Early Neonatal Mortality among Babies Born with Spina Bifida in Finland (2000–2014)

Vijaya Kancherla; Sanjida Mowla; Sari Räisänen; Mika Gissler

doi:10.1055/s-0041-1733957

American Journal of Perinatology, Table of Contents

Am J Perinatol 2023; 40(11): 1208-1216
DOI: 10.1055/s-0041-1733957

Original Article

Early Neonatal Mortality among Babies Born with Spina Bifida in Finland (2000–2014)

Vijaya Kancherla

¹Department of Epidemiology, Rollins School of Public Health, Emory University, Atlanta, Georgia

,

Sanjida Mowla

¹Department of Epidemiology, Rollins School of Public Health, Emory University, Atlanta, Georgia

,

Sari Räisänen

²School of Health, Tampere University of Applied Sciences, Tampere, Finland

,

Mika Gissler

³Information Services Department, THL Finnish Institute for Health and Welfare, Helsinki, Finland; Department of Molecular Medicine and Surgery, Karolinska Institute, Stockholm, Sweden, Academic Primary Health Care Centre, Region Stockholm, Sweden

› Author Affiliations

Abstract

Objective We examined early neonatal mortality risk, temporal trends, and selected infant and maternal factors associated with early neonatal mortality among all spina bifida-affected live births in Finland.

Study Design We linked multiregistry population-based data from the national registers in Finland for infants born with spina bifida from 2000 to 2014. Early neonatal mortality was defined as death in 0 to 6 days after birth. Early neonatal mortality risk and 95% confidence intervals (CI) was estimated by using the Poisson approximation of binomial distribution. Poisson regression was used to examine temporal trend in early neonatal mortality from 2000 to 2014 for spina bifida cases and all births in Finland. Selected infant and maternal characteristics were compared between cases that experienced early neonatal mortality and cases that did not. Exact logistic regression was used to estimate unadjusted odds ratios (uORs) and 95% confidence intervals (CIs).

Results A total of 181 babies were born alive with spina bifida in Finland during the study period; 61% had isolated spina bifida. Pooling all study years, 7.2% (95% CI: 4.2–12.4%) of all live-born cases experienced early neonatal death. There was a significant increase in early neonatal mortality among spina bifida births over the study period (p < 0.0001). Low gestational age (<37 weeks; uOR = 6.96; 95% CI: 1.86–29.01), cases occurring as a part of a syndrome (uOR = 125.67; 95% CI: 14.90 to >999.999), and advanced maternal age at gestation (≥35 years; uOR = 5.33; 95% CI: 1.21–21.87) were positively associated with early neonatal mortality.

Conclusion Using national data from Finland, we found high early neonatal mortality with increasing trend over birth period spanning 15 years (2000–2014), and unadjusted positive associations with some infant and maternal factors. Future studies should pool data from Nordic countries to increase study size allowing multivariable analysis.

Key Points

Early neonatal mortality in babies affected by spina bifida is 7% in Finland.
Early neonatal mortality trend showed a significant increase from 2000 to 2014.
Low gestational age, syndrome case status, and advanced maternal age increased early neonatal mortality risk in spina bifida.

Keywords

Finland - myelomeningocele - neonatal mortality - open spina bifida - risk factors

