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Table of Contents    
CASE REPORT
Year : 2019  |  Volume : 67  |  Issue : 6  |  Page : 1522-1524

The Effects of Lee - Silverman Voice Treatment on Voice and Swallowing Functions in a Case with Bilateral Striopallidodentate Calcinosis


Department of Speech and Language Therapy, Hacettepe University Faculty of Health Sciences, Sıhhiye, Ankara, Turkey

Date of Web Publication20-Dec-2019

Correspondence Address:
Dr. Fatma Esen Aydinli
Department of Speech and Language Therapy, Hacettepe University Faculty of Health Sciences, Sihhiye - 06100, Ankara
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.273625

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 » Abstract 


Background: Bilateral striopallidodentate calcinosis (BSPDC) is a rare neurodegenerative disorder characterized by abnormal calcium accumulation in the basal ganglia, dentate nucleus, and semioval center. Speech, voice, and swallowing abnormalities can be associated with the disease. However, there are a limited number of studies regarding the speech and swallowing characteristics of this disease in the literature. Lee-Silverman voice therapy (LSVT-LOUD) is a structured speech therapy method that can be used to treat neurogenic conditions.
Aims: The main purpose of this article was to document the effects of LSVT-LOUD on voice and swallowing functions in a case of BSPDC.
Design: Case report.
Methods: A comprehensive voice and swallowing evaluation, including objective methods, was conducted before therapy and at post-therapy follow-ups (FU) after 3 months and 6 months.
Results: The voice and swallowing parameters were substantially improved at the 3-month FU; at the 6-month FU, the improvement in swallowing function was still preserved; however, the improvement in voice function had regressed.
Conclusion: LSVT-LOUD may improve the voice and swallowing functions of patients with BSPDC. However, long-term retention of the effects of LSVT-LOUD should be investigated in future studies.


Keywords: Bilateral striopallidodentate calcinosis, Fahr disease, hypokinetic dysarthria, LSVT-LOUD, speech therapy
Key Messages: This is the first article documenting the effects of LSVT-LOUD in bilateral striopallidodentate calcinosis.


How to cite this article:
Aydinli FE, Dumbak AB. The Effects of Lee - Silverman Voice Treatment on Voice and Swallowing Functions in a Case with Bilateral Striopallidodentate Calcinosis. Neurol India 2019;67:1522-4

How to cite this URL:
Aydinli FE, Dumbak AB. The Effects of Lee - Silverman Voice Treatment on Voice and Swallowing Functions in a Case with Bilateral Striopallidodentate Calcinosis. Neurol India [serial online] 2019 [cited 2020 Jan 21];67:1522-4. Available from: http://www.neurologyindia.com/text.asp?2019/67/6/1522/273625




Bilateral striopallidodentate calcinosis (BSPDC) is a rare neurodegenerative disorder characterized by abnormal calcium accumulation in the basal ganglia, the dentate nucleus, and the semioval center.[1] The most common symptoms in these cases are movement disorders, with  Parkinsonism More Details representing approximately half of these disorders and the remaining being hyperkinetic movement disorders.[2] Although speech impairment is reported as a common symptom of the disease,[2],[3],[4] these reports are mainly based solely on clinical observations.[1],[2],[3] In addition, in terms of the speech treatment, only one study reported the results of speech therapy applied in an anarthric case.[4]

Speech characteristics in BSPDC are mostly thought to be related to hypokinetic dysarthria.[1],[2],[3] Lee-Silverman voice therapy (LSVT-LOUD) is a structured speech therapy method that was mainly developed for Parkinson's disease, but it can also be used in other neurogenic conditions.

The main purpose of this article was to reveal the effects of LSVT-LOUD on voice and swallowing functions in a case of BSPDC.

Case history

The patient was a 47-year-old male academician. He first presented at the Hacettepe University Speech and Language Department with complaints of voice and speech. It had been 5 years since he was diagnosed with BSPDC. The neurological examination findings were hypomimia, resting tremor, rigidity, bradykinesia, postural instability, and gait disturbance. The brain magnetic resonance imaging showed neurodegeneration with brain iron accumulation and cerebral atrophy. His cognitive status was normal.

After a detailed voice and swallowing evaluation, LSVT-LOUD was applied. The comprehensive voice and swallowing evaluation was repeated at follow-ups (FUs) 3 months and 6 months after therapy.


