Priapism and pyrexia of unknown origin presenting as a relapse of multiple myeloma

Abstract

Pyrexia of unknown origin (PUO) is defined as a body temperature >38.3°C on several occasions for >3 weeks with failure of diagnosis despite one week of inpatient investigations or three outpatient visits. Aetiology for PUO may be infective, inflammatory, autoimmune, malignant, miscellaneous or remains undiagnosed. We describe a patient diagnosed with multiple myeloma (ISS-stage II) who completed induction chemotherapy and presented with PUO and extensive thrombosis as a complication of relapsed myeloma (very rare; 0.2% in myeloma). A 62-year-old man, with diagnosed multiple myeloma, achieved a very good partial remission after 6 cycles of bortezomib-thalidomide-dexamethasone regimen, presented with PUO and right lower limb deep vein thrombosis. His thrombosis was managed successfully with anticoagulation. While on therapeutic anticoagulation with warfarin and being investigated extensively for PUO without a focus, he developed priapism due to bilateral internal pudendal vein thrombosis. His anticoagulation was intensified to a target INR of 2.5-3.5. He recovered without residual damage. Despite broad spectrum antimicrobial agents, trial of antimalarial and anti-tuberculosis treatment, he remained pyrexial with rising inflammatory markers (ESR, CRP). Relapsed multiple myeloma was confirmed with 30% bone marrow plasma cells and raised serum monoclonal para-protein levels of 23.78g/L. He was restarted on chemotherapy with bortezomib-lenalidomide-dexamethasone. His PUO of two months subsided with one week of chemotherapy.
 This case illustrates an atypical presentation of relapsed myeloma and highlights the importance of achieving early diagnosis with a systematic approach and making optimum treatment choice after careful evaluation of previous treatment, drug toxicities, patient’s condition and available treatment options.