The trajectory of IGF-1 across age and duration of type 1 diabetes.

BACKGROUND: Individuals with type 1 diabetes may have low IGF-1, related to insulinopenia and insulin resistance. There are few longitudinal studies of IGF-1 levels to establish its pattern in type 1 diabetes with duration and age, and to examine whether IGF-1 tracks within individuals over time. We examine age and duration trends, and the relationship of IGF-1 to gender, glycaemic control, insulin level and other factors. METHODS: Participants in the Wisconsin Diabetes Registry Study, an incident cohort study of type 1 diabetes diagnosed May 1987-April 1992, were followed for up to 18 years with IGF-1 samples up to age 45 for women and age 37 for men. RESULTS: IGF-1 is lower with type 1 diabetes than in normative samples. Although, the pattern across age resembles that in normative samples with a peak in adolescence and slow decline after age 20, the adolescent peak is delayed for women with type 1 diabetes. There was low to moderate tracking of IGF-1 within an individual. Higher insulin dose was associated with higher IGF-1 as was puberty, and female gender. Adjusted for these factors, IGF-1 declined rapidly across early diabetes duration. Lower HbA1c was most strongly related to higher IGF-1 at Tanner stages 1 and 2. CONCLUSIONS: IGF-1 is low in type 1 diabetes, with a delayed adolescent peak in women and is especially influenced by glycaemic control in early and pre-adolescence. High variability within an individual is likely a challenge in investigating associations between IGF-1 and long-term outcomes, and may explain contradictory findings.

Predictors of receiving therapy among very low birth weight 2-year olds eligible for Part C early intervention in Wisconsin.

BACKGROUND: The Individuals with Disabilities Education Act (Part C) authorizes states to establish systems to provide early intervention services (e.g., therapy) for children at risk, with the incentive of federal financial support. This study examines family and neighborhood characteristics associated with currently utilizing physical, occupational, or speech therapy among very low birthweight (VLBW) 2-year-old children who meet Wisconsin eligibility requirements for early intervention services (EI) due to developmental delay. METHODS: This cross-sectional analysis used data from the Newborn Lung Project, a regional cohort study of VLBW infants hospitalized in Wisconsin's newborn intensive care units during 2003-2004. We included the 176 children who were age two at follow-up, and met Wisconsin state eligibility requirements for EI based on developmental delay. Exact logistic regression was used to describe child and neighborhood socio-demographic correlates of parent-reported receipt of therapy. RESULTS: Among VLBW children with developmental delay, currently utilizing therapy was higher among children with Medicaid (aOR = 5.3, 95% CI: 1.3, 28.3) and concomitant developmental disability (aOR = 5.2, 95% CI: 2.1, 13.3) and lower for those living in a socially more disadvantaged neighborhood (aOR=0.48, 95% CI: 0.21, 0.98, per tertile). CONCLUSIONS: Among a sample of VLBW 2-year olds with developmental delays who are EI-eligible in WI, 4 out of 5 were currently receiving therapy, per parent report. Participation in Medicaid positively influences therapy utilization. Children with developmental difficulties who live in socially disadvantaged neighborhoods are at highest risk for not receiving therapy.

Racial disparities in health-related quality of life in a cohort of very low birthweight 2- and 3-year-olds with and without cerebral palsy.

We examine racial differences in health-related quality of life (HRQoL) among 2- and 3-year-olds born very low birthweight (VLBW, <1500 g). The sample included 611 children (290 males and 321 females) from the Newborn Lung Project, a cohort of VLBW infants hospitalized in Wisconsin's newborn intensive care units during 2003 to 2004. Of the 611 children, 14% (86/611) were black, non-Hispanic and 86% (525/611) were white, non-Hispanic and 4% (23/611) had cerebral palsy. HRQoL was measured using the Pediatric Quality of Life Inventory. Black children scored nearly four points lower (mean difference -3.6, 95% confidence interval [CI]: -6.9 to -0.3) on HRQoL than their white peers. Cerebral palsy is associated with lower HRQoL (mean difference -24.4, 95% CI: -29.3 to -19.5), especially among black children, but does not explain racial differences in HRQoL. Living in a neighborhood with lowest levels of female education is associated with lower HRQoL (mean difference -5.6, 95% CI: -9.2 to -2.1), but does not explain the racial difference in HRQoL.

Aleukemic leukemia cutis in a child preceding T-cell acute lymphoblastic leukemia.

