Literature DB >> 2395168

Microspherophakia-metaphyseal dysplasia: a 'new' dominantly inherited bone dysplasia with severe eye involvement.

A Verloes1, L Van Maldergem, P de Marneffe, J L Dufier, P Maroteaux.   

Abstract

We report a father and son affected by a hitherto unpublished bone dysplasia with moderately severe dwarfism. On initial radiographs, thickening of the diaphyses of the long bones was striking. The small bones of the extremities were almost unaffected. With age, the metaphyseal deformation became more prominent. The epiphyses became irregular and their growth was delayed (particularly the femoral heads). The femoral neck showed an unusual 'lip' on the inner edge. Later, the stubby appearance of the long bones faded and, in adulthood, only enlarged metaphyses and deformed femoral necks persisted. The vertebrae showed moderate deformation with irregular flattening, and narrowing of the spinal canal with a shortened interpedicular distance. The eye defects consisted of high grade myopia, microspherophakia, lens coloboma, lens luxation, and retinal detachment. The name 'microspherophakia-metaphyseal dysplasia' is suggested for this probably autosomal dominant bone dysplasia.

Entities:  

Mesh:

Year:  1990        PMID: 2395168      PMCID: PMC1017188          DOI: 10.1136/jmg.27.7.467

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


  8 in total

1.  Metaphyseal chondrodysplasia calcificans. A report on two cases.

Authors:  S van Creveld; K Kozlowski; K Pietron; A van der Valk
Journal:  Br J Radiol       Date:  1971-10       Impact factor: 3.039

2.  [Micrognathic dwarfism].

Authors:  P Maroteaux; C Roux; Z Fruchter
Journal:  Presse Med       Date:  1970-12-12       Impact factor: 1.228

3.  The Weissenbacher-Zweymüller, Stickler, and Marshall syndromes: further evidence for their identity.

Authors:  R M Winter; M Baraitser; K M Laurence; D Donnai; C M Hall
Journal:  Am J Med Genet       Date:  1983-10

4.  The Marshall and Stickler syndromes: objective rejection of lumping.

Authors:  S Aymé; M Preus
Journal:  J Med Genet       Date:  1984-02       Impact factor: 6.318

5.  [Kniest's disease].

Authors:  P Maroteaux; J Spranger
Journal:  Arch Fr Pediatr       Date:  1973 Aug-Sep

Review 6.  The Stickler syndrome (hereditary arthroophthalmopathy).

Authors:  J Herrmann; T D France; J W Spranger; J M Opitz; C Wiffler
Journal:  Birth Defects Orig Artic Ser       Date:  1975

7.  [Marchesani's syndrome (spherophakia-brachymorphism)].

Authors:  S Ferrier; D Nusslé; B Friedli; P E Ferrier
Journal:  Helv Paediatr Acta       Date:  1980-05

8.  The Wagner-Stickler syndrome: a study of 22 families.

Authors:  R M Liberfarb; T Hirose; L B Holmes
Journal:  J Pediatr       Date:  1981-09       Impact factor: 4.406

  8 in total

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