Literature DB >> 26183508

Dealing with congenital hepatic fibrosis? Remember COACH syndrome.

B C Acharyya1, M K Goenka, S Chatterjee, U Goenka.   

Abstract

Coach Syndrome is a rare cause of Congenital Hepatic Fibrosis associated with neurological features. COACH is a mnemonic comprising of Cerebellar vermis hypo/aplasia, Oligophrenia (developmental delay/mental retardation), Ataxia, Coloboma and Hepatic fibrosis. Here we describe a 12 years boy who presented with hepatic encephalopathy. He was subsequently found to have marked developmental delay, bilateral ptosis and ataxia. CT scan revealed brain stem molar tooth sign, ophthalmoscopy showed optic disc coloboma and elastography showed hepatic fibrosis to confirm him as a case of COACH Syndrome.

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Year:  2013        PMID: 26183508     DOI: 10.1007/s12328-013-0418-6

Source DB:  PubMed          Journal:  Clin J Gastroenterol        ISSN: 1865-7265


  9 in total

1.  Imaging findings in COACH syndrome.

Authors:  M Barzilai; N Ish-Shalom; A Lerner; T C Iancu
Journal:  AJR Am J Roentgenol       Date:  1998-04       Impact factor: 3.959

2.  An autosomal recessive mental retardation syndrome with hepatic fibrosis and renal cysts.

Authors:  E Thompson; M Baraitser
Journal:  Am J Med Genet       Date:  1986-05

3.  Hepatic fibrosis, polycystic kidney, colobomata and encephalopathy in siblings.

Authors:  A G Hunter; S J Rothman; W S Hwang; R J Deckelbaum
Journal:  Clin Genet       Date:  1974       Impact factor: 4.438

4.  COACH syndrome: report of two brothers with congenital hepatic fibrosis, cerebellar vermis hypoplasia, oligophrenia, ataxia, and mental retardation.

Authors:  M Gentile; A Di Carlo; F Susca; A Gambotto; M L Caruso; C Panella; P Vajro; G Guanti
Journal:  Am J Med Genet       Date:  1996-08-23

5.  Renal insufficiency is a component of COACH syndrome.

Authors:  S Kumar; R Rankin
Journal:  Am J Med Genet       Date:  1996-01-11

6.  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

7.  The first Japanese case of COACH syndrome.

Authors:  Fukiko Mitsui; Hiroshi Aikata; Takahiro Azakami; Yoshio Katamura; Takashi Kimura; Tomokazu Kawaoka; Hiromi Saneto; Shintaro Takaki; Nobuhiko Hiraga; Masataka Tsuge; Koji Waki; Akira Hiramatsu; Michio Imamura; Yoshiiku Kawakami; Shoichi Takahashi; Koji Arihiro; Kazuaki Chayama
Journal:  Hepatol Res       Date:  2009-03       Impact factor: 4.288

8.  Further delineation of a syndrome of cerebellar vermis hypo/aplasia, oligophrenia, congenital ataxia, coloboma, and hepatic fibrosis.

Authors:  A Verloes; C Lambotte
Journal:  Am J Med Genet       Date:  1989-02

9.  Joubert syndrome with congenital hepatic fibrosis: an entity in the spectrum of oculo-encephalo-hepato-renal disorders.

Authors:  S M Lewis; E A Roberts; M A Marcon; E Harvey; M J Phillips; S A Chuang; J R Buncic; J T Clarke
Journal:  Am J Med Genet       Date:  1994-10-01
  9 in total
  2 in total

1.  An Unusual Case of Congenital Hepatic Fibrosis with Retinitis Pigmentosa.

Authors:  Nazish Butt; Ali Akbar; Anoshia Fahad
Journal:  Cureus       Date:  2019-09-27

2.  Congenital Hepatic Fibrosis in Children and Adults: Clinical Manifestations, Management, and Outcome-Case Series and Literature Review.

Authors:  Beidi Zhu; Zunguo Du; Zhengxin Wang; Yang Li; Jiming Zhang; Haoxiang Zhu
Journal:  Gastroenterol Res Pract       Date:  2020-04-21       Impact factor: 2.260

  2 in total

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