Case Reports

AntiThyroid Peroxidase/AntiThyroglobulin AntibodyRelated Neurologic Disorder Responsive to Steroids Presenting with Pure Acute Onset Chorea

Authors: {'first_name': 'Ritwik', 'last_name': 'Ghosh'},{'first_name': 'Subhankar', 'last_name': 'Chatterjee'},{'first_name': 'Souvik', 'last_name': 'Dubey'},{'first_name': 'Alak', 'last_name': 'Pandit'},{'first_name': 'Biman Kanti', 'last_name': 'Ray'},{'first_name': 'Juli\xc3\xa1n', 'last_name': 'Benito-Le\xc3\xb3n'}

Abstract

Background: Pure acute onset chorea without encephalopathy has rarely been reported in anti-thyroid peroxidase (anti-TPO)/anti-thyroglobulin (anti-TG) antibody-related neurologic disorders responsive to steroids (ATANDS).

Case report: We report a 16-year-old female who presented with acute chorea without encephalopathy. Anti-TPO antibodies were found to be strongly positive (>1200 IU/ml) along with anti-thyroglobulin and anti-thyroid stimulating hormone receptor antibodies. After pulse intravenous methylprednisolone therapy (1 g/day for five consecutive days), all the movements seized, and she was discharged with oral prednisolone 30 mg/day with gradual tapering over next three months. After one year of follow-up, she is stable, drug-free, and never had any other problems.

Discussion: Anti-thyroid antibodies testing should be included in routine/conventional panel that is done for elucidating causes of chorea as ATANDS can be easily missed and is treatable with widely available, relatively low-cost drugs like steroids with a promising outcome.

Keywords: Anti-thyroid peroxidase/anti-thyroglobulin antibody-related neurologic disorders responsive to steroids, Anti-thyroid peroxidase antibodies, Anti-thyroglobulin antibodies, Autoimmune, Chorea, Movement Disorders, Review, Steroid-responsive encephalopathy a 
DOI: http://doi.org/10.5334/tohm.175
 Accepted on 25 May 2020            Submitted on 04 May 2020

In autoimmune thyroid disorders, albeit known to affect only 1% of the population, focal or sub-clinical autoimmune thyroid inflammation can be found in around 15% of biochemically euthyroid population [1, 2, 3]. Anti-thyroid peroxidase (anti-TPO) and anti-thyroglobulin (anti-TG) antibodies are considered diagnostic markers of autoimmune thyroid disorders [1]. Neurological manifestations associated with autoimmune thyroid disorders have been frequently under-documented in literature [4], being the most protean amongst these disorders Hashimoto’s encephalopathy [5, 6, 7]. Spectrum of this disorder an range from subtle behavioral/personality changes to movement disorders, seizures, dementia, encephalopathy, stroke, coma and death [5, 6, 7, 8]. Patients can also present with movement disorders without encephalopathy and cognitive impairment [5, 6, 7]. There is no pathognomonic clinical, serological, biochemical, electrophysiological or imaging markers [5, 6, 7]. In addition, there are no good predictors of treatment response to steroids; [9] in fact, in a recent study [9], only 31% patients completely responded to these drugs. Similarly, in other studies, only 56% and 36% patients with suspected Hashimoto’s encephalopathy responded to steroids [10, 11]. Despite this, response to steroids seems to be the only partially consistent feature of this disorder hence renamed as “steroid responsive encephalopathy associated with autoimmune thyroiditis” (SREAT) [12], but neither response to steroids nor association with thyroiditis is steadfast [13]. Termasarasab et al., [14] have recently proposed “anti-TPO/TG antibody-related neurologic disorders responsive to steroids (ATANDS)” to be the renamed entity that would include the complete spectrum. Reported movement disorders that have been associated with ATANDS can be either “encephalopathic” or “non-encephalopathic” [5, 6, 7, 14].

We had treated a patient with pure acute chorea who rapidly improved with corticosteroids. Here, we describe the case with a complete report. We also provide a review of the literature, which was performed to collect and summarize the present state of knowledge on movement disorders associated with ATANDS.

Case presentation

A 16-year-old female presented to the neurology outpatient department with complaint of acute onset involuntary weird and quirky movements of all four limbs for last four days, which were irregular, asymmetric, rapid, unpredictable, purposeless, jerky and flowing from distal to proximally and that disappeared completely during sleep. Her past medical history was unremarkable. No associated febrile episode, seizure, headache, visual disturbances, behavioral changes, personality changes, forgetfulness, attention problems, or self-care inadequacy were noted. She had no history of any drug intake for any disease or substance abuse in recent past. No history was suggestive of any connective tissue disorder or thyroid dysfunction. Nobody in family had any neurological disease. On completion of an unremarkable general survey, detailed neurological examination revealed generalized chorea involving all extremities (right > left) with classic Jack in the box tongue and Milkman’s grip signs. Precise and meticulous cognitive assessment failed to unveil any impairment. Neither motor weakness, nor sensory deficits, nor signs of meningeal irritation and cranial nerve deficits were noted. Slit lamp examination ruled out Kayser-Fleischer ring.

