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CLINICAL REPORT
Sphenoid sinusitis with intracranial extension produced by an emergent fungus Yolanda Escamilla Carpintero,a,* Mateu Espasa Soley,b M. Rosa Bella Cueto,c Mario Prenafeta Morenod Servicio de ORL, Corporación Sanitaria y Universitaria del Parc Taulí Sabadell, Barcelona, Spain UDIAT, Laboratorio de Microbiología, Corporación Sanitaria y Universitaria del Parc Taulí, Sabadell, Barcelona, Spain c Servicio de Anatomía Patológica, Corporación Sanitaria y Universitaria del Parc Taulí, Sabadell, Spain d Servicio de Radiodiagnóstico, Corporación Sanitaria y Universitaria del Parc Taulí, Sabadell, Spain a
b
Received October 14, 2009; accepted January 18, 2010
KEYWORDS Fungal sphenoid sinusitis; Sphenoid sinus surgery; Mycetoma; Phialemonium
Abstract This is a case of fungal sphenoid sinusitis in a diabetic patient with non-specific symptoms and bone erosion radiological findings in the superior and posterior sphenoid walls. Surgical treatment was performed by transnasal endoscopic approach and voriconazole orally thereafter. The histopathological study found fungus hyphal without mucosa invasion and the molecular study determined DNA to be Phialemonium curvatum, an unusual pathogen.
PALABRAS CLAVE Sinusitis fúngica esfenoidal; Cirugía endoscópica; Micetoma; Phialemonium
Sinupatia esfenoidal con extensión intracraneal causada por un hongo emergente
© 2009 Elsevier España, S.L. All rights reserved.
Resumen Sinusitis fúngica esfenoidal en paciente diabética, con clínica inespecífica y signos de erosión ósea radiológica a nivel sellar superior y retroclival. Tratada quirúrgicamente por abordaje endoscópico transnasal y voriconazol oral. La histología mostró hifas, descartando invasión mucosa y el estudio molecular detectó ADN de Phialemonium curvatum, un inusual patógeno. © 2009 Elsevier España, S.L. Todos los derechos reservados.
*Corresponding author. E-mail address:
[email protected] (Y. Escamilla Carpintero). 0001-6519/$ - see front matter © 2009 Elsevier España, S.L. All rights reserved.
Sphenoid sinusitis with intracranial extension produced by an emergent fungus
Introduction Isolated sphenoid sinus disease constitutes 2%-3%1 of sinus disease, with infection being the most common cause. There has been an increased prevalence of fungal sinusitis in recent years. There are 5 types of fungal sinusitis according to their histopathology,2 3 invasive types (with evidence of hyphae in the sinus mucosa, submucosa, vessels or bone), acute necrotising, chronic invasive and granulomatous invasive; plus 2 non-invasive types, fungal ball and allergic fungal. These may change their development when immunity is compromised. The identification of the causal fungus is not always possible due to technical reasons, and the incidence of infrequent pathogens is commonly underestimated.3
Clinical report We present a 71-year-old woman with a history of liver cirrhosis (HCV), microcytic anaemia, diabetes II, anxious-depressive syndrome, cardiac arrhythmia, arterial hypertension and mild cognitive impairment. She was admitted in hepatology due to presenting general deterioration, asthenia, severe headaches and instability of 6 months’ evolution. A complete physical examination, laboratory tests (including thyroid function), plain chest radiograph and abdominal ultrasonography were normal. A cranial CT scan showed diffuse cerebral atrophy and bilateral sphenoid sinus occupation, predominantly on the right side, with internal calcifications and sclerosis, remodelling of the bone margins, with possible intracranial extension. We requested an ENT assessment for a transsphenoidal biopsy. The CT and MRI taken, which focused on sinuses-skull base (Figures 1A and B), showed erosion of the bony margins at the level of the superior and posterior walls of the right
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sphenoid, with posterior retroclival and superior sellar extension. Sinus occupation was hyperintense on T1 and hypointense on T2, with peripheral contrast uptake and no vascular invasion at the level of the carotid and basilar arteries. We performed a right sphenoidotomy by transnasal endoscopic approach, observing the sphenoid occupied by a brown material, which was removed with suction and washing. Pathology revealed fragments of congestive respiratory mucosa, with chronic inflammation. This consisted predominantly of plasma cells and acellular eosinophilic fragments, consisting of hyphae that were positively stained by periodic Schiff acid and silver (Gomori) (Figure 2A). The samples were not correctly processed for microbiological culture. However, biopsies in paraffin underwent panfungal genome amplification and sequencing, obtaining positive results for Phialemonium curvatum. Subsequently, she was treated with voriconazole, with the symptoms disappearing. Her diabetes evolved poorly, requiring insulin treatment. The immediate postoperative controls showed a clean sphenoid sinus, with a defect at the level of the posterior and superior walls (Figure 2B). Given the initial involvement, oral voriconazole was maintained for 3 months. The CT scan showed a clean sinus, with a minimum widening of the mucosa and persistence of sclerosis in the bone walls, as well as absence in the posterior and superior walls (Figure 2B).
