J Neurocrit Care.  2017 Dec;10(2):122-125. 10.18700/jnc.170018.

Paradoxical Response after a Voriconazole Treatment in an Immunocompetent Host with a Skull Base Osteomyelitis due to Invasive Aspergillosis

• Kim JM
• Jung HB
• Bae JH
• Choi SH
• Lee TJ

• KMID: 2405768
• DOI: http://doi.org/10.18700/jnc.170018

Abstract

BACKGROUND
We describe a case of skull base osteomyelitis due to invasive aspergillosis which had been aggravated after antifungal treatment but significantly recovered by dexamethasone.
CASE REPORT
A 74-year-old male patient presented to neurology clinic complaining of sudden onset right-sided facial palsy and headache. Brain magnetic resonance imaging (MRI) and sphenoid sinus biopsy confirmed Aspergillus infection of skull base. He was treated with voriconazole for two months, but his headache was not relieved, and he additionally complained of vertigo and dysphagia. A subsequent MRI showed reduced enhancement of initial lesions, but increased thickness of surrounding dura mater. With an impression of paradoxical inflammatory response after antifungal treatment, parenteral dexamethasone was administered for one month while maintaining voriconazole. His symptoms improved thereafter.
CONCLUSION
A paradoxical inflammatory response during antifungal treatment in the skull base aspergillosis aggravates the neurological symptom by thickening the dura mater, which can be recovered by dexamethasone.

Keyword

Neuroaspergillosis; Central nervous system infections

MeSH Terms

Aged
Aspergillosis*
Aspergillus
Biopsy
Brain
Central Nervous System Infections
Deglutition Disorders
Dexamethasone
Dura Mater
Facial Paralysis
Headache
Humans
Magnetic Resonance Imaging
Male
Neuroaspergillosis
Neurology
Osteomyelitis*
Skull Base*
Skull*
Sphenoid Sinus
Vertigo
Voriconazole*
Dexamethasone
Voriconazole

Figure

• 

  Figure 1. Brain magnetic resonance imaging (MRI) and pathological analysis findings. Initial brain MRI showed a diffuse enhancement of bony structures including the clivus and basis of occiput and sphenoid, as well as the posterior fossa dura, bilateral jugular foramen, carotid canals, pterygoid muscles and right sphenoid sinus (A). Hematoxylin and Eosin staining of mucosal specimen (magnification, ×40) obtained from right sphenoid sinus revealed a large amount of non-pigmented septated hyphae with acute angle branching infiltrating mucosal layer, disclosing Aspergillus infection (B). A brain MRI performed two months after voriconazle treatment showed a decreased extent of the enhancement, but an increased thickness of the dura mater (C, arrowheads). After a combination of dexamethasone, brain MRI followed after one month showed a markedly decreased dural thickness (D).

Reference

1. Walsh TJ, Anaissie EJ, Denning DW, Herbrecht R, Kontoyiannis DP, Marr KA, et al. Treatment of aspergillosis: clinical practice guidelines of the Infectious Diseases Society of America. Clin Infect Dis. 2008; 46:327–60.

2. Thwaites GE, Nguyen DB, Nguyen HD, Hoang TQ, Do TT, Nguyen TC, et al. Dexamethasone for the treatment of tuberculous meningitis in adolescents and adults. N Engl J Med. 2004; 351:1741–51.

3. Nguyen TH, Tran TH, Thwaites G, Ly VC, Dinh XS, Ho Dang TN, et al. Dexamethasone in Vietnamese adolescents and adults with bacterial meningitis. N Engl J Med. 2007; 357:2431–40.

4. Legris T, Massad M, Purgus R, Vacher-Coponat H, Ranque S, Girard N, et al. Immune reconstitution inflammatory syndrome mimicking relapsing cryptococcal meningitis in a renal transplant recipient. Transpl Infect Dis. 2011; 13:303–8.

5. Pushker N, Meel R, Kashyap S, Bajaj MS, Sen S. Invasive aspergillosis of orbit in immunocompetent patients: treatment and outcome. Ophthalmology. 2011; 118:1886–91.

6. Miceli MH, Maertens J, Buvé K, Grazziutti M, Woods G, Rahman M, et al. Immune reconstitution inflammatory syndrome in cancer patients with pulmonary aspergillosis recovering from neutropenia: proof of principle, description, and clinical and research implications. Cancer. 2007; 110:112–20.

7. Jung J, Hong HL, Lee SO, Choi SH, Kim YS, Woo JH, et al. Immune reconstitution inflammatory syndrome in neutropenic patients with invasive pulmonary aspergillosis. J Infect. 2015; 70:659–67.