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CASE REPORT
Year : 2019  |  Volume : 5  |  Issue : 1  |  Page : 29-30

Speech Induced Primary Focal Lingual Dystonia


1 Department of Neurology, VMMC and SJH, New Delhi, India
2 Kailash Neuro Centre, Haldwani, India
3 Kainos Hospital, Rohtak, Haryana, India
4 Department of Neurology, GIPMER, New Delhi, India

Date of Web Publication30-Apr-2019

Correspondence Address:
Dr. Kamakshi Dhamija
Kainos Hospital, Rohtak, Haryana (DM Neurology, GIPMER, New Delhi)
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mamcjms.mamcjms_55_18

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  Abstract 


Isolated lingual dystonia is a rare form of focal dystonia. Speech-induced primary lingual dystonia is rarer still, with only eight cases reported worldwide till date. It is a challenge to differentiate it from psychogenic dystonia. This has therapeutic implications as directed speech therapy is an important modality of treatment in speech-induced primary lingual dystonia. We report one such case from our clinical practice.

Keywords: Focal dystonia, lingual, speech induced dystonia


How to cite this article:
Chaudhry N, Joshi L, Dhamija K, Khwaja GA. Speech Induced Primary Focal Lingual Dystonia. MAMC J Med Sci 2019;5:29-30

How to cite this URL:
Chaudhry N, Joshi L, Dhamija K, Khwaja GA. Speech Induced Primary Focal Lingual Dystonia. MAMC J Med Sci [serial online] 2019 [cited 2019 Sep 21];5:29-30. Available from: http://www.mamcjms.in/text.asp?2019/5/1/29/257427




  Introduction Top


Primary dystonias are often focal, presenting in a craniofacial distribution in adults.[1] Lingual dystonia induced by speech is a rare form of primary dystonia, usually idiopathic in nature. Patients with neurodegenerative disorders like neuroacanthocytosis and Wilson’s disease may suffer from lingual dystonia, although most of the cases of lingual dystonia have been described in association with tardive dystonia.[2] Very few cases of speech-induced lingual dystonia have been reported worldwide[3],[4],[5],[6],[7],[8],[9],[10] with only one report from India [[Table 1]].[8] We describe one such case with emphasis on differentiation from psychogenic forms of lingual dystonias.
Table 1: Speech-induced lingual dystonia cases, including our case

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  Case Report Top


A 33-year-old female presented to us with 1-year history of difficulty in speaking. Her tongue would protrude on attempt to speak, leading to slurring of speech. She experienced no difficulty in chewing or swallowing, and although unable to speak, she could sing very well. There was no history of posturing of any other body part, slowing of activities of daily living, cognitive impairment, weakness, or sensory impairment. There was a history of multiple stressors in the family, but there was no history of mood changes, feeling of worthlessness, sleep disturbances, or occurrence of symptoms only in front of others. There was also no history of any drug intake or any drug abuse.

Evaluation of the tongue movements during several tasks like spontaneous conversation, repetition of words and sentences, sequences of syllables, vowels (a, e, i, o, u), and fricative phonemes (consonants that are formed by impeding the flow of air somewhere in the vocal apparatus so that a friction sound is produced − like f, s, and others) was carried out. The tongue movement disorder was identified in all circumstances of speech and during speaking all phonemes but was absent during speaking vowels. It occurred more often in alveodental and alveolar phonemes (t, d), and less frequently in palatal (j, ch, sh) and velar (k, g, kh) phonemes, whereas it improved by slower speech and low voice intensity [Videos 1 and 2]. Routine hematological and biochemical tests were normal. Her laboratory workup was negative for Wilson’s disease, neuroacanthocytosis, and neurodegeneration with brain iron accumulation. A 1.5 T brain magnetic resonance imaging scan was also normal. Psychiatrist referral ruled out depression or any other psychiatric disorder and the patient had no response to suggestions or placebo. She was started on clonazepam and trihexyphenidyl with which she reported almost 50% benefit, which further improved on directed speech therapy.





  Discussion Top


Speech-induced primary focal lingual dystonia is an extremely rare form of focal task-specific dystonia. Because of its peculiar phenomenology, it is easy to confuse it as a psychogenic movement disorder. The dystonia is characterized by either tongue protrusion or retraction with worsening of dystonia on utterances of alveodental and alveolar phonemes and no dystonia while speaking vowels. This point helps to differentiate it from psychogenic dystonia that is an important differential. Moreover, psychogenic dystonia is characterized by nonpatterned and fluctuating postures, inconsistent examination findings, and distractibility, or with the presence of multiple somatizations and psychiatric disturbances.[2]

Till date, only eight other cases have been reported worldwide[3],[4],[5],[6],[7],[8],[9],[10] with age range of 32 to 58 years. There is female predilection (three and five cases of male and female, respectively). Only one case had the spread of dystonia from tongue to masseter[5] with rest of the cases having strictly focal involvement of the tongue. The treatment has remained challenging with most of the cases having partial improvement to drugs. Treatment protocols have included trihexyphenidyl, tetrabenezine, baclofen, levodopa, tizanidine, and benzodiazipines. Botulinum toxin was tried in only one case with no significant improvement.[10]

Our patient had multiple underlying stressors but as she had presented with patterned form of lingual dystonia, as described, isolated speech-induced lingual dystonia was considered and treated accordingly, with a fruitful result. Although psychogenic dystonia is a strong differential, a careful evaluation is essential to differentiate the two, more so, as directed speech therapy gives benefit in cases of primary dystonia, as was seen in our case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Fahn S, Jankovic J, Hallet M. Principles and Practice of Movement Disorders. 2nd edition. New York: Elsevier Saunders 2011.  Back to cited text no. 1
    
2.
Schneider SA, Aggarwal A, Bhatt M, Dupont E, Tisch S, Limousin P et al. Severe tongue protrusion dystonia: clinical syndromes and possible treatment. Neurology 2006;67:940-3.  Back to cited text no. 2
    
3.
Baik JS, Park JH, Kim JY. Primary lingual dystonia induced by speaking. Mov Disord 2004;19:1251-2.  Back to cited text no. 3
    
4.
Blunt SB, Fuller G, Kennard C, Brooks D. Orolingual dystonia with “tip of the tongue” geste. Mov Disord 1994;9:466.  Back to cited text no. 4
    
5.
Ishii K, Tamaoka A, Shoji S. A case of primary focal lingual dystonia induced by speaking. Eur J Neurol 2001;8:507.  Back to cited text no. 5
    
6.
Papapetropoulos S, Singer C. Primary focal lingual dystonia. Mov Disord 2006;21:429-30.  Back to cited text no. 6
    
7.
Tan EK, Chan LL. Sensory tricks and treatment in primary lingual dystonia. Mov Disord 2005;20:388.  Back to cited text no. 7
    
8.
Verma A, Yadav R. Isolated lingual dystonia: responding to anticholinergic treatment. J Assoc Physicians India 2011;59:526-7.  Back to cited text no. 8
    
9.
Felicio AC, Godeiro C Jr, Moriyama TS, Laureano MR, Felix EP, Borges V et al. Speech-induced lingual dystonia. Arq Neuropsiquiatr 2010;68:653-5.  Back to cited text no. 9
    
10.
Ozen B, Gunal DI, Turkmen C, Agan K, Elmaci NT. Speech-induced primary lingual dystonia: a rare focal dystonia. Neurol Sci 2011;32:155-7.  Back to cited text no. 10
    



 
 
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