Margaret C. Tyler

Health-Related Quality of Life Among Preadolescent Children With Oral Clefts: The Mother's Perspective

OBJECTIVE. The purpose of this study was to evaluate the factors that affect the health-related quality of life of preadolescent children with nonsyndromic oral clefts using the Pediatric Quality of Life Inventory instrument and to evaluate whether there were any differences related to the type of cleft or other factors. METHODS. Data for this study were derived from telephone interviews with the parents of a statewide population of children who were in the Iowa Registry for Congenital and Inherited Disorders, were aged 2 to 12 years, had nonsyndromic oral clefts, and were born in Iowa between January 1, 1990, and December 31, 2000. Twenty-minute interviews were conducted with mothers of 104 children in the spring and summer of 2003; respondents then completed and mailed back Pediatric Quality of Life Inventory surveys 2 to 3 weeks after the interviews (69% participation rate). RESULTS. After controlling for demographic characteristics, children with less severe speech problems had higher total Pediatric Quality of Life Inventory scores as well as higher physical and psychosocial health domain scores. Age and cleft type interacted, with younger children (aged 2–7 years) with a cleft lip or cleft lip and palate having higher health-related quality of life scores than children with an isolated cleft palate; however, this pattern was reversed for older children (aged 8–12 years). CONCLUSIONS. Speech and aesthetic concerns seem to have been important factors affecting the health-related quality of life for children with oral clefts. These factors seem to be more important as children get closer to adolescence (ages 8–12 years) when acceptance by peers becomes more critical.

Cost of Outpatient Medical Care for Children and Youth With Special Health Care Needs: Investigating the Impact of the Medical Home

OBJECTIVE. Our goal was to evaluate the impact of having a medical home on the outpatient medical costs of children and youth with special health care needs. DESIGN. Data from 2 sources were matched at the individual level: (1) the 2002 Iowa Consumer Assessment of Health Plans Study survey of Medicaid enrollees and (2) Iowa Medicaid administrative claims, encounter, and enrollment files. PARTICIPANTS. The subjects were 1140 children aged 6 months to 12 years for whom both sources of data were available. MAIN OUTCOME MEASURE(S). Outcomes measures included medical homeness, as developed by a scale of items in the Consumer Assessment of Health Plans Study survey, and outpatient costs, as determined from Medicaid administrative data. RESULTS. From the regression models, we found that (1) for all Medicaid-enrolled children, outpatient costs were significantly higher for female children and children and youth with special health care needs, (2) for children and youth without special health care needs, costs were significantly higher for female children, those with a personal doctor or nurse, and those with more of a medical home, and (3) for children and youth with special health care needs, costs were significantly higher for those in a lower health state, for those in health maintenance organization 2, and for older children. CONCLUSIONS. Although the degree of medical homeness was not related to outpatient costs for children and youth with special health care needs, medical homeness may affect inpatient costs more than outpatient costs for children and youth with special health care needs and should be investigated further.

Oral Clefts and Behavioral Health of Young Children

OBJECTIVES This study examined the behavioral health of young children with oral clefts, and effects of satisfaction with facial appearance, cleft team care, number of cleft-related surgeries, and socioeconomic status (SES). SUBJECTS AND METHODS The study included a population-based sample of 104 children aged 2-12 years with isolated oral clefts from the state of Iowa. Behavior was evaluated with the Child Behavior Checklist or the Pediatric Behavior Scale 30, depending on age, compared with normative samples. RESULTS Risks of behavioral problems were not significantly different from normative samples except for higher inattention/hyperactivity risks at age 6-12 years. Low satisfaction with facial appearance was associated with behavioral problems in all domains, except aggression. Team-care effects were not associated with behavioral problems. Number of cleft-related surgeries was associated with increased anxiety/depression and somatic symptom risks. Higher SES was associated with reduced inattention/hyperactivity, aggressive/oppositional behavior, and somatic symptoms. CONCLUSIONS Most children with oral clefts may have similar behavioral health outcomes to unaffected children, except for increased inattention/hyperactivity risks at older ages. However, low satisfaction with facial appearance, increased exposure to surgeries, and lower SES may significantly increase behavioral problems. Also, the findings emphasize the need to study the representation of behavioral health professionals on cleft teams and access to behavioral health care.

Prenatal diagnosis of orofacial clefts: association with maternal satisfaction, team care, and treatment outcomes.

