Invasive maxillary aspergillosis in a patient with systemic lupus erythematosus: Case report

Abstract

Introduction Invasive aspergillosis (IA) is a fungal infection caused by Aspergillus species (spp.). Aspergillosis is the most common source of opportunistic fungal infection in humans. IA can cause serious complications related to high morbidity and mortality in immunocompromised patients. Presentation of case We report a case of a 22-year-old female with a chief complaint of having a hole in the roof of her mouth. She was diagnosed with SLE in 2009. She had been consuming oral methylprednisolone ever since. In 2018, she experienced worsened symptoms and was hospitalized. She experienced swelling and bleeding of her gums and some of her teeth becoming loose and falling out, and then developing a hole in the roof of her mouth. Subsequently, she was treated with oral cyclophosphamide, oral mycophenolate sodium, and oral fluconazole. She was asked to stop taking oral methylprednisolone. In 2019, the palate biopsy was performed and showed Aspergillus spp. invading the palate. Afterward, the patient was referred to our clinic for defect closure. The patient was operated on for debridement and reconstruction of the defect. There was no recurrence of the defect or complications observed in the follow-up. The patient was satisfied with the surgical results. Discussion IA is a destructive and potentially harmful opportunistic fungal infection and treatments with surgical interventions should be well-thought-out in immunocompromised patients. Conclusion The management of IA are controlling any underlying diseases and surgical debridement or necrotomy. Generally, antifungal therapy and prompt surgical intervention are successful in managing invasive aspergillosis.

Appropriate Total Rehabilitation in the Bilateral Maxillectomy Defected Diabetic Patient Secondary to Aggressive Aspergillosis

“…The surgery for aggressive aspergillosis should be aggressive, involving not only the necrotic tissue, but also the surrounding healthy-looking tissues considering the rapid extension of infection by the Aspergillus hyphae. 7 Systemic therapy itself is not sufficient as it may not completely eradicate the fungal organisms. Maxillectomy is one of the aggressive surgery techniques that is often applied with or without orbital exenteration in aggressive fungal infection management.…”

“…Direct microscopy of specimens with Blankophor and Calcofluor is preferred. Hematoxylin eosin staining can help in identifying the fungal elements, and PAS/GMS are important to highlight the hyphae 7,12 . From CT findings, Slonimsky et al 21 showed that mucormycosis is associated with bilateral mucosal thickening of nasal and maxillary sinuses with bony dehiscence, and aspergillosis is associated with unilateral maxillary sinus mucosal thickening without bony dehiscence.…”

“…The third step is complete removal of all infected tissue and postoperative routine follow-up. The surgery for aggressive aspergillosis should be aggressive, involving not only the necrotic tissue, but also the surrounding healthy-looking tissues considering the rapid extension of infection by the Aspergillus hyphae 7 . Systemic therapy itself is not sufficient as it may not completely eradicate the fungal organisms.…”

“…Hematoxylin eosin staining can help in identifying the fungal elements, and PAS/GMS are important to highlight the hyphae. 7,12 From CT findings, Slonimsky et al 21 showed that mucormycosis is associated with bilateral mucosal thickening of nasal and maxillary sinuses with bony dehiscence, and aspergillosis is associated with unilateral maxillary sinus mucosal thickening without bony dehiscence. However, in aggressive aspergillosis, bone erosion or destruction, opacification, soft tissue mass, and necrosis are commonly identified in CT. 7 To confirm the diagnosis of aggressive aspergillosis, CT and histopathology examinations should be performed together.…”

“…5,6 Aggressive aspergillosis appears in the maxilla as a painful necrotic destructive lesion and has a yellow or grey slough. 7 Due to the rarity of this entity, aggressive aspergillosis may pose non-specific symptoms similar to other pathologic diseases, leading to difficulties in establishing a definitive diagnosis and appropriate treatment protocol. Early diagnosis and prompt treatment are essential in improving the prognosis and preventing mortality of this disease.…”