A Clicking Larynx: Diagnostic and Therapeutic Challenges
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2 The Laryngoscope VC 2017 The American Laryngological, Rhinological and Otological Society, Inc. Case Report A Clicking Larynx: Diagnostic and Therapeutic Challenges Derrek A. Heuveling, MD, PhD ; Maarten C. van Loon, MD; Rico N. P. M. Rinkel, MD A clicking larynx can be described as a clicking sensation in the neck on swallowing or when moving the head, often associated with a tender or painful area in the neck. Diagnosis and therapy are challenging. In this article, we present a case report and overview of the current literature. The clicking larynx most often is reported to be a result of a displaced cornu superior of the thyroid, an enlarged greater cornu of the hyoid bone, or a short distance between the thyroid and hyoid bone. If a possible cause is identified, surgery can be offered to the patient, although an explanation of the possible underlying anatomical cause also could be satisfying for the patient and avert surgery. Key Words: Larynx, clicking, thyroid, hyoid, swallowing. Laryngoscope, 128: , 2018 INTRODUCTION A clicking larynx can be described as a clicking sensation in the neck on swallowing or when moving the head, often associated with a tender or painful area in the neck. In 1978, Counter described the first patient with a clicking larynx due to a shortened distance between the hyoid bone and the thyroid. 1 Since then, a limited number of patients with a clicking larynx are reported. 2 8 In popular culture, this phenomenon is a puzzling question. 9,10 In this case report, we describe a patient with a clicking larynx, followed by a review of the literature to provide clinical recommendations for the daily practice. CASE REPORT A male patient aged 38 years was referred to our department with a 1-year history of a clicking sound and sensation in the throat during swallowing, mainly on the right side, in combination with a sore throat. Complaints started after a neck trauma during a water polo game. Direct laryngoscopy showed a medially displaced superior cornu of the thyroid at the left (!) side. Palpation of the neck revealed a tender area From the Department of Otolaryngology Head and Neck Surgery, VU University Medical Center (D.A.H., M.C.VL., R.N.P.M.R.), Amsterdam, The Netherlands. Editor s Note: This Manuscript was accepted for publication August 7, The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to D.A. Heuveling, MD, PhD, Department of Otolaryngology Head and Neck Surgery, VU University Medical Center, PO Box 7057, 1007 MB Amsterdam, The Netherlands. dheuveling@gmail.com DOI: /lary situated around both sides of the hyoid bone. A clicking and scratching movement was palpated on swallowing; the hyoid bone seemed to ride over the thyroid lamina on both sides. Computed tomography (CT) of the neck showed the medially displaced superior cornu of the thyroid at the left side. Videofluoroscopic swallowing exam showed an evident contact of the hyoid bone and the thyroid during laryngeal elevation (Fig. 1). A possible explanation for the patient s complaints could be a medially displaced superior cornu in combination with a shortened distance between the hyoid bone and thyroid. Because the patient s complaints resulted in a substantial discomfort and psychological distress, the patient was very motivated for a surgical treatment. We decided to perform a staged treatment plan. First, in a minimally invasive procedure, we excised the superior cornu transorally using CO2 laser. Because this did not result in a relief of symptoms, we performed a partial resection of the lamina of the thyroid on both sides via a midline neck incision (Fig. 2). The latter resulted in an immediate and complete relief of symptoms (with a follow-up of 6 months). DISCUSSION A clicking larynx is an uncommon phenomenon that may be associated with severe physical and psychological distress. Table I provides an overview of reported patients in the literature. As far as we know, only 16 patients previously have been described. 1 8 Based on these patients, the condition seemed more prevalent in women (11 of 16, 69%). The age of the patients ranged from 15 to 66 years, with a substantial number of patients being younger than 30 years (7 of 16, 44%). In all patients, a (possible) explanation for the complaints was described (Fig. 3): the clicking sound could be the 697
3 Fig. 1. Videofluoroscopic swallowing examination showing the position of the hyoid bone (black dotted line) and thyroid (white dotted line) in rest (A) and while swallowing (B). Note that the thyroid rides over the hyoid bone on swallowing (white arrow). result of friction between superior cornu of the thyroid and hyoid bone due to a displaced superior cornu (6 of 16, 38%); shortened distance between the hyoid bone and the thyroid (3 of 16, 19%); or alternatively, impingement of an enlarged greater horn of the hyoid bone and the cervical spine (6 of 16, 38%). In one patient (6%), there was abnormal bone formation in the thyrohyoid ligament for which an ossified triticeal was hypothesized. 2 Twelve patients (75%) underwent surgery, which included resection of the displaced superior cornu (n 5 6), enlarged part of the hyoid bone (n 5 3), upper part of the thyroid lamina (n 5 2), or abnormal bone formation in the thyrohyoid ligament (n 5 1). In all operated patients, this resulted in a complete resolution of symptoms, most of them immediately following surgery. However, it is important to note that there is no information about (long-term) follow-up in these patients. Of the four patients who were not operated, three patients were not willing to undergo surgery after extensive diagnostic evaluation and explanation of the possible cause of the complaints, whereas the remaining patient was lost to follow-up. Fig. 2. Peroperative view of the right superior part of the thyroid. The black dotted line indicates the line of resection. 