Correlation of sibling couple infertility history with semen parameters of infertile men, a cohort study.

INTRODUCTION: Infertility is a growing issue globally, particularly in industrialized cultures, affecting 13%-18% of couples of reproductive ages. In recent years, numerous studies have aimed to identify prognostic factors for infertility and abnormal semen analysis. To date, no study has examined the relationship between the number of infertile siblings and abnormal sperm parameters. This study aims to investigate whether the number of infertile siblings can be considered a prognostic factor for abnormal sperm parameters. METHOD: Semen samples were collected from the male partners of couples experiencing infertility issues. Study participants completed a questionnaire detailing demographic information including age and family history of infertility. Each participant provided two semen samples, with a minimum 15-day interval between collections. Sperm concentration, motility, and morphology were assessed for each sample. Clinical investigators conducted physical examinations, using an orchidometer to measure testicular size. RESULTS: The number of infertile brothers and testis volume were prognostic factors for abnormal sperm count (OR = 1.374, p-value = 0.03; OR = 0.786, p-value < 0.001; respectively) and abnormal motility (OR = 1.514, p-value = 0.018; OR = 26.74, p-value < 0.001, respectively). There was no significant association between the percentage of abnormal morphology of sperm cells and the mentioned prognostic factors. The optimal cut-off point of the number of infertile brothers for both abnormal sperm count and abnormal sperm motility was one. CONCLUSION: It is recommended that males with at least one infertile brother, undergo sperm analysis to identify individuals at risk of infertility.

Evaluation of early treatment with intravenous idursulfase and intrathecal idursulfase-IT on cognitive function in siblings with neuronopathic mucopolysaccharidosis II.

Mucopolysaccharidosis II (MPS II; Hunter syndrome; OMIM 309900) is a rare, X-linked, heterogeneous lysosomal storage disease. Approximately two-thirds of patients develop cognitive impairment, which is difficult to assess in clinical trials, partly owing to the variable nature of cognitive impairment. Analyzing data from siblings can help to minimize this heterogeneity. We report analyses of cognitive function from siblings with MPS II enrolled in clinical trials: a natural history study (NCT01822184), a randomized, open-label, phase 2/3 study of intravenous (IV) idursulfase with or without intrathecal idursulfase (idursulfase-IT; NCT02055118), and its extension (NCT2412787). Cognitive function was assessed using Differential Abilities Scales, Second Edition General Conceptual Ability (DAS-II GCA) scores; Bayley Scales of Infant and Toddler Development, Third Edition; and Vineland Adaptive Behavior Scales, Second Edition Adaptive Behavior Composite (VABS-II ABC). Seven sets of siblings (six pairs and one set of three) were included. All patients received IV idursulfase and 10 received subsequent idursulfase-IT. Younger siblings initiated IV idursulfase at an earlier age than their older sibling(s) in six of the sets; the younger sibling started treatment before 1 year of age in three sets. Monthly idursulfase-IT was generally associated with a stabilization of cognitive function: DAS-II GCA and VABS-II ABC scores were higher at age-matched assessments in the majority of those who either received idursulfase-IT earlier than their sibling or who received idursulfase-IT versus no idursulfase-IT. These data suggest that early initiation of intrathecal enzyme replacement therapy may stabilize or slow cognitive decline in some patients with neuronopathic MPS II.

Supporting siblings during the critical illness hospitalization of a child: learning from experience.

Introduction: Childhood critical illness impacts the entire family of the critically ill patient. Disruptions to usual family rhythms and routines, established relationships, physical relocations or shifts in caregivers, and the uncertainty about the patient's well-being can have significant impacts on siblings and other connected children in the family. Promoting and facilitating family interactions and engaging younger family members in the hospital experience have been shown to reduce patient and family anxiety, enhance family adaptation, and improve child and family outcomes. The critical care team can implement evidence-informed approaches to address and mitigate challenges for families and provide developmentally aligned support to impacted siblings. Aim: This conceptual paper describes the potential impacts of a critical illness hospitalization on siblings, approaches to supporting siblings, and practical interventions drawn from a synthesis of the current literature and the author's practice experience caring for critically ill children and their families. Data sources: A traditional review and narrative analysis moderated by the authors and supported by lived experience. Conclusions: There is a range of impacts of a critical illness hospitalization on siblings and young family members of the patient. Providing consistent, transparent, and supportive child, sibling, and whole family-centered care can improve the experience and outcomes for the child and family.

