Longitudinal study of growth of children with unilateral cleft lip and palate: 2 to 10 years of age.

Objective : To study the growth of children with complete unilateral cleft lip and palate (UCLP) from 2 to 10 years of age and to assess whether growth varied from that of children without UCLP (typical children). Design : Physical growth was one of the outcome measures of a National Institutes of Health-sponsored longitudinal, prospective clinical trial conducted by the University of Florida and the University of São Paulo. Setting : Hospital of Rehabilitation of Craniofacial Anomalies, University of São Paulo (HRAC-USP), Bauru, Brazil. Main Outcome Measures : Height and weight were prospectively measured for 360 healthy children with UCLP who were nonsyndromic, belonged to median socioeconomic status, and received health care at HRAC-USP. To compare growth of children with UCLP to that of typical children, growth curves for UCLP were developed and compared with World Health Organization curves for 2006 and 2007, which were used as reference for typical children. Third-degree polynomials were used to explain the relationship of length and weight with age. Confidence limits of 95% were used for the mean curve using the statistic Z ~ N (0,1). Results : Children with UCLP from 2 to 10 years old presented height and weight growth curves similar to those of typical children for both genders. Conclusion : Children with UCLP from 2 to 10 years old presented physical growth similar to that of typical children.

Symptomatic atlantoaxial instability in an adolescent with trisomy 21 (Down's syndrome).

Atlantoaxial instability (AAI) occurs in 15% of children with Trisomy 21. Health supervision guidelines were revised by the American Academy of Pediatrics in 2011 to reflect advances in care for children with special health care needs (CSHCN). Previous guidelines recommended cervical spine radiological screenings in preschool years to evaluate for atlantoaxial instability. For patients with negative screening, re-screening was recommended if they wished to compete in the Special Olympics, or became symptomatic. We present the case of an adolescent who developed a symptomatic atlantoaxial dislocation despite previous negative radiological screening at the age three (under the 2001 guidelines). This case report highlights the revisions in the 2011 guidelines for health supervision and anticipatory guidance. It underlines the need for a high index of suspicion if symptoms develop. It also addresses the need for a medical home for CSHCN, with health care providers who know the child's baseline health status.

The relationships between fatigue, quality of life, and family impact among children with special health care needs.

OBJECTIVE: To examine the relationships among pediatric fatigue, health-related quality of life (HRQOL), and family impact among children with special health care needs (CSHCNs), specifically whether HRQOL mediates the influence of fatigue on family impact. METHODS: 266 caregivers of CSHCNs were studied. The Pediatric Quality of Life Inventory Multidimensional Fatigue Scale, Pediatric Quality of Life Inventory Generic Scale, and Impact on Family Scale were used to measure fatigue, HRQOL, and family impact, respectively. Linear regressions were used to analyze the designated relationships; path analyses were performed to quantify the mediating effects of HRQOL on fatigue-family impact relationship. RESULTS: Although greater fatigue was associated with family impact (p < .05), the association was not significant after accounting for HRQOL. Path analyses indicated the direct effect of fatigue on family impact was not significant (p > .05), whereas physical and emotional functioning significantly mediated the fatigue-family impact relationship (p < .001). CONCLUSION: Fatigue is related to family impact among CSHCNs, acting through the impairment in HRQOL.

Otologic and audiologic outcomes with the Furlow and von Langenbeck with intravelar veloplasty palatoplasties in unilateral cleft lip and palate.

OBJECTIVE: Cleft palate increases the risk of chronic middle ear disease and hearing loss. The goal of this report was to determine which of two palate surgeries and which timing of palate surgery were associated with better otologic and audiologic outcomes in children with unilateral cleft lip and palate at 5 to 6 years of age. DESIGN: Subjects were randomly assigned to the von Langenbeck with intravelar veloplasty or Furlow palate repair, to palate surgery at 9 to 12 months or 15 to 18 months of age, and to the Spina or Millard lip repair. SETTING: Centralized, tertiary care craniofacial treatment center. PATIENTS: A total of 673 infants with unilateral cleft lip and palate. INTERVENTIONS: Palate and lip were repaired using established techniques. Serial otoscopic and audiometric evaluations were performed. MAIN OUTCOME MEASURES: Hearing and otoscopic findings at 5 to 6 years old. RESULTS: There were 370 children available for analysis. Hearing and need for tympanostomy tube placement did not differ by palatoplasty, age at palatoplasty, cheiloplasty, or surgeon. Risk of developing cholesteatoma or perforation was higher with Millard cheiloplasty (odds ratio  =  5.1, 95% confidence interval  =  1.44 to 18.11, p  =  .012). Type and age at palatoplasty were not significantly associated with either the rate of developing these sequelae or the rate of achieving bilaterally normal hearing and ear examinations. CONCLUSIONS: Type of palatoplasty did not influence otologic and audiologic outcomes in 5- to 6-year-olds with unilateral cleft lip and palate. The potential influence of lip repair on otologic outcomes warrants further investigation.