No cognitive domain seemed to be affected and family history was negative; therefore, Huntington’s disease, Huntington’s disease-like syndromes, dentatorubral-pallidoluysian atrophy, and deposition disorders were virtually excluded. Hence, working diagnosis kept was acute onset generalized chorea without cognitive impairment. Differentials considered were: 1) metabolic chorea, 2) rheumatic chorea, 3) dysthyroidism associated chorea, 4) autoimmune chorea, 5) vascular chorea, and 6) chorea gravidarum.

Complete hemogram, thyroid, liver, kidney functions, electrolytes, arterial blood gas analysis and HbA1C were normal. A urine beta human chorionic gonadotropin and abdominal ultrasound ruled out any pregnancy. Serologies for human immunodeficiency virus, hepatitis C, and hepatitis B were negative. 24 hours urinary copper and serum ceruloplasmin levels were within normal range. Echocardiography, serial anti-streptolysin O titers, and anti-DNase B antibodies levels ruled out possibility of Sydenham’s chorea. Anti-Nuclear Antibody (ANA) screening using HEp-2 cells, ANA profile, antiphospholipid antibodies, and antineutrophil cytoplasmic antibodies (cANCA and pANCA) were found to be negative. Autoantibodies directed against voltage gated potassium channel and anti-N-methyl-D-aspartate receptor antibodies were also negative. Magnetic resonance imaging of brain, electroencephalogram and cerebrospinal fluid analyses were otherwise normal.

Serum anti-TPO antibodies were found to be strongly positive (>1200 IU/ml) along with anti-TG and anti-thyroid stimulating hormone receptor antibodies. Patient was put on pulse intravenous methylprednisolone therapy (1 g/day for five consecutive days). All the movements seized and she was discharged with oral prednisolone 30 mg/day with gradual tapering over next three months. Tests were rerun with similar results, but anti-thyroid stimulating hormone receptor antibodies, which were within normal range this time around. At present, after one year of follow-up, she remains stable, drug-free and without any other problems.

Discussion

ATANDS with associated movement disorders have been described previously (Table 1) [15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30, 31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41, 42, 43, 44, 45, 46, 47, 48, 49, 50]. We have reported a 16-year-old female with ATANDS who presented with acute pure chorea without encephalopathy. ATANDS presenting with chorea is exceedingly rare. For example, Miranda et al., [47] described a middle-aged female with acute onset rapidly worsening choreo-athetosis with dystonia and slurred speech which came out to be a case of ATANDS. Sharan A et al., [41] reported an aged female with ATANDS, who developed abrupt onset behavioral changes along with asymmetric florid chorea. Taurin G et al., [19] narrated behavioral abnormality with psychotic features along with bilateral and axial choreic movements in an elderly female. Our patient had no behavioral abnormalities or any other extrapyramidal or cerebellar features unlike those previously mentioned cases [19, 41, 47]. In all those cases steroid resulted in good yield alike our patient [19, 41, 47].

Table 1

Movement disorders associated with anti-TPO/TG antibody-related neurologic disorders responsive to steroids.

