Invasive fungal sinusitis by Lasiodiplodia theobromae in an immunocompromised adolescent, a case report and review of the literature

Lasiodiplodia theobromae (L. theobromae) is a phytopathogenic mold, usually causes rotting and dieback in plants. It is infrequently encountered in humans, mainly as keratitis. Rarely, soft tissue infection, pneumonia, osteomyelitis and sinusitis were reported.

We report a 17-year-old female adolescent, diagnosed with acute myeloid leukemia following Hodgkin`s lymphoma who presented with neutropenic fever and left hemifacial pain, swelling and left nasal discharge. Computerized tomography scan to rule out fungal infection of sinuses and lungs revealed thickening of the left maxillary and ethmoidal sinuses` mucosa. Functional endoscopic sinus surgery demonstrated a slightly pale maxillary and ethmoidal moucosa without tissue necrosis. Surgical debridement was performed and biopsies drawn from the sinuses. Intravenous and topical liposomal amphotericin B were initiated pending cultures and molecular tests for invasive fungal infection. Two days later, 28S rDNA revealed positive signal with >99% sequence accordance for L. theobromae and which grew in many cultures. In light of the diagnosis of L. theobromae sinusitis, and based on scarce data, intavenous liposomal amphotericin B was continued beside recurrent surgical debridement. Twelve-days later the patient reported a relief in symptoms, absolute neutrophil count was normalized and subsequent biopsies returned negative. Liposomal amphotericin B was switched to voriconazole. Upon discharge, the patient had fully recovered. She completed 12 weeks of voriconazole with therapuetic blood levels.

L. theobromae sinusitis is a rare manifestation, especially in children. The best treatment protocol and duration should be further evaluated, yet it is reasonable to consider a combination of systemic anti mold azole and surgical debridement.