Waiting for child developmental and rehabilitation services: an overview of issues and needs

Concern about the length of time that children, young people, and families may have to wait to access assessment, diagnostic, interventional, therapeutic, and supportive child developmental and rehabilitation (CDR) services is widespread, but adequate data collection and research on this issue remain limited. We review key concepts and issues relevant to waiting for CDR services from the published literature, a national workshop devoted to this topic, and international experience. We conclude that gaps in data, evidence, and consensus challenge our ability to address the issue of waiting for CDR services in a systematic way. A program of research coupled with actions based on consensus-building is required. Research priorities include acquiring evidence of the appropriateness and effectiveness of different models of intervention and rehabilitation services, and documenting the experience and expectations of waiting families. Consensus-building processes are critical to identify, categorize, and prioritize \u27sentinel\u27 components of CDR service pathways: (1) to reduce the inherent complexity of the field; (2) to create benchmarks for waiting for these respective services; and (3) to develop definitions for wait-time subcomponents in CDR services. Collection of accurate and replicable data on wait times for CDR services can be used to document baseline realities, to monitor and improve system performance, and to conduct comparative and analytic research in the field of CDR services

Toward the development of the International Classification of Functioning Core Sets for children with cerebral palsy: a global expert survey

The goal of the International Classification of Functioning is to standardize the classification of health and function of children around the world. To facilitate the application of this classification, International Classification of Functioning-based tools like the Core Sets are being developed. We conducted an international survey of professional experts to identify the most relevant areas of functioning in children with cerebral palsy. The questionnaire covered each component of the classification. In total, 193 professionals completed the survey (response rate 78%). Overall, 9706 answers were linked to the classification (pediatric version) by 2 professionals. From the experts\u27 perspective, movement-related areas and social participation are the most relevant areas of functioning. Experts suggest a more comprehensive profile of functioning in particular in areas of personal capacity and social participation. The results of this survey will inform the development of the International Classification of Functioning Core Sets for children with cerebral palsy

Conceptualizing childhood health problems using survey data: a comparison of key indicators

<p>Abstract</p> <p>Background</p> <p>Many definitions are being used to conceptualize child health problems. With survey data, commonly used indicators for identifying children with health problems have included chronic condition checklists, measures of activity limitations, elevated service use, and health utility thresholds. This study compares these different indicators in terms of the prevalence rates elicited, and in terms of how the subgroups identified differ.</p> <p>Methods</p> <p>Secondary data analyses used data from the National Longitudinal Survey of Children and Youth, which surveyed a nationally representative sample of Canadian children (n = 13,790). Descriptive analyses compared healthy children to those with health problems, as classified by any of the key indicators. Additional analyses examined differences between subgroups of children captured by a single indicator and those described as having health problems by multiple indicators.</p> <p>Results</p> <p>This study demonstrates that children captured by any of the indicators had poorer health than healthy children, despite the fact that over half the sample (52.2%) was characterized as having a health problem by at least one indicator. Rates of child ill health differed by indicator; 5.6% had an activity limitation, 9.2% exhibited a severe health difficulty, 31.7% reported a chronic condition, and 36.6% had elevated service use. Further, the four key indicators captured different types of children. Indicator groupings differed on child and socio-demographic factors. Compared to children identified by more than one indicator, those identified only by the severe health difficulty indicator displayed more cognitive problems (p < 0.0001), those identified only by the chronic condition checklist had a greater likelihood of reporting allergies or asthma (p < 0.0001), and those identified as having elevated service use only were more affluent (p = 0.01) and showed better overall health (p < 0.0001). Children identified by only a single indicator were less likely to have serious health problems than those identified by two or more indicators.</p> <p>Conclusion</p> <p>We provide information useful to researchers when selecting indicators from survey data to identify children with health problems. Researchers and policy makers need to be aware of the impact of such definitions on prevalence rates as well as on the composition of children classified as being in poor health.</p

Measuring the patient perspective on latissimus dorsi donor site outcomes following breast reconstruction.