An 8-year-old boy presented with a widespread cutaneous eruption featuring macules, papules, nodules, and ulcers. The histologic infiltrate showed T lymphoblasts, but there was no sign of systemic involvement, so aleukemic leukemia cutis was diagnosed. Two months later, he developed leukemia in peripheral blood and bone marrow that was characterized as T-cell acute lymphoblastic leukemia.

A comparison of Wisconsin neonatal intensive care units with national data on outcomes and practices.

CONTEXT: Improvements in neonatal care over the past 3 decades have increased survival of infants at lower birthweights and gestational ages. However, outcomes and practices vary considerably between hospitals. OBJECTIVE: To describe maternal and infant characteristics, neonatal intensive care units (NICU) practices, morbidity, and mortality in Wisconsin NICUs, and to compare outcomes in Wisconsin to the National Institute of Child Health and Human Development network of large academic medical center NICUs. METHODS: The Newborn Lung Project Statewide Cohort is a prospective observational study of all very low birthweight (< or =1500 grams) infants admitted during 2003 and 2004 to the 16 level III NICUs in Wisconsin. Anonymous data were collected for all admitted infants (N=1463). Major neonatal morbidities, including bronchopulmonary dysplasia (BPD), intraventricular hemorrhage (IVH), necrotizing enterocolitis (NEC), and retinopathy of prematurity (ROP) were evaluated. RESULTS: The overall incidence of BPD was 24% (8%-56% between NICUs); IVH incidence was 23% (9%-41%); the incidence of NEC was 7% (0%-21%); and the incidence of grade III or higher ROP was 10% (0%-35%). CONCLUSION: The incidence rates of major neonatal morbidities in Wisconsin were similar to those of a national network of academic NICUs.

Pulmonary testing using peak flow meters of very low birth weight children born in the perisurfactant era and school controls at age 10 years.

We determined lung function at age 10 years in very low birthweight (VLBW,

School achievement in a regional cohort of children born very low birthweight.

Children born very low birthweight (VLBW, <=1500 g) have historically had lower average school achievement than their normal birthweight peers. However, perinatal care and survival have changed dramatically since prior cohorts accrued, prompting reassessment. Surfactant therapy became generally available 8/1/1990, and the use of ante- and postnatal steroids increased substantially around this time. Standardized test scores and teacher ratings in math, reading, science, and social studies were obtained at age 10 for a cohort of children admitted to six regional NICUs in Wisconsin and Iowa, 8/1/1988 - 6/30/1991. We compared achievement between the VLBW cohort and controls from the same school districts. Among VLBW children, we determined neonatal and early childhood factors associated with achievement on standardized tests (ordinal logistic regression) and teacher ratings (linear regression) and evaluated whether achievement differed by birth year. Compared to population controls, VLBW children's greatest deficits occurred in mathematics. Scores on the standardized math exam and teacher ratings of overall achievement were positively associated with birthweight, social function measured at age five, and socioeconomic status. VLBW children born in the post-surfactant era (after 8/1/1990) had lower achievement on the standardized math exam than children born during the pre-surfactant era of the prior two years. Lower achievement in the post-surfactant era may be due to greater survival among less healthy neonates or increased exposure to postnatal steroids. VLBW children more likely to struggle academically could be identified by early childhood measures, allowing for targeted interventions to mitigate their difficulties.

Vibrio cholerae (non-O1, non-O139) sepsis in a child with Fanconi anemia.

A 9-year-old female child who was a known case of Fanconi anemia was admitted to hospital because of fever and gastrointestinal symptoms. Blood culture at the time of admission yielded growth of Gram-negative curved rod that was identified as Vibrio cholerae (non-O1, non-O139), whereas repeated fecal cultures were negative for enteropathogens. To our knowledge, this is the first case of V. cholerae (non-O1, non-O139) septicemia associated with Fanconi anemia.

Racial disparities in health-related quality of life in a cohort of very-low-birth-weight 2- and 3-year-olds with and without asthma.

