A qualitative investigation of the experiences of patients living with antiphospholipid antibodies.

OBJECTIVE: Substantial morbidity and mortality affect those with antiphospholipid antibodies (aPLs) and antiphospholipid syndrome (APS), yet patient experiences remain poorly understood. This research investigated patient experiences of aPL/APS diagnosis; effects on daily life; and healthcare and treatment. METHODS: Patients aged ≥18 years with APS per the Revised Sapporo criteria or with ≥1 positive aPL on ≥2 occasions were recruited from a Canadian multidisciplinary APS clinic to participate in semi-structured in-depth interviews. Interviews were conducted virtually and transcribed verbatim for subsequent thematic analysis. RESULTS: Twenty-one patients with aPLs/APS participated; 95.2% were female, mean (SD) age was 45.6 (15.0) years. Most (71.4%) had APS, and 71.4% had aPLs/APS with SLE. Results are presented around patient experiences of aPL/APS diagnosis, effects on daily life, and healthcare and treatment. Participants described medical complications/physical symptoms and the healthcare, lifestyle, and emotional impacts experienced around the time of aPLs/APS diagnosis. In addition to the physical and psychosocial impacts of living with aPLs/APS, patients reported modified leisure activities, altered employment trajectories, and positive and negative impacts on relationships. Impacts on family planning were also a critical component of the aPL/APS lived experience; participants shared experiences of miscarriage, other pregnancy complications, and medication-related challenges (e.g., with low-molecular-weight heparin injections). Challenging aspects of aPL/APS healthcare and treatment were also discussed, particularly related to the lifestyle, physical, and emotional burden of medication use. Although a lack of resources was described, participants expressed trust in healthcare providers when making management decisions or when seeking information. Suggestions for resources included the need for additional medication-related information, examples to help contextualize management behaviours, and additional information for those with aPLs/APS without SLE. CONCLUSION: Patients highlighted how the diverse manifestations of aPLs/APS, accentuated by management-related challenges, impose considerable physical and psychosocial burdens. Results will inform the development of patient resources aligned with patient priorities.

Attitudes, subjective norms and perceived behavioural control factors influencing Canadian secondary school students' milk and milk alternatives consumption.

OBJECTIVE: The research objectives were to evaluate factors that influence Canadian secondary school students' milk and milk alternatives (MMA) consumption and to explore associations through age and gender lenses. DESIGN: A qualitative design was used, consisting of semi-structured interviews and photo-elicitation methods. Analysis was guided by the Theory of Planned Behaviour (TPB). Deductive and inductive thematic analyses were used to generate themes, charting data based on attributes such as gender and age. SETTING: Interviews were held virtually or via telephone. PARTICIPANTS: Participants were twenty-eight high school students from Ontario, Canada, diverse in terms of gender and age. RESULTS: Both desirable and undesirable beliefs about the health outcomes of consuming MMA were commonly discussed. These included health benefits such as strong bones, muscular strength, and growth, and health consequences like unwanted skin conditions, weight gain, and diseases. While boys and girls associated MMA consumption with muscular strength, boys predominantly considered this favourable, while girls discussed outcomes like unwanted skin conditions and weight gain more often. Adolescents' perspectives on taste/perceived enjoyment, environmentally friendly choices and animal welfare also influenced their MMA preferences. Parental influences were most cited among social factors, which appeared to be stronger during early adolescence. Factors involving cost, time and accessibility affected adolescents' beliefs about how difficult it was to consume MMA. CONCLUSIONS: Recommendations for shifting attitudes towards MMA are provided to address unfavourable beliefs towards these products. Interventions to increase MMA consumption among adolescents should include parents and address cost barriers.

Turning the tide on inequity through systematic equity action-analysis.

