bluuebunny/crossref_metadata_2025_split huggingface dataset:
Ascertainment Bias in Turner Syndrome: New Insights From Girls Who Were Diagnosed Incidentally in Prenatal Life
Authors: Gunther Daniel F. , Eugster Erica , Zagar Anthony J. , Bryant Constance G. , Davenport Marsha L. , Quigley Charmian A. 
Published: November 2004

Objective. To evaluate differences in phenotype and other clinical features between patients who have Turner syndrome diagnosed incidentally (on the basis of a prenatal karyotype performed for reasons unrelated to suspicion of Turner syndrome, eg, advanced maternal age) or traditionally (on the basis of either a prenatal karyotype performed for abnormal ultrasound findings or a postnatal karyotype performed for clinical findings suggesting Turner syndrome).
Methods. Analysis was performed on baseline data from 88 girls, aged 9 months to 4 years, who were randomized into a multicenter growth hormone intervention trial. Baseline information included a detailed medical history (especially of cardiac and renal anomalies), examination for presence of phenotypic features characteristic of Turner syndrome, length or height (depending on age) expressed as a standard deviation score, and weight standard deviation score. Patients were classified to either the “incidental” (N = 16) or the “traditional” (N = 72) diagnosis group as described above.
Results. The incidental group had significantly fewer total phenotypic features of Turner syndrome than the traditional group (3.6 ± 2.9 vs 6.7 ± 2.9). When subgrouped by karyotype, the proportion of patients who manifested phenotypic features was greatest in patients with a 45,X nonmosaic karyotype, lowest in the patients with 45,X/46,XX mosaicism, and intermediate in those with “other” karyotypes. Fewer congenital cardiac defects were observed in the incidental group (31%) compared with the traditional group (64%), but there was no difference between the groups in the prevalence of renal defects. Karyotype distribution differed significantly between the traditional and incidental groups: 74% versus 19% had the nonmosaic 45,X karyotype in the traditional group and 7% versus 56% had the mosaic 45,X/46,XX karyotype.
Conclusions. Patients whose Turner syndrome was diagnosed incidentally had significantly fewer phenotypic features and cardiac defects, as well as a greater proportion of mosaic karyotypes, compared with patients whose Turner syndrome was diagnosed clinically. These results support the theory that significant ascertainment bias exists in our understanding Turner syndrome, with important implications for prenatal counseling.
Neurodevelopmental Outcome and Growth at 18 to 22 Months' Corrected Age in Extremely Low Birth Weight Infants Treated With Early Erythropoietin and Iron
Authors: Ohls Robin K. , Ehrenkranz Richard A. , Das Abhik , Dusick Anna M. , Yolton Kimberly , Romano Elaine , Delaney-Black Virginia , Papile Lu-Ann , Simon Neal P. , Steichen Jean J. , Lee Kimberly G. ,   for the National Institute of Child Health and Human Development Neonatal Research Network
Published: November 2004

Background. Clinical trials evaluating the use of erythropoietin (Epo) have demonstrated a limited reduction in transfusions; however, long-term developmental follow-up data are scarce.Objective. We compared anthropometric measurements, postdischarge events, need for transfusions, and developmental outcomes at 18 to 22 months' corrected age in extremely low birth weight (ELBW) infants treated with early Epo and supplemental iron therapy with that of placebo/control infants treated with supplemental iron alone.Methods. The National Institute of Child Health and Human Development Neonatal Research Network completed a randomized, controlled trial of early Epo and iron therapy in preterm infants ≤1250 g. A total of 172 ELBW (≤1000-g birth weight) infants were enrolled (87 Epo and 85 placebo/control). Of the 72 Epo-treated and 70 placebo/control ELBW infants surviving to discharge, follow-up data (growth, development, rehospitalization, transfusions) at 18 to 22 months' corrected age were collected on 51 of 72 Epo-treated infants (71%) and 51 of 70 placebo/controls (73%) by certified examiners masked to the treatment group. Statistical significance was determined using χ2 analysis.Results. There were no significant differences between treatment groups in weight or length or in the percentage of infants weighing &lt;10th percentile either at the time of discharge or at follow-up, and no difference was found in the mean head circumference between groups. A similar percentage of infants in each group was rehospitalized (38% Epo and 35% placebo/control) for similar reasons. There were no differences between groups with respect to the percentage of infants with Bayley-II Mental Developmental Index &lt;70 (34% Epo and 36% placebo/control), blindness (0% Epo and 2% placebo/control), deafness or hearing loss requiring amplification (2% Epo and 2% placebo/control), moderate to severe cerebral palsy (16% Epo and 18% placebo/control) or the percentage of infants with any of the above-described neurodevelopmental impairments (42% Epo and 44% placebo/control).Conclusions. Treatment of ELBW infants with early Epo and iron does not significantly influence anthropometric measurements, need for rehospitalization, transfusions after discharge, or developmental outcome at 18 to 22 months' corrected age.
Health Care Preferences and Priorities of Adolescents With Chronic Illnesses
Authors: Britto Maria T. , DeVellis Robert F. , Hornung Richard W. , DeFriese Gordon H. , Atherton Harry D. , Slap Gail B. 
Published: November 2004

