Literature DB >> 23532946

Fraser syndrome: epidemiological study in a European population.

Ingeborg Barisic1, Ljubica Odak, Maria Loane, Ester Garne, Diana Wellesley, Elisa Calzolari, Helen Dolk, Marie-Claude Addor, Larraitz Arriola, Jorieke Bergman, Sebastiano Bianca, Patricia A Boyd, Elizabeth S Draper, Miriam Gatt, Martin Haeusler, Babak Khoshnood, Anna Latos-Bielenska, Bob McDonnell, Anna Pierini, Judith Rankin, Anke Rissmann, Annette Queisser-Luft, Christine Verellen-Dumoulin, David Stone, Romano Tenconi.   

Abstract

Fraser syndrome is a rare autosomal recessive disorder characterized by cryptophthalmos, cutaneous syndactyly, laryngeal, and urogenital malformations. We present a population-based epidemiological study using data provided by the European Surveillance of Congenital Anomalies (EUROCAT) network of birth defect registries. Between January 1990 and December 2008, we identified 26 cases of Fraser syndrome in the monitored population of 12,886,464 births (minimal estimated prevalence of 0.20 per 100,000 or 1:495,633 births). Most cases (18/26; 69%) were registered in the western part of Europe, where the mean prevalence is 1 in 230,695 births, compared to the prevalence 1 in 1,091,175 for the rest of Europe (P = 0.0003). Consanguinity was present in 7/26 (27%) families. Ten (38%) cases were liveborn, 14 (54%) pregnancies were terminated following prenatal detection of a serious anomaly, and 2 (8%) were stillborn. Eye anomalies were found in 20/24 (83%), syndactyly in 14/24 (58%), and laryngeal anomalies in 5/24 (21%) patients. Ambiguous genitalia were observed in 3/24 (13%) cases. Bilateral renal agenesis was present in 12/24 (50%) and unilateral in 4/24 (17%) cases. The frequency of anorectal anomalies was particularly high (42%). Most cases of Fraser syndrome (85%) are suspected prenatally, often due to the presence of the association of renal agenesis and cryptophthalmos. In the European population, a high proportion (82%) of pregnancies is terminated, thus reducing the live birth prevalence to a third of the total prevalence rate.
Copyright © 2013 Wiley Periodicals, Inc.

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Year:  2013        PMID: 23532946     DOI: 10.1002/ajmg.a.35839

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  12 in total

1.  Fraser Syndrome - a Case Report and Review of Literature.

Authors:  Adrian Dumitru; Mariana Costache; Anca Mihaela Lazaroiu; George Simion; Diana Secara; Monica Cirstoiu; Alina Emanoil; Tiberiu Augustin Georgescu; Maria Sajin
Journal:  Maedica (Bucur)       Date:  2016-03

Review 2.  Malformation syndromes associated with disorders of sex development.

Authors:  John M Hutson; Sonia R Grover; Michele O'Connell; Samuel D Pennell
Journal:  Nat Rev Endocrinol       Date:  2014-06-10       Impact factor: 43.330

3.  Syndactyly in a novel Fras1(rdf) mutant results from interruption of signals for interdigital apoptosis.

Authors:  Elizabeth A Hines; Jamie M Verheyden; Amber J Lashua; Sarah C Larson; Kelsey Branchfield; Eric T Domyan; Juan Gao; Julie F Harvey; John C Herriges; Linghan Hu; David J Mcculley; Kurt Throckmorton; Shigetoshi Yokoyama; Akihiro Ikeda; Guoliang Xu; Xin Sun
Journal:  Dev Dyn       Date:  2016-02-24       Impact factor: 3.780

4.  EUROCAT: an update on its functions and activities.

Authors:  F D Tucker; J K Morris; A Neville; E Garne; A Kinsner-Ovaskainen; M Lanzoni; M A Loane; S Martin; C Nicholl; J Rankin; A K Rissmann
Journal:  J Community Genet       Date:  2018-05-07

5.  3D Mapping Reveals a Complex and Transient Interstitial Matrix During Murine Kidney Development.

Authors:  Sarah N Lipp; Kathryn R Jacobson; David S Hains; Andrew L Schwarderer; Sarah Calve
Journal:  J Am Soc Nephrol       Date:  2021-04-19       Impact factor: 14.978

Review 6.  Congenital upper eyelid coloboma: embryologic, nomenclatorial, nosologic, etiologic, pathogenetic, epidemiologic, clinical, and management perspectives.

Authors:  Hatem A Tawfik; Mohamed H Abdulhafez; Yousef A Fouad
Journal:  Ophthalmic Plast Reconstr Surg       Date:  2015 Jan-Feb       Impact factor: 1.746

7.  Holt Oram syndrome: a registry-based study in Europe.

Authors:  Ingeborg Barisic; Ljubica Boban; Ruth Greenlees; Ester Garne; Diana Wellesley; Elisa Calzolari; Marie-Claude Addor; Larraitz Arriola; Jorieke Eh Bergman; Paula Braz; Judith Ls Budd; Miriam Gatt; Martin Haeusler; Babak Khoshnood; Kari Klungsoyr; Bob McDonnell; Vera Nelen; Anna Pierini; Annette Queisser-Wahrendorf; Judith Rankin; Anke Rissmann; Catherine Rounding; David Tucker; Christine Verellen-Dumoulin; Helen Dolk
Journal:  Orphanet J Rare Dis       Date:  2014-10-25       Impact factor: 4.123

8.  Bilateral anophthalmia and intrahepatic biliary atresia, two unusual components of Fraser syndrome: a case report.

Authors:  Muhamad Zakaria Brimo Alsaman; Sarab Agha; Hala Sallah; Rayan Badawi; Mohammad Nour Kitaz; Abdullah Assani; Hamdi Nawfal
Journal:  BMC Pregnancy Childbirth       Date:  2020-06-10       Impact factor: 3.007

Review 9.  Congenital Heart Defects and Ciliopathies Associated With Renal Phenotypes.

Authors:  George C Gabriel; Gregory J Pazour; Cecilia W Lo
Journal:  Front Pediatr       Date:  2018-06-15       Impact factor: 3.418

10.  Endoscopic Management of Lacrimal System Dysgenesis and Dacryocystoceles in Fraser Syndrome: A Case Report and Literature Review.

Authors:  Megan E Falls; Michael P Rabinowitz; Jacqueline R Carrasco; Mindy R Rabinowitz
Journal:  Allergy Rhinol (Providence)       Date:  2018-10-22
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