 » Methods Top


In the evaluation of the patient's voice, maximum phonation time (MPT) and sound pressure levels (SPL) were measured. Acoustic analysis of the patient's voice was performed using the Computerized Speech Lab (CSL) (Model 4300 B) of Kay Elemetrics (Lincoln Park, NJ, USA) with the Multi-Dimensional Voice Program (MDVP) and Cepstral analysis in Analysis of Dysphonia in Speech and Voice (ADSV). Sustained phonation of /a/and Consensus Auditory-Perceptual Evaluation of Voice/Turkish (CAPE-V) sentences and conversation were used as speech samples.[5] Patient-reported evaluation of voice impairment was conducted using the voice handicap index (VHI).[6] The Voicing Efficiency protocol of the PENTAX Medical Phonatory Aerodynamic System (PAS) Model 6600 (KayPENTAX Corp. Montvale, New Jersey) was used for the phonatoary aerodynamic evaluation.[7]

The swallowing examinations included the clinical swallowing evaluation and the videofluoroscopic swallowing study using the Modified Barium Swallow Impairment Profile (MBSImP) protocol.[8]

The LSVT-LOUD protocol was applied to the patient in 1-hour sessions for four times a week during four weeks by a certified speech and language therapist.[9]


 » Results Top


MPT increased dramatically after the therapy, but at the 6-month FU, this value was identical to the pre-therapy value. That is, MPT, which was 14.05 seconds before therapy, increased to 18.17 seconds at the 3-month FU and decreased to 14.00 seconds at the 6-month FU. [Table 1] shows SPLs for the samples of sustained phonation, reading sentences, and conversation. The most prominent increase in SPL at the third month FU was detected for the sustained phonation sample. The pre-treatment SPL value of 64 decibels (dB) was increased to 94 dB at 3-month FU, and it was 91 dB at the 6-month FU. Although the SPL scores in all samples slightly decreased at the 6th month FU, they were still higher than the pre-therapy values for the sustained phonation and reading samples. However, in terms of conversational speech, the SPL value at the 6-month FU was found to be equal to that prior to treatment. The mean peak air pressure (10.55 cmH2O) was higher prior to therapy and lower (6.85 cmH2O) at the 3-month FU. The mean airflow was 0.23l t/sn prior to therapy, and it increased to 0.31 lt/sn at the 3-month FU. These findings indicate that at the 3-month FU, the patient could phonate with a lower subglottal pressure and higher airflow, which indicated a more efficient phonatoary-aerodynamic status.
Table 1: Sound Pressure Levels in sustained phonation, sentence reading, and conversation samples

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According to the acoustic analyses of the patient's voice [Table 2], a gradual increase in fundamental frequency (F0) was observed, accompanied by a decrease in perturbation parameters in MDVP after the therapy. In addition, the cepstral peak prominence (CPP) increased in ADSV. The variability of F0 decreased according to both software, which meant a decrease in the F0 standard deviation (SD) in MDVP and a lower CPP SD score in ADSV. These findings were interpreted as better laryngeal stability after the therapy. However, the quite high F0 detected at 6-month FU was thought to indicate a hyperfunctional voice pattern.
Table 2: Acoustic analyses of voice; MDVP and ADSV results

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The patient-based evaluation clearly showed a lower voice handicap at the 3-month FU. The VHI score was found to be 35 before therapy, decreased to 12 at the 3-month FU, and was 24 at the 6-month FU.

Swallowing evaluation findings are reported in [Table 3] and [Table 4]. The penetration-aspiration scale showed improvement after therapy in all consistencies, predominantly for thin liquids. Overall impression (OI) scores of MBSImP were decreased for both oral and pharyngeal phases after therapy, with a primarily pharyngeal phase.
Table 3: Penetration-aspiration scale scores before and after therapy for each consistencies

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Table 4: Overall Impression Scores of MBSImp before and after therapy

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 » Discussion Top


LSVT-LOUD treatment is specialized for Parkinson disease as well as reported to be applied in different neurological conditions.[10] The main purpose of this method is to sustain patient's loud voice automotically in daily functional communication.

In this case, salient characteristics of speech were compatible with the hypokinetic dysartria and a trial voice therapy session lead us that patient can benefit from LSVT-LOUD. During the therapy sessions, he used his loud voice successfully in sustained phonation; however, significant problems of sensory calibration were associated in each therapy session almost for all speech samples. In addition, he had difficulty to start phonation rarely.

