Search

Background: Practice-based research networks (PBRNs) promote the conduct of research in real-world settings by engaging primary care clinicians as champion research collaborators. Card studies are brief surveys administered to patients or clinicians at the point of care. The objective of this paper is to describe the design and evaluation of a card study methodology that the WWAMI Region Practice and Research Network (WPRN) used to develop research partnerships across multiple member sites. Methods: We used a collaborative model to develop, implement and disseminate the results of a network-wide card study to assess patient preferences for weight loss in primary care. After the card study data collection was completed, we conducted individual and focus group interviews and a brief survey of participating practice champions. Results: Increased research engagement and personal and professional developments were the primary motivators for participating in the development of the card study. Increasing research activity at practices and learning information about patients were motivators for implementing the study. Their participation resulted in champions reporting increased confidence in collaborating on research projects as well as the development of new clinical services for patients. Discussion: This collaborative model positively influenced research capacity in the WPRN and may be a useful strategy for helping PBRNs conducted translational research. [ABSTRACT FROM AUTHOR]

Spinocerebellar ataxia type 7 (SCA7) is a dominantly inherited disorder characterized by cerebellum and brain stem neurodegeneration. CA7 is caused by a CAG/polyglutamine (polyQ) repeat expansion in the ataxin-7 gene. We previously reported that directed expres ion of polyQ-ataxin-7 in Bergmann glia (BG) in transgenic mice leads to ataxia and non-cell-autonomous Purkinje cell (PC) degeneration. To further define the cellular basis ofSCA7, we derived a conditional inactivation mouse model by inserting a loxP-flanked ataxin-7 cD A with 92 repeats into the translational start site of the murine prion protein (PrP) gene in a bacterial artificial chromo orne (BAC). The PrP-jloxed-SCA7-92QBACmice developed neurological disease, and exhibited cerebellar degeneration and BG proce s lo s. To inactivate polyQ-ataxin-7 expression in specific cerebellar cell types, we crossed PrP-jloxed-SCA7-92Q BAC mice with Gfa2-Cre transgenic mice (to direct Cre to BG) or Pcp2-Cre transgenic mice (which yields Cre in PCs and inferior olive). Excision of ataxin-7 from BG partially rescued the behavioral phenotype, but did not prevent BG process loss or molecular layer thinning, while excision of ataxin-7 from PC and inferior olive provided significantly greater rescue and prevented both pathological changes, revealing a non-cell-autonomous basis for BG pathology. When we prevented expression of mutant ataxin-7 in BG, PCs, and inferior olive by deriving Gfa2-Cre;Pcp2-Cre;PrP-jloxed-SCA7-92Q BA C triple transgenic mice, we noted a dramatic improvement in SCA7 disease phenotypes. These findings indicate that CA7 disease pathogenesis involves a convergence of alterations in a variety of different cell types to fully recapitulate the cerebellar degeneration. [ABSTRACT FROM AUTHOR]