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Children with medical complexity (CMC) have complex chronic conditions with significant functional impairment, contributing to high caregiving demand. This study seeks to explore impacts of parental caregiving for CMC. Fifteen caregivers of CMC followed at a tertiary care hospital participated in semi-structured interviews. Interviews were concurrently analyzed using a qualitative description framework until thematic saturation was reached. Codes were grouped by shared concepts to clarify emergent findings. Four affected domains of parental caregiver experience with associated subthemes (in parentheses) were identified: personal (identity, physical health, mental health), family (marriage, siblings, family quality of life), social (time limitations, isolating lived experience), and financial (employment, medical costs, accessibility costs). Despite substantial challenges, caregivers identified two core determinants of personal resilience: others’ support (hands-on, interpersonal, informational, material) and a positive outlook (self-efficacy, self-compassion, reframing expectations). Further research is needed to understand the unique needs and strengths of caregivers for this vulnerable population.

Background Children with medical complexity (CMC) require care coordination across hospital, home, and community settings. The use of a medical care plan supports care coordination; however, the content of the care plan is often controlled by the health care team, limiting the ability of parents to edit in real time. It has been suggested that these limitations could be improved by using online care plans shared between parents of CMC and care team members (CTM). Objectives The objective of this study was to explore the perspectives of parents and care team members of children with medical complexity following the use of an online care plan. Design/Methods This qualitative study utilized grounded theory (GT) to explore parent and CTM experiences using an online editable medical care plan. Parents of CMC were invited to use a novel online patient-facing platform to coordinate their child’s care and edit/share their child’s care plan. They were also able to invite any of their child’s CTM to use the platform. Parents could make changes to the care plan including adding, editing, or deleting information. After 6 months, parents and CTM participated in semi-structured interviews aimed at exploring their experiences using the online care plan. Consistent with GT, constant comparative analysis was used as data analysis occurred concurrently with interviews, which allowed for theoretical sampling and theory generation. Results Thirty-five semi-structured interviews with parents (n=15) and CTM (n=20) were conducted. Interviews revealed three major themes (9 sub-themes): 1) navigation of uncharted roles (trust, role sharing, responsibility), 2) requirements for success (collaborative review, electronic medical record integration, online access), and 3) cohesive care (autonomy, accessibility and convenience, same page). These themes informed the creation of a theory outlining how and why a robust online shared care plan can improve care for CMC. Both parents and CTM identified the uniqueness of this care plan located on the online platform as it promoted collaboration and trust whilst allowing for increased autonomy and convenience. Conclusion Online shared care plans, implemented in a safe and thoughtful manner, promote cohesive partnering between parents and care team members while allowing easy access to information. This facilitates care coordination and a cohesive partnering between parents and care team members involved in the care of CMC.

Background During Wave 3 of the COVID-19 pandemic, 15 community hospital paediatric inpatient units (comprising 167 beds) in Toronto were directed to close by the Greater Toronto Area (GTA) Hospital Incident Management System (IMS) Command Centre to increase adult inpatient bed capacity. All paediatric patients from closed inpatient units were redirected to a single tertiary care paediatric hospital, which increased capacity to accommodate these additional patients through activation of surge plans, while community hospitals redeployed resources to fill much needed gaps in adult care. Objectives The objective was to describe patient characteristics of all transfers during the closure to explore the impact of community paediatric inpatient unit closures on transfers to the tertiary hospital. Design/Methods A chart review of all transferred patients was conducted during the mandated closure and subsequent reopening. Transfers excluded ICU-level transfers as these were not impacted by IMS mandated closures. All transfers were categorized as requiring tertiary care (i.e. would typically be transferred) or not requiring tertiary care (i.e. only transferred due to the closure). Variables collected included sending hospital, admitting diagnosis, patient age, hospital disposition, and length of stay. Data was collected until the last paediatric unit reopened. Quality improvement project approval was granted by the institution. Results A total of 858 patients were transferred to the tertiary hospital during the 67 day closure; of those, 530 were transferred solely to increase adult bed capacity (i.e. were categorized as patients requiring non-tertiary care). The majority of patients were admitted to general paediatrics (52%), and 39% went to a surgical inpatient unit. Most patients (68%) admitted had a length of stay between 24 and 72 hours. A third of patients admitted were under 2 years old, and a third were over 12 years old. The top three diagnoses for admission were infections, gastrointestinal issues, and general surgery. Two-thirds (60%) of transfers from closed sites came from three sites. Conclusion More than half of the transfers occurred solely due to the mandated closures, and transfers returned to a stable volume once all sites re-opened. The GTA hospital system was able to respond to the mandated closure effectively through clear high-level communication, escalation processes and structures as well as responsive, real-time problem solving. Closures increased potential adult inpatient capacity by 6740 bed days and demonstrated an unprecedented system-wide approach to the provision of integrated paediatric care across the region.

Background While mental health disorders are common in the postpartum period, the mental health of parents of infants in the neonatal intensive care unit (NICU) may be further compromised. Previous research demonstrates that up to 40% of NICU parents exhibit elevated levels of depression and anxiety, and endorse trauma symptoms shortly after their baby’s birth. Objectives To describe the parental and infant characteristics and costs associated with MHS use in parents of infants recently discharged from seven Canadian level III NICUs. Design/Methods The study was nested in an ongoing clinical trial of parents of high-risk infants across seven Canadian level III NICUs. Parent and infant characteristics were obtained at enrollment and summarized. Information pertaining to MHS use was obtained from the Resource Use Questionnaire administered to parents at 4 months post-enrollment (RUQ-4m). A pricing table allocating standard costs for services was created to estimate MHS costs for the services reported across the follow-up interval. Results A total of n=213 families completed the RUQ-4m. Of these, 33 individual parents (8%; n=25 female, n=8 male) across 28 families (13%) used MHS following infant discharge from the NICU. Therapy, including psychotherapy and social work, was the most utilized service (46%), followed by psychology (24%) and psychiatry (12%). Using the pricing table to estimate costs, an average of $541 per month was spent on MHS per family. Of these costs, 34% were paid directly by a government program, while 31% were paid at least in part out-of-pocket. Three-quarters (72%) of MHS users were born in Canada compared to 47% of non-users, 72% were on a leave from work compared to 59% of non-users, and 79% were first-time parents compared to 62% of non-users. The average length of hospital admission for infants of MHS users was 100 days compared to 143 days for non-MHS users. Finally, 20% of MHS users reported past or current depression compared to 8% of non-users. Conclusion We found that just over one-tenth of families used MHS post-discharge, that those who used MHS primarily used therapy, and that mental health services cost an estimated $540 per family per month. This is the first pan-Canadian study to describe MHS utilization and cost amongst parents post-NICU discharge. Given the high rates of mental health conditions in this population, the rates of MHS use reported were lower than expected. This preliminary data may represent those who were able to access care, rather than all participants who may have required MHS.

Background Children with medical complexity (CMC) are a highly medicalized population of children due to the complexity of their clinical presentations, various diagnoses, and multiple care providers. Real-time health information can inform clinicians to make better recommendations and improve clinical outcomes. In other populations, such as children with Type 1 diabetes, online health symptom trackers (HST) were used to facilitate clinic visits and track symptoms longitudinally. To-date, HSTs have not yet been examined in a clinical setting with CMC and their families. Objectives The aims of our study were to create a standardized online tool, which supports the creation of online HSTs, and to assess their utility in clinical care from a parent and health care provider (HCP) perspective. Design/Methods Parents of CMC were invited to use a standardized online care platform called Connecting2gether for 6-months and create online HSTs that could be shared with their HCPs. Online HSTs could be added by parents from a prepopulated list with an open notes section, and the ‘Signs and Symptoms’ trackers could be customized by parents. A demographic survey was completed at baseline. At 6-months a tracker acceptability survey and in-depth semi-structured qualitative interviews were completed to assess the utility and usability of HSTs. HST usage data were also collected. Interviews were analyzed via thematic analysis whereby codes were generated to inform themes, and surveys were analyzed using descriptive methods. Results Thirty-six parents enrolled on the platform and 21 (57%) created at least one online HST during the study period. The most used HSTs were ‘Signs and Symptoms’, ‘Sleep’, and ‘How I Feel’. Majority of parents (86%) reported finding the trackers useful. Only 55% of HCPs viewed the HSTs and of those, 36% reporting using them in clinical care. Qualitative interviews revealed three themes: 1) HST Usability: HSTs were used in different settings such as in clinic and school, they were used to guide conversation as a visual over time, and they used in decision making; 2) Enhancement to HST Usage: suggestions included having more options of different symptoms; and 3) Challenges and Barriers to HST Usage: including personal preference and medical stability of the patient. Conclusion The ability of online HSTs to visually depict the change in symptoms over time in CMC was found to be a benefit from a parent and HCP perspective. HSTs were identified as a tool used in clinic to guide conversation and decision making, and as a visual to track symptoms longitudinally. However, HCPs need more guidance on how to use trackers. Future directions for online HSTs include integration into the electronic health record to increase accessibility.

