MANUSCRIPT CITATION

Saurel‐Cubizolles MJ, Marchand‐Martin L, Pierrat V, Arnaud C, Burguet A, Fresson J, et al. Maternal employment and socio‐economic status of families raising children born very preterm with motor or cognitive impairments: the EPIPAGE cohort study. Dev Med Child Neurol. 2020;62(10):1182-90. doi:10.1111/dmcn.14587. PMID: 32557556

REVIEWED BY

Kate Rawnsley, BPhysio (Hons)
Department of Physiotherapy, The University of Melbourne
Clinical Sciences, Murdoch Children’s Research Institute

Prof Jeanie Cheong, MD
Clinical Sciences, Murdoch Children’s Research Institute
Newborn Research, Royal Women’s Hospital
Obstetrics and Gynaecology, The University of Melbourne

Prof. Alicia Spittle, PhD
Department of Physiotherapy, The University of Melbourne
Clinical Sciences, Murdoch Children’s Research Institute
Newborn Research, Royal Women’s Hospital

TYPE OF INVESTIGATION

Economics of healthcare

QUESTION

In families of children born very preterm from the EPIPAGE cohort in France, does the presence of motor or cognitive impairments compared with no impairments relate to maternal employment 5 and 8 years after birth, or the socioeconomic status of the household 8 years after birth?

METHODS

• Design: Prospective cohort study
• Allocation: Not applicable
• Blinding: Not applicable
• Follow-up period: 8 years
• Setting: Maternity and neonatal units in nine regions of France
• Patients: 1885 families from the EPIPAGE cohort in France were included in the study. The cohort consisted of infants born 22-32 weeks post menstrual age (PMA) in 1997 in 9 regions of France and infants born 22-26 PMA in 1998 in 8 regions of France. 770 of the participating families had a child with a motor or cognitive impairment based upon follow-up assessments of the surviving children between 2 to 8 years. Data obtained at the oldest age was used in the final analysis. The other 1115 participating families of children without impairments were classified as the reference group.
• Intervention: At 2 years of age, questionnaires were completed by the child’s treating physician and mother. At 5 years, children underwent a standardised examination and developmental evaluation (including the Touwen neurological examination and the Kaufman Assessment Battery for Children). At 5 and 8 years, parents completed a further questionnaire about the child’s health, school situation and family characteristics. In addition, disability records were reviewed in five regions.
• Outcomes:
  • Primary outcome: Mother employed at 5 and 8 year follow up (yes/no)
  • Secondary outcomes:
    1. Change of maternal work status between pregnancy and 5-year follow up.
    2. Total monthly family income categorised into three groups (< €1500, €1500-3000, and >€3000 per month); ’financial difficulties for medical care in the past year’ (none, a little, a lot) and ‘financial difficulties for housing in the past year’ (none, a little, a lot) at 8-year follow up.
    3. Social characteristics: Maternal level of education, maternal employment during pregnancy, the socio-economic status of the family (according to the highest occupational status between the parents at birth); maternal age at birth; mother living with or without a partner, assessed at birth and at 5 and 8 year follow up; the size of the family before the birth of the index child and at ages 5 and 8  years.
• Analysis and Sample Size:
  • The family was considered the statistical unit of analysis, not the child. In families with twins, the most seriously affected child was considered for classification.
  • Characteristics at birth and at 5 and 8 years for families of children with impairments (n=770) were compared to the reference group (n= 1115).
  • At each age, multivariate logistic regression models were used to account for social context of families
  • Percentages were compared using c²
  • Missing data on outcomes and covariates were imputed by chained equations using the SAS ‘Multiple Imputation’ procedure.
  • The main outcome indicators were also analysed separately for families with singletons and twins (due to the potential impact of multiple births on maternal employment).
  • A sensitivity analysis was also conducted for females living with or without a partner (to account for family context).
• Patient follow-up: % included in analysis: At 5 years, 20% of eligible (alive, not previously withdrawn or lost to follow up) families did not return the questionnaire and at 8 years, 32% of eligible families did not return the questionnaire.

MAIN RESULTS

Response rates were lower for families with children with severe CP at 5 and 8 years (60% and 48% respectively) and for families with children with severe cognitive impairment at 8 years (45%) compared with the reference group at 5 and 8 years (80% and 68% respectively). Non-respondent mothers were more often younger at birth, living without a partner, and more socially underprivileged than respondent mothers.

Mothers of children with severe motor or cognitive impairments were less likely to have been working during pregnancy than mothers of children without impairments (60% & 69% respectively vs 41% with no impairment). At 5 years post birth, mothers of children with severe impairments, regardless of their employment status during pregnancy, had lower rates of employment than the reference group. At the 8 year follow up, results followed the same trend. In addition, families of children with an impairment had lower incomes and more frequently mentioned financial difficulties than families of children with no impairments at 8 years post birth.

