Hydroxychloroquine causes severe vacuolar myopathy in a patient with chronic graft-versus-host disease.
A 51-year-old man developed progressive debilitating limb and respiratory muscle weakness while undergoing treatment for chronic graft-versus-host disease secondary to allogeneic bone marrow transplant for mantle cell lymphoma. He had a normal serum creatine kinase level and acetylcholine receptor antibodies were negative. Electromyography showed a severe, nonirritable myopathy and a sensory motor axonal polyneuropathy. A muscle biopsy showed a necrotizing, vacuolar myopathy with many fibers containing autophagic and red-rimmed vacuoles, suggestive of an amphiphilic drug myopathy. The patient's strength and function improved significantly after discontinuation of hydroxychloroquine.
Bolaños-Meade
Javier
J
Department of Oncology, The Johns Hopkins Hospital, Baltimore, Maryland 21287, USA.
Zhou
Lan
L
Hoke
Ahmet
A
Corse
Andrea
A
Vogelsang
Georgia
G
Wagner
Kathryn R
KR
D001706:Biopsy; D016026:Bone Marrow Transplantation / Q000009:adverse effects* / Q000473:pathology; D002908:Chronic Disease; D004791:Enzyme Inhibitors / Q000009:adverse effects; D006086:Graft vs Host Disease / Q000188:drug therapy / Q000473:pathology*; D006801:Humans; D006886:Hydroxychloroquine / Q000009:adverse effects*; D007166:Immunosuppressive Agents / Q000627:therapeutic use; D020522:Lymphoma, Mantle-Cell / Q000601:surgery*; D008297:Male; D008875:Middle Aged; D018482:Muscle, Skeletal / Q000187:drug effects / Q000473:pathology* / Q000648:ultrastructure; D016896:Treatment Outcome; D014617:Vacuoles / Q000187:drug effects / Q000473:pathology* / Q000648:ultrastructure
D004791:Enzyme Inhibitors; D007166:Immunosuppressive Agents; D006886:Hydroxychloroquine