The Effects of Oxybutynin on Urinary Symptoms in Children with Williams-Beuren Syndrome

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dc.contributorSistema FMUSP-HC: Faculdade de Medicina da Universidade de São Paulo (FMUSP) e Hospital das Clínicas da FMUSP
dc.contributor.authorSAMMOUR, Zein M.
dc.contributor.authorGOMES, Cristiano M.
dc.contributor.authorBESSA JR., Jose de
dc.contributor.authorPINHEIRO, Marcello S.
dc.contributor.authorKIM, Chong A.
dc.contributor.authorHONJO, Rachel S.
dc.contributor.authorTRIGO-ROCHA, Flavio E.
dc.contributor.authorBRUSCHINI, Homero
dc.contributor.authorSROUGI, Miguel
dc.date.accessioned2013-07-30T14:39:01Z
dc.date.available2013-07-30T14:39:01Z
dc.date.issued2012
dc.description.abstractPurpose: Williams-Beuren syndrome is a genomic disorder caused by a hemizygous contiguous gene deletion on chromosome 7q11.23. Lower urinary tract symptoms are common in children with Williams-Beuren syndrome. However, there are few data on the management of voiding symptoms in this population. We report our experience using oxybutynin to treat urinary symptoms in children with Williams-Beuren syndrome. Materials and Methods: We prospectively analyzed 42 patients with Williams-Beuren syndrome and significant lower urinary tract symptoms due to detrusor overactivity diagnosed on urodynamics in a 12-week, open-label study. Urological assessment included symptomatic evaluation, the impact of lower urinary tract symptoms on quality of life, frequency-volume chart, urodynamics and urinary tract sonography. After 12 weeks of treatment with 0.6 mg/kg oxybutynin per day given in 3 daily doses, patients were assessed for treatment efficacy and side effects. Results: A total of 17 girls and 19 boys completed medical therapy and were assessed at 12 weeks. Mean +/- SD patient age was 9.2 +/- 4.3 years (range 3 to 18). The most common urinary complaint was urgency, which occurred in 31 patients (86.1%), followed by urge incontinence, which was seen in 29 (80.5%). Compared to baseline, urinary symptoms were substantially improved. The negative impact of storage symptoms on quality of life was significantly decreased from a mean +/- SD of 3.3 +/- 1.7 to 0.5 +/- 0.9 (p <0.001). Mean +/- SD maximum urinary flow improved from 14.2 +/- 15.0 to 20.5 +/- 6.4 ml per second (p <0.001). Conclusions: A total of 12 weeks of therapy with 0.6 mg/kg oxybutynin daily resulted in improvement of lower urinary tract symptoms, quality of life and maximum flow rate in most patients with Williams-Beuren syndrome.
dc.description.indexMEDLINE
dc.identifier.citationJOURNAL OF UROLOGY, v.188, n.1, p.253-257, 2012
dc.identifier.doi10.1016/j.juro.2012.03.024
dc.identifier.issn0022-5347
dc.identifier.urihttps://observatorio.fm.usp.br/handle/OPI/318
dc.language.isoeng
dc.publisherELSEVIER SCIENCE INC
dc.relation.ispartofJournal of Urology
dc.rightsrestrictedAccess
dc.rights.holderCopyright ELSEVIER SCIENCE INC
dc.subjectlower urinary tract symptoms
dc.subjectoxybutynin
dc.subjecturination disorders
dc.subjecturodynamics
dc.subjectWilliams syndrome
dc.subject.othervoiding dysfunction
dc.subject.otherdetrusor overactivity
dc.subject.otherochoa syndrome
dc.subject.otherelastin locus
dc.subject.otherincontinence
dc.subject.otherhemizygosity
dc.subject.othertolterodine
dc.subject.otherdisorder
dc.subject.otherefficacy
dc.subject.otherbladder
dc.subject.wosUrology & Nephrology
dc.titleThe Effects of Oxybutynin on Urinary Symptoms in Children with Williams-Beuren Syndrome
dc.typearticle
dc.type.categoryoriginal article
dc.type.versionpublishedVersion
dspace.entity.typePublication
hcfmusp.citation.scopus5
hcfmusp.contributor.author-fmusphcZEIN MOHAMED SAMMOUR
hcfmusp.contributor.author-fmusphcCRISTIANO MENDES GOMES
hcfmusp.contributor.author-fmusphcMARCELLO SANTOS PINHEIRO
hcfmusp.contributor.author-fmusphcCHONG AE KIM
hcfmusp.contributor.author-fmusphcRACHEL SAYURI HONJO KAWAHIRA
hcfmusp.contributor.author-fmusphcFLAVIO EDUARDO TRIGO ROCHA
hcfmusp.contributor.author-fmusphcHOMERO BRUSCHINI
hcfmusp.contributor.author-fmusphcMIGUEL SROUGI
hcfmusp.description.beginpage253
hcfmusp.description.endpage257
hcfmusp.description.issue1
hcfmusp.description.volume188
hcfmusp.lim.ref2012
hcfmusp.origemWOS
hcfmusp.origem.pubmed22595063
hcfmusp.origem.scopus2-s2.0-84862144245
hcfmusp.origem.wosWOS:000305148900091
hcfmusp.publisher.cityNEW YORK
hcfmusp.publisher.countryUSA
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