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Abstract Background To learn about the molecular etiology of strabismus, we are studying the genetic basis of 'congenital fibrosis of the extraocular muscles' (CFEOM). These syndromes are characterized by congenital restrictive ophthalmoplegia affecting muscles in the oculomotor and trochlear nerve distribution. Individuals with the classic form of CFEOM are born with bilateral ptosis and infraducted globes. When all affected members of a family have classic CFEOM, we classify the family as a CFEOM1 pedigree. We have previously determined that a CFEOM1 gene maps to the FEOM1 locus on chromosome 12cen. We now identify additional pedigrees with CFEOM1 to determine if the disorder is genetically heterogeneous and, if so, if any affected members of CFEOM1 pedigrees or sporadic cases of classic CFEOM harbor mutations in ARIX, the CFEOM2 disease gene. Results Eleven new CFEOM1 pedigrees were identified. All demonstrated autosomal dominant inheritance, and nine were consistent with linkage to FEOM1. Two small CFEOM1 families were not linked to FEOM1, and both were consistent with linkage to FEOM3. We screened two CFEOM1 families consistent with linkage to FEOM2 and 5 sporadic individuals with classic CFEOM and did not detect ARIX mutations. Conclusions The phenotype of two small CFEOM1 families does not map to FEOM1, establishing genetic heterogeneity for this disorder. These two families may harbor mutations in the FEOM3 gene, as their phenotype is consistent with linkage to this locus. Thus far, we have not identified ARIX mutations in any affected members of CFEOM1 pedigrees or in any sporadic cases of classic CFEOM.

Health care public-private partnerships (PPPs) between a government and the private sector are based on a business model that aims to leverage private-sector expertise to improve clinical performance in hospitals and other health facilities. Although the financial implications of such partnerships have been analyzed, few studies have examined the partnerships' impact on clinical performance outcomes. Using quantitative measures that reflected capacity, utilization, clinical quality, and patient outcomes, we compared a government-managed hospital network in Lesotho, Africa, and the new PPP-managed hospital network that replaced it. In addition, we used key informant interviews to help explain differences in performance. We found that the PPP-managed network delivered more and higher-quality services and achieved significant gains in clinical outcomes, compared to the government-managed network. We conclude that health care public-private partnerships may improve hospital performance in developing countries and that changes in management and leadership practices might account for differences in clinical outcomes. [ABSTRACT FROM AUTHOR]

Background: To learn about the molecular etiology of strabismus, we are studying the genetic basis of 'congenital fibrosis of the extraocular muscles' (CFEOM). These syndromes are characterized by congenital restrictive ophthalmoplegia affecting muscles in the oculomotor and trochlear nerve distribution. Individuals with the classic form of CFEOM are born with bilateral ptosis and infraducted globes. When all affected members of a family have classic CFEOM, we classify the family as a CFEOM1 pedigree. We have previously determined that a CFEOM1 gene maps to the FEOM1 locus on chromosome 12cen. We now identify additional pedigrees with CFEOM1 to determine if the disorder is genetically heterogeneous and, if so, if any affected members of CFEOM1 pedigrees or sporadic cases of classic CFEOM harbor mutations in ARIX, the CFEOM2 disease gene. Results: Eleven new CFEOM1 pedigrees were identified. All demonstrated autosomal dominant inheritance, and nine were consistent with linkage to FEOM1. Two small CFEOM1 families were not linked to FEOM1, and both were consistent with linkage to FEOM3. We screened two CFEOM1 families consistent with linkage to FEOM2 and 5 sporadic individuals with classic CFEOM and did not detect ARIX mutations. Conclusions: The phenotype of two small CFEOM1 families does not map to FEOM1, establishing genetic heterogeneity for this disorder. These two families may harbor mutations in the FEOM3 gene, as their phenotype is consistent with linkage to this locus. Thus far, we have not identified ARIX mutations in any affected members of CFEOM1 pedigrees or in any sporadic cases of classic CFEOM.

Unrepresented patients who lack decision-making capacity can remain in hospitals for weeks awaiting guardianship and subsequent discharge, resulting in medically unnecessary and costly extended stays. The guardianship process is complicated and requires collaboration between the medical and legal systems. The Rocky Mountain Regional Veteran Affairs Medical Center created a guardianship social worker position to improve the guardianship process by developing subject matter expertise, infrastructure, and standardized procedures. In this brief report, we provide preliminary data on the impact of a guardianship social worker. Time to guardianship completion, mean lengths of stay (LOS), counts of completed guardianships, and cost estimates were compared before and after the guardianship social worker position was created. The mean time to complete guardianships decreased from 121.9 to 69.8 days ( p  = .13), the mean LOS for patients awaiting guardianship decreased from 129.8 to 117.2 days ( p  = .39), and the yearly counts of completed guardianships increased from 2 to 9 ( p  = .002). Estimated inpatient cost savings per patient was $56,244. While promising, these preliminary data are limited by small case numbers and the unknown impact of the COVID-19 pandemic on long-term placement. Creating guardianship-focused social work positions has the potential to create efficiencies in the guardianship process.

Objectives: To study the association between the collection and use of clinician performance information in physician practices and patient experience in primary care., Study Design: Patient experience scores are calculated from the 2018-2019 Massachusetts Statewide Survey of Adult Patient Experience of Primary Care. Physicians were attributed to physician practices using the Massachusetts Healthcare Quality Provider database. Scores were matched to information on the collection or use of clinician performance information from the National Survey of Healthcare Organizations and Systems using practice name and location., Methods: We conducted observational multivariant generalized linear regression at the patient level where the dependent variables were 1 of 9 patient experience scores and the independent variables were 1 of 5 domains in the collection or use of performance information of the practice. Patient-level controls included self-reported general health, self-reported mental health, age, sex, education, and race/ethnicity. Practice-level controls include the size of the practice and the availability of weekend and evening hours., Results: Nearly 90% of practices in our sample collect or use clinician performance information. High patient experience scores were associated with whether any information was collected and used, especially with whether the practice shared this information internally to compare. Among practices that used clinician performance information, patient experience was not associated with whether the information was used in more aspects of care., Conclusions: The collection and use of clinician performance information were associated with better primary care patient experience among physician practices. Deliberate efforts to use clinician performance information in ways that cultivate clinicians' intrinsic motivation may be especially effective for quality improvement.

Background: Multilevel intervention research holds the promise of more accurately representing real-life situations and, thus, with proper research design and measurement approaches, facilitating effective and efficient resolution of health-care system challenges. However, taking a multilevel approach to cancer care interventions creates both measurement challenges and opportunities., Methods: One-thousand seventy two cancer care articles from 2005 to 2010 were reviewed to examine the state of measurement in the multilevel intervention cancer care literature. Ultimately, 234 multilevel articles, 40 involving cancer care interventions, were identified. Additionally, literature from health services, social psychology, and organizational behavior was reviewed to identify measures that might be useful in multilevel intervention research., Results: The vast majority of measures used in multilevel cancer intervention studies were individual level measures. Group-, organization-, and community-level measures were rarely used. Discussion of the independence, validity, and reliability of measures was scant., Discussion: Measurement issues may be especially complex when conducting multilevel intervention research. Measurement considerations that are associated with multilevel intervention research include those related to independence, reliability, validity, sample size, and power. Furthermore, multilevel intervention research requires identification of key constructs and measures by level and consideration of interactions within and across levels. Thus, multilevel intervention research benefits from thoughtful theory-driven planning and design, an interdisciplinary approach, and mixed methods measurement and analysis.