Abstract

BackgroundParaneoplastic hyperleucocytosis (PH) is sporadically seen in patients with advanced solid tumors.Case presentationWe report a female patient with disseminated melanoma metastases. Two days after the first dosage of combined immunotherapy using the cytotoxic T lymphocyte antigen-4 (CTLA-4) blocker ipilimumab and the programmed death receptor-1 (PD-1) blocker nivolumab the patient developed asymptomatic hyperleucocytosis (over 120.000 leucocytes per μl) associated with elevated granulocyte colony-stimulating factor blood levels. Hematological and infectious disorders could be ruled out. Although paraneoplastic hyperleucocytosis spontaneously resolved she died from progressive disease about 60 days after start of treatment.ConclusionsPH is extremely rare in malignant melanoma, however, most patients who developed this complication had preceding immunotherapies such as interleukin-2. The latter observation and the fact that our patient developed PH rapidly after initiation of ipilimumab and nivolumab immunotherapy indicate an immune-mediated mechanism which may trigger PH under unknown circumstances. The development of paraneoplastic hyperleucocytosis indicates a very poor prognosis.

Highlights

  • Paraneoplastic hyperleucocytosis (PH) is sporadically seen in patients with advanced solid tumors.Case presentation: We report a female patient with disseminated melanoma metastases

  • PH is extremely rare in malignant melanoma, most patients who developed this complication had preceding immunotherapies such as interleukin-2

  • Paraneoplastic hyperleucocytosis (PH; leucocytes > 100.000/μl) or paraneoplastic leukemoid reaction is sporadically seen in patients with solid tumors, especially advanced lung cancers

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Summary

Conclusions

PH is a rare complication in melanoma patients usually linked to rapid tumor progress and very limited survival. Case of malignant melanoma that developed the ability to secrete granulocyte colony-stimulating factor. Granulocyte colony-stimulating factor-producing melanoma treated with the combination of dabrafenib and trametinib. 6. Schniewind B, Christgen M, Hauschild A, et al Paraneoplastic leukemoid reaction and rapid progression in a patient with malignant melanoma: establishment of KT293, a novel G-CSF-secreting melanoma cell line. 7. Moon HW, Kim TY, Oh BR, Hwang SM, Kwon J, Ku JL, Lee DS. Effects of granulocyte-colony stimulating factor and the expression of its receptor on various malignant cells. 9. Andoh A, Yasui H, Inatomi O, Zhang Z, Deguchi Y, Hata K, Araki Y, Tsujikawa T, Kitoh K, Kim-Mitsuyama S, Takayanagi A, Shimizu N, Fujiyama Y.

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