Prospective clinical trial comparing outcome measures between Furlow and von Langenbeck Palatoplasties for UCLP.

The goal of this prospective randomized clinical trial was to compare 2 cohorts of standardized cleft patients with regard to functional speech outcome and the presence or absence of palatal fistulae. The 2 cohorts are randomized to undergo either a conventional von Langenbeck repair with intravelar velarplasty or the double-opposing Z-plasty Furlow procedure. A prospective 2 × 2 × 2 factorial clinical trial was used in which each subject was randomly assigned to 1 of 8 different groups: 1 of 2 different lip repairs (Spina vs. Millard), 1 of 2 different palatal repair (von Langenbeck vs. Furlow), and 1 of 2 different ages at time of palatal surgery (9-12 months vs. 15-18 months). All surgeries were performed by the same 4 surgeons. A cul-de-sac test of hypernasality and a mirror test of nasal air emission were selected as primary outcome measures for velopharyngeal function. Both a surgeon and speech pathologist examined patients for the presence of palatal fistulae. In this study, the Furlow double-opposing Z-palatoplasty resulted in significantly better velopharyngeal function for speech than the von Langenbeck procedure as determined by the perceptual cul-de-sac test of hypernasality. Fistula occurrence was significantly higher for the Furlow procedure than for the von Langenbeck. Fistulas were more likely to occur in patients with wider clefts and when relaxing incisions were not used.

Improving developmental screening in pediatric resident education.

OBJECTIVE: Given that pediatricians cite low competency in developmental screening, this study aims to effectively teach screening to residents. DESIGN: Using a quasi-experimental design, residents received an educational module and one-on-one teaching of 3 validated developmental screeners (Denver II, ASQ [Ages and Stages Questionnaire], and PEDS [Parents' Evaluation of Developmental Status]), with subsequent independent use with all 3 screeners with their own continuity patients. Outcome measures included changes in knowledge, skills, and preferences. RESULTS: All residents achieved significantly increased skills with all screeners. They strongly preferred the ASQ (70%), citing that this taught them normal (30.2%) and pathological (27.9%) development while negatively noting time (72.1%), scheduling issues (30.2%), and difficulties with child cooperation (20.9%). Knowledge specifics did not significantly increase. CONCLUSIONS: In-depth developmental screening education revealed marked improvement in skills and preferences. These evaluations led to full adoption of the ASQ in resident clinics. Future research must test if effective development teaching in residency leads to increased routine screenings in practice.

Quality of life information and trust in physicians among families of children with life-limiting conditions.

PURPOSE: To examine information that parents of children with life-limiting conditions want to discuss with children's physicians to assist decision-making, and whether the desire for this information is associated with parents' trust in physicians. STUDY DESIGN: A cross-sectional study using a telephone survey. PATIENTS AND METHODS: Subjects comprised a random sample of 266 parents whose children were enrolled in Florida's Medicaid Program. Parents were asked if they wanted to discuss information related to their children's treatment, including quality of life (QOL), pain relief, spiritual beliefs, clinical diagnosis/laboratory data, changes in the child's behavior due to treatment, changes in the child's appearance due to treatment, chances of recovery, and advice from the physician and family/friends. The Wake Forest Physician Trust Scale was used to measure parents' trust in physicians. We tested the relationships between parents' age, race/ethnicity, education, parent-reported children's health status, and the desired information. We also tested whether the desire for information was associated with greater trust in physicians. RESULTS: Most parents wanted information on their children's QOL (95%), followed by chance of recovery (88%), and pain relief (84%). Compared with nonHispanic whites, nonHispanic blacks and Hispanics showed a greater desire for information and a chance to discuss QOL information had greater trust in their children's physicians than other information after adjusting for covariates (P < 0.05). CONCLUSIONS: Among children with life-limiting conditions, QOL is the most frequently desired information that parents would like to receive from physicians as part of shared decision-making. Parents' desire for QOL information is associated with greater trust in their children's physicians.

The impact of insurance on satisfaction and family-centered care for CSHCN.

