Literature DB >> 22015620

Nephrological findings and genotype-phenotype correlation in Beckwith-Wiedemann syndrome.

Alessandro Mussa1, Licia Peruzzi, Nicoletta Chiesa, Agostina De Crescenzo, Silvia Russo, Daniela Melis, Luigi Tarani, Giuseppina Baldassarre, Lidia Larizza, Andrea Riccio, Margherita Silengo, Giovanni Battista Ferrero.   

Abstract

Beckwith-Wiedemann syndrome (BWS), an overgrowth disorder with several congenital abnormalities, encompasses nephrourological anomalies. The objective of the report is to analyze the latter and related genotype-phenotype correlations. The study was a retrospective review of nephrourological investigations and genotype in 67 BWS patients. Imaging and laboratory studies have been correlated with the molecular anomalies typical of BWS. Thirty-eight (56.7%) patients had a total of 61 nonmalignant nephrourological findings, including nephromegaly (n = 24), collecting system abnormalities (n = 14), cryptorchidism (n = 11), nephrolithiasis (n = 5), cysts (n = 5), and dysplasia (n = 1). Four patients had Wilms' tumor, all associated with renal hyperplasia. Renal findings were almost consistent in the BWS(IC1) group, with nephromegaly in all patients and collecting system abnormalities in half of them. BWS(UPD) and negative patients also had frequent anomalies (63.6% and 61.9% respectively), whereas only 36.0% of BWS(IC2) had renal findings (p = 0.003). Cryptorchidism was associated with abdominal wall defects (p < 0.001) appearing more frequently in BWS(IC2) (p = 0.028). Urinary tract infections were observed in 17.9% of patients, with two resulting in life-threatening sepsis. Hypercalciuria was present in 10% of cases. 55.5% of BWS patients have renal findings. Although variegate, these anomalies disclose a genotype-phenotype correlation.

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Year:  2011        PMID: 22015620     DOI: 10.1007/s00467-011-2009-4

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  28 in total

1.  Feasibility of partial nephrectomy for Wilms' tumor in children with Beckwith-Wiedemann syndrome who have been screened with abdominal ultrasonography.

Authors:  D Elizabeth McNeil; Jacob C Langer; Peter Choyke; Michael R DeBaun
Journal:  J Pediatr Surg       Date:  2002-01       Impact factor: 2.545

2.  Molecular subtypes and phenotypic expression of Beckwith-Wiedemann syndrome.

Authors:  Wendy N Cooper; Anita Luharia; Gail A Evans; Hussain Raza; Antonita C Haire; Richard Grundy; Sarah C Bowdin; Andrea Riccio; Gianfranco Sebastio; Jet Bliek; Paul N Schofield; Wolf Reik; Fiona Macdonald; Eamonn R Maher
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3.  Wiedemann-Beckwith syndrome: presentation of clinical and cytogenetic data on 22 new cases and review of the literature.

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Journal:  Hum Genet       Date:  1986-10       Impact factor: 4.132

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Journal:  J Med Genet       Date:  1994-07       Impact factor: 6.318

Review 5.  Surveillance for Wilms tumour in at-risk children: pragmatic recommendations for best practice.

Authors:  R H Scott; L Walker; Ø E Olsen; G Levitt; I Kenney; E Maher; C M Owens; K Pritchard-Jones; A Craft; N Rahman
Journal:  Arch Dis Child       Date:  2006-07-20       Impact factor: 3.791

6.  Risk of cancer during the first four years of life in children from The Beckwith-Wiedemann Syndrome Registry.

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Journal:  J Pediatr       Date:  1998-03       Impact factor: 4.406

7.  Hypercalciuria in Beckwith-Wiedemann syndrome.

Authors:  Michael Goldman; Cheryl Shuman; Rosanna Weksberg; Norman D Rosenblum
Journal:  J Pediatr       Date:  2003-02       Impact factor: 4.406

8.  Follow-up study of patients with Wiedemann-Beckwith syndrome with emphasis on the change in facial appearance over time.

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Journal:  Am J Med Genet       Date:  1994-06-01

9.  MS-MLPA is a specific and sensitive technique for detecting all chromosome 11p15.5 imprinting defects of BWS and SRS in a single-tube experiment.

