@Article{info:doi/10.2196/ijmr.9325,
author="Mastboom, Monique Josephine
and Planje, Rosa
and van de Sande, Michiel Adreanus",
title="The Patient Perspective on the Impact of Tenosynovial Giant Cell Tumors on Daily Living: Crowdsourcing Study on Physical Function and Quality of Life",
journal="Interact J Med Res",
year="2018",
month="Feb",
day="23",
volume="7",
number="1",
pages="e4",
keywords="synovitis; pigmented villonodular; giant cell tumor of tendon sheath; rare diseases; crowdsourcing; social media; patient-reported outcome measures; quality of life; health-related quality of life; social participation; surveys and questionnaires",
abstract="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). ",
issn="1929-073X",
doi="10.2196/ijmr.9325",
url="http://www.i-jmr.org/2018/1/e4/",
url="https://doi.org/10.2196/ijmr.9325",
url="http://www.ncbi.nlm.nih.gov/pubmed/29475829"
}