BACKGROUND: Collective agreement about the importance of centering equity in health research, practice, and policy is growing. Yet, responsibility for advancing equity is often situated as belonging to a vague group of 'others', or delegated to the leadership of 'equity-seeking' or 'equity-deserving' groups who are tasked to lead systems transformation while simultaneously navigating the violence and harms of oppression within those same systems. Equity efforts also often overlook the breadth of equity scholarship. Harnessing the potential of current interests in advancing equity requires systematic, evidence-guided, theoretically rigorous ways for people to embrace their own agency and influence over the systems in which they are situated. ln this article, we introduce and describe the Systematic Equity Action-Analysis (SEA) Framework as a tool that translates equity scholarship and evidence into a structured process that leaders, teams, and communities can use to advance equity in their own settings. METHODS: This framework was derived through a dialogic, critically reflective and scholarly process of integrating methodological insights garnered over years of equity-centred research and practice. Each author, in a variety of ways, brought engaged equity perspectives to the dialogue, bringing practical and lived experience to conversation and writing. Our scholarly dialogue was grounded in critical and relational lenses, and involved synthesis of theory and practice from a broad range of applications and cases. RESULTS: The SEA Framework balances practices of agency, humility, critically reflective dialogue, and systems thinking. The framework guides users through four elements of analysis (worldview, coherence, potential, and accountability) to systematically interrogate how and where equity is integrated in a setting or object of action-analysis. Because equity issues are present in virtually all aspects of society, the kinds of 'things' the framework could be applied to is only limited by the imagination of its users. It can inform retrospective or prospective work, by groups external to a policy or practice setting (e.g., using public documents to assess a research funding policy landscape); or internal to a system, policy, or practice setting (e.g., faculty engaging in a critically reflective examination of equity in the undergraduate program they deliver). CONCLUSIONS: While not a panacea, this unique contribution to the science of health equity equips people to explicitly recognize and interrupt their own entanglements in the intersecting systems of oppression and injustice that produce and uphold inequities.

Screening for Cognitive Impairment in Primary Care: Rationale and Tools.

Cognitive impairment is common and often under diagnosed in the early stages. Patients and family caregivers benefit from early diagnosis of reversible causes and longer lead time for care planning in primary dementia diagnoses. Primary care physicians are the first and best providers for diagnosing common, serious, and progressive cognitive disorders.

Economic burden of food allergy in Canada: Estimating costs and identifying determinants.

BACKGROUND: Limited data exist on the economic burden of food allergy (FA). OBJECTIVE: To assess FA-related direct (healthcare and out-of-pocket) and indirect (lost productivity) costs and their determinants in Canadian children and adults self-reporting FA. METHODS: FA-individuals self-reporting a convincing history or physician diagnosis were recruited through FA registries, an anaphylaxis registry, and advocacy associations, and electronically surveyed regarding FA-related healthcare use, out-of-pocket expenditures, and time lost from paid and unpaid labor. Direct and indirect costs (2020 Canadian dollars [CAD]) were stratified on severe reaction vs mild, moderate or no reaction, and children vs adults; multivariate regressions assessed the association between costs and sociodemographic and disease characteristics. RESULTS: Between May 2018 and July 2019, 2692 eligible individuals responded (2189 convincing history and 503 physician diagnosis only); 1020 experienced a severe reaction; 1752 were children. Per FA-individual, annual healthcare, out-of-pocket, and indirect costs were $1267, $2136, and $7950. Those with a severe reaction had higher healthcare and out-of-pocket costs than those with mild, moderate or no reaction. FA-children vs FA-adults had higher healthcare and out-of-pocket costs, and lower indirect costs. Multivariate results showed that lower age, a severe reaction ever, multiple FAs, and fair or poor general health were associated with higher healthcare and out-of-pocket costs. Higher age, lower household education and income, and fair or poor general health were associated with higher indirect costs. CONCLUSION: The economic burden of FA in Canada is substantial, particularly for those with a severe reaction ever, multiple FAs, and fair or poor general health. It is crucial that those most adversely affected are allocated appropriate resources to support disease management.

Socioeconomic factors and bacillary dysentery risk in Jiangsu Province, China: a spatial investigation using Bayesian hierarchical models.

Bacillary dysentery (BD) is an acute diarrheal disease prevalent in areas affected by socioeconomic disparities. We investigated BD risk and its associations with socioeconomic factors at the county-level in Jiangsu province, China using epidemiological and socioeconomic data from 2011-2014. We fitted four Bayesian hierarchical models with various prior specifications for random effects. As all model comparison criteria values were similar, we presented results from a reparameterized Besag-York-Mollié model, which addressed issues with the identifiability of variance captured by spatial and independent effects. Our model adjusted for year and socioeconomic status showed 18-65% decreased BD risk compared to 2011. We found a high relative risk in the northwestern and southwestern counties. Increasing the percentage of rural households, rural income per capita, health institutions per capita, or hospital beds per capita decreases the relative risk of BD, respectively. Our findings can be used to improve infectious diarrhea surveillance and enhance existing public health interventions.

