Literature DB >> 9733035

Spastic paraplegia, optic atrophy, microcephaly with normal intelligence, and XY sex reversal: a new autosomal recessive syndrome?

A S Teebi1, S Miller, H Ostrer, P Eydoux, C Colomb-Brockmann, K Oudjhane, G Watters.   

Abstract

Two female sibs of first cousin Iranian parents were found to have the syndrome of spastic paraplegia, optic atrophy with poor vision, microcephaly, and normal cognitive development. Karyotype analysis showed a normal female constitution in one and a male constitution (46,XY) in the other. The XY female showed normal female external genitalia, normal uterus and tubes, and streak gonads. SRY gene sequencing was normal. We conclude that the present family probably represents a new autosomal recessive trait of pleiotropic effects including XY sex reversal and adds further evidence for the heterogeneity of spastic paraplegia syndromes as well as sex reversal syndromes.

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Year:  1998        PMID: 9733035      PMCID: PMC1051429          DOI: 10.1136/jmg.35.9.759

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  42 in total

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Journal:  Clin Genet       Date:  1988-07       Impact factor: 4.438

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Authors:  A E Harding
Journal:  Lancet       Date:  1983-05-21       Impact factor: 79.321

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Authors:  H S Baar; A M Gabriel
Journal:  Am J Ment Defic       Date:  1966-07

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Authors:  A Abdallat; S M Davis; J Farrage; W I McDonald
Journal:  J Neurol Neurosurg Psychiatry       Date:  1980-11       Impact factor: 10.154

8.  Hereditary "pure" spastic paraplegia: a clinical and genetic study of 22 families.

Authors:  A E Harding
Journal:  J Neurol Neurosurg Psychiatry       Date:  1981-10       Impact factor: 10.154

9.  A dosage sensitive locus at chromosome Xp21 is involved in male to female sex reversal.

Authors:  B Bardoni; E Zanaria; S Guioli; G Floridia; K C Worley; G Tonini; E Ferrante; G Chiumello; E R McCabe; M Fraccaro
Journal:  Nat Genet       Date:  1994-08       Impact factor: 38.330

10.  A gene mapping to the sex-determining region of the mouse Y chromosome is a member of a novel family of embryonically expressed genes.

Authors:  J Gubbay; J Collignon; P Koopman; B Capel; A Economou; A Münsterberg; N Vivian; P Goodfellow; R Lovell-Badge
Journal:  Nature       Date:  1990-07-19       Impact factor: 49.962

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  1 in total

1.  Clinical Spectrum of Hereditary Spastic Paraplegia in Children: A study of 74 cases.

Authors:  Roshan Koul; Fathiya M Al-Murshedi; Faisal M Al-Azri; Ranjit Mani; Rana A Abdelrahim; Vivek Koul; Amna M Alfutaisi
Journal:  Sultan Qaboos Univ Med J       Date:  2013-06-25
  1 in total

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