Evaluation for Kienböck Disease Familial Clustering: A Population-Based Cohort Study

Abstract

Purpose: Kienböck's disease (KD) is rare, and its etiology remains unknown. As a result, the ideal treatment is also in question. Our primary purpose was to test the hypothesis that KD would demonstrate familial clustering in a large statewide population with comprehensive genealogical records, possibly suggesting a genetic etiologic contribution. Our secondary purpose was to evaluate for associations between KD and known risk factors for avascular necrosis.Methods: Patients diagnosed with KD were identified by searching medical records from a comprehensive statewide database, the Utah Population Database (UPDB). This database contains pedigrees dating back to the early 1800's, which are linked to 31 million medical records for 11 million patients dating from 1996 to present. Affected individuals were then mapped to pedigrees to identify high-risk families with an increased incidence of KD relative to control pedigrees. The magnitude of familial risk of KD in related individuals was calculated using Cox regression models. Association of risk factors related to KD was analyzed using conditional logistic regression. Results:We identified 394 affected individuals that are linked to 194 unrelated high-risk pedigrees with increased incidence of KD. The relative risk of developing KD was significantly *

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