Trends in Unmet Need for Genetic Counseling Among Children With Special Health Care Needs, 2001–2010 Anna Jo Smith, MS, MPH; Donald Oswald, PhD; Joann Bodurtha, MD, MPH From the Harvard Medical School, Boston, Mass (Ms Smith); Partnership for People With Disabilities, Virginia Commonwealth University, Richmond, Va (Dr Oswald); and McKusick-Nathans Institute of Genetic Medicine, Johns Hopkins School of Medicine, Baltimore, Md (Dr Bodurtha) The authors declare that they have no conflict of interest. Address correspondence to Donald Oswald, PhD, Partnership for People with Disabilities, Virginia Commonwealth University, PO Box 980405, Richmond, VA 23298 (e-mail: [email protected]). Received for publication February 16, 2015; accepted May 24, 2015.
ABSTRACT OBJECTIVE: Access to genetic counseling is increasingly important to guide families’ and clinicians’ decision making, yet there is limited research on accessibility and affordability of counseling for families with children with special health care needs (CSHCN). Our study’s objectives were to measure changes in unmet need for genetic counseling for CSHCN from 2001 to 2010 and to characterize child, family, and health system factors associated with unmet need. METHODS: We used parent-reported data from the 2001, 2005– 2006, and 2009–2010 National Survey of Children With Special Health Care Needs. We used a logistic regression model to measure the impact of survey year, child (sex, age, severity of health condition), family (primary language, household income, insurance, financial problems related to cost of CSHCN’s health care), and health system factors (region, genetic counselors per capita, having a usual source of care) on access to genetic counseling. RESULTS: Unmet need for genetic counseling increased significantly in 2009–2010 compared to 2001 (odds ratio 1.79; 95%
confidence interval [CI] 1.44–2.47). Being older (adjusted odds ratio [aOR] 1.04; 95% CI 1.02–1.06), having severe health limitations (aOR 1.72; 95% CI 1.16–2.58), being uninsured (aOR 3.56; 95% CI 2.16–5.87), and having family financial problems due to health care costs (aOR 1.90; 95% CI 1.52– 2.38) were significantly associated with greater unmet need for genetic counseling. Having a usual source of care was associated with decreased unmet need (aOR 0.55; 95% CI 0.37– 0.83). CONCLUSIONS: Unmet need for genetic counseling has increased over the past 12 years. Uninsurance and financial problems related to health care costs were the largest drivers of unmet need over time.
KEYWORDS: children with special health care needs; genetic counseling; insurance ACADEMIC PEDIATRICS 2015;-:1–7
WHAT’S NEW
cal organizations, such as the American Academy of Pediatrics (AAP), the American Psychiatric Association, and the American Heart Association, are increasingly emphasizing the importance of genetic counseling and screening in clinical practice.2–4 The AAP established a Genetics in Primary Care Institute in 2011 and integrated genetic counseling in its health supervision guidelines for several conditions.5 Genetic counseling is a key part of translating genetic advances into clinical practice. Genetic counseling may be provided by medical geneticists, masters-level genetic counselors, nurse geneticists, or other health professionals with specific training. Yet national surveys suggest that families’ access to genetic counseling varies widely. Although 1 in 15 children with special health care needs (CSHCN) is estimated to need genetic counseling, nearly one third of CSHCN do not receive needed services.6 Prior research focused on insurance as a barrier: compared to privately insured CSHCN, uninsured or publicly insured CSHCN had greater unmet
In spite of American Academy of Pediatrics’ recommendations for increased usage of genetic services, access to genetic counseling for children with special health care needs decreased from 2001 to 2010. Severe health limitations, financial problems due to health care costs, and lack of a medical home were associated with worse access.
