Literature DB >> 18190602

Some cases of common variable immunodeficiency may be due to a mutation in the SBDS gene of Shwachman-Diamond syndrome.

S Khan1, J Hinks, J Shorto, M J Schwarz, W A C Sewell.   

Abstract

Known genetic defects currently account for only a small proportion of patients meeting criteria for 'probable' or 'possible' common variable immunodeficiency (CVID). A 59-year-old male with a 12-year history of CVID on intravenous immunoglobulin (IVIG) is presented who developed bronchiectasis, cytopenias and malabsorption that are recognized complications of CVID. Work-up for his malabsorption suggested the possibility of Shwachman-Diamond syndrome, confirmed by mutation testing. With the identification of the molecular defect in Shwachman-Diamond syndrome (SDS), it is becoming clear that not all SDS patients have the prominent features of neutropenia or pancreatic malabsorption. A meta-analysis of published immunological defects in SDS suggests that four of 14 hypogammaglobulinaemic SDS patients meet criteria for 'possible' CVID. Mutations in the SBDS gene may therefore be the fifth identified molecular defect in CVID.

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Year:  2008        PMID: 18190602      PMCID: PMC2276964          DOI: 10.1111/j.1365-2249.2007.03556.x

Source DB:  PubMed          Journal:  Clin Exp Immunol        ISSN: 0009-9104            Impact factor:   4.330


  26 in total

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Authors:  G W Hall; P Dale; J A Dodge
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2.  Loss of the mouse ortholog of the shwachman-diamond syndrome gene (Sbds) results in early embryonic lethality.

Authors:  Siyi Zhang; Mingjun Shi; Chi-Chung Hui; Johanna M Rommens
Journal:  Mol Cell Biol       Date:  2006-09       Impact factor: 4.272

3.  TACI is mutant in common variable immunodeficiency and IgA deficiency.

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Review 4.  Ribosomes and marrow failure: coincidental association or molecular paradigm?

Authors:  Johnson M Liu; Steven R Ellis
Journal:  Blood       Date:  2006-02-28       Impact factor: 22.113

5.  Hematologic abnormalities in Shwachman Diamond syndrome: lack of genotype-phenotype relationship.

Authors:  Taco W Kuijpers; Mariel Alders; Anton T J Tool; Clemens Mellink; Dirk Roos; Raoul C M Hennekam
Journal:  Blood       Date:  2005-03-15       Impact factor: 22.113

6.  The Shwachman-Diamond SBDS protein localizes to the nucleolus.

Authors:  Karyn M Austin; Rebecca J Leary; Akiko Shimamura
Journal:  Blood       Date:  2005-04-28       Impact factor: 22.113

Review 7.  TACItly changing tunes: farewell to a yin and yang of BAFF receptor and TACI in humoral immunity? New genetic defects in common variable immunodeficiency.

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Journal:  Curr Opin Allergy Clin Immunol       Date:  2005-12

8.  Structural and mutational analysis of the SBDS protein family. Insight into the leukemia-associated Shwachman-Diamond Syndrome.

Authors:  Camille Shammas; Tobias F Menne; Christine Hilcenko; Stephen R Michell; Beatriz Goyenechea; Graeme R B Boocock; Peter R Durie; Johanna M Rommens; Alan J Warren
Journal:  J Biol Chem       Date:  2005-02-08       Impact factor: 5.157

9.  The Shwachman-Bodian-Diamond syndrome protein family is involved in RNA metabolism.

Authors:  Alexei Savchenko; Nevan Krogan; John R Cort; Elena Evdokimova; Jocelyne M Lew; Adelinda A Yee; Luis Sánchez-Pulido; Miguel A Andrade; Alexey Bochkarev; James D Watson; Michael A Kennedy; Jack Greenblatt; Timothy Hughes; Cheryl H Arrowsmith; Johanna M Rommens; Aled M Edwards
Journal:  J Biol Chem       Date:  2005-02-08       Impact factor: 5.157

10.  A pediatric genetic disorder diagnosed in adulthood.

Authors:  Joseph A Church
Journal:  PLoS Med       Date:  2006-01-31       Impact factor: 11.069

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  8 in total

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2.  Do ribosomal protein alterations affect ER stress response in CVID?

Authors:  S Khan
Journal:  Clin Exp Immunol       Date:  2020-02-09       Impact factor: 4.330

Review 3.  Do ribosomopathies explain some cases of common variable immunodeficiency?

Authors:  S Khan; J Pereira; P J Darbyshire; S Holding; P C Doré; W A C Sewell; A Huissoon
Journal:  Clin Exp Immunol       Date:  2010-11-09       Impact factor: 4.330

4.  Severe combined immunodeficiency (SCID) presenting with neonatal aplastic anemia.

Authors:  Angela Scott; Jason Glover; Suzanne Skoda-Smith; Troy R Torgerson; Min Xu; Lauri M Burroughs; Ann E Woolfrey; Mark D Fleming; Akiko Shimamura
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5.  Shwachman-Diamond syndrome: first molecular diagnosis in a Brazilian child.

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Journal:  Rev Bras Hematol Hemoter       Date:  2013

Review 6.  Lessons From Pediatric MDS: Approaches to Germline Predisposition to Hematologic Malignancies.

Authors:  Serine Avagyan; Akiko Shimamura
Journal:  Front Oncol       Date:  2022-03-09       Impact factor: 6.244

7.  Misdiagnosis as asphyxiating thoracic dystrophy and CMV-associated haemophagocytic lymphohistiocytosis in Shwachman-Diamond syndrome.

Authors:  Heidi Schaballie; Marleen Renard; Christiane Vermylen; Isabelle Scheers; Nicole Revencu; Luc Regal; David Cassiman; Lieve Sevenants; Ilse Hoffman; Anniek Corveleyn; Victoria Bordon; Filomeen Haerynck; Karel Allegaert; Kris De Boeck; Tania Roskams; Nancy Boeckx; Xavier Bossuyt; Isabelle Meyts
Journal:  Eur J Pediatr       Date:  2013-01-12       Impact factor: 3.860

Review 8.  Guidelines for genetic skeletal dysplasias for pediatricians.

Authors:  Sung Yoon Cho; Dong-Kyu Jin
Journal:  Ann Pediatr Endocrinol Metab       Date:  2015-12-31
  8 in total

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