Author and year of publication Age/Sex Type of movement disorder Thyroid status Anti-thyroid antibody Neuroimaging Treatment Outcome
1. Mehta AB et al., [15] 1981 17/F Torsion dystonia Primary hyperthyroidism/thyrotoxicosis Anti-TG+
Anti-TPO+		 No data available Carbimazole and radioactive iodine Euthyroid, dystonia was very mild with a slight tendency for torticollis and scoliosis to the right, and for the right outstretched arm to hyperpronate, provided drug compliant
2. Javaid A and Hilton DD [16] 1988 15/F Generalized choreo-athetosis Primary hyperthyroidism Anti-TG+
Anti-TPO+		 No data available Propranolol, carbimazole, tetrabenazine, chlorpromazine, and haloperidol Refractory chorea. Chlorpromazine and tetrabenazine only partially suppressed it. At six months it was persisting. Haloperidol almost completely abolished chorea. It returned whenever she stopped taking haloperidol. Recurrence occurred 16 months after she first presented
3. Baba M et al., [17] 1992 23/F Hemichorea Primary hyperthyroidism/Graves’ disease Anti-TG+
Anti-TPO-		 No changes Metoprolol, thiamazole and chlorpromazine Improved
4. Hernández Echebarría LE et al., [18] 2000 41/F Opsoclonus, myoclonus, and gait ataxia Euthyroid→ subclinical hypothyroidism Anti-TPO+ SPECT showed decreased perfusion in the left fronto-parietal region and in the right basal ganglia Antibiotics, acyclovir and valproate followed by L-thyroxin and steroids At one-year follow-up, CSF analysis, SPECT, and electroencephalogram were normal. Anti-TPO decreased. She remained well at the last visit, two years after the onset of neurologic symptoms.
5. Taurin G et al., [19] 2002 77/F Bilateral and axial choreic movements Primary hypothyroidism Anti-TG+
Anti-TPO+		 Cortico-subcortical atrophy L-thyroxin and oral prednisolone With 60 mg/day of prednisolone, chorea disappeared and reappeared again; on increasing dose to 80 mg/d, it disappeared. At 3 weeks, the patient was clinically normal. No relapse during 8 months of follow-up
6. Erickson JC et al., [20] 2002 34/M Myorhythmia, myoclonus, and tremor Primary hypothyroidism Anti-TG+
Anti-TPO+		 No changes IVMP followed by oral prednisolone Moderate improvement with residual mild cognitive impairment and subtle facial myorhythmia
7. Erickson JC et al., [20] 2002 38/M Palatal tremor Euthyroid Anti-TPO+
Anti-TG-		 Venous anomaly in hypothalamus IVMP followed by oral prednisolone Moderate improvement in seizures, but cognitive impairment persisting
8. Nagpal T and Pande S [21] 2004 52/F Parkinsonism and myoclonus Subclinical hypothyroidism Anti-TPO+
Anti-TG-		 Cerebral atrophy IVMP followed by oral prednisolone, PLEX, and finally by oral prednisolone No improvement with IVMP; significant improvement 10days after PLEX
9. Loh LM et al., [22] 2005 40/M Propriospinal or segmental myoclonus, Spasmodic truncal flexion Primary hyperthyroid/Graves’ disease TRAB+
Anti-TG+
TSI+
Anti-TPO-		 No changes Clonazepam and propylthiouracil Euthyroidism established and symptoms improved
10. Tan EK et al., [23] 2006 Middle aged/M Bilateral postural hand tremor and task-specific dystonia-writer’s cramp Primary hyperthyroidism/Graves’ disease Anti-TG+ No changes Carbimazole Euthyroidism achieved and symptoms improved
11. Guimaraes J et al., [24] 2007 60/M Painful legs and moving toes syndrome, bradykinesia, and dystonia Primary hypothyroidism Anti-TG+
Anti-TPO+		 Subcortical white matter lesions Oral prednisolone No improvement
12. Tan EK et al., [25] 2008 50/F Isolated orthostatic tremor Primary hyperthyroidism/Graves’ disease TRAB+
Anti-TG+
Anti-TPO+		 No changes carbimazole Complete resolution
13. Ku CR et al., [26] 2008 42/F Generalized chorea Primary hyperthyroidism/Graves’ disease TRAB+
Anti-TG-
Anti-TPO+		 No changes IVMP, propylthiouracil, propranolol, trihexyphenidyl, ropinirole, clonazepam and quetiapine Improved
14. Yu JH and Weng YM [27] 2009 17/F Chorea Primary hyperthyroidism/Graves’ disease Anti-TPO+ SPECT revealed decreased perfusion to the right anterior temporal cortex Propylthiouracil and propranolol Complete resolution
15. Broch L and Amthor KF [28] 2010 66/F Myoclonus, tremor Euthyroid Anti-TPO++ No changes Systemic steroids Improved
