TABLE 3.
Study quality | |||||||||||||
---|---|---|---|---|---|---|---|---|---|---|---|---|---|
Author (s), year | Country | Sub‐National | Birth prevalence | Birth period | Study design/data source | Study population | Specialized care a | Sample size | Data source | Diagnostic method | Numerator | Denominator | Population size |
North America | |||||||||||||
(Alonso Lotti et al., 1998) | Cuba | 13 of the 15 regions | 4.42 | 1985/03–1996/12 | RECUMAC | Live births, stillbirths | No | 520,578 | Strong | Moderate | Strong | Strong | Strong |
(Guzmán‐Huerta et al., 2012) | Mexico | UNIMEF | 10.99 b | 1995/01–2009/12 | Review of hospital charts of patients seen at the National Institute of Perinatal Medicine | Live births, stillbirths | Yes | 81,892 | Moderate | Strong | Strong | Strong | Weak |
(Kallen et al., 1993) | Mexico | NR | 2.51 b | 1978–1988 | Programa Mexicano de Registro y vigilancia epidemiogica de malformaciones congentias externas | Live births, stillbirths | No | 359,000 | Strong | Weak | Strong | Strong | Strong |
(Curran, Sigmon, & Opitz, 1974) | USA | New Jersey | 4.00 b | NR (“past 10 years,” <1973) | Records from the Margaret Hague Maternity Hospital | Live births | No | 75,000 | Moderate | Strong | Strong | Strong | Weak |
(Langlois & Scheuerle, 2015) | USA | Texas | 2.66 b | 1999–2009 | Records in the Texas Birth Defects Registry | Live births, stillbirths, elective terminations | No | 4,207,898 | Strong | Weak | Strong | Weak | Strong |
(Rasmussen et al., 1996) | USA | Boston, Massachusetts | 2.37 | 1972/02–1975/02, 1979/01–1990/12 | Brigham and Women's Hospital active malformation surveillance system | Live births, stillbirths >20 w, elective terminations | Yes | 126,316 | Moderate | Strong | Strong | Strong | Moderate |
(Stevenson, Carey, Byrne, Srisukhumbowornchai, & Feldkamp, 2012) | USA | Utah | 3.53 | 1999–2008 | UBDN | Live births, stillbirths, elective terminations | No | 509,286 | Strong | Strong | Strong | Strong | Strong |
(Waller et al., 2008) | USA | Arkansas | 5.20 | 1993–1999 | Arkansas Reproductive Health Monitoring System | Live births, stillbirths >20 w, elective terminations >20 w | No | 250,000 | Strong | Moderate | Strong | Weak | Strong |
Atlanta | 3.89 | 1968–2001 | Atlanta Congenital Defects Program | 1,129,972 | Strong | Moderate | Strong | Weak | Strong | ||||
California | 4.70 | 1983–1997 | California Birth Defects Monitoring System | 3,572,233 | Strong | Moderate | Strong | Weak | Strong | ||||
Iowa | 4.09 | 1983–2001 | Iowa Register for Congenital and Inherited Disorders | 733,196 | Strong | Moderate | Strong | Weak | Strong | ||||
New York | 3.60 | 1992–2001 | New York State Congenital Malformations Registry | 2,664,131 | Strong | Moderate | Strong | Strong | Strong | ||||
Oklahoma | 5.99 | 1994–2003 | Oklahoma Birth Defects Registry | 484,013 | Strong | Moderate | Strong | Weak | Strong | ||||
Texas | 3.87 | 1996–2002 | Texas Birth Defects Epidemiology and Surveillance Branch | 2,042,554 | Strong | Moderate | Strong | Weak | Strong | ||||
(Woolf & Turner, 1969) | USA | Salt Lake City, Utah | 13.43 b | 1951–1961 | Retrospective review of nursery records in the Latter‐day Saints Hospital | Live births | No | 59,561 | Moderate | Weak | Strong | Strong | Weak |
South America | |||||||||||||
(Barbosa‐Buck et al., 2012) | Argentina, Bolivia, Brazil, Chile, Colombia, Ecuador, Paraguay, Uruguay, and Venezuela | NR | 4.40 | 2000/01–2007/12 | ECLAMC | Live births, stillbirths >500 g | No | 1,544,496 | Strong | Moderate | Strong | Strong | Strong |
(Duarte et al., 2018) | Argentina | 24 jurisdictions | 4.75 | 2009/11–2016/12 | RENAC | Live births, stillbirths >500 g | No | 1,663,610 | Strong | Moderate | Strong | Strong | Strong |
(Kallen et al., 1993) | All South American Countries | NR | 1.93 b | 1967–1989 | ECLAMC | Live births, stillbirths | No | 2,278,000 | Strong | Weak | Strong | Strong | Strong |
