| Literature DB >> 35778857 |
Vibeke Müller1, Ulf Gerdtham1,2, Ann Alriksson-Schmidt3, Johan Jarl1.
Abstract
This study analyzes the relationship of having a child with the early-onset disability cerebral palsy (CP) and the parental decision to divorce and to have additional children. We use longitudinal matched case-control data from multiple linked Swedish National Population Registers between 2001 and 2015 and perform Cox proportional hazards regressions with interval-censoring. Although we do not find a general excess parental divorce risk on CP relative to the comparison group without CP, we find that having a child with CP increases the risk of divorce for parents with low education. We also find that having a child with CP reduces the likelihood of having additional children, especially for mothers in the older age range (maternal age at delivery >33 years) and parents with low education. The severity level of the disability, as indicated by gross motor function, is not related to the results. These findings should be understood in the Swedish context, which provides extensive welfare support (e.g., personal assistance). If future studies would find adverse results in countries with less social care and benefits, our results may indicate that it is possible to mitigate negative consequences for the family of a child with disability.Entities:
Keywords: cerebral palsy; divorce; early-onset disability; family formation; register-based study
Mesh:
Year: 2022 PMID: 35778857 PMCID: PMC9543549 DOI: 10.1002/hec.4567
Source DB: PubMed Journal: Health Econ ISSN: 1057-9230 Impact factor: 2.395
Summary statistics
| Variables | No CP | CP | Mild CP | Severe CP |
|---|---|---|---|---|
| Firstborn child | 5625 (42.6%) |
| 657 (44.6%) |
|
| Multiple birth | 33 (0.2%) |
|
|
|
| Parental education level | ||||
| Mandatory | 576 (4.4%) | 82 (4.1%) | 58 (3.9%) | 24 (4.8%) |
| Secondary | 4884 (37.0%) |
|
| 202 (40.1%) |
| Higher | 7745 (58.7%) |
|
| 278 (55.2%) |
| Maternal age at delivery | ||||
| 16–29 | 5106 (38.7%) | 732 (37.0%) | 554 (37.6%) | 178 (35.3%) |
| 30–33 | 4007 (30.3%) | 599 (30.3%) | 438 (29.8%) | 161 (31.9%) |
| 34–51 | 4092 (31.0%) | 645 (32.6%) | 480 (32.6%) | 165 (32.7%) |
| Maternal mental health diagnosis | ||||
| No | 11,952 (90.5%) |
| 1318 (89.5%) |
|
| Yes | 575 (4.4%) |
|
| 30 (6.0%) |
| Unknown | 678 (5.1%) | 101 (5.1%) | 66 (4.5%) | 35 (6.9%) |
| Maternal CCI above 0 | ||||
| No | 12,137 (91.9%) |
|
| 456 (90.5%) |
| Yes | 334 (2.5%) |
|
| 13 (2.6%) |
| Unknown | 734 (5.6%) | 106 (5.4%) | 71 (4.8%) | 35 (6.9%) |
| Mother smoked before pregnancy | ||||
| No | 10,520 (79.7%) |
|
|
|
| Yes | 1871 (14.2%) | 307 (15.5%) |
| 70 (13.9%) |
| Unknown | 814 (6.2%) |
|
|
|
| Mother smoked during pregnancy | ||||
| No | 11,603 (87.9%) |
|
|
|
| Yes | 834 (6.3%) | 142 (7.2%) | 109 (7.4%) | 33 (6.5%) |
| Unknown | 768 (5.8%) |
|
|
|
| Paternal CCI above 0 | ||||
| No | 12,309 (93.2%) | 1822 (92.2%) | 1369 (93.0%) |
|
| Yes | 372 (2.8%) | 55 (2.8%) | 34 (2.3%) | 21 (4.2%) |
| Unknown | 524 (4.0%) |
| 69 (4.7%) |
|
| Female child | 5455 (41.3%) | 822 (41.6%) | 602 (40.9%) | 220 (43.7%) |
| Mother/father immigrated | 3480 (26.4%) | 530 (26.8%) | 372 (25.3%) | 158 (31.3%) |
| Parents married | 6537 (49.5%) | 949 (48.0%) | 697 (47.4%) | 252 (50.0%) |
| Paternal mental health diagnosis | ||||
| No | 12,414 (94.0%) |
| 1372 (93.2%) |
|
| Yes | 333 (2.5%) | 55 (2.8%) | 40 (2.7%) | 15 (3.0%) |
| Unknown | 45 (3.5%) |
| 60 (4.1%) |
|
| Number of children | 13,205 | 1976 | 1472 | 504 |
Note: Authors' estimates of average characteristics using linked registry data from parents and their children born 2004–2014. Significant differences (on 0.05 level) compared to the comparison group (column 1) are displayed in bold. Mild CP encompasses individuals at GMFCS levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels.
