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Table of Contents
Year : 2020  |  Volume : 7  |  Issue : 2  |  Page : 108-111

Oromandibular dystonia secondary to methylphenidate: A case report and literature review

Department of Medicine, Federal University of Santa Maria, Santa Maria, Rio Grande Do Sul, Brasil

Date of Submission17-Nov-2019
Date of Decision04-Jan-2020
Date of Acceptance21-Apr-2020
Date of Web Publication17-Jun-2020

Correspondence Address:
Dr. Jamir Pitton Rissardo
Rua Roraima, Santa Maria, Rio Grande Do Sul
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/iahs.iahs_71_19

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Methylphenidate (MTP) is a first-line treatment for attention-deficit hyperactivity disorder (ADHD) in children and adults and as a second-line treatment for narcolepsy in adults. We report a case of a young adult male who presented with abnormal orofacial movements after MTP use for the management of ADHD. Laboratory tests were within normal limits. His family history was unremarkable and negative for neurological diseases. A dose of MTP was given during the neurological examination, and abnormal facial movements suggestive of oromandibular dystonia (DTN) were observed. MTP was withdrawn and the symptoms recovered. The oromandibular DTN secondary to MTP probably occurs due to influences in the dopaminergic pathway, and these dyskinetic movements may be associated with a disbalance state of the dopamine, in which an increase or decrease of this neurotransmitter could lead to abnormal movements.

Keywords: Dystonia, methylphenidate, oromandibular dystonia, tongue movements

How to cite this article:
Rissardo JP, Caprara AL. Oromandibular dystonia secondary to methylphenidate: A case report and literature review. Int Arch Health Sci 2020;7:108-11

How to cite this URL:
Rissardo JP, Caprara AL. Oromandibular dystonia secondary to methylphenidate: A case report and literature review. Int Arch Health Sci [serial online] 2020 [cited 2020 Sep 22];7:108-11. Available from: http://www.iahs.kaums.ac.ir/text.asp?2020/7/2/108/286990

  Introduction Top

Drug-induced movement disorders are usually associated with antipsychotic drugs. Neuroleptics are the most common cause of dystonia (DTN). Among these are the antiemetics that block central dopamine receptors, lithium, selective serotonin reuptake inhibitors, stimulants, and tricyclic antidepressants.[1]

Methylphenidate (MTP) hydrochloride was approved in 1955 by the Food and Drug Administration. It is first-line management for attention-deficit hyperactivity disorder (ADHD) in children and adults and as a second-line treatment for narcolepsy in adults.[2] In this context, MTP is a stimulant medication that is commonly associated with significant central nervous system side effects.[3] Insomnia and nervousness are the most commonly reported adverse effects, and other frequent complaints are related to the cardiovascular system such as tachycardia and palpitations. Furthermore, there have been reported cases of sudden death in both children and adults with a preexisting structural cardiac abnormality.[2]

Orofacial DTN is a rarely described adverse effect in association with single-dose MTP.[4] DTN due to MTP treatment has generally been reported during combined treatments with antiepileptic and other psychotropic drugs.[5] Herein, we will report a case of a young adult that was only using MTP and developed abnormal tongue movements.

  Case Report Top

An 18-year-old male presenting with orofacial abnormal movements was admitted to our hospital. He reported a diagnosis of ADHD since he was 9 years old, with feelings of inappropriate running, difficulty playing in silence, seems to be always on the move and talking excessively. Furthermore, his parents complained that he cannot hold his attention for the past 5 years with failing to follow through, seeming not to listen even when directly addressed, and difficulty organizing activities. His parents stated that even though he had a previous diagnosis of ADHD, he never used any medication and lost the follow-up in the psychiatric clinic.

Two days ago, the patient had a psychiatric appointment because the symptoms of ADHD were not been controlled with only behavioral adjustment, and he was struggling with the university classes. Laboratory tests were within normal limits. An electrocardiogram was normal. MTP 10 mg PO once a day was started.

After 3 days, the patient returned complaining of abnormal tongue movements. An improvement of attention-related processes was observed. However, he reported that about 1 h after every dose of the medication, his tongue seemed to have rolling movements. Besides, his parents stated that when they gave in another time the medication, tongue movements started again. His family history was unremarkable and negative for neurological diseases. On neurological examination, he had Grade 5 strength diffusely, normal deep tendon reflexes, and the absence of pyramidal signs, tremor, or bradykinesia. A dose of MTP was given during the examination, about 30 min after it was observed tongue movements, which were sustained with intermittent muscle contractions causing abnormal and repetitive movements suggestive of DTN. No other involuntary movements were present. MTP was withdrawal. The patient was referred to a psychiatric clinic, and the follow-up was lost.