BACKGROUND: There is little evidence about the long-term donor site outcome of latissimus dorsi breast reconstruction and no patient-reported outcome measures designed specifically for the procedure. METHODS: A prospective cohort of breast cancer patients having latissimus dorsi reconstruction after a mastectomy was recruited from 270 hospitals in the United Kingdom. An 18-month follow up questionnaire containing two novel scales was sent to consenting patients. The prevalence of aesthetic and functional morbidity at the donor site was described. The two new scales were refined using the Rasch measurement model and subsequently validated. RESULTS: 1,096 women completed the new scales. 78% of patients reported that no back appearance issues had bothered them "most of the time" or "all of the time" in the past two weeks. The equivalent figure for functional morbidity was 60%. Four items were eliminated following initial psychometric testing. This produced an 8-item Back Appearance scale and an 11-item Back and Shoulder Function scale. Both scales showed adequate fit to the Rasch measurement model. Higher levels of aesthetic and functional bother were observed for completely autologous procedures versus those where latissimus dorsi reconstruction was used to cover an implant (p <0.05). Higher levels of aesthetic bother were observed in women who had suffered a perioperative complication at the donor site (p = 0.003). CONCLUSION: These results can inform patients of the morbidity associated with latissimus dorsi reconstruction. The new scales can be used to compare groups undergoing different variations of the procedure and to monitor individual patients

Recursive partitioning vs computerized adaptive testing to reduce the burden of health assessments in cleft lip and/or palate : comparative simulation study

Background: Computerized adaptive testing (CAT) has been shown to deliver short, accurate, and personalized versions of the CLEFT-Q patient-reported outcome measure for children and young adults born with a cleft lip and/or palate. Decision trees may integrate clinician-reported data (eg, age, gender, cleft type, and planned treatments) to make these assessments even shorter and more accurate. Objective: We aimed to create decision tree models incorporating clinician-reported data into adaptive CLEFT-Q assessments and compare their accuracy to traditional CAT models. Methods: We used relevant clinician-reported data and patient-reported item responses from the CLEFT-Q field test to train and test decision tree models using recursive partitioning. We compared the prediction accuracy of decision trees to CAT assessments of similar length. Participant scores from the full-length questionnaire were used as ground truth. Accuracy was assessed through Pearson’s correlation coefficient of predicted and ground truth scores, mean absolute error, root mean squared error, and a two-tailed Wilcoxon signed-rank test comparing squared error. Results: Decision trees demonstrated poorer accuracy than CAT comparators and generally made data splits based on item responses rather than clinician-reported data. Conclusions: When predicting CLEFT-Q scores, individual item responses are generally more informative than clinician-reported data. Decision trees that make binary splits are at risk of underfitting polytomous patient-reported outcome measure data and demonstrated poorer performance than CATs in this study

The HAND-Q : Psychometrics of a New Patient-reported Outcome Measure for Clinical and Research Applications

Background: The perspective of the patient in measuring the outcome of their hand treatment is of key importance. We developed a hand-specific patient-reported outcome measure to provide a means to measure outcomes and experiences of care from the patient perspective, that is, HAND-Q. Methods: Data were collected from people with a broad range of hand conditions in hand clinics in six countries between April 2018 and January 2021. Rasch measurement theory analysis was used to perform item reduction and to examine reliability and validity of each HAND-Q scale. Results: A sample of 1277 patients was recruited. Participants ranged in age from 16 to 89 years, 54% were women, and a broad range of congenital and acquired hand conditions were represented. Rasch measurement theory analysis led to the refinement of 14 independently functioning scales that measure hand appearance, health-related quality of life, experience of care, and treatment outcome. Each scale evidenced reliability and validity. Examination of differential item functioning by age, gender, language, and type of hand condition (ie, nontraumatic versus traumatic) confirmed that a common scoring algorithm for each scale could be implemented. Conclusions: The HAND-Q was developed following robust psychometric methods to provide a comprehensive modular independently functioning set of scales. HAND-Q scales can be used to assess and compare evidence-based outcomes in patients with any type of hand condition.Peer reviewe

