Hydromorphone-induced choreiform dyskinesia in a patient with homozygous sickle cell disease: a reversible adverse effect.

Background: Sickle Cell Disease (SCD) is a type of chronic hemoglobinopathy with vaso-occlusive pain crises that requires aggressive pain management with opioids.Hydromorphone is commonly used in the management of pain in patients with SCD, particularly when vaso-occlusive crises require prolonged opioid therapy. While opioid-induced neurotoxicity is a well-recognized complication, usually presenting as delirium, agitation, or myoclonus, choreiform dyskinesias are rare and only infrequently reported. New-onset involuntary movements in patients with SCD may be concerning for cerebrovascular or metabolic complications, prompting extensive diagnostic evaluations. Recognition of opioid-induced choreiform dyskinesia is important, since symptoms are usually reversible with opioid dose reduction or discontinuation, thus offering early management and the avoidance of unnecessary investigations. Case Report: A 33-year-old African American woman with homozygous sickle cell disease (HbSS) was admitted for pleuritic chest pain, fever, abdominal pain, and sweating, suspected to be acute chest syndrome or a vaso-occlusive pain crisis. She had a history of recurrent vaso-occlusive crises requiring hospitalizations and opioid therapy. She had no history of cerebrovascular events, movement disorders, psychiatric illness, or exposure to dopamine-modulating medications. She had no significant family history.On day 25 of hospitalization, she developed new, involuntary, non-rhythmic choreiform movements affecting the upper limbs and face while on high-dose IV hydromorphone. She remained alert and oriented without any focal neurologic signs or metabolic issues. The hydromorphone was stopped, and the movements completely resolved within 24 hours. When trying a lower dose, the movements did not recur. Neurology advised that no further imaging or testing was needed given the rapid, full improvement. Conclusions: Dyskinesia is a rare but reversible complication of opioid therapy and may be caused by hydromorphone. It is important to recognize this side effect to prevent unnecessary testing, particularly in patients with sickle cell disease, who may otherwise undergo an extensive neurologic workup.