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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 10  |  Issue : 3  |  Page : 571-576  

The pterygoid hamulus syndrome – An important differential in orofacial pain


1 Department of Oral and Maxillofacial Surgery, Government Dental College and Hospital, Mumbai, Maharashtra, India
2 Department of Oral and Maxillofacial Radiology, Government Dental College and Hospital, Mumbai, Maharashtra, India

Date of Web Publication11-Mar-2020

Correspondence Address:
Dr. Neha Aggarwal
118, Department of Maxillofacial Surgery, Government Dental College and Hospital, Fort Area, Mumbai - 400 001, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ccd.ccd_796_18

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   Abstract 


The purpose of this review was to document cases of pterygoid hamulus (PH) syndrome and to describe the various etiology, differential diagnosis, and management strategies so far reported in literature. Here, we also present two case reports of PH syndrome. A comprehensive search in PubMed/Medline database was done using MeSH terms such as “Pterygoid Hamulus,” “Pterygoid Hamulus Syndrome,” and “Hamular Bursitis” using various Boolean operators such as “AND” and “OR”. Till date, 31 cases of this entity including the present cases have been found. Conservative management was followed in the earlier reported cases; however, most cases were treated by surgical resection.

Keywords: Bursitis, facial, hamular, pain, syndrome


How to cite this article:
Kende P, Aggarwal N, Meshram V, Landge J, Nimma V, Mathai P. The pterygoid hamulus syndrome – An important differential in orofacial pain. Contemp Clin Dent 2019;10:571-6

How to cite this URL:
Kende P, Aggarwal N, Meshram V, Landge J, Nimma V, Mathai P. The pterygoid hamulus syndrome – An important differential in orofacial pain. Contemp Clin Dent [serial online] 2019 [cited 2020 May 26];10:571-6. Available from: http://www.contempclindent.org/text.asp?2019/10/3/571/280424




   Introduction Top


Orofacial pain is a diagnostic challenge because of its varied origins. In the past, different authors have observed that irritation in the palate corresponding to the pterygoid hamulus (PH) has referred symptoms in the head-and-neck area.[1],[2] The PH, a small but significant process of the sphenoid bone, has been associated with orofacial pain that is termed as “pterygoid hamulus syndrome.” The term is used to describe pain in the palatal and pharyngeal region that is due to inflammation of the hamular region on account of bursitis or an elongated hamulus. Shankland in 1996 and Salins and Bloxham in 1989 explained the inflammation of the bursa covering tensor veli palatini tendon as hamular bursitis.[3],[4]

More often than not, it is misdiagnosed as temporomandibular disorder (TMD), impacted third molars, trigeminal and glossopharyngeal neuralgia, abnormalities of the styloid process and its associated ligaments, tumors, cysts, and infection of the middle ear.[5] Through this review article and case series, we aim to highlight the importance of PH syndrome in the differential diagnosis of orofacial pain of unknown origin.


   Case Reports Top


Case 1

A 45-year-old male patient reported with the chief complaint of pain on swallowing for 2 years.

The pain was insidious in onset, progressed gradually from mild to severe over 2 years. The pain was typically located in the palatal and pharyngeal area. The patient had visited multiple clinicians and had undergone various treatments for the same. The diagnosis was changed from temporomandibular joint (TMJ) disorder to pharyngitis to neuralgia. The patient's medical, dental, and family history was unremarkable. On extraoral examination, no tender areas could be localized. Bilateral TMJ examination was normal. Intraoral examination showed bilateral focal areas of swelling palatal to the maxillary tuberosity [Figure 1]. The swelling was hard on palpation suggestive of bony enlargement, with typical blanching erythema seen surrounding the swelling. Marked tenderness could be elicited on slight provocation in the same areas.
Figure 1: Clinical photograph showing bilateral swellings in the hamular region

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The differential diagnosis included elongated pterygoid hamular process, glossopharyngeal neuralgia, and idiopathic orofacial pain.

Radiographic examination with cone-beam computed tomography (CBCT) was performed. CBCT revealed bilateral enlarged hamuli with a lateral inclination. Precise location and measurements were obtained [Figure 2].
Figure 2: Coronal section of cone-beam computed tomography showing elongated hamuli with a lateral inclination

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All the features seen on radiographs with clinical correlation led to the final diagnosis of PH syndrome.

Case 2

A 23-year-old male patient reported with the chief complaint of pain on the left side of the face for 2–3 years.

