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Introduction: In the ICARIA-MM trial, isatuximab plus pomalidomide-dexamethasone (Pd) significantly prolonged progression-free survival, compared with Pd alone, in patients with relapsed or refractory multiple myeloma (RRMM), including those with gain or amplification of chromosome 1q, region 2, band 1 (1q21+). Case Presentation: We describe a 62-year-old Japanese man with 1q21+ RRMM, previously treated with three lines of therapy, who had a rapid and durable response to isatuximab-Pd. After the first isatuximab-Pd cycle, he had a complete response or better, which was maintained throughout 54 cycles (4.25 years). Isatuximab-Pd was generally well tolerated. The only serious AE was grade 3 cataracts. Conclusion: Our case shows that isatuximab-Pd is capable of inducing an enduring response in a patient with 1q21+ RRMM.

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