'I have to remind myself that everyone's search is different': experiences and outcomes of searching and not searching for donor connections among donor conceived adults.

STUDY QUESTION: What are the experiences and outcomes of donor conceived adults who are actively searching for, open to contact with, or not searching for donor connections? SUMMARY ANSWER: Most participants were actively searching or open to contact, and 67% had found or been found by a connection; finding or not finding experiences were complex. WHAT IS KNOWN ALREADY: There is variation among donor conceived individuals in their interest in donor connections. Individual reasons for searching for connections, and which donor connections are searched for, also vary. Most research studies have focussed on individuals who are actively searching for their donor or donor siblings. Global increases in direct-to-consumer DNA testing and social media participation mean that connections may be made to individuals unaware of their (or their relatives') involvement with donor conception. These social and technological changes have also increased the chances of donor conceived individuals being contacted without expecting or desiring contact. STUDY DESIGN, SIZE, DURATION: This study included 88 donor conceived adults, in the UK, who participated in an online multi-method survey between January and August 2022. The survey was designed in consultation with staff and volunteers from the UK's largest community networks for donor conception families (Donor Conception Network, DCN) and donor conceived people (Donor Conceived Register Registrants' Panel, DCRRP). It was piloted by five donor conceived people before its launch. Participants were recruited with assistance from DCN and DCRRP, via social media, university mailing lists, and snowballing. PARTICIPANTS/MATERIALS, SETTING, METHODS: Participants were mostly female (n = 65, 74%) and sperm donor conceived (n = 79, 90%). Of the 88 participants, 39 (44%) were actively searching for their donor connections, 44 (50%) were open to contact but not actively searching, and 5 (6%) were not searching. Questions were closed (yes/no, rating scale, or multiple choice) or open-ended, addressing experiences of donor conception, searching for connections, and finding or not finding connections. Data were analysed both quantitatively and qualitatively. MAIN RESULTS AND THE ROLE OF CHANCE: Quantitative results showed no differences between the groups on any demographic variables or in when or how they found out about being donor conceived, and no differences between active searchers and those open to contact in whether they had found their donor connections. Significant differences were found between groups in their interest in their genetic history and the perceived importance of genetics to their sense of identity, with active searchers being more interested and rating this as more important than those open to contact. Methods of searching significantly differed across groups, with active searchers using genetic testing and social media more than those open to contact. 59 participants across all groups (active searchers (n = 29, 74%), open to contact (n = 27, 61%), not open to contact (n = 3, 60%)) had found or been found by a donor connection. Experiences of finding or not finding donor connections among participants actively searching or open to contact were captured by the theme complexities, with six subthemes: uncertainties in searching and relating; searching as open-ended; different donor connections, different experiences; expectations and realities; searching and finding or not finding as catalysing change; and experiences of other donor conceived people. LIMITATIONS, REASONS FOR CAUTION: Most participants were members of relevant community organizations. As is common in research in this area, the sample was mostly female and conceived using donor sperm. Donor conceived people who are disinterested in donor connections may be unlikely to participate in research on this topic. WIDER IMPLICATIONS OF THE FINDINGS: The nature and impact of the search process itself should be considered when developing appropriate mechanisms of support for all donor conceived people, regardless of whether they are actively searching for connections or not. Further research should seek to better understand how donor conceived people with varying levels of interest in searching for donor connections differ from one another. STUDY FUNDING/COMPETING INTEREST(S): This study was funded by the UK Economic and Social Research Council [New Investigator Award ES/S015426/1]. The authors have no competing interests to declare. TRIAL REGISTRATION NUMBER: N/A.

Mismatch Negativity and P3a in Unaffected Siblings of Individuals with Autism Spectrum Disorder and the Exploration on the Neurocognitive Implications.

