Literature DB >> 28232778

Bloom's Syndrome: Clinical Spectrum, Molecular Pathogenesis, and Cancer Predisposition.

Christopher Cunniff1, Jennifer A Bassetti1, Nathan A Ellis2.   

Abstract

Bloom's syndrome is an autosomal recessive disorder characterized by prenatal and postnatal growth deficiency, photosensitive skin changes, immune deficiency, insulin resistance, and a greatly increased risk of early onset of cancer and for the development of multiple cancers. Loss-of-function mutations of BLM, which codes for a RecQ helicase, cause Bloom's syndrome. The absence of a functional BLM protein causes chromosome instability, excessive homologous recombination, and a greatly increased number of sister chromatid exchanges that are pathognomonic of the syndrome. A common founder mutation designated blmAsh is present in about 1 in 100 persons of Eastern European Jewish ancestry, and there are additional recurrent founder mutations among other populations. Missense, nonsense, and frameshift mutations as well as multiexonic deletions have all been observed. Bloom's syndrome is a prototypical chromosomal instability syndrome, and the somatic mutations that occur as a result of that instability are responsible for the increased cancer risk. Although there is currently no treatment aimed at the underlying genetic abnormality, persons with Bloom's syndrome benefit from sun protection, aggressive treatment of infections, surveillance for insulin resistance, and early identification of cancer.

Entities:  

Keywords:  BLM; Bloom's syndrome; Cancer

Year:  2016        PMID: 28232778      PMCID: PMC5260600          DOI: 10.1159/000452082

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  134 in total

1.  CHROMOSOMAL BREAKAGE IN A RARE AND PROBABLY GENETICALLY DETERMINED SYNDROME OF MAN.

Authors:  J GERMAN; R ARCHIBALD; D BLOOM
Journal:  Science       Date:  1965-04-23       Impact factor: 47.728

2.  SUMO: the glue that binds.

Authors:  Michael J Matunis; Xiang-Dong Zhang; Nathan A Ellis
Journal:  Dev Cell       Date:  2006-11       Impact factor: 12.270

3.  Regulation of gene expression by the BLM helicase correlates with the presence of G-quadruplex DNA motifs.

Authors:  Giang Huong Nguyen; Weiliang Tang; Ana I Robles; Richard P Beyer; Lucas T Gray; Judith A Welsh; Aaron J Schetter; Kensuke Kumamoto; Xin Wei Wang; Ian D Hickson; Nancy Maizels; Raymond J Monnat; Curtis C Harris
Journal:  Proc Natl Acad Sci U S A       Date:  2014-06-23       Impact factor: 11.205

4.  Successful pregnancy in a woman with Bloom syndrome.

Authors:  C A Chisholm; M J Bray; L B Karns
Journal:  Am J Med Genet       Date:  2001-08-01

5.  Back mutation can produce phenotype reversion in Bloom syndrome somatic cells.

Authors:  N A Ellis; S Ciocci; J German
Journal:  Hum Genet       Date:  2001-02       Impact factor: 4.132

6.  Bloom's syndrome. Clinical features and immunologic abnormalities of four patients.

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Journal:  Am J Dis Child       Date:  1988-10

7.  Enhanced tumor formation in mice heterozygous for Blm mutation.

Authors:  Kathleen Heppner Goss; Mary A Risinger; Jennifer J Kordich; Maureen M Sanz; Joel E Straughen; Lisa E Slovek; Anthony J Capobianco; James German; Gregory P Boivin; Joanna Groden
Journal:  Science       Date:  2002-09-20       Impact factor: 47.728

8.  Prevalence of breast and colorectal cancer in Ashkenazi Jewish carriers of Fanconi anemia and Bloom syndrome.

Authors:  Hagit N Baris; Inbal Kedar; Gabrielle J Halpern; Tamy Shohat; Nurit Magal; Mark D Ludman; Mordechai Shohat
Journal:  Isr Med Assoc J       Date:  2007-12       Impact factor: 0.892

9.  Bloom syndrome: a single complementation group defines patients of diverse ethnic origin.

Authors:  R Weksberg; C Smith; L Anson-Cartwright; K Maloney
Journal:  Am J Hum Genet       Date:  1988-06       Impact factor: 11.025

10.  Genomic instability resulting from Blm deficiency compromises development, maintenance, and function of the B cell lineage.

