Literature DB >> 7469679

[Johanson-Blizzard's syndrome: another cause of pancreatic lipomatosis (author's transl)].

J L Bresson, J Schmitz, J M Saudubray, G Lesec, J A Hummel, J Rey.   

Abstract

A new case of Johanson-Blizzard's syndrome is reported. It concerns a boy born to consanguineous parents and who died at the age of 10 months from malnutrition. Anal imperforation, alar agenesia, hair anomalies, mental retardation and external pancreatic failure were associated. Neither deafness nor hypothyroidism appeared to be present. Autopsy revealed lipomatous hypoplasia of the exocrine pancreas, hitherto unobserved in this syndrome, and probably responsible for the external pancreatic failure noted in published cases. The variability within a given family of the Johanson-Blizzard malformative syndrome is illustrated by two other cases reported in the anamnesis, one involving a brother who had died earlier with cutaneous aplasia at the fontanella and lacrimal canal malposition and one involving a second cousin who presented with isolated anal imperforation.

Entities:  

Mesh:

Year:  1980        PMID: 7469679

Source DB:  PubMed          Journal:  Arch Fr Pediatr        ISSN: 0003-9764


  5 in total

Review 1.  Johanson-Blizzard syndrome.

Authors:  J A Hurst; M Baraitser
Journal:  J Med Genet       Date:  1989-01       Impact factor: 6.318

2.  The temporal bone in the Johanson-Blizzard syndrome. A CT study.

Authors:  J Braun; A Lerner; R Gershoni-Baruch
Journal:  Pediatr Radiol       Date:  1991

3.  Johanson-Blizzard syndrome. Progression of pancreatic involvement in adulthood.

Authors:  D R Trellis; R E Clouse
Journal:  Dig Dis Sci       Date:  1991-03       Impact factor: 3.199

4.  Pancreatic lipomatosis in the Shwachman-Diamond syndrome. Identification by sonography and CT-scan.

Authors:  E Robberecht; P Nachtegaele; R Van Rattinghe; M Afschrift; M Kunnen; R Verhaaren
Journal:  Pediatr Radiol       Date:  1985

5.  X-linked dominant inherited diseases with lethality in hemizygous males.

Authors:  R Wettke-Schäfer; G Kantner
Journal:  Hum Genet       Date:  1983       Impact factor: 4.132

  5 in total

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