Geospatial analysis of cholera patterns in Nigeria: findings from a cross-sectional study.

BACKGROUND: Persistence of cholera outbreaks in developing countries calls for concern and more targeted intervention measures for long-term control. This research undertook spatial analysis of cholera incidence in Nigeria over a seventeen-year period to determine the existence of regional hotspots and predictors. METHODS: A cross-sectional study design was used for the research. Cholera data for each of the thirty-six states and the federal capital territory (FCT) were obtained from the Nigeria Centre for Disease Control (NCDC) of the Federal Ministry of Health, Nigeria. Socioeconomic data including proportion of households using solid waste disposal (unapproved dumpsite, refuse burying, refuse burning, public dumpsite, and refuse collectors), water sources (pipe borne water, well, borehole, rain water, surface waters and water vendors), sewage disposal (water closet, pit latrines, bucket/pan, public toilet and nearby bush/stream), living in a single room and earning less than minimum wage (18,000 naira) were obtained from National Population Commission. On the other hand, proportion of illiterate adults (15 years and above) and poor people; and population density were obtained from National Bureau of Statistics. Each socioeconomic data was obtained at state level. Cholera patterns were analysed at state level using Global Moran's I while specific locations of cholera clusters were determined using Local Moran's I. Stepwise multiple regression was used to determine socioeconomic predictors of cholera incidence. RESULTS: Local Moran's I revealed significant cluster patterns in 1999, 2001, 2002, 2009 and 2010 in Adamawa, Gombe, Katsina, Bauchi, Borno, Yobe, and Kano states. Households using surface water was the significant predictor (23%) of the observed spatial variations in cholera incidence. CONCLUSIONS: Persistence of cholera outbreaks in some north east and north western states calls for more targeted, long-term and effective intervention measures especially on provision of safe sources of water supply by government and other stakeholders.

The value of hackathons in integrated knowledge translation (iKT) research: Waterlupus.

BACKGROUND: Despite a growing movement toward a knowledge-user-driven research process, our understanding of the generation, implementation and evaluation of specific approaches in the integrated knowledge translation (iKT) toolbox that aim to engage health and healthcare knowledge users is limited. Health hackathons offer an innovative approach with potential to generate direct and indirect health-related outcomes benefitting participants, knowledge users and the broader population. In May 2019, our research team hosted Waterlupus, a health hackathon to improve the economic lives of individuals with systemic lupus erythematosus (SLE) in Canada. Waterlupus was held with a multi-stakeholder group of 50 participants that included advocacy organization representatives, policy-makers, researchers, physicians, individuals with lived experience and students. While the hackathon generated viable solutions with the potential to positively impact the lives of individuals with SLE, understanding how participants perceived the hackathon as an iKT tool is critical in the planning and implementation of future iKT research. METHODS: Semi-structured in-depth telephone interviews were conducted with Waterlupus participants (n = 13) between August and November 2019 to (1) explore participant experiences of the hackathon; (2) investigate participant-identified hackathon outcomes; and (3) elicit recommendations for future iKT research using health hackathons. RESULTS: Participants provided feedback on the format and organization of Waterlupus, and identified direct and indirect outcomes to knowledge users, students and researchers beyond the innovations generated at the event. While the majority (n = 11) had never participated in a hackathon prior to Waterlupus, all 13 stated they would participate in future hackathons. Positive outcomes identified include connecting with students and other SLE stakeholders, the formation of professional and support networks, increased awareness of SLE, as well as the innovations generated. Participant recommendations for future health hackathons include the addition of stakeholders from industry or technology, and the need for clear and designated roles for stakeholders to ensure efficient use of resources. CONCLUSIONS: This work contributes to a limited literature regarding the use of health hackathons for social innovation, and offers knowledge-user suggestions relevant to the implementation of future iKT events, and hackathons specifically.

Intact Protein Mass Spectrometry of Cell Culture Harvest Guides Cell Line Development for Trispecific Antibodies.