  Discussion Top

ADHD is the most common neurodevelopmental disorder, which is characterized by inattentiveness and hyperactivity/impulsivity. MTP improves ADHD symptoms and is currently considered the first-choice medication. In this context, MTP is a stimulant with a duration of action of approximately 1 h and a half-life of almost 3 h.[6] Its main mechanism of action is the norepinephrine–dopamine reuptake inhibition, which increases the concentration of these neurotransmitters in the synaptic cleft.[2] It was observed in rat models that the majority of this increase of dopamine occurs in the prefrontal cortex, which is the pathway pathophysiologically related to ADHD.[6]

DTN is defined by maintained involuntary muscle contractions, which result in twisting, abnormal, and often repetitive movements and/or postures.[7] It is a neurological movement disorder that may cause patients to visit the emergency department.[8] Furthermore, it is noteworthy that drug-induced DTN when compared to other movement disorders more commonly affects younger females using higher doses of medication.[9]

Drug-induced dyskinesias have been widely described in association with neuroleptic use or withdrawal, which are explained by the dopaminergic exposure with some therapeutic level variability in the striatum.[10] In this way, the oromandibular DTN secondary to MTP probably occurs due to influences in the dopaminergic pathway by the norepinephrine–dopamine reuptake inhibition.[11] Besides, it was already observed DTN in cases of MTP withdrawal.[7] Therefore, the dyskinetic movements may occur due to a disbalance state of the dopamine, in which an increase or decrease could lead to abnormal movements.[12]

The DTN secondary to MTP is probably a dose-dependent side effect because the majority of the reports with its occurrence are with high doses or overdoses causing toxicity.[13],[14] However, to the authors' knowledge, no report of the literature has already shown a direct relation of dose and frequency or amplitude of the abnormal movement. Moreover, it was observed in animal studies that dyskinetic movements associated with MTP happen when there is the plasmatic peak of MTP.[6],[11],[15] Hence, the prescription of lower MTP doses with small increases probably does not lead to the development of an abnormal movement.[16]

There are only a few case reports of MTP associated with DTN. A literature search was performed in Medline using a set of terms that included DTN and MTP [Table 1].[1],[3],[5],[7],[11],[13],[14],[15],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26]
Table 1: Literature review of the dystonia associated with methylphenidate use

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Another interesting fact is that DTN and convulsions when present in association with MTP therapy are sings of MTP toxicity. Doses that exceed 60 mg of the immediate-release formulation or 120 mg of the extended-release formulation can be considered toxic.[2] In these cases, benzodiazepines are considered an option to alleviate the symptoms and accelerate recovery.[23] However, the majority of the case reports in the literature with oromandibular DTN did not report the use of other drugs to improve the symptoms. We believe that this happened because the clinical manifestations occurred with fewer MTP doses, so the medication was promptly discontinued without causing an abnormal adaptation of the direct or indirect pathways.[15]

In sum, our report suggests that MTP should be listed as a probable cause of oromandibular DTN. This adverse effect, while not potentially fatal, can be very embarrassing for ADHD patients and lead to low adherence to therapy. Therefore, clinicians prescribing MTP should be aware of this possible side effect and communicate it to patients.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship


Conflicts of interest

There are no conflicts of interest.