CONCLUSION

Impairments in children born very preterm have a significant long-term effect on the employment and household income of families.  Mothers of very preterm children with severe impairments or in whom severe impairments develop, are often unemployed. Children with severe cerebral palsy were more often from families with pre-existing social disadvantage. Professionals involved in the care of these children should therefore be vigilant to the social situations of the child and families. Improved public policies for children with disabilities and their families may be required.

COMMENTARY

Developmental morbidity is high in preterm infants compared with those born at term(1), with increased risk of cerebral palsy, intellectual disability, sensory impairments and developmental delays (1). Nearly 50% of infants born before 28 weeks’ gestational age have a disability or developmental delay, with 20% having substantial neurological disability (2). Preterm births are more common among underprivileged families (3), and combined with the special requirements and financial burden of caring for a child with a disability, raises the concern regarding the long-term socioeconomic impact for these families.

Saurel-Cubizolles and colleagues (4) investigated maternal employment rates and socioeconomic status up to 8 years after the birth of a preterm child. They reported mothers of children with severe motor or cognitive impairments had lower employment rates at 5 years after birth, and more financial difficulties at 8 years after birth, compared with mothers of preterm children with no impairments. It was noted that mothers of children with severe impairment had less often been working during pregnancy. This finding, combined with other social risk indicators may suggest pre-existing social disadvantage for children who developed severe impairments. The study’s findings are in keeping with other literature regarding disability in the general population, with reduced work participation rates for mothers of children with disabilities (5-7).

The study was undertaken in France, with well-established social, schooling, child care and welfare support systems, which may limit the generalisability of these findings in less well-resourced settings. As the study did not capture data on the social security benefits received by families or disability-related childcare or schooling supports, it is not possible to identify the specific gaps in social policy. Further investigation is definitely warranted to understand the barriers and facilitators to maintaining maternal employment.

Families that responded to the questionnaires were from more privileged social circumstances than children lost to follow up. Multiple imputations were used to account for the missing data; however, the results may have underestimated the impact of impairments on the socio-economic situation of families with children born preterm.

A strength of the study was its large sample size, across 9 regions in France, which reportedly covers one third of the country’s annual births (8). Exploring the impact of locality (metropolitan vs rural), and fathers’ employment status on the socio-economic circumstances of families of children with impairments would have added further depth to the study. Given the reported associations of race/ethnicity and racism-related stress with preterm birth rates, even when social risk is accounted for (9),  it may have been interesting for authors to report ethnicity of mothers. Further exploring maternal characteristics such as this, may identify subpopulations in greater need of targeted support.

This study highlights the need for health professionals to be aware of the social circumstances of the families of preterm children and to assist families in accessing the local welfare supports. This study may assist policy makers, not only in France but globally, to improve social supports for families affected by disabilities and preserve employment for parents of these children.

REFERENCES

1. Saigal S, Doyle LW. An overview of mortality and sequelae of preterm birth from infancy to adulthood. Lancet. 2008;371(9608):261-9.
2. Doyle LW, Roberts G, Anderson PJ, Group VICS. Outcomes at age 2 years of infants< 28 weeks’ gestational age born in Victoria in 2005. J Pediatr. 2010;156(1):49-53. e1.
3. Germany L, Saurel-Cubizolles MJ, Ehlinger V, Napoletano A, Alberge C, Guyard-Boileau B, et al. Social context of preterm delivery in France in 2011 and impact on short-term health outcomes: the EPIPAGE 2 cohort study. Paediatr Perinat Epidemiol. 2015;29(3):184-95.
4. Saurel‐Cubizolles MJ, Marchand‐Martin L, Pierrat V, Arnaud C, Burguet A, Fresson J, et al. Maternal employment and socio‐economic status of families raising children born very preterm with motor or cognitive impairments: the EPIPAGE cohort study. Dev Med Child Neurol. 2020;62(10):1182-90.
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7. Susan I M, Esben M F, Mette M, Peter U. Parental social consequences of having a child with cerebral palsy in Denmark. Dev Med Child Neurol. 2015;57(8):768-75.
8. Azria E, Kayem G, Langer B, Marchand-Martin L, Marret S, Fresson J, et al. Neonatal Mortality and Long-Term Outcome of Infants Born between 27 and 32 Weeks of Gestational Age in Breech Presentation: The EPIPAGE Cohort Study. PLOS ONE. 2016;11(1).
9. Braveman PA, Heck K, Egerter S, Marchi KS, Dominguez TP, Cubbin C, et al. The role of socioeconomic factors in Black-White disparities in preterm birth. Am J Public Health. 2015;105(4):694-702.