Discussion The most frequent location of noninvasive, chronic sinusitis is the maxillary sinus, followed by the ethmoid and frontal sinuses. Sphenoid sinus location is very rare.1 Sphenoid symptoms are nonspecific, despite the critical relationships (carotid artery, dura mater and the 2nd to 6th
Figure 1 1A) CT scan: sphenoid occupation and erosion of the superior and posterior sphenoid walls. 1B) MRI: Occupation, hypointense on T2, without vascular invasion. Occupation, hyperintense on T1, with retroclival and suprasellar involvement.
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Figure 2 A) Anatomical pathology: hyphae, Gomori staining. B) Control endoscopy, radiological control image.
cranial nerves). Frontal and retro-orbital headache can be emphasised, followed by impairment of vision, retro-orbital pain, diplopia, exophthalmos and blindness. The diagnosis of sphenoid mycosis is carried out by symptoms, imaging and biopsy: CT scan shows sinus opacity that may present areas of bone density due to the deposition of calcium phosphate and sulphate crystals. Bone involvement, which does not mean tissue invasion,4 is observed more frequently in invasive forms; MRI outlines extrasinusal-intracranial-vascular involvement (in our case, it was requested due to the significant bone erosion observed in the CT scan); and biopsy, which is central to diagnosis, determines the mucosal invasion. Fungal culture is negative in more than 50% of cases, while the agent is identified by molecular biology techniques in other cases.5 Aspergillosis is the most common fungal infection, although other infectious agents could be underestimated due to the difficulty of identifying them. P. curvatum is a filamentous, dematiaceous fungus that is widely distributed in the environment. Only 19 cases have been described in the literature, most with haematogenous spread in immunocompromised patients.3 Treatment is currently performed by endoscopic transnasal or transethmoidal approaches, avoiding other more aggressive approaches (transseptal, transpalatal).
Antifungal treatment is indicated when there is bone lysis, with voriconazole being the most commonly-used agent. In our case, with no histological confirmation of invasive disease, its administration was considered due to the significant bone involvement. It was subsequently appropriate for P. curvatum.3
References 1. Castelnuovo P, Pagella F, Semino L. Endoscopic treatment of the isolated sphenoid sinus lesions. Eur Arch Otorhinolaryngol. 2005;262:142-7. 2. De Shazo R, O’Brien M, Chapin K, Soto-Aguilar M, Gardner LL, Swain R. A new classification and diagnostic criteria for invasive fungal sinusitis. Arch Otolaryngol Head Neck Surg. 1997;123:11818. 3. Rivero M, Hidalgo A, Alastruey-Izquierdo A, Cia M, Torrobas L, Rodriguez Tudela JL. Infections due to Phialemonium species: case report and review. Medical Mycology. 2009;1-9. 4. Manning SC, Holman M. Further evidence for allergic pathophysiology in allergic fungal sinusitis. Laryngoscope. 1998; 108:1485-96. 5. Hassan H. Sinusitis. Fungal Last Updated. Otolaryngology-Head and Neck Surgery. 2005;8:2.