BACKGROUND: Children born very low birth weight (VLBW) are at risk for low health-related quality of life (HRQoL), compared with normal-birth-weight peers, and racial disparities may compound the difference. Asthma is the most pervasive health problem among VLBW children and is also more common among black than white children, partly due to unfavourable environmental exposures. This study explores racial disparities in HRQoL among VLBW children and examines whether potential disparities can be explained by asthma and neighbourhood disadvantage. METHODS: The study population was the Newborn Lung Project, a cohort of infants (n=660) born VLBW in 2003-2004 in Wisconsin, USA, who were followed up at age 2-3. Multilevel linear regression models were used to examine the contributions of asthma, neighbourhood disadvantage, and other child and family socio-demographic covariates, to racial disparities in HRQoL at age 2-3. A child's HRQoL was measured using the Paediatric Quality of Life Inventory 4.0. RESULTS: VLBW, black, non-Hispanic children, on average, score nearly 4 points lower (p<0.01) on HRQoL than do white, non-Hispanic children. Including asthma reduces the difference between black and white children from -3.6 (p<0.01) to 0.08 (p>0.05). The authors found no evidence that the relationship between asthma and HRQoL differs by race. The interaction between neighbourhood disadvantage and asthma is statistically significant, with further examination suggesting that racial disparities are particularly pronounced in the most advantaged neighbourhoods. CONCLUSION: The authors found that the black disadvantage in HRQoL among 2-3-year-old VLBW children likely stems from a high prevalence of asthma. Neighbourhood attributes did not further explain the disparity, as the racial difference was particularly pronounced in advantaged neighbourhoods.

Relationship between neighborhood disadvantage and social function of Wisconsin 2- and 3-year-olds born at very low birth weight.

OBJECTIVE: To examine whether (1) neighborhood disadvantage is associated with social function in 2- and 3-year-olds born at very low birth weight (<1500 g) and (2) the association between social function and child's health-related quality of life (HRQoL) is moderated by neighborhood disadvantage. DESIGN: Cross-sectional study using the Newborn Lung Project, a cohort of infants born at very low birth weight in 2003 and 2004 in Wisconsin. SETTING: Wisconsin. PARTICIPANTS: This study includes the subgroup of 626 non-Hispanic black or white infants who were followed up at ages 24 to 43 months with parent-reported health and developmental information. MAIN EXPOSURE: An index of neighborhood disadvantage was derived by principal component analysis of 5 census tract variables (percentage of families in poverty, percentage of households with income higher than the state median, percentage of women with bachelor's degree or more, percentage of single mothers, and percentage of mothers of young children unemployed). Children were then classified (based on index tertiles) as living in either disadvantaged, middle advantage, or advantaged neighborhoods. Children's HRQoL was measured using the Pediatric Quality of Life Inventory. MAIN OUTCOME MEASURE: Social function was measured using the Pediatric Evaluation of Disability Inventory. RESULTS: Adjusting for child medical and family socioeconomic attributes, social function was lower (mean difference, -4.60; 95% confidence interval, -8.4 to -0.8) for children living in disadvantaged vs advantaged neighborhoods. We also found that the ill effects of lower HRQoL are particularly bad for children living in a disadvantaged neighborhood. CONCLUSION: Children born at very low birth weight have disparities in social function at ages 2 and 3 years that are associated with both HRQoL and neighborhood characteristics.

Daycare attendance and risk for respiratory morbidity among young very low birth weight children.

Daycare attendance and very low birth weight (VLBW, < or =1,500 g) are associated with respiratory morbidity during childhood. The objective of this study was to evaluate whether daycare attendance is associated with even higher risk for respiratory problems among VLBW children. We hypothesized that VLBW children attending daycare, in a private home or daycare center, are at higher risk for respiratory problems than VLBW children not attending daycare. We also investigated whether the effect of daycare is independent or synergistic with respiratory risk resulting from being VLBW, as indicated by having bronchopulmonary dysplasia (BPD) as a neonate. We conducted a prospective study of VLBW children followed from birth to age 2-3 (N = 715). Logistic regression was used to evaluate the relationship between daycare attendance and respiratory problems, adjusting for known neonatal risk factors for poor respiratory outcomes. Attending daycare in either a private home or in a daycare center was significantly associated with higher risk of lower respiratory infections than never attending. Attending a daycare center was also associated with higher risk for wheezy chest, cough without a cold, and respiratory medication use. While having BPD was associated with increased risk for respiratory problems, daycare attendance and BPD were not found to be synergistic risk factors for respiratory problems among VLBW children, but acted independently to increase risk. This implies that the increase in risk for respiratory problems associated with daycare attendance may be similar among VLBW children and those of normal birth weight.

Intracranial calcification in central diabetes insipidus.

Intracranial calcification is a known but extremely rare complication of diabetes insipidus. To date, only 16 patients have been reported and all had the peripheral (nephrogenic) type of diabetes insipidus. We report a child with intracranial calcification complicating central diabetes insipidus. We also report a child with nephrogenic diabetes insipidus, and compare the patterns of intracranial calcification.

