A 52-year old woman was admitted to the hospital because of headache, blurring of vision and fever. She had a history of living unrelated renal transplantation 6 years ago. Her immunosuppressive drugs consist of cyclosporine and prednisolone. There is no episode of rejection or immunosuppressive intensification for the last two years. There was history of post transplant Diabetes Mellitus since 5 years ago that was controlled with insulin. Physical examination on admission was a low grade fever, periorbital edema and erythema and chemosis and tenderness of maxillary bone suggesting orbital cellulitis. CT scan of sinuses and orbit disclosed sinusitis and soft tissue infiltration and edema and destruction of anterior bony wall of right maxillary sinus. Deep orbital structures were intact in first CT scan. Brain CT scan was normal. The presentation of patient progressed despite antibiotic therapy and a red tender nodule below the right eye appeared. Immunosuppressive drugs except prednisolone were discontinued. In examinations of nasal mucosa by nasal endoscope, there was severe erythematous and fragile bleeding mucosa. Biopsy was taken from inferior turbinate and septal mucosa. The pathologic results were nonspecific inflammation. The right eye became fixed within the orbit and there was near complete loss of vision and the patient admitted to ICU due to respiratory distress on third day of hospitalization. Another attempt to obtain tissue was done by sublabial Caldwell incision. Sinus mucosa and skin granulation tissue were sent to pathology. Drainage was established by inferior entrostomy. The pathologic review of specimens disclosed invasion by nonseptate hyphae fungi that was compatible with invasive mucormycosis. Amphotericin B was administered with a rapid escalating dose to 1.2 mg/kg and radical surgery was planned. The patient was not compliant with evacuation of orbit. Radical debridement was performed by Weber Fergusson incision and lip splitting. By extending the incision to lateral canthus, skin flap was elevated. Anterior bony wall of Maxilla excised up to inferior rim of orbit where the bone seems uninvolved. Ascending process of Maxilla and ethmoidal sinus mucosa were excised. Dissection continued to Sphenoid sinus. Superiorly, medial canthus and nasal root were excised. Involved skin with safe margins was excised and repair was performed by flap advancement of skin from neck and forehead. Despite surgical debridement and continued amphotericin B with doses more than 1mg/kg for more than 5 weeks, the disease progressed. Her allograft function remained stable with a serum creatinine around 1.6 mg/dl despite discontinued immunosuppressive drugs and administration of more than 2 gram amphotericin B. The patient refused more extensive surgical excisions and left the hospital for a more peaceful death. Teaching point: By reviewing the literature, a dozen of mucormycosis were found in renal transplant patients. Rhinocerebral and other forms of mucormycosis must be considered in immunosuppressed especially diabetics that warrants early and extensive surgical excisions.
Volume : 6
Issue : 4
Pages : 180
Department of Internal Medicine, Division of Nephrology, Urmia University of Medical Sciences, Urmia, Iran