532 WOral

ASPERGILLOSIS

Maxillofac

49532-534.

AND NECROSIS OF THE MAXILLA

Surg

1991

Aspergillosis

and Necrosis of the Maxilla: A Case Report

JOSEPH A. NAPOLI, DDS, MD,* AND J. OLIVER DONEGAN, MD, FACSt Head and neck aspergillosis, particularly of the maxillary antrum, has been reported as occurring in both healthy and immunologically compromised individuals.’ Aspergillosis occurs in both invasive and noninvasive forms; the former is more likely to occur in patients with debilitating illnesses2 and is a major cause of morbidity and mortality in immunocompromised patients. Schubert et al3 reported a case of fatal hemorrhage secondary to aspergillosis, with the site of infection appearing to be the maxillary sinus. The following case report highlights the important features of diagnosis and management of this infection. In this patient with leukemia, aspergillosis led to hemorrhage from the internal maxillary artery and necrosis of a large part of the maxilla. Case Report A 68-year-old man with recently diagnosed acute myelogenous leukemia was admitted on November 6, 1988, for placement of a Hickman catheter and institution of chemotherapy. He developed spiking fevers and neutropenia, and was treated with broad-spectrum antibiotics. Twelve days after admission, Staphylococcus epidermidis was cultured from the catheter, which was then removed. His fever spikes continued and on his 21st hospital day he developed a diffuse swelling over the right maxilla. Radiographs showed an opacified right maxillary sinus (Fig 1). The swelling over the right maxilla increased and became tender, and the patient began complaining of numbness in the right maxillary teeth. He was noted to have necrosis of the right posterior hard palate and alveolar process (Fig 2) and necrotic debris in the right maxillary mucobuccal fold. Computed tomography (CT) scan revealed opacification of the maxillary sinus

and ethmoids, with surrounding edema extending to Rosenmtiller’s fossa (Fig 3). Platelets were transfused to correct the thrombocytopenia. A biopsy of the palate revealed fungal organisms invading the vasculature (Fig 4). Filamentous organisms consistent with Aspergillus species were cultured. Amphoterocin B therapy was begun (0.5 mg/kg/d), and after 2 days the pain and swelling of the right maxilla decreased. The area of mucosal necrosis of the right posterior maxilla was well-demarcated and was felt to be secondary to thrombosis of the descending palatine artery. The patient’s condition improved with antibiotic therapy, and debridement was deferred in anticipation of bone marrow recovery with return of normal platelet and white blood cell counts. However, 8 weeks after admission, he began to bleed profusely from the oral cavity. He was taken to the operating room where he underwent debridement of necrotic soft tissue from the right posterior maxillary buccal vestibule, right palate, and maxillary sinus, as well as packing of the pterygomaxillary fossa. Hemostasis was achieved and a tracheostomy was performed. Blood loss was measured at 600 mL. The packing was removed in the operating room on the fifth postoperative day and no further bleeding occurred. Six days later, more extensive debridement, including removal of necrotic bone, was done. The posterior maxilla was approached via a Weber-Fergusson incision. The necrotic bone was clearly demarcated from viable bone and was removed en bloc. The tissue removed included the right posterior maxilla from the tuberosity to the premolar region and extended from the midpalate to the zygomatic buttress laterally and superiorly to the infraorbital foramen (Fig 5). Fungal invasion of the soft tissues attached to the bone was histologically evident. The wound healed uneventfully, and the patient developed no further evidence of aspergillosis. Amphotericin therapy was discontinued 4 weeks after surgery. A prosthesis was used to replace the posterior maxilla.

Discussion Received from the Department of Surgery, DartmouthHitchcock Medical Center, Hanover, NH. * Section of Plastic and Reconstructive Surgery, Oral and Maxillofacial Surgery. t Section of Otolaryngology and Audiology. Address correspondence and reprint requests to Dr Napoli: Section of Plastic and Reconstructive Surgery, Oral and Maxillofacial Surgery, Department of Surgery, Dartmouth-Hitchcock Medical Center, 2 Maynard Street, Hanover, NH 03756. 0 1991 American geons