Evidence suggests different mismatch negativity (MMN) and P3a responses in individuals with autism spectrum disorder (ASD). Since unaffected siblings shared aberrant neurocognition and brain connectivity with ASD probands, this study investigated MMN and P3a responses in unaffected siblings and explored its neurocognitive implications and effects modifiers. We assessed 43 unaffected siblings of ASD probands and 64 non-autistic comparisons (NTC) using MMN and P3a on both frequency and duration oddball paradigms. The amplitude and latency of MMN and P3a were compared between unaffected siblings and NTC, and validated in 67 ASD probands. In addition, the neurocognitive correlates of MMN and P3a parameters were explored in attention performance, spatial working memory (SWM), and visual research via the tasks of the Conners' Continuous Performance Test and the Cambridge Neuropsychological Test Automated Battery. Compared to NTC, unaffected siblings and ASD probands presented a shorter MMN latency. The P3a amplitude of the duration paradigm (dP3a) was correlated with fewer commission errors, fewer SWM total errors, higher detectability, and more correct responses on visual search tasks. In addition, the dP3a amplitude significantly interacted with sibship, age, and full-scale IQ to predict attention performance, SWM total errors, and total correct response on visual search. Findings suggest that unaffected siblings of ASD may have earlier brain responses upon novelty discrimination. P3a amplitude may correlate with better neurocognitive performance, but the effect was moderated by sibship, age, and intelligence.

Shared deficits of education, marital and occupational functioning in unaffected siblings of multiple affected families with major psychiatric illness.

BACKGROUND: Major psychiatric illnesses often cluster in families, and their impact on affected and unaffected members within families may reflect the consequence of both genetic and social liability. METHODS: Data was derived from 202 families with multiple affected individuals. Affected individuals (N = 259) had a diagnosis of schizophrenia, bipolar disorder, obsessive-compulsive disorder or substance use disorder. For comparison, we used the unaffected siblings from the same families (N = 229) and a matched random subset of healthy control (HC) data (N = 229) from India's National Mental Health Survey, 2016 (NMHS). We compared the three groups' educational attainment, functional marital status, and occupational status. RESULTS: The highest educational attainment was significantly different between the groups. The affected and unaffected siblings had poorer educational attainment compared to HC. Similarly, the affected and unaffected siblings more often remained single, in contrast to HC. Moreover, employment rates were significantly higher in the unaffected siblings, especially female siblings. Overall, females had spent fewer years at school, were primarily married, and were majority homemakers across the three groups compared to males. DISCUSSION: Affected and unaffected siblings had lower education and marriage rates than HC. The unaffected siblings were more likely to be employed than HC. Whether the poor educational attainment and lower marriage rates in unaffected siblings is a biological marker of shared endophenotype or the effect of the social burden of having an affected family member requires further systematic evaluation.

A Late Diagnosis of Andersen-Tawil Syndrome in Teenage Siblings.

BACKGROUND: Andersen-Tawil syndrome (ATS) is a rare autosomal dominant disorder characterized by a classic symptom triad, including periodic paralysis, ventricular arrhythmias with associated prolonged QT interval and U waves, and dysmorphic facial and skeletal features. Pathogenic variants of the KCNJ2 gene are linked to ATS. METHODS: We present two siblings with the same pathogenic mutation and facial characteristic of hypotelorism, yet with intrafamilial and sex-specific variability. RESULTS: The first patient is a 16-year-old male who presented from an outside hospital with subacute-onset weakness. The symptoms almost completely subsided the following day, with only mild proximal muscle weakness. Magnetic resonance imaging of the brain and cervical spine was unremarkable. He had one prior attack of self-resolving weakness without apparent triggering factors and a history of premature ventricular contractions and U waves seen on electrocardiogram without cardiac symptoms. On further evaluation his physical examination was significant for micrognathia, hypotelorism, and clinodactyly. Electrodiagnostic examination showed no clear evidence of polyneuropathy. Given his presentation of the typical triad of periodic weakness, dysmorphic features, and cardiac rhythm abnormalities, genetic testing was pursued revealing a pathogenic mutation of the KCNJ2 gene, indicative of ATS. Subsequent genetic testing of his older biological sister, with identical physical features but without a history of cardiac symptoms or episodic periodic paralysis, revealed the same pathogenic mutation. CONCLUSIONS: It is essential to note that ATS can manifest with a wide range of symptoms and some individuals may display only subtle or atypical signs, contributing to this challenging diagnosis.

Prevalence of mental disorders among family members of individuals on the autism spectrum: systematic review and meta-analysis.

