Community readiness and momentum: identifying and including community-driven variables in a mixed-method rural palliative care service siting model.

BACKGROUND: Health service administrators make decisions regarding how to best use limited resources to have the most significant impact. Service siting models are tools that can help in this capacity. Here we build on our own mixed-method service siting model focused on identifying rural Canadian communities most in need of and ready for palliative care service enhancement through incorporating new community-driven insights. METHODS: We conducted 40 semi-structured interviews with formal and informal palliative care providers from four purposefully selected rural communities across Canada. Communities were selected by running our siting model, which incorporated GIS methods, and then identifying locations suitable as qualitative case studies. Participants were identified using multiple recruitment methods. Interviews were transcribed verbatim and the transcripts were reviewed to identify emerging themes and were coded accordingly. Thematic analysis then ensued. RESULTS: We previously introduced the inclusion of a 'community readiness' arm in the siting model. This arm is based on five community-driven indicators of palliative care service enhancement readiness and need. The findings from the current analysis underscore the importance of this arm of the model. However, the data also revealed the need to subjectively assess the presence or absence of community awareness and momentum indicators. The interviews point to factors such as educational tools, volunteers, and local acknowledgement of palliative care priorities as reflecting the presence of community awareness and factors such as new employment and volunteer positions, new care spaces, and new projects and programs as reflecting momentum. The diversity of factors found to illustrate these indicators between our pilot study and current national study demonstrate the need for those using our service siting model to look for contextually-relevant signs of their presence. CONCLUSION: Although the science behind siting model development is established, few researchers have developed such models in an open way (e.g., documenting every stage of model development, engaging with community members). This mixed-method study has addressed this notable knowledge gap. While we have focused on rural palliative care in Canada, the process by which we have developed and refined our siting model is transferrable and can be applied to address other siting problems.

Linking Inuit Knowledge and Public Health for Improved Child and Youth Oral Health in NunatuKavut.

The oral health of Inuit children in Canada has been identified as a public health crisis. Although efforts are being made to identify and address ways to deal with this crisis, current policy and program approaches are largely entrenched within the prevailing paradigm of dental science to the exclusion of Indigenous people's understandings of health. This article reports qualitative findings of a larger study aimed at identifying, understanding, and addressing rates of oral disease among children living in NunatuKavut, a cluster of small, coastal Inuit communities located in southern Labrador, Canada. Through 18 focus groups with youth (n = 86), caregivers (n = 22), and interviews with key informant (n = 13), this study begins to elucidate southern Inuit understandings of oral health. Theorized using Two-Eyed Seeing, an Indigenous approach to balancing both Indigenous and non-Indigenous understandings of the world, the findings reported here reveal 3 themes, each of which is crosscut by historical and contemporary dimensions: 1) (w)holistic conceptualizations of health are essential to good oral health, 2) achieving optimal oral health is prohibitive for Inuit communities, and 3) community-engaged oral health service delivery is needed. Our recommendations have implications for improved oral public health service delivery for Inuit communities, in that the inclusion of Inuit perspectives on oral health should form an instrumental element of oral public health service delivery. Knowledge Transfer Statement: The results of this study may be used by clinicians and oral health educators to inform approaches to oral health service delivery within the context of Indigenous communities. It may also be used by policymakers to recognize how historical and contemporary issues of colonization relate to the formation of oral health-related policies.

GAD antibodies in probands and their relatives in a cohort clinically selected for Type 2 diabetes.

