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Table 1 Characteristics and quality appraisal of included studies

From: Long-term outcomes and quality of life of patients with Hirschsprung disease: a systematic review and meta-analysis

AuthorYearCountryStudy designAge range of patients (years)Number of patients older than 10 yearsRectosigmoid aganglionosis (n)Long-segment (n)Total colonic aganglionosis (n)Surgery approachAge at surgery (Mean, interquartile range), (year)Associated congenital disease or syndrome (n)Fecal incontinence (n)Constipation (n)Urinary system dysfunction (n)Quality of appraisal (NOS/AHRQ checklist) a
Granström et al. [25]2015SwedenCase-control20–433937/2Soave, Duhamel, Sphincteromyectomy, Ileostomy, Sigmoid colostomy.1 (1, 17)Down syndrome (n = 1), hypospadias (n = 2), congenital central hypoventilation syndrome (n = 1) and Fallot’s striad (n = 1)//3S: 3, C: 1, E: 3
Total: 7
Jarvi et al. [21]2010FinlandCase-control35–48897941Soave, Duhamel, State Rehbein, Swenson, Colonal pull-through.2.2 (0.8, 2.0)Down syndrome (n = 1), cartilage hair hypoplasia (n = 1), and multiple endocrine neoplasia type 2 (n = 1).1227/S: 3, C: 1, E: 2
Total: 6
Neuvonen et al. [22]2017FinlandCase-control18–32336673Transanal endorectal pull-through (TEPT), TEPT with laparotomy/laparoscopy, Ileoanal pull-through; Definitive endostomy.0.35 (0, 9.8)Down syndrome (n = 11), Mowat-Wilson syndrome (n = 4), cartilage-hairhypoplasia (n = 2), Currarino syndrome (n = 1), Waardenburg-Shah syndrome (n = 1), and marker-chromosome syndrome (n = 1).12/S: 4, C: 1, E: 2
Total: 7
Diseth et al. [15]1997NorwayCase-control10–201912/3Duhamel0.7 (0.1, 5)Unclear63/S: 3, C: 1, E: 3
Total: 7
Conway et al. [29]2007UKCohort study16–237863150Duhamel0.75Unclear///S: 2, C: 0, O: 2
Total: 4
Athanasakos et al. [27]2006AustraliaCross-sectional13–242350814Soave, Duhamel.UnclearDown syndrome (n = 2)115/Yes: 6, No: 0, Unclear: 5
Heikkinen et al. 1995 & Heikkinen et al. 1997 [23, 24]1995Finland & UKCohort study24–381009541Duhamel, Swenson, State-Rehbein, Soave.<  1 year old: 38, 1–3 years old: 37, older than 3 years old: 25Down syndrome (n = 1), Chondroectodermal hypoplasia (n = 1), Marfan’s syndrome (n = 1).141/S: 3, C: 1, O: 2
Total: 6
Mills et al. [30]2008CanadaCross-sectional13–181634103Duhamel, Soave, Swenson, Transnal.UnclearDown syndrome (n = 3), cardiac anomalies (n = 2). Cleft palate (n = 1), ureteric reflux (n = 1), autism (n = 1), inguinal hernia (n = 1), congenital ptosis (n = 1), hypothyroidism (n = 1), and pyloric stenosis (n = 1),110Yes: 8, No: 1, Unclear: 2
Catto-Smith et al. [28]2007AustraliaCross-sectional12–2732///Soave, Duhamel, Swenson, Unknown.UnclearDown syndrome (n = 10)7184Yes: 8, No: 1, Unclear: 2
Ieiri et al. [31]2010JapanCross-sectional19–55433443Z-shaped anastomosis (modified Duhamel procedure), Swenson, Martin.UnclearUnclear1525Yes: 7, No: 1, Unclear: 3
Gunnarsdo’ttir et al. [4, 26]2010SwedenCross-sectional18–45422983Duhamel, Duhamel-Martin modification, Ileorectal anastomosis.0.58 (0,11.6)Down syndrome (n = 6)1352Yes: 9, No: 0, Unclear: 2
Niramis et al. [32]2008ThailandCross-sectional10–1911195160Duhamel, Soave, Swenson.10–15 years old: 1 (0.5,13); older than 15 years old: 1.3 (0.6, 11)Down syndrome (n = 9), cerebral palsy (n = 4) b1711/Yes: 6, No: 2, Unclear: 3
  1. aThe NOS checklist was used to evaluate cohort studies and case-control studies, while the AHRQ checklist was used for cross-sectional studies. For cohort and case-control study, the total score of the NOS is 9, with higher score indicating higher-quality. S: Selection, C: Comparability, E: Exposure, O: Outcome. Score lower than 5 was considered low, 5–6 medium, and 7–9 high quality. For cross-sectional study, the overall quality of reports of the included studies was marginal to fair (Additional file 3). For example, most of the studies failed to report whether the patients were consecutively enrolled, which could be subjective to selection bias
  2. bIn this study, patients with associated malformations were excluded for statistical analysis