Although some voice measures returned to the baseline status at the FU, most voice parameters and swallowing function were found improved substantially at the 3-month FU. The improvements of loudness levels as well as decreasing voice handicap were similar with the findings of Parkinson patients at 3-month after the therapy. However, it was thought that the effects of LSVT-LOUD was decreased at the 6-month FU, which is shorter duration than the ideal maintenance duration up to 2 years observed for Parkinson patients.[11] One of the reason of this situation may be the fact that patient did not exercise regularly his therapy, which must be done twice a day. The patient declared that he did not exercise during the period between 3-month and 6-month FU. Similarly, reduced compliance with therapy procedures were frequently reported in some studies in patients with dysarthria.[12],[13] The other reason may be raised from the fact that different pathophysiology of Fahr disease, which may affect long-term retention performance of LSVT.[14],[15]

As far as we know, this is the first BSPDC case that a comprehensive voice and swallowing evaluation had been done. As well as, it is the first BSPDC case that a structured speech therapy method applied.

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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Manyam BV. What is and what is not 'Fahr's disease'. Parkinsonism Relat Disord 2005;11:73-80.  Back to cited text no. 1
    
2.
Manyam BV, Walters AS, Narla KR. Bilateral striopallidodentate calcinosis: Clinical characteristics of patients seen in a registry. Mov Disord 2001;16:258-64.  Back to cited text no. 2
    
3.
Millen SJ, Pulec J, Kane P. Fahr's disease. Arch Otolaryngol 1982;108:591-4.  Back to cited text no. 3
    
4.
Meera SS, Shivashankar N, Yadav R, Bhat MD. Anarthria in a patient with bilateral striopallidodentate calcinosis. Neurol India 2017;65:1144-5.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Özcebe E, Aydinli FE, Tiǧrak TK, İncebay Ö, Yilmaz T. Reliability and validity of the turkish version of the consensus auditory-perceptual evaluation of voice (CAPE-V). J Voice 2019;33:382.e1-e10.  Back to cited text no. 5
    
6.
Kiliç MA, Okur E, Yıldırım İ, Öǧüt F, Denizoǧlu İ, Kızılay A, et al. Ses Handikap Endeksi (Voice Handicap Index) Türkçe versiyonunun güvenilirliǧi ve geçerliliǧi. Kulak Burun Bogaz Ihtis Derg 2008;18:139-47.  Back to cited text no. 6
    
7.
Martin-Harris B, Brodsky MB, Michel Y, Castell DO, Schleicher M, Sandidge J. MBS measurement tool for swallow impairment—MBSImp: Establishing a standard. Dysphagia 2008;23:392-405.  Back to cited text no. 7
    
8.
Zraick RI, Smith-Olinde L, Shotts LL. Adult normative data for the KayPENTAX phonatory aerodynamic system model 6600. J Voice 2012;26:164-76.  Back to cited text no. 8
    
9.
Ramig LO, Fox C, Sapir S. Parkinson's disease: Speech and voice disorders and their treatment with the Lee Silverman Voice Treatment. Semin Speech Lang 2004;25:169-80.  Back to cited text no. 9
    
10.
Sapir S, Ramig L, Fox C. Assessment and treatment of the speech disorder in Parkinson disease. Commun Swallowing Parkinson's Disease. 1st ed. San Diego: Plural Publishing; 2011. p. 89-122.  Back to cited text no. 10
    
11.
Ramig LO, Sapir S, Countryman S, Pawlas AA, O'brien C, Hoehn M, et al. Intensive voice treatment (LSVT®) for patients with Parkinson's disease: A 2 year follow up. J Neurol Neurosurg Psychiatry 2001;71:493-8.  Back to cited text no. 11
    
12.
Sellars C, Hughes T, Langhorne P. Speech and language therapy for dysarthria due to non-progressive brain damage (Cochrane Review). Cochrane Database Syst Rev 2002:CD002088.  Back to cited text no. 12
    
13.
Wenke RJ, Theodoros D, Cornwell P. The short-and long-term effectiveness of the LSVT® for dysarthria following TBI and stroke. Brain Injury 2008;22:339-52.  Back to cited text no. 13
    
14.
Liotti M, Vogel D, New P, Ramig L, Mayberg H, Cook C, et al. A PET study of functional reorganization of premotor regions in Parkinson's disease following intensive speech and voice treatment (LSVT). Neurology 1999;52:A348-9.  Back to cited text no. 14
    
15.
Jürgens U, von Cramon D. On the role of the anterior cingulate cortex in phonation: A case report. Brain Lang 1982;15:234-48.  Back to cited text no. 15
    



 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4]



 

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