Literature DB >> 26835301

The concept and practice of Fanconi Anemia: from the clinical bedside to the laboratory bench.

Zhan-He Wu1.   

Abstract

Fanconi Anemia (FA) is characterised with multiple gene mutations, multiple types of genetic abnormalities, multiple organ involvements and multiple types of cancer risks. It is a life threatening disease commonly at 5 years old children. Research on FA is one of the fastest areas in medical research field. The identification of 15 different FA genes and the elucidation of the FA molecular pathways have translated into the understanding of the pathogenic mechanism and practically provided the directions for therapies. Studies on FA rendered invaluable information for the studies on cancers because FA possesses the unique features in many different biological aspects. Studies revealed the genetic linking between FA and cancers that FA genes are in cancers and cancers genes are in FA. As a result, FA is named as a paradigmatic disease for the understanding of cancer and aging. In clinical practice, an early and accurate diagnosis of FA before the stage of bone marrow failure, cancer/leukemia is crucial for the adequate treatment, the prevention of serious medical complications and also for the properly management in the other caring areas including paediatric, hematology, immunology, endocrinology, reproductive/IVF, obstetrics and surgery. However, an early and accurate diagnosis for FA is often difficult because FA is genetically and phenotypically heterogeneous disease. Diagnosis in more or less cases can be delayed until bone marrow failure or cancer/leukemia occurs. As a result that delayed or misdiagnosis even wrong treatment received for patients with FA are not uncommon events clinically in some regions or countries due to the lack of recognition of FA from the clinicians and the limitation in testing resource in laboratory. In this review, the new concept, brief clinical characteristics, research advancing, diagnostic guidelines/differential diagnosis, laboratory testing issues and strategies on FA are discussed.

Entities:  

Keywords:  Fanconi Anemia testing; autosomal recessive disease; bone marrow failure syndromes; cancer susceptibility; chromosomal instability syndromes

Year:  2013        PMID: 26835301      PMCID: PMC4728931          DOI: 10.3978/j.issn.2224-4336.2013.07.01

Source DB:  PubMed          Journal:  Transl Pediatr        ISSN: 2224-4336


  38 in total

1.  Malignancies and survival patterns in the National Cancer Institute inherited bone marrow failure syndromes cohort study.

Authors:  Blanche P Alter; Neelam Giri; Sharon A Savage; June A Peters; Jennifer T Loud; Lisa Leathwood; Ann G Carr; Mark H Greene; Philip S Rosenberg
Journal:  Br J Haematol       Date:  2010-04-30       Impact factor: 6.998

2.  The IVS4 + 4 A to T mutation of the fanconi anemia gene FANCC is not associated with a severe phenotype in Japanese patients.

Authors:  M Futaki; T Yamashita; H Yagasaki; T Toda; M Yabe; S Kato; S Asano; T Nakahata
Journal:  Blood       Date:  2000-02-15       Impact factor: 22.113

3.  X-linked inheritance of Fanconi anemia complementation group B.

Authors:  Amom Ruhikanta Meetei; Marieke Levitus; Yutong Xue; Annette L Medhurst; Michel Zwaan; Chen Ling; Martin A Rooimans; Patrick Bier; Maureen Hoatlin; Gerard Pals; Johan P de Winter; Weidong Wang; Hans Joenje
Journal:  Nat Genet       Date:  2004-10-24       Impact factor: 38.330

4.  Evaluation of growth and hormonal status in patients referred to the International Fanconi Anemia Registry.

Authors:  M P Wajnrajch; J M Gertner; Z Huma; J Popovic; K Lin; P C Verlander; S D Batish; P F Giampietro; J G Davis; M I New; A D Auerbach
Journal:  Pediatrics       Date:  2001-04       Impact factor: 7.124

5.  Molecular and genealogical evidence for a founder effect in Fanconi anemia families of the Afrikaner population of South Africa.

Authors:  A J Tipping; T Pearson; N V Morgan; R A Gibson; L P Kuyt; C Havenga; E Gluckman; H Joenje; T de Ravel; S Jansen; C G Mathew
Journal:  Proc Natl Acad Sci U S A       Date:  2001-05-08       Impact factor: 11.205

Review 6.  Pathophysiology and management of inherited bone marrow failure syndromes.

Authors:  Akiko Shimamura; Blanche P Alter
Journal:  Blood Rev       Date:  2010-04-24       Impact factor: 8.250

7.  Clonal chromosomal aberrations in bone marrow cells of Fanconi anemia patients: gains of the chromosomal segment 3q26q29 as an adverse risk factor.

Authors:  Holger Tönnies; Stefanie Huber; Jörn-Sven Kuhl; Antje Gerlach; Wolfram Ebell; Heidemarie Neitzel
Journal:  Blood       Date:  2003-01-02       Impact factor: 22.113

8.  Clinical variability of Fanconi anemia (type C) results from expression of an amino terminal truncated Fanconi anemia complementation group C polypeptide with partial activity.

Authors:  T Yamashita; N Wu; G Kupfer; C Corless; H Joenje; M Grompe; A D D'Andrea
Journal:  Blood       Date:  1996-05-15       Impact factor: 22.113

9.  Diagnosis of fanconi anemia: chromosomal breakage analysis.

Authors:  Anneke B Oostra; Aggie W M Nieuwint; Hans Joenje; Johan P de Winter
Journal:  Anemia       Date:  2012-05-24

10.  Towards a molecular understanding of the fanconi anemia core complex.

Authors:  Charlotte Hodson; Helen Walden
Journal:  Anemia       Date:  2012-05-22
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  3 in total

1.  PKNOX2 expression and regulation in the bone marrow mesenchymal stem cells of Fanconi anemia patients and healthy donors.

Authors:  Ilgin Cagnan; Erdal Cosgun; Ozlen Konu; Duygu Uckan; Aysen Gunel-Ozcan
Journal:  Mol Biol Rep       Date:  2018-12-04       Impact factor: 2.316

2.  Clinical and Molecular Characterization of Fanconi Anemia Patients in Turkey.

Authors:  Güven Toksoy; Dilek Uludağ Alkaya; Gülendam Bagirova; Şahin Avcı; Agharza Aghayev; Nilay Günes; Umut Altunoğlu; Yasemin Alanay; Seher Başaran; Ezgi G Berkay; Birsen Karaman; Tiraje T Celkan; Hilmi Apak; Hülya Kayserili; Beyhan Tüysüz; Zehra O Uyguner
Journal:  Mol Syndromol       Date:  2020-09-23

3.  A Case Report of Germline Compound Heterozygous Mutations in the BRCA1 Gene of an Ovarian and Breast Cancer Patient.

Authors:  Ava Kwong; Cecilia Y S Ho; Vivian Y Shin; Chun Hang Au; Tsun Leung Chan; Edmond S K Ma
Journal:  Int J Mol Sci       Date:  2021-01-17       Impact factor: 5.923

  3 in total

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