Copyright © 2020 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited. Transient Velopharyngeal Insufficiency After Calcified Stylohyoid Ligament Resection Osman Halit C¸ am, MD, Pelin Koc¸dor, MD, and Levent N. O ¨ zlu¨og˘lu, FACS y Abstract: Elongation of the stylohyoid process or calcification of the stylohyoid ligament is known as the Eagle syndrome. Mostly, it is seen incidentally on imaging or with extreme suspicion and usually patients are asymptomatic. Surgery is the preferred method in symptomatic patients. Trans- cervical or transoral methods may be preferred as surgical route. A 28-year-old female patient who had formerly underwent tonsillectomy presented with throat and ear pain. A neck computed tomography was performed, and the patient was diagnosed as Eagle Syndrome. Surgery was recommended. Patient developed transient velopharyngeal insufficiency on postoperative day 4. Ventilation exercise and follow-up was recom- mended. Complaints of the patient decreased on the 15th day. It should be kept in mind that stylohyoid ligament may be calcified in young age group and middle age group patients with dysphagia or odynophagia, and differential diagnosis should be performed. Another issue is the condition of velofaringeal insuffi- ciency which may occur due to the damage of the pharynx muscles by deep dissection during surgery. Key Words: Eagle syndrome, stylohyoid ligament, unusual complication, velopharyngeal insufficiency W att Eagle first described Eagle syndrome in 1937 as elongation of stylohyoid process or calcification of stylo- hyoid ligament. 1 To name a stylohyoid process elongation, stylo- hyoid process has to be longer than 25 mm. 2 Although its etiology is not fully known, familial cases have been reported that suggest an autosomal dominant transition. 3 Most patients with an incidental diagnosis of elongated stylo- hyoid process may have no pain. The most common cause of admission is pain, and it usually refers to the ear. Pain is often severe during swallowing and may sometimes refer to the temporal region. Patients are usually diagnosed after consulting several clinicians. In the physical examination of these patients, the palpation of the tonsillar fossa is severely painful. CLINICAL REPORT A 28-year-old female patient referred to our clinic with unbearable throat and ear pain. The patient had consulted four otolaryngologists with 1 year and underwent tonsillectomy in January 2019. After the patient’s insistence, computed neck tomography was performed. Based on tomography results, the patient was diagnosed as bilat- erally elongated and calcified stylohyoid ligament (Eagle Syn- drome) that was dominant on the left side. (Fig. 1A and B) Under general anesthesia, a parallel entry was made to the left plica palatoglossus, and the constrictor pharyngeus and salpingo- pharyngeus muscle were incised. Then, soft tissues were peeled off by sharp dissection, and the calcified ligament was taken into traction and resected transorally from the lowest point. (Fig. 1C) Muscles were sutured with 3/0 vicryl and mucosa with 4/0 vicryl. The operation was completed without any complication. On postoperative day 4, the patient stated that she could not say words beginning with ‘‘s’’ or ‘‘h’’ or containing these letters and felt a slight congestion in her ear. In the examination, it was seen that the sutures were still in place, and it was found that when the patient closes her nose, she was able to say these words. On the post- operative day 8, Voice Handicap Index-10 was administrated and the score was found 21. 4 (Supplementary Table 1, http://links.lww.- com/SCS/B357) Valsalva Maneuver and speech exercises were recommended. Voice Handicap Index-10 was reapplied on the postoperative day 26 and the score was 1. DISCUSSION There are several theories about pain etiology in Eagle syndrome. One of them is based on fibrosis in the stylohyoid ligament apex after tonsillectomy. 5 Considering that our patient underwent ton- sillectomy in order to minimize her pain, our case does not support this theory. Another theory for pain is the compression of the cranial nerves and the subsequent pressure pain due to this compression. 6 Considering our patient’s admittance to several hospitals with these complaints for about 1 year, we think the pain was more likely to have emerged with this mechanism. The fact that the immediate resolution of postoperative pain is another finding that supports this theory. The most common complications that develop in the postopera- tive period in patients undergoing transoral surgery are first bite syndrome, numbness, infection, significant vascular injury, cranial nerve injuries, and recurrences. 7 A method is described in the literature to minimize these complications, which follows the ramus mandibula border via transoral route and argues to be relatively safer. 8 In our patient, none of these complications that are well described in the literature developed. From the Ear, Nose, and Throat Department, Bas¸kent University, Istanbul Hospital, Istanbul; and y Ear, Nose, and Throat Department, Bas¸kent University, Ankara Hospital, Ankara, Turkey. Received November 6, 2019. Accepted for publication March 2, 2020. Address correspondence and reprint requests to Osman Halit C¸ am, MD, Bas¸kent U ¨ niversitesi Tip Faku¨ltesi, Istanbul Sag˘lik Uygulama ve, Hastanesi Altunizade Mahallesi, 7, Kisikli Caddesi, Oymaci Sk., No: 2 34662 U ¨ sku¨dar, Istanbul, Turkey; E-mail: osman.cam@gmail.com The authors report no conflicts of interest. Supplemental digital contents are available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s Web site (www.jcraniofa- cialsurgery.com). Copyright # 2020 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000006467 FIGURE 1. A. 3D Computed tomography. White arrow: Hyoid bone, Blue arrow: Calcified stylohyoid ligament, Orange arrow: Skip calcification. B. 3D Computed tomography. Coronal section of Neck Computed Tomography. Orange arrows: Right and left calcified stylohyoid ligament. C, Asterix: Alice Clamp, Up Arrow: Intubation Tube, Left Arrow: Calcified Stylohyoid Ligament, Star: Uvula. Brief Clinical Studies The Journal of Craniofacial Surgery Volume 31, Number 6, September 2020 e540 # 2020 Mutaz B. Habal, MD