BMJ Paediatrics Open最新文献

Background: The Rome criteria define childhood functional constipation but do not address refractory constipation. Attempts to define refractory constipation lack consensus. The interchangeable use of 'refractory' and 'intractable' or 'therapy-resistant' constipation and lack of understanding of the therapeutic ceilings before this diagnosis complicates the definition.

Aim: To conduct an online cross-sectional study among medical professionals and researchers across a range of countries, to propose a consensus definition, terminology and duration of medically unresponsive constipation.

Method: An expert-designed questionnaire was disseminated via Google Forms in a two-stage study over 2 months targeting paediatric gastroenterology professionals globally and Latin American clinicians with a translated version. The questionnaire had seven critical questions containing details needed to define medically unresponsive constipation. The study protocol was approved by the ethics review panel.

Results: The survey involved 1079 participants: 87 from various countries in the first phase and 992 from Latin America in the second. There were 619 (57.3%) general paediatricians and 462 (43 %) paediatric gastroenterologists. The preferred term to indicate poorly responding constipation was 'therapy-resistant constipation' (47.8%), followed by 'refractory constipation' (43.6%). The majority of respondents (92.9%) agreed on considering a time frame for defining refractory constipation, with 37.7% suggesting 2-3 months. 467 (43.2%) recommended including failure despite maximum laxative therapy with two agents should be considered as previous therapy failure. Compliance with therapy was deemed essential for successful treatment by 91.1%, assessed through detailed history-taking (47.4%) or medical/pharmacy records (29.4%).

Conclusion: Based on the professional views collected in this study, we propose the term 'therapy-resistant constipation' and it can be defined as constipation that is not responding to a maximum dose of at least two laxatives of different classes for a minimum of 3 months with good compliance in a secondary or tertiary care facility.

Future time perspectives (FTP) and future-related concerns in adolescence remain underexplored. We aimed to identify factors associated with limited FTP and describe future-related concerns. Data were drawn from a population-based sample of adolescents aged 14-17 participating in the SEROCoV-KIDS cohort study, in Geneva, Switzerland (October 2022). Of 329 adolescents, 56/329 (17%) reported limited FTP; determinants included prior low mental well-being, limited social support, sexual minority identity, academic difficulties and excessive screen time. Adolescents' main future-oriented concerns encompassed failure, education and climate change. These results underscore the need to address future perspectives/concerns among young people and implement interventions that strengthen adolescent resilience.

Background: Receiving communication of positivity for metabolic diseases at Expanded Newborn Screening can be extremely stressful for parents, both in case of false positive and true positive cases. However, little is known about the predictors of distress and differential impact on mothers and fathers.

Methods: In this longitudinal study, 169 fathers and 171 mothers referred to one of the Italian metabolic centres for communication of positivity completed a survey including General Health Questionnaire-12, Emotion Thermometers (measuring stress, anxiety, depression, anger and need for help), Impact of Event Scale-Revised, Multidimensional Scale of Perceived Social Support and Emotion Regulation Questionnaire. Perceived severity and control of the children's health were also assessed. The survey was completed in person after the first session at metabolic centres and online after 1, 3 and 6 months.

Results: Nearly 80% of parents reported a clinical level of distress and anxiety after the communication of positivity, one-third of them reported post-traumatic symptoms and more than half of parents reported a need for help. After 6 months, there are still more than 30% of parents with a clinical level of distress and anxiety, 6% with post-traumatic symptoms and more than 20% who continue to express a need for help. No gender difference was reported and no differences emerged between pre-COVID-19 and post-COVID-19 periods for parental distress and post-traumatic symptoms.Social support, perceived severity and control of the child's health-but not gender or previous parental experience-predicted the post-traumatic symptoms at baseline while at 6 months the only significant predictor was perceived severity.

Conclusion: Adequate psychological support should be provided from the initial communication for both parents and for true positive, false positive and variants of uncertain significance/heterozygous carrier cases.

Background: The COVID-19 pandemic placed increased pressure on service provision and healthcare worker (HCW) wellness. As the crisis of the pandemic receded, paediatric healthcare staff required an appropriate response to facilitate individual and organisational recovery, to minimise long-term HCW burn-out and to be better equipped for future crisis in paediatric healthcare.

Objective: To explore the experiences of HCWs working during the COVID-19 pandemic in an acute paediatric hospital to determine an appropriate leadership response in the postcrisis work environment.

Methods: Qualitative research design using responses from open-ended questions from 133 clinical and non-clinical staff (89% clinical) from an Irish paediatric teaching hospital. Responses were thematically analysed.