Background. Efforts to make health care for adolescents with chronic illnesses more patient-centered must be grounded in an understanding and clear measures of adolescents' preferences and priorities.Objective. To develop a measure of health care preferences of adolescents with chronic illnesses and to determine demographic, developmental, and health factors associated with adolescents' preferences.Design. Mixed-method questionnaire development and survey.Setting. Subspecialty clinics of a tertiary care children's hospital.Participants. All adolescents (age: 11–19 years) with juvenile rheumatoid arthritis, sickle cell disease, inflammatory bowel disease, or cystic fibrosis of at least 2-year duration who were being treated at the participating center were eligible to participate, and 155 of 251 did so (62%). The participants had a mean age of 15.5 ± 2.4 years, 45% were male, and 75% were white.Intervention. None.Main Outcome Measures. Ratings of 65 items related to quality of care and 17 items related to physician-patient communication styles.Results. An 82-item questionnaire, devised from qualitative analysis of focus group results, contained 65 Likert scale items that adolescents considered important for health care quality and 17 forced-choice items related to adolescents' preferences for communication. Among the first 65 items, the group of questions related to physician trust and respect had the highest rating of 5.24 ± 0.62 of 6, followed by patient power and control (mean rating: 4.72 ± 0.77) and then caring and closeness in the patient-doctor relationship (mean rating: 4.19 ± 0.91). For the communication items, the adolescents, on average, preferred communication directly to them rather than to their parents and were nearly neutral regarding physicians' inquiries about personal issues.Conclusions. Participants rated aspects of interpersonal care (especially honesty, attention to pain, and items related to respect) as most important in their judgments of quality. As in most previous studies of adults, technical aspects of care were also rated highly, suggesting that adolescents understand and value both scientific and interpersonal aspects of care.
Prevalence, Stability, and Predictors of Clinically Significant Behavior Problems in Low Birth Weight Children at 3, 5, and 8 Years of Age
Authors: Gray Ronald F. , Indurkhya Alka , McCormick Marie C. 
Published: November 2004

Objectives. To examine the prevalence, stability, and predictors of clinically significant behavior problems in 869 preterm low birth weight (LBW) infants at 3, 5, and 8 years of age.Methods. A prospective cohort study was conducted. Clinically significant behavior problems were assessed using dichotomized total problem Child Behavior Checklist scores in LBW children at ages 3, 5, and 8 years. Baseline sociodemographic and obstetric data were collected. Maternal General Health Questionnaire performed at 40 weeks' gestation was dichotomized at a score of 12 to give a measure of maternal psychological distress. Prevalence and stability of behavior problems at ages 3, 5, and 8 were determined and potential predictors of behavior problems at age 3, 5, and 8 were examined using multiple logistic regression.Results. Prevalence of behavior problems remained at ∼20% at 3, 5, and 8 years. Stability of behavior problems between different ages was ∼50%. Significant behavior problems at ages 3, 5, and 8 were predicted by maternal psychological distress at 40 weeks (odds ratio [OR]: 1.59; 95% confidence interval [CI]: 1.21–2.09), maternal cigarette smoking during pregnancy (OR: 1.57; 95% CI: 1.20–2.04), Hispanic ethnicity (OR: 2.00; 95% CI: 1.24–3.24), and maternal age (OR: 0.97; 95% CI: 0.94–0.99).Conclusions. This sample had double the prevalence of behavior problems expected in the general child population. These problems showed stability over time. Cigarette smoking in pregnancy, maternal psychological distress at 40 weeks' gestation, maternal age, and Hispanic ethnicity all were significant predictors of the development of behavior problems from ages 3 to 8. These findings have implications for health policies on smoking and postnatal depression.
Thimerosal Exposure in Infants and Developmental Disorders: A Prospective Cohort Study in the United Kingdom Does Not Support a Causal Association
Authors: Heron Jon , Golding Jean ,   and the ALSPAC Study Team
Published: November 2004