BACKGROUND: Children with special health care needs (CSHCN) have worse health outcomes and satisfaction compared with children with typical needs. Although individual characteristics influence satisfaction and family-centered care, additional effects of health insurance and state child health policies are unknown. OBJECTIVES: To determine if satisfaction and family-centered care varied among CSHCN, after adjusting for individual characteristics, according to insurance type and state child health policies. METHODS: We performed descriptive and multivariate analyses by using demographic, insurance, and satisfaction data from the 2006 National Survey of Children With Special Health Care Needs (N = 40723). Additional state data included Medicaid and State Children's Health Insurance Program (SCHIP) characteristics and the supply of pediatricians. We supplemented the national findings with survey data from Florida's SCHIP comprehensive care program (CMS-Duval ["Ped-I-Care"]) for CSHCN (N = 300). RESULTS: Nationally, 59.8% of parents were satisfied with their child's health services, and two thirds (65.7%) received family-centered care. Adjusting for individual predictors, those uninsured and those with public insurance were less satisfied (odds ratios [ORs]: 0.45 and 0.83, respectively) and received less family-centered care (ORs: 0.43 and 0.80, respectively) than privately insured children. Of note, satisfaction increased with state Medicaid spending. Survey data from Ped-I-Care yielded significantly higher satisfaction (91.7%) compared with national levels of satisfaction in the SCHIP (54.2%) and similar rates of family-centered care (65.6%). These results suggest that satisfaction is based more on experiences with health systems, whereas family-centered care reflects more on provider encounters. CONCLUSIONS: Insurance type affects both satisfaction and family-centered care for CSHCN, and certain state-level health care characteristics affect satisfaction. Future studies should focus on interventions in the health care system to improve satisfaction and patient encounters for family-centered care.

Longitudinal study of growth of children with unilateral cleft-lip palate from birth to two years of age.

OBJECTIVE: To study the growth of children with complete unilateral cleft lip and palate (UCLP) from birth to 2 years of age and to construct specific UCLP growth curves. DESIGN: Physical growth was a secondary outcome measure of a National Institutes of Health-sponsored longitudinal, prospective clinical trial involving the University of Florida (United States) and the University of São Paulo (Brazil). PATIENTS: Six hundred twenty-seven children with UCLP, nonsyndromic, both genders. METHODS: Length, weight, and head circumference were prospectively measured for a group of children enrolled in a clinical trial. Median growth curves for the three parameters (length, weight, head circumference) were performed and compared with the median for the National Center for Health Statistics (NCHS) curves. The median values for length, weight, and head circumference at birth and 6, 12, 18, and 24 months of age were plotted against NCHS median values and statistically compared at birth and 24 months. SETTING: Hospital de Reabilitação de Anomalias Craniofaciais, Universidade de São Paulo, Bauru, Brazil (HRAC-USP). RESULTS: At birth, children of both genders with UCLP presented with smaller body dimensions in relation to NCHS median values, but the results suggest a catch-up growth for length, weight, and head circumference for girls and for weight (to some degree) and head circumference for boys. CONCLUSIONS: Weight was the most compromised parameter for both genders, followed by length and then head circumference. There was no evidence of short stature. This study established growth curves for children with UCLP.

Managed care organization characteristics and outpatient specialty care use among children with chronic illness.

BACKGROUND: Limited information is available about managed care organization (MCO) characteristics that influence outpatient physician specialist use among children with chronic conditions. OBJECTIVE: To examine the association between MCO characteristics and outpatient physician specialist use among children with chronic conditions who were receiving care in MCOs in which primary care providers (PCPs) served as gatekeepers for referrals and who were publicly insured. DESIGN AND METHODS: A total of 2333 children who had been diagnosed with a chronic condition and had functional limitations, an increased need for or use of health care services beyond what children normally use, and/or dependence on medications or home medical equipment were included in the study. The odds of an outpatient physician specialist visit 1 year after study entry were examined as a function of child health and sociodemographic characteristics, MCO characteristics, the child's prior specialty care use, and provider availability in the MCO service delivery area. RESULTS: Children cared for in MCOs with lower percentages of PCPs paid on a fee-for-service basis (odds ratio: 0.95; 95% confidence interval: 0.92-0.98), with higher percentages of pediatricians in the PCP network (odds ratio: 1.17; 95% confidence interval: 1.07-1.29), and offering financial incentives for meeting quality of care standards (odds ratio: 1.71; 95% confidence interval: 1.28-2.29) had greater odds of outpatient physician specialist visits. Black children had odds of specialty care that were approximately one half those of white children. Children with prior physician specialist use were 52% more likely to have a physician specialist visit in the year after study entry. The children's diagnoses and condition consequences were not related significantly to the odds of a specialty visit. CONCLUSIONS: Specific MCO characteristics were associated with greater specialty care use among a group of low-income children with chronic conditions. Such information should be used to improve the structure of managed care arrangements for these vulnerable children.

Adolescents' preventive care experiences before entry into the State Children's Health Insurance Program (SCHIP).