(Orioli et al., 1995) | South America | NR | 1.64 | 1967–1981 | ECLAMC | Live births | No | 852,893 | Strong | Strong | Strong | Strong | Strong |
South America | NR | 2.00 | 1982–1992 | No | 2,054,682 | Strong | Strong | Strong | Strong | Strong | |||
(Sánchez, Brito‐Arreaza, Alvarez‐Arratia, & Ramírez, 1991) | Venezuela | Ciudad Bolívar | 14.25 | 1978/04–1990/08 | Congenital malformations surveillance program at Ruiz y Paez Hopital | Live births until 1979–12, live births, stillbirths thereafter | No | 70,152 | Moderate | Strong | Strong | Strong | Weak |
Europe | |||||||||||||
(Coi et al., 2019) | Austria | Styria | 1.62 | 1991–2012 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 247,210 | Strong | Moderate | Strong | Strong | Strong |
Belgium | Antwerp | 5.49 | 1991–2014 | No | 400,634 | Strong | Moderate | Strong | Strong | Strong | |||
Croatia | Zagreb | 3.73 | 1991–2015 | No | 160,988 | Strong | Moderate | Strong | Strong | Moderate | |||
(Andersen Jr & Hauge, 1989) | Denmark | Fyn | 1.28 | 1970/01/01–1983/12/31 | County hospital records | Live births, stillbirths | No | 77,977 | Moderate | Moderate | Strong | Strong | Weak |
(Coi et al., 2019) | Denmark | Odense | 5.22 | 2000–2014 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 76,625 | Strong | Moderate | Strong | Strong | Weak |
(Kallen et al., 1993) | Denmark | NR | 0.61 b | 1983–1988 | Danish National Board of Health: Registry of Congenital Malformations | Live births, stillbirths | No | 328,000 | Strong | Weak | Strong | Strong | Strong |
(Coi et al., 2019) | France | Auvergne | 3.89 | 1991–2015 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 334,612 | Strong | Moderate | Strong | Strong | Strong |
France | Isle de Reunion | 5.94 | 2001–2015 | No | 218,796 | Strong | Moderate | Strong | Strong | Strong | |||
France | Paris | 6.11 | 1991–2015 | No | 768,885 | Strong | Moderate | Strong | Strong | Strong | |||
(Stoll, Dott, Roth, & Alembik, 1989) | France | City of Strasbourg (urban area) and “Département du Bas‐Rhin” (rural area) | 6.64 | 19 79/01–1986/12 | Registry of all newborn children in Strasbourg and Department du Bas‐Rhin | Live births, stillbirths | No | 105,374 | Strong | Strong | Strong | Strong | Moderate |
(Coi et al., 2019) | Germany | Saxony Anhalt | 4.76 | 1991–2015 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 357,516 | Strong | Moderate | Strong | Strong | Strong |
(Kallen et al., 1993) | Italy | NR | 3.42 b | 1978–1988 | Italian birth defects monitoring system (IPIMC) | Live births, stillbirths | No | 1,256,000 | Strong | Weak | Strong | Strong | Strong |
(Camera, 1980) | Italy | Genoa | 1.86 b | 1960–1980/02 | Records of osteochondroplasias encountered in the maternity ward of a single hospital | NR | No | 53,700 | Moderate | Weak | Weak | Weak | Weak |
(Camera & Mastroiacovo, 1988) | Italy | NR | 3.70 | 1978–1985 | IMMSBD | Live births, stillbirths | No | 838,717 | Strong | Strong | Strong | Strong | Strong |
(Coi et al., 2019) | Italy | Emilia Romagna | 5.70 | 1991–2015 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 806,485 | Strong | Moderate | Strong | Strong | Strong |
(Coi et al., 2019) | Tuscany | 5.06 | 1991–2015 | No | 672,268 | Strong | Moderate | Strong | Strong | Strong | |||
(Orioli et al., 1995) | Italy | NR | 3.61 | 1978–1991 | IPIMC | Live births, stillbirths | No | 1,494,756 | Strong | Weak | Strong | Strong | Strong |
(Coi et al., 2019) | Ireland | Cork&Kerry | 3.34 | 1996–2015 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 179,563 | Strong | Moderate | Strong | Strong | Strong |
Malta | NR | 6.35 | 1991–2015 | No | 110,174 | Strong | Moderate | Strong | Strong | Moderate | |||
Netherlands | Northern region | 3.01 | 1991–2015 | No | 465,261 | Strong | Moderate | Strong | Strong | Strong | |||