The pain was severe, radiating to the ipsilateral ear and temporal region. The pain was aggravated on swallowing and relieved on medications. The patient was on intermittent pain relief medications and was earlier diagnosed with myofascial pain dysfunction syndrome. The patient's medical, dental, and family history was unremarkable. Extraoral examination revealed no abnormalities. Bilateral TMJ examination was normal with no prominent signs and symptoms of TMJ disorder. Intraoral examination showed focal areas of slight bony enlargement with typical blanching palatal to the maxillary tuberosity on the left side. Marked tenderness could be elicited on slight provocation in the same area.

The differential diagnosis included elongated pterygoid hamular process, glossopharyngeal neuralgia, and idiopathic orofacial pain.

Radiographic investigations with intraoral periapical radiograph were advised, which demonstrated a prominent left PH [Figure 3]. Further evaluation with CBCT was performed for precise location and measurements in both axial and coronal sections. Both radiographic and clinical findings led to the final diagnosis of PH syndrome.
Figure 3: Intraoral periapical radiograph showing an elongated hamulus

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Surgical resection of the elongated hamuli was planned under local anesthesia in both cases. Intraorally, a longitudinal incision over the bony prominence, posteromedial to the maxillary tuberosity, was made. Dissection was done to isolate and expose the elongated hamulus on the left side, which was located along the palatal mucogingival line [Figure 4]. The hamulus was excised with a bur, and bony margins were smoothened. Primary closure was obtained. Postoperative healing was uneventful. The patients reported with relief of symptoms thereafter and were kept on regular follow-up up to 2 years with no recurrence [Figure 5].
Figure 4: (a) Intraoperative photograph showing the exposed hamulus. (b) Intraoperative photograph showing the excised hamulus. (c) Intraoperative photograph after primary closure

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Figure 5: Intraoral post operative photograph showing good healing

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   Review of Literature Top


In literature, there have been various anatomic and radiographic studies to locate the spatial position and angulation of PH.[6],[7],[8] Anatomic implications of this structure with respect to age and function have been studied.[9] Some authors also correlated PH length to obstructive sleep apnea.[10] There are very few cases of PH syndrome reported in literature. A comprehensive search in PubMed/Medline database was done using MeSH terms such as “Pterygoid Hamulus,” “Pterygoid Hamulus Syndrome,” and “Hamular Bursitis” using various Boolean operators such as “AND” and “OR”. To the author's knowledge, till date, 31 cases of this entity including the present cases have been found [Table 1].[1],[2],[3],[4],[5],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24] The age group in our review ranged from 5 to 62 years. Male preponderance was seen. All cases reported were symptomatic except the two cases reported by Wooten et al. who showed elongated hamulus as an asymptomatic mass in the soft palate region. Conservative management was followed in the earlier reported cases; however, most cases were treated by surgical resection. Similar technique was followed in our cases owing to the elongated hamulus. There is also a variation in terminologies of the entity such as “Pterygoid Hamular Syndrome,” Hamular Bursitis,” and “Hamulus Hypertrophia.”
Table 1: Literature review

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   Discussion Top


The PH can be responsible for atypical pain in the oral cavity and pharynx. Its close topographical relationship to the upper dental arch and pharynx renders it of interest to all specialties that are involved with this region. There have been various efforts to describe the mechanism of hamular pain, but the precise etiology is not known.

Careful assessment of this region helped us to trace and manage the ambiguous and conflicting symptoms related to PH elongation in the oral cavity. Due to the rarity of the disease, its exclusion from the diagnosis becomes a rule rather than the exception. This leads to delay in treatment. In both the cases, the patients suffered for 2–3 years before reaching a final diagnosis. Often, they are misdiagnosed as Eagle's syndrome, TMDs, geniculate ganglion neuralgia, glossopharyngeal neuralgia, cyst and tumors, otitis media, foreign bodies, burning mouth syndrome, and impacted third molars. In our cases, the diagnosis was made based on clinical and radiological findings. Clinical features commonly seen include palatal pain with the offending side more erythematous than the opposite side, firm swelling or enlargement and reddness of the palatal mucosa over the hamulus, sharp localized pain in the hamular region, ear pain, difficulty and pain with swallowing. Along with these clinical features, conventional radiographic imaging such as lateral cephalometric, submentovertex, and tomography and advanced imaging including computed tomography scan in axial and coronal planes with three-dimensional views can be used.