  References Top

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Verghese C, Abdijadid S. Methylphenidate. StatPearls: StatPearls Publishing; 2019.  Back to cited text no. 2
LeRiger M, Williams J, Duncan-Wiebe G, Shukry M. Acute masseter dystonia in a pediatric patient receiving aripiprazole and methylphenidate following induction of general anesthesia. Paediatr Anaesth 2017;27:863-4.  Back to cited text no. 3
Anand B. Methylphenidate induced tongue movements. Arch Ment Health 2019;20:26.  Back to cited text no. 4
  [Full text]  
Senecky Y, Lobel D, Diamond GW, Weitz R, Inbar D. Isolated orofacial dyskinesia: A methylphenidate-induced movement disorder. Pediatr Neurol 2002;27:224-6.  Back to cited text no. 5
Wargin W, Patrick K, Kilts C, Gualtieri CT, Ellington K, Mueller RA, et al. Pharmacokinetics of methylphenidate in man, rat and monkey. J Pharmacol Exp Ther 1983;226:382-6.  Back to cited text no. 6
Grau-López L, Daigre C, Mercado N, Casas M, Roncero C. Dystonia in methylphenidate withdrawal: A case report. J Addict Med 2017;11:154-6.  Back to cited text no. 7
Rissardo JP, Caprara AL. Cervical and axial dystonia secondary to mirtazapine: A case report and literature review. Ann Mov Disord 2020;3:47.  Back to cited text no. 8
Rissardo JP, Caprara AL. Buspirone-associated movement disorder: A literature review. Prague Med Rep 2020;121:5-24.  Back to cited text no. 9
Rosenberg D, Rosenberg DR, Holttum J, Gershon S. Textbook of Pharmacotherapy for Child and Adolescent Psychiatric Disorders. New York: Psychology Press; 1994.  Back to cited text no. 10
Husain A, Chapel J, Malek-Ahmadi P. Methylphenidate, neuroleptics and dyskinesia-dystonia. Can J Psychiatry 1980;25:254-8.  Back to cited text no. 11
Rissardo JP, Caprara AL. Comment: Dystonia and asterixis in acute thalamic infarct: Proposed mechanism. Ann Mov Disord 2019;2:138.  Back to cited text no. 12
Rylander G. Psychoses and the punding and choreiform syndromes in addiction to central stimulant drugs. Psychiatr Neurol Neurochir 1972;75:203-12.  Back to cited text no. 13
Waugh JL. Acute dyskinetic reaction in a healthy toddler following methylphenidate ingestion. Pediatr Neurol 2013;49:58-60.  Back to cited text no. 14
Yilmaz AE, Donmez A, Orun E, Tas T, Isik B, Sonmez FM. Methylphenidate-induced acute orofacial and extremity dyskinesia. J Child Neurol 2013;28:781-3.  Back to cited text no. 15
Rissardo JP, Caprara AL. Oromandibular dystonia and botulinum neurotoxin: An overview. Med J DY Patil Vidyapeeth 2019;12:554.  Back to cited text no. 16
Fann WE. Use of methylphenidate to counteract acute dystonic effects of phenothiazines. Am J Psychiatry 1966;122:1293-4.  Back to cited text no. 17
Gay CT, Ryan SG. Paroxysmal kinesigenic dystonia after methylphenidate administration. J Child Neurol 1994;9:45-6.  Back to cited text no. 18
Boogerd W, Beijnen JH. Methylphenidate for cerebral palsy with choreoathetosis. Ann Intern Med 2000;132:510.  Back to cited text no. 19
McLaren JL, Cauble S, Barnett RJ. Aripiprazole induced acute dystonia after discontinuation of a stimulant medication. J Clin Psychopharmacol 2010;30:77-8.  Back to cited text no. 20
Eftekhari K, Choe CH, Vagefi MR, Gausas RE, Eckstein LA. Oral methylphenidate for the treatment of refractory facial dystonias. Ophthalmic Plast Reconstr Surg 2015;31:e65-6.  Back to cited text no. 21
Guler G, Yildirim V, Kutuk MO, Toros F. Dystonia in an adolescent on risperidone following the discontinuation of methylphenidate: A case report. Clin Psychopharmacol Neurosci 2015;13:115-7.  Back to cited text no. 22
Tekin U, Soyata AZ, Oflaz S. Acute focal dystonic reaction after acute methylphenidate treatment in an adolescent patient. J Clin Psychopharmacol 2015;35:209-11.  Back to cited text no. 23
Pérez CA, Garcia SS, Yu RD. Extrapyramidal symptoms as a result of risperidone discontinuation during combination therapy with methylphenidate in a pediatric patient. J Child Adolesc Psychopharmacol 2016;26:182.  Back to cited text no. 24
Meyers KJ, Upadhyaya HP, Goodloe R, Kryzhanovskaya LA, Liles-Burden MA, Kellier-Steele NA, et al. Evaluation of dystonia in children and adolescents treated with atomoxetine within the truven marketscan database: A retrospective cohort study. Expert Opin Drug Saf 2018;17:467-73.  Back to cited text no. 25
Attalla M. Methylphenidate: Signal for trismus. React 1770;3:2019.  Back to cited text no. 26


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