Abbreviations: CP, Cerebral Palsy; CCI, Charlson Comorbidity Index.
FIGURE 1Proportional Hazards (PH) assumption test for having a child with cerebral palsy (CP) in divorce risk analysis. CP this figure shows the log‐log survival curves for the treatment and comparison groups (i.e., having a child without CP or with CP on the left, and no CP, mild CP or severe CP on the right) regarding the risk of the divorce/separation. The hazard curves are adjusted for all covariates from the full specification (see columns 3 and 4 in Table 2). A parallel development of the plots indicates that the PH assumption is fulfilled. Mild CP encompasses individuals at gross motor function classification system (GMFCS) levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels.
Hazard ratios of time to parental divorce
| CP diagnosis | HR |
| 95% CI | |
|---|---|---|---|---|
| (1) Baseline | No CP | 1.00 (Ref.) | ||
| CP | 1.07 | 0.267 | [0.95, 1.19] | |
| (2) Covariates predicting CP | No CP | 1.00 (Ref.) | ||
| CP | 1.03 | 0.574 | [0.92, 1.15] | |
| (3) Full specification | No CP | 1.00 (Ref.) | ||
| CP | 1.03 | 0.617 | [0.92, 1.15] | |
| (4) Full specification accounting for severity of CP | No CP | 1.00 (Ref.) | ||
| Mild CP | 1.05 | 0.414 | [0.93, 1.19] | |
| Severe CP | 0.95 | 0.664 | [0.77, 1.18] | |
| Observations | 15,181 |
Note: Authors' estimates of time to divorce from Cox PH model for interval‐censored survival‐time data, using registry data of 15,181 children born 2004–2014 and their parents. Specification (1) to (3) stepwise include more controls to a model differentiating between parents of a child without CP or with CP. Specification (1) includes no controls, specification (2) includes variables predicting CP in our sample (i.e., firstborn child, multiple birth, parental education level, maternal age at delivery, maternal mental health, maternal CCI (Charlson Comorbidity Index), mother smoked before pregnancy, mother smoked during pregnancy, paternal CCI), and specification (3) adds child's birth year, child's sex, marital status, immigration background, region of residence, and paternal mental health to the controls used in specification (2). Specification (4) differentiates between no CP, mild CP and severe CP and includes the controls from specification (3). Mild CP encompasses individuals at GMFCS levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels.
Abbreviations: CI, Confidence Interval; CP, Cerebral Palsy; HR, Hazard Ratio.
FIGURE 2Smoothed hazard function and cumulated failure estimates of divorce. Cerebral palsy (CP). This figure shows smoothed hazard rates and cumulated failure estimates of divorce/separation for parents of a child with no CP, mild CP, or severe CP, based on the analysis in column 4, Table 2. The smoothed HR on the left show the likelihood of divorce/separation across the severity groups in each period, given that a divorce did not happen in any previous period. The cumulated failure rates display the proportion of divorced parents across time. Mild CP encompasses individuals at gross motor function classification system (GMFCS) levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels.
Sensitivity analysis: Risk of parental divorce
| (1) | (2) | |||||
|---|---|---|---|---|---|---|
| Full specification | Full specification accounting for severity of CP | |||||
| TR |
| 95% CI | TR |
| 95% CI | |
| CP diagnosis | ||||||
| No CP | 1.00 (Ref.) | 1.00 (Ref.) | ||||
| CP | 0.96 | 0.532 | [0.85, 1.09] | |||
| Mild CP | 0.94 | 0.377 | [0.82, 1.08] | |||
| Severe CP | 1.03 | 0.780 | [0.82, 1.31] | |||
| Observations | 15,181 | 15,181 | ||||
Note: Authors' estimates of time to divorce using registry data of 15,181 children born 2004–2014 and their parents and controls for the covariates of the full specification (see column 3 and 4 of Table 2) and displays the result when running a lognormal parametric model. Mild CP encompasses individuals at GMFCS levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels.