Norway | NR | 2.39 | 1999–2012 | No | 836,535 | Strong | Moderate | Strong | Strong | Strong | |||
Poland | Wielkopolska | 4.47 | 1999–2015 | No | 626,876 | Strong | Moderate | Strong | Strong | Strong | |||
Spain | Basque County | 2.72 | 1991–2015 | No | 441,896 | Strong | Moderate | Strong | Strong | Strong | |||
Spain | Valencia region | 2.69 | 2007–2015 | No | 446,903 | Strong | Moderate | Strong | Strong | Strong | |||
(Martínez‐Frías et al., 1991) | Spain | 16 of the 17 Spanish Regions (Comunidades Autonomas) | 2.53 | 1976/04–1988/12 | ECEMC | Live births | No | 710,815 | Strong | Moderate | Strong | Strong | Strong |
(Gustavson & Jorulf, 1975) | Sweden | Uppsala | 6.75 b | 1970/02–1974/08 | Prospective collection of neonatal disorders and anomalies of the skeleton at the University Hospital in Uppsala | Live births, stillbirths | No | 14,816 | Moderate | Strong | Strong | Strong | Weak |
(Kallen et al., 1993) | Sweden | NR | 1.64 b | 1965–1989 | Swedish register of congenital malformations | Live births, stillbirths | No | 2,375,000 | Strong | Weak | Strong | Strong | Strong |
(Coi et al., 2019) | Switzerland | Vaud | 3.63 | 1991–2015 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 192,684 | Strong | Moderate | Strong | Strong | Strong |
UK | Wessex | 4.07 | 1994–2015 | No | 615,000 | Strong | Moderate | Strong | Strong | Strong | |||
UK | Wales | 3.48 | 1998–2015 | No | 602,776 | Strong | Moderate | Strong | Strong | Strong | |||
UK | South West England | 3.12 | 2005–2015 | No | 545,302 | Strong | Moderate | Strong | Strong | Strong | |||
UK | Northern England | 3.03 | 1991–2015 | No | 824,745 | Strong | Moderate | Strong | Strong | Strong | |||
UK | Thames Valley | 1.94 | 1991–2015 | No | 411,928 | Strong | Moderate | Strong | Strong | Strong | |||
(Gardner, 1977) | UK | Edinburgh | 1.93 b | 1964/04–1968/10 | Edinburgh Register of the Newborn | Live births, stillbirths | No | 51,836 | Strong | Strong | Strong | Strong | Weak |
2.73 b | 1968/11–1973/12, 1968/11–1972/11 | Birth records at the Simpson Memorial Maternity Pavilion of the Royal Infirmary and at the Eastern General Hospital | Live births, stillbirths | No | 36,569 | Moderate | Strong | Strong | Strong | Weak | |||
(Harris & Patton, 1971) | UK | Manchester | 6.26 b | 1951–1969 | Reassessment of cases of achondroplasia from birth records at St. Mary's Hospital, Manchester | Live births, stillbirths | No | 63,934 | Moderate | Moderate | Strong | Strong | Weak |
(Sokal, Tata, & Fleming, 2014) | UK | Whole country | 7.56 b | 1990–2009 | Prospectively collected primary care data from THIN | Live births | No | 794,169 | Moderate | Weak | Strong | Strong | Strong |
(Stevenson, 1957) | UK | Belfast | 28.34 | 1938/01–1956/06 | Records of the Royal Maternity Hospital | Live births, stillbirths | No | 31,753 | Moderate | Moderate | Strong | Strong | Weak |
(Coi et al., 2019) | Ukraine | OMNI‐net | 6.00 | 2005–2015 | EUROCAT | Live births, stillbirths ≥20 w, elective terminations | No | 333,189 | Strong | Moderate | Strong | Strong | Strong |
Northern Africa/Middle East | |||||||||||||
(Golalipour, Ahmadpour‐Kacho, & Vakili, 2005) | Iran | Gorgan | 40.02 | 1998/01–1999/08 | Prospective collection of congenital malformation frequency at a referral hospital in Gorgan | Live births, stillbirths | Yes | 9,996 | Moderate | Moderate | Strong | Strong | Weak |
(Golalipour, Kaviany, Golalipour, Mirfazeli, & Behnampour, 2018) | Iran | Gorgan, Golestan Provincein | 33.44 | 2007/03–2011 | Prospective collection of frequencies of congenital limb defects in 3 hospitals in Gorgan | Live births | Yes | 32,895 | Strong | Moderate | Strong | Strong | Weak |
(Alaani, Al‐Fallouji, Busby, & Hamdan, 2012) | Iraq | Fallujah | 16.53 b | 2009/11–2010/09 | Records from a single pediatric clinic | Live births | Yes | 6,049 | Moderate | Weak | Strong | Strong | Weak |