Responsiveness to nonsteroidal anti-inflammatory drugs and a long history of symptoms helped us in ruling out neuralgias. Absence of any extraoral muscular or TMJ findings ruled out TMJ disorders. However, Dupont and Brown found positive findings of hamular pain in a study of 493 TMD patients.[21]

Some authors have suggested infiltration of local anesthesia in the region as an excellent diagnostic aid.[8] In our cases, the presence of a tender erythematous bony prominence excluded the need for local anesthetic infiltration. In addition, the mere presence of an elevated soft-tissue area is not enough for diagnosis as authors have previously found asymptomatic elongated hamulus as a swelling in this region.[2]

There have been various pathogeneses of PH syndrome postulated by authors. Sasaki et al. suggested that the abnormal PH is the cause of mechanical invigoration to the adjacent tissues, thus interrupting the normal action of the tensor veli palatini muscle, which can further cause bursitis.[19] Furthermore, these events also invigorate the lesser and greater palatine nerve, glossopharyngeal nerve, and facial nerve, which may mimic neuralgia-like symptoms. In addition, tensor veli palatini dysfunction may also lead to symptoms in the meatus, which was seen in both our cases.

Ramirez et al. suggested that such referred expression focuses on the common neural interconnection between the oro-masticatory and the otic system.[5] They also suggested a bio-psychosocial aspect in their two case reports which reported with concomitant TMD symptoms and were managed for both.

The position, length, and medio-lateral inclination of the hamulus are of concern for the adequate function of several muscles such as tensor veli palatini, palatopharyngeus, and pharyngeal constrictors.[24] Due to availability of the advanced imaging modalities such as CBCT, measurements of the length and inclination have become possible. This not only assists in diagnosis, but is also of immense use in treatment. The length of the PH in our cases was found to be within the range of 4.9–7.2 mm, with the positions of PHs inclined laterally in the coronal plane. The average length of the left hamulus was 5.0 mm and the that of the right was 4.9 mm as found by Eyrich et al. in normal individuals, whereas in the CBCT study by Orhan et al., it was found to be 5.40 and 5.48 mm on the right and left sides, respectively.[17],[10] Excessive length of the hamulus is not the only etiological factor in this syndrome. According to Charbeneau and Blanton, other relationships can exist, as follows: (1) the medial pterygoid plate and therefore its hamular process, may have been located more inferiorly than usual with respect to the palate; or (2) the mucosa of the soft palate, could be thinner.[13] However, bilateral occurrence is a strong evidence of anatomic entity as the etiology in one of our cases.

Treatment may be medicinal, minimally invasive including injections, or invasive such as surgical resection. For conventional minimally invasive treatment, infiltration of 1 ml of dexamethasone 4 mg per ml has been advised to mitigate the inflammation. In addition, anti-inflammatory medications such as ibuprofen, 600 mg–800 mg, every 6 h should be prescribed. Patients should be re-evaluated on a regular basis. If there is improvement of symptoms, injections should be repeated and the anti-inflammatory medication should be extended and maintained, with regular follow-ups.

If conservative treatment proves unsuccessful, or the length of PH is above the normal defined limits, surgical management should be considered. If osteophytes, a prominent hamular process or bursa fibrosis, are present, the surgical treatment should be followed.

If inflammatory or fibrotic changes of the bursa are not present, such as in our case, a hamulotomy should be accomplished, or the hamulus should be removed, by the use of a rongeur. One reported complication of hamulotomy is interruption of the tensor veli palatini function over this osseous hook that results in tubic dysfunction with otic expression as hypoacusis besides loss of a palate-pharynx seal during speaking and swallowing. [Table 2] describes a clinical guide enumerating the symptoms, differential diagnosis, and treatment options for this entity.
Table 2: Clinical guide for the diagnosis and management of pterygoid hamulus syndrome

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   Conclusion Top


PH syndrome, a rare cause of orofacial pain, was reported in this article. Through this article, we have presented a literature appraisal on the incidence, diagnosis, and treatment of this condition. The palatal and pharyngeal areas warrant particular clinical surveillance during the differential diagnosis of orofacial pain. As the treatment for this entity is markedly different from that for the other pain conditions of this region, clinicians should contemplate a potential diagnosis of pterygoid hamular syndrome.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patients have given their consent for their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Hertz RS. Pain resulting from elongated pterygoid hamulus: Report of case. J Oral Surg 1968;26:209-10.  Back to cited text no. 1
    
2.
Wooten JW, Tarsitano JJ, Reavis DK. The pterygoid hamulus: A possible source for swelling erythema, and pain: Report of three cases. J Am Dent Assoc 1970;81:688-90.  Back to cited text no. 2
    
3.
Salins PC, Bloxham GP. Bursitis: A factor in the differential diagnosis of orofacial neuralgias and myofascial pain dysfunction syndrome. Oral Surg Oral Med Oral Pathol 1989;68:154-7.  Back to cited text no. 3
    
4.
Shankland WE 2nd. Bursitis of the hamular process. Part II: Diagnosis, treatment and report of three case studies. Cranio 1996;14:306-11.  Back to cited text no. 4
    