Vaccinationis a critical tool in the prevention of COVID‐19 infection for individuals and for communities. The mRNA vaccines contain polyethylene glycol (PEG) as a stabilizer. Currently, in North America, only the BNT162b2 (Pfizer‐BioNTech) mRNA vaccine is approved for individuals aged 12–17. Most patients treated with contemporary regimens for acute lymphoblastic leukemia receive PEG‐asparaginase (PEG‐ASNase) and 10%–30% will develop allergic reactions. Optimizing access and safety for vaccine administration for these patients is critical. This report describes a process developed to support COVID vaccination in a cohort of adolescents and young adults with a history of PEG‐ASNase allergy.

Context: Acceptance and Commitment Therapy (ACT) is an emerging treatment for improving psychological well-being. Objective: To summarize research evaluating the effects of ACT on psychological well-being in children with special health care needs (SHCN) and their parents. Data Sources: An electronic literature search was conducted in PubMed, Web of Science, Ovid/EMBASE and PsycINFO (January 2000–April 2021). Study Selection: Included were studies that assessed ACT in children with SHCN (ages 0–17y) and/or parents of children with SHCN and had a comparator group. Data Extraction: Descriptive data were synthesized and presented in a tabular format, and data on relevant outcomes (e.g., depressive symptoms, stress, avoidance and fusion) were used in the meta-analyses to explore the effectiveness of ACT (administered independently with no other psychological therapy) compared to no treatment. Results: Ten studies were identified (child (7) and parent (3)). In children with SHCN, ACT was more effective than no treatment at helping depressive symptoms (standardized mean difference [SMD] = −4.27, 95% CI: −5.20, −3.34; p < 0.001) and avoidance and fusion (SMD = −1.64, 95% CI: −3.24, −0.03; p = 0.05), but not stress. In parents of children with SHCN, ACT may help psychological inflexibility (SMD = −0.77, 95% CI: −1.07, −0.47; p < 0.01). Limitations: There was considerable statistical heterogeneity in three of the six meta-analyses. Conclusions: There is some evidence that ACT may help with depressive symptoms in children with SHCN and psychological inflexibility in their parents. Research on the efficacy of ACT for a variety of children with SHCN and their parents is especially limited, and future research is needed.

IntroductionHaving an infant admitted to the neonatal intensive care unit (NICU) is associated with increased parental stress, anxiety and depression. Enhanced support for parents may decrease parental stress and improve subsequent parent and child outcomes. The Coached, Coordinated, Enhanced Neonatal Transition (CCENT) programme is a novel bundled intervention of psychosocial support delivered by a nurse navigator that includes Acceptance and Commitment Therapy-based coaching, care coordination and anticipatory education for parents of high-risk infants in the NICU through the first year at home. The primary objective is to evaluate the impact of the intervention on parent stress at 12 months.Methods and analysisThis is a multicentre pragmatic randomised controlled superiority trial with 1:1 allocation to the CCENT model versus control (standard neonatal follow-up). Parents of high-risk infants (n=236) will be recruited from seven NICUs across three Canadian provinces. Intervention participants are assigned a nurse navigator who will provide the intervention for 12 months. Outcomes are measured at baseline, 6 weeks, 4, 12 and 18 months. The primary outcome measure is the total score of the Parenting Stress Index Fourth Edition Short Form at 12 months. Secondary outcomes include parental mental health, empowerment and health-related quality of life for calculation of quality-adjusted life years (QALYs). A cost-effectiveness analysis will examine the incremental cost of CCENT versus usual care per QALY gained. Qualitative interviews will explore parent and healthcare provider experiences with the intervention.Ethics and disseminationResearch ethics approval was obtained from Clinical Trials Ontario, Children’s Hospital of Eastern Ontario Research Ethics Board (REB), The Hospital for Sick Children REB, UBC Children’s and Women’s REB and McGill University Health Centre REB. Results will be shared with Canadian level III NICUs, neonatal follow-up programmes and academic forums.Trial registration numberClinicalTrials.gov Registry (NCT03350243).

Background and Objective Approximately 73% of children with severe neurological impairment (SNI) can experience episodes of pain and irritability often of unknown origin (PIUO). Limited research exists on how these experiences of PIUO may affect parental caregivers and families. The primary objective of this study was to understand the parental caregiver experience of caring for a child with SNI who experiences persistent PIUO. Methods We conducted a qualitative study using semi-structured interviews to explore the experience of parental caregivers of children with SNI. Interview guide questions focused on exploring pain behaviours, the diagnostic process, pharmacological and non-pharmacological management, healthcare-team support, discussion surrounding irritability, and family impact. Interviews were conducted until thematic saturation was reached. Interviews were audio-recorded, transcribed verbatim, and coded and analyzed by 2 independent reviewers using thematic analysis. Results 15 parental caregivers were interviewed, with 93% being mothers and 33% being a visible minority. Interviews revealed 3 major themes: 1) Variations in Clinical Care for PIUO; 2) The Experience and Challenges of Living with PIUO); 3) Managing the Impact of PIUO on Parental Well Being. Interviews demonstrated that parent and child can be viewed as a dyad, in which the child's experience is inherently linked to the parental experience. Conclusion Parental caregivers described caring for a child with persistent PIUO as physically and emotionally exhausting, and negatively impacting family quality of life. Interviews highlighted avenues of future exploration for clinical care, including both enhanced management pathways for children and supportive resources for education and coping for parents.

The start of a parenting journey in the neonatal intensive care unit (NICU) presents many stressors to parents. Previous research has shown parents of infants admitted to the NICU experience heightened stress, anxiety, and depression. Mental health support varies across Canadian NICUs with mixed results. One promising intervention that has not been explored in the NICU is Acceptance and Commitment Therapy (ACT), a behavioural therapy that has had positive mental health-related outcomes in similar parental populations. ACT differs from previous mental health interventions such as traditional Cognitive Behavioural Therapy (CBT) as it involves mindfulness and acceptance to increase psychological flexibility. Increased psychological flexibility is linked to greater emotional well-being, a higher quality of life, and decreased stress, anxiety, and depression. There is a need for research investigating the utility of ACT in improving mental health outcomes for parents of preterm infants.

Un nourrisson est à haut risque d’allergie alimentaire si lui ou un membre de sa famille immédiate présente une affection atopique (comme l’eczéma). Il faut promouvoir et soutenir l’allaitement, quels que soient les enjeux reliés à la prévention des allergies alimentaires, mais chez les nourrissons dont la mère ne peut pas allaiter ou choisit de ne pas le faire, il n’est pas recommandé d’utiliser une préparation en particulier (p. ex., les préparations hydrolysées) pour prévenir les allergies alimentaires. Lorsque les préparations à base de lait de vache sont introduites dans l’alimentation d’un nourrisson, il faut s’assurer de maintenir une ingestion régulière (pas nécessairement plus de 10 mL par jour) pour éviter la perte de tolérance. Chez les nourrissons à haut risque, des données concluantes indiquent que l’introduction précoce d’aliments allergènes (vers l’âge de six mois, mais pas avant l’âge de quatre mois) peut prévenir les allergies alimentaires courantes, notamment les allergies aux arachides et aux œufs. Lorsqu’un aliment allergène a été introduit, il est important d’en maintenir une ingestion régulière (p. ex., quelques fois par semaine) pour maintenir la tolérance. Il est possible d’introduire les aliments allergènes courants sans faire de pause de quelques jours entre chaque nouvel aliment. Par ailleurs, le risque d’une grave réaction lors de la première exposition est très faible chez le nourrisson. Il n’est pas recommandé de procéder au dépistage préventif en cabinet avant d’introduire des aliments allergènes. Aucune recommandation ne peut être formulée pour l’instant sur le rôle des modifications à l’alimentation de la mère pendant la grossesse ou l’allaitement, ou sur les suppléments de vitamine D, d’oméga 3, de prébiotiques ou de probiotiques pour prévenir les allergies alimentaires.

Primary Subject area Complex Care Background Children with medical complexity (CMC) are a highly medicalized population of children who require specialized care across various settings including the hospital, home and community, making care coordination challenging. Care-maps, a visual representation of the people and places involved in a patient’s care, are one such tool to facilitate care coordination (Figure 1). To date, care-maps have not yet been used in a clinical environment, examined in real time or used via a standardized approach. Objectives The aims of our study were to develop a shareable standardized online tool that supports the parental creation of a care-map, and to assess the utility of care-maps in clinical care from a parent, health care provider (HCP), and community perspective. Design/Methods Parents of CMC were invited to use a standardized online care platform called Connecting2gether for 6-months and create online care-maps that could be shared with their HCPs and other community members (i.e., teachers, secondary caregivers). Demographics and internet usage surveys were completed at baseline and an acceptability survey was completed at 6-months. Surveys were analyzed using descriptive methods and care-maps were analyzed via descriptive visual analysis. Results Thirty-seven parents enrolled on the platform and 25 (70%) created a care-map and used it for the duration of the study. Of the 25, 14 (66%) went back and made revisions and 17 (80%) reported using it in clinic, home or school. Visual analysis demonstrated 11 categories (bubbles) that were commonly included. All care-maps included a Medical Team, School/Daycare and Family and Friends category, which automatically populated. The majority of care-maps included a central child bubble with the child’s photo (92%), and Community Medical Services (i.e. rehab centers) (60%). Less frequent categories included Home Care (28%), Goals (16%), and 12% included What I Like, Funding, and Community/Foundation individual bubbles. Some parents reported initial uncertainty, but at end-of-study, some reported care-maps as the most useful feature of the platform. Fifty seven percent (12/23) of HCPs viewed the created care-map and only 20% used it in the child’s care. The majority (83%) of HCPs specifically valued seeing the big picture of the child’s care, found it easy to navigate and the detail it provided. Conclusion The ability of care-maps to illustrate the intricate web of medical and non-medical care supporting CMCs in their daily life provides insight and value for parents, HCPs and non-HCPs. Care-maps were found to be valuable from the perspective of HCPs. Parents reported initial uncertainty, highlighting the importance of the HCP promoting the use of care-maps with their patients and families.

