Case Report : Botulinum Toxin Treatment in Oromandibular Dystonia

보툴리눔 톡신을 이용한 구강하악 근긴장이상증의 치료 증례

  • Ryu, Ji-Won (Department of Oral medicine, College of Dentistry, Chosun University) ;
  • Hong, Seong-Ju (Department of Oral medicine, College of Dentistry, Chosun University) ;
  • Bae, Kook-Jin (Department of Oral medicine, College of Dentistry, Chosun University) ;
  • Yoon, Chang-Lyuk (Department of Oral medicine, College of Dentistry, Chosun University) ;
  • Ahn, Jong-Mo (Department of Oral medicine, College of Dentistry, Chosun University)
  • 유지원 (조선대학교 치과대학 구강내과학교실) ;
  • 홍성주 (조선대학교 치과대학 구강내과학교실) ;
  • 배국진 (조선대학교 치과대학 구강내과학교실) ;
  • 윤창륙 (조선대학교 치과대학 구강내과학교실) ;
  • 안종모 (조선대학교 치과대학 구강내과학교실)
  • Published : 2009.12.30

Abstract

Oromandibular dystonia is a focal neurological movement disorder characterized by involuntary sustained and often painful muscle contraction, usually producing repetitive movements or abnormal positions of the mouth, jaw and.or tongue. Patients suffering from oromandibular dystonia often experience difficulties in chewing, swallowing and speaking, resulting from the impairment of mandibular movements. At present there is no etiologic treatment for oromandibular dystonia, because the pathophysiology of primary and focal dystonia is still incompletely understood. Many treatments such as medication, behavioral therapy, surgery are suggested to decrease the involuntary movements. But these success rates are relatively low and they have a lot of complications. many studies suggested that chemodenervation with botulinum toxin is the most effective treatment for oromandibular dystonia. We reported the 2 cases which were treated oromandibular dystonia with botulinum toxin and reviewed the orofacial movement disorders(especially oromandibular dystonia) and botulinum toxin treatment for oromanfibular dystonia.

구강하악 근긴장이상증(oromandibular dystonia)은 국소적인 근긴장이상증의 한 형태로, 저작근, 안면근 또는 혀 근육의 지속적이고 반복적인 근경련이 발생하여 불수의적인 개구 및 폐구, 악골의 측방 및 후퇴운동이 나타나는 것으로 정의내릴 수 있다. 구강하악 근긴장이상증에 이환된 환자의 경우, 저작, 연하 및 발음을 하는데 불편감을 가지게 되고, 그 결과 하악 운동에 지장을 초래하게 된다. 현재까지는 근긴장이상증에 대한 병태생리가 뚜렷히 입증된 바가 없어, 원인에 관련한 치료는 이루어지지 않고 있는 상태이다. 약물요법, 행동요법, 외과적 처치 등 다양한 방법이 구강하악 근긴장이상증의 치료법으로 제시되고 있으나, 성공률이 그리 높은 편은 아니며, 많은 부작용을 초래할 수 있다. 최근 들어 근긴장이상증에 이환된 근육에 보툴리눔 톡신을 이용한 치료법이 주목을 받고 있는 추세이다. 본 증례를 통해 구강하악 근긴장이상증을 중심으로 한 구강안면 운동장애에 대하여 전반적으로 고찰을 시행하고 해당 질환에서의 보툴리눔 톡신 치료법에 대하여 알아보고자 한다.