5.
Ramirez LM, Ballesteros LE, Sandoval GP. Hamular bursitis and its possible craniofacial referred symptomatology: Two case reports. Med Oral Patol Oral Cir Bucal 2006;11:E329-33.  Back to cited text no. 5
    
6.
Khoubivand M, Ghaffari R, Anjomshoa M. Morphological evaluation of pterygoid hamulus through MPR cone-beam computed tomography image. Bull Env Pharmacol Life Sci 2015;4:144-50.  Back to cited text no. 6
    
7.
Orhan K, Sakul BU, Oz U, Bilecenoglu B. Evaluation of the pterygoid hamulus morphology using cone beam computed tomography. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e48-55.  Back to cited text no. 7
    
8.
Langenegger JJ, Lownie JF, Cleaton-Jones PE. The relationship of the greater palatine foramen to the molar teeth and pterygoid hamulus in human skulls. J Dent 1983;11:249-56.  Back to cited text no. 8
    
9.
Krmpotić-Nemanić J, Vinter I, Marusić A. Relations of the pterygoid hamulus and hard palate in children and adults: Anatomical implications for the function of the soft palate. Ann Anat 2006;188:69-74.  Back to cited text no. 9
    
10.
Oz U, Orhan K, Aksoy S, Ciftci F, Özdoǧanoǧlu T, Rasmussen F. Association between pterygoid hamulus length and apnea hypopnea index in patients with obstructive sleep apnea: A combined three-dimensional cone beam computed tomography and polysomnographic study. Oral Surg Oral Med Oral Pathol Oral Radiol 2016;121:330-9.  Back to cited text no. 10
    
11.
Gores RJ. Pain due to long hamular process in the edentulous patient. Report of two cases. J Lancet 1964;84:353-4.  Back to cited text no. 11
    
12.
Panzoni E, Clauser C. The pain syndrome of the pterygoid hamulus. Clinical contribution. Riv Ital Stomatol 1978;47:12-7.  Back to cited text no. 12
    
13.
Charbeneau TD, Blanton PL. The pterygoid hamulus. A consideration in the diagnosis of posterior palatal lesions. Oral Surg Oral Med Oral Pathol 1981;52:574-6.  Back to cited text no. 13
    
14.
Hjørting-Hansen E, Lous I. Hamulus pterygoid syndrome. Tandlaegebladet 1987;91:833-7.  Back to cited text no. 14
    
15.
Hjørting-Hansen E, Lous I. The pterygoid hamulus syndrome. Ugeskr Laeger 1987;149:979-82.  Back to cited text no. 15
    
16.
Kronman JH, Padamsee M, Norris LH. Bursitis of the tensor veli palatini muscle with an osteophyte on the pterygoid hamulus. Oral Surg Oral Med Oral Pathol 1991;71:420-2.  Back to cited text no. 16
    
17.
Eyrich GK, Locher MC, Warnke T, Sailer HF. The pterygoid hamulus as a pain-inducing factor. A report of a case and a radiographic study. Int J Oral Maxillofac Surg 1997;26:275-7.  Back to cited text no. 17
    
18.
Dias G. Pterygoid hamulus bursitis: One cause of craniofacial pain. J Prosthet Dent 1997;78:111-2.  Back to cited text no. 18
    
19.
Sasaki T, Imai Y, Fujibayashi T. A case of elongated pterygoid hamulus syndrome. Oral Dis 2001;7:131-3.  Back to cited text no. 19
    
20.
Fu Y, Peng J, Chen W. The pterygoid hamulus syndrome with the main discomfort of pharynx. Lin Chuang Er Bi Yan Hou Ke Za Zhi 2004;18:134-5.  Back to cited text no. 20
    
21.
Dupont JS Jr., Brown CE. Comorbidity of pterygoid hamular area pain and TMD. Cranio 2007;25:172-6.  Back to cited text no. 21
    
22.
Cho JY, Cheon KY, Shin DW, Chun WB, Lee H. Pterygoid hamulus bursitis as a cause of craniofacial pain: A case report. J Korean Assoc Oral Maxillofac Surg 2013;39:134-8.  Back to cited text no. 22
    
23.
Roode GJ, Bütow KW. Pterygoid Hamulus syndrome – Undiagnosed. SADJ 2014;69:70-1.  Back to cited text no. 23
    
24.
Bandini M, Corre P, Huet P, Khonsari RH. A rare cause of oral pain: The pterygoid hamulus syndrome. Rev Stomatol Chir Maxillofac Chir Orale 2015;116:380-3.  Back to cited text no. 24
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

  [Table 1], [Table 2]



 

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