Characteristics of Movement Disorders in Patients with Autoimmune GFAP Astrocytopathy
Abstract
:1. Introduction
2. Patients and Methods
2.1. Patients
2.2. Measurement of CSF GFAP-IgG
2.3. Statistical Analysis
3. Results
3.1. Demographics, Clinical Features, CSF Characteristics, and Neuroimaging in Autoimmune GFAP-A Patients
3.2. Types and Frequencies of Movement Disorders in GFAP-A Patients
3.3. Characteristics of GFAP-A Patients with Movement Disorders
3.4. Literature Review of Movement Disorders in GFAP-A Patients
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
References
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Variables | Patients (%) | Median (Range) |
---|---|---|
Demographics | ||
Age (N = 87) | 51 (5–83) | |
Male | 56/87 (64) | |
Concomitant tumor | 14/87 (16) | |
Concomitant autoimmune disease | 2/87 (2) | |
Period from onset to admission (days) (N = 85) | 12 (1–1054) | |
Clinical findings | ||
Movement disorder | 74/87 (85) | |
Urinary dysfunction | 67/87 (77) | |
Altered consciousness | 66/87 (76) | |
Nuchal rigidity and/or Kernig’s sign | 56/87 (64) | |
Fever | 54/85 (64) | |
Hyperreflexia | 50/86 (58) | |
Headache | 43/85 (51) | |
Cognitive dysfunction | 42/87 (48) | |
Psychosis | 37/87 (43) | |
Papillary edema | 17/48 (43) | |
Weakness | 32/87 (37) | |
Sensory disturbance | 24/87 (28) | |
Respiratory failure | 19/86 (22) | |
Convulsion | 17/87 (20) | |
Cerebrospinal fluid findings | ||
Cell counts (cells/μL) (N = 86) | 79 (4–472) | |
Protein concentrations (mg/dL) (N = 86) | 146 (30–320) | |
Oligoclonal IgG bands | 43/61 (70) | |
Coexisting neural autoantibodies | ||
Anti-MOG antibodies | 2/57 (4) | |
Anti-GAD antibodies | 2/8 (25) | |
Anti-NMDAR antibodies | 2/42 (5) | |
Anti-AQP4 antibodies | 0/66 (0) | |
Brain MRI findings | ||
T2/FLAIR hyperintensity lesions | 76/86 (88) | |
Hyperintensities in white matter | 49/84 (58) | |
Hyperintensities in basal ganglia | 36/84 (43) | |
Hyperintensities in cerebellum | 5/84 (6) | |
Gadolinium enhancement lesions | 53/78 (68) | |
LPRGE patterns | 42/78 (54) | |
Spinal cord MRI findings | ||
Intramedullary T2 hyperintensity lesions | 29/71 (41) | |
Gadolinium enhancement lesions | 24/51 (47) | |
Intramedullary enhancement lesions | 12/51 (24) | |
Meningeal enhancement lesions | 13/51 (25) | |
Therapies | ||
Corticosteroid therapies | 85/87 (98) | |
Period from onset to steroid initiation (day) (N = 83) | 24 (7–1084) |
Movement Disorders | Patients (%) |
---|---|
Ataxia | 43/87 (49) |
Tremor | 39/87 (45) |
Myoclonus | 32/87 (37) |
Dyskinesia | 2/87 (2) |
Opsoclonus | 2/87 (2) |
Rigidity | 2/87 (2) |
Myokymia | 1/87 (1) |
Choreoathetosis | 1/87 (1) |
Variables | With MDs | Without MDs | p | ||
---|---|---|---|---|---|
Patients (%) | Median (Range) | Patients (%) | Median (Range) | ||
Demographics | |||||
Age | 54 (5–83), N = 74 | 30 (17–76), N = 13 | 0.005 | ||
Male | 50/74 (68) | 6/13 (46) | 0.208 | ||
Concomitant tumor | 11/74 (15) | 3/12 (25) | 0.400 | ||
Concomitant autoimmune disease | 2/74 (3) | 0/13 (0) | 1.000 | ||
Period from onset to admission (days) | 12 (1–1054), N = 74 | 8 (1–39), N = 13 | 0.139 | ||
Clinical features | |||||
Urinary disturbance | 59/74 (80) | 8/13 (62) | 0.165 | ||