Full Text

References

References
1 Bakaniene I, Prasauskiene A, Vaiciene-Magistris N. Health-related quality of life in children with myelomeningocele: a systematic review of the literature. Child Care Health Dev 2016; 42 (05) 625-643
2 Rocque BG, Bishop ER, Scogin MA. et al. Assessing health-related quality of life in children with spina bifida. J Neurosurg Pediatr 2015; 15 (02) 144-149
3 Wiener JS, Suson KD, Castillo J. et al. Bowel management and continence in adults with spina bifida: results from the National Spina Bifida Patient Registry 2009-15. J Pediatr Rehabil Med 2017; 10 (3-4): 335-343
4 Alabi NB, Thibadeau J, Wiener JS. et al. Surgeries and health outcomes among patients with spina bifida. Pediatrics 2018; 142 (03) e20173730
5 Bol KA, Collins JS, Kirby RS. National Birth Defects Prevention Network. Survival of infants with neural tube defects in the presence of folic acid fortification. Pediatrics 2006; 117 (03) 803-813
6 Kancherla V, Druschel CM, Oakley Jr GP. Population-based study to determine mortality in spina bifida: New York State Congenital Malformations Registry, 1983 to 2006. Birth Defects Res A Clin Mol Teratol 2014; 100 (08) 563-575
7 Shin M, Kucik JE, Siffel C. et al. Improved survival among children with spina bifida in the United States. J Pediatr 2012; 161 (06) 1132-1137
8 Wang Y, Hu J, Druschel CM, Kirby RS. Twenty-five-year survival of children with birth defects in New York State: a population-based study. Birth Defects Res A Clin Mol Teratol 2011; 91 (12) 995-1003
9 Wong LY, Paulozzi LJ. Survival of infants with spina bifida: a population study, 1979-94. Paediatr Perinat Epidemiol 2001; 15 (04) 374-378
10 Atta CA, Fiest KM, Frolkis AD. et al. Global birth prevalence of spina bifida by folic acid fortification status: a systematic review and meta-analysis. Am J Public Health 2016; 106 (01) e24-e34
11 Turner MJ. Neural tube defects and folic acid food fortification in Europe. Am J Public Health 2018; 108 (05) 601-602
12 De Wals P, Tairou F, Van Allen MI. et al. Spina bifida before and after folic acid fortification in Canada. Birth Defects Res A Clin Mol Teratol 2008; 82 (09) 622-626
13 Lopez KN, Nembhard WN, Wang Y. et al. Birth defect survival for Hispanic subgroups. Birth Defects Res 2018; 110 (04) 352-363
14 Pace ND, Siega-Riz AM, Olshan AF. et al; National Birth Defects Prevention Study. Survival of infants with spina bifida and the role of maternal prepregnancy body mass index. Birth Defects Res 2019; 111 (16) 1205-1216
15 Wang Y, Hu J, Druschel CM. A retrospective cohort study of mortality among children with birth defects in New York State, 1983-2006. Birth Defects Res A Clin Mol Teratol 2010; 88 (12) 1023-1031
16 Kancherla V, Ma C, Grant G, Lee HC, Hintz SR, Carmichael SL. Factors associated with early neonatal and first-year mortality in infants with myelomeningocele in California from 2006 to 2011. Am J Perinatol 2020; 37: 1234-1242
17 Finnish Institute for Health and Welfare. 2015 Register of congenital malformations. Accessed February 18, 2021 at: https://thl.fi/en/web/thlfi-en/statistics/information-on-statistics/register-descriptions/register-of-congenital-malformations
18 National Center on Birth Defects and Developmental Disabilities. 1996 Birth defects and genetic diseases branch 6-digit code. Accessed February 18, 2021 at: https://www.cdc.gov/ncbddd/birthdefects/documents/macdpcode0807.pdf
19 Finnish Institute for Health and Welfare. 2016 Care register for health care. Accessed February 18, 2021 at: https://thl.fi/en/web/thlfi-en/statistics/information-on-statistics/register-descriptions/care-register-for-health-care
20 Finnish Institute for Health and Welfare. 2019 Medical birth register. Accessed February 18, 2021 at: https://thl.fi/en/web/thlfi-en/statistics/information-on-statistics/register-descriptions/newborns
21 Official Statistics of Finland (OSF), ( n.d.). Educational structure of population. Accessed February 18, 2021 at: http://www.stat.fi/til/vkour/meta_en.html
22 Official Statistics of Finland (OSF), (n.d.). Income distribution statistics. Accessed February 18, 2021 at: https://www.stat.fi/til/tjt/meta_en.html
23 Finnish Institute for Health and Welfare. 2020 Congenital malformations 2017. Accessed February 18, 2021 at: https://thl.fi/en/web/thlfi-en/statistics/information-on-statistics/quality-descriptions/congenital-anomalies
24 Blencowe H, Kancherla V, Moorthie S, Darlison MW, Modell B. Estimates of global and regional prevalence of neural tube defects for 2015: a systematic analysis. Ann N Y Acad Sci 2018; 1414 (01) 31-46
