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Atypical presentation of psychogenic movement disorder with jaw-opening dystonia: two cases reports
Abstracts / Parkinsonism and Related Disorders 22 (2016) e87ee141
Methods: We conducted investigation with anamnesis and treatments’ data, appropriate scales for dystonia and Pittsburgh Sleep Quality Index (PSQI), Visual Analogue Scale, McGill questionnaire and Hospital Anxiety and Depression Scale (HADS). The study involved randomly selected 70 dystonia patients (20 generalized dystonia, 25 cervical dystonia and 25 facial dystonia: blepharospasm, oromandibular dystonia and hemifacial spasm) and 10 patients that had treated with DBS last year. . For all patients we did another investigation 6 months after first one and for DBS patients before DBS and 6 months after operation. In addition we try to see factors that were connected with pain and sleep problems. Results: The pain prevalence was 83% and for sleep disturbances 80%. After 6 months the pain was reduced in group treated with Botulinum toxin (from 76% to 20%). After DBS we observed significant decrease in frequency concerning sleep problems (from 90 % to 10% of patients) and pain (from 90% to 20%) (p<0,05). We found that both symptoms were associated with depression. Conclusions: There is rather high frequency of the pain and sleep problems at dystonia patients and they should have to be assessed regularly in our clinical practice. DBS helps in relieving of the pain and sleep problems. References: 1. Eichenser SR, Stebbins GT, Comella CL. Beyond a motor disorder: a prospective evaluation of sleep quality in cervical dystonia. Parkinsonism Relat Disord 2014;20:405-408. 2. Kuyper DJ, Parra V, Aerts S et al. Nonmotor manifestations of dystonia: a systematic review. Mov Disord 2011;26:1206-1217.
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Objectives: Jaw-opening dystonia is a rare condition, and only a limited number of cases have been reported in the literature. Dystonia is rarely psychogenic and jaw-opening dystonia is extremely rare. Our aim is to report atypical types of psychogenic movement disorder presenting jawopening dystonia. Methods: Two cases reports and description of clinical characteristics. Results: We report two patients with clinically established psychogenic dystonia. Patient 1 is a female that presented sudden-onset, involuntary opening of mouth one day ago. She developed clinically non-fixed, continual jaw-opening dystonia and hypertonia of jaw and mouth disappeared with distraction. Psychological examination showed severe stressful condition and depression. Patient 2 is a female that presented with involuntary opening of mouth and irregular limb tremors at postnatal state after delivery. She also developed clinically similar type of jawopening dystonia, which disappeared with distraction. Psychological examination showed postpartum depression. Conclusions: To our knowledge, this is the first case report of psychogenic jaw opening dystonia. As like these cases, if there is abrupt-onset, notfixed, continual jaw-opening dystonia with clinically inconsistent and incongruous characteristics, it should be considered diagnosis of psychogenic jaw-opening dystonia. P 3.076. TOBACCO INCREASES SYMPTOMS
DYSTONIA
RISK
BUT
MAY
AMELIORATE
Clara Hellberg, Andreas Puschmann. Department for Neurology and Rehabilitational Medicine, Lund, Sweden
P 3.074. A CASE OF HEMIFACIAL SPASM IN YOUNG ADULT Inessa Gabrielyan 1, K. Harutyunyan 2, H. Amirjanyan 3, G. Avagyan 1, H. S. Khachaturyan 4, A. Nazaryan 1, H. Hambardzumyan 1, Manvelyan 1. 1 Department of Neurology, Yerevan State Medical University, Yerevan, Armenia; 2 Department of Neurology, Armenian Republican Medical Center, Yerevan, Armenia; 3 Department of Neurology, Nor Hachn Polyclinic, Kotayk Marz, Armenia; 4 Department of Neurology, Echmiadzin Medical Center, Echmiadzin, Armenia Objectives: Hemifacial spasm is a neuromuscular disorder characterized by frequent involuntary contractions of the muscles on one side of the face. The causes include vascular compression, facial nerve compression by a mass, brainstem lesion such as stroke or multiple sclerosis, and secondary
Objectives: To characterize the role of tobacco use as a risk factor for dystonia and its possible direct impact on dystonia symptoms. Methods: We asked 105 dystonia patients, 117 PD patients and 116 controls about previous and present tobacco consumption. Standardized questionnaires were used. Results were dichotomized into “ever” or “never” users. Dystonia patients who used tobacco after dystonia onset were also asked about alterations in symptoms immediately following tobacco use. Fisher’s Exact Test was used for statistical analyses. Results: A majority of the dystonia patients were “ever” users of tobacco, and this proportion was significantly higher among the dystonia patients compared to “ever” users of tobacco among the PD patients (p¼0.007) or controls (p¼0.014). There was no significant difference between PD patients and controls (p¼0.794; Table).
