Transdermal opioid patch in treatment of paroxysmal autonomic instability with dystonia with multiple cerebral insults

Abstract

Rationale: Paroxysmal autonomic instability with dystonia (PAID) is an underdiagnosed syndrome that describes a collection of symptoms following diverse cerebral insults, such as traumatic brain injury, hydrocephalus, hemorrhagic stroke, or brain anoxia. It is manifested by systemic high blood pressure, hyperthermia, tachycardia, tachypnea, diaphoresis, intermittent agitation, and certain forms of dystonia. Patient concerns: A semi-comatose 46-year-old man was transferred from the regional rehabilitation hospital with various complaints involving fluctuating vital signs, including uncontrolled hyperthermia, hypertension, tachycardia, and tachypnea, and dystonia in all extremities. The patient underwent brain surgery for astrocytoma in 1996. The patient also had a history of first ischemic stroke on the basal ganglia in 2008 and a second one in the same area in 2017. Diagnosis: The laboratory, electrocardiography, and radiologic findings were normal. Brain imaging indicated an old infarction on the basal ganglia with hydrocephalus. Tractography using diffusion tensor imaging showed discontinuity of multiple tracts, and electrophysiologic tests, such as evoked potentials, displayed an absent response. Based on the dysautonomic symptoms and brain evaluations, the physiatrist diagnosed the patient with PAID. Interventions: Bromocriptine, propranolol, and clonazepam were administered sequentially, but autonomic instability persisted. Then, intravenous opioid was administered, and fluctuations in body temperature, heart rate, and respiratory rate, as well as decerebrate-type dystonia were improved. However, simultaneously, drug-induced severe hypotension developed (systolic blood pressure, 57 mm Hg). Subsequently, a transdermal opioid (fentanyl) patch for PAID was applied once every 3 days. Outcomes: Ultimately, all vital signs and dystonia were managed without further complications, and the patient was discharged. Lessons: A patient diagnosed with PAID following multiple cerebral insults was observed, whose condition was controlled by application of opioid patch rather than by intravenous or oral routes. A transdermal opioid patch, such as fentanyl patch, can thus be effective in the treatment of patients with PAID following multiple cerebral insults.

Case Report: Paroxysmal autonomic instability with dystonia syndrome: a rare complication of tuberculous meningitis

“…6 It is commonly observed after a severe traumatic brain injury (TBI) (79.4%); occurring in almost one-third of all cases of TBI but may also arise from paraneoplastic limbic encephalopathy (9.7%), cerebrovascular disorders (5.4%), hydrocephalus (2.6%), tumor (0.6%), and rarely after central nervous system infections (0.3%) like in pneumococcal meningoencephalitis and tuberculous meningitis, as in our case. 2,4,7 The pathophysiology of PAIDS is still not well understood but likely best explained by the Disconnection Theory, wherein a brain injury causes dysfunction of autonomic centers in the diencephalon. 5 Due to the disconnection of higher • Improvement of neurologic status and was neurologically stable after 3 months of follow-up sympathetic inhibitory regions (e.g.…”