Khoueir N, Verillaud B, Herman P
Département d'otorhinolaryngologie/chirurgie cervico-faciale/chirurgie de la base du crâne, université Paris-Diderot, hôpital Lariboisière, AP-HP, Paris, France; Département d'otorhinolaryngologie/chirurgie cervico-faciale, hôpital universitaire Hôtel-Dieu de France, université Saint Joseph, faculté de médecine, Beirut, Lebanon.
Département d'otorhinolaryngologie/chirurgie cervico-faciale/chirurgie de la base du crâne, université Paris-Diderot, hôpital Lariboisière, AP-HP, Paris, France.
Eur Ann Otorhinolaryngol Head Neck Dis. 2019 Apr;136(2):119-121. doi: 10.1016/j.anorl.2018.11.009. Epub 2018 Dec 4.
Chronic invasive fungal rhinosinusitis (CIFR) is a rare entity generally observed in immunodepressed subjects. The pathogen most frequently identified is Aspergillus spp. Imaging generally reveals invasive pseudoneoplastic features. We report a case of Scedosporium apiospermum (S. apiospermum) CIFR with an atypical clinical and radiological presentation.
A 72-year-old immunocompetent man presented with chronic headache, neck pain and bilateral limitation of lateral gaze. Imaging revealed an isolated left sphenoidal lesion with marked bone changes and an extradural abscess over the clivus. Large endoscopic sphenoidotomy with type II rhinopharyngectomy was performed and the diagnosis of S. apiospermum CIFR was based on histological examination and fungal culture. The patient refused all medical treatment and did not present any signs of recurrence after 1 year of follow-up.
S. apiospermum is a fungal species rarely isolated in CIFR. The present case was revealed by an atypical clinical presentation including isolated sphenoidal infection complicated by bilateral abducens nerve paralysis and extradural abscess. Imaging was also unusual, revealing features of fibrous dysplasia or bacterial osteomyelitis rather than the typical pseudoneoplastic appearance. The patient was successfully treated by surgery alone, which may therefore be sufficient treatment in immunocompetent subjects.