Group
Number of patients
Average age
Men (%)
Women (%)
Ever used tobacco (%)
Never used tobacco (%)
Dystonia PD Controls
105 117 116
50.9 years 61.6 years 64.7 years
29 (27.6%) 71 (60.7%) 50 (43.1%)
76 (72.4%) 46 (39.3%) 66 (56.9%)
70 (66.7%) 56 (47.9%) 58 (50.0%)
35 (33.3%) 61 (52.1%) 58 (50.0%)
causes such as trauma. A 38 year old male presented to our hospital with a history of transient contractions of facial muscles on the left side for about one month. Patient also noted occasional headaches and fatigue. Methods: On examination left sided hemifacial spasm with frequent clonic contractions was evident. Neurological examination otherwise was normal. Laboratory analyzes were within normal range. Results: Brain MRI scan was performed, which revealed an extensive tumor in brainstem. The findings were consistent with a pontine glioma. Radiotherapy was initiated. On repeated MRI after 3 months follow up no progression was noticed. Conclusions: Even though there are few neurological signs, the possibility of serious underlying cause must always be kept in mind. P 3.075. ATYPICAL PRESENTATION OF PSYCHOGENIC MOVEMENT DISORDER WITH JAW-OPENING DYSTONIA: TWO CASES REPORTS Won Tae Yoon. Department of Neurology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
Forty-two patients reported that they still used tobacco after their onset of symptoms. One patient reported worsening, while nine reported improvement of symptoms with reduced muscle tension or tremor, starting immediately to 15 minutes after tobacco use, and lasting for 10 minutes to several hours. Conclusions: Tobacco may increase the risk of developing dystonia, but some patients report symptomatic improvement during and after tobacco use. The improvement may be due to a pharmacological effect or the relaxing situation when tobacco was consumed. Previous studies reported both improvement 1,4 and worsening 2,3 of dystonia symptoms with tobacco use, and suggested a possible influence of nicotine on other neurotransmitters. References 1. Lees AJ. Hemidystonia relieved by nicotine. Lancet 1984;8407:871. 2. Murase N, Kaji R, Sakamoto T, et al. Nicotine-sensitive writer's cramp. Movement disorders : official journal of the Movement Disorder Society 2000;15:1276-1279. 3. Prashantha DK, Pal PK. Smoking induced worsening of dystonia. A case report. Mov Disord 2009;24:1857-1858.
Methods: We conducted investigation with anamnesis and treatments’ data, appropriate scales for dystonia and Pittsburgh Sleep Quality Index (PSQI), Visual Analogue Scale, McGill questionnaire and Hospital Anxiety and Depression Scale (HADS). The study involved randomly selected 70 dystonia patients (20 generalized dystonia, 25 cervical dystonia and 25 facial dystonia: blepharospasm, oromandibular dystonia and hemifacial spasm) and 10 patients that had treated with DBS last year. . For all patients we did another investigation 6 months after first one and for DBS patients before DBS and 6 months after operation. In addition we try to see factors that were connected with pain and sleep problems. Results: The pain prevalence was 83% and for sleep disturbances 80%. After 6 months the pain was reduced in group treated with Botulinum toxin (from 76% to 20%). After DBS we observed significant decrease in frequency concerning sleep problems (from 90 % to 10% of patients) and pain (from 90% to 20%) (p<0,05). We found that both symptoms were associated with depression. Conclusions: There is rather high frequency of the pain and sleep problems at dystonia patients and they should have to be assessed regularly in our clinical practice. DBS helps in relieving of the pain and sleep problems. References: 1. Eichenser SR, Stebbins GT, Comella CL. Beyond a motor disorder: a prospective evaluation of sleep quality in cervical dystonia. Parkinsonism Relat Disord 2014;20:405-408. 2. Kuyper DJ, Parra V, Aerts S et al. Nonmotor manifestations of dystonia: a systematic review. Mov Disord 2011;26:1206-1217.