Keywords

References

  1. Tolosa E, Marti MJ. Blepharospasm-oromandibular dystonia syndrome (Meige's syndrome): clinical aspects. Adv Neurol 1988;49:73–84
  2. Jankovic J. Etiology and differential diagnosis of blepharospasm and oromandibular dystonia. Adv Neurol 1988;49:103–116
  3. Cardoso F, Jankovic J. Peripherally induced tremor and parkinsonism. Arch Neurol 1995;52(3):263–270 https://doi.org/10.1001/archneur.1995.00540270055019
  4. Michelotti A, Silva R, Paduano S, Cimino R, Farella M. Oromandibular dystonia and hormonal factors:twelve years follow-up of a case report. J Oral Rehabil. 2009;36(12):916-921 https://doi.org/10.1111/j.1365-2842.2009.02007.x
  5. Berardelli A, Rothwell JC, Hallett M, Thompson PD, Manfredi M, Marsden CD. The pathophysiology of primary dystonia. Brain. 1998;121:1195-1212 https://doi.org/10.1093/brain/121.7.1195
  6. Balasubramaniam R, Ram S. Orofacial movement disorders. Oral Maxillofac Surg Clin North Am 2008;20(2):273-285 https://doi.org/10.1016/j.coms.2007.12.010
  7. Blanchet PJ, Abdillahi O, Beauvais C, Rompre PH, Lavigne GJ. Prevalence of spontaneous oral dyskinesia in the elderly: a reappraisal. Mov Disord 2004; 19(8):892–896 https://doi.org/10.1002/mds.20130
  8. Klawans HL, Tanner CM, Goetz CG. Epidemiology and pathophysiology of tardive dyskinesias. Adv Neurol 1988;49:185–197
  9. Bassett A, Remick RA, Blasberg B. Tardive dyskinesia: an unrecognized cause of orofacial pain. Oral Surg Oral Med Oral Pathol 1986;61(6):570–572 https://doi.org/10.1016/0030-4220(86)90095-2
  10. 유지원, 윤창륙, 조영곤, 안종모. 구강안면 운동장애의 임상적 증상 발현. 대한구강내과학회지 2008;33(4):375-382
  11. Logroscino G, Livrea P, Anaclerio D, et al. Agreement among neurologists on the clinical diagnosis of dystonia at different body sites. J Neurol Neurosurg Psychiatry 2003;74(3):348–350 https://doi.org/10.1136/jnnp.74.3.348
  12. Derogatis LR, Lipman RS, Rickels K, Uhlenhuth EH, Covi L. The Hopkins Symptom Checklist (HSCL): a self-report symptom inventory. Behav Sci 1974;19(1):1–15 https://doi.org/10.1002/bs.3830190102
  13. Epidemiologic Study of Dystonia in Europe (ESDE) Collaborative Group. Sex related influences on the frequency and age of onset of primary dystonia. Neurology. 1999;53:1871–1873 https://doi.org/10.1212/WNL.53.8.1871
  14. Watts MW, Tan EK, Jankovic J. Bruxism and cranial-cervical dystonia: is there a relationship? Cranio 1999;17(3):196–201 https://doi.org/10.1080/08869634.1999.11746095
  15. Thompson PD, Obeso JA, Delgado G, Gallego J, Marsden CD. Focal dystonia of the jaw and the differential diagnosis of unilateral jaw and masticatory spasm. J Neurol Neurosurg Psychiatry 1986;49(6):651–656 https://doi.org/10.1136/jnnp.49.6.651
  16. Wang A, Jankovic J. Hemifacial spasm: clinical findings and treatment. Muscle Nerve 1998;21(12):1740–1747 https://doi.org/10.1002/(SICI)1097-4598(199812)21:12<1740::AID-MUS17>3.0.CO;2-V
  17. Jankovic J, Ford J. Blepharospasm and orofacialcervical dystonia: clinical and pharmacological findings in 100 patients. Ann Neurol 1983;13(4):402–411 https://doi.org/10.1002/ana.410130406
  18. Osborne TE, Grace EG, Schwartz MK. Severe degenerative changes of the temporomandibular joint secondary to the effects of tardive dyskinesia: a literature review and case report. Cranio 1989;7(1):58–62 https://doi.org/10.1080/08869634.1989.11678269
  19. Clark GT, Stiles A, Lockerman LZ, et al. A critical review of the use of botulinum toxin in orofacial pain disorders. Dent Clin North Am 2007;51(1):245–261 https://doi.org/10.1016/j.cden.2006.09.003
  20. Tan EK, Jankovic J. Botulinum toxin A in patients with oromandibular dystonia. Long term follow up. Neurology.1999;53:2102-2107 https://doi.org/10.1212/WNL.53.9.2102
  21. Bridayasiri R, Cardoso F, Truong DD. Botulinum toxin in blepharospasm and oromandibular dystonia:comparing different botulinum toxin preparations. Eur J Neurol. 2006;13:21–29
  22. Singer C, Papapetropoulos S. A comparison of jawclosing and jaw-opening idiopathic oromandibular dystonia. Parkinsonism Relat Disord. 2006;12:115–118 https://doi.org/10.1016/j.parkreldis.2005.07.007