16. Salazar R et al., [29] 2012 59/M Opsoclonus and gait ataxia Euthyroid Anti-TG+
Anti-TPO+		 No changes IVIG/IVMP After three months of therapy with corticosteroids improved, but not with IVIG
17. Liu MY et al., [30] 2012 75/M Paroxysmal kinesigenic dyskinesia Unknown Anti-TPO+ No changes IVMP followed by oral prednisone taper Back to baseline in 20days
18. Inoue K et al., [31] 2012 63/F Micrography, parkinsonian gait and tremor Euthyroid Anti-TPO+ White matter ischemic changes IVMP followed by oral prednisolone Improved
19. Ryan SA et al., [32] 2012 48/M Myoclonus Subclinical hypothyroidism Anti-TPO++
Anti-TG-		 No changes Oral prednisolone Improved
20. Park J et al., [33] 2012 16/M Asymmetric chorea Primary hyperthyroidism/Graves’ disease TRAB+
Anti-TG+		 No changes Propylthiouracil and propranolol Improved
21. Nakavachara P et al., [34] 2013 14/M Choreo-athetosis Primary hyperthyroidism/Graves’ disease Anti-TPO+
Anti-TG+		 No data available Methimazole, propranolol, and IV potassium Improved
22. Kaminska A et al., [35] 2013 23/F Hemi-chorea Primary hyperthyroidism/Graves’ disease TRAB+
Anti-TPO+		 No changes Thiamazole, prednisolone, haloperidol, thioridazine Subsidence of symptoms
23. Ghoreishi E et al., [36] 2013 32/M Palatal myoclonus Euthyroid Anti-TG+
Anti-TPO+		 Bilateral striatal hyperintensity on T2WI Oral prednisolone Improved
24. Philip R et al., [37] 2014 18/M Myoclonus and tremor Primary hyperthyroidism Anti-TPO+ Non-specific white matter changes and pituitary hyperplasia IVMP followed by oral prednisolone and L-thyroxin Significant improvement
25. Saygi S et al., [38] 2014 12/M Motor tics Euthyroid Anti-TPO++
Anti-TG+		 No changes Oral prednisolone Improved
26. Rozankovic PB et al., [39] 2015 27/F Myoclonus-Dystonia and choreo-athetosis Euthyroid Anti-TPO+ No changes IVMP followed by oral Prednisolone Complete resolution
27. Lee HJ et al., [40] 2015 30/M Ocular flutter, limb and gait ataxia, myoclonus, and truncal titubation Euthyroid Anti-TG+
Anti-TPO-		 No changes IVMP followed by oral prednisolone Improved
28. Sharan A et al., [41] 2015 78/F Choreiform movements Euthyroid Anti-TPO+ Atrophy IVMP followed by oral prednisolone Improved
29. Sheetal SK et al., [42] 2016 66/M Action-myoclonus, parkinsonism (corticobasal disease-variant-like) Euthyroid Anti-TPO+
Anti-TG-		 Small right thalamic hematoma IVMP followed by oral prednisolone Improved
30. Ramcharan K et al., [43] 2016 34/F Bilateral hand postural tremor Euthyroid Anti-TG+
TRAB+
Anti-TPO-		 No changes Oral prednisolone Improved
31. Correia I et al., [44] 2016 61/F Limb myoclonus Primary hypothyroidism Anti-TPO+ SPECT revealed hypoperfusion in frontal, temporal, and parietal regions with left predominance L-thyroxin, IVMP followed by oral prednisolone and azathioprine Resolution
32. Kelly DM et al., [45] 2017 64/M Abdominal tremor and abdominal wall dyskinesia Primary hyperthyroidism Anti-TPO+ No changes Carbimazole and supportive Complete resolution
33. Keshavaraj A and Anpalagan J [46] 2018 23/F Oro-lingual dyskinesia Primary hyperthyroidism Anti-TPO+
TRAB+		 No changes IVMP, carbimazole, and propranolol Significant improvement
34. Miranda M et al., [47] 2018 34/F Choreo-athetosis, dystonia and ataxia Euthyroid Anti-TG+
Anti-TPO+		 No changes IVMP and IVIG Complete resolution
35. Miranda M et al., [47] 2018 61/M Myoclonus-dystonia Euthyroid Anti-TPO+ No changes IVMP, IVIG, PLEX and rituximab Incomplete resolution
36. Mohd Fauz NA et al., [48] 2019 50/F Relapsing-remitting opsoclonus-myoclonus-ataxia syndrome Subclinical hyperthyroidism Anti-TG-
Anti-TPO+		 Lesions in cortical and subcortical regions, pons and midbrain IVMP and PLEX Improved
37. Delhasse S et al., [49] 2019 60/F Complex dyskinesia, high-amplitude myoclonic jerks, mild chorea, and postural tremor Primary hyperthyroid/Graves’ disease TRAB+
Anti-TG+
Anti-TPO+
Isotope scan+		 No changes Carbimazoleàpropylthiouracilàradio-active iodine Complete resolution
38. ShreeS et al., [50] 2020 60/F Tremor, myoclonus and catatonia Euthyroid Anti-TPO+ No changes IVMP followed by oral prednisolone. Improved

