Fanconi anemia and its diagnosis.

Fanconi anemia (FA) is a genetically and phenotypically heterogeneous recessive disorder characterized by diverse congenital malformations, progressive pancytopenia, and predisposition to both hematologic malignancies and solid tumors. Congenital anomalies vary from patient to patient and may affect skeletal morphogenesis as well as any of the major organ systems. Although this highly variable phenotype makes accurate diagnosis on the basis of clinical manifestations difficult in some patients, laboratory study of chromosomal breakage induced by diepoxybutane (DEB) or other crosslinking agents provides a unique cellular marker for the diagnosis of the disorder either prenatally or postnatally. Diagnosis based on abnormal response to DNA crosslinking agents can be used to identify the pre-anemia patient as well as patients with aplastic anemia or leukemia who may or may not have the physical stigmata associated with the syndrome. This overview will present our current knowledge regarding the varied phenotypic manifestations of FA and procedures for diagnosis based upon abnormal DNA damage responses.

[1]  R. Gale,et al.  Hematologic abnormalities in Fanconi anemia: an International Fanconi Anemia Registry study. , 1994, Blood.

[2]  A. Auerbach Fanconi anemia diagnosis and the diepoxybutane (DEB) test. , 1993, Experimental hematology.

[3]  B. Alter Cancer in Fanconi anemia, 1927–2001 , 2003, Cancer.

[4]  P. Rosenberg,et al.  Cancer risks in Fanconi anemia: findings from the German Fanconi Anemia Registry , 2008, Haematologica.

[5]  A. Auerbach,et al.  Fanconi anemia: prenatal diagnosis in 30 fetuses at risk. , 1985, Pediatrics.

[6]  R. Chaganti,et al.  Prenatal and postnatal diagnosis and carrier detection of Fanconi anemia by a cytogenetic method. , 1981, Pediatrics.

[7]  W. Dameshek,et al.  Familial hypoplastic anemia of childhood; report of eight cases in two families with beneficial effect of splenectomy in one case. , 1947, American journal of diseases of children.

[8]  A. Auerbach,et al.  International Fanconi Anemia Registry: relation of clinical symptoms to diepoxybutane sensitivity. , 1989 .

[9]  W. Ebell,et al.  Clonal Chromosomal Aberrations in Bone Marrow Cells of Fanconi Anemia Patients: Results and Implications , 2007 .

[10]  J. Kurtzberg,et al.  Hematopoietic reconstitution in a patient with Fanconi's anemia by means of umbilical-cord blood from an HLA-identical sibling. , 1989, The New England journal of medicine.

[11]  A. Rossi,et al.  Fanconi anemia in brothers initially diagnosed with VACTERL association with hydrocephalus, and subsequently with Baller-Gerold syndrome. , 1996, American journal of medical genetics.

[12]  A. Auerbach,et al.  International Fanconi Anemia Registry: First Report , 1989 .

[13]  J. Liu,et al.  Fanconi anemia presenting unexpectedly in an adult kindred with no dysmorphic features. , 1991, The American journal of medicine.

[14]  P. Rosenberg,et al.  Cancer incidence in persons with Fanconi anemia. , 2003, Blood.

[15]  S. Batish,et al.  GST genotype may modify clinical phenotype in patients with Fanconi anaemia , 2005, British journal of haematology.

[16]  J. Shah,et al.  High incidence of head and neck squamous cell carcinoma in patients with Fanconi anemia. , 2003, Archives of otolaryngology--head & neck surgery.

[17]  A. Auerbach,et al.  Clastogen-induced chromosomal breakage as a marker for first trimester prenatal diagnosis of Fanconi anemia , 1986, Human Genetics.

[18]  R. Gale,et al.  Hematologic Abnormalities in Fanconi Anemia: An International Fanconi Anemia Registry Study , 1994 .

[19]  J. Satagopan,et al.  Re: Human Papillomavirus DNA and p53 Polymorphisms in Squamous Cell Carcinomas From Fanconi Anemia Patients , 2004 .

[20]  J. Wagner,et al.  Successful hematopoietic stem cell transplantation for Fanconi anemia from an unaffected HLA-genotype-identical sibling selected using preimplantation genetic diagnosis. , 2004, Blood.

[21]  David A. Williams,et al.  Small pituitary size in children with Fanconi Anemia , 2007, Pediatric blood & cancer.

[22]  Marianne Berwick,et al.  A 20-year perspective on the International Fanconi Anemia Registry (IFAR). , 2003, Blood.

[23]  C. Strom,et al.  Preimplantation diagnosis for Fanconi anemia combined with HLA matching. , 2001, JAMA.

[24]  T. M. Schroeder,et al.  Spontane Chromosomenaberrationen bei familiärer Panmyelopathie , 2004, Humangenetik.

[25]  A D Auerbach,et al.  Phenotypic consequences of mutations in the Fanconi anemia FAC gene: an International Fanconi Anemia Registry study. , 1997, Blood.

[26]  S. Batish,et al.  Evaluation of Growth and Hormonal Status in Patients Referred to the International Fanconi Anemia Registry , 2001, Pediatrics.

[27]  N. Akar,et al.  Spectrum of anomalies in Fanconi anaemia. , 1984, Journal of medical genetics.

[28]  H. Hoehn,et al.  Comparative evaluation of diepoxybutane sensitivity and cell cycle blockage in the diagnosis of Fanconi anemia. , 1995, Blood.

[29]  D. D’Alessio,et al.  Abnormalities in glucose tolerance are common in children with fanconi anemia and associated with impaired insulin secretion , 2008, Pediatric blood & cancer.

[30]  J. Satagopan,et al.  A note on competing risks in survival data analysis , 2004, British Journal of Cancer.

[31]  H. Broxmeyer,et al.  Prenatal identification of potential donors for umbilical cord blood transplantation for Fanconi anemia , 1990, Transfusion.

[32]  P. Giampietro,et al.  Diagnosis of Fanconi anemia in patients without congenital malformations: an international Fanconi Anemia Registry Study. , 1997, American journal of medical genetics.

[33]  C. Stratakis,et al.  Endocrine abnormalities in patients with Fanconi anemia. , 2007, The Journal of clinical endocrinology and metabolism.

[34]  P. Giampietro,et al.  The need for more accurate and timely diagnosis in Fanconi anemia: a report from the International Fanconi Anemia Registry. , 1993, Pediatrics.

[35]  R. Mueller,et al.  Improved growth velocity during thyroid hormone therapy in children with Fanconi anemia and borderline thyroid function , 2008, Pediatric blood & cancer.

[36]  J. Wagner,et al.  Somatic mosaicism in Fanconi anemia: Evidence of genotypic reversion in lymphohematopoietic stem cells , 2001, Proceedings of the National Academy of Sciences of the United States of America.

[37]  M. Freedman,et al.  Fanconi's anaemia and pregnancy , 1991, British journal of haematology.

[38]  C. Mathew,et al.  Somatic Mosaicism in Fanconi Anemia: Molecular Basis and Clinical Significance , 1997, European journal of human genetics : EJHG.