Computerized adaptive testing to screen children for emotional and behavioral problems by preventive child healthcare

BACKGROUND: Questionnaires to detect emotional and behavioral problems (EBP) in Preventive Child Healthcare (PCH) should be short which potentially affects validity and reliability. Simulation studies have shown that Computerized Adaptive Testing (CAT) could overcome these weaknesses. We studied the applicability (using the measures participation rate, satisfaction, and efficiency) and the validity of CAT in routine PCH practice. METHODS: We analyzed data on 461 children aged 10-11 years (response 41%), who were assessed during routine well-child examinations by PCH professionals. Before the visit, parents completed the CAT and the Child Behavior Checklist (CBCL). Satisfaction was measured by parent- and PCH professional-report. Efficiency of the CAT procedure was measured as number of items needed to assess whether a child has serious problems or not. Its validity was assessed using the CBCL as the criterion. RESULTS: Parents and PCH professionals rated the CAT on average as good. The procedure required at average 16 items to assess whether a child has serious problems or not. Agreement of scores on the CAT scales with corresponding CBCL scales was high (range of Spearman correlations 0.59-0.72). Area Under Curves (AUC) were high (range: 0.95-0.97) for the Psycat total, externalizing, and hyperactivity scales using corresponding CBCL scale scores as criterion. For the Psycat internalizing scale the AUC was somewhat lower but still high (0.86). CONCLUSIONS: CAT is a valid procedure for the identification of emotional and behavioral problems in children aged 10-11 years. It may support the efficient and accurate identification of children with overall, and potentially also specific, emotional and behavioral problems in routine PCH

Esophageal atresia: Historical evolution of management and results in 371 patients

Background. It has been more than 50 years since the first successful surgical reconstruction of esophageal atresia was performed in The Netherlands. We reviewed the historical changes in management and treatment results of patients born with esophageal atresia. Methods. We developed and analyzed a database of 371 consecutive patients treated for esophageal atresia in our center between 1947 and 2000. Results. The mean birthweight decreased from 2,723 g (1947 to 1968) to 2,494 g (1994 to 2000), the mean gestational age decreased from 39 weeks (1947 to 1968) to 37 weeks (1994 to 2000). The number of patients with associated congenital malformations increased from 34% (1947 to 1968) to 66% (1994 to 2000). Most patients underwent primary repair of their atresia. Clinically significant tracheomalacia was present in 34 of 269 patients (13%). Gastroesophageal reflux was present in 90 of 277 patients (33%). Mortality decreased from 61% (1947 to 1968) to 11% (1994 to 2000). Conclusions. The patients who are treated nowadays for esophageal atresia in a pediatric surgical center are born earlier, weigh less, and have more associated anomalies than those treated 50 years ago. Still, the mortality rate is much lower thanks to earlier diagnosis, better supportive care and improved surgical techniques. Therefore, further significant reduction will be difficult to achieve. (C) 2002 by The Society of Thoracic Surgeon

Young adult patients with a history of pediatric disease: impact on course of life and transition into adulthood

PURPOSE: To assess the course of life of young adults who grew up with a chronic or life-threatening disease, and to compare their course of life with that of peers from the general population. Optimal transition from pediatric to adult health care requires knowledge of the psychosocial history of patients grown up with a pediatric disease. METHODS: A total of 508 young adults from the general Dutch population and 650 patients, aged 18-30 years, participated: 348 survivors of childhood cancer, 93 patients with anorectal malformations, 72 patients with Hirschsprung's disease, 61 patients with oesophageal atresia, 76 patients with end-stage renal disease. They completed the Course of Life Questionnaire, which retrospectively assesses the achievement of developmental milestones (autonomy, psychosexual and social development), and risk behavior (antisocial behavior, substance use and gambling). RESULTS: The young adults grown up with a chronic or life-threatening disease proved to have achieved significantly fewer milestones, or at older age than their peers, in all course-of-life domains. The course of life of young adults grown up with esophageal atresia was not delayed compared with that of their peers, whereas that of survivors of childhood cancer and patients with end-stage renal disease was delayed most. CONCLUSIONS: Health care providers should help to minimize the harm for children who grow up with a chronic or life-threatening disease by encouraging parents to stimulate social contacts and autonomy. Attention should especially be directed at children and adolescents growing up with childhood cancer or with end-stage renal diseas

Quality of life in adult survivors of correction of esophageal atresia

Hypothesis: Long-term quality of life (QOL) in adults after correction of esophageal atresia (EA) is comparable with that of healthy adults. Design: Outcome study with a consecutive sample and follow-up after 16 years or longer. Setting: Pediatric surgical center (academic center) in a tertiary hospital. Patients and Methods: A questionnaire was sent to 119 consecutive surviving adults who were treated for EA between 1947 and 1986. The questionnaire was to assess sociodemographic characteristics and generic and disease-specific QOL and contained 3 open-ended questions about the consequences of the disease in daily life. Clinical characteristics were collected from patient case notes. Results: Ninety-seven (82%) of the 119 questionnaires were completed and returned. When comparing the generic QOL of patients who had EA with that of healthy subjects, we found no differences in overall physical and mental health. The presence of concomitant congenital anomalies did not influence generic QOL. A third of the patients reported that EA had had negative effects on their daily lives. Gastrointestinal symptoms such as dysphagia were most often mentioned (23%). Conclusion: In general, after EA correction, patients perceive their generic and disease-specific QOL to be good. The presence of concomitant congenital anomalies did not influence generic QOL. However, a third of patients reported that the disease had had negative consequence