Results: Paediatric HCWs experienced frustration, uncertainty, anxiety and stress, during the pandemic crisis. Perceived organisational contributors included communication inconsistencies, inadequate support and resources, including staff shortages. This exposed remaining staff to high risk for long-term burn-out as the pandemic recedes. Three themes were developed detailing this: support, communication and trust.

Conclusion: This research supports the long-standing need to increase mental health service investment and to implement an appropriate response to regain and maintain a healthy workforce, post-COVID-19. The organisational response should address the biopsychosocial needs of the individual and paediatric healthcare organisations should work dynamically, creatively and collaboratively to ensure the psychological safety of their workforce.

Background: Children and young people (CYP) with primary brain tumour (PBT) are at high risk for developing late effects, potentially affecting long-term quality of life (QoL). In low-income and middle-income countries, QoL has not been studied in depth in CYP. In the present study, CYP treated for PBTs in Pakistan were evaluated regarding (A) mean change in QoL scores pretreatment and 12 months post-treatment and (B) predictors of change in QoL scores 12 months post-treatment.

Methods: A prospective cohort study was conducted from November 2020 to July 2023. CYP aged 5-21 years, with newly diagnosed PBTs, were recruited from tertiary care centres in Karachi, Pakistan. QoL was assessed using the Paediatric Quality of Life Inventory 4 generic and brain tumour module, pretreatment and at 12 months post-treatment, by a trained psychologist.

Results: A total of 48 patients diagnosed with PBTs were enrolled in the study. At the 12-month post-treatment, 25 (52%) of the patients were reassessed, while 23 (48%) were lost to follow-up. There was no significant difference in mean global QoL scores of patients at 12 months post-treatment. On multivariable analysis, there was a statistically significant improvement in mean global QoL scores among those who underwent total surgical tumour resection (beta 7.7; 95% CI 0.9, 14.5) and maximum safe surgical tumour resection (beta 10.6; 95% CI 4.7, 16.6). However, there was a significant decline in mean global QoL scores among those who had hydrocephalous at diagnosis managed with a shunt and/or external ventricular drain (EVD) (beta -10.0; 95% CI -14.5, -5.5).

Conclusion: This study found a decline in mean global QoL scores among those with hydrocephalous at diagnosis who were managed with a shunt and/or EVD but an improvement in those who underwent total or maximum safe surgical tumour resection. Larger-scale studies are needed to comprehensively evaluate and validate these outcomes.

Background: The cause of acute paediatric hepatitis of unknown aetiology (2022) has not been established despite extensive investigation.

Objective: To summarise the evidence for and against a causal role for human adenovirus (HAdv), adeno-associated virus 2 (AAV-2) and SARS-CoV-2 in outbreaks of paediatric hepatitis in 2022.

Methods: We appraised and summarised relevant evidence for each of the Bradford Hill criteria for causality using quantitative (statistical modelling) and qualitative (narrative coherence) approaches. Each team member scored the evidence base for each criterion separately for HAdv, AAV-2 and SARS-CoV-2; differences were resolved by discussion. We additionally examined criteria of strength and temporality by examining the lagged association between SARS-CoV-2 positivity, respiratory HAdv positivity, positive faecal HAdv specimens and excess A&E attendances in 1-4 years for liver conditions in England.

Results: Assessing criteria using the published literature and our modelling: for HAdv three Bradford Hill criteria (strength, consistency and temporality) were partially met; and five criteria (consistency, coherence, experimental manipulation, analogy and temporality) were minimally met. For AAV-2, the strength of association criterion was fully met, five criteria (consistency, temporality, specificity, biological gradient and plausibility) were partially met and three (coherence, analogy and experimental manipulation) were minimally met. For SARS-CoV-2, five criteria (strength of association, plausibility, temporality, coherence and analogy) were fully met; one (consistency) was partially met and three (specificity, biological gradient and experimental manipulation) were minimally met.

Conclusion: Based on the Bradford Hill criteria and modelling, HAdv alone is unlikely to be the cause of the recent increase in hepatitis in children. The causal link between SARS-CoV-2, and to a lesser degree AAV-2, appears substantially stronger but remains unproven. Hepatitis is a known complication of multisystem inflammatory syndrome in children following COVID-19, and SARS-CoV-2 has been linked to increased susceptibility to infection post-COVID-19, which may suggest complex causal pathways including a possible interaction with AAV-2 infection/reactivation in hosts that are genetically susceptible or sensitised to infection.

