Fetal alcohol syndrome surveillance: age of syndrome manifestation in case ascertainment.

BACKGROUND: Fetal alcohol syndrome (FAS) is a leading cause of developmental disability (Abel & Sokol, ). Active public health surveillance through medical record abstraction has been used to estimate FAS prevalence rates, typically based on birth cohorts. There is an extended time for FAS characteristics to become apparent in infants and young children, and there are often delays in syndrome recognition and documentation. This methodological study analyzes the age at case ascertainment in a large surveillance program. METHODS: The Fetal Alcohol Syndrome Surveillance (FASSLink) Project, funded by the Centers for Disease Control and Prevention, sought to estimate FAS prevalence rates in eight U.S. states. FASSLink used linked abstractions from multiple health care records of suspected cases of FAS. The present study analyzed data from this effort to determine the child's age in months at confirming abstraction. RESULTS: The average age at abstraction for confirmed/probable FAS cases (n = 422) was 48.3 (±19.5) months with a range of 0 to 94 months. Age of ascertainment varied by state and decreased with each birth year; the number of cases ascertained also decreased in a steep stepwise gradient over the 6 birth years in the study. CONCLUSION: FAS surveillance efforts should screen records of children who are much older than is typical in birth defects surveillance. To best establish rates of FAS using medical records abstraction, surveillance efforts should focus on 1-year birth cohorts followed for a fixed number of years or, if using multi-year cohorts, should implement staggered end dates allowing all births to be followed for up to 8 years of age.

Autism Spectrum Disorder (ASD) Prevalence in Somali and Non-Somali Children.

The current study presents results from an autism spectrum disorder (ASD) public health surveillance project conducted in Minneapolis. The study was designed to compare ASD prevalence in Somali children (ages 7-9) to that of non-Somali children. The study adapted methodology used by the Centers for Disease Control and Prevention's Autism and Developmental Disabilities Monitoring Network. Results indicated that Somali (1 in 32) and White (1 in 36) children were about equally likely to be identified with ASD, but more likely to be identified with ASD than Black and Hispanic children. Somali children with ASD were significantly more likely to have an intellectual disability than children with ASD in all other racial and ethnic groups.

Long-term neurologic and neurosensory sequelae in adult survivors of a childhood brain tumor: childhood cancer survivor study.

PURPOSE: To describe the neurologic and neurosensory deficits in children with brain tumors (BTs), compare incidence of these deficits with that of a sibling control group, and evaluate the factors associated with the development of these deficits. PATIENTS AND METHODS: Detailed questionnaires were completed on 1,607 patients diagnosed between 1970 and 1986 with a primary CNS tumor. Neurosensory and neurologic dysfunctions were assessed and results compared with those of a sibling control group. Medical records on all patients were abstracted, including radiotherapy dose and volume. RESULTS: Seventeen percent of patients developed neurosensory impairment. Relative to the sibling comparison group, patients surviving BTs were at elevated risk for hearing impairments (relative risk [RR], 17.3; P = <.0001), legal blindness in one or both eyes (RR, 14.8; P = <.0001), cataracts (RR, 11.9; P = <.0001), and double vision (RR, 8.8; P = <.0001). Radiation exposure greater than 50 Gy to the posterior fossa was associated with a higher likelihood of developing any hearing impairment. Coordination and motor control problems were reported in 49% and 26%, respectively, of survivors. Children receiving at least 50 Gy to the frontal brain regions had a moderately elevated risk for motor problems (RR, 2.0; P <.05). Seizure disorders were reported in 25% of patients, including 6.5% who had a late first occurrence. Radiation dose of 30 Gy or more to any cortical segment of the brain was associated with a two-fold elevated risk for a late seizure disorder. CONCLUSION: Children surviving BTs are at significant risk for both early and late neurologic or neurosensory sequelae. These sequelae need to be prospectively monitored.

Final height and body mass index among adult survivors of childhood brain cancer: childhood cancer survivor study.