Objective Prenatal diagnosis of an orofacial cleft is thought to allow mothers greater opportunity to become prepared for the special needs of an infant with a cleft and plan for the care of their child. Using a population-based sample, we determined which children were more likely to be diagnosed prenatally, and whether early diagnosis was associated with maternal satisfaction and treatment outcomes. Design Interviews were completed with 235 (49% of eligible) mothers of children ages 2 to 7 with orofacial clefts initially enrolled in the National Birth Defects Prevention Study from the Arkansas, Iowa, and New York sites. Maternal satisfaction with information, support, and treatment outcomes was compared between women who received a prenatal diagnosis and those who did not. Results Of 235 infants with clefts, 46 (19.6%) were identified prenatally. One third of mothers were somewhat or not satisfied with information provided by medical staff. Satisfaction did not vary by timing of the diagnosis. Infants diagnosed prenatally were no more likely to have received care provided by a recognized multidisciplinary cleft team (76%) than were infants diagnosed at birth (78%). Speech problems and facial appearance as rated by the mother did not vary by timing of the diagnosis. Conclusions Timing of the cleft diagnosis did not alter maternal satisfaction with information, whether care was provided by a designated cleft team, or maternal perception of facial appearance or speech. Further research should determine whether prenatal diagnoses alter maternal anxiety or influence postnatal morbidity.

Differential use of the CAHPS® 0–10 global rating scale by medicaid and commercial populations

The primary objective of this study was to investigate whether Medicaid managed care enrollees and commercially insured health plan participants respond differently to the CAHPS® 2.0 health plan survey global ratings of health care, personal doctor or nurse, and health plan. A secondary objective was to examine whether and how these differences may vary by alternative approaches to collapsing the 0-10 response scale. This study is a secondary analysis of CAHPS 2.0 health plan survey data collected in 1999 and 2000. Data on 2,142 Iowa Medicaid managed care enrollees and 1,051 commercially insured State of Iowa employees were analyzed. Differences in responses between the Medicaid-enrolled and commercially insured respondents were modeled using multinomial logistic regression, adjusting for demographics, health status and CAHPS composite measures. Results of these analyses indicated that Medicaid enrollees were significantly more likely than State of Iowa employees to use the extreme ends of the CAHPS global rating scales, particularly in the approaches when the category representing the highest end of the scale was defined as a score of 10 for the analysis. Thus, the choice of cut points for collapsing the 0-10 scales influenced statistical differences on CAHPS global ratings of care, doctor and health plan between Medicaid and privately insured populations. In conclusion, a populations use of the extremes of the global rating scales should be considered when comparing or combining CAHPS data for different populations. If response contraction bias is present, a format such as the alternative approach presented here (using categories 0-4, 5-8, 9, 10) that captures that bias may be preferable to the CAHPS format, which has been shown to maximize plan differentiation.

Separation Anxiety in Children Ages 4 Through 9 With Oral Clefts

Background Psychosocial research on children with oral clefts (OCs), i.e., clefts of the lip, palate, or lip and palate, has suggested that these children may have higher rates of anxiety in general, but overall results have been equivocal. In this study we estimated the prevalence of separation anxiety disorder (SAD) in a population-based sample of children with OCs and identified associated risk factors. Methods Parents of 279 children with OCs, identified through three state birth defect registries, responded to a postal survey that included the Separation Anxiety subscale of the Screen for Child Anxiety-Related Emotional Disorders (SCARED) and items regarding the childs OC and its sequelae, parent and child health, parent mental health, family structure, and socioeconomic status indicators. Associations with SA were evaluated using bivariate and multivariate statistics. Results One quarter (24%) of the children screened positive for SAD, which was substantially higher than the U.S. child population estimates of 3% to 5%. OC-related impairments in speaking and eating more than doubled the risk of SAD, as did lower socioeconomic status and lower maternal health rating. Maternal mental health and marital status remained in the logistic model but were not significant. Conclusions Children with OCs appear to be at increased risk for SAD as measured by the SCARED instrument. OC-related problems with speech and eating more than doubled the risk of SAD. The use of targeted screening tools may help refine our understanding of psychosocial adjustment in children with OCs.

Interdisciplinary craniofacial teams compared with individual providers: is orofacial cleft care more comprehensive and do parents perceive better outcomes?

