A published case report describes a rare presentation of Aspergillus osteomyelitis of the mandible in a 13-year-old boy with underlying hematologic malignancy, underscoring the ongoing diagnostic and therapeutic challenges posed by invasive fungal disease in high-risk pediatric populations.
Reporting in SAGE Pathology, clinicians from Farhat Hached University Hospital in Sousse, Tunisia, noted that aspergillosis primarily affects the respiratory tract. Extrapulmonary involvement—especially bony infection of the jaws—is rare, representing only about 18% of reported Aspergillus osteomyelitis cases involving maxillofacial structures. Mandibular involvement is even less frequent, they stated, typically arising in severely immunocompromised individuals.
The reported patient had a history of juvenile idiopathic arthritis complicated by B-cell lymphoma, treated initially with European Organization for Research and Treatment for Cancer Average Risk 2 protocol chemotherapy. His clinical course included probable invasive pulmonary aspergillosis, managed successfully with voriconazole. Following marrow relapse and salvage therapy with FLAG-Daunorubicin, the patient developed febrile neutropenia and progressive swelling of the left mandible.
Imaging findings revealed submandibular inflammatory changes, myositis, and ultimately cortical destruction of teeth 36–37 with a vestibular sequestrum visible on Cone Beam CT. Intraoral examination revealed grayish ischemic mucosa and exposed necrotic bone. These features initially raised concern for neutropenic ulceration or bacterial osteomyelitis, though the patient’s immunosuppressed state prompted suspicion for fungal infection.
Surgical debridement yielded a 1.5-cm sequestrum. Histopathologic evaluation confirmed invasive fungal osteomyelitis, revealing septate, branching hyphae consistent with Aspergillus, highlighted by Periodic Acid-Schiff (PAS) and Grocott’s methenamine silver (GMS) staining. Notably, fungal elements were identified only within the sequestrum, consistent with chronic localized infection rather than diffuse mandibular invasion.
The patient continued antifungal treatment using voriconazole, which had already been started as prevention, along with systemic antibiotics and local care measures. Mucosal healing was seen 15 days after the surgery. The area had fully closed, and there were no signs of ongoing infection, indicating that combined medical and surgical treatment was successful.
The authors emphasize that oral aspergillosis progresses through early mucosal discoloration and necrosis before advancing to bone destruction and perforation, often mimicking more common conditions such as bacterial osteomyelitis, mucormycosis, malignancy, or neutropenic ulcers. Therefore, getting a biopsy and culture as soon as possible is key to making a definitive diagnosis, particularly when initial imaging reveals sequestration or progressive cortical loss.
According to the authors, only six prior cases of mandibular aspergillus osteomyelitis have been reported in the literature, ranging from immunocompetent adults to children undergoing chemotherapy. Gingival necrosis or bone exposure is common, often with concurrent pulmonary aspergillosis in immunocompromised hosts.
The report also raises questions about antifungal prophylaxis efficacy, noting that significant mandibular involvement developed despite ongoing voriconazole. The authors emphasize the need for further evaluation of prophylactic strategies, especially in pediatric patients with profound, recurrent immunosuppression.
“Early diagnosis and the prompt initiation of aggressive antifungal therapy and surgical debridement are crucial for favorable outcomes,” wrote the authors, who reported having no conflicts. “This approach has demonstrated greater efficacy in eradicating infection and is associated with a lower relapse rate, aligning with current clinical guidelines and corroborating the findings of this case report.”
Source: Sage Open Pathology