F: female; M: male; Anti-TPO: anti-thyroid peroxidase antibody; Anti-TG: anti-thyroglobulin antibody; TSI: thyroid stimulating immunoglobulin; TRAB: TSH receptor antibody; CSF: cerebrospinal fluid; SPECT: Single-photon emission computed tomography; IV: intravenous; MP: methylprednisolone; IG: immunoglobulin; PLEX: plasma exchange; MRI: magnetic resonance imaging; ‘+’: high/positive titer; ‘–’: low/negative titer.

Etiopathogenetic factors for chorea are believed to be a) a hypersensitivity of dopaminergic receptors to dopamine due to a thyrotoxic state; [17, 51] b) derangements in cerebral perfusion as reported in cases of acute onset chorea associated with other etiologies [27, 52, 53] and substantiated by single-photon emission computed tomography and positron emission tomography imaging; [54, 55] c) autoimmune central nervous system vasculitis; [8, 56] and d) anti-thyroid antibody mediated effects on neurons [7, 57, 58, 59]. However, for neurological manifestations like chorea, anti-thyroid antibodies are extremely sensitive, but lack specificity [60]. And whether they are pathogenic or just a marker of the disease or just an epiphenomenon, remains elusive [6, 7, 60, 61]. Presence of anti-thyroid antibodies in general population is well established even in absence of neurologic disorders and usually acting as a confounding factor in diagnosis [60]. Further, levels of anti-thyroid antibodies do not correlate well with disease severity and often persist in high levels after the treatment and clinical response [6, 62, 63].

In conclusion, our experience with the current case and our review of the literature strongly suggest that ATANDS/SREAT can rarely present with movement disorders alone and may be a definite separate entity. Anti-thyroid antibody testing should be included in routine/conventional panel that is done for elucidating causes of chorea as this disorder can be easily missed and is treatable with widely available, relatively low-cost drugs like steroids with a promising outcome.

Funding Information

This research was supported by FEDER funds.

R. Ghosh, S. Chatterjee, S. Dubey, A. Pandit, B. Kanti Ray report no relevant disclosures. J. Benito-León is supported by the National Institutes of Health, Bethesda, MD, USA (NINDS #R01 NS39422), European Commission (grant ICT-2011-287739, NeuroTREMOR), the Ministry of Economy and Competitiveness (grant RTC-2015-3967-1, NetMD—platform for the tracking of movement disorder), and the Spanish Health Research Agency (grant FIS PI12/01602 and grant FIS PI16/00451).

Competing Interests

The authors have no competing interests to declare.

References

  1. Stathatos N, Daniels GH. Autoimmune thyroid disease. Current opinion in rheumatology. 2012; 24: 70–75. DOI: https://doi.org/10.1097/BOR.0b013e32834ddb27 

  2. Antonelli A, Ferrari SM, Corrado A, Di Domenicantonio A, Fallahi P. Autoimmune thyroid disorders. Autoimmunity reviews. 2015; 14: 174–180. DOI: https://doi.org/10.1016/j.autrev.2014.10.016 

  3. Meena A, Nagar P. Pregnancy Outcome in Euthyroid Women with Anti-Thyroid Peroxidase Antibodies. Journal of obstetrics and gynaecology of India. 2016; 66: 160–165. DOI: https://doi.org/10.1007/s13224-014-0657-6 

  4. Afreen E, Humayun M, Sheikh A. Neurological spectrum of diseases associated with autoimmune thyroid disease: a case series to recognize diagnostically helpful signs. (P1.347). Neurology. 2018; 90: P1.347. 

  5. Montagna G, Imperiali M, Agazzi P, et al. Hashimoto’s encephalopathy: A rare proteiform disorder. Autoimmunity reviews. 2016; 15: 466–476. DOI: https://doi.org/10.1016/j.autrev.2016.01.014 

  6. Zhou JY, Xu B, Lopes J, Blamoun J, Li L. Hashimoto encephalopathy: literature review. Acta neurologica Scandinavica. 2017; 135: 285–290. DOI: https://doi.org/10.1111/ane.12618 

  7. de Holanda NC, de Lima DD, Cavalcanti TB, Lucena CS, Bandeira F. Hashimoto’s encephalopathy: systematic review of the literature and an additional case. J Neuropsychiatry Clin Neurosci. 2011; 23: 384–390. DOI: https://doi.org/10.1176/jnp.23.4.jnp384 

  8. Nolte KW, Unbehaun A, Sieker H, Kloss TM, Paulus W. Hashimoto encephalopathy: a brainstem vasculitis? Neurology. 2000; 54: 769–770. DOI: https://doi.org/10.1212/WNL.54.3.769 

  9. Mattozzi S, Sabater L, Escudero D, et al. Hashimoto encephalopathy in the 21st century. Neurology. 2020; 94: e217–e224. DOI: https://doi.org/10.1212/WNL.0000000000008785 

  10. Castillo P, Woodruff B, Caselli R, et al. Steroid-responsive encephalopathy associated with autoimmune thyroiditis. Archives of neurology. 2006; 63: 197–202. DOI: https://doi.org/10.1001/archneur.63.2.197 

  11. Litmeier S, Prüss H, Witsch E, Witsch J. Initial serum thyroid peroxidase antibodies and long-term outcomes in SREAT. Acta neurologica Scandinavica. 2016; 134: 452–457. DOI: https://doi.org/10.1111/ane.12556 

  12. Mahad DJ, Staugaitis S, Ruggieri P, et al. Steroid-responsive encephalopathy associated with autoimmune thyroiditis and primary CNS demyelination. J Neurol Sci. 2005; 228: 3–5. DOI: https://doi.org/10.1016/j.jns.2004.08.015 

