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Letters to the Editors

Acute Laryngeal Dystonia Associated With Aripiprazole

Goga, Joshana K. PharmD, BCPP; Seidel, Laura MD; Walters, J. Ken PharmD; Khushalani, Sunil MD; Kaplan, Desmond MD

Author Information
Journal of Clinical Psychopharmacology: December 2012 - Volume 32 - Issue 6 - p 837-839
doi: 10.1097/JCP.0b013e318272d468
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To the Editors

An acute dystonic reaction presents as a sudden painful muscular spasm forming twisting abnormal postures. One form of this reaction is laryngeal dystonia with involuntary contractions of the vocal cords. A wide variety of symptoms may develop including interruptions in speech to life-threatening complications of asphyxia.1–4

Approximately 3% to 10% of patients exposed to neuroleptics will experience an acute dystonic reaction.5–7 Haloperidol and fluphenazine long-acting injections have the highest incidence of these reactions. In comparison, novel neuroleptics seem to pose a lower risk of acute dystonia although several cases have been reported.6–11 The risk of acute laryngeal dystonia associated with the use of neuroleptics seems to be rare, with only 1 case report in the use of ziprasidone.6

Fifty percent of dystonic reactions occur within 48 hours of initiation or an increase in dose of the neuroleptic, whereas 90% occur within 5 days.1–7 Additional risk factors include family history of dystonia, recent history of cocaine or alcohol use, or treatment with a potent intravenous dopamine D2 receptor antagonist (eg, fluphenazine or haloperidol). These reactions are more common in children, teenagers, and young male adults.1–7 The authors report the first case of acute laryngeal dystonia with the use of aripiprazole in an adolescent female.

A 16-year-old white female patient was admitted to a private acute care psychiatric hospital for symptoms of aggression exacerbated by an altercation at school in which she struck a peer. Her chief complaints upon admission were, “I am emotionally upset. I feel unsafe. I don’t really know if I want to hurt myself and I don’t know if I want to hurt this girl at school.” Her feelings of helplessness and worthlessness peaked before admission since having intercourse with several men.

On assessment, she presented with decreased concentration and attention; sad mood; loss of interest in pleasure; feelings of hopelessness, worthlessness, and guilt; suicidal ideation with crying spells; irritable mood; flashbacks; intrusive re-experiencing; nightmares; hypervigilance; avoidance; aggressive behaviors in the form of yelling and threatening others; anger; compulsive cutting; and self-mutilation.

One of her stressors included a recent release from an outpatient residential treatment center. The patient had been a resident at this center for 8 months in 2009 and was discharged home for 3 months and required inpatient psychiatric admission in January 2010 for aggressive behavior, which led to readmission to the residential treatment center until 1 week before this admission. The course of her illness has been chronic, with acute exacerbations leading to hospitalizations such as this one.

Her previous diagnoses include depression, bipolar I or II, anxiety disorder, posttraumatic stress disorder, with a history of rape 1 year ago and sexual molestation when she was 7 years old, and attention-deficit/hyperactivity disorder. Her treating diagnosis at the time of admission included bipolar disorder not otherwise specified with borderline traits.

She has had trials of several medications for the management of her symptoms, including lithium, sertraline, buproprion, and quetiapine complicated by adverse effects of weight gain and chest pain with lithium, oversedation with quetiapine and unknown reactions with the other agents leading to their discontinuation. Four months before this admission, aripiprazole, 5 mg, had been added to topiramate, 25 mg twice daily, for mood lability and naproxen, 375 mg twice daily, for pain after cutting attempts. Three days before this admission, aripiprazole was increased to 10 mg daily.

Her preadmission laboratory work was within normal limits. Her last normal menstrual period was 14 days before admission. She presented with old scars from self-inflicted wounds on the left arm. She denied any pain at time of admission. A complete blood cell count showed all values within normal limits. The result of a pregnancy test was negative. Alcohol toxicology report was negative. A comprehensive metabolic panel showed a low value for potassium of 3.4. All other values were within normal limits. Urinalysis results showed all values within normal limits. A urine toxicology report showed none detected. Her temperature was 95.3°F, orally; pulse rate, 80 beats per minute; respirations, 16 per minute; and blood pressure, 121/70 mm Hg. The patient’s height was 68 inches and her weight was 157 lbs.

During the third day of hospitalization, the patient was found on the gymnasium floor. On assessment by medical staff, she presented with dyspnea, dysphonia, tongue and throat tightening, cogwheel rigidity, and dizziness. Her vitals included blood pressure of 118/74 mm Hg, heart rate of 100 beats per minute, and variations in her oxygen saturation from 92% to 100%. Her temperature was normal, and all laboratory values were unremarkable except for sodium of 148 meq/L. She was alert and oriented. Aripiprazole was discontinued; and Stat orders of benzotropine, 2 mg intramuscular, with diazepam, 5 mg oral, were administered. Symptoms of cogwheel rigidity and shortness of breath improved quickly after intramuscular medication administration, and symptoms of chest pain improved over 3 days with the aid of oral benztropine, 1 mg every 4 hours, as needed. Recurrence of symptoms was not noted.

The patient’s symptoms are congruent to those that have been reported in past cases of acute laryngeal dystonia.12–19 Previous cases have described dyspnea; extreme subjective distress, such as clutching the throat; dystonias involving muscles of the head and neck, including torticollis, retrocollis, tongue and jaw stiffness, oculogyric crisis, and opisthotonus. Neuroleptic malignant syndrome can be ruled out in the patient with the absence of fever, change in vital signs, change in the level of consciousness combined with rapid relief of symptoms seen after anticholinergic administration. The patient’s age and the dose increase of aripiprazole from 5 mg to 10 mg just before admission seem to be her main risk factors for the presentation of acute dystonia. There are no significant pharmacokinetic drug interactions between topiramate, aripiprazole, and naproxen. Pharmacodynamic interactions may include increased sedation.

One of the main limitations of this case report includes the complexity of her diagnosis. The possibility of a psychogenic etiology of the patient’s acute presentation on the gymnasium floor cannot be ruled out given her diagnosis of borderline traits.

Acute laryngeal dystonia is a potentially lethal adverse effect of one of the most commonly prescribed drug classes in psychiatry. The true incidence likely has been underreported especially with novel neuroleptics. This is the second report of acute laryngeal dystonia with novel neuroleptics and the first with aripiprazole. Early recognition of laryngeal dystonia associated with neuroleptic medications can prevent life-threatening complications. Educating medical staff to this easily treatable reaction will improve overall quality of health care. This case introduces the need for awareness of the risk of acute laryngeal dystonia in adolescent female patients receiving aripiprazole.

Joshana K. Goga, PharmD, BCPP

Sheppard Pratt Health System

University of Maryland School of Pharmacy

Towson, MD

[email protected]

Laura Seidel, MD

Sheppard Pratt Health System

Towson, MD

J. Ken Walters, PharmD

Sheppard Pratt Health System

University of Maryland School of Pharmacy

Towson, MD

Sunil Khushalani, MD

Desmond Kaplan, MD

Sheppard Pratt Health System

Towson, MD

AUTHOR DISCLOSURE INFORMATION

The authors declare no conflicts of interest.

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