Browsing by Author "Li, Zhiyi"

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    Healthcare Resource Use Associated With Tumor-Induced Osteomalacia: A Literature Review
    (Oxford University Press, 2024) Jan de Beur, Suzanne M.; Dahir, Kathryn M.; Imel, Erik A.; Zanchetta, María Belén; Williams, Angela; Li, Zhiyi; Webb, Neil; Crowe, Victoria; Johnson, Ben; Carpenter, Thomas O.; Medicine, School of Medicine
    Context: Tumor-induced osteomalacia (TIO) is an ultra-rare, paraneoplastic syndrome caused by tumors that secrete fibroblast growth factor 23 (FGF23). Initial signs and musculoskeletal symptoms can be nonspecific and unrecognized, leading to long delays in diagnosis and treatment, and resulting in severe and progressive disability in patients with TIO. Objective: This review aimed to identify published evidence on healthcare resource use in TIO to better understand the burden of the disease. Evidence acquisition: A targeted literature review was conducted to identify publications reporting on disease characteristics and healthcare resource use associated with TIO. Evidence synthesis: In total, 414 publications were included in the review, of which 376 were case reports. From the case reports, data on 621 patients were extracted. These patients had a mean (SD) age of 46.3 (15.8) years; 57.6% were male. Mean time from first symptoms to diagnosis of TIO was 4.6 (4.7) years and, in cases where imaging tests were reported, patients underwent a mean of 4.1 (2.7) procedures. Tumor resection was attempted in 81.0% of patients and successful in 67.0%. Fracture was reported in 49.3% of patients. Results from association analyses demonstrated that longer time to diagnosis was associated with poorer tumor resection outcomes and a higher probability of tumor recurrence. Unfavorable tumor resection outcomes were associated with greater use of pharmacologic treatment and a greater likelihood of orthopedic surgery. Conclusion: TIO is associated with a substantial healthcare resource burden. Improvements in the diagnostic process could lead to better management of TIO, thereby benefiting patients and reducing that burden.
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    Real-World Clinical and Healthcare Resource Burden Among Burosumab-Naïve Patients With Familial Hypophosphatemia
    (Oxford University Press, 2024-10-24) Imel, Erik A.; Li, Zhiyi; Heerssen, Heather M.; Princic, Nicole; Schwartz, Hana; Zhao, Yang; Dahir, Kathryn M.; Medicine, School of Medicine
    Objective: To examine the real-world clinical and healthcare resource burden of familial hypophosphatemia (FH). Methods: In a retrospective, observational cohort study using MarketScan claims data from 2017 to 2021, clinical characteristics and healthcare resource utilization (HCRU) and costs were compared between burosumab-naïve pediatric and adult patients with ≥ 1 FH diagnosis code and matched controls without FH. Patient characteristics were evaluated at baseline, and disease characteristics, HCRU, and costs were evaluated over a 12-month follow-up period. Outcomes were analyzed descriptively. Costs were additionally analyzed using multivariate regression models. Results: Overall, 570 patients with FH and 1710 non-FH matched controls were included. Approximately 10% of study participants were aged < 18 years. Patients with FH had 7.8-fold higher mean baseline comorbidity (Charlson Comorbidity Index). The prevalence of morbidities over the 12-month follow-up period was higher in patients with FH than controls, including renal disease (33% vs 3%), arthralgia (25% vs 10%), osteoarthritis (17% vs 6%), and delayed growth/walking difficulty (16% vs 2%; all P < .001). All-cause HCRU was significantly greater for patients with FH than controls over follow-up, including the proportion of patients with at least one inpatient admission (60% vs 4%), outpatient emergency room visit (52% vs 16%), and outpatient pharmacy prescription (96% vs 71%; all P < .001). The mean annual total healthcare cost per patient was also 22.6-fold higher for patients with FH than controls (adjusted cost difference = $129 643; P < .001). Differences were apparent across all age groups. Conclusion: Compared with non-FH matched controls, burosumab-naïve patients with FH experienced multiple morbidities and had substantially higher HCRU and costs.