Literature DB >> 10745040

A locus for primary ciliary dyskinesia maps to chromosome 19q.

M Meeks1, A Walne, S Spiden, H Simpson, H Mussaffi-Georgy, H D Hamam, E L Fehaid, M Cheehab, M Al-Dabbagh, S Polak-Charcon, H Blau, A O'Rawe, H M Mitchison, R M Gardiner, E Chung.   

Abstract

Primary ciliary dyskinesia is an autosomal recessive condition characterised by chronic sinusitis, bronchiectasis, and subfertility. Situs inversus occurs in 50% of cases (Kartagener syndrome). It has an estimated incidence of 1 in 20 000 live births. The clinical phenotype is caused by defective ciliary function associated with a range of ultrastructural abnormalities including absent dynein arms, absent radial spokes, and disturbed ciliary orientation. The molecular genetic basis is unknown. A genome scan was performed in five Arabic families. Using GENEHUNTER, a maximal multipoint lod score (HLOD) of 4.4 was obtained on chromosome 19q13.3-qter at alpha (proportion of linked families) = 0.7. A 15 cM critical region is defined by recombinations at D19S572 and D19S218. These data provide significant evidence for a PCD locus on chromosome 19q and confirm locus heterogeneity.

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Year:  2000        PMID: 10745040      PMCID: PMC1734555          DOI: 10.1136/jmg.37.4.241

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


  25 in total

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Journal:  Trends Genet       Date:  1995-10       Impact factor: 11.639

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Authors:  M A Sleigh
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Authors:  B A Afzelius
Journal:  Am J Hum Genet       Date:  1981-11       Impact factor: 11.025

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Authors:  M Gebbia; G B Ferrero; G Pilia; M T Bassi; A Aylsworth; M Penman-Splitt; L M Bird; J S Bamforth; J Burn; D Schlessinger; D L Nelson; B Casey
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  15 in total

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Authors:  Saeko Takada; Curtis G Wilkerson; Ken-ichi Wakabayashi; Ritsu Kamiya; George B Witman
Journal:  Mol Biol Cell       Date:  2002-03       Impact factor: 4.138

2.  Linkage analysis localises a Kartagener syndrome gene to a 3.5 cM region on chromosome 15q24-25.

Authors:  M Geremek; E Zietkiewicz; S R Diehl; B Z Alizadeh; C Wijmenga; M Witt
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3.  The human dynein intermediate chain 2 gene (DNAI2): cloning, mapping, expression pattern, and evaluation as a candidate for primary ciliary dyskinesia.

Authors:  G Pennarun; C Chapelin; E Escudier; A M Bridoux; F Dastot; V Cacheux; M Goossens; S Amselem; B Duriez
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4.  Radial spoke proteins of Chlamydomonas flagella.

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Journal:  J Cell Sci       Date:  2006-02-28       Impact factor: 5.285

5.  Identification of predicted human outer dynein arm genes: candidates for primary ciliary dyskinesia genes.

Authors:  G J Pazour; N Agrin; B L Walker; G B Witman
Journal:  J Med Genet       Date:  2005-06-03       Impact factor: 6.318

6.  Gene expression studies in cells from primary ciliary dyskinesia patients identify 208 potential ciliary genes.

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7.  Split cord malformation and situs inversus totalis: case report and review of the literature.

Authors:  R Shane Tubbs; John C Wellons; W Jerry Oakes
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8.  Split cord malformation and situs inversus totalis: case report and review of the literature.

Authors:  R Shane Tubbs; John C Wellons; W Jerry Oakes
Journal:  Childs Nerv Syst       Date:  2003-11-26       Impact factor: 1.475

9.  Carrier status for 3 most frequent CFTR mutations in Polish PCD/KS patients: lack of association with the primary ciliary dyskinesia phenotype.

Authors:  Urszula Skrzypczak; Ewa Rutkiewicz; Andrzej Pogorzelski; Michał Witt; Ewa Zietkiewicz
Journal:  J Appl Genet       Date:  2007       Impact factor: 2.653

10.  Mutations in radial spoke head protein genes RSPH9 and RSPH4A cause primary ciliary dyskinesia with central-microtubular-pair abnormalities.

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Journal:  Am J Hum Genet       Date:  2009-02-05       Impact factor: 11.025

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