The objectives of this study were 1) to compare final height and body mass index (BMI) between adult survivors of childhood brain cancer and age- and sex-matched population norms, 2) to quantify the effects of treatment- and cancer-related factors on the risk of final height below the 10th percentile (adult short stature) or having a BMI of 30 kg/m(2) or more (obesity). Treatment records were abstracted and surveys completed by 921 adults aged 20-45 yr who were treated for brain cancer as children and were participants in the multicenter Childhood Cancer Survivor Study. Nearly 40% of childhood brain cancer survivors were below the 10th percentile for height. The strongest risk factors for adult short stature were young age at diagnosis and radiation treatment involving the hypothalamic-pituitary axis (HPA). The multivariate odds ratio for adult short stature among those 4 yr of age or younger at diagnosis, relative to ages 10-20 yr, was 5.67 (95% confidence interval, 3.6-8.9). HPA radiation exposure increased the risk of adult short stature in a dose-response fashion (trend test, P < 0.0001). Adjuvant chemotherapy was not an independent risk factor for adult short stature. BMI distribution in survivors did not differ appreciably from that of population norms; however, in females, young age at diagnosis and HPA radiation dose (trend test, P < 0.001) were associated with risk of obesity. Except for patients treated with surgery only, survivors of childhood brain cancer are at very high risk for adult short stature, and this risk increases with radiation dose involving the HPA. We did not find a corresponding elevated risk for obesity.

What factors are associated with state performance on provision of transition services to CSHCN?

OBJECTIVE: To examine whether individual, condition-related, and system-related characteristics are associated with state performance (high, medium, low) on the provision of transition services to children with special health care needs (CSHCN). METHODS: We conducted descriptive, bivariate, and multivariable analyses of 16876 children aged 12 to 17 years by using data from the 2005-2006 National Survey of Children With Special Health Care Needs. Polytomous logistic regression was used to compare the characteristics of CSHCN residing within high-, medium-, and low-performance states, with low-performance states serving as the reference group. RESULTS: Compared with non-Hispanic white CSHCN, Hispanic (adjusted odds ratio [aOR]: 0.25 [95% confidence interval (CI): 0.17-0.37]) and non-Hispanic black (aOR: 0.44 [95% CI: 0.30-0.62]) CSHCN were less likely to reside in a high-performance than in a low-performance state. Compared with CSHCN who had a medical home or adequate insurance coverage, CSHCN who did not have a medical home or adequate insurance coverage were less likely to reside in a high-performance than in a low-performance state (aOR: 0.73 [95% CI: 0.57-0.95]; aOR: 0.73 [95% CI: 0.58-0.93], respectively). CONCLUSIONS: Key factors found to be important in a state's performance on provision of transition services to CSHCN were race/ethnicity and having a medical home and adequate insurance coverage. Efforts to support the Maternal and Child Health Bureau's integration of system-level factors in quality-improvement activities, particularly establishing a medical home and attaining and maintaining adequate insurance, are likely to help states improve their performance on provision of transition services.

Endocrine and cardiovascular late effects among adult survivors of childhood brain tumors: Childhood Cancer Survivor Study.

BACKGROUND: Survivors of childhood brain tumors (CBTs) are at high risk for a variety of late adverse effects. Most research on long-term effects of CBTs has been comprised of single-institution case series without comparison groups. Research on CBT late effects often is focused on neurologic and sensory outcomes, with less emphasis on other potential targets such as the endocrine and circulatory systems. The current study was conducted to contrast the incidence of endocrine and cardiovascular conditions among CBT survivors as a function of treatment and to determine the risk of occurrence of these conditions relative to a sibling comparison group. METHODS: As part of the Childhood Cancer Survivor Study (CCSS), treatment data were collected from medical records and self-reported late effects were ascertained from a survey questionnaire of 1,607 CBT patients who survived their disease for 5 or more years. For comparison purposes, questionnaire data were also collected from 3418 randomly selected siblings of participants in CCSS. RESULTS: One or more endocrine conditions were reported by 43% of CBT survivors. Compared with siblings, CBT survivors had a significantly increased risk of late-onset (>/= 5 years postdiagnosis) hypothyroidism (relative risk [RR] = 14.3; 95% confidence interval [95% CI] 9.7-21.0), growth hormone deficiency (RR = 277.8; 95% CI 111.1-694.9), the need for medications to induce puberty (RR = 86.1; 95% CI 31.1-238.2), and osteoporosis (RR = 24.7; 95% CI 9.9-61.4). One or more cardiovascular conditions were reported by 18% of CBT survivors, with an elevated late-onset risk for stroke (RR = 42.8; 95% CI 16.7-109.8), blood clots (RR = 5.7; 95% CI 3.2-10.0), and angina-like symptoms (RR = 2.0; 95% CI 1.5-2.7). Very few late effects were evident among those treated with surgery only, but risks were consistently elevated for those treated with radiation and surgery, and higher still for those who also received adjuvant chemotherapy. CONCLUSIONS: Childhood brain tumor survivors are at a significantly increased risk for several adverse endocrine and cardiovascular late effects, particularly if they were treated with radiation and chemotherapy. Lifetime medical surveillance and follow-up for potential toxicities are necessary because treatment-related complications may occur many years after therapy.