Table 1.
Key characteristics of the included studies
| Author | Year of Publication | Country | Study design | Number of patients pre-OP | S-HD | L-HD | TCA | Surgical procedure | EC pre-OP | EC post-OP | Congenital anomalies |
|---|---|---|---|---|---|---|---|---|---|---|---|
| Carcassonne et al | 1989 | France | Cohort study | 32 | 22 | 7 | 2 | Swenson, Soave, Duhamel | 11 | 4 | / |
| Carneiro et al | 1992 | UK | Cohort study | 76 | 61 | 8 | 7 | Swenson, Soave, Duhamel | 24 | 7 | Down' syndrome (n = 8), cardiac defects (n = 8), gastrointestinal (n = 4), genitourinary (n = 4), central nervous system (n = 3) |
| Downey et al | 2015 | USA | Cohort study | 24 | 21 | 2 | 4 | Soave, Duhamel | 7 | 7 | Down Syndrome (n = 7), Turner Syndrome (n = 1), cardiac defects (n = 4), central hypoventilation syndrome (n = 1), esophageal atresia (n = 1), duodenal atresia (n = 1), omphalocele (n = 1), renal duplication (n = 1) |
| Elhalaby et al | 1995 | USA | Cohort study | 168 | 124 | 27 | 10 | Swenson, Soave, Duhamel | 21 | 44 | Down' Syndrome (n = 16), other (n = 31) |
| Fortuna et al | 1996 | USA | Cohort study | 82 | 66 | 11 | 5 | Soave, Duhamel | 10 | 20 | Down' Syndrome (n = 7) |
| Foster et al | 1990 | USA | Cohort study | 63 | 44 | 5 | 9 | Swenson, Soave, Duhamel | 6 | 4 | / |
| Harrison et al | 1986 | USA | Cohort study | 139 | 95 | 21 | 14 | Swenson, Soave, Duhamel, Myectomy | 23 | 13 | Down's Syndrome (n = 14), cleft lip and palate (n = 3), supernumary digits (n = 3), congenital hip dislocation (n = 2), congenital heart disease (n = 2), mental retardation (n = 2), microcephalus (n = 1), hydrocephalus (n = 1), growth hormone deficiency (n = 1), deafness (n = 1), facial nerve palsy (n = 1), microphthalmus (n = 1), brachial cleft cyst (n = 1), duplication of small bowel (n = 1), sucrase or maltase deficiency (n = 1), agenesis of the left kidney (n = 1), imperforate anus type II (n = 1), undescended testis (n = 1), bilateral tallpes equinovarus (n = 1), Legg-Calvé-Syndrome (n = 1), Ichthyosis congenita (n = 1), Siemens' Syndrome (n = 1) |
| Ikeda and Goto | 1984 | Japan | Nationwide study | 1628 | 1239 | 186 | 137 | Swenson, Soave, Duhamel | 475 | 180 | Down's Syndrome (n = 47), cardiac anomalies (n = 41), inguinal hernia (n = 11), malrotation (n = 10), cleft lip and palate (n = 9), Meckel's diverticulum (n = 8), mental retardation (n = 8), poly- or syndactyria (n = 6), hydrocephalus (n = 6), atresia ani (n = 5), megaureter (n = 2), others (n = 80) |
| Jung | 1995 | Korea | Cohort study | 137 | 90 | 19 | 4 | Duhamel | 35 | 12 | genitourinary anomalies (n = 15), Down's Syndrome (n = 2), cardiac defects (n = 5), gastrointestinal anomalies (n = 7), gross malformation of face and skull (n = 5), skeletal anomalies (n = 3), others (n = 7) |
| Kleinhaus et al | 1979 | USA | Nationwide study | 1196 | 299 | 609 | 90 | Swenson, Soave, Duhamel | 179 | 99 | / |