IgG-like multispecific antibodies with asymmetric constructs have become widely used formats for therapeutic applications in recent years. Correct assembly of the subunits in this class of therapeutics is a critical quality attribute (CQA) with direct impact on biological activity. Therefore, early drug development efforts such as clone selection during cell line development must be guided by information on potential chain mispairing to enable timely decision making and risk mitigation. Here we describe a high-throughput analytical platform based on denaturing size-exclusion ultraperformance liquid chromatography (UPLC) coupled with intact protein mass spectrometry for profiling of mispairing and other product-related impurities, including half antibodies. This method can be performed directly on the clarified cell culture harvest fluid without the need for Protein A purification or other sample preparations and provides unbiased information on the product quality of the clones and the effect of growth conditions in a fast and cost-effective manner. Screening large numbers of clones expressing different trispecific antibody (tsAb) constructs revealed that although chain mispairing primarily depends on the antibody sequence and structure, it is also a characteristic of the clone. In addition, different growth conditions may affect the type and distribution of half antibodies and mispaired species impurities but not the quality ranking of the clones.

Newborn Screening for Mucopolysaccharidoses: Results of a Pilot Study with 100 000 Dried Blood Spots.

OBJECTIVE: To test, in a newborn screening (NBS) laboratory, the performance of liquid chromatography-tandem mass spectrometry (LC-MS/MS) to assay 5 enzymatic activities in dried blood spots (DBS) for NBS of 5 lysosomal storage diseases (mucopolysaccharidosis [MPS]-II, MPS-IIIB, MPS-IVA, MPS-VI, and MPS-VII). STUDY DESIGN: Three mm punches from de-identified DBS were obtained from the Washington NBS laboratory and submitted to the 5-plex LC-MS/MS assay. Screen cut-offs were established by analyzing the enzymatic activity in patients confirmed to have the MPS disorder. DNA sequencing of the relevant gene was performed on a second DBS punch for all samples with enzyme activity below 10% of the mean daily activity. RESULTS: (1) For MPS-II, 18 below cut-off samples, 1 pathogenic genotype, and 2 "high risk" genotypes; (2) For MPS-IIIB, no below cut-off samples; (3) For MPS-IVA, 8 below cut-off samples, 4 non-pathogenic genotypes, 4 genotypes unobtainable; (4) For MPS-VI, 4 below cut-off samples and no high-risk genotypes; (5) For MPS-VII, 1 below cut-off sample confirmed by genotype and clinical report to be affected. CONCLUSIONS: These results establish that the number of initial screen positive samples is low and manageable. Thus, population newborn screening for these conditions is feasible in a state newborn screening laboratory.

Water risk perceptions across the life-course of women in Kenya.

Inadequate access to water, sanitation and hygiene (WaSH) exposes many vulnerable populations, especially women and girls, to preventable diseases around the world. This paper reports findings from a photovoice project that explored water-related risk perceptions and health outcomes among women in Nyanchwa, Kenya. Thirteen women in four age categories were recruited for this study in July 2016 using the 'snowball' technique. From the results, inadequate access to WaSH was associated with increased water collection burden on women and children; environmental pollution; poor educational outcomes; loss of time due to water collection and poor sanitation infrastructure. Some barriers to change identified include financial barriers and inadequate government support. The identified risks and barriers are important considerations for the design, evaluation and mainstreaming of WaSH programs in resource constrained settings.

Trajectories of Depressive Symptoms and Perceived Stress From Pregnancy to the Postnatal Period Among Canadian Women: Impact of Employment and Immigration.

Objectives. To identify trajectory patterns of maternal depressive symptoms and perceived stress from midpregnancy to 2 years postpartum and determine relationships with selected sociodemographic factors including income, education, immigration, and postpartum employment. Methods. Pregnant women (n = 3307) recruited from the general population in 4 regions in Canada provided 6 waves of data from pregnancy to 2 years postpartum. The study was conducted from 2009 to 2015. Results. We determined 5 trajectory groups distinguished by time and magnitude for both depressive symptoms and perceived stress. Immigrants living in Canada for more than 5 up to 10 years, but not more recent arrivals, were at higher risk for persistent stress and depression independent of income status. Being employed at 1 year postpartum was associated with a lower likelihood of postpartum depression and perceived stress, while mothers reporting work exhaustion were substantially more likely to experience persistent depression and stress. Conclusions. The study highlighted the heterogeneous nature of depressive symptoms and perceived stress. Targeting interventions toward women 5 to 10 years after immigration and those experiencing exhaustion from postpartum work may be particularly beneficial.