Abbreviations: CP, Cerebral Palsy; HR, Hazard Ratio; TR, Time Ratio; CI, Confidence Interval.
Heterogeneity analysis: Risk of parental divorce
| (1) | ||||
|---|---|---|---|---|
| Full specification | ||||
| HR |
| 95% CI | ||
| (1) | CP diagnosis × maternal age at delivery | |||
| CP × 16–29 years | 1.02 | 0.836 | [0.87, 1.18] | |
| CP × 30–33 years | 1.14 | 0.285 | [0.90, 1.43] | |
| CP × 34–51 years | 0.96 | 0.759 | [0.77, 1.21] | |
|
| 0.603 | |||
| (2) | CP diagnosis × parental education | |||
| CP × mandatory |
| 0.004 | [1.17, 2.33] | |
| CP × secondary | 0.95 | 0.496 | [0.81, 1.11] | |
| CP × higher | 1.04 | 0.708 | [0.86, 1.24] | |
|
|
| |||
| (3) | CP diagnosis × parental marital status | |||
| CP × unmarried | 1.08 | 0.298 | [0.94, 1.23] | |
| CP × married | 0.95 | 0.616 | [0.79, 1.15] | |
|
| 0.310 | |||
| (4) | CP diagnosis × firstborn child | |||
| CP × No | 1.06 | 0.444 | [0.91, 1.23] | |
| CP × Yes | 0.99 | 0.933 | [0.84, 1.17] | |
|
| 0.568 | |||
| Observations | 15,181 | |||
Note: Authors' estimates from Cox PH models for interval‐censored survival‐time data of durations to parental divorce using registry data of 15,181 children born 2004–2014 and their parents. All columns control for all covariates considered in the full model specification (see column 3, Table 2). Significant effects on 0.05 level are displayed in bold. The estimates display the linear combination of parameters (i.e., the direct effect of having a child with CP multiplied with the effect of having a child with CP interacted with maternal age at delivery, education level, marital status, firstborn child).
Abbreviations: CI, Confidence Interval; CP, Cerebral Palsy; HR, Hazard Ratio.
FIGURE 3Proportional Hazards (PH) assumption test for having a child with cerebral palsy (CP) in likelihood of additional children analysis. CP this figure shows the log‐log survival curves for the treatment and comparison groups (i.e., having a child without CP or with CP on the left, and no CP, mild CP or severe CP on the right) regarding the likelihood of having additional children. The hazard curves are adjusted for all covariates from the full specification (see columns 3 and 4 in Table 2). A parallel development of the plots indicates that the PH assumption is fulfilled. The decreasing hazards show that the likelihood of having additional children decreases over time. Mild CP encompasses individuals at gross motor function classification system (GMFCS) levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels.
Hazard ratios|hazard rates (HR) of time to additional children
| CP diagnosis | HR |
| 95% CI | |
|---|---|---|---|---|
| (1) Baseline | No CP | 1.00 (Ref.) | ||
| CP |
| 0.000 | [0.77, 0.90] | |
| (2) Covariates predicting CP | No CP | 1.00 (Ref.) | ||
| CP |
| 0.000 | [0.72, 0.86] | |
| (3) Full specification | No CP | 1.00 (Ref.) | ||
| CP |
| 0.000 | [0.72, 0.86] | |
| (4) Full specification accounting for severity of CP | No CP | 1.00 (Ref.) | ||
| Mild CP |
| 0.000 | [0.71, 0.87] | |
| Severe CP |
| 0.007 | [0.68, 0.94] | |
| Observations | 15,181 |
Note: Authors' estimates of time to additional children from Cox PH model for interval‐censored survival‐time data, using registry data of 15,181 children born 2004–2014 and their parents. Specification (1) to (3) stepwise include more controls to a model differentiating between parents of a child without CP or with CP. Specification (1) includes no controls, specification (2) includes variables predicting CP in our sample (i.e., firstborn child, multiple birth, parental education level, maternal age at delivery, maternal mental health, maternal CCI (Charlson Comorbidity Index), mother smoked before pregnancy, mother smoked during pregnancy, paternal CCI), and specification (3) adds child's birth year, child's sex, marital status, immigration background, region of residence, and paternal mental health to the controls used in specification (2). Specification (4) differentiates between no CP, mild CP and severe CP and includes the controls from specification (3). Mild CP encompasses individuals at GMFCS levels I‐III or individuals with subtypes of CP that are more likely to be associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected (GMFCS IV‐V) and who require wheelchairs or individuals with subtypes of CP that are more likely to be associated with higher GMFCS‐levels. Significant effects on 0.05 level are displayed in bold.