Parenting a child on the autism spectrum presents particular challenges that can lead to increased stress, anxiety, and depression among family members. Therefore, we aimed to investigate the prevalence of mental disorders in first-degree relatives of individuals on the autism spectrum. This article adheres to the Preferred Reporting Items for Systematic Review and Meta-Analyses Protocols (PRISMA-P) guidelines, including studies indexed in PubMed/Medline, Embase, PsycINFO, Biblioteca Virtual em Saúde (BVS), and SciELO. Nineteen articles met eligibility criteria for the systematic review. Using a random-effects model (N = 93,876), we found a pooled prevalence of affective disorders of 13% in mothers of people on the autism spectrum (95% CI 7-21%; I2 = 99%, p < 0.01). Additionally, another random-effects model pointed out that first-degree relatives of people on the autism spectrum (N = 93,263) were more likely to present affective disorders than relatives of people with neurotypical development (N = 152,455) (pooled OR: 2.17; 95% CI 1.81-2.61). Careful assessment for mental disorders in parents and siblings of individuals on the autism spectrum is crucial to ensure appropriate treatment for these family members. This approach can also contribute to optimizing care for the individuals on the autism spectrum.

Parent-mediated intervention in infants with an elevated likelihood for autism reduces dwell time during a gaze-following task.

Cognitive markers may in theory be more sensitive to the effects of intervention than overt behavioral measures. The current study tests the impact of the Intervention with the British Autism Study of Infant Siblings-Video Interaction for Promoting Positive Parenting (iBASIS-VIPP) on an eye-tracking measure of social attention: dwell time to the referred object in a gaze following task. The original two-site, two-arm, assessor-blinded randomized controlled trial (RCT) of this intervention to increase parental awareness, and responsiveness to their infant, was run with infants who have an elevated familial likelihood for autism (EL). Fifty-four EL infants (28 iBASIS-VIPP intervention, 26 no intervention) were enrolled, and the intervention took place between 9 months (baseline) and 15 months (endpoint), with gaze following behavior measured at 15 months. Secondary intention to treat (ITT) analysis showed that the intervention was associated with significantly reduced dwell time to the referent of another person's gaze (ß = -0.32, SE = 0.14, p = 0.03) at 15-month treatment endpoint. Given the established link between gaze following and language, the results are considered in the context of a previously reported, non-significant and transient trend toward lower language scores at the treatment endpoint (Green et al. (2015) The Lancet Psychiatry, 2(2), 133-140). Future intervention trials should aim to include experimental cognitive measures, alongside behavioral measures, to investigate mechanisms associated with intervention effects.

Predictors of the quality of life in siblings of children with chronic diseases.

PURPOSE: Advances in medical sciences have contributed to the increase in children living with chronic diseases. The diagnosis of a chronic disease in a child can affect the quality of life of all family members, including siblings. Therefore, this study aimed to identify the predictors of quality of life among siblings aged 8 to 12 years who have a brother or sister with chronic diseases. METHODS: In this case-control study conducted in Rasht, 85 siblings of children with chronic diseases as the case group and 172 siblings of healthy children as the control group were recruited using cluster sampling. The two groups were matched for age and gender. Data were collected using the Pediatric Quality of Life Inventory (PedsQL TM) and a demographic questionnaire. The collected data were analyzed using Mann-Whitney U, Kruskal-Wallis, and logistic regression analysis tests via SPSS version 16. RESULTS: The mean quality of life scores in the case and control groups were 88.86 ± 10.33 and 87.39 ± 10.89, respectively, indicating a desirable level within both groups. No statistically significant difference was found in this regard between the two groups. Based on the logistic regression analysis, three variables of number of family members (P = 0.03), mother's education level (P = 0.025), and family's income level (P = 0.023) were identified as significant predictors of quality of life. In this regard, it was found that the number of family members and the mother's education level were positively related to the quality of life, increasing the mean scores by 2.32 and 1.48, respectively. Conversely, a higher family income level was associated with a decrease in quality of life scores by 0.543. CONCLUSIONS: The study found that the quality of life among siblings of children with chronic diseases is good, comparable to that of their peers. The findings imply that the chronicity of the disease does not significantly impair siblings' adaptation and overall well-being. PRACTICE IMPLICATIONS: The findings of this study can be used in various fields, including healthcare management, nursing clinical services, nursing education, and nursing research.

Effects of a behavioral intervention with foster families: A randomized controlled trial.

