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A severe case of non-infective myositis six weeks post intramuscular injections of Onabotulinum toxin A (Botox) in a young man with tetraplegia: case report

Abstract

Introduction

Myositis of unknown aetiology might be a very rare complication of intramuscular injections of onabotulinum toxin A (Botox) for spasticity treatment.

Case presentation

We describe a case of significant myositis of unknown aetiology in a 17-year-old man, who was admitted for rehabilitation 4 months after his initial spinal cord injury (SCI) as a result of a mountain bike accident. He has an incomplete tetraplegia, C4 AIS B international Standards for Neurological Classification for Spinal Cord Injury (ISNCSCI) [1] due to C5 vertebra 3 column fracture [2]. He had severe spasticity of his lower limb muscles treated with Botox, following which, he required two acute hospital transfers for diagnosis and management of myositis.

Discussion

This is a severe unusual presentation of myositis caused by intramuscular botulinum toxin for treatment of spasticity, in the frequent setting of spasticity where intramuscular botulinum toxin injections are routinely used.

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Fig. 1: Creatine kinase and C reactive protein trends.
Fig. 2: Lower limbs Magnetic Resonance Imaging T2 sequence.
Fig. 3: Frozen sections with haematinin-eosin staining showing infarcted necrotic muscle, without inflammation.

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Acknowledgements

Ms Nicole Whitehead, Physiotherapist at Spinal Injuries Unit, Royal Rehab, Ryde, NSW 2112, Australia for assessing the patient before and after injections and offering ongoing advice regarding spasticity management. Dr Hwei Choo Soh, Senior Staff Specialist, Department of Anatomical Pathology, Royal North Shore Hospital, St Leonards NSW 2065, Australia, for the provision and interpretation of the histopathological slides. Royal North Shore Hospital Consulting specialists: Dr David Hunter, Consulting Rheumatologist, Dr Dimitri Papadimitriou, Consulting Orthopaedic Surgeon and Dr Melanie Figtree, Consulting Infectious Diseases Specialist. Dr Andreea Heriseanu, DClinPsych/PhD, who assisted in the proofreading of the manuscript. No financial assistance was received in support of the study.

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Authors and Affiliations

Authors

Contributions

REH was responsible for extracting and analysing the clinical information, writing the report, interpreting results, and updating reference lists. PC was responsible for providing the histopathology slides and MRI findings, liaising with all treating specialists, conducting the literature search and contributing to the writing of the report and the reference list.

Corresponding authors

Correspondence to Roxana Edith Heriseanu or Priyadarshini Chari.

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The authors declare no competing interests.

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Appendices

Appendix A

Tardieu Scale [15]

This scale quantifies muscle spasticity by assessing the response of the muscle to stretch applied at specified velocities. Grading is always performed at the same time of day, in a constant position of the body for a given limb. For each muscle group, reaction to stretch is rated at a specified stretch velocity with 2 parameters x and v. Measurements take place at 3 velocities to stretch (V1, V2, and V3). Responses are recorded at each velocity as X/Y, with X indicating the quality of muscle reaction from 0 to 5 rating, and Y indicating the degree of angle at which the muscle reaction occurs.

By moving the limb at different velocities, the response to stretch can be more easily gauged since the stretch reflex responds differently to velocity.

Velocities:

V1: As slow as possible, slower than the natural drop of the limb segment under gravity.

V2: Speed of limb segment falling under gravity.

V3: As fast as possible, faster than the rate of the natural drop of the limb segment under gravity.

Scoring:

0 No resistance throughout the course of the passive movement.

1 Slight resistance throughout the course of passive movement, no clear catch at a precise angle.

2 Clear catch at a precise angle, interrupting the passive movement, followed by release.

3 Fatigable clonus with less than 10 s when maintaining the pressure and appearing at the precise angle.

4 Unfatigable clonus with more than 10 s when maintaining the pressure and appearing at a precise angle.

5 Joint is immovable.

Example:

When testing spasticity of the hamstring at the speed V1, place the patient in the supine position. Flex the hip to 90 degrees, with the opposite hip extended (as for popliteal angle test). Beginning with the knee flexed, extend the knee as slowly as possible. If a clear catch interrupts the passive movement at −70 degrees of extension, followed by a release facilitating further extension to −50 degrees of extension, then the Tardieu V1 score would be 2/−70. The rating would be repeated for V2 and V3 velocities. Evaluating movement of a part at different velocities may help distinguish passive stiffness from spasticity.

Appendix B

Literature search:

Search Databases: Allergan Product Literature Database Search Date: 14-MAY-2020

Search Parameters: (BOTOX OR BOTOX VISTA OR BOTOX Cosmetic OR VISTABEL OR VISTABEX) AND (myositis OR inflammatory myopathy).

Search Limits: Meta-Analyses, Systematic Reviews, Clinical Trials, Observational Studies, Case Reports, English, Humans Allergan’s Product Literature Database contains publications compiled from publicly accessible literature databases (i.e., MEDLINE®, EMBASE.com®, etc.).

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Heriseanu, R.E., Chari, P. A severe case of non-infective myositis six weeks post intramuscular injections of Onabotulinum toxin A (Botox) in a young man with tetraplegia: case report. Spinal Cord Ser Cases 7, 76 (2021). https://doi.org/10.1038/s41394-021-00442-1

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