PedsQL relates to function and behavior in very low and normal birth weight 2- and 3-year-olds from a regional cohort.

STUDY OBJECTIVE: To compare PedsQL scores in young children who were very low (< or =1,500 g) or normal birth weight (>2,500 g) and to examine the relationship of the PedsQL score to behavioral and functional scores. METHODS: The PedsQL, Achenbach Child Behavior Checklist and the PEDI functional scales were telephone administered to parents of a regional cohort of 672 very low birth weight and 455 normal birth weight children, 2- and 3-years old. PedsQL scales were regressed on behavior, function and health conditions. RESULTS: Mean (SD) overall PedsQL score was 91 (8.4) for normal birth weight and 87 (12) for very low birth weight children, and changed little when standardized to the race/ethnicity and maternal education of corresponding Wisconsin births. Mobility function and the CBCL explained 58% of the variance in PedsQL, but the relationship was curvilinear. CONCLUSION: The PedsQL is sensitive to health problems of very low birth weight in young children. The PedsQL is quite strongly related to mobility and behavior problems, but scales these differently than do standard instruments. Parents either do not think of subtle issues with child function and behavior without specific prompting or do not perceive them as problems affecting quality of life.

Permissive hypercapnia and risk for brain injury and developmental impairment.

OBJECTIVE: Permissive hypercapnia is a respiratory-care strategy that is used to reduce the risk for lung injury. The goal of this study was to evaluate whether permissive hypercapnia is associated with higher risk for intraventricular hemorrhage and early childhood behavioral and functional problems than normocapnia among very low birth weight infants. METHODS: Very low birth weight infants from a statewide cohort were eligible for this study when they were born at <32 weeks' gestational age and survived at least 24 hours. Infants were classified as receiving a permissive hypercapnia, normocapnia, or unclassifiable respiratory strategy during the first 24 hours after birth according to an algorithm based on Pco(2) values and respiratory-treatment decisions that were abstracted from medical charts. Intraventricular hemorrhage diagnosis was also abstracted from the medical chart. Behavioral and functional outcomes were assessed by parent interview at 2 to 3 years. Logistic regression was used to evaluate the relationship between intraventricular hemorrhage and respiratory strategy; ordinary linear regression was used to evaluate differences in behavior and function scores between children by respiratory strategy. RESULTS: Infants who received a permissive hypercapnia strategy were not more likely to have intraventricular hemorrhage than those with normocapnia. There were no differences in any of the behavioral or functional scores among children according to respiratory strategy. There was a significant interaction between care strategy and 1-minute Apgar score, indicating that infants with lower Apgar scores may be at higher risk for intraventricular hemorrhage with permissive hypercapnia. CONCLUSIONS: This study suggests that permissive hypercapnia does not increase risk for brain injury and impairment among very low birth weight children. The interaction between respiratory strategy and Apgar score is a potential worrisome exception to this conclusion. Future research should further evaluate the effect of elevated Pco(2) levels among those who are sickest at birth.

Primary immunodeficiency disorders: survey of pediatricians in Kuwait.

INTRODUCTION: Early diagnosis of primary immunodeficiency disorders (PID) is critical so life saving interventions can be implemented to avoid significant morbidity and mortality. Unfortunately, they are frequently misdiagnosed, which results into significant delay in diagnosis. This study aimed to determine the knowledge and practice of pediatricians in Kuwait about PID. MATERIALS AND METHODS: A 66-item self-administered questionnaire was designed and distributed to the pediatricians working at all six governmental hospitals to measure their knowledge and practice about PID. A total of 244 pediatricians (78.4%; 143 males and 101 females) participated in the study. The mean age of participants was 40 years, and the mean number of years working in pediatrics was 13 years. The mean overall score was 59.6%, whereas the mean score in clinical presentation section was 63%, in associated diseases and syndromes section 58%, and in laboratory investigations section 51%. Only 26% of the participants answered correctly at least 2/3 of the questions (67% of the questions). CONCLUSION: This survey demonstrates that there is universal deficiency in both the knowledge and practice of pediatricians in the field of PID. Implementation of strategies to improve the awareness of pediatricians about PID is critical so early therapeutic interventions can be done to improve the health and prevent morbidity and mortality.

Acute pyelonephritis and renal scarring in Kuwaiti children: a follow-up study using 99mTc DMSA renal scintigraphy.