GENETICS PLAYS AN increasingly important role in clinical practice. Many conditions, such as intellectual disabilities and cancer, are now understood to have genetic bases, which can affect prognostic assessment and preventive care. Our knowledge of genetics can guide treatment decisions, especially in pediatrics; for example, modified diets for metabolic disorders and mutation-specific therapies for cystic fibrosis have helped turn previously fatal childhood diseases into manageable conditions.1 Accordingly, mediACADEMIC PEDIATRICS Copyright ª 2015 by Academic Pediatric Association
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need for genetic counseling.7,8 However, the last major studies on access to genetic counseling were published over a decade ago, before state and federal initiatives to decrease uninsurance—and before the AAP and other organizations’ recommendations for greater integration into clinical practice.9 In other types of pediatric specialty care, recent research has shown that, independent of insurance status, financial problems related to health care costs,10 poverty,11 race,12 and severity of children’s health limitations11 are associated with unmet need, but the impact of these family and child factors on access to genetic counseling has not been studied. Health system factors may also influence access to genetic counseling. Pediatric specialists, including geneticists and genetic counselors, tend to cluster at academic health centers, so access may be insufficient in rural areas or other places with few counselors per capita.13 For example, previous research found that CSHCN living in Western states had greater rates of unmet need for genetic counseling than CSHCN elsewhere in the country.14 Previous studies did not assess provider supply, which is highly related to access in other areas of pediatrics.15 Other health system factors, such as having a medical home, may improve access to genetic counseling for CSHCN.7 The aims of our study were to investigate whether access to genetic counseling improved over time and to examine the impact of child, family, and health system factors on access to genetic counseling. To our knowledge, no previous study has comprehensively examined the relative impact of child, family, and health system factors on access to genetic counseling.
METHODS DATA SOURCE We used data from the 2001, 2005–2006, and 2009– 2010 National Survey of Children With Special Health Care Needs (NS-CSHCN) to investigate 1) trends in access to genetic counseling over time, 2) child, family, and health system factors associated with access to genetic counseling, and 3) trends in the impact of child, family, and health system factors on access to genetic counseling over time. The NS-CSHCN is random-digit-dial survey of families stratified by state and weighted to be representative of all noninstitutionalized CSHCN aged 0 to 17 years in the United States and in each state. Interviews were conducted in English, Spanish, Mandarin, Cantonese, Vietnamese, and Korean. Combining the 3 survey years produced a data set including 119,831 interviews. The NS-CSHCN response rate was 61% in 2001, 56% in 2005–2006, and 26% in 2009–2010. We applied survey weights developed by the National Center for Health Statistics to account for possible nonresponse bias in the 2009–2010 survey.16,17 Additional details about the surveys are available elsewhere.18 MAIN OUTCOMES AND COVARIATES We used 2 questions from the NS-CSHCN to measure parent-reported access to genetic counseling. We estimated
need for genetic counseling on the basis of the response to the question, “During the past 12 months, was there any time when you or other family members needed genetic counseling for advice about inherited conditions related to [your child]’s medical, behavioral, or other health conditions?” Parents who responded yes to that question were then asked, “Did you or your family receive all the genetic counseling that was needed?” Parents who answered no to this second question were considered to have an unmet need for genetic counseling. We measured need for genetic counseling and the unmet need for genetic counseling in each survey year and examined trends over time. In addition to survey year, we analyzed the impact of child, family, and health system characteristics on unmet need for genetic counseling. We imputed missing demographic data using a random digit generator, which occurred for 0.2% to 2.6% of cases across covariates. Child variables were age, sex, race/ethnicity (Hispanic, black non-Hispanic, white non-Hispanic, other non-Hispanic), and health limitations (none, very little, some, a great deal). The health limitation variable was based on parents’ responses to the question, “Do [your child]’s (medical, behavioral, or other health conditions, emotional, developmental, or behavioral problems) affect [his/her] ability to do things a great deal, some or very little, [or not at all]?” Family variables included income (below 100% federal poverty level [FPL], 100% to 200% FPL, 200% to 400% FPL, above 400% FPL), child insurance status (insured, noninsured), family financial problems related to the cost of the CSHCN’s health care (yes, no), and primary language spoken at home (English, not English). We classified CSHCN as insured if they had public insurance, private insurance, both public and private insurance, or other comprehensive insurance. CSHCN were classified as uninsured if they were uninsured at the time of the survey. Health system variables were census region (Northeast, Midwest, South, and West), usual source of care (yes, no), and number of genetic counselors per 1 million population. We obtained the number of genetic counselors from a 2009–2010 database maintained by the National Society of Genetic Counselors (K. Maude, e-mail communication, 2014). We then divided the overall number of counselors by the state population in millions (2010 census) to calculate a per capita measure of genetic counselors. All included variables are considered comparable across survey years by the Data Resource Center for Child and Adolescent Health.19,20 We included only 1 measure of medical home (usual source of care) in our analysis because questions on other components of medical homes changed significantly between surveys. The 2009–2010 NS-CSHCN included a cell phone sample in addition to the traditional landline sample. We include sample type as a covariate in the full model. ANALYSIS We examined the frequency of need for genetic counseling and unmet need in each survey year. We compared access to genetic counseling across survey years using linear and logistic regression. We also analyzed access to