25 Johnson CY, Honein MA, Dana Flanders W, Howards PP, Oakley Jr GP, Rasmussen SA. Pregnancy termination following prenatal diagnosis of anencephaly or spina bifida: a systematic review of the literature. Birth Defects Res A Clin Mol Teratol 2012; 94 (11) 857-863
26 Bakker MK, Kancherla V, Canfield MA. et al. Analysis of mortality among neonates and children with spina bifida: An international registry-based study, 2001–2012. Paediatr Perinat Epidemiol 2019; 33 (06) 436-448
27 EUROCAT. Prevalence Charts and Tables. 2020 . Accessed February 18, 2021 at: https://eu-rd-platform.jrc.ec.europa.eu/eurocat/eurocat-data/prevalence
28 Borgstedt-Bakke JH, Fenger-Grøn M, Rasmussen MM. Correlation of mortality with lesion level in patients with myelomeningocele: a population-based study. J Neurosurg Pediatr 2017; 19 (02) 227-231
29 Preis K, Swiatkowska-Freund M, Janczewska I. Spina bifida–a follow-up study of neonates born from 1991 to 2001. J Perinat Med 2005; 33 (04) 353-356
30 Glinianaia SV, Morris JK, Best KE. et al. Long-term survival of children born with congenital anomalies: a systematic review and meta-analysis of population-based studies. PLoS Med 2020; 17 (09) e1003356
31 Oakeshott P, Hunt GM, Poulton A, Reid F. Expectation of life and unexpected death in open spina bifida: a 40-year complete, non-selective, longitudinal cohort study. Dev Med Child Neurol 2010; 52 (08) 749-753
32 Westlung K. Mortality from congenital malformations of the central nervous system in Norway, 1951-65. Life insurance companies' institute for medical statistics at the Oslo city hospitals, report no. 12. Br J Prev Soc Med 1969; 23 (01) 28-33
33 den Quden AL, Hirasing RA, Buitendijk SE, de Jong-van den Berg LT, de Walle HE, Cornel MC. [Prevalence, clinical aspects and prognosis of neural tube defects in The Netherlands]. Ned Tijdschr Geneeskd 1996; 140 (42) 2092-2095
34 Haase J, Green A, Hauge M, Holm NV, Mathiasen H. A cohort study of neural tube defects (NTD) in Denmark covering the first seven years of life. Childs Nerv Syst 1987; 3 (02) 117-120
35 Hewitt D. Geographical variations in the mortality attributed to spina bifida and other congenital malformations. Br J Prev Soc Med 1963; 17: 13-22
36 Rogers SC, Morris M. Infant mortality from spina bifida, congenitalhydrocephalus, monstrosity, and congenital dseases of the cardiovascular system in England and Wales. Ann Hum Genet 1971; 34 (03) 295-305
37 Sípek A, Gregor V, Horácek J, Masátová D. [Infant mortality due to birth defects in the Czech Republic in 1994-2004]. Ceska Gynekol 2006; 71 (05) 380-388
38 Sunderland R, Emery JL. The mortality and birth rates of spina bifida during a period of treatment, selection and antenatal screening in Sheffield, 1963-1978. Z Kinderchir Grenzgeb 1979; 28 (04) 294-301
39 Williams J, Mai CT, Mulinare J. et al; Centers for Disease Control and Prevention. Updated estimates of neural tube defects prevented by mandatory folic acid fortification - United States, 1995-2011. MMWR Morb Mortal Wkly Rep 2015; 64 (01) 1-5
40 Toivonen KI, Lacroix E, Flynn M. et al. Folic acid supplementation during the preconception period: a systematic review and meta-analysis. Prev Med 2018; 114: 1-17
41 Gissler M, Shelley J. Quality of data on subsequent events in a routine Medical Birth Register. Med Inform Internet Med 2002; 27 (01) 33-38
42 Gissler M, Teperi J, Hemminki E, Meriläinen J. Data quality after restructuring a national medical registry. Scand J Soc Med 1995; 23 (01) 75-80
43 Haikonen K, Lunetta P, Lillsunde PM, Sund R. Methodological challenges in using the Finnish Hospital Discharge Register for studying fire-related injuries leading to inpatient care. BMC Med Inform Decis Mak 2013; 13: 36
44 Sund R. Quality of the Finnish Hospital Discharge Register: a systematic review. Scand J Public Health 2012; 40 (06) 505-515
45 Teperi J. Multi method approach to the assessment of data quality in the Finnish Medical Birth Registry. J Epidemiol Community Health 1993; 47 (03) 242-247
46 Arbour L, Melnikov V, McIntosh S, Olsen B, Osborne G, Vaktskjold A. The current state of birth outcome and birth defect surveillance in northern regions of the world. Int J Circumpolar Health 2009; 68 (05) 443-458
47 Spina Bifida Association. 2019 Guidelines for the care of people with spina bifida. Accessed February 18, 2021 at: http://spinabifidaassociation.org/wp-content/uploads/2018/10/GFPWSB-Update-102518.pdf
48 Van Der Vossen S, Pistorius LR, Mulder EJ. et al. Role of prenatal ultrasound in predicting survival and mental and motor functioning in children with spina bifida. Ultrasound Obstet Gynecol 2009; 34 (03) 253-258