Literature DB >> 9744479

Mutation 1091delC is highly prevalent in Spanish Sanfilippo syndrome type A patients.

M Montfort1, L Vilageliu, N Garcia-Giralt, S Guidi, M J Coll, A Chabás, D Grinberg.   

Abstract

The gene resposible for Sanfilippo syndrome type A, a lysosomal disorder caused by deficiency of sulfamidase, was recently cloned and more than 40 mutations were identified. This paper presents the mutation analysis and clinical findings in 11 Spanish patients in whom 19 of the 22 mutant alleles have been identified. This is the first report on mutations in Spanish Sanfilippo A patients. Seven different mutations were found, four of which (Q85R, R206P, A354P, and L386R) were not previously described. Mutation 1091delC was the most prevalent, accounting for nearly one-half of the mutated alleles, while mutations R245H and R74C were not found. Haplotype analysis suggests a founder effect as the cause of the high frequency of 1091delC in this population.

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Year:  1998        PMID: 9744479     DOI: 10.1002/(SICI)1098-1004(1998)12:4<274::AID-HUMU9>3.0.CO;2-F

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  8 in total

1.  Allelic heterogeneity in Spanish patients with Sanfilippo disease type B. Identification of eight new mutations.

Authors:  M J Coll; C Antón; A Chabás
Journal:  J Inherit Metab Dis       Date:  2001-02       Impact factor: 4.982

2.  Liver production of sulfamidase reverses peripheral and ameliorates CNS pathology in mucopolysaccharidosis IIIA mice.

Authors:  Albert Ruzo; Miquel Garcia; Albert Ribera; Pilar Villacampa; Virginia Haurigot; Sara Marcó; Eduard Ayuso; Xavier M Anguela; Carles Roca; Judith Agudo; David Ramos; Jesús Ruberte; Fatima Bosch
Journal:  Mol Ther       Date:  2011-10-18       Impact factor: 11.454

3.  A founder mutation in the CLCNKB gene causes Bartter syndrome type III in Spain.

Authors:  Juan Rodríguez-Soriano; Alfredo Vallo; Gustavo Pérez de Nanclares; José Ramón Bilbao; Luis Castaño
Journal:  Pediatr Nephrol       Date:  2005-05-05       Impact factor: 3.714

4.  Natural History and Molecular Characteristics of Korean Patients with Mucopolysaccharidosis Type III.

Authors:  Min-Sun Kim; Aram Yang; Eu-Seon Noh; Chiwoo Kim; Ga Young Bae; Han Hyuk Lim; Hyung-Doo Park; Sung Yoon Cho; Dong-Kyu Jin
Journal:  J Pers Med       Date:  2022-04-21

Review 5.  Sanfilippo syndrome: a mini-review.

Authors:  M J Valstar; G J G Ruijter; O P van Diggelen; B J Poorthuis; F A Wijburg
Journal:  J Inherit Metab Dis       Date:  2008-04-04       Impact factor: 4.982

6.  Natural history of Sanfilippo syndrome in Spain.

Authors:  Verónica Delgadillo; Maria del Mar O'Callaghan; Laura Gort; Maria Josep Coll; Mercedes Pineda
Journal:  Orphanet J Rare Dis       Date:  2013-12-06       Impact factor: 4.123

Review 7.  Epidemiology of Mucopolysaccharidoses Update.

Authors:  Betul Çelik; Saori C Tomatsu; Shunji Tomatsu; Shaukat A Khan
Journal:  Diagnostics (Basel)       Date:  2021-02-10

8.  Structure of sulfamidase provides insight into the molecular pathology of mucopolysaccharidosis IIIA.

Authors:  Navdeep S Sidhu; Kathrin Schreiber; Kevin Pröpper; Stefan Becker; Isabel Usón; George M Sheldrick; Jutta Gärtner; Ralph Krätzner; Robert Steinfeld
Journal:  Acta Crystallogr D Biol Crystallogr       Date:  2014-04-30
  8 in total

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