The aim of this study was to determine the prevalence of renal scarring in a group of Kuwaiti Arab children with their first documented acute pyelonephritis (APN). Eighty-two Kuwaiti Arab children (10 males and 72 females) who had abnormal (99m)Tc DMSA renal scan findings of acute pyelonephritis were prospectively studied with the same imaging modality 6 months after treatment to identify those who developed renal scarring. A micturition cystourethrogram (MCUG) was performed for all of the children 1 month after diagnosis. Children were divided into 3 age groups (<2 years, 2-5 years and above 5 years). The follow-up DMSA renal scans 6 months after diagnosis revealed normalization of renal changes in 56% (46 patients), much improvement with residual renal abnormality in 6% (5 patients), and persistent parenchymal defects in 38% (31 patients). Vesicoureteric reflux (VUR) was found in 32% of children (26/82) and the majority were between grade I and III. Thirteen of those with VUR (50%) developed renal scars on follow-up. Fifty-three percent of the scarred kidneys (19/36) were drained by non-refluxing ureters. In this study, children older than 2 years had less VUR yet were more susceptible to APN and to the development of renal scars. Girls were more prone to developing APN and renal scarring than boys. This work shows that APN is a serious cause for renal scarring in our patients, particularly if associated with other risk factors such as recurrent infections and the female sex.

Vitamin D status of mothers and their neonates in Kuwait.

BACKGROUND: There has been a resurgence of nutritional rickets in children in many developing countries and some of the developed countries. Children between 6 and 18 months old are commonly affected. In order to find out the association between vitamin D and rickets we studied the vitamin D status of the neonates and their mothers in Kuwait. METHODS: Two hundred and fourteen full-term pregnant mothers and their neonates were selected from two hospitals in Kuwait. All mothers had normal vaginal delivery. On the day of delivery 2.5 mL of maternal blood and 2.5 mL of cord blood samples were withdrawn. Serum 25-hydroxyvitamin D (25OHD) was determined in duplicate by radioimmunoassay using an Incstar kit. Quality control analyses were done using several between and within run experiments. RESULTS: A total of 128 mother-neonate pairs were selected from the Al-Adan hospital and 86 from the Maternity Hospital. The mean age and parity of the mothers were similar in both hospitals. The mean (+/- SD) 25OHD levels of the mothers and the neonates in the Adan hospital were 13.3 (6.5) ng/mL and 8.2 (6.5) ng/mL, respectively. The corresponding values in the Maternity Hospital were 17.6 (12.4) and 8.1 (7.3) ng/mL for the mothers and the neonate, respectively. Serum 25OHD of the mothers and their newborn infants were highly correlated (r = 0.790, P < 0.001). CONCLUSIONS: Results demonstrate that 40% of the mothers and 60% of the neonates are vitamin D deficient on the day of delivery. The vitamin D of the mothers and neonates are highly correlated (r = 0.790, P < 0.001).

Vesicoureteric reflux in Kuwaiti children with first febrile urinary tract infection.

The prevalence of vesicoureteric reflux (VUR) in children with urinary tract infection (UTI) varies among different racial groups. The purpose of this study was to determine the frequency of VUR and associated renal changes in a group of Arab Kuwaiti children with their first documented febrile UTI and to compare our findings with those reported from other racial groups. One hundred and seventy-four children (38 males and 136 females) fulfilled the study criteria and were divided into three age groups (<1 year, 1-5 years, and >5 years). Patients in each group had both micturating cystourethrography (MCUG) and 99m-Tc-dimercaptosuccinic acid (DMSA) renal scan after diagnosis. VUR was detected in 39 children (22%). Two-thirds of cases had mild reflux (grade I and II). Females ( n=32) had more reflux than males ( n=7) (24% vs. 18%). Sixty-three patients (36%) had abnormal (DMSA) renal scans (acute pyelonephritis [AP] or renal scars). Of these, 79% were children below 5 years. Abnormal DMSA scans were found in 4 of 38 males (11%) versus 59 of 136 females (43%). Abnormal scans in children with VUR were seen in 1 of 7 males (14%) versus 19 of 32 females (59%). In total, the combination of abnormal scan with VUR occurred in 1 of 38 males (3%) and in 19 of 136 females (14%), whereas abnormal scan without demonstrable VUR was seen in 3 of 38 males (8%) versus 40 of 136 females (29%). Our data showed that the frequency of VUR in Arab Kuwaiti children with febrile UTI is midway between Caucasian and other racial groups. In this study, males had a lower-risk profile than females, the latter having a higher rate of reflux as well as a higher rate of abnormal DMSA scans, irrespective of demonstrable VUR.