Preventing Contractions in Neuroleptic Malignant Syndrome and Dystonia

To the Editor: We noted with interest the recent discussion by Bridget Craddock, M.D., and Nick Craddock, M.D., Ph.D., of contractures in patients with neuroleptic malignant syndrome (1). The authors highlighted the importance of this complication, the need for prophylactic physical therapy, and the difficulty of management with physical therapy alone.

We recently treated a similar patient and wish to remind the readers of the Journal of an adjunctive treatment.

Ms. A, a 57-year-old woman with DSM-IV schizoaffective disorder, was seen in consultation after she developed fever, tremor, rigidity, and confusion. She had received intramuscular haloperidol decanoate, 100 mg, both 2 and 5 weeks earlier, plus oral haloperidol, which had been discontinued 1 weeks earlier. A week after the first injection, Ms. A developed severe parkinsonism (akinesia, bradykinesia, 4–5-H resting tremor, rigidity, short shuffling steps, and impaired postural reflexes); severe generalized dystonia (forced conjugate downgaze, marked neck flexion, and marked plantar flexion and inversion at the ankles); and neuroleptic malignant syndrome (confusion, catalepsy, palilalia, marked diaphoresis, marked fluctuations in blood pressure and pulse, and intermittent fever to 39.5ºC with numerous negative cultures). She also had bilateral grasp and palmomental reflexes. Serum creatine kinase was 1252 U/liter 2 weeks after admission, without recent intramuscular injections. White blood cell count was normal.

Initial treatment for Ms. A’s movement disorder included neuroleptic discontinuation, anticholinergics (stopped because of urinary retention) and oral lorazepam (stopped because of worsened confusion). She was then treated with intravenous hydration, subcutaneous heparin, ECT, and oral bromocriptine, 10 mg t.i.d. (later replaced by levodopa). However, dystonia was still severe; passive dorsiflexion of the ankles with substantial effort could reach only about 90º.

Physical therapy was begun, and Ms. A was also treated with 100 units of botulinum toxin A injected into each gastrocnemius under electromyographic guidance. Effort required for passive range of motion of the ankles substantially decreased, and the severity of her lower extremity dystonia began to decrease within a few days after the injections. She continued to improve over the next several weeks and had no contracture or symptomatic weakness at discharge.

Ms. A met research criteria for neuroleptic malignant syndrome, complicated by prominent parkinsonian and dystonic features (2). Her prolonged course is attributable to the long effective half-life of the depot neuroleptic (2). We wish to highlight the availability of botulinum toxin for treatment of dystonia. Botulinum toxin injections are recognized as first-line treatment for several focal dystonias, although utility for limb dystonia (as in Ms. A) is less thoroughly studied (3). Although botulinum toxin therapy requires substantial clinical expertise, it offers an additional therapeutic option in preventing contracture associated with neuroleptic-induced dystonia or neuroleptic malignant syndrome.