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. 2021 Jul 2;16(1):294.
doi: 10.1186/s13023-021-01883-5.

Economic burden and health-related quality of life in tenosynovial giant-cell tumour patients in Europe: an observational disease registry

J Lopez-Bastida  1 , I Aranda-Reneo  2 , B Rodríguez-Sánchez  3 , L M Peña-Longobardo  4 , X Ye  5 , P Laeis  6 , E M Fronk  6 , E Palmerini  7 , A Leithner  8 , M A J Van de Sande  9
Affiliations

Economic burden and health-related quality of life in tenosynovial giant-cell tumour patients in Europe: an observational disease registry

J Lopez-Bastida et al. Orphanet J Rare Dis. .

Abstract

Background: Tenosynovial Giant-Cell Tumour (TGCT) is a benign clonal neoplastic proliferation arising from the synovium, causing a variety of symptoms and often requiring repetitive surgery. This study aims to define the economic burden-from a societal perspective-associated with TGCT patients and their health-related quality of life (HRQOL) in six European countries.

Methods: This article analyses data from a multinational, multicentre, prospective observational registry, the TGCT Observational Platform Project (TOPP), involving hospitals and tertiary sarcoma centres from six European countries (Austria, France, Germany, Italy, the Netherlands, and Spain). It includes information on TGCT patients' health-related quality of life and healthcare and non-healthcare resources used at baseline (the 12-month period prior to the patients entering the registry) and after 12 months of follow-up.

Results: 146 TGCT patients enrolled for the study, of which 137 fulfilled the inclusion criteria. Their mean age was 44.5 years, and 62% were female. The annual average total costs associated with TGCT were €4866 at baseline and €5160 at the 12-month follow-up visit. The annual average healthcare costs associated with TGCT were €4620 at baseline, of which 67% and 18% corresponded to surgery and medical visits, respectively. At the 12-month follow-up, the mean healthcare costs amounted to €5094, with surgery representing 70% of total costs. Loss of productivity represented, on average, 5% of the total cost at baseline and 1.3% at follow-up. The most-affected HRQOL dimensions, measured with the EQ-5D-5L instrument, were pain or discomfort, mobility, and the performance of usual activities, both at baseline and at the follow-up visit. Regarding HRQOL, patients declared a mean index score of 0.75 at baseline and 0.76 at the 12-month follow-up.

Conclusion: The results suggest that TGCT places a heavy burden on its sufferers, which increases after one year of follow-up, mainly due to the healthcare resources required-in particular, surgical procedures. As a result, this condition has a high economic impact on healthcare budgets, while the HRQOL of TGCT patients substantially deteriorates over time.

Keywords: Cost-of-illness; Economic burden; Europe; Health-related quality of life; Informal care; Productivity loss; Tenosynovial giant-cell tumour (TGCT).

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Conflict of interest statement

IAR has received grants by Daiichi-Sankyo, Biogen and Lilly. LPL has received grants by Daiichi-Sankyo and Biogen. JLB has received grants by Daiichi-Sankyo, Biogen, Lilly. EP has served on an advisory board for Takeda, Amgen, Daiichi Sankyo, Lilly, Eusa Pharma, and Deciphera, has received other research support from Bristol Myers Squibb, Pfizer, PharmaMar, and Daiichi Sankyo, and travel support from Lilly, PharmaMar, Takeda. AL reports institutional education grants by Johnson & Johnson, Alphamed, Globus, and Implantec. MvS reports institutional grants by Daiichi-Sankyo. YX, LP, and FE are employees of Daiichi-Sankyo.

Figures

Fig. 1
Fig. 1
Share of each cost category out of the total cost at baseline. Units: percentage; Note: the percentage represents the weight of each cost category out of the total cost. GP = general practitioner; MRI: Magnetic Resonance Imaging; TGCT: Tenosynovial Giant-Cell Tumour
Fig. 2
Fig. 2
Share of each cost category out of the total cost, at the 12-months visit. Units: percentage; Note: the percentage represents the weight of each cost category out of the total cost. GP = general practitioner; MRI: Magnetic Resonance Imaging; TGCT: Tenosynovial Giant-Cell Tumour
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