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Background: It is unclear whether functional outcomes improve or deteriorate with age following surgery for Hirschsprung's disease. The aim of this cross-sectional study was to determine the long-term functional outcomes and quality of life (QoL) in patients with Hirschsprung's disease.

Methods: Patients with pathologically proven Hirschsprung's disease older than 7 years were included. Patients with a permanent stoma or intellectual disability were excluded. Functional outcomes were assessed according to the Rome IV criteria using the Defaecation and Faecal Continence questionnaire. QoL was assessed by means of the Child Health Questionnaire Child Form 87 or World Health Organization Quality of Life questionnaire 100. Reference data from healthy controls were available for comparison.

Results: Of 619 patients invited, 346 (55·9 per cent) responded, with a median age of 18 (range 8-45) years. The prevalence of constipation was comparable in paediatric and adult patients (both 22·0 per cent), and in patients and controls. Compared with controls, adults with Hirschsprung's disease significantly more often experienced straining (50·3 versus 36·1 per cent; P = 0·011) and incomplete evacuation (47·4 versus 27·2 per cent; P < 0·001). The prevalence of faecal incontinence, most commonly soiling, was lower in adults than children with Hirschsprung's disease (16·8 versus 37·6 per cent; P < 0·001), but remained higher than in controls (16·8 versus 6·1 per cent; P = 0·003). Patients with poor functional outcomes scored significantly lower in several QoL domains.

Conclusion: This study has shown that functional outcomes are better in adults than children, but symptoms of constipation and soiling persist in a substantial group of adults with Hirschsprung's disease. The persistence of defaecation problems is an indication that continuous care is necessary in this specific group of patients.

Titel:	Long-term functional outcomes and quality of life in patients with Hirschsprung's disease.
Autor/in / Beteiligte Person:	Meinds, RJ ; van der Steeg AFW ; Sloots, CEJ ; Witvliet, MJ ; de Blaauw I ; van Gemert WG ; Trzpis, M ; Broens, PMA
Zeitschrift:	The British journal of surgery, Jg. 106 (2019-03-01), Heft 4, S. 499-507
Veröffentlichung:	2021- : [Oxford] : Oxford University Press on behalf of BJS Foundation Ltd. ; <i>Original Publication</i>: [Bristol, England : Baltimore : John Wright & Sons ; Williams & Wilkins Co., 2019
Medientyp:	academicJournal
ISSN:	1365-2168 (electronic)
DOI:	10.1002/bjs.11059
Schlagwort:	Adolescent Adult Age Factors Analysis of Variance Case-Control Studies Child Female Follow-Up Studies Hirschsprung Disease diagnosis Humans Logistic Models Male Prognosis Recovery of Function Risk Assessment Time Factors Treatment Outcome Hirschsprung Disease surgery Quality of Life Rectum surgery Surveys and Questionnaires
Sonstiges:	Nachgewiesen in: MEDLINE Sprachen: English Publication Type: Comparative Study; Journal Article Language: English [Br J Surg] 2019 Mar; Vol. 106 (4), pp. 499-507. <i>Date of Electronic Publication: </i>2019 Jan 17. MeSH Terms: Quality of Life* ; Surveys and Questionnaires* ; Hirschsprung Disease / surgery ; Rectum / surgery ; Adolescent ; Adult ; Age Factors ; Analysis of Variance ; Case-Control Studies ; Child ; Female ; Follow-Up Studies ; Hirschsprung Disease / diagnosis ; Humans ; Logistic Models ; Male ; Prognosis ; Recovery of Function ; Risk Assessment ; Time Factors ; Treatment Outcome References: J Pediatr Surg. 2017 Mar;52(3):449-453. (PMID: 27836370) ; Gastroenterology. 2016 Mar 25;:. (PMID: 27144630) ; Ann Surg. 2010 Dec;252(6):977-81. (PMID: 21107107) ; Eur J Pediatr Surg. 2008 Feb;18(1):38-43. (PMID: 18302068) ; Eur J Pediatr Surg. 2013 Apr;23(2):94-102. (PMID: 23572464) ; Scand J Gastroenterol. 2018 Jun - Jul;53(7):790-796. (PMID: 29703095) ; J Pediatr Surg. 2010 Jun;45(6):1213-20. (PMID: 20620323) ; J Pediatr Surg. 1997 Oct;32(10):1443-6. (PMID: 9349764) ; Expert Rev Pharmacoecon Outcomes Res. 2007 Aug;7(4):393-401. (PMID: 20528422) ; Soc Sci Med. 1998 Jun;46(12):1569-85. (PMID: 9672396) ; Ann Surg. 2017 Mar;265(3):622-629. (PMID: 28169931) ; Arch Dis Child. 2011 Apr;96(4):398-406. (PMID: 20371581) ; Semin Pediatr Surg. 2012 Nov;21(4):336-43. (PMID: 22985839) ; J Gastroenterol Hepatol. 2007 Dec;22(12):2273-82. (PMID: 18031392) ; Eur J Pediatr. 2019 Jan;178(1):33-39. (PMID: 30264351) ; J Pediatr Surg. 2010 Dec;45(12):2398-402. (PMID: 21129554) ; Aliment Pharmacol Ther. 2010 May;31(9):938-49. (PMID: 20180788) ; J Pediatr Surg. 2017 Sep;52(9):1446-1450. (PMID: 27765267) ; J Pediatr Surg. 2014 Oct;49(10):1488-92. (PMID: 25280652) ; J Pediatr Surg. 2017 Sep;52(9):1458-1464. (PMID: 28094015) ; Pediatr Surg Int. 2006 Dec;22(12):987-90. (PMID: 17006709) ; Eur J Pediatr Surg. 2016 Apr;26(2):192-9. (PMID: 25643245) ; World J Gastroenterol. 2009 Jul 14;15(26):3276-82. (PMID: 19598304) ; J Adolesc Health. 2006 Jul;39(1):4-13. (PMID: 16781955) ; Pediatr Surg Int. 2006 Apr;22(4):319-25. (PMID: 16485101) ; Curr Opin Pediatr. 2015 Jun;27(3):364-9. (PMID: 25944307) ; J Pediatr Surg. 2008 May;43(5):899-905. (PMID: 18485963) ; Int J Colorectal Dis. 2017 Apr;32(4):475-483. (PMID: 27913883) ; Eur J Pediatr Surg. 2018 Feb;28(1):81-88. (PMID: 28958095) ; Gastroenterology. 2016 Feb 18;:. (PMID: 27144627) Entry Date(s): Date Created: 20190118 Date Completed: 20190905 Latest Revision: 20231012 Update Code: 20240513 PubMed Central ID: PMC6590339

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Long-term functional outcomes and quality of life in patients with Hirschsprung's disease.