Abbreviations: CI, Confidence Interval; CP, Cerebral Palsy; HR, Hazard Ratio.
FIGURE 4Smoothed hazard function and cumulated failure estimates of having additional children cerebral palsy (CP) this figure shows smoothed hazard ratios|hazard rates (HR) and cumulated failure estimates of divorce/separation for parents of a child with no CP, mild CP, or severe CP, based on the analysis in column 4, Table 5. The smoothed HR on the left show the likelihood of having an additional child after the birth of the child in our sample with no CP, mild CP, or severe CP in each period, given that the parents did not had an additional child in any previous period. The cumulated failure rates display the proportion of divorced parents across time. Mild CP encompasses individuals at gross motor function classification system (GMFCS) levels I‐III or individuals with subtypes of CP that are more likely to be associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected (GMFCS IV‐V) and who require wheelchairs or individuals with subtypes of CP that are more likely to be associated with higher GMFCS‐levels.
Sensitivity analysis: Likelihood of having additional children
| (1) | (2) | |||||
|---|---|---|---|---|---|---|
| Full specification | Full specification with mild and severe CP | |||||
| HR |
| 95% CI | HR |
| 95% CI | |
| CP diagnosis | ||||||
| No CP | 1.00 (ref.) | 1.00 (ref.) | ||||
| CP |
| 0.000 | [0.74, 0.87] | |||
| Mild CP |
| 0.000 | [0.72, 0.88] | |||
| Severe CP |
| 0.011 | [0.71, 0.96] | |||
| Observations | 15,181 | 15,181 | ||||
Note: Authors' estimates of time to additional children using registry data of 15,181 children born 2004–2014 and their parents and controls for the covariates of the full specification (see column 3 and 4 of Table 5) and displays the result when running a Gompertz parametric model. Mild CP encompasses individuals at GMFCS levels I‐III or individuals with subtypes of CP that are associated with lower GMFCS‐levels whereas severe CP encompasses individuals whose gross motor function is more severely affected at GMFCS levels IV‐V or individuals with subtypes of CP that are associated with higher GMFCS‐levels. Significant effects on 0.05 level are displayed in bold.
Abbreviations: CI, Confidence Interval; CP, Cerebral Palsy; HR, Hazard Ratio; TR, Time Ratio.
Heterogeneity analysis: Likelihood of additional children
| HR |
| 95% CI | ||
|---|---|---|---|---|
| (1) | CP diagnosis × maternal age at delivery | |||
| CP × 16–29 years |
| 0.001 | [0.70, 0.91] | |
| CP × 30–33 years | 0.88 | 0.098 | [0.75, 1.02] | |
| CP × 34–51 years |
| 0.000 | [0.53, 0.77] | |
|
|
| |||
| (2) | CP diagnosis × parental education | |||
| CP × mandatory |
| 0.007 | [0.38, 0.86] | |
| CP × secondary |
| 0.003 | [0.71, 0.93] | |
| CP × higher |
| 0.000 | [0.70, 0.89] | |
|
| 0.270 | |||
| (3) | CP diagnosis × parental marital status | |||
| CP × unmarried |
| 0.000 | [0.67, 0.85] | |
| CP × married |
| 0.006 | [0.73, 0.95] | |
|
| 0.212 | |||
| (4) | CP diagnosis × firstborn child | |||
| CP × No |
| 0.006 | [0.68, 0.94] | |
| CP × Yes |
| 0.000 | [0.71, 0.87] | |
|
| 0.867 | |||
| Observations | 15,181 | |||
Note: Authors' estimates from Cox PH models for interval‐censored survival‐time data of durations to having additional children using registry data of 15,181 children born 2004–2014 and their parents. All columns control for all covariates considered in the full model specification (see column 3, Table 5). Significant effects on 0.05 level are displayed in bold. The estimates display the linear combination of parameters (i.e., the direct effect of having a child with CP multiplied with the effect of having a child with CP interacted with maternal age at delivery, education level, marital status, firstborn child).
Abbreviations: CI, Confidence Interval; CP, Cerebral Palsy; HR, Hazard Ratio.