698 To understand the etiology of the clicking sound and sensation, neck trauma probably provides the best explanation, but a history of neck trauma was identified only in a minority (25%) of the patients. Other explanations include an altering shape of the thyroid, ossification of the, or an increase in laxity of the supporting ligaments of the joints during aging. These explanations could not fully be supported because a substantial number of patients were adolescents and young adults. Predisposing factors for altering the position of the hyoid bone, which may contribute to a clicking larynx, are neck dissection, 11 mandibular advancement operations, 12 resection of the styloid process, 8 and mouth breathing. 13 However, of the reported patients, there only was one patient in whom the complaints occurred several years after resection of a styloid process. 8 Therefore, in most patients a satisfying etiology could not be given. In patients with a clicking larynx, a thorough examination of the neck should be performed. Careful palpation and manipulation of the hyoid bone and the thyrohyoid region on swallowing may reveal a clicking sound or crepitus that may be helpful for understanding the underlying mechanism. Direct laryngoscopy will be normal in most patients. Computed tomography of the neck may show a displaced superior cornu or an enlarged greater horn of the hyoid bone. If the clicking occurs on head turns, a CT with the head rotated may provide additional information. 5,8 Videofluoroscopic swallowing examination provides additional dynamic information and may demonstrate a short distance between the hyoid bone and thyroid (see Fig. 1). A clicking larynx is different than hyoid bone syndrome or Eagle syndrome, in which discomfort is present in the absence of a clicking sensation. CONCLUSION The clicking larynx may be related to one of the structures of the larynx or hyoid bone and can result in severe physical and psychological distress. It is a multifactorial symptom for which careful examination and diagnostic testing should be performed. If a possible
4 699 Author N Age M/F TABLE I. Overview of Reported Patients With a Clicking Feeling With or Without Pain in the Neck on Swallowing or Head Movements. Prior Neck Trauma or HN surgery (Possible) Cause Description of Surgery Follow-up Counter M None Shortened distance hyoid bone and thyroid Ilankovan M None Abnormal bone forming a joint with the superior cornu of the thyroid and the greater horn of the hyoid bone Excision of upper part of thyroid lamina Resection of abnormal bone Makura et al F None Enlarged greater horn of hyoid bone Resection of enlarged part of hyoid bone Hilali et al F None Enlarged greater horn of hyoid bone Surgery was proposed, but patient was satisfied after explanation of the cause Smith et al F None Posteriorly and medially displaced superior cornu Excision of superior cornu 27 F Neck trauma Shortened distance hyoid bone and thyroid Excision of upper part of thyroid lamina 18 M Neck trauma Posteriorly and medially displaced superior cornu Excision of superior cornu 39 F None Posteriorly and medially displaced superior cornu Excision of superior cornu and posterior edge thyroid ala excision (staged) 25 M None Posteriorly and medially displaced superior cornu Excision of superior cornu 51 F Neck trauma Posteriorly and medially displaced superior cornu Excision of superior cornu 15 F Neck trauma Posteriorly and medially displaced superior cornu Excision of superior cornu and hyoid trim (staged) Jungheim 3 60 F None Probably an enlarged greater horn of hyoid bone Patient not motivated for surgery? et al F None Probably an enlarged greater horn of hyoid bone No surgery? 57 M None Probably a very prominent ala of the thyroid No surgery after explanation of complaints? Singh et al F None Enlarged greater horn of hyoid bone Resection enlarged part of hyoid bone Dieckhoff 1 64 F Resection of styloid et al. 8 process 3 years before complaints? 5 no information provided; 5 complete resolution of symptoms; F 5 female; HN 5 head and neck; M 5 male; N 5 number. Enlarged greater horn of hyoid bone Resection of enlarged part of hyoid bone?
5 cause is identified, surgery can be offered to the patient, although explanation of the possible underlying anatomical cause also could be satisfying for the patient and avert surgery. Fig. 3. Slightly lateral view on the hyoid bone and thyroid with the possible causes of a clicking larynx: 1) short distance between hyoid bone and superior part of thyroid ; 2) displaced superior cornu; 3) enlarged greater horn of hyoid bone; and 4) abnormal bone formation in the thyrohyoid ligament. The black dotted lines indicate the line of resection during surgery. BIBLIOGRAPHY 1. Counter RT. A clicking larynx. J Laryngol Otol 1978;92: Ilankovan V. An anomaly of the thyro-hyoid articulation. J Laryngol Otol 1987;101: Makura ZG, Nigam CB. The clunking neck. J Laryngol Otol 1995;109: Hilali AS, Saleh HA, Hickey SA. Clicking hyoid. J R Soc Med 1997;90: Smith ME, Berke GS, Gray SD, et al. Clicking in the throat: cinematic fiction or surgical fact? Arch Otolaryngol Head Neck Surg 2001;127: Jungheim M, Miller S, Ptok M. [Clicking larynx syndrome: a literature review and multiple-case report]. [Article in German]. HNO 2013;61: Singh V, Priya K, Bhagol A, et al. Clicking hyoid: a rare case report and review. Natl J Maxillofac Surg 2015;6: Dieckhoff FJ. Clicking Larynx. Fortschr Rontgenstr 2015;187: Steady Health. Clicking in adams apple when swallowing. Available at: Accessed August 3, No More Panic. Clicking when swallowing? Available at: nomorepanic.co.uk/showthread.php?t= Accessed August 3, Hirai H, Omura K, harada H, et al. Sequential evaluation of swallowing function in patients with unilateral neck dissection. Head Neck 2010;32: Jiang C, Yi Y, Jiang C, et al. Pharyngeal airway space and hyoid bone position after different orthognathic surgeries in Skeletal Class II patients. J Oral Maxillofac Surg 2017;75: Bibby RE. The hyoid bone position in mouth breathers and tongue-thrusters. Am J Orthod 1984;85:
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