Literature DB >> 12368986

Cerebellar vermis defect, oligophrenia, congenital ataxia, and hepatic fibrocirrhosis without coloboma and renal abnormalities: report of three cases.

G Coppola1, P Vajro, S De Virgiliis, E Ciccimarra, L Boccone, A Pascotto.   

Abstract

We describe 3 children (2 siblings aged 10 and 3 years, and 1 sporadic case aged 13 years) with cerebellar vermis defect associated with oligophrenia, congenital ataxia, and hepatic fibrocirrhosis. Differently from what is reported in COACH syndrome, coloboma and renal involvement were absent. Since in one patient hepatic involvement was subclinical and early therapy seemed to prevent disease progression, the presence of liver disease should be carefully investigated in any patient with ataxia and midline cerebellar defects.

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Year:  2002        PMID: 12368986     DOI: 10.1055/s-2002-34492

Source DB:  PubMed          Journal:  Neuropediatrics        ISSN: 0174-304X            Impact factor:   1.947


  3 in total

1.  MKS3/TMEM67 mutations are a major cause of COACH Syndrome, a Joubert Syndrome related disorder with liver involvement.

Authors:  Francesco Brancati; Miriam Iannicelli; Lorena Travaglini; Annalisa Mazzotta; Enrico Bertini; Eugen Boltshauser; Stefano D'Arrigo; Francesco Emma; Elisa Fazzi; Romina Gallizzi; Mattia Gentile; Damir Loncarevic; Vlatka Mejaski-Bosnjak; Chiara Pantaleoni; Luciana Rigoli; Carmelo D Salpietro; Sabrina Signorini; Gilda Rita Stringini; Alain Verloes; Dominika Zabloka; Bruno Dallapiccola; Joseph G Gleeson; Enza Maria Valente
Journal:  Hum Mutat       Date:  2009-02       Impact factor: 4.878

2.  Mutations in 3 genes (MKS3, CC2D2A and RPGRIP1L) cause COACH syndrome (Joubert syndrome with congenital hepatic fibrosis).

Authors:  D Doherty; M A Parisi; L S Finn; M Gunay-Aygun; M Al-Mateen; D Bates; C Clericuzio; H Demir; M Dorschner; A J van Essen; W A Gahl; M Gentile; N T Gorden; A Hikida; D Knutzen; H Ozyurek; I Phelps; P Rosenthal; A Verloes; H Weigand; P F Chance; W B Dobyns; I A Glass
Journal:  J Med Genet       Date:  2009-07-01       Impact factor: 6.318

3.  Fatty liver disease and hypertransaminasemia hiding the association of clinically silent Duchenne muscular dystrophy and hereditary fructose intolerance.

Authors:  Giulia Paolella; Pasquale Pisano; Raffaele Albano; Lucio Cannaviello; Carolina Mauro; Gabriella Esposito; Pietro Vajro
Journal:  Ital J Pediatr       Date:  2012-10-31       Impact factor: 2.638

  3 in total

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