Primary Subject area Mental Health Background Chronic childhood health conditions are known to have an impact on the well-being of family members. Parental caregivers face well-defined adverse health outcomes, though less is known about the health impacts on siblings. Objectives To assess clinical health outcomes in siblings of children with chronic health condition(s) compared to siblings of healthy children or normative data. Design/Methods We searched Ovid MEDLINE, EMBASE, the Cochrane Central Register of Controlled Trials, PsycInfo, and CINAHL through June 4, 2020. We included English-language studies that: (1) reported clinically diagnosable mental or physical health outcomes of siblings of children (0-18 years old) diagnosed with any childhood chronic health condition; (2) included a comparison group; and (3) used an experimental or observational study design. Risk of bias was assessed using the Newcastle-Ottawa Scale. Results We included 28 studies of the 9053 screened, comprising 10 cohort studies and 18 cross-sectional studies. Studies from 11 different countries reported most commonly on siblings of children with disabilities (12 studies), cancer (8 studies), or psychiatric disorders (4 studies). Siblings of children with chronic conditions had greater depression rating scale scores than their comparison groups (standardized mean difference 0.49; 95% CI 0.33-0.65; P < .001 [5 studies]) (Fig. 1), whereas anxiety scores did not differ significantly (standardized mean difference 0.24; 95% CI -0.03-0.52; P = .08 [6 studies]) (Fig. 2). Studies that reported on prevalence of psychiatric diagnoses, rather than rating scale scores, had mixed results, either indicating increased risk (3 studies) or no increased risk (4 studies) among exposed siblings. We did not meta-analyze effects for mortality (3 studies) or physical health outcomes (dental caries [1 study], traumatic brain injury [1 study], sexually transmitted infection [1 study], overweightness/obesity [1 study]) given the limited number of studies and between-study heterogeneity. Included studies were rated as high quality (12 studies) or of moderate quality (16 studies). Conclusion Siblings of children with chronic health conditions may be at an increased risk of depression. Our findings suggest the need for targeted interventions to support the psychological well-being of siblings of children with chronic health conditions.

Primary Subject area Hospital Paediatrics Background The coronavirus (COVID-19) pandemic has broad implications for children and families. Healthcare experience and delivery has changed significantly, and changes will likely continue for some time. Particular attention has been paid to delays in accessing timely pediatric care leading to unintended morbidity. Objectives This study aimed (1) to describe the broader spectrum of unintended negative consequences by describing the courses of care altered by the COVID-19 pandemic from the clinician's perspectives and (2) to identify thematic similarities to inform clinical practice change. Design/Methods All full-time doctors, dentists, and nurse practitioners working at a tertiary care children’s hospital in Canada were surveyed every two weeks throughout the initial phase of the COVID-19 pandemic. We asked them to identify and describe clinical cases in which they perceived a negative outcome associated with hospital or societal changes due to the COVID-19 pandemic. Analysis followed a qualitative case series methodology using a narrative synthesis approach to determine similarities and associated themes. Results Two-hundred and twelve clinicians reported 116 cases. Several broad themes emerged, including (1) timeliness of care, (2) disruption of child and family-centred care, (3) new pressures in the provision of safe and efficient care and (4) inequity in the experience of the COVID-19 pandemic. Within each of these themes, subthemes emerged, highlighting its impact on (1) patients, (2) their families and (3) healthcare providers. Table 1 provides examples of cases within each theme. Conclusion The broad consequences of the COVID-19 pandemic impact patients, families, healthcare providers and the healthcare system. Understanding this breadth is necessary as we strive to deliver safe, high quality, family-centred pediatric care in this new era. As the pandemic continues, we need to consider carefully how to provide elective and ambulatory care, including surgery, in this era of social distancing. Particular attention is needed to understand particular aspects, including vulnerable children and the clinician experience of the COVID-19 pandemic.

BACKGROUND AND OBJECTIVES Caregivers of children with medical complexity (CMC) face many stressors related to their child’s medical condition(s). Financial stress and its impact on housing has been reported to be a challenge among this population. However, unique housing challenges specific to CMC, including disability accommodations in the home and housing space and layout, have yet to be examined in the literature. METHODS We conducted 20 individual semistructured interviews with parents of CMC. Interviews were recorded, coded, and analyzed by using thematic analysis to emphasize, examine, and record patterns of meaning within the data. RESULTS Eighteen mothers and 2 fathers participated in individual interviews. Two major themes and subthemes (in parentheses) were identified: (1) the impact of health on housing (housing preferences, housing possibilities, and housing outcome as a trade-off) and (2) the impact of housing on health (health of the caregiver and health of the child). Parents had preferences regarding the location and layout of their home specific to their child’s illness and medical needs. In addition, parents indicated their child’s illness affected their income and home ownership status, which in turn shaped their housing possibilities. The location and layout of the family home was often the result of a trade-off between the caregiver’s housing preferences and possibilities. CONCLUSIONS Housing outcomes among CMC are often the result of a trade-off between housing preferences and possibilities, both of which are influenced by the child’s health status. Policy changes targeting housing accessibility and affordability are vital to support the health of CMC.

BACKGROUND AND OBJECTIVES: The coronavirus disease (COVID-19) pandemic has broad implications for children and families. Particular attention has been paid to delays in accessing timely pediatric care leading to unintended morbidity. In this study, we aim to describe the broader spectrum of unintended negative consequences for pediatric patients and families due to recent health care and societal changes. METHODS: All full-time doctors, dentists, and nurse practitioners working at a tertiary care children’s hospital in Canada were surveyed every 2 weeks throughout the initial phase of the COVID-19 pandemic to identify clinical cases in which they perceived a negative outcome associated with hospital or societal changes as a result of the COVID-19 pandemic. Analysis followed a qualitative case series methodology using a narrative synthesis approach to determine similarities and associated themes. RESULTS: One hundred and forty-one clinicians, representing 26 hospital divisions, reported 57 unique cases in the first 6 weeks of the study. Thematic analysis of the first 50 reported cases was used to identify 6 primary themes focusing on health care quality domains as described by the Agency for Healthcare Research and Quality (safe, effective, patient-centered, timely, efficient, and equitable care). CONCLUSIONS: In this preliminary case analysis, we describe the broad social and clinical impact of COVID-19 on hospitalized pediatric patients and their families. These themes highlight the unintended consequence on families, siblings, disease diagnosis, and hospital-based care provision. Recognition and understanding of the broad implications of the COVID-19 pandemic are necessary as we strive to deliver safe, high-quality, family-centered pediatric care in this new era.

BACKGROUND: Risk factors for severe outcomes of SARS-CoV-2 infection are not well established in children. We sought to describe pediatric hospital admissions associated with SARS-CoV-2 infection in Canada and identify risk factors for more severe disease. METHODS: We conducted a national prospective study using the infrastructure of the Canadian Paediatric Surveillance Program (CPSP). Cases involving children who were admitted to hospital with microbiologically confirmed SARS-CoV-2 infection were reported from Apr. 8 to Dec. 31 2020, through weekly online questionnaires distributed to the CPSP network of more than 2800 pediatricians. We categorized hospital admissions as related to COVID-19, incidental, or for social or infection control reasons and determined risk factors for disease severity in hospital. RESULTS: Among 264 hospital admissions involving children with SARS-CoV-2 infection during the 9-month study period, 150 (56.8%) admissions were related to COVID-19 and 100 (37.9%) were incidental infections (admissions for other reasons and found to be positive for SARS-CoV-2 on screening). Infants (37.3%) and adolescents (29.6%) represented most cases. Among hospital admissions related to COVID-19, 52 (34.7%) had critical disease, 42 (28.0%) of whom required any form of respiratory or hemodynamic support, and 59 (39.3%) had at least 1 underlying comorbidity. Children with obesity, chronic neurologic conditions or chronic lung disease other than asthma were more likely to have severe or critical COVID-19. INTERPRETATION: Among children who were admitted to hospital with SARS-CoV-2 infection in Canada during the early COVID-19 pandemic period, incidental SARS-CoV-2 infection was common. In children admitted with acute COVID-19, obesity and neurologic and respiratory comorbidities were associated with more severe disease.

