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The Effects of Lee - Silverman Voice Treatment on Voice and Swallowing Functions in a Case with Bilateral Striopallidodentate Calcinosis
Correspondence Address: Source of Support: None, Conflict of Interest: None DOI: 10.4103/0028-3886.273625
Keywords: Bilateral striopallidodentate calcinosis, Fahr disease, hypokinetic dysarthria, LSVT-LOUD, speech therapy
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.
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]
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.
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.
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.
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.
[Table 1], [Table 2], [Table 3], [Table 4]
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