Am J Perinatol 2021; 38(12): 1263-1270
DOI: 10.1055/s-0040-1712165
Original Article

Factors Associated with Early Neonatal and First-Year Mortality in Infants with Myelomeningocele in California from 2006 to 2011

Vijaya Kancherla
1   Department of Epidemiology, Emory University, Rollins School of Public Health, Atlanta, Georgia
,
Chen Ma
2   Division of Neonatal and Developmental Medicine, Department of Pediatrics, Stanford University School of Medicine, Stanford, California
,
Gerald Grant
3   Department of Neurosurgery, Stanford University School of Medicine, Stanford, California
,
Henry C. Lee
2   Division of Neonatal and Developmental Medicine, Department of Pediatrics, Stanford University School of Medicine, Stanford, California
4   California Perinatal Quality Care Collaborative, Stanford, California
,
Susan R. Hintz
2   Division of Neonatal and Developmental Medicine, Department of Pediatrics, Stanford University School of Medicine, Stanford, California
4   California Perinatal Quality Care Collaborative, Stanford, California
,
Suzan L. Carmichael
2   Division of Neonatal and Developmental Medicine, Department of Pediatrics, Stanford University School of Medicine, Stanford, California
5   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Stanford University, Stanford, California
› Author Affiliations
Funding This study received its financial support from the National Institute on Minority Health and Health Disparities (grant number R01 MD007796).

Abstract

Objective The aim of this study is to examine factors associated with early neonatal (death within first 7 days of birth) and infant (death during the first year of life) mortality among infants born with myelomeningocele.

Study Design We examined linked data from the California Perinatal Quality Care Collaborative, vital records, and hospital discharge records for infants born with myelomeningocele from 2006 to 2011. Survival probability was calculated using Kaplan–Meier Product Limit method and 95% confidence intervals (CI) using Greenwood's method; Cox proportional hazard models were used to estimate unadjusted and adjusted hazard ratios (HR) and 95% CI.

Results Early neonatal and first-year survival probabilities among infants born with myelomeningocele were 96.0% (95% CI: 94.1–97.3%) and 94.5% (95% CI: 92.4–96.1%), respectively. Low birthweight and having multiple co-occurring birth defects were associated with increased HRs ranging between 5 and 20, while having congenital hydrocephalus and receiving hospital transfer from the birth hospital to another hospital for myelomeningocele surgery were associated with HRs indicating a protective association with early neonatal and infant mortality.

Conclusion Maternal race/ethnicity and social disadvantage did not predict early neonatal and infant mortality among infants with myelomeningocele; presence of congenital hydrocephalus and the role of hospital transfer for myelomeningocele repair should be further examined.

Key Points

  • Mortality in myelomeningocele is a concern

  • Social disadvantage was not associated with death

  • Hospital-based factors should be further examined



Publication History

Received: 17 December 2019

Accepted: 14 April 2020

Article published online:
30 May 2020

© 2020. Thieme. All rights reserved.

Thieme Medical Publishers
333 Seventh Avenue, New York, NY 10001, USA.

 
  • References

  • 1 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
  • 2 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
  • 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 Avagliano L, Massa V, George TM, Qureshy S, Bulfamante GP, Finnell RH. Overview on neural tube defects: from development to physical characteristics. Birth Defects Res 2019; 111 (19) 1455-1467
  • 5 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
  • 6 Bowman RM, McLone DG, Grant JA, Tomita T, Ito JA. Spina bifida outcome: a 25-year prospective. Pediatr Neurosurg 2001; 34 (03) 114-120
  • 7 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
  • 8 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
  • 9 Alabi NB, Thibadeau J, Wiener JS. et al. Surgeries and health outcomes among patients with spina bifida. Pediatrics 2018; 142 (03) e20173720
  • 10 Wong LY, Paulozzi LJ. Survival of infants with spina bifida: a population study, 1979-94. Paediatr Perinat Epidemiol 2001; 15 (04) 374-378
  • 11 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
  • 12 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
  • 13 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
  • 14 Lopez KN, Nembhard WN, Wang Y. et al. Birth defect survival for Hispanic subgroups. Birth Defects Res 2018; 110 (04) 352-363
  • 15 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
  • 16 American Academy of Pediatrics Committee on Fetus And Newborn. Levels of neonatal care. Pediatrics 2012; 130 (03) 587-597
  • 17 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
  • 18 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
  • 19 Wang Y, Liu G, Canfield MA. et al; National Birth Defects Prevention Network. Racial/ethnic differences in survival of United States children with birth defects: a population-based study. J Pediatr 2015; 166 (04) 819-26.e1 , 2
  • 20 Spina Bifida Association. Guidelines for the care of people with spina bifida. Accessed July 19, 2019 at: http://spinabifidaassociation.org/wp-content/uploads/2018/10/GFPWSB-Update-102518.pdf
  • 21 Kancherla V, Ma C, Grant G. et al. Factors associated with timeliness of surgical repair among infants with myelomeningocele: California perinatal quality care collaborative, 2006. Am J Perinatol 2011
  • 22 Marengo LK, Hoyt AT, Canfield MA. The utility of the national death index as a supplemental data source in ascertaining 5-year mortality among Texas heterotaxy cases. J Registry Manag 2014; 41 (01) 4-6
  • 23 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