Objective The primary objective was to examine whether children with orofacial clefts received more comprehensive care and whether their parents perceived better outcomes if the care was delivered by interdisciplinary teams compared with individual providers. Design Data about services received and outcomes were collected from mothers of children with orofacial clefts. Participants Mothers of children born between 1998 and 2003 with orofacial clefts from Arkansas, Iowa, and New York who participated in the National Birth Defects Prevention Study were eligible. Main Outcome Measure(s) Services and treatments received and maternal perception of cleft care, health status, aesthetics, and speech were evaluated by team care status. Results Of 253 children, 24% were not receiving team care. Of those with cleft lip and palate, 86% were enrolled in team care. Compared with children with team care, those without had fewer surgeries and were less likely to have seen a dentist, received a hearing test, or had a genetic consultation. Mothers of children lacking team care were twice as likely to give lower ratings for overall cleft care; maternal perceptions of global health, facial appearance, and speech did not differ by team care status. Conclusions Recommended care tended to be received more often among those with team care. A larger, longitudinal study might answer questions about whether team care provides the best care and the role that type and severity of the condition and racial/ethnic differences play in the services received and outcomes experienced.

Maternal reports of satisfaction with care and outcomes for children with craniosynostosis.

Craniosynostosis is a congenital deformity caused by premature fusion of cranial suture(s). Surgical outcomes of craniosynostosis have been well documented; however, limited published data are available regarding access to care and social and psychological health outcomes for children with craniosynostosis. The authors conducted a population-based, multistate study of maternal perceptions of care and outcomes for children with craniosynostosis born from January 1998 through December 2003. Mothers of 147 children were contacted and asked to complete a structured telephone interview; 82 mothers (55.8%) participated. Overall, the mean ages of children and their mothers at the time of interview were 4.4 and 34.8 years, respectively. Mothers interviewed tended to be white and, at a minimum, high school graduates. Most mothers reported being satisfied with the craniosynostosis-related information and support provided by medical care providers, and access to primary and specialty care was most often rated as satisfactory. Mothers tended to report the physical health of their child to be good; however, delays in cognitive and emotional development, hearing loss, and speech problems were identified. In addition, statistically significant differences among mothers in each state were noted for reports of the quality of team care, primary care, learning interventions, and perceived need for additional surgeries. These results suggest some limitations in craniosynostosis-related care and provide guidance to medical and surgical care providers in making need-based improvements in craniosynostosis surgery and care. Future studies are recommended to replicate this evaluation in additional populations.

Type of Oral Cleft and Mothers’ Perceptions of Care, Health Status, and Outcomes for Preadolescent Children

Objective: To evaluate the outcomes of care for children by type of oral cleft. Design: Data were collected through structured telephone interviews during 2003 in Iowa with mothers of 2- to 12-year-old children with oral clefts. Interviews with mothers of children with clubfoot and statewide data on Iowa children were used for comparison. Participants: Participants included mothers of children in Iowa born between 1990 and 2000 with nonsyndromic oral clefts. Children were identified by the statewide Iowa Registry for Congenital and Inherited Disorders. Main Outcome Measures: Rating of cleft care, severity of condition, health status, esthetic outcome, speech, and school performance were evaluated by type of oral cleft. Results: Children with cleft lip and palate were most likely to have their clefts rated as very severe. Children with palatal involvement were reported to have a lower health status and were almost twice as likely to be identified as having a special health care need compared with either children with cleft lip or children statewide. Children with cleft lip had more esthetic concerns; children with palatal involvement had the most speech concerns. Conclusions: Although mothers generally believed their children had received high-quality care, ratings of the childrens current health status and outcomes of care varied significantly by type of cleft (cleft lip, cleft palate, and cleft lip and palate). Differences observed in this population-based study support the proposition that cleft type should be considered when examining outcomes of care for children with oral clefts.

Demographic Characteristics, Care, and Outcomes for Children with Oral Clefts in Three States Using Participants from the National Birth Defects Prevention Study:

Objective: The primary objective of this study was to evaluate whether there were differences in the characteristics and outcomes of care for children with oral clefts (OCs) among population-based samples in three states. Design: Data on the health status and on speech and esthetic outcomes were collected using structured telephone interviews conducted during 2005–2006 with mothers of children with OCs aged 2 to 7 in Arkansas, Iowa, and New York. Participants: Mothers of children born with nonsyndromic OCs on or after January 1, 1998, and on or before December 31, 2003, in Arkansas, Iowa, or New York. Subjects were identified through their participation in the ongoing National Birth Defects Prevention Study. Main Outcome Measures: Demographic characteristics, rating of cleft care, severity of condition, health status, esthetic outcomes, and speech problems were evaluated by state of residence. Results: Children with OCs from Arkansas were from lower income families, and their parents were less likely to be married. Children with OCs from Arkansas were more likely to have special health care needs and to require mental health care. Few differences were found across states in type of cleft, severity of cleft, or outcomes of cleft care. Conclusions: Combining results from population-based samples across multiple studies increases the variability of sample characteristics. Including multiple states can be an efficient way to learn more about the outcomes of medical care for less common conditions such as oral cleft.