Oral Facial Dystonia (Meige or Brueghel Syndrome) Induced by Paliperidone Palmitate

Oral Facial Dystonia (Meige or Brueghel Syndrome) Induced by Paliperidone Palmitate

To the Editor: Meige syndrome is a focal oral facial dystonia involving symmetrical blepharospasm and oromandibular dystonia. It is also known as Brueghel syndrome and was first described more than 100 years ago. The typical symptom of Meige syndrome is difficulty keeping the eyelids open. This syndrome has been reported to be induced by several atypical antipsychotics, including newer drugs such as perospirone.1 Here, a case is reported of Meige syndrome emerging with the atypical antipsychotic paliperidone. To the author’s knowledge, this is the first report of Meige syndrome induced by paliperidone.

Case report. A 64-year-old woman with a history of schizoaffective disorder (DSM-5 criteria) since age 25 years was admitted for refractory persecutory delusional ideation. She had been on many different combinations of benzodiazepines, antidepressants, mood stabilizers, and antipsychotics in the past with no known extrapyramidal effects. Her personal history included mild head trauma 30 years prior, plus hypothyroidism and chronic hepatitis C, which were both under control. Her parents had died from cancer, but there was no psychiatric illness in her family. Brain magnetic resonance imaging revealed no changes, while a neuropsychological assessment suggested mild deficits in attention, memory, and prefrontal functions. Because of her potential liver impairment, she was given paliperidone extended release 12 mg once a day. Total remission of symptoms was achieved in 2 weeks. A monthly long-acting injectable formulation of paliperidone palmitate 150 mg was introduced before discharge to guarantee maximum therapeutic adherence.

One month later, the patient presented with bilateral blepharospasm and oromandibular dystonia. To estimate probability of causation between paliperidone intake and these symptoms, the Naranjo Scale2 was administered, and the patient scored a 7. Paliperidone was discontinued, and the patient was started on daily clozapine 100 mg. After 6 months of follow-up, all signs of Meige syndrome had disappeared.

Paliperidone is an atypical antipsychotic drug that has been available in Portugal for a couple of years, although it has been approved for the treatment of schizoaffective disorder in the United States for more than half a decade. Paliperidone is the 9-hydroxy metabolite of risperidone, with similar pharmacologic properties as the antagonist effect at dopamine D2 and serotonin 5-HT2A receptors. Furthermore, paliperidone safety has been assessed in elderly patients3 and undergoes limited hepatic metabolism,4 so it was theoretically a good choice for the treatment of the patient presented here. Meige syndrome is a tardive dystonia that usually appears after antipsychotic and anticholinergic drugs have been used long-term for its treatment. Although paliperidone long-acting injectable has been associated with lower incidence of extrapyramidal symptoms,5 it can cause cranial dystonia (eg, rabbit syndrome) when administered in oral solid formulation.6

This report suggests that paliperidone also can be related to Meige syndrome, a more disabling condition, even with short-term use. It is the author’s belief that the patient’s neurologic findings made her particularly vulnerable to the paliperidone-induced extrapyramidal symptoms. A careful selection of antipsychotic medication in every patient is recommended, as even the most modern treatments can cause undesirable iatrogenic effects.

References

1. Takahashi S, Suzuki M, Uchiyama M. A case of schizophrenia with Meige syndrome induced by perospirone successfully treated with biperiden. J Neuropsychiatry Clin Neurosci. 2013;25(1):E28. PubMed doi:10.1176/appi.neuropsych.12020029

2. Naranjo CA, Busto U, Sellers EM, et al. A method for estimating the probability of adverse drug reactions. Clin Pharmacol Ther. 1981;30(2):239-245.  doi:10.1038/clpt.1981.154

3. Citrome L. Paliperidone: quo vadis? Int J Clin Pract. 2007;61(4):653-662. PubMed doi:10.1111/j.1742-1241.2007.01321.x

4. Kane J, Canas F, Kramer M, et al. Treatment of schizophrenia with paliperidone extended-release tablets: a 6-week placebo-controlled trial. Schizophr Res. 2007;90(1-3):147-161. PubMed doi:10.1016/j.schres.2006.09.012

5. Gopal S, Liu Y, Alphs L, et al. Incidence and time course of extrapyramidal symptoms with oral and long-acting injectable paliperidone: a posthoc pooled analysis of seven randomized controlled studies. Neuropsychiatr Dis Treat. 2013;9:1381-1392. PubMed doi:10.2147/NDT.S49944

6. Teng PR, Lai TJ. Paliperidone-related rabbit syndrome. J Clin Psychopharmacol. 2011;31(3):379-380. PubMed doi:10.1097/JCP.0b013e318219263d

João Gama Marques, MDa

joaogamamarques@gmail.com

aCli­nica de Psicoses Esquizofrénicas, Hospital Júlio de Matos, Centro Hospitalar Psiquiátrico de Lisboa and Cli­nica Universitária de Psiquiatria e Psicologia Médica, Faculdade de Medicina, Universidade de Lisboa, Lisboa, Portugal

Potential conflicts of interest: None.

Funding/support: None.

Informed consent: Informed consent was obtained from the patient to publish the case report.

Published online: January 26, 2017.

Prim Care Companion CNS Disord 2017;19(1):16l01997

https://doi.org/10.4088/PCC.16l01997

© Copyright 2017 Physicians Postgraduate Press, Inc.