Generic and disease-specific health-related quality of life in patients with Hirschsprung disease: A systematic review and meta-analysis

Table 1 Characteristics of included studies

Ref.	Study design	Sample size	Self- report	Parental proxy report	HRQoL instrument	Age range (yr)	Mean age (yr)	Sex (% male)	Operation-technique	Segment- length (% short)	Syndromal comorbidity (%)	Stoma present (%)	Postoperative complications (%)
Amin et al[50], 2018	Cohort	46	0	46	PedsQL	0-18	4.2	71		80
Baayen et al[70], 2017	Cohort	50	50	0	HAQL	12-16						6
Cavusoglu et al[46], 2012	Case-control	12	6	12	PedsQL	2-12	4.7	84
Collins et al[26], 2017	Cohort	60	0	60	PedsQL	2-10	6.4	82	13% Duhamel 80% Trans- anal PT	78	12	13
Espeso et al[21], 2019	Cohort	63			HAQL	6-18	10.8	70	22% Duhamel 78% Trans- anal PT	79	0	0	44
Hartman et al[53], 2007	Cohort	121	121	0	HAQL / TACQoL	8-16	11.4	82		69	0	4
Hartman et al[49], 2008	Cohort	152	152	0	TACQoL	8-16	11.6	80		68	0	3
Khalil et al[47], 2015	Cohort	53	0	53	PedsQL	5-7	5.9	70					3
Lane et al[52], 2016	Cohort	118	0	118	PedsQL	0-17
Meinds et al[17], 2019	Cohort	150	0	150	CHQ-CF87	8-17		79	62% Duhamel 16% Trans- anal PT	84		0	31
Mills et al[25], 2008	Cohort	51	37	44	PedsQL	3-21	9.8	82	15% Duhamel 77% Trans- anal PT	66	6
Nah et al[33], 2018	Case-control	44	21	42	PedsQL	2-20	9.1	75		73
Neuvonen et al[23], 2017	Case-control	39	27	12	PedsQL	0-17		75	96% Trans- anal PT	84	24	5	47
Roorda et al[61], 2018	Cohort	18 /14	7	7	HAQL / CHQ-CF87 / CHQ-PF50	4-17		77	67% Duhamel	0	13	6
Sood et al[48], 2018	Cohort	58	38	58	PedsQL	11-18	14.5	84	7% Duhamel 86% Trans- anal PT	84	12	7	28
Townley et al[67], 2019	Cohort	56	0	56	PedsQL	0-13	5.4	80	84% Duhamel 16% Trans- anal PT	77		14
Xi et al[50], 2018	Case-control	50	0	50	TACQoL	6	6.0	76	100% Trans- anal PT

HRQoL: Health-related quality of life; HAQL: Hirschsprung disease/anorectal malformation quality of life.