BACKGROUND: The health and well-being of children in foster care are of high concern. A resource with which to disrupt maladaptation and promote healing are positive relationships among children and caregivers within the foster home. The research question was: Can an online intervention improve family hardiness and sibling relationships within foster care families? OBJECTIVES: To: (1) establish feasibility and acceptability of an online behavioral intervention within the foster family, (2) explore the effects of the intervention on relational quality outcomes, and (3) decompose the mechanisms driving improved family hardiness through mediation analysis. PARTICIPANTS AND SETTING: 95 currently fostering families across the US, participated in a 4-week, online, self-paced, behavioral intervention with an emphasis on the relationship between children residing in the home. METHODS: We employed a randomized control trial design with multiple regression analysis. Stress and relational quality outcomes were measured through psychometrically validated questionnaires on family hardiness, preparedness, and sibling relationships. RESULTS: Significant increase in family hardiness (Cohen's d = 0.97, p < 0.001) were found compared to the control group. Measures of a positive sibling relationship score increased significantly in the intervention group (d = 0.76, p < 0.002), mediating 32 % of the total effect in hardiness score. Sibling relationship served as a mediator for increasing indicators of family hardiness. CONCLUSIONS: Providing families the sibling-inclusive intervention caused an increase in positive indicators of sibling relationship and overall family hardiness. Researchers should consider the implications of supporting foster siblings and the affects their inclusion may have on outcomes for children in foster care.

Siblings at home: Moderating influence of sibling presence and composition on the relation between problem behaviors and Theory of Mind in early childhood.

Despite the prevalence of internalizing and externalizing difficulties in children, the impact of these problem behaviors on the development of important social cognitive skills, such as Theory of Mind (ToM), is not well-understood. Indeed, many studies that have explored relations between problem behaviors and ToM report inconsistent findings. A possible reason for these disparities may be a lack of accounting for social protective factors within the home, such as the presence and number of siblings. Here, we explored the moderating influence of sibling presence and number on the relation between problem behaviors (i.e., internalizing and externalizing) and ToM. A total of 184 children (88 boys; Mage = 64.6 months, SD = 10.39) completed six well-validated ToM tasks while mothers reported on their children's externalizing and internalizing behaviors. Children who had siblings living in the same home exhibited higher ToM than children without siblings. In addition, both sibling presence and number of siblings moderated the relation between children's externalizing behaviors and ToM, such that in children without siblings externalizing behaviors were negatively associated with ToM. In contrast, children with siblings had similar ToM regardless of externalizing behaviors. As well, children with relatively fewer siblings and higher externalizing behaviors displayed lower ToM than children with relatively more siblings and higher externalizing behaviors. We did not detect a moderating effect of sibling presence or number on the relation between internalizing behaviors and ToM. These findings provide support for siblings' protective utility within the context of children's social cognition.

Experiences of siblings and parents of children with congenital heart disease and exploration of siblings' support needs.

Research with siblings of children with congenital heart disease (CHD) is scarce, although more than one-third of them experience limitations on their quality of life. This interview study aims to explore the diagnosis-associated experience of German siblings of children with CHD, their interest in a potential intervention, and potential key topics and contextual conditions of such an intervention. Interviews with 10 siblings aged 10 to 21 and a respective parent were conducted from August to October 2021, resulting in 20 interviews. Negative experiences associated with CHD included concerns regarding hospitalization, health deterioration, and the death of the child with CHD, as well as burdens including reduced family activities, less parental attention and support, and extended family meals. Positive experiences included perceived positive consequences of CHD, such as strong family cohesion and empathy toward people with chronic illnesses. Furthermore, siblings experienced enhanced coping mechanisms, such as having conversations with friends and family about the high prevalence of CHD and successful treatment or using distractions such as entertainment or study. Siblings' reported interest in a future intervention included empathy, peer support, and studying medical information on CHD. These findings should be used for counseling and developing tailored interventions to support these siblings.

Two brothers with human papillomavirus-positive oropharyngeal squamous cell carcinoma of the tonsil: A case report.

Oropharyngeal squamous cell carcinoma is a distinct subtype of head and neck cancer that has become increasingly linked to human papillomavirus over the last four decades. Described is the case of two brothers diagnosed with human papillomavirus-positive oropharyngeal squamous cell carcinoma 6 years apart. The first brother, R.M., presented with an 8-month history of tonsillar swelling, found to be stage III human papillomavirus-positive oropharyngeal squamous cell carcinoma. Despite delayed treatment with chemoradiation, he developed metastatic disease and succumbed to his illness. The second brother, K.M., presented only 3 weeks after the development of neck swelling given his family history, which was also diagnosed as stage III human papillomavirus-positive oropharyngeal squamous cell carcinoma. Following prompt chemoradiation and neck dissection, K.M. has remained in remission for 9 years. Literature has yet to characterize this degree of familial clustering among human papillomavirus-positive oropharyngeal squamous cell carcinomas. Hence, this introduces the possibility of a genetic predisposition to human papillomavirus's oncogenesis in the oropharynx. This case emphasizes the importance for clinicians to stay vigilant of the family history of human papillomavirus, as well as poses significant implications for future research investigating the interaction of genetic aberrations on human papillomavirus's oncogenic process.