| Le-Nguyen et al | 2019 | Canada | Case–control study | 171 | 130 | 21 | 14 | Swenson, Soave, Duhamel, Myectomy | 25 | 33 | Down's Syndrome (n = 9), Mowat-Wilson-Syndrome (n = 2) |
| Li et al | 2006 | China | Cohort study | 252 | 147 | 4 | 1 | Soave | 20 | 34 | / |
| Menezes and Puri | 2006 | Ireland | Cohort study | 259 | 209 | 50 | Swenson, Soave, Duhamel, Myectomy | 43 | 56 | Down's Syndrome (n = 39), others (n = 15) | Menezes and Puri |
| Moore et al | 1996 | South Africa | Cohort study | 178 | 123 | 41 | 14 | Swenson, Soave, Duhamel | 30 | 19 | / |
| Neuvonen et al | 2015 | Finland | Cohort study | 146 | 121 | 10 | 15 | Soave | 24 | 64 | Down's Syndrome (n = 25), cartilage-hair hypoplasia (n = 6), cardiac defects (n = 5), Mowat-Wilson-Syndrome (n = 5), Haddad-Syndrome (n = 3), vesicoureteral reflux (n = 3), anorectal malformation (n = 2), hypertelorism (n = 2), undescended testis (n = 1), pyloric stenosis (n = 1), choanal atresia (n = 1), cleft palare (n = 1), vena Galeni malformation (n = 1), trigonocephalia (n = 1), club fett (n = 1), Currarino-Syndrome (n = 1), Waardenbourg-Syndrome (n = 1), marker Chromosome Syndrome (n = 1) |
| Pini Prato et al | 2019 | Italy | Case–control study | 385 | 273 | 27 | 74 | Soave, Duhamel | 66 | 87 | Down's Syndrome (n = 23), anomalies of kidney and urinary tract (n = 6), CHD (n = 8), visual impairment (n = 7), cerebral abnormality (n = 1), other (n = 3) |
| Pini Prato et al | 2008 | Italy | Cohort study | 112 | 80 | 6 | 22 | Swenson, Soave, Duhamel | 39 | 25 | urogenital anomalies (n = 9), central nervous system (n = 8), cardiac defects (n = 5), ocular anomalies (n = 4), Down's syndrome (n = 2), others (n = 17) |
| Ramesh et al | 1999 | Malaysia | Cohort study | 40 | 27 | 10 | 3 | Rehbein, Swenson, Duhamel, TERPT, Myectomy | 5 | 3 | Down's Syndrome (n = 3) |
| Reding et al | 1997 | Belgium | Cohort study | 59 | 45 | 3 | 8 | Swenson, Duhamel, Myectomy | 13 | 17 | Down's Syndrome (n = 7), genitourinanry anomalies (n = 4), cardiac defects (n = 3), Waardenburg-Syndrome (n = 1), others (n = 2) |
| Rescorla et al | 1992 | USA | Cohort study | 260 | 174 | 61 | 25 | Swenson, Soave, Duhamel, Myectomy | 15 | 32 | Down's Syndrome (n = 23) |
| Sauer et al | 2005 | Canada | Cohort study | 24 | 19 | 2 | 3 | Soave, Duhamel | 7 | 0 | / |
| Singh et al | 2003 | Australia | Nationwide study | 126 | 76 | 22 | 7 | Soave, Duhamel | 15 | 17 | Down's Syndrome (n = 13), cardiac defects (n = 2), development delayed (n = 2), others (n = 16) |
| Surana et al | 1994 | Ireland | Cohort study | 135 | 98 | 25 | 12 | Swenson, Soave, Duhamel | 25 | 20 | Down's Syndrome (n = 17) |
| Teitelbaum et al | 1988 | USA | Cohort study | 80 | 68 | 5 | 7 | Swenson, Soave, Duhamel, Myectomy | 15 | 5 | Down's Syndrome (n = 13), cardiac defects (n = 10), central nervous system (n = 6), gastrointestinal anomalies (n = 3), genitourinary anomalies (n = 2), maxillofacial anomalies (n = 3), skelettal anomalies (n = 2) |
