Life-threatening mandibular angle gigantic osteoma presenting with severe dyspnoea

Abstract

Osteoma is a benign osteogenic tumour. Solitary osteoma of the jaws usually involves the mandible and commonly remains asymptomatic. Purpose of this article is to report a case of life-threatening gigantic mandibular osteoma in an edentulous woman in her 70s developed in the lingual side of the mandibular angle presenting at emergency department with dyspnoea and discuss the correct management of the patient and the surgical approach for space occupying mass in the pharapharyngeal space.

Background

Osteoma is a benign osteogenic tumour that occurs from proliferation of compact bone, cancellous bone or their combination; osteoma of the jaws is a common tumour that usually involves the mandible and commonly remains asymptomatic. The growth of a mandibular osteoma is slow, and the symptom related to the osteoma is the asymmetry due to hypertrophy of the mandible; osteoma may appear in routine panoramic X-ray, too.¹

In the international literature, cases of dyspnoea related to the presence of mandibular osteoma are very rare; dyspnoea is a common but dangerous symptom in the elderly and commonly a reason for emergency.²

In the present article, we report a case of a life-threatening mandibular gigantic osteoma presenting with severe dyspnoea in elderly patient and our surgical care with postoperative follow-up.

Case presentation

An edentulous woman in her 70s was referred to the emergency department for dyspnoea. Haemogasanalysis revealed hypoxaemia and hypercapnia, with an initial acidosis (PaO₂ 55 mm Hg

PaCO₂ 50 mm Hg). At the clinical intraoral examination, an asymmetry was observed with the left lingual side of the mandible and mandibular floor shifted, with a displacement of the left oropharingeal wall and reduced airway. CT scan revealed a radiopaque mass of 47×18×23 mm occupying the pharapharyngeal space. The vertical component was remarkable by a craniocaudal dimension of 23 mm. As emergency treatment, patient was immediately sedated in the emergency room and intubated to administer oxygen and initiated pharmacological therapy with corticosteroids and antibiotics.

In the following days, the patient saturation improved, and she retrieved consciousness and reported a mandibular injury at 19 years old without surgical treatment, and she complained of dysphagia and dyspnoea. The intervention was proposed to the patient that accepted and further scheduled.

Investigations

• Complete haemogram and routine urine.

• Serum calcium.

• CT.

Treatment

Surgical excision via a left submandibular cervical approach was then decided. Traditional nasotracheal intubation was used; with a minimally invasive incision 2 cm below the jaw line, the mass was identified at the mandibular angle and removed with bur and chisels (figure 1). Histological diagnosis resulted peripheral osteoma. Postoperative hospitalisation required intensive care unit and was uneventful.

Figure 1.

Oral view, asymmetry on the left lingual face of the mandible (A); 3D view of the CT scan, underlying the extention of the mass in the parapharygeal space (B); surgical view (C); 3D view CT-scan 1 year after surgery (D).

Outcome and follow-up

Patient was dischargerd 10 days after surgery, and postoperative ambulatory visits showed complete recovery and correct respiratory functions as reported in figure 2 (PaO₂ 80 mm Hg, PaCO₂ 41 mm Hg at discharge). One year after surgery, CT scan showed no relapse and correct recovery (figure 1).

Figure 2.

Preoperative CT scan with airway compression (A); postoperative CT scan with recovery (B).

Discussion

The growth of space-occupying mass in the pharapharyngeal space is a life-threatening matter, especially in the elderly patients, that requires a clear surgery, possibly a minimally invasive surgery.³

Peripheral osteomas usually have a slow growth. For this reason, diagnosis may be performed when the osteoma becomes symptomatic or may be accidentally evidenced with radiological investigations with other purposes. Small osteoma may present itself with no symptoms for many years, while giant maxillofacial osteomas may disfigure the face or interfere with temporomandibular functions; Ortega Beltrá et al⁴ reported a case of ankylosis of the temporomandibular joint treated with conservative treatment and radiological follow-up; Kashima reported two cases of peripheral osteoma with a significative external deformity: external deformity of the osteoma may lead to an early diagnosis, and usually no other symptoms are reported.⁵ Badauy et al⁶ reported a similar case with a significative lingual expansion of the osteoma.

Tarsitano and Marchetti⁷ reported a case of mandibular osteoma associated with obstructive sleep apnoea syndrome; Sadeghi et al⁸ reported a case of giant osteoma presenting as dyspnoea.

As many mandibular osteoma are reported in international literature, few and small in dimensions are present in the mandibular angle; the mandibular angle is thought to be one of the most common sites for osteoma because of the attachment of muscles.

Due to the giant volume of the neoformation, we decided for a skin access to fully dominate the osteoma and perform a complete radical surgery. At the discharge, the patient presented no signs of dysphagia and the following months recovered well. Kerckhaert et al⁹ reported a similar case with a mandibular enlargment, treated with endoscopical corrective surgery. Endoscopic corrective surgery may be performed for small osteoma or bone exostosis; in case of endoscopic surgery for gigantic osteoma, as the one reported, the intervention may not be sufficient to remove the bone mass. The nature of the mass has a great influence on the management of enlarging tumours; mandibulotomy is currently not advocated for benign tumours, while the transcervical approach is suitable for the safe removal of even large masses in most cases.

Ochiai et al¹⁰ reported the surgery of a peripheral osteoma with piezosurgery; this device my be of use for small osteoma, but for the gigantic ones, this technique requires too much time, and in this submandibular surgery, the time should be spared and care dedicated to soft tissue management.

Many minimally invasive or endoscopic approaches have been reported in literature¹¹; however, in cases of gigantic neoformation and to perform a complete resection, open surgery and intensive care are suggested. Even if osteoma represent a benign neoformation, in cases of huge dimensions, it may be a threat for the patient life, as the case reported. In these cases, surgeon should avoid intraoral interventions and perform an open access and resective surgery; intensive care is advocated.

Patient’s perspective.

I went to the emergency because I couldn’t breathe: the doctors were thorough and I caught my breath after the surgery.

Learning points.

• Do not understimate osteogenic tumour as emergency case.

• For parapharyngeal open surgery, the intensive care is advocated.

• Consider the osteoma in cases of facial asymmetry.

• Histopathological examination after surgery is mandatory.

Ethics statements

Patient consent for publication

Consent obtained directly from patient(s).