  13. Ferracci F, Carnevale A. The neurological disorder associated with thyroid autoimmunity. J Neurol. 2006; 253: 975–984. DOI: https://doi.org/10.1007/s00415-006-0170-7 

  14. Termsarasab P, Pitakpatapee Y, Frucht SJ, Srivanitchapoom P. Steroid-responsive Encephalopathy Associated with Autoimmune Thyroiditis (SREAT) Presenting with Pure Cerebellar Ataxia. Tremor Other Hyperkinet Mov (N Y). 2018; 8: 585. DOI: https://doi.org/10.5334/tohm.420 

  15. Mehta AB, Marsden CD. Latent idiopathic torsion dystonia provoked by thyrotoxicosis. J Neurol Neurosurg Psychiatry. 1981; 44: 93–94. DOI: https://doi.org/10.1136/jnnp.44.1.93 

  16. Javaid A, Hilton DD. Persistent chorea as a manifestation of thyrotoxicosis. Postgraduate medical journal. 1988; 64: 789–790. DOI: https://doi.org/10.1136/pgmj.64.756.789 

  17. Baba M, Terada A, Hishida R, Matsunaga M, Kawabe Y, Takebe K. Persistent hemichorea associated with thyrotoxicosis. Internal medicine (Tokyo, Japan). 1992; 31: 1144–1146. DOI: https://doi.org/10.2169/internalmedicine.31.1144 

  18. Hernandez Echebarria LE, Saiz A, Graus F, et al. Detection of 14-3-3 protein in the CSF of a patient with Hashimoto’s encephalopathy. Neurology. 2000; 54: 1539–1540. DOI: https://doi.org/10.1212/WNL.54.7.1539 

  19. Taurin G, Golfier V, Pinel JF, et al. Choreic syndrome due to Hashimoto’s encephalopathy. Mov Disord. 2002; 17: 1091–1092. DOI: https://doi.org/10.1002/mds.10230 

  20. Erickson JC, Carrasco H, Grimes JB, Jabbari B, Cannard KR. Palatal tremor and myorhythmia in Hashimoto’s encephalopathy. Neurology. 2002; 58: 504–505. DOI: https://doi.org/10.1212/WNL.58.3.504 

  21. Nagpal T, Pande S. Hashimoto’s encephalopathy: response to plasma exchange. Neurology India. 2004; 52: 245–247. 

  22. Loh LM, Hum AY, Teoh HL, Lim EC. Graves’ disease associated with spasmodic truncal flexion. Parkinsonism Relat Disord. 2005; 11: 117–119. DOI: https://doi.org/10.1016/j.parkreldis.2004.08.004 

  23. Tan EK, Chan LL. Movement disorders associated with hyperthyroidism: expanding the phenotype. Mov Disord. 2006; 21: 1054–1055. DOI: https://doi.org/10.1002/mds.20883 

  24. Guimaraes J, Santos L, Bugalho P. Painful legs and moving toes syndrome associated with Hashimoto’s disease. European journal of neurology. 2007; 14: 343–345. DOI: https://doi.org/10.1111/j.1468-1331.2006.01513.x 

  25. Tan EK, Lo YL, Chan LL. Graves disease and isolated orthostatic tremor. Neurology. 2008; 70: 1497–1498. DOI: https://doi.org/10.1212/01.wnl.0000310405.36026.92 

  26. Ku CR, Park HJ, Hong SJ, et al. A Case of Graves’ Disease Presenting with Chorea. J Korean Endocr Soc. 2008; 23: 342–346. DOI: https://doi.org/10.3803/jkes.2008.23.5.342 

  27. Yu JH, Weng YM. Acute chorea as a presentation of Graves disease: case report and review. The American journal of emergency medicine. 2009; 27: 369.e361–369.e363. DOI: https://doi.org/10.1016/j.ajem.2008.05.031 

  28. Broch L, Amthor KF. A 66-year-old woman with confusion, aphasia, and myoclonus. Tidsskrift for den Norske laegeforening: tidsskrift for praktisk medicin, ny raekke. 2010; 130: 1041–1043. DOI: https://doi.org/10.4045/tidsskr.09.0697 

  29. Salazar R, Mehta C, Zaher N, Miller D. Opsoclonus as a manifestation of Hashimoto’s encephalopathy. J Clin Neurosci. 2012; 19: 1465–1466. DOI: https://doi.org/10.1016/j.jocn.2012.02.012 

  30. Liu MY, Zhang SQ, Hao Y, Zheng HM. Paroxysmal kinesigenic dyskinesia as the initial symptom of Hashimoto encephalopathy. CNS neuroscience & therapeutics. 2012; 18: 271–273. DOI: https://doi.org/10.1111/j.1755-5949.2012.00297.x 