Objective. There is an established link between exposure to mercury and impaired childhood cognitive development and early motor skills. Thimerosal (also known as thiomersal), a preservative used in a number of children's vaccines, contains ethylmercury (an organic compound of mercury), and there has been concern that this exposure to mercury may be of some detriment to young children. The aim of this research was to test in a large United Kingdom population–based cohort whether there is any evidence to justify such concerns.Methods. We used population data from a longitudinal study on childhood health and development. The study has been monitoring &gt;14 000 children who are from the geographic area formerly known as Avon, United Kingdom, and were delivered in 1991–1992. The age at which doses of thimerosal-containing vaccines were administered was recorded, and measures of mercury exposure by 3, 4, and 6 months of age were calculated and compared with a number of measures of childhood cognitive and behavioral development covering the period from 6 to 91 months of age.Results. Contrary to expectation, it was common for the unadjusted results to suggest a beneficial effect of thimerosal exposure. For example, exposure at 3 months was inversely associated with hyperactivity and conduct problems at 47 months; motor development at 6 months and at 30 months; difficulties with sounds at 81 months; and speech therapy, special needs, and “statementing” at 91 months. After adjustment for birth weight, gestation, gender, maternal education, parity, housing tenure, maternal smoking, breastfeeding, and ethnic origins, we found 1 result of 69 to be in the direction hypothesized—poor prosocial behavior at 47 months was associated with exposure by 3 months of age (odds ratio: 1.12; 95% confidence interval: 1.01-1.23) compared with 8 results that still supported a beneficial effect.Conclusions. We could find no convincing evidence that early exposure to thimerosal had any deleterious effect on neurologic or psychological outcome.
Integrating Adherence to Highly Active Antiretroviral Therapy Into Children's Daily Lives: A Qualitative Study
Authors: Hammami Naïma , Nöstlinger Christiana , Hoerée Tom , Lefèvre Pierre , Jonckheer Tyl , Kolsteren Patrick 
Published: November 2004

Objective. To acquire a deeper understanding of factors that influence adherence to highly active antiretroviral therapy (HAART) in a pediatric population.
Methods. We performed a qualitative study of adherence in children who receive HAART in a Belgian pediatric acquired immune deficiency syndrome referral center. Eleven primary caregivers were interviewed to assess their child's adherence and influencing factors. The interview guidelines were developed on the basis of an extensive literature review. Adherence to treatment was assessed using caregivers' self-report and laboratory results. Content analysis for common items was performed, and statements of adherent and less-adherent patients were compared.
Results. Three main factors influenced adherence. Adherent patients were found to internalize the medical information to a stronger extent than less-adherent patients. Adherent patients showed stronger motivation to stick to the medical regimen on the basis of personal cost-benefit analyses, ie, perceived benefits outweighed the costs or difficulties experienced. Adherent patients developed greater problem-solving capacities, ie, ways to deal with practical complications of medication intake. The interviews revealed a fourth, more dynamic component: knowledge, motivation, and capacities evolved in a progressive way, related to individual stages of coping with human immunodeficiency virus (HIV).
Conclusions. The data suggest that coping with HIV and the process of establishing good adherence may be interrelated. Caregivers who accept the disease may be more likely to internalize the received information and thus develop a stronger motivation to fight for the child's life. Problem-solving skills sustain this adherence, and medication becomes a priority in the adherent caregivers' daily lives. On the contrary, less-adherent caregivers may be situated at less advanced stages of the coping process. Thus, tailor-made approaches adapted to the individual HIV-related coping strategies need to be developed to improve adherence in children and caregivers.
Prognosis for Ambulation in Cerebral Palsy: A Population-Based Study
Authors: Wu Yvonne W. , Day Steven M. , Strauss David J. , Shavelle Robert M. 
Published: November 2004

