Literature DB >> 2002486

Cleft lip and palate, sensorineural deafness, and sacral lipoma in two brothers: a possible example of the disorganisation mutant.

R B Lowry1, S L Yong.   

Abstract

We report two brothers of Chinese origin who have an apparently unique syndrome of cleft lip/palate, profound sensorineural deafness, and a sacral lipoma. Additional findings which were not common to both were aberrant digital appendages on the heel and thigh of one boy and an anterior sacral meningocele and dislocated hip in the other. Intelligence is normal in both. Both boys suffer from functional constipation but biopsy studies showed no evidence of Hirschsprung's disease. The parents, who are normal, are not related. Inheritance is probably autosomal or X linked recessive. A possible link with the disorganisation mouse mutant is discussed.

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Mesh:

Year:  1991        PMID: 2002486      PMCID: PMC1016785          DOI: 10.1136/jmg.28.2.135

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


  3 in total

1.  Developmental anomalies in mice resulting from action of the gene, disorganization, a semi-dominant lethal.

Authors:  K P HUMMEL
Journal:  Pediatrics       Date:  1959-01       Impact factor: 7.124

2.  Syndromes with cleft lip and cleft palate.

Authors:  M M Cohen
Journal:  Cleft Palate J       Date:  1978-10

Review 3.  A possible human homologue for the mouse mutant disorganisation.

Authors:  R M Winter; D Donnai
Journal:  J Med Genet       Date:  1989-07       Impact factor: 6.318

  3 in total
  6 in total

1.  Another human homologue for the mouse mutant disorganisation.

Authors:  R C Hennekam
Journal:  J Med Genet       Date:  1992-01       Impact factor: 6.318

2.  Two-hit model for sporadic congenital anomalies in mice with the disorganization mutation.

Authors:  J L Crosby; D S Varnum; J H Nadeau
Journal:  Am J Hum Genet       Date:  1993-05       Impact factor: 11.025

3.  Lipomatous myelomeningocele, athyrotic hypothyroidism, and sensorineural deafness: a new form of syndromal deafness?

Authors:  H L Peters; A Bankier
Journal:  J Med Genet       Date:  1998-11       Impact factor: 6.318

4.  Human malformations similar to those in the mouse mutation disorganization (Ds).

Authors:  N H Robin; O O Adewale; D McDonald-McGinn; J H Nadeau; E H Zackai; M Bućan
Journal:  Hum Genet       Date:  1993-11       Impact factor: 4.132

5.  Limb body wall complex, amniotic band sequence, or new syndrome caused by mutation in IQ Motif containing K (IQCK)?

Authors:  Paul Kruszka; Annette Uwineza; Leon Mutesa; Ariel F Martinez; Yu Abe; Elaine H Zackai; Rebecca Ganetzky; Brian Chung; Roger E Stevenson; Robert S Adelstein; Xuefei Ma; James C Mullikin; Sung-Kook Hong; Maximilian Muenke
Journal:  Mol Genet Genomic Med       Date:  2015-05-06       Impact factor: 2.183

6.  The Orthopaedic Management of Human Disorganization Syndrome.

Authors:  Kevin Smit; Judy So; Emily Schaeffer; Linlea Armstrong; Cindy Verchere; Kishore Mulpuri
Journal:  J Am Acad Orthop Surg Glob Res Rev       Date:  2020-06-15
  6 in total

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