Fungal Sphenoiditis in a Patient With ANCA-associated Vasculitis

Cover Page


Cite item

Full Text

Open Access Open Access
Restricted Access Access granted
Restricted Access Subscription or Fee Access

Abstract

Fungal sinusitis comprises a heterogeneous group of diseases differing in etiology, clinical presentation, and pathogenesis. One such form is a mycetoma, a noninvasive form characterized by the accumulation of fungal hyphae and debris within a sinus cavity, without invasion of the mucosa. Isolated sphenoid sinus mycetoma is relatively rare, and its pathophysiology remains incompletely understood. The patient’s immune status plays a critical role in the pathogenesis of fungal sinusitis, as immunosuppression is a major risk factor for the transformation of noninvasive mycetoma into an invasive form, potentially resulting in severe complications. Therefore, immune status assessment in patients with noninvasive fungal sinusitis, particularly in the presence of comorbidities and immunosuppressive therapy, is essential for determining optimal treatment strategies. This article presents a clinical case of fungal sphenoiditis in a female patient with granulomatosis with polyangiitis (ANCA-associated vasculitis) undergoing immunosuppressive therapy. The case is notable for the combination of noninvasive fungal sphenoiditis with a high risk of invasive disease due to immunosuppression. Given the elevated risk, the patient underwent endoscopic sphenoethmoidectomy followed by systemic antifungal therapy. This case highlights the importance of early surgical intervention and appropriate systemic antifungal treatment in preventing infection progression and complications in patients with noninvasive fungal sinusitis, especially in the context of immunosuppression.

Full Text

Restricted Access

About the authors

Maria A. Shumikhina

City Clinical Hospital No. 52

Author for correspondence.
Email: masha_myxa@mail.ru
ORCID iD: 0009-0001-1557-0220