BACKGROUND: Adolescence has traditionally been thought of as a time of good health. However, adolescents comprise an important group with unique needs among State Children's Health Insurance Program (SCHIP) enrollees. Throughout the 1990s, there was increasing evidence of unacceptably high morbidity and mortality among adolescents from injuries, suicide, sexually transmitted diseases, substance abuse, and other conditions associated with risk behaviors. The establishment of relationships with the health care system can ensure prompt treatment and help promote healthy behaviors, assuming that the adolescent feels comfortable seeking help for his or her health-related concerns. However, health care systems typically are not designed to ensure that adolescents receive the primary and preventive care that might ameliorate the negative consequences of health-damaging behaviors. OBJECTIVES: The purpose of this study was to examine the following hypotheses. 1) Adolescents with special health care needs, those engaging in risk behaviors, and those who were insured before program enrollment would be more likely than those who were healthy and those not engaging in risk behaviors to have a preventive care visit in the year preceding the interview. No differences would be observed in the odds of preventive care visits based on age, race/ethnicity, and gender. 2) No differences would be observed in the receipt of risk-behavior counseling for those with a preventive care visit based on the adolescents' sociodemographic and health characteristics. 3) Adolescents who were older would be more likely to engage in risk behaviors than younger adolescents. There would be no differences in reports of risk behaviors based on gender, race/ethnicity, and children with special health care needs status. METHODS: Adolescents 12 to 19 years old and newly enrolled in SCHIP were eligible for the study. Telephone interviews were conducted within 3 months after enrollment with parents of adolescents to obtain sociodemographic information and information about the adolescents' health by using the Children with Special Health Care Needs screener. Interviews also were conducted with the adolescents themselves to obtain information about the adolescents' risk behaviors and experiences with preventive care before SCHIP enrollment. RESULTS: Interviews were completed with 1872 parents. In addition, a total of 918 interviews were completed with adolescents. Approximately 73% of adolescents reported engaging in at least one risk behavior. Approximately 69% reported having a primary care visit during the last year with 46% of those reporting that the visit was private. Of those reporting a primary care visit, between 41% and 53% reported receiving counseling along 1 of the 5 content dimensions of anticipatory guidance. Older adolescents were more likely to engage in risk behaviors than younger adolescents. Hispanic adolescents were approximately 30% less likely than white non-Hispanic adolescents to report engaging in risk behaviors. In terms of having a preventive care visit, adolescents with a special need were twice as likely to have a visit when compared with their healthy counterparts. Hispanics and black non-Hispanics were half as likely to have a preventive care visit (odds ratios of 0.59 and 0.54, respectively) than white non-Hispanics. Those engaging in risk behaviors were almost 50% less likely to report private preventive care visits than those reporting no risk behaviors. Privacy during the preventive care visit was associated with a greater odds of receiving counseling for risk behaviors in general, sexual activity, and emotional health and relationships. Depending on the type of counseling, those with private preventive care visits were 2 to 3 times more likely to receive the counseling than those whose visits were not private. In addition, those engaging in risk behaviors were 1.45 to almost 2 times more likely to receive counseling than those not engaging in any risk behaviors. CONCLUSIONS AND IMPLICATIONS: Based on our findings, health plans health plans and providers involved in SCHIP are likely to serve adolescents who have had limited opportunities for private preventive care visits and counseling during such visits. The most underserved are likely to be black and Hispanic adolescents who may have had no preventive care at all compared with their white non-Hispanic counterparts. State agencies, health plans, and providers need to follow established guidelines for adolescent health care that emphasize the provision of counseling for risk behaviors for all adolescents, not just those engaging in risk behaviors or those with special health care needs. Moreover, providers need to seek opportunities to ensure privacy for the adolescents during their preventive care visits so that much-needed counseling can be provided. Particular attention needs to be given to adolescents from minority groups to encourage them to seek preventive care.

Managed care organizational characteristics and health care use among children with special health care needs.

OBJECTIVE: To examine the relationship between features of managed care organizations (MCOs) and health care use patterns by children. DATA SOURCES: Telephone survey data from 2,223 parents of children with special health care needs, MCO-administrator interview data, and health care claims data. STUDY DESIGN: Cross-sectional survey data from families about the number of consequences of their children's conditions and from MCO administrators about their plans' organizational features were used. Indices reflecting the MCO characteristics were developed using data reduction techniques. Hierarchical models were developed to examine the relationship between child sociodemographic and health characteristics and the MCO indices labeled: Pediatrician Focused (PF) Index, Specialist Focused (SF) Index, and Fee for Service (FFS) Index, and outpatient use rates and charges, inpatient admissions, emergency room (ER) visits, and specialty consultations. DATA COLLECTION/EXTRACTION METHODS: The telephone and MCO-administrator survey data were linked to the enrollment and claims files. PRINCIPAL FINDINGS: The child's age, gender, and condition consequences were consistent predictor variables related to health care use and charges. The PF Index was associated with decreased outpatient use rates and charges and decreased inpatient admissions. The SF Index was associated with increased ER visits and decreased specialty consultations, while the FFS Index was associated with increased outpatient use rates and charges. CONCLUSION: After controlling for sociodemographic and health characteristics, the PF, SF, and FFS indices were significantly associated with children's health care use patterns.