%0 Journal Article
%@ 1929-073X
%I JMIR Publications
%V 7
%N 1
%P e4
%T The Patient Perspective on the Impact of Tenosynovial Giant Cell Tumors on Daily Living: Crowdsourcing Study on Physical Function and Quality of Life
%A Mastboom,Monique Josephine
%A Planje,Rosa
%A van de Sande,Michiel Adreanus
%+ Department of Orthopedics, Leiden University Medical Center, University of Leiden, Albinusdreef 2, Leiden, 2333 ZA, Netherlands, 31 715264088, mjlmastboom@lumc.nl
%K synovitis
%K pigmented villonodular
%K giant cell tumor of tendon sheath
%K rare diseases
%K crowdsourcing
%K social media
%K patient-reported outcome measures
%K quality of life
%K health-related quality of life
%K social participation
%K surveys and questionnaires
%D 2018
%7 23.02.2018
%9 Original Paper
%J Interact J Med Res
%G English
%X Background: Tenosynovial giant cell tumor (TGCT) is a rare, benign lesion affecting the synovial lining of joints, bursae, and tendon sheaths. It is generally characterized as a locally aggressive and often recurring tumor. A distinction is made between localized- and diffuse-type. The impact of TGCT on daily living is currently ill-described. Objective: The aim of this crowdsourcing study was to evaluate the impact of TGCT on physical function, daily activities, societal participation (work, sports, and hobbies), and overall quality of life from a patient perspective. The secondary aim was to define risk factors for deteriorated outcome in TGCT. Methods: Members of the largest known TGCT Facebook community, PVNS is Pants!!, were invited to an e-survey, partially consisting of validated questionnaires, for 6 months. To confirm disease presence and TGCT-type, patients were requested to share histological or radiological proof of TGCT. Unpaired t tests and chi-square tests were used to compare groups with and without proof and to define risk factors for deteriorated outcome. Results: Three hundred thirty-seven questionnaires, originating from 30 countries, were completed. Median age at diagnosis was 33 (interquartile range [IQR]=25-42) years, majority was female (79.8% [269/337]), diffuse TGCT (70.3% [237/337]), and affected lower extremities (knee 70.9% [239/337] and hip 9.5% [32/337]). In 299 lower-extremity TGCT patients (32.4% [97/299]) with disease confirmation, recurrence rate was 36% and 69.5% in localized and diffuse type, respectively. For both types, pain and swelling decreased after treatment; in contrast, stiffness and range of motion worsened. Patients were limited in their employment (localized 13% [8/61]; diffuse 11.0% [21/191]) and sport-activities (localized 58% [40/69]; diffuse 63.9% [147/230]). Compared with general US population, all patients showed lower Patient-Reported Outcomes Measurements Information System-Physical Function (PROMIS-PF), Short Form-12 (SF-12), and EuroQoL 5 Dimensions 5 Levels (EQ5D-5L) scores, considered clinically relevant, according to estimated minimal important difference (MID). Diffuse versus localized type scored almost 0.5 standard deviation lower for PROMIS-PF (P<.001) and demonstrated a utility score of 5% lower for EQ-5D-5L (P=.03). In localized TGCT, recurrent disease and ≥2 surgeries negatively influenced scores of Visual Analog Scale (VAS)-pain/stiffness, SF-12, and EQ-5D-5L (P<.05). In diffuse type, recurrence resulted in lower score for VAS, PROMIS-PF, SF-12, and EQ-5D-5L (P<.05). In both types, patients with treatment ≤1year had significantly lower SF-12. Conclusions: TGCT has a major impact on daily living in a relatively young and working population. Patients with diffuse type, recurrent disease, and ≥2 surgeries represent lowest functional and quality of life outcomes. Physicians should be aware that TGCT patients frequently continue to experience declined health-related quality of life and physical function and often remain limited in daily life, even after treatment(s). 
%M 29475829
%R 10.2196/ijmr.9325
%U http://www.i-jmr.org/2018/1/e4/
%U https://doi.org/10.2196/ijmr.9325
%U http://www.ncbi.nlm.nih.gov/pubmed/29475829