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Article: Hirschsprung's disease: A more generalised neuropathy?

TitleHirschsprung's disease: A more generalised neuropathy?
Authors
KeywordsGlial cell-derived neurotrophic factor
Hirschsprung's disease
Neurotrophin factor 3
Oto-acoustic emission
Issue Date2001
PublisherWB Saunders Co. The Journal's web site is located at http://www.elsevier.com/locate/jpedsurg
Citation
Journal Of Pediatric Surgery, 2001, v. 36 n. 2, p. 296-300 How to Cite?
AbstractBackground/Purpose: Aberrant neural crest migration is implicated in the pathogenesis of hindgut aganglionosis in Hirschsprung's disease (HSCR). Neural crest progenitors also give rise to a variety of cell types outside the gut, including nerves of the inner ear and peripheral nervous system. This study aims to determine whether such nerves are also affected in HSCR. Methods: Pure tone audiometry and oto-acoustic emission tests were performed on 53 patients with Hirschsprung's disease and 28 age-matched controls. Forty-two children with Hirschsprung's disease and 30 age-matched controls also underwent neurologic evaluation, including quantitative peripheral tests of sensory function. Results: Moderate to severe sensorineural hearing loss (1 profound [Waardenburg syndrome] and 3 high frequency) was detected in 4 children (8%) from the Hirschsprung's group. Abnormal (absent or reduced) oto-acoustic emission was detected in 77 of 102 ears (41 of 51 patients) in the HSCR group and 8 out of 48 ears (6 of 24 children) in the control group (75% v 17%; P < .0001). Two HSCR children had marked abnormalities on tests of peripheral nerve function. Significantly higher thresholds for vibration sensation were detected in HSCR versus controls (P < .05). Conclusion: Hirschsprung's disease may have a more generalised neuropathy than just that involving the affected bowel. Copyright © 2001 by W.B. Saunders Company.
Persistent Identifierhttp://hdl.handle.net/10722/84354
ISSN
2015 Impact Factor: 1.733
2015 SCImago Journal Rankings: 0.802
ISI Accession Number ID
References

 

DC FieldValueLanguage
dc.contributor.authorCheng, Wen_HK
dc.contributor.authorAu, DKKen_HK
dc.contributor.authorKnowles, CHen_HK
dc.contributor.authorAnand, Pen_HK
dc.contributor.authorTam, PKHen_HK
dc.date.accessioned2010-09-06T08:51:57Z-
dc.date.available2010-09-06T08:51:57Z-
dc.date.issued2001en_HK
dc.identifier.citationJournal Of Pediatric Surgery, 2001, v. 36 n. 2, p. 296-300en_HK
dc.identifier.issn0022-3468en_HK
dc.identifier.urihttp://hdl.handle.net/10722/84354-
dc.description.abstractBackground/Purpose: Aberrant neural crest migration is implicated in the pathogenesis of hindgut aganglionosis in Hirschsprung's disease (HSCR). Neural crest progenitors also give rise to a variety of cell types outside the gut, including nerves of the inner ear and peripheral nervous system. This study aims to determine whether such nerves are also affected in HSCR. Methods: Pure tone audiometry and oto-acoustic emission tests were performed on 53 patients with Hirschsprung's disease and 28 age-matched controls. Forty-two children with Hirschsprung's disease and 30 age-matched controls also underwent neurologic evaluation, including quantitative peripheral tests of sensory function. Results: Moderate to severe sensorineural hearing loss (1 profound [Waardenburg syndrome] and 3 high frequency) was detected in 4 children (8%) from the Hirschsprung's group. Abnormal (absent or reduced) oto-acoustic emission was detected in 77 of 102 ears (41 of 51 patients) in the HSCR group and 8 out of 48 ears (6 of 24 children) in the control group (75% v 17%; P < .0001). Two HSCR children had marked abnormalities on tests of peripheral nerve function. Significantly higher thresholds for vibration sensation were detected in HSCR versus controls (P < .05). Conclusion: Hirschsprung's disease may have a more generalised neuropathy than just that involving the affected bowel. Copyright © 2001 by W.B. Saunders Company.en_HK
dc.languageengen_HK
dc.publisherWB Saunders Co. The Journal's web site is located at http://www.elsevier.com/locate/jpedsurgen_HK
dc.relation.ispartofJournal of Pediatric Surgeryen_HK
dc.subjectGlial cell-derived neurotrophic factoren_HK
dc.subjectHirschsprung's diseaseen_HK
dc.subjectNeurotrophin factor 3en_HK
dc.subjectOto-acoustic emissionen_HK
dc.titleHirschsprung's disease: A more generalised neuropathy?en_HK
dc.typeArticleen_HK
dc.identifier.openurlhttp://library.hku.hk:4550/resserv?sid=HKU:IR&issn=0022-3468&volume=36&issue=2&spage=296&epage=300&date=2001&atitle=Hirschsprung%27s+disease:+a+more+generalised+neuropathy?en_HK
dc.identifier.emailAu, DKK: kinkwau@hkucc.hku.hken_HK
dc.identifier.emailTam, PKH: paultam@hkucc.hku.hken_HK
dc.identifier.authorityAu, DKK=rp00385en_HK
dc.identifier.authorityTam, PKH=rp00060en_HK
dc.description.naturelink_to_subscribed_fulltexten_US
dc.identifier.doi10.1053/jpsu.2001.20695en_HK
dc.identifier.pmid11172419en_HK
dc.identifier.scopuseid_2-s2.0-0035141282en_HK
dc.identifier.hkuros71957en_HK
dc.relation.referenceshttp://www.scopus.com/mlt/select.url?eid=2-s2.0-0035141282&selection=ref&src=s&origin=recordpageen_HK
dc.identifier.volume36en_HK
dc.identifier.issue2en_HK
dc.identifier.spage296en_HK
dc.identifier.epage300en_HK
dc.identifier.isiWOS:000166697800017-
dc.publisher.placeUnited Statesen_HK
dc.identifier.scopusauthoridCheng, W=7402169228en_HK
dc.identifier.scopusauthoridAu, DKK=7004909224en_HK
dc.identifier.scopusauthoridKnowles, CH=26643431700en_HK
dc.identifier.scopusauthoridAnand, P=35472567900en_HK
dc.identifier.scopusauthoridTam, PKH=7202539421en_HK

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