Benefits of multi-day supplementation with probiotic kefir in Rasmussen encephalitis: the first case report

ABSTRACT

Introduction

Rasmussen encephalitis (RE) is a rare inflammatory disease, characterized by unilateral hemispheric atrophy, focal intractable seizures, progressive hemiparesis, and neurological deficits.

Case report

The patient is a young man under pharmacotherapy for epilepsy, exhibiting classical abnormal movements, which are consider typical hallmarks of RE. During clinical care sessions, he presented many episodes of tonic-clonic seizures involving sudden loss of consciousness followed by a post-ictal phase with weakness and interaction difficulty. During the kefir supplementation, the patient presented only short-term absence seizures, quickly returning to activities. Additionally, he presented cognitive and language improvement, being more responsive to commands. The daily diary control of patient’s mother and caregiver at school reported an impressive reduction in number and severity of seizures, becoming less aggressive and more involved in school activities. The serum biochemical markers showed that kefir administration caused a significant decrease of pro-inflammatory and a simultaneous increase of anti-inflammatory cytokine levels. In parallel, after treatment, this probiotic reduced reactive oxygen species levels, increased NO bioavailability, revealing antiapoptotic and antigenotoxic effects. Regarding the microbiological analysis, kefir increased Lactobacillus and Bifidobacterium species.

Conclusion

To our knowledge, this is the first case reporting remarkable beneficial effects of the probiotic kefir in RE. This case report strongly suggests kefir supplementation as a potential and safe-effective adjuvant therapeutic strategy in the control and treatment of RE.

GRAPHICAL ABSTRACT

KEYWORDS:

• Rasmussen encephalitis
• seizure
• microbiota
• oxidative stress
• case report

Disclosure statement

No potential conflict of interest was reported by the authors.

Consent to participate

The mother’s patient (legal guardian) voluntarily agreed with his participation after understanding the purpose and nature of the study and provided informed written consent to publish clinical data and videos.

Availability of data and material

The data that support the findings of this study are available from the corresponding author (BPC), upon request.

Author contributions

VRL, RA, and LZC carried out the experiments, provided acquisition, analysis, and interpretation of the data. RBD, SSM, and ECV participated in the critical revision of the manuscript for important intellectual content. TMCP and BPC made substantial contributions to the concept, design, and supervision of the study, interpretation of data and drafted the manuscript. All authors read and approved the final version of the manuscript. In addition, the authors agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Additional information

Funding

This work was supported by the State of Espírito Santo Research Foundation (FAPES) and by the National Research Council of Science and Technology (CNPq) FAPES (Fundação Estadual de Amparo à Pesquisa do Estado do Espírito Santo) supported BPC (FAPES-Universal, N° 21/2018, Termo Outorga 120/2019) and ECV (CNPq/FAPES N° 24/2018; Termo Outorga 569/2018). Research grants: BPC supported by FAPES under [grant 552/2018]; SSM, TMCP and ECV supported by CNPq (Conselho Nacional de Desenvolvimento Científico e Tecnológico) under [grants 312056/2018-5, 309277/2019-1 and 305740/2019-9].

Notes on contributors

Valéria R. Lemos

Valéria R. Lemos: Physiotherapist, PhD in Pharmaceutical Sciences at Vila Velha University (UVV), Brazil.

Rafaela Aires

Rafaela Aires: Physiotherapist, PhD in Physiological Sciences at Federal University of Espírito Santo (UFES), Brazil.

Larissa Z. Côco

Larissa Z. Côco: Pharmacist, PhD student in Pharmaceutical Sciences at Vila Velha University (UVV), Brazil.

Renan B. Domingues

Renan B. Domingues: Neurologist, PhD in Medicine at University of São Paulo (USP), Brazil. Post-Doctorate in Neurology at the Cerebrovascular Diseases Service at the University of Lille, France.

Silvana S. Meyrelles

Silvana S. Meyrelles: Nurse, PhD in Physiological Sciences at Federal University of Espírito (UFES), Brazil. Post-Doctorate in Cardiovascular Physiology at the University of Iowa, USA.

Elisardo C. Vasquez

Elisardo C. Vasquez: Biomedical, PhD in Physiology at University of São Paulo (USP), Brazil. Currently Full Professor of the Graduate Program of Pharmaceutical Sciences at Vila Velha University (UVV), Brazil.

Thiago M. C. Pereira

Thiago M. C. Pereira: Pharmacist, PhD in Physiological Sciences at Federal University of Espírito Santo (UFES), Brazil. Post-Doctorate in Pharmacology at University of Santiago de Compostela, Spain.

Bianca P. Campagnaro

Bianca P. Campagnaro: Biologist, PhD in Physiological Sciences at Federal University of Espírito Santo (UFES), Brazil. Researcher at the Graduate Program of Pharmaceutical Sciences at Vila Velha University (UVV), Brazil.