(Al‐Ani et al., 2012) | Iraq | Al‐Anbar governorate | 52.25 | 2010/10–2011/10 | WICCARS | Live births | Yes | 5,742 | Strong | Moderate | Strong | Strong | Weak |
(Al‐Janabi, 2007) | Iraq | Al‐Anbar governorate | 241.60 | 2000/07–2002/06 | Prospective collection of congenital malformation frequency at the Maternal and Children Hospital in Al‐Anbar governate | Live births, stillbirths | No | 12,831 | Moderate | Moderate | Strong | Strong | Weak |
(Al‐Obaidi, Mahmood, & Al‐Dalla Ali, 2013) | Iraq | Ramadi | 66.93 | 2009/02–2009/10 | Prospective collection of congenital malformation frequency at the Maternity and Children Teaching Hospital in Ramadi | Live births, stillbirths | No | 1,494 | Moderate | Moderate | Strong | Strong | Weak |
(Al‐Rubaii, Al‐Tufaily, & Fakhri, 2009) | Iraq | Babylon | 62.82 b | 2007/01–2008/01 | Records from Babylon Maternity and Pediatrics Teaching Hospital | Live births | No | 9,551 | Moderate | Moderate | Strong | Weak | Weak |
(Taboo, 2012) | Iraq | Mosul | 34.21 b | 2009/01–2010/12 | Prospective study of congenital abnormalities at Lahore General Hospital | Live births, stillbirths | No | 46,775 | Moderate | Moderate | Strong | Strong | Weak |
(Madi, Al Naggar, Al Awadi, & Bastaki, 2005) | Kuwait | Al‐Jahra Region | 12.92 b | 2000/01–2001/12 | Data from the newborn register at AL‐Jahra Hospital | Live births, stillbirths | No | 7,739 | Moderate | Strong | Strong | Strong | Weak |
(Bittar, 1998) | Libanon | Southern sector of Beirut, Baalbak, Hermel and South Lebanon | 25.87 | 1991/02–1993/07 | Prospective collection of congenital malformation frequency at a large hospital in south Beirut | Live births, stillbirths | No | 3,865 | Moderate | Moderate | Strong | Strong | Weak |
(Al‐Jama, 2001) | Saudi Arabia | Al‐Khobar | 6.77 | 1992/01–1997/12 | Retrospective examination of delivery room records | Singleton live births | Yes | 14,762 | Moderate | Strong | Strong | Strong | Weak |
(Sallout et al., 2015) | Saudi Arabia | Riyadh | 48.14 | 2007/01–2012/12 | Prospective collection of data on congenital anomalies in the obstetrics and gynecology ultrasound unit King Fahad Medical City | Live births | Yes | 29,084 | Moderate | Moderate | Strong | Strong | Weak |
(Al‐Gazali et al., 2003) | UAE | Al Ain Medical District | 10.51 | 1996/01–2000/12 | Active malformation surveillance system in Al Ain Medical District | Live births, stillbirths | No | 38,048 | Strong | Strong | Strong | Strong | Weak |
Sub‐Saharan Africa | |||||||||||||
(Charlotte, Aurore, Charlotte, Esther, & Eugene, 2015) | Camaroon | NR | 16.53 b | 2008/01–2012/06 | Prospective collection of congenital malformation frequency at Doala General Hospital | Live births, stillbirths | Yes | 6,048 | Moderate | Moderate | Strong | Strong | Weak |
(Ekanem et al., 2008) | Nigeria | Cross River and Akwa Ibom states | 3.13 | 1980–2003 | Records from University of Calabar Teaching Hospital, St Luke's Hospital Anua, Uyo, and St Mary's Hospital Uruakpan | NR | Yes | 127,929 | Moderate | Weak | Weak | Weak | Moderate |
(Ekanem, Bassey, Mesembe, Eluwa, & Ekong, 2011) | Nigeria | Port Harcourt, Rivers state | 12.60 b | 1990–2003 | Records from 2 major hospitals in Port Harcourt | NR | Yes | 39,693 | Moderate | Weak | Weak | Weak | Weak |
(Sunday‐Adeoye, Okonta, & Egwuatu, 2007) | Nigeria | Afiko, Ebonyi State | 38.62 b | 1980/01–1999/12 | Records births at the Mater Misericordiae Hospital | NR | No | 33,659 | Strong | Moderate | Strong | Strong | Weak |
(Delport, Christianson, Van den Berg, Wolmarans, & Gericke, 1995) | South‐Africa | Pretoriaz | 5.76 | 1986/05–1989/04 | Prospective collection of congenital malformation frequency at the Kalafong Hospital | Live births | Yes | 17,351 | Moderate | Moderate | Strong | Strong | Weak |
South‐East Asia/Oceania | |||||||||||||
