Literature DB >> 7639308

Ophthalmologic findings in a patient with cerebellar ataxia, hypogonadotropic hypogonadism, and chorioretinal dystrophy.

F Salvador1, J García-Arumí, B Corcóstegui, T Minoves, F Tarrus.   

Abstract

PURPOSE: To determine the ophthalmologic findings in a 39-year-old woman with the Boucher-Neuhäuser syndrome, which is a disorder of autosomal recessive inheritance characterized by the triad of spinocerebellar ataxia, chorioretinal dystrophy, and hypogonadotropic hypogonadism.
METHODS: The patient underwent clinical and electrophysiologic examinations and fluorescein angiography.
RESULTS: The main clinical findings were extensive areas of retinal pigment epithelial and choriocapillaris atrophy affecting the posterior pole and midperiphery of both eyes. Results of electroretinographic examination showed subnormal photopic and scotopic responses.
CONCLUSIONS: The Boucher-Neuhäuser syndrome should be included in the differential diagnosis of patients with chorioretinal degeneration, particularly if there are neurologic or endocrinologic manifestations.

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Year:  1995        PMID: 7639308     DOI: 10.1016/s0002-9394(14)72612-1

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  8 in total

Review 1.  Two sibs with chorioretinal dystrophy, hypogonadotrophic hypogonadism, and cerebellar ataxia: Boucher-Neuhäuser syndrome.

Authors:  P Rump; B C Hamel; A J Pinckers; P A van Dop
Journal:  J Med Genet       Date:  1997-09       Impact factor: 6.318

2.  Compound heterozygous PNPLA6 mutations cause Boucher-Neuhäuser syndrome with late-onset ataxia.

Authors:  A Deik; B Johannes; J C Rucker; E Sánchez; S E Brodie; E Deegan; K Landy; Y Kajiwara; S Scelsa; R Saunders-Pullman; C Paisán-Ruiz
Journal:  J Neurol       Date:  2014-09-30       Impact factor: 4.849

3.  Two adult females with a distinct familial mental retardation syndrome: non-progressive neurological symptoms with ataxia and hypotonia, similar facial appearance, hypergonadotrophic hypogonadism, and retinal dystrophy.

Authors:  J P Fryns; C Van Lingen; K Devriendt; E Legius; P Raus
Journal:  J Med Genet       Date:  1998-04       Impact factor: 6.318

Review 4.  Effects of gonadotrophin-releasing hormone outside the hypothalamic-pituitary-reproductive axis.

Authors:  D C Skinner; A J Albertson; A Navratil; A Smith; M Mignot; H Talbott; N Scanlan-Blake
Journal:  J Neuroendocrinol       Date:  2009-03       Impact factor: 3.627

5.  The gonadotropin-releasing hormone type I receptor is expressed in the mouse cerebellum.

Authors:  Asher J Albertson; Heather Talbott; Qi Wang; Dane Jensen; Donal C Skinner
Journal:  Cerebellum       Date:  2008       Impact factor: 3.847

6.  Ophthalmologic findings of Boucher-Neuhäuser syndrome.

Authors:  Sun Im Yu; Jung Lim Kim; Sul Gee Lee; Hyun Woong Kim; Sang Jin Kim
Journal:  Korean J Ophthalmol       Date:  2008-12

7.  Novel variants in PNPLA6 causing syndromic retinal dystrophy.

Authors:  Shijing Wu; Zixi Sun; Tian Zhu; Richard G Weleber; Paul Yang; Xing Wei; Mark E Pennesi; Ruifang Sui
Journal:  Exp Eye Res       Date:  2020-10-22       Impact factor: 3.467

8.  CHORIORETINAL CHANGES IN A GENETICALLY CONFIRMED CASE OF BOUCHER-NEUHÄUSER SYNDROME.

Authors:  Brittany B DeNaro; Elona Dhrami-Gavazi; David M Rubaltelli; K Bailey Freund; Winston Lee; Lawrence A Yannuzzi; Stephen H Tsang; Joann J Kang
Journal:  Retin Cases Brief Rep       Date:  2021-03-01
  8 in total

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