Post-traumatic near-complete aseptic necrosis of the maxilla: a case report and review of the literature
Osteonecrosis of the mandible and the maxilla is known; however, aseptic necrosis of the maxilla after traumatic fracture is hardly reported. This case aims to help clinicians realize the need to closely follow up and treat such patients with trauma as it can lead to osteonecrosis of the bone and cause difficulty in daily activities. We have also carried out a thorough review of the literature to give a concise understanding of all disease processes that may lead to osteonecrosis of the maxilla. Such an article has not previously been published.
Post-traumatic necrosis of the maxilla is rare.1 Only two case reports have been published describing necrosis of the maxilla after traumatic fracture.1,2 Osteonecrosis in the maxilla or anywhere else in the body develops when the vascularity is compromised. This may occur from a traumatic fracture; however, it is also a known complication after surgical osteotomy.3,4 Other known causes include infections, tumours and radiation.5-9 Recently, bisphosphonate-associated osteonecrosis has been reported as an emerging oral complication in cancer patients.10-13 We report a case of aseptic necrosis of the maxilla after mid-face trauma.
A 38-year-old male with no known comorbidities presented to the maxillofacial dental clinic with difficulty in chewing on the right side of his jaw. He complained that he was able to freely move a portion of his right upper gum, including the teeth, separately from the rest of the jaw. This caused difficulty in chewing. He had no other complaints. He had been in a road traffic accident 2 months previously where he was sitting in a car in the passenger seat without a seatbelt and was struck against the dashboard. He received first aid from a nearby primary healthcare centre and had, among other injuries, trauma to his mid-face, which on examination was diagnosed as a fracture of the right maxilla. He was advised to see a maxillofacial or dental surgeon but, as the bruising and pain over his cheek subsided without any difficulty and he recovered from his injuries without any deformity, he did not follow this advice. He did not have an infection in the cheek and felt completely well. However, in the last few weeks he started noticing the abnormal mobility of his jaw.
A non-contrast CT scan of the face was performed which showed near-complete necrosis of the right maxillary bone. The body of the maxilla with the maxillary sinus, the alveolar process, zygomatic process and palatine process were completely absent (Figure 1a–d). Only the most superior part of the right maxilla above the maxillary sinus, which included the infraorbital foramen, was present (Figure 1c) and the frontal process of the maxilla was intact; these were forming the rim and walls of the orbit. Only a fine lace-like remnant of bone was seen along the outermost margins of the maxilla. The absence of alveolar processes gave the appearance of a floating upper right incisor and canine and molar teeth (Figure 1a–d).
No soft-tissue swelling or mass was present and no sclerosis, fracture or evidence of previous surgery was visible. On the basis of these findings a diagnosis of aseptic necrosis of the maxilla was made. He was referred to the maxillofacial clinic where he was prescribed prophylactic antibiotics. A maxillary necrosectomy with maxillary reconstruction was offered; however, at the last follow-up he still had not undergone surgery.
To the best of our knowledge, osteonecrosis of the maxilla from traumatic fracture has only previously been reported twice.1,2 In one case described by Cornah and O'Hare,1 the patient had extensive maxillary fractures associated with Le Fort I and II-type fractures. This patient also developed maxillary infection. The maxilla was excised and appeared to have undergone complete necrosis.1 In the other case, the patient sustained a Le Fort I-type fracture that was inadequately treated and resulted in a large maxillary defect.2
Osteonecrosis of the maxilla occurs when the vascularity is compromised.14 The vascular supply of the maxilla arises from the descending palatine artery and internal maxillary artery.14 There are a few known conditions that can affect this vascularity resulting in osteonecrosis of the maxilla. The causes can be broadly classified into septic and aseptic necrosis. Septic osteonecrosis can occur from a variety of infections and aseptic necrosis includes malignancy, radiation, trauma, surgery and bisphosphonates therapy.5-9
Septic necrosis of the maxilla can be caused by fungal infections such as mucormycosis and aspergillosis.5,6 Viral infections such as herpes zoster7,8,15 affect immunocompromised patients, including those who have uncontrolled diabetes, lymphomas, organ transplants and long-term corticosteroid and immunosuppressive therapy.5,6 Infections such as mucormycosis form thrombi within the blood vessels and result in decreased blood supply and necrosis.5 Osteonecrosis from herpes occurs when the varicella zoster virus reactivates from latency and becomes an active viral replication. Our patient did not have any signs or symptoms of infections in his post-traumatic period and he was not immunocompromised.
Malignancy and radiation in the region of the maxillary sinus can result in aseptic necrosis of the maxilla. Malignancy, such as squamous cell carcinoma of the maxillary sinus, can cause direct destruction of the maxilla and also occlude the vasculature, resulting in osteonecrosis.14 Radiation of tumours in the head and neck region can also result in osteonecrosis, which is considered as the worst long-term complication of radiotherapy.9,12
Le Fort I osteotomy has become a routine procedure in elective orthognathic surgery.4 Although rare, ischaemia is one of the known complications of this procedure. In a study involving 1000 patients with Le Fort I osteotomy, ischaemic complications affected 10 patients, of whom 2 (0.2%) experienced partial aseptic necrosis of the alveolar process.4 One theory suggests that the blood flow to the maxilla after osteotomy is reduced, especially in diabetic patients, which may be a cause of necrosis in osteotomy patients.16
Recently, there have been reports in medical literature supporting the association between bisphosphonate use and osteonecrosis.11 This is most commonly reported in patients who are receiving bisphosphonates for metastatic disease.11 Bisphosphonates inhibit resorption by osteoclasts and cause induction of apoptosis.17 The mandible is the most common bone affected followed by the maxilla.11 Osteonecrosis in patients taking bisphosphonate can show focal lytic changes on conventional radiographs and CT.11 A positive association is seen between osteonecrosis and length of intravenous bisphosphonate therapy, oral hygiene and dental extraction.11-13
Lesser-known causes of osteonecrosis include Wegener's granulomatosis, which is an inflammatory disease affecting blood vessels.5 Acute necrotizing ulcerative gingivitis arising from the gingiva can involve the maxilla,5 while Gorham's disease is another rare disorder of unknown aetiology that results in destruction and resorption of bone from proliferation of non-neolastic vascular or lymphamatous tissue.18
Surgical reconstruction of the maxilla is a daunting task for the surgeon because it needs to include the restoration of the original form and function that has been lost.19 The masticatory function, the resonance of the voice and the architecture of the face has to be restored so that the quality of life after the operation is optimal.19 Many local flaps have been described which achieve soft-tissue closure of the defect. Local flaps of the palatal mucosa from the contralateral side or ipsilateral cheek mucosa can be used to close small defects. Pedical regional flaps are used for larger defects; the most common is the ipsilateral temporalis myofascial flap. Microvascular free-tissue transfer from the radical forearm flap and the latissimus dorsi flaps have also been advocated, but they rarely provide a functional solution. Osseous reconstruction of the maxilla has been attempted with little success.20 Titanium mesh implants have also been tried as carriers for osseous tissue and the bone can then be used to place dental osseointegrated implants.20 The reconstruction of the maxilla and the surrounding tissues using a prosthetic maxillary obturator device fabricated to include the lost dental arch still remains the method of choice because it achieves the best possible results compared with the other methods.20
Trauma to the face is common in road traffic accidents. Mid-face trauma can result in fractures of the maxilla. Such injuries should be properly managed given that they can result in osteonecrosis of the maxilla. Although rare, it should not be taken lightly as it can lead to significant anxiety for the patient and necessitate major surgery to fill the defect.
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