Literature DB >> 2801777

Spinocerebellar ataxia, hypogonadotropic hypogonadism, and choroidal dystrophy (Boucher-Neuhäuser syndrome)

E R Limber1, G H Bresnick, R M Lebovitz, R E Appen, E F Gilbert-Barness, R M Pauli.   

Abstract

We describe two families (including one previously reported) in which cerebellar or spinocerebellar ataxia, hypogonadotropic hypogonadism, and choroidal dystrophy result from abnormal function of an autosomal recessive gene. Review of the literature adds one other family with this disorder. These three examples confirm the existence of this traid as a specific, pleiotropic, single-gene syndrome. Careful ophthalmologic evaluation of patients with ataxia and hypogonadotropic hypogonadism may identify additional cases.

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Year:  1989        PMID: 2801777     DOI: 10.1002/ajmg.1320330325

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  7 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.  PNPLA6 mutations cause Boucher-Neuhauser and Gordon Holmes syndromes as part of a broad neurodegenerative spectrum.

Authors:  Matthis Synofzik; Michael A Gonzalez; Charles Marques Lourenco; Marie Coutelier; Tobias B Haack; Adriana Rebelo; Didier Hannequin; Tim M Strom; Holger Prokisch; Christoph Kernstock; Alexandra Durr; Ludger Schöls; Marcos M Lima-Martínez; Amjad Farooq; Rebecca Schüle; Giovanni Stevanin; Wilson Marques; Stephan Züchner
Journal:  Brain       Date:  2013-12-19       Impact factor: 13.501

Review 4.  Boucher-Neuhäuser syndrome: cerebellar degeneration, chorioretinal dystrophy and hypogonadotropic hypogonadism: two novel cases and a review of 40 cases from the literature.

Authors:  A A Tarnutzer; C Gerth-Kahlert; D Timmann; D I Chang; F Harmuth; P Bauer; D Straumann; M Synofzik
Journal:  J Neurol       Date:  2014-10-31       Impact factor: 4.849

5.  Multiple pituitary hormone deficiencies in a patient with spinocerebellar ataxia: magnetic resonance imaging and hormonal studies.

Authors:  E Bhatia; R Shukla; R K Gupta; U K Misra
Journal:  J Endocrinol Invest       Date:  1993-09       Impact factor: 4.256

6.  Cerebellar ataxia, hypogonadism and chorioretinopathy: molecular analysis of an Italian family.

Authors:  R Rizzi; V Carelli; L Monari; M Mochi; R Liguori; M Sensi; S Cocozza; A Filla; P Montagna
Journal:  Ital J Neurol Sci       Date:  1998-02

7.  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
  7 in total

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