Association

0278-2391191/4905-0017$3.00/0

of Oral and Maxillofacial

Sur-

The spores of Aspergillus species are ubiquitous and commonly enter the body via the respiratory tract. Aspergilfus fumigatus is the most common infecting species followed by Aspergillus niger and Aspergillus flavus. Under certain conditions, Aspergillus species may become pathogenic in otherwise healthy individuals. Local factors, such as sinus obstruction, may promote infection and growth of Aspergillus species is also favored by hypoxia

NAPOLI AND DONEGAN

FIGURE I. Waters’ view radiograph showing opacification of the right maxillary sinus.

and anaerobic conditions.’ Beck-Mannagetta et al indicated that antral aspergillosis can be induced by the presence of endodontic filling material, citing experimental evidence that revealed growth acceleration of Aspergillus in the presence of zinc oxide, a common component of root canal tilling material.6 In the majority of cases the infection is opportunistic, occurring in patients with debilitating illness, particularly if the disease or its treatment results in granulocytopenia.3 In addition to the paranasal sinuses, other sites of infection within the head and neck region include the ear and orbit. When in-

FIGURE 2. of necrosis.

View of palate showing sharply demarcated region

533

FIGURE 3. Computed tomography scan showing soft-tissue density within right maxillary sinus and swelling of the overlying tissue.

volvement of the paranasal sinuses occurs, signs and symptoms are nonspecific. They may include mucoid or purulent nasal drainage, fever, pain, facial swelling, and nasal obstruction, unilateral proptosis, sensory nerve disturbances, and epistaxis.“5 Immunocompromised patients may present difftculty in diagnosis because of the presence of concomitant infections and their impaired immune response. Because of the propensity for airway involvement, evidence of pulmonary infection should be sought. Radiologic findings also are nonspecific. The

FIGURE 4. Photomicrograph of palatal biopsy specimen showing fungal organisms (hematoxylin-eosin, original magnification X400).

ASPERGILLOSIS

AND NECROSIS OF THE MAXILLA

available sooner. Vascular invasion by fungal organisms is a characteristic histologic feature. Management involves the use of systemic antifungal agents and surgery, ie drainage and debridement. Schubert et al3 reported four cases in patients undergoing bone marrow transplantation. Two cases were managed successfully with antibiotics and surgery and one case with antibiotics alone. The fourth case resulted in fatal hemorrhage despite combined treatment. A case of aspergillosis associated with AIDS has also been reported.* Initial improvement with medical therapy does not obviate the need for surgical care; local vascular invasion can progress and result in hemorrhage, as demonstrated in this case. Tolerance of surgery and ability to recover from the infection will be determined by the patient’s medical condition and hematologic profile. References

FIGURE 5.

Surgical specimen containing first molar.

usual finding is a unilateral homogeneous sinus opacity. An intraluminal soft-tissue mass may also be noted. Evidence of bone destruction is not common. In a restrospective study of 142 cases of aspergillosis of the paranasal sinuses, Kopp et al reported that 58% demonstrated the presence of radiopaque structures that consisted of tertiary calcium phosphate, calcium sulfate, and heavy metal salts.7 This feature was not present in our patient. Histologic and microbiologic data are needed for definitive diagnosis, the former generally being

1. Meikle D, Yarington CT, Winterbauer RV: Aspergillosis of the maxillary sinuses in otherwise heathy patients. Laryngoscope 95776, 1985 2. Gonty AA, Page LR: Aspergillosis of the maxillary sinus: Review of the literature and a report of a case. Oral Surg 43:350, 1977 3. Schubert MM, Peterson DE, Meyers JD, et al: Head and neck aspergillosis in patients undergoing bone marrow transfusion: Report of four cases and review of the literature. Cancer 57: 1092, 1986 4. Roser SM, Canalis RF, Hanna CJ: Aspergillosis of the maxillary antrum. J Oral Med 31:91, 1976 5. Babajews A: Aspergillosis mycetoma of the maxillary annum. Br J Oral Surg 20:299, 1982 6. Beck-Mannagetta J, Necek D: Radiologic findings in aspergillosis of the maxillary sinus. Oral Surg 62:345, 1986 7. Kopp W, Fotter R, Ebner F, et al: Radiologic aspects of aspergillosis in the paranasal sinuses. Eur J Radio1 6: 178, 1986 8. Rubin MM, Jui V, Sadoff RS: Oral aspergillosis in a patient with acquired immunodeficiency syndrome. J Oral Maxillofac Surg 48997, 1990