Due to advances in medical care and innovations in health technology, many children with life-limiting conditions are now living longer. These children are often referred to as ‘children with medical complexity (CMC)’, and they are characterized by chronic conditions, increased health care utilization, and technology dependence. Their complexity of care and inherent fragility lead to higher risk for medication errors, both in-community and in-hospital. High rates of care fragmentation, miscommunication, and polypharmacy in CMC increase opportunities for error, particularly as children transition between health care settings and practitioners. Data on the factors contributing to higher risk of medication errors in this population and how they can be effectively addressed are lacking. This practice point provides clinical guidance for health care professionals to ensure medication safety when caring for CMC, with focus on practical strategies for outpatient and inpatient care.

OBJECTIVES: Health care providers (HCPs) require ongoing support to meet the evolving care needs of children with medical complexity (CMC). Project Extension for Community Healthcare Outcomes (ECHO) is a model for delivering technology-enabled medical education and cultivating a community of practice. In this study, we focused on developing, implementing, and evaluating the first ECHO program dedicated to the care of CMC. Specific objectives were to evaluate the program feasibility (participation and acceptability) and impact on perceived HCP knowledge, self-efficacy, and clinical practice after 6 months. METHODS: A needs assessment was conducted to inform an interprofessional CMC curriculum. This curriculum was delivered through monthly virtual TeleECHO clinics (didactic and case-based learning) from January 2018 to 2020. The program was available at no cost to HCPs throughout Ontario. Surveys were distributed at baseline and 6 months to assess program acceptability, knowledge, self-efficacy, and practice impact by using 7-point Likert scales. Descriptive and inferential data analyses were conducted. RESULTS: Twenty-four clinics were completed with a mean of 19 ± 6 attendees. Acceptability scores (n = 27) ranged from 5.0 ± 1.1 to 6.4 ± 0.6. Participants reported an improvement in their knowledge and self-efficacy across all probed topics and skills (P values ranged from CONCLUSIONS: Project ECHO is a feasible and acceptable model for virtual education of interprofessional HCPs in managing CMC. This program has the potential to increase system capacity to provide quality care to CMC close to home.

The COVID-19 pandemic has had dramatic effects on the lives of children globally. However, socially vulnerable children have been particularly impacted. Certain populations have increased vulnerabilities, including children and youth experiencing homelessness. Increased infection risk due to congregant living and challenges with physical distancing are contributing factors. An urgent need exists for a wholistic approach to care with unique cross-sectoral partnerships across disciplines. A recognition of the unintended consequence of the COVID-19 pandemic on this population is urgently required by all those supporting children. Families should receive direct support in clinical settings to identify their social needs. Partnership with community agencies and advocacy for appropriate isolation facilities for patients experiencing homelessness are critical.

Introduction/Background Pain and irritability of unknown origin (PIUO) is reported to effect 73% of children with severe neurological impairments (SNI), and is a source of stress for children and families alike. Currently, there is no consensus among clinicians on how to manage PIUO and it is often difficult to determine the source of pain. Lacking an explanation for the source leaves clinicians unable to effectively treat the pain and increases a caregiver’s obstacles in providing care. Limited research exists on the effect of PIUO on children with SNI and their families. Objectives To explore and characterize the overall experience of PIUO for children with SNI and their families. Design/Methods Semi-structured interviews were conducted with parental caregivers of children with SNI who experience PIUO and are followed by the Complex Care Program at SickKids. Interview guide topics included pain expression and management, healthcare-team support and family coping. Interviews were conducted until saturation was reached. Interviews were audio-recorded, transcribed verbatim, coded and analyzed by two independent reviewers using an inductive six-step thematic analysis process on NVivo software. Results Fifteen caregivers were interviewed, with 93% being mothers and 33% being a visible minority. Interviews revealed two major themes and associated subthemes (in parentheses): 1) Day-to-day life with PIUO (pain expression and frequency, management, and quality of life) and, 2) Areas for improvement (diagnostic process, resources and support, healthcare-team interactions). Characterizing the PIUO experience is an important area of research as findings can be used to guide clinical teams in providing holistic family-centered care to children with SNI. The findings support the need for clinical innovation by adjusting practice guidelines through the creation and implementation of an integrated clinical pathway to identify treatable causes of pain and irritability in children with SNI. Conclusion Diagnostic tests for PIUO are often inconclusive and stressful for patient-families. Limited pharmacological and non-pharmacological treatments exist for PIUO. Parental caregivers describe the experience as emotionally challenging and requested support for coping. Future research should focus on interventions for PIUO in children with SNI and reducing caregiver stress and burden.

BACKGROUND AND OBJECTIVES:Children with medical complexity (CMC) often have multiple life-limiting conditions with no unifying diagnosis and an unclear prognosis and are at high risk for morbidity and mortality. Advance care planning (ACP) conversations need to be uniquely tailored to this population. Our primary objective for this study was to develop an in-depth understanding of the ACP experiences from the perspectives of both parents and health care providers (HCPs) of CMC.METHODS:We conducted 25 semistructured interviews with parents of CMC and HCPs of various disciplines from a tertiary pediatric hospital. Interview guide questions were focused on ACP, including understanding of the definition, positive and negative experiences, and suggestions for improvement. Interviews were conducted until thematic saturation was reached. Interviews were audio recorded, transcribed verbatim, coded, and analyzed using content analysis.RESULTS:Fourteen mothers and 11 HCPs participated in individual interviews. Interviews revealed 4 major themes and several associated subthemes (in parentheses): (1) holistic mind-set, (2) discussion content (beliefs and values, hopes and goals, and quality of life), (3) communication enhancers (partnerships in shared decision-making, supportive setting, early and ongoing conversations, consistent language and practice, family readiness, provider expertise in ACP discussions, and provider comfort in ACP discussions), and (4) the ACP definition.CONCLUSIONS:Family and HCP perspectives revealed a need for family-centered ACP for CMC and their families. Our results aided the development of a family-centered framework to enhance the delivery of ACP through a holistic mind-set, thoughtful discussion content, and promoting of conversation enhancers.

Improved survival of children with life-threatening conditions has resulted in more children in need of caregiving support. For example, a child assisted by medical technology may need constant monitoring of a tracheostomy at home, often a role taken on by parents. Increased caregiving is also required for children with certain developmental or psychiatric conditions that have self-injurious or high-risk behaviours. Caregiving for a child with high needs can be a fulfilling experience, but can also pose substantial risks of burnout as well as poor mental and physical health. Caregivers face increased stress, financial burden and sometimes stigma and social isolation. Those stressors may increase the likelihood of depression and anxiety, as well as of premature maternal cardiovascular disease and mortality.1 Even if caregivers do not meet criteria for a mental illness but are struggling with high emotional distress, their parenting skills and ability to promote secure attachment may be affected. Children of parents with mental illness have elevated risk for poor growth, feeding difficulties, depression, behavioural challenges and poor learning.2 A child with a disability and high needs may be even more susceptible to these detrimental effects. Some adverse outcomes may be reversible with support for the caregiver. For example, therapy for depressed mothers has been reported to improve the child’s mental health, social and academic functioning and the mother–child interaction.3 4 Likewise, children exposed to adverse experiences—most commonly parental mental illness—have been shown to have improved language and behaviour, decreased injuries and more uptake of immunisation …

Infants at high risk for developing a food allergy have either an atopic condition (such as eczema) themselves or an immediate family member with such a condition. Breastfeeding should be promoted and supported regardless of issues pertaining to food allergy prevention, but for infants whose mothers cannot or choose not to breastfeed, using a specific formula (i.e., hydrolyzed formula) is not recommended to prevent food allergies. When cow’s milk protein formula has been introduced in an infant’s diet, make sure that regular ingestion (as little as 10 mL daily) is maintained to prevent loss of tolerance. For high-risk infants, there is compelling evidence that introducing allergenic foods early—at around 6 months, but not before 4 months of age—can prevent common food allergies, and allergies to peanut and egg in particular. Once an allergenic food has been introduced, regular ingestion (e.g., a few times a week) is important to maintain tolerance. Common allergenic foods can be introduced without pausing for days between new foods, and the risk for a severe reaction at first exposure in infancy is extremely low. Pre-emptive in-office screening before introducing allergenic foods is not recommended. No recommendations can be made at this time about the role of maternal dietary modification during pregnancy or lactation, or about supplementing with vitamin D, omega 3, or pre- or probiotics as means to prevent food allergy.

African Americans have a lower marriage rate and report lower relationship quality than whites. Experiencing stress associated with racial discrimination and financial strain may impact the intimate relationships of African American fathers, yet this topic is underexplored. We used a clinic-based sample of African American fathers in Baltimore, MD, and Washington, DC (N = 203), to examine (1) the relationship between socioeconomic factors and experiences of racial discrimination and (2) the extent to which racial discrimination, financial stress, and perceived stress are associated with marital status and intimate relationship quality for African American fathers. Education was positively associated with the racial discrimination, and relationship quality was negatively associated with perceived stress and racial discrimination. Perceived stress mediates the relationship between racial discrimination and relationship quality. Addressing interpersonal and institutional racism may impact relationship quality for African American men.

An Icteric Tongue A 12-year-old boy presented with a 4-day history of sore throat and dark urine. He was found to have jaundice with an icteric tongue, and the hemoglobin level was 6.1 g per decili...

