Literature DB >> 7468650

Non-Majewski short rib-polydactyly syndrome.

D O Sillence.   

Abstract

Clinical, radiographic, and morphologic studies of non-Majewski short rib-polydactyly cases show a spectrum of abnormalities. While the findings of Yang et al reported in this journal suggest that a unique change in chondrocytes, namely PAS-positive inclusions, might distinguish one group of patients from another, experience with 4 other cases with radiographic findings similar to the case reported by Yang and colleagues does not confirm the finding. Therefore, the full significance of their observation must await further morphologic studies of these patients. The clinical findings are extremely variable, and no feature is pathognomonic of either of the groups of patients. Similarly, the radiographic findings show a spectrum of severity. The original cases of Saldino and Noonan have a more severe ossification defect in the long bones and pelvis than any of these other cases. While genetic heterogeneity might be invoked to explain this wide spectrum of findings in these patients, at present, variability in expression in just as likely a hypothesis.

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Year:  1980        PMID: 7468650     DOI: 10.1002/ajmg.1320070215

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  9 in total

1.  Mutations in DYNC2H1, the cytoplasmic dynein 2, heavy chain 1 motor protein gene, cause short-rib polydactyly type I, Saldino-Noonan type.

Authors:  N Badiner; S P Taylor; K Forlenza; R S Lachman; M Bamshad; D Nickerson; D H Cohn; D Krakow
Journal:  Clin Genet       Date:  2017-03-13       Impact factor: 4.438

2.  Complex consanguinity associated with short rib-polydactyly syndrome III and congenital infection-like syndrome: a diagnostic problem in dysmorphic syndromes.

Authors:  L I al-Gazali; L Sztriha; A Dawodu; E Varady; M Bakir; A Khdir; J Johansen
Journal:  J Med Genet       Date:  1999-06       Impact factor: 6.318

3.  Short rib syndrome without polydactyly.

Authors:  K Chen; P A McKeever; I D Young
Journal:  J Med Genet       Date:  1989-05       Impact factor: 6.318

4.  Perinatally lethal short rib-polydactyly syndromes. 1. Variability in known syndromes.

Authors:  D Sillence; K Kozlowski; J Bar-ziv; A Fuhrumann-Rieger; W Fuhrmann; F Pascu
Journal:  Pediatr Radiol       Date:  1987

5.  Parental consanguinity and the Majewski syndrome.

Authors:  I L Black; J Fitzsimmons; E Fitzsimmons; A J Thomas
Journal:  J Med Genet       Date:  1982-04       Impact factor: 6.318

6.  Lethal short rib-polydactyly syndromes: further evidence for their overlapping in a continuous spectrum.

Authors:  M L Martínez-Frías; E Bermejo; M Urioste; H Huertas; I Arroyo
Journal:  J Med Genet       Date:  1993-11       Impact factor: 6.318

7.  Atrioventricular septal defect and type A postaxial polydactyly without other major associated anomalies: a specific association.

Authors:  S E Levin; R Dansky; S Milner; A Benatar; K Govendrageloo; J du Plessis
Journal:  Pediatr Cardiol       Date:  1995 Sep-Oct       Impact factor: 1.655

8.  Short rib-polydactyly syndrome: a single or heterogeneous entity? A re-evaluation prompted by four new cases.

Authors:  R Bernstein; J Isdale; M Pinto; J Du Toit Zaaijman; T Jenkins
Journal:  J Med Genet       Date:  1985-02       Impact factor: 6.318

9.  A lethal short rib syndrome without polydactyly.

Authors:  R M Winter
Journal:  J Med Genet       Date:  1988-05       Impact factor: 6.318

  9 in total

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