"Mend the rift" therapeutic model for working with sibling sexual abuse: Professionals' perspectives.

BACKGROUND: Sibling sexual abuse (SSA) is considered the most prevalent and longest-lasting type of interfamilial sexual abuse. The psychological implications of SSA may be felt throughout the harmed siblings' lifespan. Nevertheless, SSA receives very little therapeutic attention. OBJECTIVE: The present study explores how professionals dealing with the phenomenon see how to work with such cases. PARTICIPANTS AND SETTING: Fifty-two professionals working in the field of SSA participated in the study and completed closed and open-ended questionnaires. METHODS: A mixed-method research approach was implemented to quantitatively and qualitatively analyze the professionals' responses. RESULTS: The findings addressed three main intervention aspects. The first comprised the key unique therapeutic themes of SSA interventions that differentiate the SSA therapeutic process from other child sexual abuse cases. These included complex familial dynamics (e.g., secrecy climate, loyalty conflict, and intergenerational transmission of trauma) and at-home risk. The second addressed the therapeutic goals, including rebuilding family relationships and promoting safety strategies at home. The third was the SSA intervention structure, defining the structural elements of a holistic therapeutic model for family care. CONCLUSIONS: Based on the findings, a therapeutic model for family intervention in SSA cases was proposed, aiming to define service characteristics, determine who should be involved in the therapeutic process, and identify who should lead the intervention. The research contributes to the advancement of the theoretical and practical knowledge necessary to address the inherent familial complexities in SSA cases and the enhancement of the therapeutic orientation for professionals involved in such cases.

Familial Spontaneous Keloids: Examining Thoracic Manifestations in Two Brothers.

Keloids are complex fibroproliferative disorders with diverse clinical presentations. Spontaneous keloids (SKs) represent a rare subtype that emerges without any known preceding traumatic event. This report presents a case of familial spontaneous keloids appearing on the thoracic region in two brothers with no prior history of trauma or keloid occurrence in other family members. The lesions exhibited progressive growth over several years but responded to cycles of triamcinolone treatment. This case underscores an unusual spontaneous occurrence of keloids in the thoracic region of two siblings, highlighting the potential genetic predisposition in the aetiology of these lesions. Additionally, this instance reinforces the concept that keloids can develop spontaneously without any apparent trauma in the affected area.

All-cause mortality and cause-specific death in U.S. long-lived siblings: data from the Long Life Family Study.

BACKGROUND: This study compared the mortality risk of long-lived siblings with the U.S. population average and their spouse controls, and investigated the leading causes of death and the familial effect in death pattern. METHODS: In the Long Life Family Study (LLFS), 1,264 proband siblings (Mean age 90.1, SD 6.4) and 172 spouses (83.8, 7.2) from 511 U.S.-based families were recruited and followed over 12 years. Their survival function was compared with a birth cohort-, baseline age-, sex-, and race-matched pseudo sample from U.S. census data. To examine underlying and contributing causes, we examined in detail 338 deaths with complete death adjudication at the University of Pittsburgh Field Center through the year 2018. A familial effect on survival and death pattern was examined using mixed effect models. RESULTS: The LLFS siblings had better survival than the matched U.S. population average. They also had slightly but not significantly better survival than their spouses' (HR=1.18 [95%CI 0.94-1.49]) after adjusting for age and sex. Age at death ranged from 75-104 years, mean 91.4. The leading causes of death were cardiovascular disease (33.1%), dementia (22.2%), and cancer (10.7%). Mixed effect model shows a significant random effect of family in survival, with adjustment of baseline age and sex. There was no significant familial effect in the underlying cause of death or conditions directly contributing to death among siblings recruited by the University of Pittsburgh Field Center. CONCLUSION: Our findings demonstrate a higher survival in the LLFS siblings than the U.S. census data, with a familial component of survival. We did not find significant correspondence in causes of death between siblings within families.

