Ernest Syndrome: A Systematic Review of the Literature

Aims: To conduct a systematic review of the available evidence regarding the clinical characteristics and treatment of Ernest syndrome. Methods: A systematic search was carried out in the EBSCOhost, Embase, MEDLINE, ScienceDirect, Scopus, Web of Science, and Trip databases. The free terms “stylomandibular ligament” and “Ernest syndrome” were used, and the Boolean operator “OR” was used for connection of the terms. The research protocol was registered in PROSPERO (CRD42018112914). The results of this study are presented according to the PRISMA statement, and risk of bias was assessed according to the Quality Assessment Tool for Quantitative Studies of the Effective Public Health Practice Project. Results: A total of 57 articles were found, 5 of which met the selection criteria and were included in this review. The selected articles represent a population of 81 patients with primarily unilateral pain in the periauricular and mandibular areas. Diagnosis was established according to clinical history, recognition of pain on palpation of the mandibular angle, and remission following infiltration with local anesthesia. Concerning the treatments applied, infiltration with corticosteroids was highly successful, as was partial resection of the stylomandibular ligament and part of the styloid process. Furthermore, in refractory cases, the application of radiofrequency thermoneurolysis was described. Conclusion: The analyzed studies allow a better understanding of Ernest syndrome and the proposal of a tendinosis model for this condition. However, the evidence is scarce, and it is therefore necessary to carry out additional studies with better methodologic designs.

Ernest syndrome; inflammation; pain; stylomandibular ligament; temporomandibular disorders

1. Okeson JP (ed). Management of Temporomandibular Disorders and Occlusion, ed 7. St. Louis: Mosby, 2012.

2. Goldstein GR, Scopp IW. Radiographic interpretation of calcified stylomandibular and stylohyoid ligaments. J Prosthet Dent 1973;30:330–334.

3. Fuentes R, Ottone N, Cantin M, Bucchi C. Análisis de los términos utilizados en la literature científica para referirse a los ligamentos extracapsulares de la articulacíon témporomandibular: Parte II: Ligamentum stylomandibulare y raphe pterymandibulare. Int J Morphol 2014;32:1289–1295.

4. Bumann A, Lotzmann U. TMJ Disorders and orofacial pain. The role of dentistry in a multidisciplinary diagnostic approach.In: Rateitschak KH, Wolf HF (ed). Color Atlas of Dental Medicine. Thieme, 2002.

5. De Paz FJ, Rueda C, Barbosa M, García M, Pastor JF. Biometry and statistical analysis of the styloid process. Anat Rec (Hoboken) 2012;295:742–747.

6. Shivakumar HR, Batra J, Upasi AP, Rai KK. Evaluation of elongated styloid process in patients with oral submucous fibrosis using panoramic radiographs. J Maxillofac Oral Surg 2014; 13:556–559.

7. Lavine MH, Stoopack JC, Jerrold TL. Calcification of the stylohyoid ligament. Oral Surg Oral Med Oral Pathol 1968; 25:55–58.

8. Naik SM, Naik SS. Tonsillo-styloidectomy for Eagle’s syndrome: A review of 15 cases in KVG Medical College Sullia. Oman Med J 2011;26:122–126.

9. Mupparapu M, Robinson MD. The mineralized and elongated styloid process: A review of current diagnostic criteria and evaluation strategies. Gen Dent 2005;53:54–59.

10. Patil S, Ghosh S, Vasudeva N. Morphometric study of the styloid process of temporal bone. J Clin Diagn Res 2014;8: AC04–AC06.

11. Janfaza P. Surgical Anatomy of the Head and Neck. Cambrige, MA: Harvard University, 2011.

12. Beukes J, Reyneke JP, Becker PJ. Medial pterygoid muscle and stylomandibular ligament: The effects on postoperative stabili-ty. Int J Oral Maxillofac Surg 2013;42:43–48.

13. Dimitroulis G. Temporomandibular disorders: A clinical up-date. BMJ 1998;317:190–194.

14. Schiffman E, Ohrbach R, Truelove E, et al. Diagnostic Criteria for Temporomandibular Disorders (DC/TMD) for clinical and research applications: Recommendations of the International RDC/TMD Consortium Network and Orofacial Pain Special Interest Group. J Oral Facial Pain Headache 2014;28:6–27.

15. Peck CC, Goulet JP, Lobbezoo F, et al. Expanding the taxonomy of the diagnostic criteria for temporomandibular disorders. J Oral Rehabil 2014;41:2–23.

16. Shankland WE. Anterior throat pain syndromes: Causes for undiagnosed craniofacial pain. Cranio 2010;28:50–59.

17. Peñarrocha-Oltra D, Ata-Ali J, Ata-Ali F, Peñarrocha- Diago MA, Peñarrocha M. Treatment of orofacial pain in patients with stylomandibular ligament syndrome (Ernest syndrome). Neurologia 2013;28:294–298.

18. Moher D, Liberati A, Tetzlaff J, Altman DG, PRISMA Group. Preferred Reporting Items for Systematic Reviews and Meta-Analyses: The PRISMA Statement. PLoS Med 2009;6: e1000097

19. Thomas BH, Ciliska D, Dobbins M, Micucci S. A process for systematically reviewing the literature: Providing the research evidence for public health nursing interventions. Worldviews Evid Based Nurs 2004;1:176–184.

20. Ernest EA. The Ernest syndrome: An insertion tendinosis of the stylomandibular ligament. J Neuro Orthop Med Surg 1987;7:427–438.

21. Shankland WE. Ernest syndrome as a consequence of stylomandibular ligament injury: A report of 68 patients. J Prosthet Dent 1987;57:501–506.

22. Wilk SJ. Surgical management of refractory craniomandibular pain using radiofrequency thermolysis: A report of thirty patients. Cranio 1994;12:93–99.

23. Sataloff RT, Price DB. Distention of the stylomandibular ligament as a cause of styloid pain syndrome. Ear Nose Throat J 1984;63:412–415.

24. Ernest EA 3rd, Martinez ME, Rydzewski DB, Salter EG. Photomicrographic evidence of insertion tendonosis: The etiologic factor in pain for temporal tendonitis. J Prosthet Dent 1991;65:127–131.

25. Thoma KH. Case report no. 41. Calcification of the stylo-man-dibular ligament. Am J Orthod Oral Surg 1940;26:812.

26. López-Rodríguez MA, Varela M, Camba MA. Aplicaciones de la radiogrecuencia en el tratamiento del dolor crónico benigno. Unarevisión de las publicaciones de los últimos 6 años. Rev Soc Esp Dolor 2001;8:397–411.