A 51-year-old woman of African origin was referred to the Service d’Odontologie of the Hôpital Pitié-Salpêtrière (GHPS) in Paris, France, by her general practitioner. She described a 72-month history of pain in the left retroangulomandibular region, radiating to the ipsilateral ear and dentomaxillar region, of undetermined cause. The patient was referred to the specialized orofacial pain clinic of this service.

Medical history

Anamnesis revealed that the pain began in 2013, when the patient started suffering from pain in the oropharyngeal region, radiating to the left maxilla and ear region, which was perceived as a foreign-body sensation, associated with painful chewing and swallowing, and noise in the ipsilateral ear. Acetaminophen (1000 mg, p.o., t.i.d.) provided only modest pain relief. After consulting a dentist in 2014, tooth #28 was extracted, without any improvement of her condition. Thereafter, she was referred to an otolaryngologist (ORL), who suspected sinusitis and treated it with antibiotics and corticosteroids, but no improvement was observed. The ORL then performed sinus meatotomy in 2015, but again no improvement of the symptoms was noticed. Finally, the patient consulted another dentist, who extracted tooth #25 and later tooth #38, after an episode of swelling of the left mandibular region unresponsive to antibiotic therapy (amoxicillin).

Additional findings

The patient had undergone three C-sections and a hysterectomy. She also reported chronic stress attributed to a complicated marital life, which was not under medical treatment. In an episode of domestic violence in 2003, she was punched by her husband in the left retroangulomandibular region and behind the ascendant branch of the mandible (ramus). This was the only traumatic injury reported by the patient.

After this 3-year period of unsuccessful medical nomadism, in 2016 she consulted the orofacial pain clinic of the Hôpital Pitié-Salpêtrière in Paris, France. After a rigorous clinical and radiological examination by a senior expert practitioner (Y.B.), Eagle’s syndrome was diagnosed.

Clinical findings

The patient reported a foreign-body sensation in the oropharyngeal region, and painful chewing and swallowing, mostly with solid foods, which had worsened over time. The pain was experienced almost every day, sometimes spontaneously, and it was enhanced by rotation of the head to the left. It could also be elicited by palpation of the tonsillar fossa. The pain was described as “aching during mouth opening” and “dull as a pharyngitis,” and was associated with a noise in the ispilateral ear. Pain quality was assessed by the DN4 questionnaire for neuropathic pain [7], which revealed only mechanical allodynia. The severity of the pain was rated 5/10 using the Numeric Pain Rating Scale (NPRS), and was partially relieved by acetaminophen (1000 mg, p.o., t.i.d.).

Orofacial evaluation according to DC recommendations [8] revealed a slight tumefaction at the left angle of the mandible without any modification in the appearance of the skin. Digital palpation revealed painful left masseter, mylohyoid muscle and the posterior part of the digastric muscles. Ear and nose examination was unremarkable. A left deviation of the mandibula during mouth opening was also observed. Intraoral examination revealed an asymmetry at the level of the palatine tonsils. The ipsilateral (left) tonsil seemed bent inside, and palpation of the oropharynx highlighted the left voluminous styloid process visible on orthopantomogram (see below).

Radiographic findings

Visual inspection and panoramic radiography (Fig. 1) confirmed missing teeth #25, #28, and #38, and no dental/periodontal disease treatment. A radiopacity was visible distal of tooth #37, suggesting the presence of a residual root of #38 or an osseous condensation with no concomitant inflammatory process. As suspected by palpation, both SP were long, with the left one being more elongated. Cone beam computed tomography (CBCT) (Fig. 2) revealed a pseudo-articulation in the left SP: a superior mineralized segment joined to an inferior mineralized segment by a single pseudo-articulation with respective maximal dimensions of 37.3 × 13.1 mm and 32.5 ×10.5 mm, located above the inferior border of the mandible. This kind of SP corresponds to type II (pseudo-articulated) in Langlais’s classification [9] (Table 1).

Fig. 1
figure 1

Orthopantomogram: teeth #25, #28, and #38 were extracted. Radiopacity (dotted arrow) is visible distal of #37, suggesting a residual root of #38 as a result of incomplete surgical treatment. Note that both styloid processes (solid arrows) are prolonged, but the left one was more prolonged and voluminous than the right

Fig. 2
figure 2

Dental cone beam computed tomography. A Right side: asymptomatic, B Left side, in which the styloid process corresponds to type II of Langlais’s classification; a superior mineralized segment is joined to an inferior mineralized one by a single pseudo-articulation that is located above the inferior border of the mandible

Table 1 Left: criteria for positive diagnosis of ES20–22; Right: differential diagnosis of ES1

Based on the medical history, clinical and radiographic findings, the final diagnosis of Eagle’s syndrome was made.

Management of the Eagle’s syndrome

The patient was then referred to the ORL service of Hôpital Pitié-Salpêtrière, where a left stylectomy was decided, and performed using a classical external access technique, by an expert surgeon (F.T.) under general anesthesia [10]. At the 3-month follow-up visit, the pain was almost completely relieved. A slight limitation of mouth opening and mild pain sensation at left temporomandibular joint were still present, thus physical therapy was prescribed. At the second, third, and fourth recall visits at 2.5, 3, and 4 years after surgery respectively, total pain relief was observed and the functional discomfort had disappeared.

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