Catatonia during deep brain stimulator implantation complicated by intracranial hemorrhage.

Catatonia is a syndrome of motor dysregulation characterized by fluctuating stupor, mutism, negativism, catalepsy, automatic obedience, and stereotypy. Other motor signs include gegenhalten, mitgehen, waxy flexibility, echophenomena, and ambitendency. The diagnosis requires two to four features. The pathophysiology of catatonia remains unclear but may involve dysfunction of frontal circuitry and motor regulatory brain areas. Catatonia is most closely associated with mood disorders but also results from medications (e.g., dopamine blockers and serotonergic agents), metabolic derangement, seizures, or focal structural brain lesions. We present a patient with Parkinson’s disease (PD) who developed catatonia during deep brain stimulation (DBS) surgery complicated by intracranial hemorrhage. The patient was a 60-year-old right-handed woman with levodopa-responsive PD for 12 years. Prior to surgery, she was taking levodopa 800 mg, entacapone 800 mg, and ropinirole 8 mg daily but experienced debilitating motor fluctuations, abrupt ‘‘off’’ periods, and dyskinesias, despite adjustments to her medication regimen. She had a history of nonmelancholic depression treated with citalopram 20 mg daily. During presurgical evaluation, she had no dementia on neuropsychological testing or evidence of an active mood disorder. Off-on Unified Parkinson’s Disease Rating Scale (UPDRS) testing demonstrated at least a 32% improvement to her standard morning levodopa dose. She was deemed an appropriate DBS candidate and underwent surgical placement of DBS electrodes into each subthalamic nucleus using microelectrode recordings. The surgery was conducted in the off medication state, over 12 hours after her last dose of PD medication. Mild hypertension during the procedure was controlled with low doses of metoprolol. Before DBS electrode placement, the patient spoke only when asked questions or to complain of intermittent left arm pain. During right electrode placement, she gradually developed dystonic flexion posturing of the left arm and intermittent flexion of the right arm and left leg. When asked questions or given commands, she was mute. She had gegenhalten in both arms and eyelids upon passive manipulation. She had no motor weakness. Other catatonic features included a protruding grimace (schnauzkrampf), bilateral grasp reflexes, and mitgehen in the right arm. Differential considerations included a worsening of her parkinsonian ‘‘off’’ state versus catatonia. Lorazepam 2 mg was given intravenously. Her Bush-Francis catatonia rating scale score was 24 prior to the injection and decreased to 10 after 4 min. Rigidity decreased and her grimace and mitgehen disappeared. She spoke a few words softly and followed simple commands. These symptoms remained unchanged as the left DBS electrode was placed. Postoperative noncontrast head CT (see Fig. 1) revealed an acute intraparenchymal hematoma in the right frontal lobe measuring 7.0 cm by 2.8 cm. There was a small amount of associated extra-axial blood. There was mild adjacent mass effect with effacement of the cerebral sulci and frontal horn of the right lateral ventricle with subfalcine herniation and associated mild midline shift to the left. She was transferred to neurosurgical intensive care where her Bush-Francis score 4 hours after surgery was 28. Her features of stupor, rigidity, negativism, grimacing, and mitgehen had returned. She was managed supportively for 10 days without change in her examination. A trial of high dose lorazepam or electroconvulsive therapy (ECT) was not pursued. She was then transferred to a subacute care facility closer to home where her subsequent course is unknown. We know of no other cases of catatonia associated with DBS surgery or treatment. In the intraoperative setting, the main differential consideration was whether her exam represented a worsening of her parkinsonian ‘‘off’’ state. Her rigidity and paucity of spontaneous movements were consistent with this diagnosis. However, the mutism, negativism, gegenhalten, and grimacing were inconsistent with previous ‘‘off’’ examinations. The positive lorazepam challenge supported the diagnosis of catatonia. We cannot infer a specific mechanism for the development of catatonia in our patient. Although the frontal lobe hemorrhage is an obvious etiologic candidate, there may have been contributions from multiple factors, including the direct surgical manipulations of basal ganglia regions and the patient’s demographic predisposition to catatonia. Despite her history of PD and mood disorder, she had never been catatonic prior FIG. 1. Postoperative three dimensionally reconstructed image (left) and noncontrast head CT (right). [Color figure can be viewed in the online issue, which is available at www.interscience.wiley.com.]