Isolated lingual involvement in Wilson’s disease

Choudhary, Neera; Joshi, Laxmikant; Duggal, Ashish; Puri, Vinita; Khwaja, Geeta Anjum

doi:10.4103/0976-3147.154578

Cited by 8 publications

(7 citation statements)

References 5 publications

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“…Due to the low prevalence of lingual dystonia, it has previously been reported on in single case reports [15,26,33,35,36,37,38,39,40,41,42] or case series [7,8,9,10,11,12,13]. This study is the largest series of cases with lingual dystonia reported thus far.…”

Section: Discussionmentioning

confidence: 81%

Botulinum Neurotoxin Therapy for Lingual Dystonia Using an Individualized Injection Method Based on Clinical Features

Yoshida

2019

Toxins

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Lingual dystonia is a debilitating type of oromandibular dystonia characterized by involuntary, often task-specific, contractions of the tongue muscle activated by speaking or eating. Botulinum neurotoxin (BoNT) has been used to treat lingual dystonia; however, it is known to cause serious complications, such as dysphasia and aspiration. The purpose of this study was to evaluate the efficacy and adverse effects of individualized BoNT therapy for lingual dystonia. One-hundred-and-seventy-two patients (102 females and 70 males, mean age: 46.2 years) with lingual dystonia were classified into four subtypes based on symptoms of involuntary tongue movements: protrusion (68.6%), retraction (16.9%), curling (7.6%), and laterotrusion (7.0%). Patients were treated with BoNT injection into the genioglossus and/or intrinsic muscles via individualized submandibular and/or intraoral routes. Results were compared before and after BoNT therapy. Botulinum neurotoxin was injected in 136 patients (mean: 4.8 injections). Clinical sub-scores (mastication, speech, pain, and discomfort) in a disease-specific rating scale were reduced significantly (p < 0.001) after administration. Comprehensive improvement after BoNT injection, assessed using the rating scale, was 77.6%. The curling type (81.9%) showed the greatest improvement, while the retraction type showed the least improvement (67.9%). Mild and transient dysphasia occurred in 12.5% of patients (3.7% of total injections) but disappeared spontaneously within several days to two weeks. No serious side effects were observed. With careful diagnosis of subtypes and a detailed understanding of lingual muscle anatomy, individualized BoNT injection into dystonic lingual muscles can be effective and safe.

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Section: Discussionmentioning

confidence: 81%

Botulinum Neurotoxin Therapy for Lingual Dystonia Using an Individualized Injection Method Based on Clinical Features

Yoshida

2019

Toxins

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“…1 Phenomenology varies among dystonia, chorea, bradykinesia, or tremor and is commonly misdiagnosed. [1][2][3][4][5][6] Isolated lingual involvement in WD has previously been described by Liao et al 2 and Kumar et al, 3 but as irregular contractions and dystonia, respectively. We agree with El Otmani et al that such arrhythmic, irregular to-and-fro tongue movements are best described as "undulating."…”

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confidence: 99%

Undulating Tongue: A Window for Diagnosing Wilson's Disease

Agarwal

Garg

Qavi

2020

Movement Disord Clin Pract

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“…To date, prevalence estimates for lingual dystonia have not been reported. Lingual dystonia has previously been described mostly in single case reports ( 13 , 16 , 18 , 25 – 33 ) or case series ( 6 – 11 ). Thus, methodologically sound studies of lingual dystonia are lacking.…”

Section: Discussionmentioning

confidence: 99%

“…Except for embouchure dystonia, task-specific occupation-related dystonia in the orofacial region has been reported only in auctioneers ( 22 ), bingo callers ( 23 ), and those who recite Islamic prayers ( 24 ). Previous reports of lingual dystonia have been single case reports ( 13 , 16 , 18 , 25 – 33 ) or case series ( 6 – 11 ). Most reports have been published as Letters to the Editor or Clinical Notes.…”

Section: Introductionmentioning

confidence: 99%

Clinical and Phenomenological Characteristics of Patients with Task-Specific Lingual Dystonia: Possible Association with Occupation

Yoshida

2017

Front. Neurol.

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BackgroundLingual dystonia is a subtype of oromandibular dystonia, which is a movement disorder characterized by involuntary sustained or intermittent contraction of the masticatory and/or tongue muscles. Lingual dystonia interferes with important daily activities, such as speaking, chewing, and swallowing, resulting in vocational and social disability.ObjectiveThe aim of this study was to investigate a possible relationship between occupation and the development of lingual dystonia.MethodsPhenomenological and clinical characteristics of 95 patients [53 females (55.8%) and 42 males (44.2%), mean age 48.0 years] with task-specific, speech-induced lingual dystonia were analyzed. Structured interviews were carried out to obtain information regarding primary occupation, including overtime work and stress during work. The factors that might have influenced the development of lingual dystonia were estimated using multivariate logistic regression analysis of the 95 patients with lingual dystonia and 95 controls [68 females (71.6%) and 27 males (28.4%), mean age 47.2 years] with temporomandibular disorders.ResultsOverall, 84.2% of the patients had regular occupations; 73.8% of the patients with regular occupations reported working overtime more than twice a week, and 63.8% of them experienced stress at the workplace. Furthermore, 82.1% of the patients had engaged in occupations that required them to talk to customers or other people under stressful situations over prolonged periods of time for many years (mean: 15.6 years). The most common occupation was sales representative (17.9%), followed by telephone operator (13.7%), customer service representative (10.5%), health care worker (9.5%), waiter or waitress (5.3%), receptionist (5.3%), and cashier (5.3%). Twenty-nine patients (30.5%) had tardive lingual dystonia. Logistic regression analyses revealed that frequent requirements for professional speaking (p = 0.011, odds ratio: 5.66), high stress during work (p = 0.043, odds ratio: 5.4), and neuroleptic use (p = 0.032, odds ratio: 2.52) were significant contributors to the manifestation of lingual dystonia.ConclusionProfessions in which conversations in stressful situations are unavoidable may trigger lingual dystonia. Therefore, speech-induced lingual dystonia can be regarded as occupational dystonia in certain cases.

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Isolated lingual involvement in Wilson’s disease

Cited by 8 publications

References 5 publications

Botulinum Neurotoxin Therapy for Lingual Dystonia Using an Individualized Injection Method Based on Clinical Features

Botulinum Neurotoxin Therapy for Lingual Dystonia Using an Individualized Injection Method Based on Clinical Features

Undulating Tongue: A Window for Diagnosing Wilson's Disease

Clinical and Phenomenological Characteristics of Patients with Task-Specific Lingual Dystonia: Possible Association with Occupation

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