Literature DB >> 474623

Genetic and diagnostic considerations in three families with abnormalities of facial expression and congenital urinary obstruction: "The Ochoa syndrome".

B R Elejalde.   

Abstract

Seven patients (4 females, and 3 males) born in unrelated families, one of them consanguineous (first cousins), were affected by peculiar facies and gestures while smiling and crying, and by hydronephrosis, hydroureter and intravesical stenosis of the ureter, abnormal caliber of the urether in the prostatic and membranous portions, urethral valves, abnormal bladder with trabeculation, and diverticula associated with severe hypertrophy of the mucosa with sclerotic changes. The genetic analysis of these families indicates that the condition is probably autosomal dominant, with variable expressivity and incomplete penetrance. The syndrome represents alteration of facial and urinary developmental fields. The peculiar facies allows early recognition of the condition, and this can be helpful for early assessment and treatment, leading perhaps to a better prognosis.

Entities:  

Mesh:

Year:  1979        PMID: 474623     DOI: 10.1002/ajmg.1320030114

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  9 in total

Review 1.  Dysfunctional voiding syndromes and vesicoureteral reflux.

Authors:  Y L Homsy
Journal:  Pediatr Nephrol       Date:  1994-02       Impact factor: 3.714

2.  Ochoa syndrome: a spectrum of urofacial syndrome.

Authors:  Ozgu Aydogdu; Berk Burgu; Fuat Demirel; Tarkan Soygur; Zeynep Birsin Ozcakar; Fatos Yalcinkaya; Serdar Tekgul
Journal:  Eur J Pediatr       Date:  2009-08-11       Impact factor: 3.183

3.  Exome capture and massively parallel sequencing identifies a novel HPSE2 mutation in a Saudi Arabian child with Ochoa (urofacial) syndrome.

Authors:  Wisam Al Badr; Suha Al Bader; Edgar Otto; Friedhelm Hildebrandt; Todd Ackley; Weiping Peng; Jishu Xu; Jun Li; Kailey M Owens; David Bloom; Jeffrey W Innis
Journal:  J Pediatr Urol       Date:  2011-03-29       Impact factor: 1.830

4.  Expanding the HPSE2 Genotypic Spectrum in Urofacial Syndrome, A Disease Featuring a Peripheral Neuropathy of the Urinary Bladder.

Authors:  Glenda M Beaman; Filipa M Lopes; Aybike Hofmann; Wolfgang Roesch; Martin Promm; Emilia K Bijlsma; Chirag Patel; Aykut Akinci; Berk Burgu; Jeroen Knijnenburg; Gladys Ho; Christina Aufschlaeger; Sylvia Dathe; Marie Antoinette Voelckel; Monika Cohen; Wyatt W Yue; Helen M Stuart; Edward A Mckenzie; Mark Elvin; Neil A Roberts; Adrian S Woolf; William G Newman
Journal:  Front Genet       Date:  2022-06-23       Impact factor: 4.772

Review 5.  Clinical and genetic characteristics for the Urofacial Syndrome (UFS).

Authors:  Yaqin Tu; Ping Yang; Jia Yang; Yuchen Xu; Fei Xiong; Qilin Yu; Weikuan Gu; Dinel Pond; Nancy Mendelsohn; Guus A M A Lachmeijer; Shu Zhang; Cong-Yi Wang
Journal:  Int J Clin Exp Pathol       Date:  2014-04-15

Review 6.  Can a congenital dysfunctional bladder be diagnosed from a smile? The Ochoa syndrome updated.

Authors:  Bernardo Ochoa
Journal:  Pediatr Nephrol       Date:  2003-11-25       Impact factor: 3.714

7.  A Tribute to Bernardo Ochoa, MD.

Authors:  Ricardo González
Journal:  Front Pediatr       Date:  2017-04-27       Impact factor: 3.418

8.  Heparanase 2, mutated in urofacial syndrome, mediates peripheral neural development in Xenopus.

Authors:  Neil A Roberts; Adrian S Woolf; Helen M Stuart; Raphaël Thuret; Edward A McKenzie; William G Newman; Emma N Hilton
Journal:  Hum Mol Genet       Date:  2014-04-01       Impact factor: 6.150

9.  Genetic mapping to 10q23.3-q24.2, in a large Italian pedigree, of a new syndrome showing bilateral cataracts, gastroesophageal reflux, and spastic paraparesis with amyotrophy.

Authors:  M Seri; R Cusano; P Forabosco; R Cinti; F Caroli; P Picco; R Bini; V B Morra; G De Michele; M Lerone; M Silengo; I Pela; C Borrone; G Romeo; M Devoto
Journal:  Am J Hum Genet       Date:  1999-02       Impact factor: 11.025

  9 in total

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