IntroductionTechnological and medical advances have led to a growing population of children with medical complexity (CMC) defined by substantial medical needs, healthcare utilisation and morbidity. These children are at a high risk of missed, fragmented and/or inappropriate care, and families bear extraordinary financial burden and stress. While small in number (Methods and analysisOur primary objective is to evaluate the CCKO intervention using a randomised waitlist control design. The waitlist approach involves rolling out an intervention over time, whereby all participants are randomised into two groups (A and B) to receive the intervention at different time points determined at random. Baseline measurements are collected at month 0, and groups A and B are compared at months 6 and 12. The primary outcome is the family-prioritized Family Experiences with Coordination of Care (FECC) survey at 12 months. The FECC will be compared between groups using an analysis of covariance with the corresponding baseline score as the covariate. Secondary outcomes include reports of child and parent health outcomes, health system utilisation and process outcomes.Ethics and disseminationResearch ethics approval has been obtained for this multicentre RCT. This trial will assess the effect of a large population-level complex care intervention to determine whether dedicated key workers and coordinated care plans have an impact on improving service delivery and quality of life for CMC and their families.Trial registration numberNCT02928757.

To assess health outcomes of parents caring for children with chronic illnesses compared with parents of healthy children.We searched OvidSP MEDLINE, EBM Reviews-Cochrane Central Register of Controlled Trials, EMBASE, and EBSCOHost CINAHL through September 2019. Included were English-language studies reporting health conditions or mortality of parents of affected children compared with healthy controls.Of 12 181 screened publications, 26 met inclusion criteria. Eight studies reported on anxiety, 23 on depression, 1 on mortality, and 1 on cardiovascular disease. Parents of chronically ill children had greater anxiety (standardized mean difference 0.42; 95% CI 0.24-0.60; P .001) and depression scores (standardized mean difference 0.35; 95% CI 0.26-0.45; P .001) than parents of healthy children. Thirty-five percent of parents of affected children met cut-offs for clinical depression, compared with 19% in the control (relative risk 1.75; 95% CI 1.55-1.97). Fifty-seven percent of such parents met cut-offs for anxiety, compared with 38% in the control (relative risk 1.40; 95% CI 1.18-1.67). One study of mothers of children with congenital anomalies reported a greater mortality risk than a comparison (adjusted hazard ratio 1.22; 95% CI 1.15-1.29), and another reported that these mothers experience an increased risk of cardiovascular disease (adjusted hazard ratio 1.15; 95% CI 1.07-1.23).Parents of chronically ill children experience poorer mental health (more anxiety and depression), and mothers of those with congenital anomalies may have greater risk of cardiovascular disease and mortality than parents of unaffected children. Our findings suggest a need for targeted interventions to attenuate adverse parental caregiver health outcomes.PROSPERO CRD42018094657 (https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=94657).

BACKGROUND: Children with medical complexity (CMC) require specialized care from multiple health care providers across various settings; hospital, home and community. Communication and care coordination are essential to provide streamlined, accessible care. Currently a child’s executive medical summary, known as a care plan, is thought to be the gold standard in assisting with care coordination. However, there are many limitations regarding care plan use such as: lack of shared ownership, limited capability to update in real-time, and lack of universal access. Previous literature indicates that care plans should be cloud-based and comprehensive, allowing for more functions other than the executive medical summary alone. OBJECTIVES: To determine the requirements and design features that are needed (e.g. care plans and care maps) in a cloud-based care coordination platform in order to best serve parents and health care providers (HCP) of CMC. DESIGN/METHODS: 10 parents of CMC and 10 HCPs participated in phase one of a feasibility study aiming to design, test and implement a patient-facing care coordination platform. Participants were recruited through purposive sampling and provided informed consent. Participants were shown screen-shots of the virtual platform, had the various functionalities explained to them verbally and were interviewed throughout the process about what they were shown and their likes/dislikes using an open ended semi-structured interview guide. The interviews were audio recorded and transcribed verbatim with data analysis occurring simultaneously. Data was coded individually by three members of the research team who then met to review emerging codes and came to consensus on codes and emerging themes. RESULTS: Parents of CMC were enthusiastic about the prospect of having a cloud-based care plan that they would be able to update in real-time. Most HCPs were weary of allowing parents the ability to make changes to the care plans, suggesting various mechanisms that would increase their comfort. Parents and HCPs also provided various suggestions regarding content that should be added to the platform such as: additional care plan sub-headings and symptom trackers. CONCLUSION: Parents and HCPs provided many ways in which the patient-facing virtual care platform could be improved prior to usability testing. Care plans continue to be viewed as a valuable tool and other novel concepts for care provision emerged.

BACKGROUND: Medical technologies and technological advances have resulted in a growing number of children with medical complexity (CMC), many of whom would not have survived previously. Despite these advances CMC are still at high risk of morbidity and mortality during childhood. Advance Care Planning (ACP) is defined by the Canadian Paediatric Society as “the process of discussing life-sustaining treatments and establishing long-term care goals.” Currently the pediatric literature regarding ACP has been largely limited to the intensive care setting and the oncology population. There is a dearth of information focussing on ACP for CMC and that includes bereaved family caregiver’s views. Bereaved caregivers have the unique ability to reflect upon ACP through their child’s whole disease course and end of life experience. OBJECTIVES: To explore the ACP experiences of bereaved family caregivers of CMC who have experienced the entire illness trajectory, including their child’s death. DESIGN/METHODS: A qualitative approach was applied, allowing for in-depth data collection through semi-structured interviews. Purposive sampling was used to recruit bereaved caregivers of CMC until thematic saturation was reached. The interview guide was developed through expert consultation and was refined iteratively throughout the interviews. Questions assessed caregivers’ experiences with ACP discussions, their feelings about those conversations and their perceptions about whether ACP affected the end of life experience. Each participant provided written, informed consent and interviews were recorded and transcribed verbatim. Three research team members used content analysis to independently code the interviews. RESULTS: 13 bereaved caregivers of CMC completed 12 interviews ranging from 40–80 minutes in length. All caregivers of CMC had participated in ACP discussions and sometimes found them to be overwhelming and frustrating in the moment. However, all caregivers reported that they now understand and appreciate the importance of the discussions. Four major themes emerged from the data describing caregiver’s feelings and experiences regarding Advance Care Planning: 1) influencers of the ACP experience, 2) positive experiences, 3) negative experiences and 4) the influence of ACP on end of life. CONCLUSION: Bereaved caregivers provided a unique perspective, highlighting the importance and the benefits of ACP discussions. They also revealed various ways in which ACP conversations could be improved. These insights will be helpful in guiding educational tools for health care providers working with CMC in the future.

To describe the process of obtaining consensus of outcome priorities between families of children with medical complexity (CMC) and their healthcare providers (HCPs) for the purpose of evaluating changes to service delivery.The consensus of outcomes involved surveying families of CMC and HCPs and an in-person consensus meeting. Priorities were obtained from the survey using a stratified ranking approach ensuring equal representation among unequally sized subgroups. An in-person meeting was held using the survey results to inform Delphi voting.Families of CMC (n=40) and HCPs (n=74) responded to the survey. Consensus generated three main target areas (child health, family health, experience of care) covered by 15 specific outcomes needed to evaluate care. Differences between family and HCP perceptions of importance were found for child self-care, play, social skills, and recreation as well as emotional health (for both parent and child) outcomes.Families of CMC and HCPs identified common priorities for outcome evaluation of CMC initiatives. Outcomes that differ in importance between families of CMC and HCPs should be studied further.Families of children with medical complexity and their providers can reach consensus on important outcomes. Stratifying subgroups ensures diverse representation, which is important to outcome prioritization.CONSENSO EN LOS OBJETIVOS ENTRE LA FAMILIA Y LOS PROFESIONALES DE LA SALUD PARA NIÑOS CON COMPLEJIDAD MÉDICA: OBJETIVO: Describir el proceso para lograr un consenso sobre los objetivos prioritarios entre las familias de niños con complejidad médica (NCCM) y los profesionales de la salud (PS) con el fin de evaluar cambios en la prestación de servicios. METODO: La búsqueda del consenso en los objetivos involucró una encuesta a las familias de NCCM y PS y luego una reunión en persona para lograr un consenso. Se identificaron las prioridades en la encuesta utilizando una clasificación estratificada para garantizar una representación equitativa entre los subgrupos de tamaño desigual. Luego se llevó a cabo una reunión en persona, utilizando los resultados de la encuesta para crear una votación tipo Delphi. RESULTADOS: Las familias de NCCM (n = 40) y los PS (n = 74) respondieron a la encuesta. El consenso generó tres áreas principales (salud del niño/a, salud familiar, experiencia de atención) cubiertas por 15 resultados específicos necesarios para evaluar la atención. Se encontraron diferencias en la percepción de importancia de los objetivos entre la familia y los profesionales de la salud en las áreas del autocuidado del niño, el juego, las habilidades sociales y la recreación, así como los objetivos para la salud emocional (tanto para padres como para niños). INTERPRETACIÓN: Las familias de NCCM y los PS identificaron prioridades comunes para la evaluación de resultados de iniciativas para hacer cambios en la atención médica. Las diferencias entre los objetivos de las familias de NCCM y PS deben estudiarse con más detalle.RESULTADOS CONSENSUAIS ENTRE FAMÍLIA-PROFISSIONAL PARA CRIANÇAS COM COMPLEXIDADE MÉDICA: OBJETIVO: Descrever o processo de obter consenso dos resultados prioritários entre famílias de crianças com alta complexidade médica (ACM) e profissionais de saúde (PSs) para avaliar mudanças na prestação de serviços. MÉTODO: A obtenção de consenso dos resultados envolvidos foi obtida entrevistando ACMs e PSAs e por meio de um encontro pessoalmente. Prioridades foram obtidas a partir da entrevista usando uma pontuação estratificada assegurando representação igual entre grupos com tamanhos diferentes. Um encontro pessoalmente aconteceu usando os resultados da pesquisa para informar a votação Delphi. RESULTADOS: Famílias de AMCs (n=40) and PSs (n=74) responderam às questões. O consenso gerou três áreas principais (Criança, família, experiência de cuidado) cobertas por 15 resultados específicos necessários para avaliar o cuidado. Diferenças entre percepções da família e de PSs e as percepções de importância foram obtidas para auto-cuidado da criança, brincar, habilidades sociais, e recreação, assim como saúde emocional (para pais e filhos). INTERPRETAÇÃO: Famílias de AMCs e PSs identificaram prioridades comuns para avaliacão de resultados em iniciativas de ACM. Resulados que diferem em importância entre famílias de AMCs e PSs devem ser considerados em futuros estudos.

