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Try out PMC Labs and tell us what you think. Learn More. View Supplementary Video 1. Involuntary vocalizations are a striking feature of several neurological conditions, often associated with movement disorders.
They range from simple and brief sounds e. Constant grunting and groaning are involuntary and uncontrollable vocalizations, typical for several types of dementia, and have also been associated with advanced progressive supranuclear palsy PSP. Attempts to distract attention from the active inhibitory process led to reemergence of the groaning behavior. The phenomenological classification and pathophysiological implications of the involuntary groaning behavior are discussed.
The groaning behavior, which was constantly present and only abated during deep sleep, was distressing, particularly for the patient's family. However, the most striking feature was the groaning behavior.
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Importantly, although the patient reported that the sound was generated involuntarily, he was able to selectively suppress the involuntary groaning; employing, for example, breathing techniques Video S1, segment B. Distracting the patient from the effortful inhibitory process led to reemergence of the involuntary sounds. There was no association of the involuntary groaning behavior with attempted changes in medication.
For example, discontinuation of all dopaminergic medication over a period of four weeks led to deterioration of parkinsonism, without changes in groaning frequency or intensity. Similarly, discontinuation of quetiapine over several months did not lead to any changes in involuntary groaning behavior.
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In the presented case, constant groaning was the most striking feature and the reason for referral to our clinic. Although the association of groaning with neurodegeneration e. However, two clinical observations from the case here provide crucial insights into both of these aspects. First, the generation of voluntary speech and groaning behavior did not occur in succession, but in parallel. Importantly, the presence of involuntary groaning interfered with the production of voluntary speech. This implies that the two different types of vocalizations are generated by two different neural resources that are concurrently active, but the vocal motor apparatus cannot be driven by both generators simultaneously.
Second, selective inhibition of the involuntary vocalizations did not abolish the ability to speak. Thus, our case illustrates two important points regarding the neurocognitive architecture for vocal control, first, dissociation between two pathways for vocalizations. The possibility of selective inhibition confirms the dissociation between two vocal generators. Hughlings Jackson proposed the distinction between voluntary vs. Among these different phenomena, constant groaning is thought to belong to a range of purposeless behaviors, such as continuous chattering and motor perseverations, as a result of reduced—prefrontal—cortical inhibitory control on subcortical structures.
However, this effortful control would only be exerted during the brief periods of concentration. Research Project: A. Conception, B. Organization, C. Execution; 2. Statistical Analysis: A. De, B. Execution, C. Male groaning sounds and Critique; 3. Manuscript Preparation: A. Writing the First Draft, B.
Review and Critique. Ethical Compliance Statement : We confirm that we have read the Journal's position on issues involved in ethical publication and affirm that this work is consistent with those guidelines.
The authors confirm that the approval of an institutional review board was not required for this work. Funding Sources and Conflicts of Interest: There are no funders to report for this submission.
The authors declare that there are no conflicts of interest relevant to this work. Video S1. Segment A : Examination revealed supranuclear vertical gaze palsy, particularly for downward gaze.
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Groaning behavior was constant throughout the examination, even when patient attempts to keep his mouth closed. There was bradykinesia. Applause was positive. Postural reflexes were clearly affected. Segment B : The patient can suppress the involuntary groaning behavior with concentration and breathing exercises. Table S1. Overview of involuntary vocalizations, their phenomenology and associated conditions.
We are much obliged to the patient and his family for written permission to publish this case description. Relevant disclosures and conflicts of interest are listed at the end of this article. National Center for Biotechnology InformationU. Mov Disord Clin Pract. Published online Mar Author information Article notes Copyright and information Disclaimer.
Tina Mainka, : ed. Corresponding author.
Keywords: groaning, involuntary vocalization, parkinsonism, progressive supranuclear palsy. This article has been cited by other articles in PMC. Associated Data Supplementary Materials Videos accompanying this article are available in the supporting information here.
Author Roles 1. Disclosures Ethical Compliance Statement : We confirm that we have read the Journal's position on issues involved in ethical publication and affirm that this work is consistent with those guidelines.
Supporting information Videos accompanying this article are available in the supporting information here. for additional data file. Acknowledgements We are much obliged to the patient and his family for written permission to publish this case description.
Notes Relevant disclosures and conflicts of interest are listed at the end of this article. References 1. Characteristic constant groaning in late stage progressive supranuclear palsy: a case report. Parkinsonism Relat Disord. The neural control of vocalization in mammals: a review. J Voice. On affections of speech from disease of the brain. Arch Gerontol Geriatr.
Voluntary inhibition of involuntary groaning in progressive supranuclear palsy
Progressive supranuclear palsy. A heterogeneous degeneration involving the brain stem, basal ganglia and cerebellum with vertical gaze and pseudobulbar palsy, nuchal dystonia and dementia.
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