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A Complex Case of Rhinocerebral Zygomycosis: Challenges in Diagnosis and Treatment

This case study explores an 82-year-old male with a history of low-grade B-cell lymphoma, experiencing peri-orbital cellulitis that complicated into a life-threatening condition. Initially treated with antibiotics, the patient's condition worsened, displaying signs of cerebral involvement and leading to the diagnosis of rhinocerebral zygomycosis. Despite aggressive medical intervention, including surgical debridement and antifungal therapy, the prognosis remained grave due to the disease's invasive nature and the patient's immunocompromised status.

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A Complex Case of Rhinocerebral Zygomycosis: Challenges in Diagnosis and Treatment

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  1. Teaching Case of the Week Dr. W. A. Ciccotelli Sept 14, 2005

  2. The Patient • 82 y M • Past Hx • Low grade B cell lymphoma • Pancytopenia/transfusion dependent • Interstitial lung dz • HTN • Ex-smoker

  3. The Patient • Meds • Amlodipine • Prednisone (taperingx 4 mos) • NKDA • 2-4x EtOH/wk

  4. The Case • Referred to ID for peri-orbital cellulitis • 3 day Hx of progressive • R eye swelling • R frontal headache • Reactive clear discharge • FB sensation • No fever/chills • No other ocular symptoms • Vision ok • On cefotaxime 36 hrs

  5. The Case • Afebrile, VSS • Peri-orbital cellulitis • R eye proptosis, mild ptosis, chemosis • Loss of EOM R eye • CNs normal otherwise • Visual acuity normal

  6. The Case • WBC 4.9, Hgb 99, plts 54, grans 1.7 • Lytes N • Cr 123 • TSH 1.1 • Panculture neg • CXR: unchanged chronic interstitial pattern

  7. The Case • CT scan head • R pre-septal edema • Minimal proptosis R eye • R Maxillary & ethmoidal sinusitis • R nasal septum deviation • No bony lesions • No retro-orbital masses • ENT consulted

  8. The Case • Not responding on Cefotaxime • Febrile • New diplopia • Worsening peri-orbital cellulitis

  9. The Case • Abx changed to Clinda/Cipro • MRI head • Small fluid collection lat. R eye ?abscess • Maxillary & ethmoid sinusitis (L & R) • Meninges inflammatory changes in R middle cranial fossa • No cavernous vein thrombosis • Nasal culture: commensal flora

  10. The Case • Now really bad! • Delirious • Febrile • Clonus in lower ext. • R Facial droop

  11. The Case • Urgent ethmoidectomy • necrotic sinus • painless procedure • LP aseptic meningitis • ANCAs neg • Lipo Ampho B started 5 mg/kg/day

  12. The Case • Repeat MRI • Early cerebritis R temporal operculum • Ongoing inflammatory changes of all sinuses • Inflammatory changes around R orbit, masticator space, cavernous sinus

  13. Case Resolution • Further CNS deterioration • Sinus Bx • Broad ribbon like non-septate fungal filament on microscopy • ZN & PAS stains confirm non-septate hyphae • Dx of Rhinocerebral zygomycosis • Lipo Ampho B to 10 mg/kg/day • Family withdrew care given degree of surgery needed

  14. Zygomycosis • Mucorales order • Ubiquitous in environment • Thick walled non-septate hyphae with right angle branching • Rare & mimics other invasive mould infections • Inherent resistance to antifungal agents • Angioinvasive disease

  15. Zygomycosis • Multiple clinical forms • Cutaneous • Pulmonary • Gastrointestinal • Rhinocerebral • Sino-orbital • Disseminated • Direct inoculation, inhalation, ingestion of spores

  16. Zygomycosis • Immunocompromised state hallmarks • DM ketoacidosis • Neutropenia • Chemotherapy • BMT patients • Lymphoma/leukemia • Trauma with exposure to contaminated soil

  17. Zygomycosis • Dx is difficult & delayed • Poor recovery from culture • Non specific presentation • Not on everyone’s DDx • Mimics other invasive molds (Aspergillus) • Dx generally made with invasive testing for histopathological sampling • Dx commonly made at autopsy • Yet increasingly problematic in Heme-Onc patients over 1990s

  18. Zygomycosis • Treatment is multifaceted • Immune reconstitution • Aggressive surgical debridement • Ampho B • Prayer • Posaconazole as oral alternative • Despite this still highly fatal (mortality 50-80%)

  19. Zygomycosis • Prognosis is poor • Late Dx • Not able to recover immune system • Disseminated • Death usually from hemorrhage • Best prognosis • Limited disease • Early surgery • Non Heme-Onc patients

  20. Zygomycosis • Tip offs • Right patient population (esp neutropenia) • Unexplained thrombosis • Necrotic eschar • Unexplained hemorrhage • Common clinical situations • Culture neg despite real disease • Not responsing to reasonable Abx

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