Grâce aux progrès de la médecine et aux innovations en technologies de la santé, de nombreux enfants atteints d’affections au pronostic réservé vivent désormais plus longtemps. Ces enfants sont souvent qualifiés d’« enfants ayant des problèmes médicaux complexes », parce qu’ils ont des affections chroniques, utilisent davantage les soins de santé et sont dépendants de la technologie. La complexité de leurs soins et leur fragilité intrinsèque entraînent des risques plus élevés d’erreurs de médication, autant dans un contexte ambulatoire qu’hospitalier. Le taux élevé de fragmentation des soins, de mauvaise communication et de polypharmacie chez les enfants ayant des problèmes médicaux complexes accroissent les possibilités d’erreurs, particulièrement au moment de changer de milieu de soins et de praticiens. Il n’existe pas de données sur les facteurs qui contribuent à un risque plus élevé d’erreurs de médication dans cette population et sur les meilleures mesures pour les éviter. Le présent point de pratique fournit des directives cliniques aux professionnels de la santé afin de s’assurer d’une utilisation sécuritaire des médicaments auprès des enfants ayant des problèmes médicaux complexes et s’attarde sur des stratégies pratiques applicables à la fois en soins ambulatoires et hospitaliers.

Key Points Question What is the diagnostic yield of genome sequencing in children with unexplained medical complexity and prior negative results of genetic testing? Findings In this cohort study that included 138 individuals from 49 families, genome sequencing detected all genomic variation previously identified by conventional genetic testing and resulted in a new diagnosis for 31% of patients. Meaning This study suggests that, because of its high yield, comprehensive nature, and increasingly competitive costs, genome sequencing is a potentially first-tier genetic test for children with unexplained medical complexity., Importance Children with medical complexity (CMC) represent a growing population in the pediatric health care system, with high resource use and associated health care costs. A genetic diagnosis can inform prognosis, anticipatory care, management, and reproductive planning. Conventional genetic testing strategies for CMC are often costly, time consuming, and ultimately unsuccessful. Objective To evaluate the analytical and clinical validity of genome sequencing as a comprehensive diagnostic genetic test for CMC. Design, Setting, and Participants In this cohort study of the prospective use of genome sequencing and comparison with standard-of-care genetic testing, CMC were recruited from May 1, 2017, to November 30, 2018, from a structured complex care program based at a tertiary care pediatric hospital in Toronto, Canada. Recruited CMC had at least 1 chronic condition, technology dependence (child is dependent at least part of each day on mechanical ventilators, and/or child requires prolonged intravenous administration of nutritional substances or drugs, and/or child is expected to have prolonged dependence on other device-based support), multiple subspecialist involvement, and substantial health care use. Review of the care plans for 545 CMC identified 143 suspected of having an undiagnosed genetic condition. Fifty-four families met inclusion criteria and were interested in participating, and 49 completed the study. Probands, similarly affected siblings, and biological parents were eligible for genome sequencing. Exposures Genome sequencing was performed using blood-derived DNA from probands and family members using established methods and a bioinformatics pipeline for clinical genome annotation. Main Outcomes and Measures The primary study outcome was the diagnostic yield of genome sequencing (proportion of CMC for whom the test result yielded a new diagnosis). Results Genome sequencing was performed for 138 individuals from 49 families of CMC (29 male and 20 female probands; mean [SD] age, 7.0 [4.5] years). Genome sequencing detected all genomic variation previously identified by conventional genetic testing. A total of 15 probands (30.6%; 95% CI 19.5%-44.6%) received a new primary molecular genetic diagnosis after genome sequencing. Three individuals had novel diseases and an additional 9 had either ultrarare genetic conditions or rare genetic conditions with atypical features. At least 11 families received diagnostic information that had clinical management implications beyond genetic and reproductive counseling. Conclusions and Relevance This study suggests that genome sequencing has high analytical and clinical validity and can result in new diagnoses in CMC even in the setting of extensive prior investigations. This clinical population may be enriched for ultrarare and novel genetic disorders. Genome sequencing is a potentially first-tier genetic test for CMC., This cohort study evaluates the analytical and clinical validity of genome sequencing as a comprehensive diagnostic genetic test for children with medical complexity.

Background Children with medical complexity (CMC) are defined by their medical fragility, dependence on assistive technology and substantial care needs. Family caregivers of CMC have unique challenges, such as prolonged hospitalizations and poor care coordination, which result in extensive caregiver stress. There is a great need to quantify the level of psychosocial stress and resilience in these caregivers to allow for appropriate allocation of health care resources. The Psychosocial Assessment Tool (PAT) is a brief parent-reported screening tool for measuring psychosocial risk in caregivers of pediatric populations. This is the first study to use the PAT in children with medical complexity. Objectives To quantify psychosocial risk in family caregivers of children diagnosed with medical complexity. To identify predictors of caregiver distress based on their sociodemographic factors. It was hypothesized that the extensive health demands of CMC will result in high amounts of chronic, ongoing caregiver distress relative to the previously studied pediatric populations. Design/Methods This cross-sectional study was conducted at The Hospital for Sick Children, Toronto, Canada. Family caregivers of children with medical complexity completed the PAT questionnaires during regularly scheduled Long-Term Ventilation and Complex Care clinic visits. Based on the overall PAT scores, caregivers were stratified as “Universal” low risk ( Results 136 [103 females (76%)] family caregivers completed the study. Mean PAT score was 1.17 (SD = 0.740). 61 (44.85%) caregivers were classified as Universal risk, 60 (44.12%) as Targeted risk, and 15 (11.03%) as Clinical risk. Compared to previously studied pediatric populations, our CMC have the second-highest overall PAT scores, which are also substantially weighted towards the higher risk categories (Table 1). Multiple linear regression analysis demonstrated that subjective report of financial hardship by caregivers is a significant predictor of total PAT scores (p < 0.05). Conclusion Family caregivers of children with medical complexity report PAT scores amongst the highest of all pediatric populations. These caregivers experience significant psychosocial distress, demonstrated by larger proportions of caregivers in the Targeted and Clinical risk categories. Therefore, psychosocial interventions including financial assistance are urgently needed in this population.

Key Points Question How do bereaved family caregivers describe their experiences with advance care planning for children with medical complexity? Findings In this qualitative study that included 13 parents of children with medical complexity, participants emphasized the importance of involving trusted health care professionals and incorporating parental expertise to guide advance care planning. The relative shock parents experienced regarding the timing of the child’s death, despite recurrent experiences with life-threatening events, and the multiple losses they experienced when the child with a large health care team died were important themes. Meaning The perspectives of bereaved parents in this study revealed important themes that should be considered for future study of advance care planning for children with medical complexity., This qualitative study explores the experiences of bereaved family caregivers with advance care planning for children with medical complexity., Importance Advance care planning (ACP) is the process of discussing values and preferences for care to help inform medical decision-making. Children with medical complexity (CMC) often have a shortened life span with an unpredictable clinical course and timing of death; however, there is a paucity of literature that describes the experience of ACP from the perspective of bereaved family caregivers of CMC. Objective To explore the experiences of bereaved family caregivers with ACP for CMC. Design, Setting, and Participants This qualitative study included 12 interviews with 13 bereaved family caregivers of CMC whose deaths had occurred in the 5 years before study commencement (2013-2018). Participants were recruited at a single tertiary care pediatric center; CMC were treated by the Complex Care or Long-term Ventilation clinic in Toronto, Ontario, Canada. Data were collected from July to October 2018. Thematic analysis with an inductive approach was used. Exposures Qualitative interviews were conducted using purposive sampling of bereaved family caregivers using semistructured interviews that were recorded and transcribed. Interviews were conducted until saturation was reached. Main Outcomes and Measures Transcripts were analyzed to create themes that characterized caregiver experiences with ACP. Results A total of 13 family caregivers were interviewed in 12 interviews, all of whom were parents (12 [92%] women, 1 [8%] man) of a deceased child (aged 7 months to 12 years). Themes were divided in the 3 following categories, which align with the Donabedian model for health service quality: (1) structure of care, (2) ACP process, and (3) end-of-life experience. Notable subthemes for this population included the importance of accounting for parental expertise in the child’s care, recurrent experiences with life-threatening events, relative shock of the timing of death, and the multiple losses that caregivers experienced. Conclusions and Relevance In this study, parental experiences revealed that there are key aspects of the structure of the child’s care, process around ACP, and end-of-life care experiences that provide important reflections on ACP that warrant future study.

