Literature DB >> 15326626

Spondylometaphyseal dysplasia with cone-rod dystrophy.

Brent A Walters1, Michael L Raff, James Ver Hoeve, Rachel Tesser, Leonard O Langer, Thomas D France, Ian A Glass, Richard M Pauli.   

Abstract

The co-occurrence of ophthalmologic abnormality and intrinsic skeletal dysplasia is uncommon. We describe eight instances of a unique form of spondylometaphyseal dysplasia (SMD) associated with cone-rod dystrophy (although documentation is insufficient to be certain of that diagnosis in some). This is a new, syndromic form of SMD for which there is evidence for autosomal recessive transmission. Recognition of the specific bony features described here should precipitate comprehensive ophthalmologic assessment, since vision impairment becomes significantly disabling with age.

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Year:  2004        PMID: 15326626     DOI: 10.1002/ajmg.a.30145

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  5 in total

1.  Mutations in PCYT1A cause spondylometaphyseal dysplasia with cone-rod dystrophy.

Authors:  Guilherme L Yamamoto; Wagner A R Baratela; Tatiana F Almeida; Monize Lazar; Clara L Afonso; Maria K Oyamada; Lisa Suzuki; Luiz A N Oliveira; Ester S Ramos; Chong A Kim; Maria Rita Passos-Bueno; Débora R Bertola
Journal:  Am J Hum Genet       Date:  2014-01-02       Impact factor: 11.025

2.  Mutations in the PCYT1A gene are responsible for isolated forms of retinal dystrophy.

Authors:  Francesco Testa; Mariaelena Filippelli; Raffaella Brunetti-Pierri; Giuseppina Di Fruscio; Valentina Di Iorio; Mariateresa Pizzo; Annalaura Torella; Maria Rosaria Barillari; Vincenzo Nigro; Nicola Brunetti-Pierri; Francesca Simonelli; Sandro Banfi
Journal:  Eur J Hum Genet       Date:  2017-03-08       Impact factor: 4.246

3.  Mutations in PCYT1A, encoding a key regulator of phosphatidylcholine metabolism, cause spondylometaphyseal dysplasia with cone-rod dystrophy.

Authors:  Julie Hoover-Fong; Nara Sobreira; Julie Jurgens; Peggy Modaff; Carrie Blout; Ann Moser; Ok-Hwa Kim; Tae-Joon Cho; Sung Yoon Cho; Sang Jin Kim; Dong-Kyu Jin; Hiroshi Kitoh; Woong-Yang Park; Hua Ling; Kurt N Hetrick; Kimberly F Doheny; David Valle; Richard M Pauli
Journal:  Am J Hum Genet       Date:  2014-01-02       Impact factor: 11.025

4.  A new form or a variant of SMD type A4.

Authors:  Ivo Marik; Olga Hudakova; Sarka Petrasova; Lukasz Kuszel; Malwina Czarny-Ratajczak; Kazimierz Kozlowski
Journal:  J Appl Genet       Date:  2012-04-24       Impact factor: 3.240

Review 5.  Cone rod dystrophies.

Authors:  Christian P Hamel
Journal:  Orphanet J Rare Dis       Date:  2007-02-01       Impact factor: 4.123

  5 in total

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