Supplementary material to "Healthcare resource use associated with tumor-induced osteomalacia: a literature review"

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 non-specific and unrecognized,
leading to long delays in diagnosis and treatment, which results in severe
and progressive disability in patients with TIO. This review aimed to
identify published evidence on healthcare resource use in TIO to better
understand the burden of the disease. A targeted literature
review was conducted to identify publications reporting on disease
characteristics and healthcare resource use associated with
TIO. 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 (standard deviation) age of 46.3
(15.8) years; 57.6% were male. The 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%. The
fracture was reported in 49.3% of patients. Results from association
analyses demonstrated that a 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. 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.