Authors:  Manuela Priolo; Angela Sparago; Corrado Mammì; Flavia Cerrato; Carmelo Laganà; Andrea Riccio
Journal:  Eur J Hum Genet       Date:  2008-01-23       Impact factor: 4.246

10.  Beckwith-Wiedemann syndrome: role of ultrasound in its management.

Authors:  K J Shah
Journal:  Clin Radiol       Date:  1983-05       Impact factor: 2.350

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  18 in total

1.  Refractory arterial hypertension and renal failure combined with cerebral seizures and pancytopenia in a 5-year-old girl with bilateral nephromegaly: Answers.

Authors:  Patrick Hundsdoerfer; Uwe Querfeld
Journal:  Pediatr Nephrol       Date:  2015-08-11       Impact factor: 3.714

2.  Beckwith-Wiedemann syndrome in diverse populations.

Authors:  Kelly A Duffy; Brian J Sajorda; Alice C Yu; Evan R Hathaway; Katheryn L Grand; Matthew A Deardorff; Jennifer M Kalish
Journal:  Am J Med Genet A       Date:  2019-02-04       Impact factor: 2.802

3.  Revisiting Wilms tumour surveillance in Beckwith-Wiedemann syndrome with IC2 methylation loss, reply.

Authors:  Frédéric Brioude; Raoul Hennekam; Jet Bliek; Carole Coze; Thomas Eggermann; Giovanni B Ferrero; Christian Kratz; Yves Le Bouc; Saskia M Maas; Deborah J G Mackay; Eamonn R Maher; Alessandro Mussa; Irene Netchine
Journal:  Eur J Hum Genet       Date:  2018-02-15       Impact factor: 4.246

4.  (Epi)genotype-phenotype correlations in Beckwith-Wiedemann syndrome.

Authors:  Alessandro Mussa; Silvia Russo; Agostina De Crescenzo; Andrea Freschi; Luciano Calzari; Silvia Maitz; Marina Macchiaiolo; Cristina Molinatto; Giuseppina Baldassarre; Milena Mariani; Luigi Tarani; Maria Francesca Bedeschi; Donatella Milani; Daniela Melis; Andrea Bartuli; Maria Vittoria Cubellis; Angelo Selicorni; Margherita Cirillo Silengo; Lidia Larizza; Andrea Riccio; Giovanni Battista Ferrero
Journal:  Eur J Hum Genet       Date:  2015-04-22       Impact factor: 4.246

Review 5.  Genetics of pediatric renal tumors.

Authors:  Brigitte Royer-Pokora
Journal:  Pediatr Nephrol       Date:  2012-03-30       Impact factor: 3.714

6.  Characterization of the Beckwith-Wiedemann spectrum: Diagnosis and management.

Authors:  Kelly A Duffy; Christopher M Cielo; Jennifer L Cohen; Christina X Gonzalez-Gandolfi; Jessica R Griff; Evan R Hathaway; Jonida Kupa; Jesse A Taylor; Kathleen H Wang; Arupa Ganguly; Matthew A Deardorff; Jennifer M Kalish
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7.  Urological Findings in Beckwith-Wiedemann Syndrome With Chromosomal Duplications of 11p15.5: Evaluation and Management.

Authors:  Carmen C Tong; Kelly A Duffy; David I Chu; Dana A Weiss; Arun K Srinivasan; Douglas A Canning; Jennifer M Kalish
Journal:  Urology       Date:  2016-09-07       Impact factor: 2.649

8.  Primary renal diffuse large B-Cell lymphoma causing haemodialysis-dependent nephromegaly in a child.

Authors:  Andrew Michael South
Journal:  BMJ Case Rep       Date:  2018-09-26

9.  The effectiveness of Wilms tumor screening in Beckwith-Wiedemann spectrum.

Authors:  Alessandro Mussa; Kelly A Duffy; Diana Carli; Jessica R Griff; Riccardo Fagiano; Jonida Kupa; Garrett M Brodeur; Giovanni Battista Ferrero; Jennifer M Kalish
Journal:  J Cancer Res Clin Oncol       Date:  2019-10-04       Impact factor: 4.553

10.  Diagnosis of Beckwith-Wiedemann syndrome in children presenting with Wilms tumor.

Authors:  Suzanne P MacFarland; Kelly A Duffy; Tricia R Bhatti; Rochelle Bagatell; Naomi J Balamuth; Garrett M Brodeur; Arupa Ganguly; Peter A Mattei; Lea F Surrey; Frank M Balis; Jennifer M Kalish
Journal:  Pediatr Blood Cancer       Date:  2018-06-22       Impact factor: 3.167

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