Objectives. To determine independent predictors of ambulation among children with cerebral palsy and to develop a simple tool that estimates the probability that a child will walk.
Methods. In a retrospective study of all children with cerebral palsy who were not yet walking at 2 to 3 \batchmode \documentclass[fleqn,10pt,legalpaper]{article} \usepackage{amssymb} \usepackage{amsfonts} \usepackage{amsmath} \pagestyle{empty} \begin{document} \({1}/{2}\) \end{document} years of age, while receiving services from the California Department of Developmental Services during the years 1987–1999, we analyzed medical and functional data obtained annually by Department of Developmental Services physicians and social workers. Using logistic regression analyses, we determined independent predictors of a child's ability to walk well alone at least 20 feet, without assistive devices, by age 6. We then estimated the probabilities of walking at various levels of ability over time, using multistate survival analysis.
Results. Of 5366 study subjects, 2295 (43%) were evaluated at age 6; 12.8% could walk independently and 18.4% walked with support. Independent predictors of successful ambulation included early motor milestones such as sitting (odds ratio: 12.5; 95% confidence interval: 5.8–27.2) and pulling to a stand (odds ratio: 28.5; 95% confidence interval: 13.4–60.4) when compared with lack of rolling at age 2, cerebral palsy type other than spastic quadriparesis (odds ratio: 2.2; 95% confidence interval: 1.5–3.1), and preserved visual function (odds ratio: 2.4; 95% confidence interval: 1.1–5.4). Our ambulation charts depict the probability of remaining nonambulatory, transitioning to 1 of 3 possible ambulatory states, or expiring at all subsequent ages through age 14.
Conclusion. The ambulation charts provide a simple straightforward way to estimate the probability that a child with cerebral palsy who is nonambulatory at 2 to 3 \batchmode \documentclass[fleqn,10pt,legalpaper]{article} \usepackage{amssymb} \usepackage{amsfonts} \usepackage{amsmath} \pagestyle{empty} \begin{document} \({1}/{2}\) \end{document} years of age will eventually walk with or without support.
Group A Streptococci Among School-Aged Children: Clinical Characteristics and the Carrier State
Authors: Martin Judith M. , Green Michael , Barbadora Karen A. , Wald Ellen R. 
Published: November 2004

Objective. A 4-year longitudinal study of school-aged children was conducted to describe the clinical characteristics and epidemiologic features of infections with group A streptococci (GAS).Methods. Between 1998 and 2002, surveillance throat cultures were performed twice per month (October to May) for a cohort of elementary school children in Pittsburgh, Pennsylvania. In addition, throat cultures were obtained during any respiratory illness. Erythromycin and clindamycin susceptibility testing was performed for all isolates. Molecular typing was performed with field-inversion gel electrophoresis. Representative isolates from each field-inversion gel electrophoresis group were emm typed. Strict definitions were used to characterize each GAS infection. Children were classified into 4 categories each year, ie, single episode, recurrent episodes, carriers of GAS, and no infections.Results. A total of 48 to 100 children per year were studied for 4 years; 61 (49%) were male. The mean age was 9.6 years (range: 5–15 years). A total of 5658 throat cultures were performed; 878 (15.5%) were positive for GAS. Antimicrobial agents were used to treat 209 episodes of infection. Thirteen emm types were observed during the 4-year period. GAS were isolated most often from children who were carriers; isolates from single episodes were next most common. Children carried a single emm type for a mean of 10.8 weeks (range: 3–34 weeks). Carriers were likely to be classified again as carriers in subsequent years and frequently switched emm types. Sixty-two percent of the children had ≥1 year with no infections.Conclusions. GAS infections are common among school-aged children. The majority of positive throat cultures observed in this longitudinal study were obtained from children who were carriers of GAS. Carriers switched emm types but tended to become carriers repeatedly during the study. Practitioners should consider treating children known to be GAS carriers when they develop a new illness that is consistent with streptococcal pharyngitis, because they may acquire new emm types and be at risk for rheumatic heart disease.
Inequality of Access to Surgical Specialty Health Care: Why Children With Government-Funded Insurance Have Less Access Than Those With Private Insurance in Southern California
Authors: Wang Edward C. , Choe Meeryo C. , Meara John G. , Koempel Jeffrey A. 
Published: November 2004

