Early Identification and Treatment of Communication and Swallowing Deficits in Parkinson Disease

Ciucci, Michelle R.; Grant, Laura M.; Rajamanickam, Eunice S. Paul; Hilby, Breanna L.; Blue, Katherine V.; Jones, Corinne A.; Kelm‐Nelson, Cynthia A.

doi:10.1055/s-0033-1358367

Cited by 48 publications

(26 citation statements)

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“…In the case of PD, patients have reduced vocal quality (e.g., breathiness or roughness), intensity (loudness), inflection, and articulation [19, 39, 40], even in the early stages of disease progression [1]. Changes in vocalizations in animal models of PD have been demonstrated in several model species.…”

Section: Discussionmentioning

confidence: 99%

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Vocalization development in common marmosets for neurodegenerative translational modeling

Jones

Duffy

Hoffman

et al. 2018

Neurological Research

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Objectives In order to facilitate the study of vocalizations in emerging genetic common marmoset models of neurodegenerative disorders, we aimed to analyze call type changes across age in a translational research environment. We hypothesized that acoustic parameters of vocalizations would change with age, reflecting growth of the vocal apparatus and a maturation of control needed to make adult-like calls. Methods Nineteen developing common marmosets were longitudinally video- and audio-recorded between the ages of 1–149 days in a naturalistic setting without any vocalization elicitation protocol. Vocalizations were coded for call type (cry, tsik, trill, phee, and trill-phee) and analyzed for duration (sec), minimum & maximum frequency (Hz), and bandwidth (Hz). Mixed model linear regressions were performed to assess the effects of age on call parameters listed above for each call type. Results Cries decreased in duration (p=0.038), maximum frequency (p=0.047), and bandwidth (p=0.023) with age. Tsik calls decreased in duration (p=0.002) and increased in minimum frequency (p=0.004) and maximum frequency (p=0.005) with age. Trill calls increased in duration (p=0.003), and trillphee bandwidth (p=0.031) decreased with age. Discussion Our results demonstrate that development of common marmoset vocalizations is call type-dependent and that changes in acoustic parameters can be detected without complex vocalization elicitation paradigms or specialized audio recording equipment. Thus, we demonstrate the feasibility of a naturalistic protocol to collect and objectively analyze marmoset vocalizations longitudinally. This approach may be useful for studying vocal communication deficits in genetic models of neurodegenerative disorders.

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Section: Discussionmentioning

confidence: 99%

“…These changes can occur early in disease progression, especially in Parkinson’s disease (PD) and Alzheimer’s disease (AD) [1, 2], in some cases even before diagnosis [3, 4]. Animal models of these diseases are important for translational medicine, including development and testing of new drug and behavioral treatments [5, 6, 7, 8].…”

Section: Introductionmentioning

confidence: 99%

Vocalization development in common marmosets for neurodegenerative translational modeling

Jones

Duffy

Hoffman

et al. 2018

Neurological Research

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“…However, treatment is often not initiated until the patient is seen by a speech-language pathologist; this may not occur until late in the progression of the disease, as many patients do not report symptoms despite having dysphagia [2]. Early identification of swallowing deficits in individuals with PD may allow for earlier, preventative intervention and thus improved health and quality of life outcomes [12]. Nonetheless, swallowing changes in the early stages of PD are not well described.…”

Section: Introductionmentioning

confidence: 99%

Multimodal Swallowing Evaluation with High-Resolution Manometry Reveals Subtle Swallowing Changes in Early and Mid-Stage Parkinson Disease