Risk for Maternal Depressive Symptoms and Perceived Stress by Ethnicities in Canada: From Pregnancy Through the Preschool Years.

OBJECTIVE: Past cross-sectional studies have reported that mothers from ethnic minorities experience higher levels of prenatal and post-partum psychosocial distress compared with mothers from ethnic majorities. However, no studies have examined how the pattern varies longitudinally in a Canadian population of heterogeneous ethnicity. METHODS: We analyzed data from 3,138 mothers participating in the Canadian Healthy Infant Longitudinal Development (CHILD) Study, a longitudinal multi-center study incorporating 10 distinct waves of psychosocial data collection from pregnancy until the index child was aged 5 y. Maternal self-identified ethnicity was grouped as White Caucasian, First Nations, Black, Southeast Asian, East Asian, South Asian, Middle Eastern, Hispanic and mixed ethnicity. We performed a multi-level regression to determine whether mothers of specific minority ethnicities were more likely to experience higher levels of distress (i.e. depressive symptoms and perceived stress) compared to white Caucasian mothers. RESULTS: Mothers self-identifying as Black or First Nations had consistently higher distress scores than mothers from other ethnicities across all data collection times. After adjusting for relevant variables (history of depression, education, household income, marital status, and social support), First Nations mothers had a 20% increase in the mean scores of depressive symptoms compared to White Caucasian Mothers. CONCLUSIONS: Increased levels of perinatal and post-partum distress were seen in only some ethnic minority groups. Studies should avoid collapsing all categories into ethnic minority or majority and may need to consider how ethnicity interacts with other sociodemographic factors such as poverty.

Effect of peer support interventions on cardiovascular disease risk factors in adults with diabetes: a systematic review and meta-analysis.

BACKGROUND: Peer support by persons affected with diabetes improves peer supporter's diabetes self-management skills. Peer support interventions by individuals who have diabetes or are affected by diabetes have been shown to improve glycemic control; however, its effects on other cardiovascular disease risk factors in adults with diabetes are unknown. We aimed to estimate the effect of peer support interventions on cardiovascular disease risk factors other than glycemic control in adults with diabetes. METHODS: We conducted a systematic review and meta-analysis of randomized controlled trials comparing peer support interventions to a control condition in adults affected by diabetes that measured any cardiovascular disease risk factors [Body Mass Index, smoking, diet, physical activity, cholesterol level, glucose control and blood pressure]. Quality was assessed by Cochrane's risk of bias tool. We calculated standardized mean difference effect sizes using random effects models. RESULTS: We retrieved 438 citations from multiple databases including OVID MEDLINE, Cochrane database and Scopus, and author searches. Of 233 abstracts reviewed, 16 articles met inclusion criteria. A random effects model in a total of 3243 participants showed a positive effect of peer support interventions on systolic BP with a pooled effect size of 2.07 mmHg (CI 0.35 mmHg to 3.79 mmHg, p = 0.02); baseline pooled systolic blood pressure was 137 mmHg. There was a non-significant effect of peer support interventions on diastolic blood pressure, cholesterol, body mass index, diet and physical activity. Cardiovascular disease risk factors other than glycemic control outcomes were secondary outcomes in most studies and baseline values were normal or mildly elevated. Only one study reported smoking outcomes. CONCLUSIONS: We found a small (2 mmHg) positive effect of peer support interventions on systolic blood pressure in adults with diabetes whose baseline blood pressure was on average minimally elevated. Additional studies need to be conducted to further understand the effect of peer support interventions on high-risk cardiovascular disease risk factors in adults with diabetes.

Can knowledge exchange support the implementation of a health-promoting schools approach? Perceived outcomes of knowledge exchange in the COMPASS study.

