Journal of Oral Medicine and Pain

Korean Academy of Orofacial Pain and Oral Medicine (대한안면통증구강내과학회)
권호 표지
DOI QR코드

DOI QR Code

A Primary Sjögren's Syndrome Patient Presented with Severe General Toothache

  • Lee, Yeon-Hee (Department of Oral Medicine and Oral Diagnosis, School of Dentistry and Dental Research Institute, Seoul National University) ;
  • Kho, Hong-Seop (Department of Oral Medicine and Oral Diagnosis, School of Dentistry and Dental Research Institute, Seoul National University)
  • Received : 2015.08.10
  • Accepted : 2015.09.02
  • Published : 2015.09.30
Download PDF
⟨ Previous Next ⟩

Abstract

$Sj{\ddot{o}}gren's$ syndrome (SS) is an autoimmune disease characterized by an autoimmune exocrinopathy involving mainly salivary and lacrimal glands. Apart from manifestations due to involvement of exocrine glands, patients with SS can present with muscular and neurological manifestations. Here, we report a rare case of a 59-year-old woman with primary SS, who presented with severe general toothache and masticatory muscle myalgia successfully treated with clonazepam. Although it was not certain that these symptoms could be originated from focal muscle dystonia or neurological changes that are associated with primary SS, our case suggested that comprehensive evaluation including neuromuscular examinations in the oral and maxillofacial area is needed in patients with SS.

Keywords

References

  1. Baldini C, Talarico R, Tzioufas AG, Bombardieri S. Classification criteria for Sjogren's syndrome: a critical review. J Autoimmun 2012;39:9-14. https://doi.org/10.1016/j.jaut.2011.12.006
  2. List T, Stenström B, Lundström I, Dworkin SF. TMD in patients with primary Sjögren syndrome: a comparison with temporomandibular clinic cases and controls. J Orofac Pain 1999;13:21-28.
  3. Vrethem M, Lindvall B, Holmgren H, Henriksson KG, Lindstrom F, Ernerudh J. Neuropathy and myopathy in primary Sjogren's syndrome: neurophysiological, immunological and muscle biopsy results. Acta Neurol Scand 1990;82:126-131.
  4. Asmussen K, Andersen V, Bendixen G, Schiodt M, Oxholm P. A new model for classification of disease manifestations in primary Sjogren's syndrome: evaluation in a retrospective long-term study. J Intern Med 1996;239:475-482. https://doi.org/10.1046/j.1365-2796.1996.418817000.x
  5. Kanellopoulos P, Baltoyiannis C, Tzioufas AG. Primary Sjogren's syndrome associated with inclusion body myositis. Rheumatology (Oxford) 2002;41:440-444. https://doi.org/10.1093/rheumatology/41.4.440
  6. Teixeira F, Moreira I, Silva AM, Vasconcelos C, Farinha F, Santos E. Neurological involvement in primary Sjogren syndrome. Acta Reumatol Port 2013;38:29-36.
  7. Chai J, Logigian EL. Neurological manifestations of primary Sjogren's syndrome. Curr Opin Neurol 2010;23:509-513. https://doi.org/10.1097/WCO.0b013e32833de6ab
  8. Papageorgiou SG, Kontaxis T, Bonakis A, Kalfakis N. Orofacial dystonia related to Sjogren's syndrome. Clin Rheumatol 2007;26:1779-1781. https://doi.org/10.1007/s10067-006-0519-4
  9. de Leeuw R. Orofacial pain. Guidelines for assessment, diagnosis, and management. 5th ed. Chicago: Quintessence; 2013. pp. 1-23.
  10. Okeson JP. Bell's oral and facial pain. 7th ed. Chicago: Quintessence;2014. pp. 287-326.
  11. Fricton JR, Kroening R, Haley D, Siegert R. Myofascial pain syndrome of the head and neck: a review of clinical characteristics of 164 patients. Oral Surg Oral Med Oral Pathol 1985;60:615-623. https://doi.org/10.1016/0030-4220(85)90364-0
  12. Simons DG, Travell JG, Simons LS. Travell & Simons' myofascial pain and dysfunction: the trigger point manual. Vol. 1. 2nd ed. Philadelphia: Lippincott Williams & Wilkins; 1999. pp. 329-364.
  13. Herman CR, Schiffman EL, Look JO, Rindal DB. The effectiveness of adding pharmacologic treatment with clonazepam or cyclobenzaprine to patient education and self-care for the treatment of jaw pain upon awakening: a randomized clinical trial. J Orofac Pain 2002;16:64-70.
  14. Lindvall B, Bengtsson A, Ernerudh J, Eriksson P. Subclinical myositis is common in primary Sjogren's syndrome and is not related to muscle pain. J Rheumatol 2002;29:717-725.
  15. Fasano A, Bove F, Lang AE. The treatment of dystonic tremor: a systematic review. J Neurol Neurosurg Psychiatry 2014;85:759-769. https://doi.org/10.1136/jnnp-2013-305532
  16. Dellemijn PL, Fields HL. Do benzodiazepines have a role in chronic pain management? Pain 1994;57:137-152. https://doi.org/10.1016/0304-3959(94)90217-8
  17. Pavlakis PP, Alexopoulos H, Kosmidis ML, et al. Peripheral neuropathies in Sjögren's syndrome: a critical update on clinical features and pathogenetic mechanisms. J Autoimmun 2012;39:27-33. https://doi.org/10.1016/j.jaut.2012.01.003
  18. Briani C, Cacciavillani M, Gasparotti R. MRI evidence of trigeminal sensory neuropathy in Sjogren's syndrome. Clin Neurophysiol 2013;124:1703-1705. https://doi.org/10.1016/j.clinph.2013.01.012
  19. Flanagan EP, Kaufmann TJ, Keegan BM. Sjogren's syndrome with trigeminal neuropathy: motor involvement. Pract Neurol 2013; 13:340-342. https://doi.org/10.1136/practneurol-2013-000560