Rasch Analysis of Patient- and Parent-Reported Outcome Measures in the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set for Cleft Lip and Palate

Objectives: The aim of this study was to evaluate the psychometric performance of the patient- and parent-reported measures in the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set for Cleft Care, and to identify ways of improving concept coverage. Methods: Data from 714 patients with cleft lip and/or palate, aged 8 to 9, 10 to 12.5, and 22 years were collected between November 2015 and April 2019 at Erasmus University Medical Center, Boston Children's Hospital, Duke Children's Hospital, and from participating sites in the CLEFT-Q Phase 3 study. The Standard Set includes 9 CLEFT-Q scales, the Nasal Obstruction Symptom Evaluation (NOSE) questionnaire, the Child Oral Health Impact Profile–Oral Symptoms Scale (COHIP-OSS), and the Intelligibility in Context Scale (ICS). Targeting, item-fit statistics, thresholds for item responses, and measurement precision (PSI) were analyzed using Rasch measurement theory. Results: The proportion of the sample to score within each instruments range of measurement varied from 69% (ICS) to 92% (CLEFT-Q teeth and COHIP-OSS). Specific problems with individual items within the NOSE and COHIP-OSS questionnaires were noted, such as poor item fit to the Rasch model and disordered thresholds (6 of 10). Reliability measured with PSI was above 0.82 for the ICS and all but one CLEFT-Q scale (speech distress). PSIs were lowest for the COHIP-OSS (0.43) and NOSE questionnaire (0.35). Conclusion: The patient- and parent-reported components within the facial appearance, psychosocial function, and speech domains are valid measures; however, the facial function and oral health domains are not sufficiently covered by the CLEFT-Q eating and drinking, NOSE, and COHIP-OSS, and these questionnaires may not be accurate enough to stratify cleft-related outcomes

General population normative scores for interpreting the BODY-Q

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Development of an international standard set of patient-centred outcome measures for overall paediatric health: a consensus process

Objective: To develop an Overall Pediatric Health Standard Set (OPH-SS) of outcome measures that captures what matters to young people and their families and recognising the biopsychosocial aspects of health for all children and adolescents regardless of health condition. Design: A modified Delphi process. Setting: The International Consortium for Health Outcomes Measurement convened an international Working Group (WG) comprised of 23 international experts from 12 countries in the field of paediatrics, family medicine, psychometrics as well as patient advisors. The WG participated in 11 video-conferences, through a modified Delphi process and 9 surveys between March 2018 and January 2020 consensus was reached on a final recommended health outcome standard set. By a literature review conducted in March 2018, 1136 articles were screened for clinician and patient-reported or proxy-reported outcomes. Further, 4315 clinical trials and 12 paediatric health surveys were scanned. Between November 2019 and January 2020, the final standard set was endorsed by a patient validation (n=270

A multilab study of bilingual infants: Exploring the preference for infant-directed speech

From the earliest months of life, infants prefer listening to and learn better from infant-directed speech (IDS) compared with adult-directed speech (ADS). Yet IDS differs within communities, across languages, and across cultures, both in form and in prevalence. This large-scale, multisite study used the diversity of bilingual infant experiences to explore the impact of different types of linguistic experience on infants’ IDS preference. As part of the multilab ManyBabies 1 project, we compared preference for North American English (NAE) IDS in lab-matched samples of 333 bilingual and 384 monolingual infants tested in 17 labs in seven countries. The tested infants were in two age groups: 6 to 9 months and 12 to 15 months. We found that bilingual and monolingual infants both preferred IDS to ADS, and the two groups did not differ in terms of the overall magnitude of this preference. However, among bilingual infants who were acquiring NAE as a native language, greater exposure to NAE was associated with a stronger IDS preference. These findings extend the previous finding from ManyBabies 1 that monolinguals learning NAE as a native language showed a stronger IDS preference than infants unexposed to NAE. Together, our findings indicate that IDS preference likely makes similar contributions to monolingual and bilingual development, and that infants are exquisitely sensitive to the nature and frequency of different types of language input in their early environments