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. 2017 Jul;88(7):595-602.
doi: 10.1136/jnnp-2016-315461. Epub 2017 Apr 24.

Psychiatric associations of adult-onset focal dystonia phenotypes

Brian D Berman  1   2 , Johanna Junker  3   4 , Erika Shelton  1 , Stefan H Sillau  1 , H A Jinnah  5 , Joel S Perlmutter  6 , Alberto J Espay  7 , Joseph Jankovic  8 , Marie Vidailhet  9   10 , Cecilia Bonnet  9   10 , William Ondo  11 , Irene A Malaty  12 , Ramón Rodríguez  13 , William M McDonald  14 , Laura Marsh  15 , Mateusz Zurowski  16 , Tobias Bäumer  17 , Norbert Brüggemann  3   4
Affiliations

Psychiatric associations of adult-onset focal dystonia phenotypes

Brian D Berman et al. J Neurol Neurosurg Psychiatry. 2017 Jul.

Abstract

Background: Depression and anxiety frequently accompany the motor manifestations of isolated adult-onset focal dystonias. Whether the body region affected when this type of dystonia first presents is associated with the severity of these neuropsychiatric symptoms is unknown.

Objectives: The aim of this study was to determine whether depression, anxiety and social anxiety vary by dystonia onset site and evaluate whether pain and dystonia severity account for any differences.

Methods: Patients with isolated focal dystonia evaluated within 5 years from symptom onset, enrolled in the Natural History Project of the Dystonia Coalition, were included in the analysis. Individual onset sites were grouped into five body regions: cervical, laryngeal, limb, lower cranial and upper cranial. Neuropsychiatric symptoms were rated using the Beck Depression Inventory, Hospital Anxiety and Depression Scale and Liebowitz Social Anxiety Scale. Pain was estimated using the 36-Item Short Form Survey.

Results: Four hundred and seventy-eight subjects met our inclusion criteria. High levels of depression, anxiety and social anxiety occurred in all groups; however, the severity of anxiety and social anxiety symptoms varied by onset site group. The most pronounced differences were higher anxiety in cervical and laryngeal, lower anxiety in upper cranial and higher social anxiety in laryngeal. Increases in pain were associated with worse neuropsychiatric symptom scores within all groups. Higher anxiety and social anxiety in laryngeal and lower anxiety in upper cranial persisted after correcting for pain and dystonia severity.

Conclusion: Anxiety and social anxiety severity vary by onset site of focal dystonia, and this variation is not explained by differences in pain and dystonia severity.

Keywords: Isolated focal dystonia; anxiety; depression; pain.

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Conflict of interest statement

Competing interests: None declared.

Figures

Figure 1
Figure 1
Depression, anxiety and social anxiety by dystonia onset region. Isolated focal dystonia patients were grouped into five regions of dystonia onset and excluded if they reported more than one body part affected at onset. Overall, F tests for group differences are shown for each scale. Significant difference of *p<0.05 or **p<0.005 in pairwise comparisons with Tukey-Kramer adjustments for multiple comparisons. Error bars represent 95% CIs. BDI, Beck Depression Inventory (2nd edition); HADS-D/A, Hospital Anxiety and Depression Scale–Depression/Anxiety; LSAS, Liebowitz Social Anxiety Scale.
Figure 2
Figure 2
Proportion of depression, anxiety and social anxiety by dystonia onset region. Isolated focal dystonia patients were grouped into five regions of dystonia onset and excluded if they reported more than one body part was affected at time of onset. Significant difference of *p <0.05 or **p <0.005 in pairwise comparisons using two-way χ2 tests or Fisher exact tests. Error bars represent 95% CIs. BDI, Beck Depression Inventory (2nd edition); HADS-D/A, Hospital Anxiety and Depression Scale–Depression/Anxiety; LSAS, Liebowitz Social Anxiety Scale.
Figure 3
Figure 3
Relationship of pain with depression, anxiety and social anxiety by dystonia onset region. Isolated focal dystonia patients were grouped into five regions of dystonia onset and excluded if they reported more than one body part was affected at the time of onset. Significance of linear fit regression model is noted as *p<0.05 or **p<0.005. BDI, Beck Depression Inventory (2nd edition); HADS-D/A, Hospital Anxiety and Depression Scale–Depression/Anxiety; LSAS, Liebowitz Social Anxiety Scale; SF-36, 36-Item Short Form Survey.
Figure 4
Figure 4
Relationship of dystonia severity with depression, anxiety and social anxiety by dystonia onset region. Isolated focal dystonia patients were grouped into five regions of dystonia onset and excluded if they reported more than one body part was affected at the time of onset. Significance of linear fit regression model is noted as *p<0.05 or **p<0.005. BDI, Beck Depression Inventory (2nd edition); GDRS, Global Dystonia Rating Scale; HADS-D/A, Hospital Anxiety and Depression Scale–Depression/Anxiety; LSAS, Liebowitz Social Anxiety Scale.
Figure 5
Figure 5
Depression, anxiety and social anxiety by dystonia onset region after correcting for pain and dystonia severity. Isolated focal dystonia patients were grouped into five regions of dystonia onset and excluded if they reported more than one body part affected at onset. Overall F tests for group differences are shown for each scale. Significant difference of *p <0.05 or **p<0.005 in pairwise comparisons with Tukey-Kramer adjustments for multiple comparisons. Error bars represent 95% CIs. BDI, Beck Depression Inventory (2nd edition); HADS-D/A, Hospital Anxiety and Depression Scale–Depression/Anxiety; LSAS, Liebowitz Social Anxiety Scale.
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