Literature DB >> 3275766

Achondrogenesis type I: delineation of further heterogeneity and identification of two distinct subgroups.

Z Borochowitz1, R Lachman, G E Adomian, G Spear, K Jones, D L Rimoin.   

Abstract

Achondrogenesis has traditionally been divided into type I (Parenti-Fraccaro) and type II (Langer-Saldino). We studied the clinical, radiologic, and morphologic features of 17 cases previously diagnosed as achondrogenesis type I to define whether there is even further heterogeneity. On radiographic analysis, two distinct groups of patients were defined based on the presence or absence of rib fractures and ossification of the vertebral pedicles, ischium, and fibula. Two distinct chondroosseous morphologic patterns were observed that directly correlated with the radiographic grouping. One group had round vacuolated chondrocytes with inclusion bodies; the other had collagenous rings around the chondrocytes. We conclude that achondrogenesis type I (Parenti-Fraccaro) consists of two distinct disorders: type IA, which corresponds to the cases originally published by Houston et al. and Harris et al., and type IB, which corresponds to the case originally published by Fraccaro. Analysis of Parenti's case suggests the diagnosis of achondrogenesis type II. All three types of achondrogenesis appear to be inherited as autosomal recessive traits.

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Year:  1988        PMID: 3275766     DOI: 10.1016/s0022-3476(88)80113-6

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  9 in total

1.  The boneless neonate: a severe form of achondrogenesis type I.

Authors:  H J Jaeger; A Schmitz-Stolbrink; J Hulde; M Novak; K Roggenkamp; K Mathias
Journal:  Pediatr Radiol       Date:  1994

Review 2.  Achondrogenesis type 1B.

Authors:  A Superti-Furga
Journal:  J Med Genet       Date:  1996-11       Impact factor: 6.318

3.  A defect in the metabolic activation of sulfate in a patient with achondrogenesis type IB.

Authors:  A Superti-Furga
Journal:  Am J Hum Genet       Date:  1994-12       Impact factor: 11.025

Review 4.  Atelosteogenesis type 2.

Authors:  R Newbury-Ecob
Journal:  J Med Genet       Date:  1998-01       Impact factor: 6.318

5.  Type II achondrogenesis-hypochondrogenesis: morphologic and immunohistopathologic studies.

Authors:  M Godfrey; D R Keene; E Blank; H Hori; L Y Sakai; L A Sherwin; D W Hollister
Journal:  Am J Hum Genet       Date:  1988-12       Impact factor: 11.025

6.  Atelosteogenesis type II is caused by mutations in the diastrophic dysplasia sulfate-transporter gene (DTDST): evidence for a phenotypic series involving three chondrodysplasias.

Authors:  J Hästbacka; A Superti-Furga; W R Wilcox; D L Rimoin; D H Cohn; E S Lander
Journal:  Am J Hum Genet       Date:  1996-02       Impact factor: 11.025

7.  Identification of loss-of-function mutations of SLC35D1 in patients with Schneckenbecken dysplasia, but not with other severe spondylodysplastic dysplasias group diseases.

Authors:  T Furuichi; H Kayserili; S Hiraoka; G Nishimura; H Ohashi; Y Alanay; J C Lerena; A D Aslanger; H Koseki; D H Cohn; A Superti-Furga; S Unger; S Ikegawa
Journal:  J Med Genet       Date:  2009-06-08       Impact factor: 6.318

8.  Skeletal Dysplasias That Cause Thoracic Insufficiency in Neonates: Illustrative Case Reports.

Authors:  Mehmet Sah İpek; Cihan Akgul Ozmen
Journal:  Medicine (Baltimore)       Date:  2016-04       Impact factor: 1.889

9.  Achondrogenesis type 1A: clinical, histologic, molecular, and prenatal ultrasound diagnosis.

Authors:  Sara Vanegas; Luz Fernanda Sua; Jaime López-Tenorio; Diana Ramírez-Montaño; Harry Pachajoa
Journal:  Appl Clin Genet       Date:  2018-05-25
  9 in total

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