Literature DB >> 2251013

A new syndrome with cerebro-oculo-skeletal-renal involvement.

M C Silengo1, M Lerone, A Pelizza, R Gatti, A Barabino, G Romeo.   

Abstract

We report on an infant male who presented with microcephaly of prenatal onset, schizencephaly, decorticated disturbance of the neurological function, congenital optic atrophy, abnormal eye movements and nystagmus. In addition, he had a skeletal dysplasia with predominant acromelic involvement and a renal disease characterized by both nephritic and nephrotic changes. The natural history of his condition included severe postnatal failure to thrive, lack of development of psychomotor milestones, intractable seizures, terminal renal insufficiency with early death. Such spectrum of phenotypic abnormalities has never been reported before and we suggest that it may represent a new syndromic entity. The differential diagnosis with the oculo-skeletal-renal syndromes, with the osteodysplastic primordial dwarfism of the Taybi-Linder type and with the Hutterite cerebro-osteo-nephrodysplasia, is discussed.

Entities:  

Mesh:

Year:  1990        PMID: 2251013     DOI: 10.1007/bf02129073

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  6 in total

1.  A new syndrome with ocular, skeletal and renal involvement.

Authors:  M Cirillo Silengo; G Lopez Bell; M Biagioli; A Guala; G Porcellini; P Franceschini
Journal:  Pediatr Radiol       Date:  1987

2.  Hutterite cerebro-osteo-nephrodysplasia: autosomal recessive trait in a Lehrerleut Hutterite family from Montana.

Authors:  J M Opitz; R B Lowry; T M Holmes; K Morgan
Journal:  Am J Med Genet       Date:  1985-11

3.  Studies of microcephalic primordial dwarfism III: an intrauterine dwarf with platyspondyly and anomalies of pelvis and clavicles--osteodysplastic primordial dwarfism type III.

Authors:  F Majewski; M Stoeckenius; H Kemperdick
Journal:  Am J Med Genet       Date:  1982-05

4.  Familial nephropathy associatdd with retinitis pigmentosa, cerebellar ataxia and skeletal abnormalities.

Authors:  F Mainzer; R M Saldino; M B Ozonoff; H Minagi
Journal:  Am J Med       Date:  1970-10       Impact factor: 4.965

5.  [Congenital familial dwarfism with cephaloskeletal dysplasia (Taybi-Linder syndrome)].

Authors:  B Lavollay; C Faure; G Filipe; G Branca; Y Huet de Barochez
Journal:  Arch Fr Pediatr       Date:  1984-01

6.  [Familial nephropathy with retinitis pigmentosa and peripheral dysostosis].

Authors:  L Diekmann; C Louis; E Schulte-Kemna
Journal:  Helv Paediatr Acta       Date:  1977-11
  6 in total
  2 in total

1.  A genetic screen identifies putative targets and binding partners of CREB-binding protein in the developing Drosophila eye.

Authors:  Jason Anderson; Rohan Bhandari; Justin P Kumar
Journal:  Genetics       Date:  2005-07-05       Impact factor: 4.562

2.  CREB binding protein functions during successive stages of eye development in Drosophila.

Authors:  Justin P Kumar; Tazeen Jamal; Alex Doetsch; F Rudolf Turner; Joseph B Duffy
Journal:  Genetics       Date:  2004-10       Impact factor: 4.562

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.