Gastroesophageal reflux - Prevalence in adults older than 28 years after correction of esophageal atresia

Objective: To study the incidence of gastroesophageal reflux (GER) related complications after correction of esophageal atresia (EA). Summary Background Data: The association of EA and GER in children is well known. However, little is known about the prevalence of GER and its potential complications in adults who have undergone correction of EA as a child. Methods: Prospective analysis of the prevalence of GER and its complications over 28 years after correction of EA by means of a questionnaire, esophagogastroscopy, and histologic evaluation of esophageal biopsies. Results: The questionnaire was returned by 38 (95%) of 40 patients. A quarter of the patients had no complaints. Swallowing solid food was a problem for 13 patients (34%), and mashed foods for 2 (5%). Heartburn was experienced by 7 patients (18%), retrosternal pain by 8 (21%). However, none of the patients were using antireflux. medication. Twenty-three patients (61%) agreed to undergo esophagogastroscopy, which showed macroscopic Barrett esophagus in 1 patient, which was confirmed by histology. One patient developed complaints of dysphagia at the end of the study. A squamous cell esophageal carcinoma was diagnosed and treated by transthoracic subtotal esophagectomy. Conclusions: This study shows a high incidence of GER-related complications after correction of EA, but it is still very disputable if all EA patients should be screened at an adult ag

Esophagitis and Barrett esophagus after correction of esophageal atresia

Background: Gastroesopliageal reflux is a frequent problem after esophageal atresia (EA) repair. Our aim was to determine the prevalence of esophagitis and Barrett esophagus more than 10 years after repair of EA. Methods: Ninety-two patients treated between 1973 and 1985 were included in this prospective study. A questionnaire was completed by 86 patients; esophagogastroscopy was performed in 49 patients. Results: Only 36 patients had no complaints at all. Thirty-one patients complained of difficulties swallowing solid food; 23 complained of heartburn. Esophagogastroscopy revealed grade 3 esophagitis in 2 patients and a macroscopic image of Barrett esophagus in 2. Histology showed esophagitis in 30 patients, gastric inetaplasia in 3, and no intestinal metaplasia (Barrett esophagus). Conclusions: For epidemiologic reasons, that is, the short interval of follow-up (10 years) and the low compliance of the study group, larger numbers are needed to decide if routine long-term endoscopic screening after repair of EA is necessary. For now, it cannot yet be recommended. The prevalence of symptoms of gastroesophageal reflux disease in this study group is higher than that in the general population, but we found no severe complications of gastroesophageal reflux in the pediatric age group. (c) 2005 Elsevier Inc. All rights reserve

Transcription Factor 2I Regulates Neuronal Development via TRPC3 in 7q11.23 Disorder Models

Williams syndrome (WS) and 7q11.23 duplication syndrome (Dup7q11.23) are neurodevelopmental disorders caused by the deletion and duplication, respectively, of ~ 25 protein-coding genes on chromosome 7q11.23. The general transcription factor 2I (GTF2I, protein TFII-I) is one of these proteins and has been implicated in the neurodevelopmental phenotypes of WS and Dup7q11.23. Here, we investigated the effect of copy number alterations in Gtf2i on neuronal maturation and intracellular calcium entry mechanisms known to be associated with this process. Mice with a single copy of Gtf2i (Gtf2i+/Del) had increased axonal outgrowth and increased TRPC3-mediated calcium entry upon carbachol stimulation. In contrast, mice with 3 copies of Gtf2i (Gtf2i+/Dup) had decreases in axon outgrowth and in TRPC3-mediated calcium entry. The underlying mechanism was that TFII-I did not affect TRPC3 protein expression, while it regulated TRPC3 membrane translocation. Together, our results provide novel functional insight into the cellular mechanisms that underlie neuronal maturation in the context of the 7q11.23 disorders

Narrowing the difference in dose delivery for IOERT and IOBT for locally advanced and locally recurrent rectal cancer

Purpose: Intra-operative radiotherapy (IORT) has been used as a tool to provide a high-dose radiation boost to a limited volume of patients with fixed tumors with a likelihood of microscopically involved resection margins, in order to improve local control. Two main techniques to deliver IORT include high-dose-rate (HDR) brachytherapy, termed 'intra-operative brachytherapy' (IOBT), and electrons, termed 'intra-operative electron radiotherapy' (IOERT), both having very different dose distributions. A recent paper described an improved local recurrence-free survival favoring IOBT over IOERT for patients with locally advanced or recurrent rectal cancer and microscopically irradical resections. Although several factors may have contributed to this result, an important difference between the two techniques was the higher surface dose delivered by IOBT. This article described an adaptation of IOERT technique to achieve a comparable surface dose as dose delivered by IOBT. Material and methods: Two steps were taken to increase the surface dose for IOERT: 1. Introducing a bolus to achieve a maximum dose on the surface, and 2. Re-normalizing to allow for the same prescribed dose at reference depth. Conclusions: We describe and propose an adaptation of IOERT technique to increase surface dose, decreasing the differences between these two techniques, with the aim of further improving local control. In addition, an alternative method of dose prescription is suggested, to consider improved comparison with other techniques in the future