Examining emotional and behavioural trajectories in siblings of children with life-limiting conditions.

BACKGROUND: Healthy siblings of children with life-limiting conditions often experience emotional and behavioural struggles over the course of the ill child's condition(s). Resources to support these siblings are limited due to a lack of understanding about their needs. Therefore, this study was designed to characterize the emotional and behavioural trajectories among siblings of children with progressive, life-limiting genetic, metabolic, or neurological conditions over a 12-month observation period. METHODS: Seventy siblings were recruited from a large-survey based study (Charting the Territory) that examined the bio-psychosocial health outcomes of parents and siblings. Linear mixed effect models were used to assess the association between siblings' emotions and behaviour trajectories and selected demographic variables. Siblings' emotions and behaviour were measured with Child Behaviour Checklist (CBCL). RESULTS: Siblings' mean age was 11.2 years at baseline and Internalizing, Externalizing, and Total Behaviour Problems mean scores were within normal ranges across time. However, 7-25% of siblings had scores within the clinical range. Brothers had higher levels of Internalizing Problems than sisters, whereas sisters had higher levels of Externalizing Problems than brothers. When treatment was first sought for the ill child less than a year prior to study participation, siblings had higher levels of Internalizing and Externalizing Problems compared with siblings who participated more than one year after treatment was sought. CONCLUSION: Healthy siblings experience emotional and behavioural problems early in the child's disease trajectory. Although these problems improve with time, our findings show that brothers and sisters experience different types of challenges. Therefore, timely support for siblings is important as they navigate through the uncertainties and challenges.

"I Don't Get to Play With My Mum Anymore": Experiences of Siblings Aged 8-12 of Children With Cancer: A Qualitative Study.

Background: Siblings of children with cancer have been shown to experience disruption in multiple domains including family, school, and friendships. Existing literature on siblings' experiences focuses on older children or on a broad range of ages. Aim: To explore the experience of siblings aged 8-12 years when their brother or sister is diagnosed with cancer. Method: A qualitative design incorporating phenomenology as the theoretical framework was used. Participants were recruited from across Australia via notices on social media sites and by the distribution of flyers. We used thematic analysis to analyze the data. Data were collected via semistructured interviews conducted either in person or online. Findings: A total of 13 siblings (7 boys and 6 girls) aged between 8 and 12 years (M = 9.8, SD = 1.6) were interviewed. Seven main themes were identified. These were "It was really hard": Reactions to the cancer diagnosis; "I'm really angry": Emotional and Physical Responses to siblings' treatment; "I pretend teddy is real": Play as an outlet; "It was very lonely": Missing their siblings; "I missed out on a lot of fun": Disruption of activities: School, sports, playdates, and parties; Change and Transition and "Making a difficult situation worse": COVID-19 Pandemic. Discussion: Findings extend the current understanding showing that younger siblings' developmental and cognitive skills impact their experiences of childhood cancer. Younger siblings outlined the many losses they experienced which demonstrated a need for a comprehensive and tailored program to support young siblings aged under 12 of children with cancer.

Evaluating iSibWorks: A virtual cognitive-behavioural intervention for siblings of children with disabilities.

Objectives: 1) Examine if participation in iSibWorks, a group-based virtual intervention for siblings of children with disabilities, impacted siblings' perception of quality of life (QoL) and social support; and 2) Explore siblings' feedback on iSibWorks. Methods: Thirty-eight children participated in iSibWorks and completed questionnaires (Pediatric Quality of Life [PedsQL™], Social Support Scale for Children [SSSC]) one week pre- and post-intervention. Conventional content analysis was used to explore siblings' open-ended responses on a post-participation feedback form. Results: No significant differences in PedsQL™ and SSSC scores were observed after participating in iSibWorks. Despite this, siblings had positive feedback about iSibWorks and discussed: 1) Engaging in group learning and activities, 2) Meeting other siblings, and 3) Applying iSibWorks content to their daily life. Conclusion: Factors related to the COVID-19 pandemic such as family stress, school closures, virtual learning, and social distancing likely impacted study results. Although there were no significant changes in QoL and social support, siblings found iSibWorks to be fun, meaningful, and engaging. Innovation: Siblings of children with disabilities can experience psychosocial challenges and there are few virtual interventions designed for this population. iSibWorks was adapted to address this gap and increase access and support for siblings of children with disabilities.