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Castleman's disease is a lymphoproliferative disease that is rare and hard to diagnose. As a result of untypical manifestations, missed diagnosis and misdiagnosis of Castleman's disease are common in clinical practice. In this paper, we report one patient with clinical manifestations of painless submaxillary lymphadenectasis. The patient was finally diagnosed with Castleman's disease by clinical, laboratory, and pathological examinations. This report may promote the understanding and diagnosis of Castleman's disease for clinicians.