  31. Inoue K, Kitamura J, Yoneda M, Imamura E, Tokinobu H. Hashimoto’s encephalopathy presenting with micrographia as a typical feature of parkinsonism. Neurological sciences: official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology. 2012; 33: 395–397. DOI: https://doi.org/10.1007/s10072-011-0750-x 

  32. Ryan SA, Kennedy C, Harrington HJ. Steroid-responsive encephalopathy associated with autoimmune thyroiditis presenting as confusion, dysphasia, and myoclonus. Case reports in medicine. 2012; 2012: 782127. DOI: https://doi.org/10.1155/2012/782127 

  33. Park J, Kim JG, Park SP, Lee HW. Asymmetric chorea as presenting symptom in Graves’ disease. Neurological sciences: official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology. 2012; 33: 343–345. DOI: https://doi.org/10.1007/s10072-011-0679-0 

  34. Nakavachara P, Suppakrucha M, Likasitwattanakul S. Thyrotoxic periodic paralysis and chorea: two uncommon neuromuscular complications in an adolescent with newly diagnosed Graves disease. Int J Pediatr Endocrinol. 2013; 2013: P145–P145. DOI: https://doi.org/10.1186/1687-9856-2013-S1-P145 

  35. Kaminska A, Kaminski M, Nowaczewska M, Ksiazkiewicz B, Junik R. Chorea, convulsive seizures and cognitive impairment in a patient with autoimmune thyroid disease. Neurological sciences: official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology. 2013; 34: 263–264. DOI: https://doi.org/10.1007/s10072-012-0990-4 

  36. Ghoreishi E, Shahidi GA, Rohani M, Nabavi M, Aghaei M, Akhoundi FH. Palatal-Myoclonus as a Presentation of Hashimoto Encephalopathy: an interesting case report. Iranian journal of psychiatry. 2013; 8: 149–151. 

  37. Philip R, Saran S, Gutch M, Gupta K. An unusual presentation of Hashimoto’s encephalopathy. Indian journal of endocrinology and metabolism. 2014; 18: 113–115. DOI: https://doi.org/10.4103/2230-8210.126589 

  38. Saygi S, Ozkale Y, Erol I. Tic disorder probably associated with steroid responsive encephalopathy with autoimmune thyroiditis (SREAT). Indian journal of pediatrics. 2014; 81: 1105–1107. DOI: https://doi.org/10.1007/s12098-014-1374-y 

  39. Rozankovic PB, Rozankovic M, Susak I, Vlahovic I, Sporis D. Steroid-responsive autoimmune encephalopathy associated with autoimmune thyroiditis (SREAT) presenting with myoclonus-dystonia syndrome. J Neurol Sci. 2015; 354: 110–111. DOI: https://doi.org/10.1016/j.jns.2015.04.021 

  40. Lee HJ, Jeon K, Lee JY, et al. Ocular flutter and ataxia without cognitive impairment associated with steroid-responsive encephalopathy associated with autoimmune thyroiditis (SREAT). J Neurol Sci. 2015; 359: 86–87. DOI: https://doi.org/10.1016/j.jns.2015.10.037 

  41. Sharan A, Sengupta S, Mukhopadhyay S, Ghosh B. Hashimoto’s Encephalopathy Presenting with Chorea. The Journal of the Association of Physicians of India. 2015; 63: 83–84. DOI: https://doi.org/10.4103/0028-3886.170064 

  42. Sheetal SK, Mathew R, Peethambaran B. Hashimoto’s encephalopathy as a treatable cause of corticobasal disease. Annals of Indian Academy of Neurology. 2016; 19: 285–286. DOI: https://doi.org/10.4103/0972-2327.176859 

  43. Ramcharan K, Hosein N, Teelucksingh JD, Rampersad F, Teelucksingh S. Prominent Bilateral Hand Tremor in Hashimoto’s Encephalopathy: A Video Demonstration. Tremor Other Hyperkinet Mov (N Y). 2016; 6: 419. DOI: https://doi.org/10.5334/tohm.305 

  44. Correia I, Marques IB, Ferreira R, Sousa L. Encephalopathy Associated with Autoimmune Thyroid Disease: A Potentially Reversible Condition. Case reports in medicine. 2016; 2016: 9183979. DOI: https://doi.org/10.1155/2016/9183979 

  45. Kelly DM, Lynch T, Casserly LF. Abdominal tremor in thyrotoxicosis. Neurology. 2017; 89: 1424–1425. DOI: https://doi.org/10.1212/WNL.0000000000004403 

  46. Keshavaraj A, Anpalagan J. Reversible Orolingual Dyskinesia: An Unknown Presentation of Hashimoto’s Toxic Encephalopathy. Journal of Case Reports. 2018: 169–171. DOI: https://doi.org/10.17659/01.2018.0045 

