Summary
Thymic neuro endocrine tumor (tNET) are extremely rare malignancies with poor prognosis, requiring investigation of novel therapeutic approaches. 177Lu-DOTATATE is a successful systemic treatment modality in patients with metastatic gastroenteropancreatic but it role in tNET is not yet well established. Here we report a case of a 39-year-old man with refractory bone marrow infiltration of a tNET, treated by 4 cycles of peptide receptor radionuclide therapy (PRRT) with 177Lu DOTATATE. Since the first cycle, clinical symptoms were substantially decreased, without any severe subacute haematological toxicity. Three months after the end of PRRT, both 68Ga-DOTATOC and 18F-FDG PET confirmed a partial response, already suggested by 177Lu-DOTATATE treatment scan with a significant decrease of the bone marrow uptake between the first and fourth cycle. This report highlights that PRRT could be an effective therapeutic option for advanced bone metastatic disease tNET, with the significant benefit of alleviation of bone pain and radiologic response, without severe or irreversible haematotoxicity.
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Filosso PL, Yao X, Ahmad U, Zhan Y, Huang J, Ruffini E, Travis W, Lucchi M, Rimner A, Antonicelli A, Guerrera F, Detterbeck F, European Society of Thoracic Surgeons Thymic Group Steering C (2015) Outcome of primary neuroendocrine tumors of the thymus: a joint analysis of the international Thymic malignancy interest group and the European Society of Thoracic Surgeons databases. J Thorac Cardiovasc Surg 149(1):103–109 e102. https://doi.org/10.1016/j.jtcvs.2014.08.061
Strosberg J, El-Haddad G, Wolin E, Hendifar A, Yao J, Chasen B, Mittra E, Kunz PL, Kulke MH, Jacene H, Bushnell D, O’Dorisio TM, Baum RP, Kulkarni HR, Caplin M, Lebtahi R, Hobday T, Delpassand E, Van Cutsem E, Benson A, Srirajaskanthan R, Pavel M, Mora J, Berlin J, Grande E, Reed N, Seregni E, Oberg K, Lopera Sierra M, Santoro P, Thevenet T, Erion JL, Ruszniewski P, Kwekkeboom D, Krenning E, Investigators N-T (2017) Phase 3 trial of (177)Lu-Dotatate for Midgut neuroendocrine tumors. N Engl J Med 376(2):125–135. https://doi.org/10.1056/NEJMoa1607427
Brabander T, van der Zwan WA, Teunissen JJM, Kam BLR, Feelders RA, de Herder WW, van Eijck CHJ, Franssen GJH, Krenning EP, Kwekkeboom DJ (2017) Long-Term Efficacy, Survival, and Safety of [(177)Lu-DOTA(0),Tyr(3)]octreotate in Patients with Gastroenteropancreatic and Bronchial Neuroendocrine Tumors. Clin Cancer Res 23(16):4617–4624. https://doi.org/10.1158/1078-0432.CCR-16-2743
Bergsma H, Konijnenberg MW, Kam BL, Teunissen JJ, Kooij PP, de Herder WW, Franssen GJ, van Eijck CH, Krenning EP, Kwekkeboom DJ (2016) Subacute haematotoxicity after PRRT with (177)Lu-DOTA-octreotate: prognostic factors, incidence and course. Eur J Nucl Med Mol Imaging 43(3):453–463. https://doi.org/10.1007/s00259-015-3193-4
Sabet A, Khalaf F, Haslerud T, Al-Zreiqat A, Sabet A, Simon B, Poppel TD, Biersack HJ, Ezziddin S (2013) Bone metastases in GEP-NET: response and long-term outcome after PRRT from a follow-up analysis. Am J Nucl Med Mol Imaging 3(5):437–445
Sabet A, Khalaf F, Yong-Hing CJ, Sabet A, Haslerud T, Ahmadzadehfar H, Guhlke S, Grunwald F, Biersack HJ, Ezziddin S (2014) Can peptide receptor radionuclide therapy be safely applied in florid bone metastases? A pilot analysis of late stage osseous involvement. Nuklearmedizin 53(2):54–59. https://doi.org/10.3413/Nukmed-0614-13-08
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Cottereau, A.S., Bricaire, L., Arrondeau, J. et al. Prolonged response to 177Lu-DOTATATE therapy of a bone marrow infiltration in a refractory thymic neuro endocrine tumor. Invest New Drugs 38, 1196–1199 (2020). https://doi.org/10.1007/s10637-019-00865-6
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DOI: https://doi.org/10.1007/s10637-019-00865-6