Authors:  Holger Babbe; Jennifer McMenamin; Elias Hobeika; Jing Wang; Scott J Rodig; Michael Reth; Philip Leder
Journal:  J Immunol       Date:  2009-01-01       Impact factor: 5.422

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  72 in total

Review 1.  Osteosarcoma: Molecular Pathogenesis and iPSC Modeling.

Authors:  Yu-Hsuan Lin; Brittany E Jewell; Julian Gingold; Linchao Lu; Ruiying Zhao; Lisa L Wang; Dung-Fang Lee
Journal:  Trends Mol Med       Date:  2017-07-20       Impact factor: 11.951

2.  First Two Cases of Bloom Syndrome in Russia: Lack of Skin Manifestations in a BLM c.1642C>T (p.Q548X) Homozygote as a Likely Cause of Underdiagnosis.

Authors:  Evgeny N Suspitsin; Farida I Sibgatullina; Lydia V Lyazina; Evgeny N Imyanitov
Journal:  Mol Syndromol       Date:  2017-01-17

3.  BLM's balancing act and the involvement of FANCJ in DNA repair.

Authors:  Srijita Dhar; Robert M Brosh
Journal:  Cell Cycle       Date:  2018-09-23       Impact factor: 4.534

Review 4.  RecQ and Fe-S helicases have unique roles in DNA metabolism dictated by their unwinding directionality, substrate specificity, and protein interactions.

Authors:  Katrina N Estep; Robert M Brosh
Journal:  Biochem Soc Trans       Date:  2017-12-22       Impact factor: 5.407

5.  Germline BLM mutations and metastatic prostate cancer.

Authors:  Elisa M Ledet; Emmanuel S Antonarakis; William B Isaacs; Tamara L Lotan; Colin Pritchard; A Oliver Sartor
Journal:  Prostate       Date:  2019-11-05       Impact factor: 4.104

Review 6.  DNA repair fidelity in stem cell maintenance, health, and disease.

Authors:  Chinnadurai Mani; P Hemachandra Reddy; Komaraiah Palle
Journal:  Biochim Biophys Acta Mol Basis Dis       Date:  2019-04-04       Impact factor: 5.187

Review 7.  Recommendations for Childhood Cancer Screening and Surveillance in DNA Repair Disorders.

Authors:  Michael F Walsh; Vivian Y Chang; Wendy K Kohlmann; Hamish S Scott; Christopher Cunniff; Franck Bourdeaut; Jan J Molenaar; Christopher C Porter; John T Sandlund; Sharon E Plon; Lisa L Wang; Sharon A Savage
Journal:  Clin Cancer Res       Date:  2017-06-01       Impact factor: 12.531

Review 8.  Fanconi anaemia and cancer: an intricate relationship.

Authors:  Grzegorz Nalepa; D Wade Clapp
Journal:  Nat Rev Cancer       Date:  2018-01-29       Impact factor: 60.716

9.  Diagnosis of Bloom Syndrome in a Patient with Short Stature, Recurrence of Malignant Lymphoma, and Consanguineous Origin.

Authors:  Jakub Trizuljak; Terezie Petruchová; Ivona Blaháková; Zuzana Vrzalová; Věra Hořínová; Martina Doubková; Jozef Michalka; Jiří Mayer; Šárka Pospíšilová; Michael Doubek
Journal:  Mol Syndromol       Date:  2020-03-21

Review 10.  Review: Ewing Sarcoma Predisposition.

Authors:  Pablo Gargallo; Yania Yáñez; Antonio Juan; Vanessa Segura; Julia Balaguer; Bárbara Torres; Silves Oltra; Victoria Castel; Adela Cañete
Journal:  Pathol Oncol Res       Date:  2019-10-26       Impact factor: 3.201

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