e137
Objectives: Jaw-opening dystonia is a rare condition, and only a limited number of cases have been reported in the literature. Dystonia is rarely psychogenic and jaw-opening dystonia is extremely rare. Our aim is to report atypical types of psychogenic movement disorder presenting jawopening dystonia. Methods: Two cases reports and description of clinical characteristics. Results: We report two patients with clinically established psychogenic dystonia. Patient 1 is a female that presented sudden-onset, involuntary opening of mouth one day ago. She developed clinically non-fixed, continual jaw-opening dystonia and hypertonia of jaw and mouth disappeared with distraction. Psychological examination showed severe stressful condition and depression. Patient 2 is a female that presented with involuntary opening of mouth and irregular limb tremors at postnatal state after delivery. She also developed clinically similar type of jawopening dystonia, which disappeared with distraction. Psychological examination showed postpartum depression. Conclusions: To our knowledge, this is the first case report of psychogenic jaw opening dystonia. As like these cases, if there is abrupt-onset, notfixed, continual jaw-opening dystonia with clinically inconsistent and incongruous characteristics, it should be considered diagnosis of psychogenic jaw-opening dystonia. P 3.076. TOBACCO INCREASES SYMPTOMS
DYSTONIA
RISK
BUT
MAY
AMELIORATE
Clara Hellberg, Andreas Puschmann. Department for Neurology and Rehabilitational Medicine, Lund, Sweden
P 3.074. A CASE OF HEMIFACIAL SPASM IN YOUNG ADULT Inessa Gabrielyan 1, K. Harutyunyan 2, H. Amirjanyan 3, G. Avagyan 1, H. S. Khachaturyan 4, A. Nazaryan 1, H. Hambardzumyan 1, Manvelyan 1. 1 Department of Neurology, Yerevan State Medical University, Yerevan, Armenia; 2 Department of Neurology, Armenian Republican Medical Center, Yerevan, Armenia; 3 Department of Neurology, Nor Hachn Polyclinic, Kotayk Marz, Armenia; 4 Department of Neurology, Echmiadzin Medical Center, Echmiadzin, Armenia Objectives: Hemifacial spasm is a neuromuscular disorder characterized by frequent involuntary contractions of the muscles on one side of the face. The causes include vascular compression, facial nerve compression by a mass, brainstem lesion such as stroke or multiple sclerosis, and secondary
Objectives: To characterize the role of tobacco use as a risk factor for dystonia and its possible direct impact on dystonia symptoms. Methods: We asked 105 dystonia patients, 117 PD patients and 116 controls about previous and present tobacco consumption. Standardized questionnaires were used. Results were dichotomized into “ever” or “never” users. Dystonia patients who used tobacco after dystonia onset were also asked about alterations in symptoms immediately following tobacco use. Fisher’s Exact Test was used for statistical analyses. Results: A majority of the dystonia patients were “ever” users of tobacco, and this proportion was significantly higher among the dystonia patients compared to “ever” users of tobacco among the PD patients (p¼0.007) or controls (p¼0.014). There was no significant difference between PD patients and controls (p¼0.794; Table).
Group
Number of patients
Average age
Men (%)
Women (%)
Ever used tobacco (%)
Never used tobacco (%)
Dystonia PD Controls
105 117 116
50.9 years 61.6 years 64.7 years
29 (27.6%) 71 (60.7%) 50 (43.1%)
76 (72.4%) 46 (39.3%) 66 (56.9%)
70 (66.7%) 56 (47.9%) 58 (50.0%)
35 (33.3%) 61 (52.1%) 58 (50.0%)
causes such as trauma. A 38 year old male presented to our hospital with a history of transient contractions of facial muscles on the left side for about one month. Patient also noted occasional headaches and fatigue. Methods: On examination left sided hemifacial spasm with frequent clonic contractions was evident. Neurological examination otherwise was normal. Laboratory analyzes were within normal range. Results: Brain MRI scan was performed, which revealed an extensive tumor in brainstem. The findings were consistent with a pontine glioma. Radiotherapy was initiated. On repeated MRI after 3 months follow up no progression was noticed. Conclusions: Even though there are few neurological signs, the possibility of serious underlying cause must always be kept in mind. P 3.075. ATYPICAL PRESENTATION OF PSYCHOGENIC MOVEMENT DISORDER WITH JAW-OPENING DYSTONIA: TWO CASES REPORTS Won Tae Yoon. Department of Neurology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea
Forty-two patients reported that they still used tobacco after their onset of symptoms. One patient reported worsening, while nine reported improvement of symptoms with reduced muscle tension or tremor, starting immediately to 15 minutes after tobacco use, and lasting for 10 minutes to several hours. Conclusions: Tobacco may increase the risk of developing dystonia, but some patients report symptomatic improvement during and after tobacco use. The improvement may be due to a pharmacological effect or the relaxing situation when tobacco was consumed. Previous studies reported both improvement 1,4 and worsening 2,3 of dystonia symptoms with tobacco use, and suggested a possible influence of nicotine on other neurotransmitters. References 1. Lees AJ. Hemidystonia relieved by nicotine. Lancet 1984;8407:871. 2. Murase N, Kaji R, Sakamoto T, et al. Nicotine-sensitive writer's cramp. Movement disorders : official journal of the Movement Disorder Society 2000;15:1276-1279. 3. Prashantha DK, Pal PK. Smoking induced worsening of dystonia. A case report. Mov Disord 2009;24:1857-1858.