Survival after rhino-orbital-cerebral mucormycosis in an immunocompetent patient

Ophthalmology. 2000 Mar;107(3):555-8. doi: 10.1016/s0161-6420(99)00142-6.

Abstract

Objective: Rhino-orbital-cerebral mucormycosis is usually associated with a poor prognosis and is almost exclusively seen in immunocompromised patients. We report the third documented case of rhino-orbital-cerebral mucormycosis caused by Apophysomyces elegans (a new genus of the family Mucoraceae first isolated in 1979) in an immunocompetent individual. Orbital exenteration and radical debridement of involved adjacent structures combined with intravenous liposomal amphotericin resulted in patient survival.

Design: Interventional case report.

Method: A 59-year-old immunocompetent white man sustained a high-pressure water jet injury to the right inner canthus while cleaning an air conditioner filter. He later had "orbital cellulitis" develop that did not respond to antibiotics and progressed to orbital infarction. Imaging studies and biopsy results led to a diagnosis of mucormycosis. Tissue culture grew Apophysomyces elegans, a new genus of the family Mucoraceae first isolated in 1979. Orbital exenteration and radical debridement of involved adjacent structures, combined with intravenous liposomal amphotericin, resulted in patient survival.

Results: After orbital exenteration and debridement of involved adjacent structures along with intravenous liposomal amphotericin, our patient has remained free from relapse with long-term follow-up.

Conclusions: The agent causing this case of rhino-orbital-cerebral mucormycosis (Apophysomyces elegans) contrasts with the three genera most commonly responsible for mucormycosis (Rhizopus, Mucor, and Absidia) in that infections with this agent tend to occur in warm climates, by means of traumatic inoculation, and in immunocompetent patients. Rhino-orbital-cerebral mucormycosis should be considered in all patients with orbital inflammation associated with multiple cranial nerve palsies and retinal or orbital infarction, regardless of their immunologic status. A team approach to management is recommended for early, appropriate surgery and systemic antifungal agents.

Publication types

  • Case Reports

MeSH terms

  • Amphotericin B / therapeutic use
  • Brain Diseases / diagnosis
  • Brain Diseases / microbiology*
  • Brain Diseases / therapy
  • Debridement
  • Eye Infections, Fungal* / diagnosis
  • Eye Infections, Fungal* / microbiology
  • Eye Infections, Fungal* / therapy
  • Humans
  • Immunocompetence
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Mucorales / isolation & purification*
  • Mucormycosis* / diagnosis
  • Mucormycosis* / microbiology
  • Mucormycosis* / therapy
  • Nose Diseases / diagnosis
  • Nose Diseases / microbiology*
  • Nose Diseases / therapy
  • Orbit Evisceration
  • Orbital Diseases / diagnosis
  • Orbital Diseases / microbiology*
  • Orbital Diseases / therapy
  • Tomography, X-Ray Computed

Substances

  • Amphotericin B