Literature DB >> 2208770

Rhizomelic chondrodysplasia punctata and survival beyond one year: a review of the literature and five case reports.

T D Wardinsky1, R A Pagon, B R Powell, B McGillivray, M Stephan, J Zonana, A Moser.   

Abstract

Rhizomelic chondrodysplasia punctata (RCDP), a peroxisomal disorder, is considered to be a lethal neonatal autosomal recessive chondrodysplasia. We report five patients, three of whom survived beyond 1 year, and we summarize the findings in 21 patients from a literature review who survived beyond 1 year. In those patients that survive, there is a high association of spasticity, psychomotor retardation, growth failure, seizures, thermoregulatory instability, feeding difficulty, and recurrent otitis media and pneumonia. Three of our five patients had no radiographic evidence of vertebral body clefts, a finding which has previously been considered invariable in RCDP. Three of our patients had distinctive facies that differ from the classic Conradi-Hunermann facies.

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Year:  1990        PMID: 2208770     DOI: 10.1111/j.1399-0004.1990.tb03554.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

1.  A novel mutation in the PEX12 gene causing a peroxisomal biogenesis disorder.

Authors:  Jana Konkoľová; Robert Petrovič; Ján Chandoga; Edita Halasová; Petra Jungová; Daniel Böhmer
Journal:  Mol Biol Rep       Date:  2015-06-21       Impact factor: 2.316

Review 2.  Punctate epiphyses: a radiological sign not a disease.

Authors:  A K Poznanski
Journal:  Pediatr Radiol       Date:  1994

3.  Ultrastructure and immunocytochemistry of hepatic peroxisomes in rhizomelic chondrodysplasia punctata.

Authors:  J L Hughes; A Poulos; D I Crane; C W Chow; L J Sheffield; D Sillence
Journal:  Eur J Pediatr       Date:  1992-11       Impact factor: 3.183

4.  Chondrodysplasia punctata with a mild clinical course.

Authors:  J M Nuoffer; J P Pfammatter; A Spahr; H Toplak; R J Wanders; R B Schutgens; U N Wiesmann
Journal:  J Inherit Metab Dis       Date:  1994       Impact factor: 4.982

5.  Rhizomelic Chondrodysplasia Punctata Type 1 Caused by a Novel Mutation in the PEX7 Gene.

Authors:  Abdullah Çim; Salih Coşkun; Orhan Görükmez; Hatice Yüksel; Ünal Uluca; Erminia Di Pietro; François Plourde; Nancy Elise Braverman
Journal:  J Clin Res Pediatr Endocrinol       Date:  2015-03

6.  Rhizomelic chondrodysplasia punctata: Role of EEG as a biomarker of impending epilepsy.

Authors:  Debopam Samanta
Journal:  eNeurologicalSci       Date:  2019-12-04
  6 in total

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