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  • Hindi, N.Hospital Fundación Jiménez Díaz,Health Research Institute Jiménez Díaz Foundation (author)

Efficacy of immune checkpoint inhibitors in alveolar soft-part sarcoma : results from a retrospective worldwide registry

  • Article/chapterEnglish2023

Publisher, publication year, extent ...

  • 2023

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  • LIBRIS-ID:oai:lup.lub.lu.se:1ede7563-37b1-4de7-adae-2096dcfb68ba
  • https://lup.lub.lu.se/record/1ede7563-37b1-4de7-adae-2096dcfb68baURI
  • https://doi.org/10.1016/j.esmoop.2023.102045DOI

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  • Language:English
  • Summary in:English

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  • Subject category:art swepub-publicationtype
  • Subject category:ref swepub-contenttype

Notes

  • Background: Conventional cytotoxic drugs are not effective in alveolar soft-part sarcoma (ASPS). Immune checkpoint (programmed cell death protein 1/programmed death-ligand 1) inhibitors (ICIs) are promising drugs in ASPS. A worldwide registry explored the efficacy of ICI in ASPS. Materials and methods: Data from adult patients diagnosed with ASPS and treated with ICI for advanced disease in expert sarcoma centers from Europe, Australia and North America were retrospectively collected, including demographics and data related to treatments and outcome. Results: Seventy-six ASPS patients, with a median age at diagnosis of 25 years (range 3-61 years), were registered. All patients received ICI for metastatic disease. Immunotherapy regimens consisted of monotherapy in 38 patients (50%) and combination in 38 (50%) (23 with a tyrosine kinase inhibitor). Among the 68 assessable patients, there were 3 complete responses and 34 partial responses, translating into an overall response rate of 54.4%. After a median follow-up of 36 months [95% confidence interval (CI) 32-40 months] since the start of immunotherapy, 45 (59%) patients have progressed on ICI, with a median progression-free survival (PFS) of 16.3 months (95% CI 8-25 months). Receiving ICI in first line (P = 0.042) and achieving an objective response (P = 0.043) correlated with a better PFS. Median estimated overall survival (OS) from ICI initiation has not been reached. The 12-month and 24-month OS rates were 94% and 81%, respectively. Conclusions: This registry constitutes the largest available series of ASPS treated with ICI. Our results suggest that the ICI treatment provides long-lasting disease control and prolonged OS in patients with advanced ASPS, an ultra-rare entity with limited active therapeutic options.

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  • Razak, A.Princess Margaret Hospital University of Toronto (author)
  • Rosembaum, E.Memorial Sloan-Kettering Cancer Center (author)
  • Jonczak, E.University of Miami (author)
  • Hamacher, R.University Hospital Essen (author)
  • Rutkowski, P.The Maria Sklodowska-Curie Memorial Cancer Center and Institute of Oncology (author)
  • Bhadri, V. A.Cancer Institute of New South Wales (author)
  • Skryd, A.University of Miami (author)
  • Brahmi, M.Claude Bernard University Lyon 1 (author)
  • Alshibany, A.Princess Margaret Hospital University of Toronto (author)
  • Jagodzinska-Mucha, P.The Maria Sklodowska-Curie Memorial Cancer Center and Institute of Oncology (author)
  • Bauer, S.University Hospital Essen (author)
  • Connolly, EmmaCancer Institute of New South Wales (author)
  • Gelderblom, H.Leiden University Medical Centre (author)
  • Boye, K.Oslo university hospital (author)
  • Henon, C.Institut Gustave Roussy (author)
  • Bae, S.Peter MacCallum Cancer Centre (author)
  • Bogefors, K.Lund University,Lunds universitet,Medicinsk onkologi,Sektion I,Institutionen för kliniska vetenskaper, Lund,Medicinska fakulteten,Lymfom - Klinisk forskning,Forskargrupper vid Lunds universitet,LUCC: Lunds universitets cancercentrum,Övriga starka forskningsmiljöer,Medical oncology,Section I,Department of Clinical Sciences, Lund,Faculty of Medicine,Lymphoma - Clinical Research,Lund University Research Groups,LUCC: Lund University Cancer Centre,Other Strong Research Environments,Skåne University Hospital(Swepub:lu)med-kbf (author)
  • Vincenzi, B.University Campus Bio-Medico (author)
  • Martinez-Trufero, J.Miguel Servet University Hospital (author)
  • Lopez-Martin, J. A.12 de Octubre University Hospital (author)
  • Redondo, A.University Hospital La Paz (author)
  • Valverde, C.Vall d'Hebron University Hospital (author)
  • Blay, J. Y.Claude Bernard University Lyon 1 (author)
  • Moura, D. S.Health Research Institute Jiménez Díaz Foundation (author)
  • Gutierrez, A.Hospital Universitario Son Espases (author)
  • Tap, W.Memorial Sloan-Kettering Cancer Center (author)
  • Martin-Broto, JavierHealth Research Institute Jiménez Díaz Foundation,Hospital Fundación Jiménez Díaz (author)
  • Hospital Fundación Jiménez DíazHealth Research Institute Jiménez Díaz Foundation (creator_code:org_t)

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