Literature DB >> 21910247

New case of Primrose syndrome with mild intellectual disability.

Renata Posmyk1, Ryszard Leśniewicz, Monika Chorąży, Sławomir Wołczyński.   

Abstract

We report on a 27-year-old man, who represents the sixth and the youngest published case of Primrose syndrome. Primrose syndrome (PS) (OMIM#295090) is an extremely rare entity of unknown etiology characterized by the progressive wasting of distal muscles of the legs, the small muscles of the hands resulting in contractures, the presence of intellectual disability, hearing problems, cataracts, brain calcification, and the ossification of ear cartilage. All the main manifestations were present in our patient. Despite the phenotypic similarity to five other cases, our patient had mild intellectual disability. Additionally we found hypergonadotropic hypogonadism and a low bone density due to progressive osteoporosis. We discuss our observations in relation to previously published cases, and we stress the need for the detail and phenotypic descriptions of further cases as PS remains rare, and the genetic basis is still undiscovered.
Copyright © 2011 Wiley Periodicals, Inc.

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Year:  2011        PMID: 21910247     DOI: 10.1002/ajmg.a.34257

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


  2 in total

1.  Mutations in ZBTB20 cause Primrose syndrome.

Authors:  Viviana Cordeddu; Bert Redeker; Emilia Stellacci; Aldo Jongejan; Alessandra Fragale; Ted E J Bradley; Massimiliano Anselmi; Andrea Ciolfi; Serena Cecchetti; Valentina Muto; Laura Bernardini; Meron Azage; Daniel R Carvalho; Alberto J Espay; Alison Male; Anna-Maja Molin; Renata Posmyk; Carla Battisti; Alberto Casertano; Daniela Melis; Antoine van Kampen; Frank Baas; Marcel M Mannens; Gianfranco Bocchinfuso; Lorenzo Stella; Marco Tartaglia; Raoul C Hennekam
Journal:  Nat Genet       Date:  2014-07-13       Impact factor: 38.330

2.  Primrose syndrome: Characterization of the phenotype in 42 patients.

Authors:  Daniela Melis; Daniel Carvalho; Tina Barbaro-Dieber; Alberto J Espay; Michael J Gambello; Blanca Gener; Erica Gerkes; Marrit M Hitzert; Hanne B Hove; Sandra Jansen; Petr E Jira; Katherine Lachlan; Leonie A Menke; Vinodh Narayanan; Damara Ortiz; Eline Overwater; Renata Posmyk; Keri Ramsey; Alessandro Rossi; Renata Lazari Sandoval; Constance Stumpel; Kyra E Stuurman; Viviana Cordeddu; Peter Turnpenny; Pietro Strisciuglio; Marco Tartaglia; Sheela Unger; Todd Waters; Clare Turnbull; Raoul C Hennekam
Journal:  Clin Genet       Date:  2020-04-20       Impact factor: 4.438

  2 in total

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