ObjectiveTo quantify psychosocial risk in family caregivers of children with medical complexity using the Psychosocial Assessment Tool (PAT) and to investigate potential contributing sociodemographic factors.DesignCross-sectional study.SettingFamily caregivers completed questionnaires during long-term ventilation and complex care clinic visits at The Hospital for Sick Children, Toronto, Ontario, Canada.PatientsA total of 136 family caregivers of children with medical complexity completed the PAT questionnaires from 30 June 2017 through 23 August 2017.Main outcome measuresMean PAT scores in family caregivers of children with medical complexity. Caregivers were stratified as ‘Universal’ low risk, ‘Targeted’ intermediate risk or ‘Clinical’ high risk. The effect of sociodemographic variables on overall PAT scores was also examined using multiple linear regression analysis. Comparisons with previous paediatric studies were made using T-test statistics.Results136 (103 females (76%)) family caregivers completed the study. Mean PAT score was 1.17 (SD=0.74), indicative of ‘Targeted’ intermediate risk. Sixty-one (45%) caregivers were classified as Universal risk, 60 (44%) as Targeted risk and 15 (11%) as Clinical risk. Multiple linear regression analysis revealed an overall significant model (p=0.04); however, no particular sociodemographic factor was a significant predictor of total PAT scores.ConclusionFamily caregivers of children with medical complexity report PAT scores among the highest of all previously studied paediatric populations. These caregivers experience significant psychosocial risk, demonstrated by larger proportions of caregivers in the highest-risk Clinical category.

Purpose: RAC3 is an underexamined member of the Rho GTPase gene family that is expressed in the developing brain and linked to key cellular functions. De novo missense variants in the homolog RAC1 were recently associated with developmental disorders. In the RAC subfamily, transforming missense changes at certain shared residues have been observed in human cancers and previously characterized in experimental studies. The purpose of this study was to determine whether constitutional dysregulation of RAC3 is associated with human disease. Methods: We discovered a RAC3 variant in the index case using genome sequencing, and searched for additional variants using international data-sharing initiatives. Functional effects of the variants were assessed using a multifaceted approach generalizable to most clinical laboratory settings. Results: We rapidly identified five individuals with de novo monoallelic missense variants in RAC3, including one recurrent change. Every participant had severe intellectual disability and brain malformations. In silico protein modeling, and prior in vivo and in situ experiments, supported a transforming effect for each of the three different RAC3 variants. All variants were observed in databases of somatic variation in cancer. Conclusions: Missense variants in RAC3 cause a novel brain disorder, likely through a mechanism of constitutive protein activation.

BACKGROUND Genetic testing is often pursued in children with medical complexity (CMC), in an attempt to establish a unifying diagnosis, understand pathogenicity and disease progression, guide care and inform reproductive planning. CMC are defined by at least one chronic condition, technology dependence, multiple subspecialist involvement, and high healthcare utilization. Despite multiple efforts to confirm clinical suspicion of an underlying genetic condition, many remain undiagnosed. Whole genome sequencing (WGS) is becoming increasingly available as an informative diagnostic tool. The application of genomic technology to this population has the potential to increase the proportion of CMC for whom diagnoses are established, in an effort to reduce time and emotional burden of the diagnostic process, and reduce health care system costs. OBJECTIVES The main purpose of this study was to optimize the clinical implementation of state-of-the-art genome diagnostics for CMC, in terms of diagnostic yield. DESIGN/METHODS We conducted a prospective study using patients followed by the Complex Care program at a large urban tertiary care center. Research ethics board approval was obtained. Of 435 patients screened, 114 were eligible for inclusion as an underlying genetic condition was clinically suspected but not established to date by conventional genetic testing. To date, 21 participants were evaluated through a clinical genetic assessment, previous genetic testing review and peripheral blood-derived DNA sequence. A laboratory team identified candidate genetic variants associated with patients’ clinical symptoms, as well as other paediatric medically actionable variants. When found, these variants were validated as clinically significant by comparing the child’s DNA to his parents’. WGS diagnostic yield was then determined by calculating the proportion of cases for which a genetic diagnosis was established. RESULTS Of the 21 patients recruited, nine WGS analysis were completed thus far. Among these, four participants were diagnosed with established diseases, two of which were considered as novel diseases. One case was identified with a possible diagnosis, however, the interpretation of this clinical phenotype remains of unknown significance. The other four patients of the study remained undiagnosed. Given these preliminary results, the diagnostic yield of WGS was predicted at 44% in CMC. This can be compared to a previous study performed at our center in which the diagnostic rate for chromosomal microarray alone was reported to be 8% and microarray plus targeted gene sequencing 13%. CONCLUSION This study has shown WGS to be feasible and achieve a higher diagnostic yield in our complex care population. As detection rates improve and laboratory costs decrease overtime, WGS will undoubtedly become a more informative diagnostic tool, particularly in this population. Optimizing the application of this increasingly sophisticated genomic technology warrants further consideration.

BACKGROUND Children with medical complexity (CMC) represent a growing population in the paediatric healthcare system. CMCs’ multiple health needs, illness severity, and fragility lead to a high degree of caregiver burnout, parental employment loss, and other social and financial consequences. Healthcare providers must consider a holistic view of the family, including consideration of social determinants of health [SDOH]: socio-economic status, employment, quality of housing, availability of social support, and access to healthcare services. This study addresses a gap in current research by exploring how caring for a CMC exacerbates social inequalities, and how these challenges may be mitigated. OBJECTIVES 1. What are the SDOH impacting CMC and their families? 2. How do families report the impact of their child’s medical complexity on their social circumstances? DESIGN/METHODS A qualitative description approach was used to enable rich data collection through semi-structured interviews until thematic saturation was reached. Nine interviews were conducted with CMC caregivers who were chosen by purposeful sampling. The interview guide was developed by expert consultation and iteratively refined. Ethics approval and written consent were obtained. Interviews were recorded and transcribed verbatim. Three team members independently coded the interviews for recurrent themes to inform qualitative content analysis. RESULTS Caregiving for a CMC is all-consuming, requiring parents to take on roles including planner, medical professional, medical educator, and advocate. Parents of CMC report three major areas negatively impacted by caregiving: 1) physical and mental health; 2) personal relationships; and 3) finances. Additionally, three themes emerged describing enablers for resiliency: 1) the CMC’s health status when well or stable in hospital; 2) acceptance of one’s limitations as a caregiver and of the family’s ‘new normal’; and 3) broad supports including medical, personal, financial, and educational. CONCLUSION The wellbeing of CMC families is impacted by medical and social factors. An interdisciplinary model of care may offset some of the caregiver’s advocacy and medical educator responsibilities. Paediatricians can support families of CMC by providing comprehensive care for all components of the child’s health status, and by including routine assessment of the family’s SDOH. Finally, explicit discussion about parental expectations and caregiver burnout helps foster a positive therapeutic relationship with the family.

BACKGROUND Medical and technological advances have resulted in a growing cohort of children with medical complexity (CMC) who are reaching adulthood. These children are diagnostically heterogenous and require frequent hospitalizations, intensive community services, and medical technology at home to maintain their health. Transitioning CMC to the adult healthcare system has proven challenging for patients and their caregivers, who are comfortable and empowered in the paediatric setting. This multi-faceted process requires the expertise of an interdisciplinary team and one transition can take up to 100 hours of coordination. Although each patient is diagnostically unique, common requirements for transition arise. A standardized approach to transition would empower caregivers to be better advocates for CMC in the adult healthcare system. In order to implement an approach, a baseline understanding of this growing population is required. OBJECTIVES 1. To obtain demographic, medical, and transition-related data for CMC approaching transition to assess whether current transition practices are aligned with critical transition milestones and events, as detailed by key informants 2. To identify optimal targets for intervention in the transition process and produce a standardized toolkit to address gaps DESIGN/METHODS This was a cross-sectional chart review study. It was conducted at one of the largest Canadian complex care programs, which follows 400+ children. Prior to chart review, critical transition-related parameters and milestones were ascertained from interviews with key informants, such as transition coordination experts, the medical lead of the complex care program, family physicians, social workers, and nurse practitioners, who are the most involved with transitioning patients clinically. This previously siloed knowledge was centralized and converted into a standardized clinical toolkit including an age-stratified checklist and caregiver handouts. Having determined key transition parameters, 51 CMC were identified between the ages of 14 and 17 (inclusive) and were included in the study. Demographic, medical, and transition-related data was collected from care plans and clinical notes. Data was subsequently stratified by age and care location. Descriptive statistics were generated for each key transition parameter. RESULTS This cohort of 51 CMC (51% male, 49% female) displayed steady growth between years. On average, these CMC had 9 diagnoses, 6 subspecialists, actively used 8 medications, and were dependent on one home-technology. Only 50% of patients had discussed transition-related topics and 76% of patients did not have a designated transition lead. Although discussion of transition increased with age, many crucial transition-related topics, both in medical and social categories, were not routinely addressed. From a medical perspective, over 40% of patients had not found a family physician, who serve as the primary medical care coordinators in the adult healthcare system. In the final year before transition, less than 50% of patients had been referred to new adult subspecialists and of these patients, half had not met these new providers. Adult social support programs, which provide critical funding for medications, respite, and home care, represent the other major component of transition. At age 17, 50% of patients had not applied for any provincial disability and respite funding, which can take up to 2 years to receive approval for. CONCLUSION This study has identified areas for improvement in medical and social aspects of transition-related planning that may benefit from a standardized clinical toolkit. These are highly complex patients who are not meeting transition-related milestones before turning 18, and are entering the adult healthcare system unprepared. Without approved funding and established medical follow-up, patients and caregivers suffer. Due to the highly heterogenous nature of this patient population, a standardized approach to transition, in tandem with further research and education, is needed.

