M J L Mastboom1, F G M Verspoor2, D F Hanff3, M G J Gademan4, P D S Dijkstra5, H W B Schreuder6, J L Bloem7, R J P van der Wal8, M A J van de Sande9. 1. Orthopaedic Surgery, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: mjlmastboom@lumc.nl. 2. Orthopaedic Surgery, Radboud University Medical Center, Nijmegen, the Netherlands. Electronic address: floorverspoor@gmail.com. 3. Radiology, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: d.f.hanff@lumc.nl. 4. Epidemiology, Clinical Epidemiology, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: m.g.j.gademan@lumc.nl. 5. Orthopaedic Surgery, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: p.d.s.dijkstra@lumc.nl. 6. Orthopaedic Surgery, Radboud University Medical Center, Nijmegen, the Netherlands. Electronic address: bart.schreuder@radboudumc.nl. 7. Radiology, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: j.l.bloem@lumc.nl. 8. Orthopaedic Surgery, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: r.j.p.van_der_wal@lumc.nl. 9. Orthopaedic Surgery, Leiden University Medical Center, Leiden, the Netherlands. Electronic address: majvandesande@lumc.nl.
Abstract
AIM: Current development of novel systemic agents requires identification and monitoring of extensive Tenosynovial Giant Cell Tumours (TGCT). This study defines TGCT extension on MR imaging to classify severity. METHODS: In part one, six MR parameters were defined by field-experts to assess disease extension on MR images: type of TGCT, articular involvement, cartilage-covered bone invasion, and involvement of muscular/tendinous tissue, ligaments or neurovascular structures. Inter- and intra-rater agreement were calculated using 118 TGCT MR scans. In part two, the previously defined MR parameters were evaluated in 174 consecutive, not previously used, MR-scans. TGCT severity classification was established based on highest to lowest Hazard Ratios (HR) on first recurrence. RESULTS: In part one, all MR parameters showed good inter- and intra-rater agreement (Kappa≥0.66). In part two, cartilage-covered bone invasion and neurovascular involvement were rarely appreciated (<13%) and therefore excluded for additional analyses. Univariate analyses for recurrent disease yielded positive associations for type of TGCT HR12.84(95%CI4.60-35.81), articular involvement HR6.00(95%CI2.14-16.80), muscular/tendinous tissue involvement HR3.50(95%CI1.75-7.01) and ligament-involvement HR4.59(95%CI2.23-9.46). With these, a TGCT severity classification was constructed with four distinct severity-stages. Recurrence free survival at 4 years (log rank p < 0.0001) was 94% in mild localized (n56, 1 recurrence), 88% in severe localized (n31, 3 recurrences), 59% in moderate diffuse (n32, 12 recurrences) and 36% in severe diffuse (n55, 33 recurrences). CONCLUSION: The proposed TGCT severity classification informs physicians and patients on disease extent and risk for recurrence after surgical treatment. Definition of the most severe subgroup attributes to a universal identification of eligible patients for systemic therapy or trials for novel agents.
AIM: Current development of novel systemic agents requires identification and monitoring of extensive Tenosynovial Giant Cell Tumours (TGCT). This study defines TGCT extension on MR imaging to classify severity. METHODS: In part one, six MR parameters were defined by field-experts to assess disease extension on MR images: type of TGCT, articular involvement, cartilage-covered bone invasion, and involvement of muscular/tendinous tissue, ligaments or neurovascular structures. Inter- and intra-rater agreement were calculated using 118 TGCT MR scans. In part two, the previously defined MR parameters were evaluated in 174 consecutive, not previously used, MR-scans. TGCT severity classification was established based on highest to lowest Hazard Ratios (HR) on first recurrence. RESULTS: In part one, all MR parameters showed good inter- and intra-rater agreement (Kappa≥0.66). In part two, cartilage-covered bone invasion and neurovascular involvement were rarely appreciated (<13%) and therefore excluded for additional analyses. Univariate analyses for recurrent disease yielded positive associations for type of TGCT HR12.84(95%CI4.60-35.81), articular involvement HR6.00(95%CI2.14-16.80), muscular/tendinous tissue involvement HR3.50(95%CI1.75-7.01) and ligament-involvement HR4.59(95%CI2.23-9.46). With these, a TGCT severity classification was constructed with four distinct severity-stages. Recurrence free survival at 4 years (log rank p < 0.0001) was 94% in mild localized (n56, 1 recurrence), 88% in severe localized (n31, 3 recurrences), 59% in moderate diffuse (n32, 12 recurrences) and 36% in severe diffuse (n55, 33 recurrences). CONCLUSION: The proposed TGCT severity classification informs physicians and patients on disease extent and risk for recurrence after surgical treatment. Definition of the most severe subgroup attributes to a universal identification of eligible patients for systemic therapy or trials for novel agents.
Authors: Francisco Manuel Cañete Sánchez; Leonardo Gabriel Romero Robles; Xavier Louis Boulvard Chollet; María Mangas Losada; Puy Garrastachu; Antonio Cabrera Villegas; Rafael Ramírez Lasanta; Roberto Delgado Bolton Journal: Mol Imaging Radionucl Ther Date: 2022-06-27
Authors: F G M Verspoor; M J L Mastboom; G Hannink; R G Maki; A Wagner; E Bompas; J Desai; A Italiano; B M Seddon; W T A van der Graaf; J-Y Blay; M Brahmi; L Eberst; S Stacchiotti; O Mir; M A J van de Sande; H Gelderblom; P A Cassier Journal: Sci Rep Date: 2019-10-10 Impact factor: 4.379
Authors: Nicholas M Bernthal; Geert Spierenburg; John H Healey; Emanuela Palmerini; Sebastian Bauer; Hans Gelderblom; Eric L Staals; Julio Lopez-Bastida; Eva-Maria Fronk; Xin Ye; Petra Laeis; Michiel A J van de Sande Journal: Orphanet J Rare Dis Date: 2021-04-29 Impact factor: 4.123