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Background and Purpose: The recent off-label use of botulinum neurotoxin (BoNT) for intragastric obesity treatment has led to 67 cases of systemic botulism in Türkiye, Germany, Austria and Switzerland. This case report highlights the potential risks and adverse effects associated with this treatment., Case Report: A 36-year-old female presented to the emergency room with shortness of breath, fatigue, difficulty in eating and holding her head, constipation and double vision after receiving intragastric BoNT injection for obesity treatment. She had bilateral orbicularis oculi weakness, facial diplegia, weak tongue, masseter, neck and extremity muscles. Electromyography showed a presynaptic type neuromuscular junction disorder. The patient was admitted to the intensive care unit and administered botulinum heptavalent equine-derived antitoxin, but the medication had to be stopped due to a reaction. The patient was started on pyridostigmine for symptomatic treatment and was transferred to an inpatient clinic after minimal improvement. She was discharged after 7 days of follow-up., Conclusion: Clinicians should be cautious of the potential risks of intragastric BoNT injection for obesity treatment and consider systemic botulism as a potential adverse effect. Antitoxin treatment should be considered in clinically progressing patients despite negative botulinum toxin testing., (© 2023 European Academy of Neurology.)