| Teitelbaum et al | 2000 | USA | Case–control study | 181 | 134 | 26 | 16 | Soave | 27 | 55 | Down's Syndrome (n = 11), cardiac defects (n = 11), others (6) |
| Suita et al | 1996 | Japan | Nationwide study | 1121 | 877 | 135 | 109 | Swenson, Soave, Duhamel Myectomy | 326 | 188 | Down's Syndrome (n = 80), cardiac defects (n = 67), malrotation (n = 19), mental retardation (n = 10), intestinal atresia (n = 9), cleft lip and palate (n = 8), Ondine's Syndrome (n = 8), anomalies of the limb (n = 7), anorectal malformation (n = 6), inguinal hernia (n = 3), Meckel's diverticulum (n = 3) |
| Suita et al | 2005 | Japan | Nationwide study | 1103 | 856 | 143 | 104 | Swenson, Soave, Duhamel, TERPT, Myectomy | 191 | 117 | Down's Syndrome (n = 507), cardiac defects (n = 202), mental retardation (n = 49), malrotation (n = 29), anomalies of the limb (n = 21), anorectal malformation (n = 20), Ondine's Syndrome (n = 19), cleft lip and palate (n = 18), ingulinal hernia (n = 14), dwarfism (n = 14), microcephalus (n = 13), Meckel's diverticulum (n = 11), intestinal atresia (n = 9), hydrocephalus (n = 7), undescended testis (n = 7), vesico-ureteral reflux (n = 7) |
| Ali | 2010 | Egypt | Cohort study | 28 | 15 | 6 | 0 | Soave | 7 | 4 | Down's Syndrome (n = 4), cardiac defects (n = 3), hypospadias (n = 1) |
| Chiengkriwate et al | 2007 | Thailand | Case–control study | 50 | 41 | 9 | 0 | Duhamel | 9 | 18 | Down's syndrome (n = 2), cardiac defects (n = 1), cerebral palsy (n = 1) |
| Giuliani et al | 2011 | Italy | Cohort study | 70 | 60 | 10 | 0 | Soave, Duhamel | 10 | 2 | Down's Syndrome (n = 3), hypothyreodism (n = 1), plurimalformative syndrome (n = 1) |
| Hackham et al | 2004 | USA | Cohort study | 66 | 52 | 14 | 0 | Soave, Duhamel | 13 | 17 | Down's syndrome (n = 5), cardiac defects (n = 5), pulmonary defects (n = 4), chromosomal anomalies (n = 2), orthopedic anomalies (n = 1), diaphragmatic hernia (n = 1), imperforate anus (n = 1) |
| Haricharan et al | 2008 | USA | Cohort study | 52 | 40 | 12 | 0 | TERPT | 3 | 19 | Down's Syndrome (n = 4), malrotation (n = 1), unilateral renal agenesis (n = 1) |
| Langer et al | 2003 | Canada, USA, Mexico | Cohort study | 141 | 110 | 7 | 0 | Soave | 20 | 9 | Down's Syndrome (n = 4), cardiac defects (n = 3), central hypoventilation syndrome (n = 1) |
| Lin et al | 2020 | China | Case–control study | 95 | 78 | 17 | 0 | Soave | 19 | 15 | 0 |
| Mattioli et al | 2008 | Italy | Cohort study | 46 | 38 | 8 | 0 | Swenson, Soave, Duhamel | 11 | 4 | Urogenital anomalies (n = 5), central nervous system (n = 4), Down's Syndrome (n = 3), cardiac defects (n = 2), Ondine's Syndrome (n = 1), metabolic disorders (n = 1), eye malformations (n = 1) |
| Mir et al | 2001 | Turkey | Cohort study | 10 | 9 | 1 | 0 | Duhamel | 1 | 2 | Down's syndrome (n = 1), umbilical hernia (n = 1) |
| Ouladsaiad et al | 2016 | Morocco | Cohort study | 15 | 13 | 2 | 0 | TERPT | 6 | 4 | / |