  47. Miranda M, Bustamante ML, Campero M, et al. Movement disorders in non-encephalopathic Hashimoto’s thyroiditis. Parkinsonism Relat Disord. 2018; 55: 141–142. DOI: https://doi.org/10.1016/j.parkreldis.2018.06.008 

  48. Mohd Fauzi NA, Abdullah S, Tan AH, Mohd Ramli N, Tan CY, Lim SY. Relapsing encephalopathy with dancing eyes and jerky limbs. Parkinsonism Relat Disord. 2019. DOI: https://doi.org/10.1016/j.parkreldis.2019.02.025 

  49. Delhasse S, Debove I, Arnold-Kunz G, Ghika JA, Chabwine JN. Erratic movement disorders disclosing Graves’ disease and paralleling thyroid function but not autoantibody levels. The Journal of international medical research. 2019:300060518816873. DOI: https://doi.org/10.1177/0300060518816873 

  50. Shree R, Madhaw G, Manchanda R, Radhakrishnan D, Kumar N. Steroid responsive catatonia: A case of Hashimoto’s encephalopathy. Annals of Movement Disorders. 2020; 3: 51–55. DOI: https://doi.org/10.4103/AOMD.AOMD_32_19 

  51. Klawans HL, Jr., Shenker DM. Observations on the dopaminergic nature of hyperthyroid chorea. Journal of neural transmission. 1972; 33: 73–81. DOI: https://doi.org/10.1007/BF01244729 

  52. Watanabe T, Onda H. Hemichorea with antiphospholipid antibodies in a patient with lupus nephritis. Pediatric nephrology (Berlin, Germany). 2004; 19: 451–453. DOI: https://doi.org/10.1007/s00467-003-1388-6 

  53. Lai PH, Tien RD, Chang MH, et al. Chorea-ballismus with nonketotic hyperglycemia in primary diabetes mellitus. AJNR American journal of neuroradiology. 1996; 17: 1057–1064. 

  54. Forchetti CM, Katsamakis G, Garron DC. Autoimmune thyroiditis and a rapidly progressive dementia: global hypoperfusion on SPECT scanning suggests a possible mechanism. Neurology. 1997; 49: 623–626. DOI: https://doi.org/10.1212/WNL.49.2.623 

  55. Pari E, Rinaldi F, Premi E, et al. A follow-up (1)(8)F-FDG brain PET study in a case of Hashimoto’s encephalopathy causing drug-resistant status epilepticus treated with plasmapheresis. J Neurol. 2014; 261: 663–667. DOI: https://doi.org/10.1007/s00415-013-7228-0 

  56. George A, Abdurahman P, James J. Spastic paraparesis, abnormal muscle biopsy and positive antithyroid antibodies. The Journal of the Association of Physicians of India. 2007; 55: 585–586. 

  57. Blanchin S, Coffin C, Viader F, et al. Anti-thyroperoxidase antibodies from patients with Hashimoto’s encephalopathy bind to cerebellar astrocytes. Journal of neuroimmunology. 2007; 192: 13–20. DOI: https://doi.org/10.1016/j.jneuroim.2007.08.012 

  58. Takahashi S, Mitamura R, Itoh Y, Suzuki N, Okuno A. Hashimoto encephalopathy: etiologic considerations. Pediatric neurology. 1994; 11: 328–331. DOI: https://doi.org/10.1016/0887-8994(94)90011-6 

  59. Oide T, Tokuda T, Yazaki M, et al. Anti-neuronal autoantibody in Hashimoto’s encephalopathy: neuropathological, immunohistochemical, and biochemical analysis of two patients. J Neurol Sci. 2004; 217: 7–12. DOI: https://doi.org/10.1016/j.jns.2003.08.005 

  60. Saravanan P, Dayan CM. Thyroid autoantibodies. Endocrinology and metabolism clinics of North America. 2001; 30: 315–337, viii. DOI: https://doi.org/10.1016/S0889-8529(05)70189-4 

  61. Kirshner HS. Hashimoto’s encephalopathy: a brief review. Current neurology and neuroscience reports. 2014; 14: 476. DOI: https://doi.org/10.1007/s11910-014-0476-2 

  62. Olmez I, Moses H, Sriram S, Kirshner H, Lagrange AH, Pawate S. Diagnostic and therapeutic aspects of Hashimoto’s encephalopathy. J Neurol Sci. 2013; 331: 67–71. DOI: https://doi.org/10.1016/j.jns.2013.05.009 

  63. Cook MK, Malkin M, Karafin MS. The use of plasma exchange in Hashimoto’s encephalopathy: A case report and review of the literature. Journal of clinical apheresis. 2015; 30: 188–192. DOI: https://doi.org/10.1002/jca.21353