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연제번호 : P 2-127 북마크
제목 Clinically diagnosed iatrogenic botulinum toxin intoxification : a case report
소속 Seoul St. Mary`s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea, Department of Rehabilitation Medicine1, Yeouido St. Mary`s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea, Department of Rehabilitation Medicine2, Eunpyeong St. Mary`s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea, Department of Rehabilitation Medicine3
저자 Sehee Kim 1*, Daye Kim2, So yeon Jun1, Jihye Park3, Young Jin Ko1†
Introduction
Botulism is caused by the exotoxin of Clostridium botulinum, which acts by blocking the presynaptic release of acetylcholine. Although safety has been verified, it has been reported that iatrogenic botulism(IB) can be caused by systemic spread of botulinum toxin which is used therapeutically or cosmetically. Although it is nearly impossible to conclusively prove IB because of the diversity of underlying diseases and dosage of patients, Nerve conduction study (NCS) and electromyographic (EMG) examination can be useful support to diagnose IB.
Case report
A 52-year-old male patient came to the emergency room. Five days before admission, the patient underwent botulinum toxin injection to mitigate right posterior neck pain and headache. Ptosis developed in the right eye a day after. On the day before admission, the pronunciation of patient became sloppy and slowing food became difficult. On the morning of hospitalization, suspicious symptoms of laryngospasm occurred and it became very difficult to breathe. The vocal cord gap of the patient was narrowed compared to normal adult male. Intravenous immunoglobulin therapy and intravenous pyridostigmine therapy started, but his symptoms gradually worsened and later that day, the patient underwent tracheostomy. Five days later he fell into respiratory failure and ventilator care was started. Strength of proximal muscle of upper extremity and neck flexor muscle was generally trace grade according to the manual muscle test(MMT).
NCS and EMG examination were done at hospital day 83 to support the confirmation of his diagnosis. Motor NCS presented decreased amplitudes of compound muscle action potential(CMAP) in right common peroneal nerve, spinal accessory nerve and axillary nerve(Table 1). Needle EMG showed abnormal spontaneous activities at rest in several muscles. Increased insertional activities were observed at rest in first dorsal interosseous muscle (Table2). Repetitive nerve stimulation test presented decrement pattern more than 10% of CMAP in right spinal accessory nerve recorded at trapezius muscle and right axillary nerve recorded at middle deltoid muscle on resting state (Figure1). Based on his history, he was diagnosed with IB.
Comprehensive rehabilitation therapy including gait training, pulmonary rehabilitation and dysphagia therapy was continued. After a month, the patient was tolerable enough to exchange tracheostomy to retainer. At the time of discharge, the patient could walk independently.
Conclusion
Early diagnosis of botulism is crucial because it is life-threatening with airway involvement. NCS and EMG examination supported the diagnosis of IB. Treatment of IB requires prevention of sequelae and management of symptoms such as dyspnea in the acute phase. In this case, pyridostigmine was applied to relieve severe sequelae of IB. In our case, the patient gradually recovered over several weeks after comprehensive rehabilitation therapy and application of pyridostigmine.
Motor nerve conduction study showed decreased amplitudes of compound muscle action potential(CMAP)s in right common peroneal nerve recorded at extensor digiotrum brevis muscle, spinal accessory nerve recorded at trapezius muscle and axillary nerve recorded at middle deltoid muscle.
Needle EMG study presents Abnormal spontaneous activities at rest in right cervical paraspinalis, middle deltoid, biceps, flexor carpi radialis and extensor digitorum communis muscles. Increased insertional activities were shown at rest in first dorsal interosseous muscle.
Repetitive nerve stimulation test. decrement pattern more than 10% of CMAP in right spinal accessory nerve recorded at trapezius muscle and right axillary nerve recorded at middle deltoid muscle on resting state were observed.