor: 11.025

  7 in total

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