Background: The aetiology of congenital heart disease (CHD) is multifactorial. Environmental risk factors have emerged as an important modifiable determinant of several congenital cardiac conditions. Previous studies have shown a strong relationship between CHD and air pollution. Much less is known about the influence of ambient temperature on CHD. The primary objective of this meta-analysis was to examine the association between exposure to extreme heat events (EHE) during pregnancy and CHD in the offspring.

Methods: comprehensive literature search was conducted using electronic databases such as Scopus and PubMed, along with a review of secondary references. This process yielded eight studies that met the inclusion criteria: four from the USA, two from China, one from Israel and one from Canada. The risk of bias was assessed using the Newcastle Ottawa scale. Mixed-effect regression was used to pool the estimates of individual studies. Heterogeneity was measured by I². Results were visualised by a forest plot, a Baujat plot and an albatross plot. Subgroup analyses were performed for climate zones, exposure definitions and the study region. As a sensitivity analysis, a leave-one-out meta-analysis was performed to assess the robustness of the estimates.

Results: Exposure to EHE during pregnancy increased the risk for CHD in the offspring (OR=1.12, 95% CI 1.04 to 1.34). Studies from the USA observed the smallest effect (OR=1.01, 95% CI 0.91 to 1.13), studies in the continental climate zone observed a slightly larger effect (OR=1.07, 95% CI 0.97 to 1.19), and studies from the temperate climate zone observed the largest effect (OR=1.35, 95% CI 1.23 to 1.48). Subgroup analysis was conducted with respect to exposure, effect definition and region. The residual heterogeneity measures were 88% (exposure), 61% (effect definition) and 38% (Region).

Conclusion: The association between prenatal EHE exposure and future risk of CHD has important implications for pregnant mothers, infant health and health policy. Future studies should explore the additional burden of CHD contributed by current trends in global ambient temperature in other parts of the world.

Prospero registration number: CRD42023455934.

Objective: Lack of physical activity (PA) and sedentary behaviour (SB) have emerged as critical global health concerns in children and are believed to be associated with functional constipation (FC). The present study aims to explore this potential association.

Design: A comprehensive search of PubMed, Scopus, Web of Science, Embase, Cochrane Library and PsycInfo databases was conducted through 2023 using terms related to constipation and PA and SB in ages 0-18 years. Titles and abstracts were screened against eligibility criteria. Constipation was diagnosed using Rome (II-IV) criteria. Full-text reviews were reviewed, and data were extracted. Risk of Bias in Non-randomized Follow-up Studies of Exposure quality assessment tool was used to evaluate the risk of bias of studies.

Main outcome measures: We assessed the association between lack of PA/SB and FC.

Results: A total of 2170 titles were screened. Nine studies encompassing 3849 children from six countries were included. Of these, four were community/school-based studies, one was a birth cohort, three were case series and two were hospital-based case-controlled studies. All 10 studies assessed the association between PA and FC. Only three showed an association between lack of PA and FC. Five studies evaluated the effects of SB on FC, and only two reported a positive association. The methods used to assess PA/SB differed across the studies. All nine studies included in the systematic review were rated as having high risk of bias.

Conclusions: Despite numerous studies suggesting a link between insufficient PA/SB and FC, this systematic review did not uncover compelling evidence supporting such an association.

Objective: Children with special healthcare needs (SHCN) due to a chronic health condition perform more poorly at school compared with their classmates. We aimed to estimate the effects of past, current, transient, emerging and persistent SHCN on school performance in primary school children.

Methods: Data from the German population-based prospective cohort study ikidS were used. The children withSHCN screener was administered before school entry (T1) and at the end of first (T2) and third grade (T3). Grades for German, maths and science (range: 1 (Very Good) to 6 (Failure)) were obtained at the end of third grade (age 8-9 years), and an average grade was calculated. Associations between the timing of SHCN and average grade were estimated by mixed linear regression models adjusted for potential confounding variables.

Results: 751 children were included, and 21% had ever SHCN. Children with ever SHCN had poorer school performance than children with never SHCN (adjusted mean difference in average grade [95% CI]: 0.17 [0.06; 0.28]). SHCN in the third year were associated with a poorer average grade (0.29 [0.16; 0.41]) compared with healthy children. Only emerging (0.31 [0.15; 0.48]) and persistent (0.25 [0.07; 0.43]) SHCN were associated with average grade.

Conclusions: This study demonstrates the negative effect of current, emerging and persistent SHCN on academic performance in primary school children. Consequently, students should be regularly assessed for SHCN during school age. Timely interventions may help reduce the adverse effects of chronic health conditions on academic achievements in childhood.