Cervical Castleman's disease associated with benign M-proteinemia: report of a case

We report the unusual case of a 77-year-old man with cervical Castleman's disease associated with benign M-proteinemia. The patient was found to have an anterior cervical tumor during a follow-up examination after surgery for early gastric cancer, at which time blood biochemistry tests revealed M-proteinemia at the position of beta-globulin with a high level of IgG and low levels of IgA and IgM. Serum protein immunoelectrophoresis revealed M-protein consisting of the IgG-k chain, and urine protein immuno-electrophoresis demonstrated Bence Jones protein consisting of the chain. No abnormalities were noted in bone marrow aspiration or bone scintigram. The results of imaging and fine-needle aspiration biopsy of the tumor led to a pre-operative diagnosis of cervical malignant lymphoma with suspected M-proteinemia derived from this disease, and resection of the anterior cervical tumor with bilateral neck dissection was performed. Histopathological examination subsequently demonstrated four mixed-type tumors of Castleman's disease. The pathogenesis of Castleman's disease remains unknown; however, we speculate that the complication of benign M-proteinemia in this patient was not incidental, but caused by an underlying immunological abnormality of the B cells.