Objective. More than 25 million children in the United States are dependent on federal and state medical insurance programs for their health care needs. In California, 3.25 million children depend on Medi-Cal for their health insurance. In Southern California alone, the figure is as high as 1.81 million. However, 9.30 million children nationally and 1.55 million in California have no health insurance. Various public policies that would increase enrollment in these programs are being discussed to address this problem. However, before their implementation, it is important to understand what impact such policies would have on the actual delivery of health care to this patient population. In California, 2 predominant health care delivery models exist for Medi-Cal: a fee-for-service (so-called regular or straight Medi-Cal) and a managed care plan. One third of the children in Medi-Cal in the state are enrolled in the fee-for-service plan with the remainder in the managed care plan, whereas in Southern California, this figure is slightly lower at 28% in the fee-for-service plan. The objective of this study was to determine the number of otolaryngologists in Southern California who would offer a new patient appointment for an evaluation for tonsillectomy for a child with commercial insurance versus government-funded (Medi-Cal) insurance through direct contact with the physician and to determine whether the surgeon would offer to perform the procedure or refer the patient to another institution and to identify the specific reason(s) for any disparity in access to health care.
Methods. A written questionnaire was sent via regular mail to 303 otolaryngologists in the Southern California area in 2003.
Results. A total of 100 fully completed questionnaires were received. Ninety-seven surgeons would offer an office appointment to a child with commercial insurance as compared with only 27 for a child with Medi-Cal. Of those 27 surgeons, 8 would then refer the child to another physician to perform the surgery, and only 19 would actually offer to perform surgery, if indicated. Reasons provided for not offering an office appointment or surgery for the child with Medi-Cal include excessive paperwork and/or administrative burdens (96%), low monetary reimbursement for the surgery (92%), and low monetary reimbursement for the office visit (87%).
Conclusions. There is a tremendous inequality of access to surgical specialty health care for children with government-funded insurance when compared with those with commercial insurance in Southern California. Physicians indicate that this disparity is related to excessive administrative burdens and low monetary reimbursement. The implications of our findings on public health care policies are discussed.
Palivizumab Use in Very Premature Infants in the Neonatal Intensive Care Unit
Authors: Wu Shou-Yien , Bonaparte Joel , Pyati Suma 
Published: November 2004

Objective. The purpose of this study was to determine the ability of young hospitalized premature (born ≤30 weeks’ gestational age) infants to achieve serum levels of palivizumab that are protective against RSV infection.
Methods. Palivizumab, 15 mg/kg per dose intramuscularly, was administered every 28 days to stable premature infants who were hospitalized in the neonatal intensive care unit starting at 1 month of postnatal life. Palivizumab concentrations were assayed in serum samples that were drawn from infants who remained in the hospital at 14 days (midpoint concentration) and at 28 days (trough concentration) after each dose was administered.
Results. The gestational age of the 24 infants who were enrolled was 27.5 ± 1.8 weeks (mean ± standard deviation), and birth weight was 928 ± 159 g. Midpoint palivizumab concentrations in the 24 infants after the first dose were 45.6 ± 13.0 μg/mL; 71% (17 of 24) of the infants maintained optimal palivizumab concentrations (≥40 μg/mL). The concentrations dropped subsequently; trough concentrations just before the second dose were 32.2 ± 10.5 μg/mL, and only 23% (5 of 22) of the infants had concentrations in the optimal range. Sixteen infants were given 2 doses and 6 were given three doses of palivizumab while in the neonatal intensive care unit. Midpoint concentrations after the second dose were significantly higher than those after the first dose. Likewise, trough concentrations before the third dose were 51.9 ± 7.8 μg/mL and higher than those before the second dose; the concentrations were &gt;40 μg/ml in all 6 infants tested.
Conclusions. Very premature infants had sustained optimal protective serum concentrations only after the second dose of palivizumab; 77% of infants tested had trough concentrations &lt;40 μg/mL before the second dose. Additional studies are needed to establish the optimal timing of the initial dose and optimal dosing interval of palivizumab in this most vulnerable population.