BACKGROUND: Despite the potential population-level impact of a health-promoting schools approach, schools face challenges in implementation, indicating a gap between school health research and practice. Knowledge exchange provides an opportunity to reduce this gap; however, there has been limited evaluation of these initiatives. This research explored researchers' and knowledge users' perceptions of outcomes associated with a knowledge exchange initiative within COMPASS, a longitudinal study of Canadian secondary students and schools. Schools received annual tailored summaries of their students' health behaviours and suggestions for action and were linked with knowledge brokers to support them in taking action to improve student health. METHODS: Qualitative semi-structured interviews were conducted with COMPASS researchers (n = 13), school staff (n = 13), and public health stakeholders (n = 4) to explore their experiences with COMPASS knowledge exchange. Key issues included how knowledge users used school-specific findings, perceived outcomes of knowledge exchange, and suggestions for change. RESULTS: Outcomes for both knowledge users and researchers were identified; interestingly, knowledge users attributed more outcomes to using school-specific findings than knowledge brokering. School and public health participants indicated school-specific findings informed their programming and planning. Importantly, knowledge exchange provided a platform for partnerships between researchers, schools, and public health units. Knowledge brokering allowed researchers to gain feedback from knowledge users to enhance the study and a better understanding of the school environment. Interestingly, COMPASS knowledge exchange outcomes aligned with Samdal and Rowling's eight theory-driven implementation components for health-promoting schools. Hence, knowledge exchange may provide a mechanism to help schools implement a health-promoting schools approach. CONCLUSIONS: This research contributes to the limited literature regarding outcomes of knowledge brokering in public health and knowledge exchange in school health research. However, since not all schools engaged in knowledge brokering, and not all schools that engaged discussed these outcomes, further research is needed to determine the amount of engagement required for change and examine the process of COMPASS knowledge brokering to consider how to increase school engagement.

"Now What?" Perceived Factors Influencing Knowledge Exchange in School Health Research.

Increasing the uptake of school health research into practice is pivotal for improving adolescent health. COMPASS, a longitudinal study of Ontario and Alberta secondary students and schools (2012-2021), used a knowledge exchange process to enhance schools' use of research findings. Schools received annual summaries of their students' health behaviors and suggestions for action and were linked with a knowledge broker to support them in making changes to improve student health. The current research explored factors that influenced COMPASS knowledge exchange activities. Semistructured interviews were conducted with researchers (n = 13), school staff (n = 13), and public health stakeholders (n = 4). Interestingly, knowledge users focused more on factors that influenced their use of COMPASS findings than factors that influenced knowledge brokering. The factors identified by participants are similar to those that influence implementation of school health interventions (e.g., importance of school champions, competing priorities, inadequate resources). While knowledge exchange offers a way to reduce the gap between research and practice, schools that need the most support may not engage in knowledge exchange; hence, we must consider how to increase engagement of these schools to ultimately improve student health.

Psychosocial impacts of the lack of access to water and sanitation in low- and middle-income countries: a scoping review.

The lack of access to safe water and adequate sanitation has implications for the psychosocial well-being of individuals and households. To review the literature on psychosocial impacts, we completed a scoping review of the published literature using Medline, Embase, and Scopus. Fifteen studies met the inclusion criteria and were reviewed in detail. Of the included studies, six were conducted in India, one in Nepal, one in Mexico, one in Bolivia, two in Ethiopia, one in Zimbabwe, one in South Africa, and two in Kenya. Four interrelated groups of stressors emerged from the review: physical stressors, financial stressors, social stressors, and stressors related to (perceived) inequities. Further, gender differences were observed, with women carrying a disproportionate psychosocial burden. We argue that failure to incorporate psychosocial stressors when estimating the burden or benefits of safe water and sanitation may mask an important driver of health and well-being for many households in low- and middle-income countries. We propose further research on water-related stressors with particular attention to unique cultural norms around water and sanitation, short and long term psychosocial outcomes, and individual and collective coping strategies. These may help practitioners better understand cumulative impacts and mechanisms for addressing water and sanitation challenges.

The sharing of ballistics data across Europe and neighbouring territories.

The current study explored the use of ballistic examinations and cross-border information sharing across 14 European countries. The presented data were collected using a mixed methods technique consisting of semi-structured interviews and questionnaires that were completed by participants. The results painted a very heterogeneous picture of the use of automated ballistic systems across these countries, as well as how ballistic analyses are integrated in the fight against gun-enabled crime. Three super-ordinates themes emerged from the thematic analysis: use of automated ballistic systems; Ballistic evidence recovery and analysis; knowledge exchange and best practices. The ability to draw firm conclusions regarding the value of ballistics comparison systems, either on a national or cross-border basis, is hampered by inconsistencies regarding data recording practices and definitions. Therefore, key recommendations are suggested to establish better cross border cooperation between member states and develop a better understanding of data sharing procedures.

