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A Brain-Heart Interaction: Bickerstaff's brainstem encephalitis with Takotsubo Cardiomyopathy.
QJM. 2020 Jul 08 [Online ahead of print]QJM

Abstract

INTRODUCTION

Bickerstaff's brainstem encephalitis (BBE) is a neurological condition characterised by ataxia, opthalmoplegia and altered sensorium. Neurological diseases, such as Guillain-Barre syndrome, have previously been reported in association with Takotsubo cardiomyopathy. We describe a case of Takotsubo cardiomyopathy associated with BBE.

METHODS/RESULTS

A 62-year-old Chinese female with no significant medical history was admitted after a fall due to transient bilateral lower limb numbness. A day after her initial presentation, she developed altered sensorium and further deteriorated within hours - with bulbar weakness, increasing oxygen requirements requiring intubation and cardiogenic shock needing noradrenaline support. She also had rapid atrial fibrillation treated with amiodarone. Her cardiac enzymes were elevated (high sensitivity troponin I 2137pg/ml; upper limit 15.6pg/ml) and echocardiography demonstrated apical ballooning and dyskinesis with hypercontractility of the base in the left ventricle consistent with Takotsubo cardiomyopathy. Examination revealed bulbar weakness with poor gag and cough reflexes, ataxia and complex opthalmoplegia. Her reflexes were brisk and pupils mydriatic with sluggish pupillary response. Magnetic resonance imaging revealed a gadolinium-enhancing lesion in the brainstem on T1 sequence and anti-GQ1b antibody was positive. She was diagnosed with BBE and treated with intravenous immunoglobulin with good functional recovery. Repeat echocardiography demonstrated interval improvement in apical ballooning.

CONCLUSION

This is the first described case of Takotsubo cardiomyopathy associated with BBE. The pathogenesis of Takotsubo cardiomyopathy in relation to BBE is postulated to be related to dysautonomia with sympathetic overactivity and excessive catecholamine release. In cases of hemodynamic instability in BBE, Takotsubo cardiomyopathy should be considered.

Authors+Show Affiliations

Department of Medicine, National University Health System, Singapore.Division of Neurology, Department of Medicine, National University Health System, Singapore.Department of Medicine, Yong Loo Lin School of Medicine, National University of Singapore.Division of Neurology, Department of Medicine, National University Health System, Singapore. Department of Medicine, Yong Loo Lin School of Medicine, National University of Singapore.Division of Neurology, Department of Medicine, National University Health System, Singapore. Department of Medicine, Yong Loo Lin School of Medicine, National University of Singapore.Division of Neurology, Department of Medicine, National University Health System, Singapore.Division of Neurology, Ng Teng Fong General Hospital, Singapore.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

32642763

Citation

See, Faith Huiwen, et al. "A Brain-Heart Interaction: Bickerstaff's Brainstem Encephalitis With Takotsubo Cardiomyopathy." QJM : Monthly Journal of the Association of Physicians, 2020.
See FH, Goh Y, Sia CH, et al. A Brain-Heart Interaction: Bickerstaff's brainstem encephalitis with Takotsubo Cardiomyopathy. QJM. 2020.
See, F. H., Goh, Y., Sia, C. H., Tan, B. Y., Yeo, L. L., Vijayan, J., & Aftab, A. (2020). A Brain-Heart Interaction: Bickerstaff's brainstem encephalitis with Takotsubo Cardiomyopathy. QJM : Monthly Journal of the Association of Physicians. https://doi.org/10.1093/qjmed/hcaa218
See FH, et al. A Brain-Heart Interaction: Bickerstaff's Brainstem Encephalitis With Takotsubo Cardiomyopathy. QJM. 2020 Jul 8; PubMed PMID: 32642763.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - A Brain-Heart Interaction: Bickerstaff's brainstem encephalitis with Takotsubo Cardiomyopathy. AU - See,Faith Huiwen, AU - Goh,Yihui, AU - Sia,Ching-Hui, AU - Tan,Benjamin Yong-Qiang, AU - Yeo,Leonard Leong-Litt, AU - Vijayan,Joy, AU - Aftab,Ahmad, Y1 - 2020/07/08/ PY - 2020/06/21/received PY - 2020/06/23/accepted PY - 2020/7/10/entrez KW - Anti-Gq1b syndrome KW - Bickerstaff’s brainstem encephalitis KW - Catecholamine KW - Takotsubo cardiomyopathy JF - QJM : monthly journal of the Association of Physicians JO - QJM N2 - INTRODUCTION: Bickerstaff's brainstem encephalitis (BBE) is a neurological condition characterised by ataxia, opthalmoplegia and altered sensorium. Neurological diseases, such as Guillain-Barre syndrome, have previously been reported in association with Takotsubo cardiomyopathy. We describe a case of Takotsubo cardiomyopathy associated with BBE. METHODS/RESULTS: A 62-year-old Chinese female with no significant medical history was admitted after a fall due to transient bilateral lower limb numbness. A day after her initial presentation, she developed altered sensorium and further deteriorated within hours - with bulbar weakness, increasing oxygen requirements requiring intubation and cardiogenic shock needing noradrenaline support. She also had rapid atrial fibrillation treated with amiodarone. Her cardiac enzymes were elevated (high sensitivity troponin I 2137pg/ml; upper limit 15.6pg/ml) and echocardiography demonstrated apical ballooning and dyskinesis with hypercontractility of the base in the left ventricle consistent with Takotsubo cardiomyopathy. Examination revealed bulbar weakness with poor gag and cough reflexes, ataxia and complex opthalmoplegia. Her reflexes were brisk and pupils mydriatic with sluggish pupillary response. Magnetic resonance imaging revealed a gadolinium-enhancing lesion in the brainstem on T1 sequence and anti-GQ1b antibody was positive. She was diagnosed with BBE and treated with intravenous immunoglobulin with good functional recovery. Repeat echocardiography demonstrated interval improvement in apical ballooning. CONCLUSION: This is the first described case of Takotsubo cardiomyopathy associated with BBE. The pathogenesis of Takotsubo cardiomyopathy in relation to BBE is postulated to be related to dysautonomia with sympathetic overactivity and excessive catecholamine release. In cases of hemodynamic instability in BBE, Takotsubo cardiomyopathy should be considered. SN - 1460-2393 UR - https://www.unboundmedicine.com/medline/citation/32642763/A_Brain-Heart_Interaction:_Bickerstaff's_brainstem_encephalitis_with_Takotsubo_Cardiomyopathy L2 - https://academic.oup.com/qjmed/article-lookup/doi/10.1093/qjmed/hcaa218 DB - PRIME DP - Unbound Medicine ER -
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