Jones

Ciucci

2016

JPD

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Background Parkinson disease (PD) has detrimental effects on swallowing function. Treatment options are largely behavioral; thus, patients would benefit from an earlier start to therapy. Early swallowing changes in PD are not well-known, so patients do not typically receive swallowing treatment until later in the progression of PD. Objective We used predictive modeling to determine what quantitative swallowing variables best differentiate individuals with early to mid-stage PD from healthy controls. Methods Participants included twenty-six individuals with early to mid-stage PD and 26 healthy, age- and sex-matched controls. Swallowing was evaluated by simultaneous high-resolution manometry and videofluoroscopy as well as the Sydney Swallow Questionnaire (SSQ). Binomial logistic regression was performed on 4 sets of data: 1) high-resolution manometry only; 2) videofluoroscopy only; 3) SSQ only; and 4) all data combined. Results A model from a combined data set had the highest accuracy in differentiating individuals with PD from controls. The model included maximum pressure in the velopharynx (soft palate), pressure variability in the velopharynx, and the SSQ item concerning difficulty with saliva swallowing. No significant models could be generated using the videofluoroscopy data. Conclusions Individuals with PD show quantitative changes in pressure generation and are able to self-assess aspects of swallowing function in the early and mid-stages of PD, even in the absence of swallowing changes seen on videofluoroscopy. A multimodal approach for the assessment of swallowing may be more accurate for determining subtle swallowing changes that occur in the early stages of PD.

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“…As the disease progresses, up to 90% of individuals have significant voice deficits that negatively impact quality of life and are difficult to treat [1–4]. Importantly, there is evidence that these changes in voice manifest prior to the onset of cardinal signs of the disease (resting tremor, bradykinesia and muscle rigidity) that are typically associated with significant striatal dopamine loss [5].…”

Section: Introductionmentioning

confidence: 99%

Atp13a2 expression in the periaqueductal gray is decreased in the Pink1 -/- rat model of Parkinson disease

Kelm‐Nelson

Stevenson

Ciucci

2016

Neuroscience Letters

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Vocal communication deficits are common in Parkinson disease (PD). Widespread alpha-synuclein pathology is a common link between familial and sporadic PD, and recent genetic rat models based on familial genetic links increase the opportunity to explore vocalization deficits and their associated neuropathologies. Specifically, the Pink1 knockout (−/−) rat presents with early, progressive motor deficits, including significant vocal deficits, at 8 months of age. Moreover, this rat model exhibits alpha-synuclein pathology compared to age-matched non-affected wildtype (WT) controls. Aggregations are specifically dense within the periaqueductal gray (PAG), a brainstem region involved in the coordination of emotional and volitional control of vocalizations. Here, we investigated changes in gene expression within the PAG at 8 months of age in Pink1 −/− rats compared to WT. Our data demonstrate that Pink1 −/− rat mRNA expression levels of alpha-synuclein are comparable to WT. However, Pink1 −/− rats show significantly decreased levels of Atp13a2, a transmembrane lysosomal P5-type ATPase suggesting a potential mechanism for the observed abnormal aggregation. We found no difference in the expression of glucocerebrosidase (Gba) or the CASP8 and FADD-like apoptosis regulator (Cflar). Further, we show that mRNA expression levels of dopaminergic markers including Th, D1 and D2 receptor as well as GABA signaling markers including Gaba-A and glutamate decarboxylase 2 (Gad2) do not differ between genotypes. However, we found that glutamate decarboxylase 1 (Gad1) is significantly reduced in this PD model suggesting possible disruption of neurotransmission within the PAG. These results are the first to suggest the hypothesis that alpha-synuclein aggregation in this model is not a result of increased transcription, but rather a deficit in the breakdown and clearance, and that the observed vocal deficits may be related to impaired neural transmission. Altogether, these findings are consistent with the hypothesis that differences in neural substrate sensitivity contribute to the early pathogenesis of vocalizations and motivation to communicate in the Pink1 −/− rat model of PD. Our results suggest novel therapeutic pathways, including the lysosomal degradation pathway, which can be used in to further study the pathogenesis and treatment of vocal dysfunction PD.

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Early Identification and Treatment of Communication and Swallowing Deficits in Parkinson Disease

Cited by 48 publications

References 100 publications

Vocalization development in common marmosets for neurodegenerative translational modeling

Vocalization development in common marmosets for neurodegenerative translational modeling

Multimodal Swallowing Evaluation with High-Resolution Manometry Reveals Subtle Swallowing Changes in Early and Mid-Stage Parkinson Disease

Atp13a2 expression in the periaqueductal gray is decreased in the Pink1 -/- rat model of Parkinson disease

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