MD

Russian Federation, Moscow

Tatyana A. Shirokikh

City Clinical Hospital No. 52

Email: tshirokih83@yandex.ru
ORCID iD: 0009-0000-1360-3992

MD

Russian Federation, Moscow

Pavel V. Azarov

City Clinical Hospital No. 52

Email: azarovp@mail.ru
ORCID iD: 0009-0004-7408-7847

MD

Russian Federation, Moscow

References

  1. deShazo RD, O’Brien M, Chapin K, et al. A new classification and diagnostic criteria for invasive fungal sinusitis. Arch Otolaryngol Head Neck Surg. 1997;123(11):1181–1188. doi: 10.1001/archotol.1997.01900110031005
  2. deShazo RD, Chapin K, Swain RE. Fungal sinusitis. N Engl J Med. 1997;337(4):254–259. doi: 10.1056/NEJM199707243370407
  3. Ferguson BJ. Definitions of fungal rhinosinusitis. Otolaryngol Clin North Am. 2000;33(2):227–235. doi: 10.1016/s0030-6665(00)80002-x
  4. Nicolai P, Lombardi D, Tomenzoli D, et al. Fungus ball of the paranasal sinuses: experience in 160 patients treated with endoscopic surgery. Laryngoscope. 2009;119(11):2275–2279. doi: 10.1002/lary.20578
  5. Willinger B, Obradovic A, Selitsch B, et al. Detection and identification of fungi from fungus balls of the maxillary sinus by molecular techniques. J Clin Microbiol. 2003;41(2):581–585. doi: 10.1128/JCM.41.2.581-585.2003
  6. Ni Mhurchu E, Ospina J, Janjua AS, et al. Fungal rhinosinusitis: a radiological review with intraoperative correlation. Can Assoc Radiol J. 2017;68(2):178–186. doi: 10.1016/j.carj.2016.12.009
  7. Wang ZM, Kanoh N, Dai CF, et al. Isolated sphenoid sinus disease: an analysis of 122 cases. Ann Otol Rhinol Laryngol. 2002;111(4):323–327. doi: 10.1177/000348940211100407
  8. Mensi M, Piccioni M, Marsili F, et al. Risk of maxillary fungus ball in patients with endodontic treatment on maxillary teeth: A case-control study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;103(3):433–436. doi: 10.1016/j.tripleo.2006.08.014
  9. Grosjean P, Weber R. Fungus balls of the paranasal sinuses: a review. Eur Arch Otorhinolaryngol. 2007;264(5):461–470. EDN: GSLXPJ doi: 10.1007/s00405-007-0281-5
  10. Kochetkov PA, Ordian AB, Lunicheva AA. On the pathogenesis of isolated non-invasive fungal sphenoiditis. Medical Council. 2018;(8):52–57. EDN: UZQHEZ doi: 10.21518/2079-701X-2018-8-52-57
  11. Lee TJ, Huang SF, Chang PH. Characteristics of isolated sphenoid sinus aspergilloma: report of twelve cases and literature review. Ann Otol Rhinol Laryngol. 2009;118(3):211–217. doi: 10.1177/000348940911800309
  12. Pagella F, Pusateri A, Matti E, et al. Sphenoid sinus fungus ball: our experience. Am J Rhinol Allergy. 2011;25:276–280. doi: 10.2500/ajra.2011.25.3639
  13. Aribandi M, McCoy VA, Bazan C. Imaging features of invasive and noninvasive fungal sinusitis: a review. RadioGraphics. 2007;27(5):1283–1296. doi: 10.1148/rg.275065189
  14. Turner JH, Soudry E, Nayak JV, Hwang PH. Survival outcomes in acute invasive fungal sinusitis: a systematic review and quantitative synthesis of published evidence. Laryngoscope. 2013;123(5):1112–1118. doi: 10.1002/lary.23912
  15. Watkinson JC, Clarke RW. Scott-Brown’s otorhinolaryngology and head and neck surgery: Vol. 1. basic sciences, endocrine surgery, rhinology. 8th edition. CRC Press: Boca Raton; 2018. 1402 p. doi: 10.1201/9780203731031
  16. Charles PE, Dalle F, Aho S, et al. Serum procalcitonin measurement contribution to the early diagnosis of candidemia in critically ill patients. Intensive Care Med. 2006;32(10):1577–1583. EDN: JZPRND doi: 10.1007/s00134-006-0306-3
  17. Dufour X, Kauffmann-Lacroix C, Ferrie JC, et al. Paranasal sinus fungus ball: Epidemiology, clinical features and diagnosis. A retrospective analysis of 173 cases from a single medical center in France, 1989–2002. Med Mycol. 2006;44(1):61–67. doi: 10.1080/13693780500235728
  18. Gungor A, Adusumilli V, Corey JP. Fungal sinusitis: progression of disease in immunosuppression — a case report. Ear Nose Throat J. 1998;77(3):207–211. EDN: CSEZIH doi: 10.1177/014556139807700311
  19. Ota R, Katada A, Bandoh N, et al. A case of invasive paranasal aspergillosis that developed from a non-invasive form during 5-year follow-up. Auris Nasus Larynx. 2010;37(2):250–254. doi: 10.1016/j.anl.2009.06.003
  20. Ferguson BJ. Fungus balls of the paranasal sinuses. Otolaryngol Clin North Am. 2000;33(2):389–398. doi: 10.1016/s0030-6665(00)80013-4
  21. Adelson RT, Marple BF. Fungal rhinosinusitis: state-of-the-art diagnosis and treatment. J Otolaryngol. 2005;34(Suppl 1):S18–S23.
  22. Leroux E, Valade D, Guichard, JP, Herman P. Sphenoid fungus balls: clinical presentation and long-term follow-up in 24 patients. Cephalalgia. 2009;29(11):1218–1223. doi: 10.1111/j.1468-2982.2009.01850.x
  23. Toussain G, Botterel F, Alsamad IA, et al. Sinus fungal balls: characteristics and management in patients with host factors for invasive infection. Rhinology. 2012;50(3):269–276. doi: 10.4193/Rhin11.223
  24. Dufour X, Kauffmann-Lacroix C, Ferrie JC, et al. Paranasal sinus fungus ball and surgery: a review of 175 cases. Rhinology. 2005;43(1):34–39.

Supplementary files

Supplementary Files
Action
1. JATS XML
2. Fig. 1. Temporal changes in inflammatory markers (C-reactive protein, procalcitonin).

Download (125KB)
3. Fig. 2. Multislice computed tomography of the paranasal sinuses (2023), sagittal and coronal views. A hyperdense lesion with a solid component (fungal ball) and an area of calcification (arrow) is observed in the right sphenoid sinus, which is characteristic of a mycetoma. No signs of bony destruction are detected.

Download (120KB)
4. Fig. 3. Intraoperative endoscopic view. Endoscopic transnasal sphenoethmoidectomy was performed on the right side. A fungal ball occupying the lower third of the right sphenoid sinus was visualized.

Download (146KB)
5. Fig. 4. Intraoperative endoscopic view after removal of the fungal ball. The sinus was irrigated with an antiseptic solution and 0.9% sodium chloride. No residual fungal material was detected on inspection with 30° and 70° endoscopes; the mucosa appeared edematous.

Download (115KB)
6. Fig. 5. Multislice computed tomography of the paranasal sinuses (2024), coronal and sagittal views. A wide communication between the right sphenoid sinus and the ethmoid air cells remains along the anterior wall, up to 7 mm. No mucosal thickening or sinus contents are observed. No bony destruction is noted.

Download (161KB)
7. Fig. 6. Endoscopic image (2024), 0° endoscope. No sinus content is observed in the right sphenoid sinus. The mucosa appears pale pink, smooth, and glistening. The sphenoid sinus ostium remains widely patent.

Download (91KB)
8. Fig. 1. Temporal changes in inflammatory markers (C-reactive protein, procalcitonin).

Download (125KB)