| Parahita et al | 2018 | Indonesia | Cohort study | 100 | 93 | 7 | 0 | Soave, Duhamel | 9 | 15 | / |
| Singh et al | 2007 | Canada | Cohort study | 52 | 46 | 6 | 0 | Swenson | 5 | 6 | Down's Syndrome (n = 5) |
| Wang et al | 2004 | China | Cohort study | 61 | 58 | 3 | 0 | Soave | 22 | 13 | / |
| Wu et al | 2009 | China | Cohort study | 97 | 89 | 8 | 0 | Swenson | 17 | 19 | / |
| Zhang et al | 2014 | China | Cohort study | 127 | 113 | 14 | 0 | TERPT | 43 | 2 | / |
| Adigüzel et al | 2017 | Turkey | Cohort study | 50 | 41 | 0 | 0 | Soave | 11 | 10 | Down's Syndrome (n = 3), cardiac defects (n = 2), cleft palate (n = 1) |
| Chung et al | 2019 | China | Cohort study | 96 | 96 | 0 | 0 | Soave, Duhamel | 10 | 20 | cardiac defects (n = 14), Down's syndrome (n = 9), neurological disorders (n = 8), genitourinary anomalies (n = 6), gastrointestinal anomalies (n = 5), endocrine diseases (n = 6), others (n = 10) |
| Fujiwara et al | 2007 | Japan | Cohort study | 35 | 35 | 0 | 0 | Soave | 0 | 7 | 0 |
| Gao et al | 2001 | China | Cohort study | 34 | 34 | 0 | 0 | Soave | 5 | 2 | Down's Syndrome (n = 1), undescended testis (n = 1) |
| Langer et al | 1999 | USA | Cohort study | 9 | 9 | 0 | 0 | Soave | 2 | 2 | Down's Syndrome (n = 1) |
| Pratap et al | 2007 | Nepal | Cohort study | 65 | 65 | 0 | 0 | Soave | 30 | 3 | / |
| Sapin et al | 2006 | France | Cohort study | 21 | 21 | 0 | 0 | Swenson, Soave | 1 | 1 | central hypoventilation syndrome (n = 1) |
| Weidner and Waldhausen | 2003 | USA | Cohort study | 15 | 15 | 0 | 0 | Swenson | 1 | 2 | Down's Syndrome (n = 1) |
| Zakaria et al | 2012 | Egypt | Cohort study | 40 | 40 | 0 | 0 | Soave | 9 | 3 | / |
| Escobar et al | 2005 | USA | Cohort study | 36 | 0 | 0 | 36 | Soave, Duhamel | 3 | 11 | Ondine's Syndrome (n = 3), UDT (n = 3), Down's Syndrome (n = 2), cardiac defects (n = 2), cleft lip and palate (n = 3), corneal opacification (n = 1), hydrocephalus (n = 1), optic nerve hypoplasia (n = 1), hypospadias (n = 1), other (n = 4) |
| Hoehner et al | 1998 | Canada | Cohort study | 29 | 0 | 0 | 29 | Soave, Duhamel | 1 | 16 | Down's Syndrome (n = 3), colonic atresia (n = 2), Warden's Syndrome (n = 1), solitary kidney (n = 1) |
| Menezes et al | 2008 | Ireland, Italy | Cohort study | 58 | 0 | 0 | 58 | Swenson, Soave, Duhamel | 13 | 31 | Down's Syndrome (n = 2), cardiac defects (n = 4), renal anomalies (n = 4), mental retardation (n = 3), visual defects (n = 2), Ondine's Syndrome (n = 1) |
| Wildhaber et al | 2005 | USA | Cohort study | 25 | 0 | 0 | 25 | Swenson, Soave, Duhamel | 5 | 11 | / |
| Yan et al | 2020 | China | Cohort study | 35 | 0 | 0 | 35 | Soave | 17 | 10 | Cardiac defects (n = 2), Meckel's diverticulum (n = 1), primary immunodeficiency disease (n = 1) |
| Yeh et al | 2014 | China | Cohort study | 9 | 0 | 0 | 9 | Duhamel | 3 | 5 | Hypoplastic right heart syndrome (n = 1), imperforate anus (n = 1), urogenital sinus, vaginal atresia, Pallister-Hall-syndrome (n = 1) |