Literature DB >> 20556422

Eponym: Johanson-Blizzard syndrome.

Nima Rezaei1, Mozhgan Sabbaghian, Zhifeng Liu, Martin Zenker.   

Abstract

Johanson-Blizzard syndrome is a very rare autosomal recessive disorder caused by mutations in the Ubiquitin-Protein Ligase E3 Component N-Recognin 1 (UBR1) gene. The syndrome is characterized by exocrine pancreatic insufficiency and a wide range of additional clinical features, including aplasia or hypoplasia of the alae nasi, oligodontia, sensorineural hearing loss, hypothyroidism, scalp defects, mental retardation, and developmental delay. Several other abnormalities in different organs, particularly anorectal, urogenital, and cardiac anomalies have been reported since the first description of this syndrome four decades ago. UBR1 gene defects are underlying the disease. Only symptomatic treatment is available. Exocrine pancreas insufficiency plus abnormal alae nasi is pathognomonic for Johanson-Blizzard syndrome.

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Year:  2010        PMID: 20556422     DOI: 10.1007/s00431-010-1240-5

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  31 in total

Review 1.  Genetic basis and pancreatic biology of Johanson-Blizzard syndrome.

Authors:  Martin Zenker; Julia Mayerle; André Reis; Markus M Lerch
Journal:  Endocrinol Metab Clin North Am       Date:  2006-06       Impact factor: 4.741

Review 2.  Johanson-Blizzard syndrome.

Authors:  J A Hurst; M Baraitser
Journal:  J Med Genet       Date:  1989-01       Impact factor: 6.318

3.  Johanson-Blizzard syndrome: autopsy findings with special emphasis on hypopituitarism and review of the literature.

Authors:  William H Hoffman; Jeffrey R Lee; Kalman Kovacs; Harold Chen; Farivar Yaghmai
Journal:  Pediatr Dev Pathol       Date:  2007 Jan-Feb

4.  Johanson-Blizzard syndrome--a case study, behavioral manifestations, and successful treatment strategies.

Authors:  John F Prater; Karyn D'Addio
Journal:  Biol Psychiatry       Date:  2002-03-15       Impact factor: 13.382

5.  Novel UBR1 gene mutation in a patient with typical phenotype of Johanson-Blizzard syndrome.

Authors:  Gholam Hossein Fallahi; Mozhgan Sabbaghian; Manijeh Khalili; Nima Parvaneh; Martin Zenker; Nima Rezaei
Journal:  Eur J Pediatr       Date:  2010-06-17       Impact factor: 3.183

6.  Johanson-Blizzard syndrome facial anomaly and its correction using a microsurgical bone graft and tripartite osteotomy.

Authors:  S Kobayashi; K Ohmori; J Sekiguchi
Journal:  J Craniofac Surg       Date:  1995-09       Impact factor: 1.046

7.  Deficiency of UBR1, a ubiquitin ligase of the N-end rule pathway, causes pancreatic dysfunction, malformations and mental retardation (Johanson-Blizzard syndrome).

Authors:  Martin Zenker; Julia Mayerle; Markus M Lerch; Andreas Tagariello; Klaus Zerres; Peter R Durie; Matthias Beier; Georg Hülskamp; Celina Guzman; Helga Rehder; Frits A Beemer; Ben Hamel; Philippe Vanlieferinghen; Ruth Gershoni-Baruch; Marta W Vieira; Miroslav Dumic; Ron Auslender; Vera L Gil-da-Silva-Lopes; Simone Steinlicht; Manfred Rauh; Stavit A Shalev; Christian Thiel; Arif B Ekici; Andreas Winterpacht; Yong Tae Kwon; Alexander Varshavsky; André Reis
Journal:  Nat Genet       Date:  2005-11-20       Impact factor: 38.330

8.  Johanson-blizzard syndrome: loss of glucagon secretion response to insulin-induced hypoglycemia.

Authors:  Tsutomu Takahashi; Miwa Fujishima; Satoko Tsuchida; Masamichi Enoki; Goro Takada
Journal:  J Pediatr Endocrinol Metab       Date:  2004-08       Impact factor: 1.634

Review 9.  The ubiquitin system: from basic mechanisms to the patient bed.

Authors:  Aaron Ciechanover; Kazuhiro Iwai
Journal:  IUBMB Life       Date:  2004-04       Impact factor: 3.885

10.  Johanson-Blizzard syndrome caused by identical UBR1 mutations in two unrelated girls, one with a cardiomyopathy.

Authors:  Mariet Elting; Ariana Kariminejad; Marie-Louise de Sonnaville; Jaap Ottenkamp; Susanne Bauhuber; Bita Bozorgmehr; Martin Zenker; Jan M Cobben
Journal:  Am J Med Genet A       Date:  2008-12-01       Impact factor: 2.802

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  3 in total

Review 1.  The N-end rule pathway.

Authors:  Takafumi Tasaki; Shashikanth M Sriram; Kyong Soo Park; Yong Tae Kwon
Journal:  Annu Rev Biochem       Date:  2012-04-10       Impact factor: 23.643

2.  A Case with Complete Pancreatic Aplasia Suggestive of Johanson-Blizzard Syndrome.

Authors:  Seyed Ali Jafari; Roozbeh Moghaddar; Mohammad Bahadoram; Hamid Reza Kianifar; Mehran Beiraghi Tosi
Journal:  J Clin Diagn Res       Date:  2016-08-01

Review 3.  Is a fatty pancreas a banal lesion?

Authors:  Andrzej Smereczyński; Katarzyna Kołaczyk
Journal:  J Ultrason       Date:  2016-09-07
  3 in total

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