BACKGROUND Project ECHO® is an innovative, interactive model for medical education and healthcare delivery that expands access and capacity to provide evidence-informed care. This model virtually connects specialist interprofessional teams with community healthcare providers (HCPs). It aims to increase knowledge, build capacity, and ultimately, improve patient outcomes by bringing expertise to the community. This project focuses on the first paediatric implementation of Project ECHO for children with medical complexity. This is the first Project ECHO for this population. OBJECTIVES In preparation for this novel program launch in January 2018, a needs assessment was conducted with the objectives of: (1) informing the complex care-specific ECHO curriculum; and (2) assessing community partner preferences for program format and delivery modality. DESIGN/METHODS An online survey (38 items; 15-minutes) was distributed via targeted emails to professional networks, associations, and allied health organizations related to complex care throughout Ontario. The survey assessed interest in specific educational topics as well as program format preferences. An online survey and data management tool (REDCap) was used to build the survey and ensure data quality and security. RESULTS 49 HCPs completed the survey for Paediatric Project ECHO for Children with Medical Complexity. Profession breakdown was: 29% paediatricians, 19% dieticians, 16% nurses, 14% nurse practitioners with the remaining being allied health care professionals, including occupational therapists, social workers and psychologists. Overall, 39% of participants practiced in academic hospitals, 27% in non-academic hospitals, and 35% in other community settings. In terms of program format, 63% preferred weekly 1- or 2-hour educational sessions versus longer sessions. Top medical topics rated by HCPs were: management of nutrition, pain or irritability of unknown origin, and challenging behaviours in children with complex care needs. Other topics of interest included: developmental review, transition to adult care, advanced care planning, funding opportunities, and social supports for the patient as well as caregivers and siblings. CONCLUSION HCPs who expressed interest in Paediatric Project ECHO for Children with Medical Complexity are generally experienced professionals who preferred short educational sessions. The themes that emerged from this needs assessment spanned the biological, psychological and social domains of management of complex care patients. Survey results are being used to inform the development of an evidence-based paediatric complex care curriculum that meets the needs of community HCPs. This project is funded by the MOHLTC.

BACKGROUND Children with medical complexity (CMC), are defined by at least one chronic condition, functional limitation requiring technology support, multiple subspecialist involvement, and high healthcare utilization. A potentially significant social determinant that has yet to be examined in this population is that of “housing need.” According to the Canada Mortgage and Housing Corporation, “housing need” refers to housing that is crowded, unaffordable, or in need of major repairs. Given the known associations between housing and health, we hypothesize that housing need could be particularly relevant in CMC. Population-specific housing considerations may include in-home medical services, housing space, and disability accommodations. OBJECTIVES 1) Determine the prevalence of housing need in families with CMC and 2) Explore the experience and meaning of housing need of caregivers of CMC. DESIGN/METHODS We conducted a mixed methods study using questionnaires and semi-structured interviews. Recruitment occurred through a tertiary paediatric hospital. Research ethics board approval was obtained. All participants were English-speaking primary caregivers of CMC living in the same household. Questionnaires were completed in person, by phone, or online via REDCap, a secure data collection application. Questionnaires explored safety, affordability, and accommodations and were analyzed using descriptive statistics. Following the questionnaire, participants were invited to be interviewed in person or by phone. Interview questions were developed iteratively and were analyzed using grounded theory. RESULTS Of the 354 eligible caregivers, 93 participated in the questionnaire. A total of 36 caregivers (40%) reported some difficulties paying for housing each month. In addition, 56 participants (62%) described unmet need for accommodations (i.e. a lift, toilet accommodations, etc.). A total of 65 participants (83%) reported that their child’s condition(s) affected their preferred housing location, while 55 participants (70.5%) felt it affected their preferred housing type. Of the fifteen interviews completed, several themes emerged including limited housing options for families with CMC and financial, physical, and mental health consequences of housing challenges. In addition, caregivers felt counseling was lacking on 1) safe evacuation and 2) housing requirements, such as space and equipment, as children age. CONCLUSION This is the first in-depth exploration of housing need amongst families of CMC. Challenges identified include affordability, disability accommodations, and restrictions on housing type and location due to children’s illnesses. Clinicians can support families of CMC by offering counseling on evacuation planning and common housing requirements for CMC.

BACKGROUND: Caring for a child with a chronic medical condition can adversely affect parental health and well-being. Family-centered care requires clinical teams to consider novel ways of providing holistic care to the entire family by understanding the unique challenges of caring for a child with a chronic medical condition. Addressing parental well-being not only improves parental capacity to care for their child, but may also optimize long-term health outcomes for the child themselves. Acceptance and Commitment Therapy (ACT) is an established behavioural therapy that has been used with caregivers in the paediatric setting. Through ACT, individuals learn skills to increase psychological flexibility, cognitive diffusion, and mindfulness. ACT aims to help with coping and adjustment, but its impact has not been systematically appraised in parents of children with chronic medical conditions. OBJECTIVES: To synthesize available research evidence on the efficacy of ACT for parents of children with medical conditions. DESIGN/METHODS: A comprehensive literature search (December 2008 to October 2018) in PubMed, Cochrane Library, EMBASE, Web of Science, and Google Scholar was conducted. Abstracts were included if: 1) they were original articles and 2) they assessed the efficacy and outcomes of ACT on parents caring for children with chronic medical conditions. RESULTS: In total, 66 abstracts were identified. Nine publications describing ACT as an intervention aimed at improving parent-oriented outcomes met the final inclusion criteria. Five studies were randomized control trials. Medical conditions included children with: chronic pain (n=4), acquired brain injuries (n=2), autism spectrum disorder (n=1), cerebral palsy (n=1), and life-threatening illness (cancer or life-saving cardiac surgery) (n=1). All nine studies found improvements in caregiver related outcomes following ACT treatment. Improvements were seen in psychological flexibility (n=5), family functioning (n=3), parenting (n=1), and depression (n=2). Significant, medium to large effect sizes were found for measures of psychological flexibility, dysfunctional parenting styles (laxness and over reactivity). A medium effect size was found for family functioning. CONCLUSION: ACT improved outcomes for parents caring for children with chronic medical conditions. The majority of studies concentrated in paediatric patients with chronic pain or neurologic conditions. Less is known about the efficacy of ACT in improving parent-related outcomes in other paediatric disease groups however, further research in this area is warranted.

OBJECTIVE: To compare health care use and costs for youth with chronic health conditions before and after transfer from pediatric to adult health care services. METHODS: Youth born in Ontario, Canada, between April 1, 1989, and April 1, 1993, were assigned to 11 mutually exclusive, hierarchically arranged clinical groupings, including “complex” chronic conditions (CCCs), non-complex chronic conditions (N-CCCs), and chronic mental health conditions (CMHCs). Outcomes were compared between 2-year periods before and after transfer of pediatric services, the subjects’ 18th birthday. RESULTS: Among 104 497 youth, mortality was highest in those with CCCs, but did not increase after transfer (1.3% vs 1.5%, P = .55). Costs were highest among youth with CCCs and decreased after transfer (before and after median [interquartile range]: $4626 [1253–21 435] vs $3733 [950–16 841], P < .001);Costs increased slightly for N-CCCs ($569 [263–1246] vs $589 [262–1333], P < .001), and decreased for CMHCs ($1774 [659–5977] vs $1545 [529–5128], P < .001). Emergency department visits increased only among youth with N-CCCs (P < .001). High-acuity emergency department visits increased CCCs (P = .04) and N-CCCs (P < .001), but not for CMHC (P = .59), who had the highest visit rate. Among the 11 individual conditions, costs only increased in youth with asthma (P < .001), and decreased (P < .05) in those with neurologic impairment, lupus, inflammatory bowel disease, and mood/affective disorders. CONCLUSIONS: Pediatric transfer to adult care is characterized by relatively stable short-term patterns of health service use and costs among youth with chronic conditions.