ACADEMIC PEDIATRICS
UNMET NEED FOR GENETIC COUNSELING
genetic counseling stratified by race/ethnicity, insurance status, and household income. To understand the multifactorial influences on access to genetic counseling, we added child, family, and health system variables to our logistic regression model. We report unadjusted (ie, bivariate) and adjusted (ie, full model) odds ratios for each variable. We considered a P value of
ABSTRACT OBJECTIVE: Access to genetic counseling is increasingly important to guide families’ and clinicians’ decision making, yet there is limited research on accessibility and affordability of counseling for families with children with special health care needs (CSHCN). Our study’s objectives were to measure changes in unmet need for genetic counseling for CSHCN from 2001 to 2010 and to characterize child, family, and health system factors associated with unmet need. METHODS: We used parent-reported data from the 2001, 2005– 2006, and 2009–2010 National Survey of Children With Special Health Care Needs. We used a logistic regression model to measure the impact of survey year, child (sex, age, severity of health condition), family (primary language, household income, insurance, financial problems related to cost of CSHCN’s health care), and health system factors (region, genetic counselors per capita, having a usual source of care) on access to genetic counseling. RESULTS: Unmet need for genetic counseling increased significantly in 2009–2010 compared to 2001 (odds ratio 1.79; 95%
confidence interval [CI] 1.44–2.47). Being older (adjusted odds ratio [aOR] 1.04; 95% CI 1.02–1.06), having severe health limitations (aOR 1.72; 95% CI 1.16–2.58), being uninsured (aOR 3.56; 95% CI 2.16–5.87), and having family financial problems due to health care costs (aOR 1.90; 95% CI 1.52– 2.38) were significantly associated with greater unmet need for genetic counseling. Having a usual source of care was associated with decreased unmet need (aOR 0.55; 95% CI 0.37– 0.83). CONCLUSIONS: Unmet need for genetic counseling has increased over the past 12 years. Uninsurance and financial problems related to health care costs were the largest drivers of unmet need over time.
KEYWORDS: children with special health care needs; genetic counseling; insurance ACADEMIC PEDIATRICS 2015;-:1–7
WHAT’S NEW
cal organizations, such as the American Academy of Pediatrics (AAP), the American Psychiatric Association, and the American Heart Association, are increasingly emphasizing the importance of genetic counseling and screening in clinical practice.2–4 The AAP established a Genetics in Primary Care Institute in 2011 and integrated genetic counseling in its health supervision guidelines for several conditions.5 Genetic counseling is a key part of translating genetic advances into clinical practice. Genetic counseling may be provided by medical geneticists, masters-level genetic counselors, nurse geneticists, or other health professionals with specific training. Yet national surveys suggest that families’ access to genetic counseling varies widely. Although 1 in 15 children with special health care needs (CSHCN) is estimated to need genetic counseling, nearly one third of CSHCN do not receive needed services.6 Prior research focused on insurance as a barrier: compared to privately insured CSHCN, uninsured or publicly insured CSHCN had greater unmet
In spite of American Academy of Pediatrics’ recommendations for increased usage of genetic services, access to genetic counseling for children with special health care needs decreased from 2001 to 2010. Severe health limitations, financial problems due to health care costs, and lack of a medical home were associated with worse access.
GENETICS PLAYS AN increasingly important role in clinical practice. Many conditions, such as intellectual disabilities and cancer, are now understood to have genetic bases, which can affect prognostic assessment and preventive care. Our knowledge of genetics can guide treatment decisions, especially in pediatrics; for example, modified diets for metabolic disorders and mutation-specific therapies for cystic fibrosis have helped turn previously fatal childhood diseases into manageable conditions.1 Accordingly, mediACADEMIC PEDIATRICS Copyright ª 2015 by Academic Pediatric Association
1
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need for genetic counseling.7,8 However, the last major studies on access to genetic counseling were published over a decade ago, before state and federal initiatives to decrease uninsurance—and before the AAP and other organizations’ recommendations for greater integration into clinical practice.9 In other types of pediatric specialty care, recent research has shown that, independent of insurance status, financial problems related to health care costs,10 poverty,11 race,12 and severity of children’s health limitations11 are associated with unmet need, but the impact of these family and child factors on access to genetic counseling has not been studied. Health system factors may also influence access to genetic counseling. Pediatric specialists, including geneticists and genetic counselors, tend to cluster at academic health centers, so access may be insufficient in rural areas or other places with few counselors per capita.13 For example, previous research found that CSHCN living in Western states had greater rates of unmet need for genetic counseling than CSHCN elsewhere in the country.14 Previous studies did not assess provider supply, which is highly related to access in other areas of pediatrics.15 Other health system factors, such as having a medical home, may improve access to genetic counseling for CSHCN.7 The aims of our study were to investigate whether access to genetic counseling improved over time and to examine the impact of child, family, and health system factors on access to genetic counseling. To our knowledge, no previous study has comprehensively examined the relative impact of child, family, and health system factors on access to genetic counseling.