Altered consciousness | 55/74 (74) | 11/13 (85) | 0.726 | ||
Nuchal rigidity and/or Kernig’s sign | 46/74 (62) | 10/13 (77) | 0.364 | ||
Fever | 44/72 (61) | 10/13 (77) | 0.358 | ||
Hyperreflexia | 43/73 (59) | 7/13 (54) | 0.768 | ||
Cognitive disfunction | 37/74 (50) | 5/13 (38) | 0.553 | ||
Headache | 34/72 (47) | 9/13 (69) | 0.228 | ||
Psychosis | 31/74 (42) | 6/13 (46) | 0.771 | ||
Papillary edema | 16/35 (46) | 1/5 (20) | 0.373 | ||
Weakness | 28/74 (38) | 4/13 (31) | 0.760 | ||
Sensory disturbance | 22/74 (30) | 2/13 (15) | 0.501 | ||
Respiratory failure | 14/73 (19) | 5/13 (38) | 0.150 | ||
Convulsion | 14/74 (19) | 3/10 (30) | 0.712 | ||
Cerebrospinal fluid | |||||
Cell counts (cells/μL) | 79 (4–378), N = 73 | 74 (10–472), N = 13 | 0.109 | ||
Protein concentrations (mg/dL) | 136 (54–320), N = 73 | 156 (30–241), N = 13 | 0.376 | ||
Oligoclonal IgG bands | 39/56 (70) | 4/5 (80) | 1.000 | ||
Coexisting neural autoantibodies | |||||
Anti-MOG antibodies | 2/50 (4) | 0/7 (0) | 1.000 | ||
Anti-GAD antibodies | 1/7 (14) | 1/1 (100) | 0.250 | ||
Anti-NMDAR antibodies | 2/38 (5) | 0/4 (0) | 1.000 | ||
Brain MRI | |||||
T2/FLAIR hyperintensity lesions | 64/73 (88) | 12/13 (92) | 1.000 | ||
Hyperintensities in white matter | 43/71 (61) | 6/13 (46) | 0.371 | ||
Hyperintensities in basal ganglia | 27/71 (38) | 9/13 (70) | 0.065 | ||
Hyperintensities in cerebellum | 4/71 (6) | 1/13 (8) | 0.578 | ||
Gadolinium enhancement lesions | 47/67 (70) | 6/11 (55) | 0.316 | ||
LPRGE | 36/67 (54) | 6/11 (55) | 1.000 | ||
Spinal cord MRI | |||||
Intramedullary T2 hyperintensity lesions | 25/60 (42) | 4/11 (36) | 1.000 | ||
Gadolinium enhancement lesions | 21/44 (48) | 3/7 (45) | 1.000 | ||
Intramedullary enhancement lesions | 11/44 (25) | 1/7 (14) | 1.000 | ||
Meningeal enhancement lesions | 11/44 (25) | 2/7 (29) | 1.000 | ||
Therapies | |||||
Corticosteroid therapies | 72/74 (97) | 13/13 (100) | 1.000 | ||
Period from onset to steroid initiation (days) | 25 (7–1084), N = 70 | 14 (8–60), N = 13 | 0.136 |
Movement Disorders | Number of Reports | References |
---|---|---|
Ataxia | 23 | [1,2,3,4,5,8,9,13,15,16,17,19,22,23,24,25,26,27,32,36,38,40,41] |
Tremor | 22 | [1,2,6,8,13,14,20,21,24,26,27,28,29,30,31,33,34,35,36,37,38,39] |
Myoclonus | 12 | [6,8,13,14,15,16,25,32,33,35,36,37] |
Dyskinesia | 6 | [4,5,6,17,23,41] |
Hyperekplexia | 3 | [6,32,35] |
Dystonia | 2 | [6,23] |
Parkinsonism | 2 | [6,14] |
Chorea | 1 | [13] |
Axial stiffness | 1 | [35] |
Grimacing | 1 | [18] |
Oral movements | 1 | [18] |
Catatonia | 1 | [18] |
Oculogyric crises | 1 | [35] |
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Kimura, A.; Takekoshi, A.; Shimohata, T. Characteristics of Movement Disorders in Patients with Autoimmune GFAP Astrocytopathy. Brain Sci. 2022, 12, 462. https://doi.org/10.3390/brainsci12040462
Kimura A, Takekoshi A, Shimohata T. Characteristics of Movement Disorders in Patients with Autoimmune GFAP Astrocytopathy. Brain Sciences. 2022; 12(4):462. https://doi.org/10.3390/brainsci12040462
Chicago/Turabian StyleKimura, Akio, Akira Takekoshi, and Takayoshi Shimohata. 2022. "Characteristics of Movement Disorders in Patients with Autoimmune GFAP Astrocytopathy" Brain Sciences 12, no. 4: 462. https://doi.org/10.3390/brainsci12040462