Pilot study of newborn screening for six lysosomal storage diseases using Tandem Mass Spectrometry.

BACKGROUND: There is current expansion of newborn screening (NBS) programs to include lysosomal storage disorders because of the availability of treatments that produce an optimal clinical outcome when started early in life. OBJECTIVE: To evaluate the performance of a multiplex-tandem mass spectrometry (MS/MS) enzymatic activity assay of 6 lysosomal enzymes in a NBS laboratory for the identification of newborns at risk for developing Pompe, Mucopolysaccharidosis-I (MPS-I), Fabry, Gaucher, Niemann Pick-A/B, and Krabbe diseases. METHODS AND RESULTS: Enzyme activities (acid α-glucosidase (GAA), galactocerebrosidase (GALC), glucocerebrosidase (GBA), α-galactosidase A (GLA), α-iduronidase (IDUA) and sphingomyeline phosphodiesterase-1 (SMPD-1)) were measured on ~43,000 de-identified dried blood spot (DBS) punches, and screen positive samples were submitted for DNA sequencing to obtain genotype confirmation of disease risk. The 6-plex assay was efficiently performed in the Washington state NBS laboratory by a single laboratory technician at the bench using a single MS/MS instrument. The number of screen positive samples per 100,000 newborns were as follows: GAA (4.5), IDUA (13.6), GLA (18.2), SMPD1 (11.4), GBA (6.8), and GALC (25.0). DISCUSSION: A 6-plex MS/MS assay for 6 lysosomal enzymes can be successfully performed in a NBS laboratory. The analytical ranges (enzyme-dependent assay response for the quality control HIGH sample divided by that for all enzyme-independent processes) for the 6-enzymes with the MS/MS is 5- to 15-fold higher than comparable fluorimetric assays using 4-methylumbelliferyl substrates. The rate of screen positive detection is consistently lower for the MS/MS assay compared to the fluorimetric assay using a digital microfluidics platform.

Peer Support Interventions for Adults With Diabetes: A Meta-Analysis of Hemoglobin A1c Outcomes.

PURPOSE: Peer support intervention trials have shown varying effects on glycemic control. We aimed to estimate the effect of peer support interventions delivered by people affected by diabetes (those with the disease or a caregiver) on hemoglobin A1c (HbA1c) levels in adults. METHODS: We searched multiple databases from 1960 to November 2015, including Ovid MEDLINE, the Cochrane Central Register of Controlled Trials, CINAHL, and Scopus. We included randomized controlled trials (RCTs) of adults with diabetes receiving peer support interventions compared with otherwise similar care. Seventeen of 205 retrieved studies were eligible for inclusion. Quality was assessed with the Cochrane risk of bias tool. We calculated the standardized mean difference (SMD) of change in HbA1c level from baseline between groups using a random effects model. Subgroup analyses were predefined. RESULTS: Seventeen studies (3 cluster RCTs, 14 RCTs) with 4,715 participants showed an improvement in pooled HbA1c level with an SMD of 0.121 (95% CI, 0.026-0.217; P = .01; I2 = 60.66%) in the peer support intervention group compared with the control group; this difference translated to an improvement in HbA1c level of 0.24% (95% CI, 0.05%-0.43%). Peer support interventions showed an HbA1c improvement of 0.48% (95% CI, 0.25%-0.70%; P <.001; I2 = 17.12%) in the subset of studies with predominantly Hispanic participants and 0.53% (95% CI, 0.32%-0.73%; P <.001; I2 = 9.24%) in the subset of studies with predominantly minority participants; both were clinically relevant. In sensitivity analysis excluding cluster RCTs, the overall effect size changed little. CONCLUSIONS: Peer support interventions for diabetes overall achieved a statistically significant but minor improvement in HbA1c levels. These interventions may, however, be particularly effective in improving glycemic control for people from minority groups, especially those of Hispanic ethnicity.