METHODS DATA SOURCE We used data from the 2001, 2005–2006, and 2009– 2010 National Survey of Children With Special Health Care Needs (NS-CSHCN) to investigate 1) trends in access to genetic counseling over time, 2) child, family, and health system factors associated with access to genetic counseling, and 3) trends in the impact of child, family, and health system factors on access to genetic counseling over time. The NS-CSHCN is random-digit-dial survey of families stratified by state and weighted to be representative of all noninstitutionalized CSHCN aged 0 to 17 years in the United States and in each state. Interviews were conducted in English, Spanish, Mandarin, Cantonese, Vietnamese, and Korean. Combining the 3 survey years produced a data set including 119,831 interviews. The NS-CSHCN response rate was 61% in 2001, 56% in 2005–2006, and 26% in 2009–2010. We applied survey weights developed by the National Center for Health Statistics to account for possible nonresponse bias in the 2009–2010 survey.16,17 Additional details about the surveys are available elsewhere.18 MAIN OUTCOMES AND COVARIATES We used 2 questions from the NS-CSHCN to measure parent-reported access to genetic counseling. We estimated
need for genetic counseling on the basis of the response to the question, “During the past 12 months, was there any time when you or other family members needed genetic counseling for advice about inherited conditions related to [your child]’s medical, behavioral, or other health conditions?” Parents who responded yes to that question were then asked, “Did you or your family receive all the genetic counseling that was needed?” Parents who answered no to this second question were considered to have an unmet need for genetic counseling. We measured need for genetic counseling and the unmet need for genetic counseling in each survey year and examined trends over time. In addition to survey year, we analyzed the impact of child, family, and health system characteristics on unmet need for genetic counseling. We imputed missing demographic data using a random digit generator, which occurred for 0.2% to 2.6% of cases across covariates. Child variables were age, sex, race/ethnicity (Hispanic, black non-Hispanic, white non-Hispanic, other non-Hispanic), and health limitations (none, very little, some, a great deal). The health limitation variable was based on parents’ responses to the question, “Do [your child]’s (medical, behavioral, or other health conditions, emotional, developmental, or behavioral problems) affect [his/her] ability to do things a great deal, some or very little, [or not at all]?” Family variables included income (below 100% federal poverty level [FPL], 100% to 200% FPL, 200% to 400% FPL, above 400% FPL), child insurance status (insured, noninsured), family financial problems related to the cost of the CSHCN’s health care (yes, no), and primary language spoken at home (English, not English). We classified CSHCN as insured if they had public insurance, private insurance, both public and private insurance, or other comprehensive insurance. CSHCN were classified as uninsured if they were uninsured at the time of the survey. Health system variables were census region (Northeast, Midwest, South, and West), usual source of care (yes, no), and number of genetic counselors per 1 million population. We obtained the number of genetic counselors from a 2009–2010 database maintained by the National Society of Genetic Counselors (K. Maude, e-mail communication, 2014). We then divided the overall number of counselors by the state population in millions (2010 census) to calculate a per capita measure of genetic counselors. All included variables are considered comparable across survey years by the Data Resource Center for Child and Adolescent Health.19,20 We included only 1 measure of medical home (usual source of care) in our analysis because questions on other components of medical homes changed significantly between surveys. The 2009–2010 NS-CSHCN included a cell phone sample in addition to the traditional landline sample. We include sample type as a covariate in the full model. ANALYSIS We examined the frequency of need for genetic counseling and unmet need in each survey year. We compared access to genetic counseling across survey years using linear and logistic regression. We also analyzed access to
ACADEMIC PEDIATRICS
UNMET NEED FOR GENETIC COUNSELING
genetic counseling stratified by race/ethnicity, insurance status, and household income. To understand the multifactorial influences on access to genetic counseling, we added child, family, and health system variables to our logistic regression model. We report unadjusted (ie, bivariate) and adjusted (ie, full model) odds ratios for each variable. We considered a P value of