AIMS: A subset of patients who present as if they have Type 2 diabetes have positive pancreatic autoantibodies, and have been referred to as having latent autoimmune diabetes in adults (LADA). We assessed the prevalence and clinical characteristics of patients with glutamic acid decarboxylase antibodies (GADA) in a cohort clinically selected for Type 2 diabetes and determined the presence of diabetes and GADA in their first-degree relatives. METHODS: GADA were measured in 2059 subjects, not known to be related, and clinically selected as having Type 2 diabetes for genetic studies. Clinical characteristics were compared in GADA positive and GADA negative subjects. Diabetes and GAD antibody status were compared in 208 first-degree relatives of GADA positive and GADA negative probands. RESULTS: Of the subjects, 136 (7%) were GADA positive. Compared with the GADA negative subjects, they were slimmer (P < 0.001), diagnosed at a younger age (P = 0.011) and progressed to insulin faster (P < 0.001). Thirty-three per cent of GADA positive subjects had a first-degree relative with diabetes compared with 42% of GADA negative subjects (P = 0.034). The overall prevalence of GADA was similar in the first-degree relatives of GADA positive and GADA negative probands (4 v 5%), and 19 of 22 (86%) diabetic relatives of GADA positive probands were GADA negative. CONCLUSION: Despite clinically selecting a Type 2 diabetes cohort, 7% were GADA positive with an altered phenotype. These GADA positive patients had a strong family history of non-autoimmune diabetes. This suggests that, in this subgroup of patients, autoimmune pancreatic beta-cell destruction occurs on a background of Type 2 diabetes genetic susceptibility.

Molecular genetics and phenotypic characteristics of MODY caused by hepatocyte nuclear factor 4alpha mutations in a large European collection.

AIMS/HYPOTHESIS: Heterozygous mutations in the gene of the transcription factor hepatocyte nuclear factor 4alpha (HNF-4alpha) are considered a rare cause of MODY with only 14 mutations reported to date. The description of the phenotype is limited to single families. We investigated the genetics and phenotype of HNF-4alpha mutations in a large European Caucasian collection. METHODS: HNF-4alpha was sequenced in 48 MODY probands, selected for a phenotype of HNF-1alpha MODY but negative for HNF-1alpha mutations. Clinical characteristics and biochemistry were compared between 54 HNF-4alpha mutation carriers and 32 familial controls from ten newly detected or previously described families. RESULTS: Mutations in HNF-4alpha were found in 14/48 (29%) probands negative for HNF-1alpha mutations. The mutations found included seven novel mutations: S34X, D206Y, E276D, L332P, I314F, L332insCTG and IVS5nt+1G>A. I314F is the first reported de novo HNF-4alpha mutation. The average age of diagnosis was 22.9 years with frequent clinical evidence of sensitivity to sulphonylureas. Beta cell function, but not insulin sensitivity, was reduced in diabetic mutation carriers compared to control subjects (homeostasis model assessment of beta cell function 29% p<0.001 vs controls). HNF-4alpha mutations were associated with lower apolipoprotein A2 (p=0.001), A1 (p=0.04) and total HDL-cholesterol (p=0.02) than in control subjects. However, in contrast to some previous reports, levels of triglycerides and apolipoprotein C3 were normal. CONCLUSIONS/INTERPRETATION: HNF-4alpha mutations are common when no HNF-1alpha mutation is found in strictly defined MODY families. The HNF-4alpha clinical phenotype and beta cell dysfunction are similar to HNF-1alpha MODY and are associated with reduced apolipoprotein A2 levels. We suggest that sequencing of HNF-4alpha should be performed in patients with clinical characteristics of HNF-1alpha MODY in whom mutations in HNF-1alpha are not found.

Serum amino acids in patients with mutations in the hepatocyte nuclear factor-1 alpha gene.

AIMS: Knockout mice lacking both copies of the hepatocyte nuclear factor 1 (HNF1) gene have altered serum levels of amino acids and generalized aminoaciduria. The aim of our study was to test whether alterations in serum amino acid levels were found in patients with mutations in the hepatocyte nuclear factor-1 alpha (HNF-1alpha) gene compared with controls. METHODS: Fasting serum from 20 patients with HNF-1alpha mutations and 20 age, sex and body mass index-matched controls was analysed for 16 amino acids. Means were compared between the two groups and Z scores calculated. RESULTS: There was no significant difference between patients with HNF-1alpha mutations and controls in serum levels of phenylalanine, arginine, citrulline or lysine as suggested by knockout mice models. Although serum levels of eight amino acids were different in the two groups, these were not significant after Bonferroni correction. CONCLUSIONS: The alterations in serum amino acid levels seen in mice models are not seen in patients with mutations in the HNF-1alpha gene. This suggests differences in mouse and man in the regulation of amino acid transport and has not provided us with a phenotypic marker to use before confirmatory genetic testing.