(Oberklaid, Danks, & Jensen, 1979) | Australia | Victoria | 3.85 | 1969–1975 | Royal Children's Hospital records and surveys of all pediatricians, radiologists, orthopedic surgeons in Victoria (1968–1970). Newspaper and television publicity, Little Peoples' Association of Australasia, and personal visits to rural areas to ascertain additional cases. | NR | No | 492,889 | Weak | Weak | Weak | Weak | Strong |
(Kallen et al., 1993) | Australia | NR | 4.93 b | 1981–1989 | Data from Australian National data systems for (1) congenital malformations and (2) for pregnancies resulting from in vitro fertilization. | Live births, stillbirths | No | 1,946,000 | Strong | Weak | Strong | Strong | Strong |
(Jaikrishan et al., 2013) | India | NR | 11.38 | 1995/08–2011/06 | Prospective collection of congenital malformation frequency in 7 government hospitals serving people from high and normal national radiation areas | Live births, stillbirths >28 w | No | 140,558 | Moderate | Moderate | Strong | Strong | Moderate |
(Kusumalatha et al., 2017) | India | Kakinada | 14.40 b | 2016/01–2016/12 | Hospital‐based cross‐sectional study | Live births, stillbirths | No | 13,893 | Moderate | Moderate | Strong | Strong | Weak |
(Rasheed & Haseeb, 2016) | India | Maharashtra | 14.26 | 1994/03–1995/04 | Prospective collection of frequencies of congenital anomalies at Marden Medical Complex | Live births, stillbirths | Yes | 7,012 | Moderate | Moderate | Strong | Strong | Weak |
(Higurashi et al., 1990) | Japan | Tokyo | 10.92 | 1972/07–1985/12 | Records from consecutive births in a single large maternity hospital in Tokyo | Live births | No | 27,472 | Moderate | Moderate | Strong | Strong | Weak |
(Peng, 1988) | Malaysia | State of Kedah | 10.12 | 1984/04–1987/03 | Records of live births occurring in Alor Setar General Hospital | Live births | Yes | 19,769 | Moderate | Moderate | Strong | Strong | Weak |
(Qadir, Amir, & Bano, 2017) | Pakistan | Mardan | 10.58 b | 2016/05–2017/04 | Prospective collection of frequencies of congenital anomalies at Government Medical College and Hospital | NR | No | 9,453 | Moderate | Moderate | Weak | Weak | Weak |
(Tariq, 2010) | Pakistan | Lahore | 34.82 b | 2007/01–2007/12 | Prospective study of congenital abnormalities at Al‐Batool Teaching Hospital of Obstetrics and Gynecology | NR | No | 2,872 | Moderate | Moderate | Weak | Weak | Weak |
(Nasreen, Naib, & Ibrar, 2016) | Pakistan | Peshawar | 0.00 b | 2007/06–2009/06 | Prospective collection of data on congenital anomalies at Khyber Teaching Hospital | NR | No | 6,297 | Moderate | Moderate | Weak | Weak | Weak |
(Jaruratanasirikul et al., 2016) | Thailand | Songkhla, Trang and Phatthalung | 2.68 b | 2009/01–2013/12 | Records from Bureau of Policy and Strategy, Ministry of Public Health | Live births, stillbirths, elective terminations | No | 186,393 | Strong | Moderate | Strong | Strong | Strong |
Abbreviations: ECEMC, Spanish Collaborative Study of Congenital Malformations; ECLAMC, Latin American Collaborative Study of Congenital Malformations; EUROCAT, European network of population‐based registries for the epidemiological surveillance of congenital anomalies; g, grams; IMMSBD, Italian birth defects monitoring system; NR, not reported; OMNI‐NET, Ukraine Birth Defects Program; RECUMAC, Registry of Congenital Malformations; RENAC, Records from National Network of Congenital Anomalies of Argentina; THIN, the Health Improvement Network; UBDN, Utah Birth Defect Network; UK, United Kingdom; UNIMEF, Department of Maternal Fetal Medicine; USA, United States of America; W, weeks; WICCARS, Western Iraq Center for Congenital Anomalies Registry and Surveillance.
Yes: Women who gave birth at a referral center or tertiary hospital. No: Women who gave birth in other settings.
Calculated per 100,000 births based on raw data provided in the article.