Parasellar Aspergillus granuloma extending from the sphenoid sinus: Report of two cases

ELSEVIER

PARASELLAR ASPERGLLUS GRANULOMA EXTENDING FROM THE SPHENOID SINUS: REPORT OF Two CASES Hiroki Kurita, M.D.; Yoshiaki Shiokawa, M.D.; Kazuhide Furuya, M.D.; Hiromu Segawa, M.D.; and Keiji Sano, M.D. Department of Neurosurgery, Fuji Brain Institute and Hospital, Shizuoka, Japan

Kurita H, Shiokawa Y, Furuya K, Segawa H, Sano K. Parasellar

Aspergillus granuloma extendingfromthe sphenoidsinus:report

of two cases.SurgNeural1995;44:489-94. BACKGROUND

Sphenoid sinus aspergillosis is a rare diseaseknown to show an aggressive course with high mortality. Early diagnosis, though difficult, is required to prevent lethal fungal meningoencephalitis. CASE

REPORT

We describe two cases of parasellar Aspergillus granuloma extending from the sphenoid sinus clinically indistinguishable from intracranial neoplasms.In the first patient, the fungus colony was visualized by computed tomography (CT) and magnetic resonance imaging (MRl) as a calcified concretion and total removal was curative. In the second patient, partial removal and subsequent antifungal therapy had minimal effect. CONCLUSIONS

The prognosis of the patients with this diseasedepends on prompt surgical treatment before intradural invasion occurs, and CT and MRl are useful diagnostic maneuvers for detecting calcified Aspergillus colonies. KEY

WORDS

Sphenoid

sinus, Aspergillus, granuloma,

parasellar

region.

A

spergillus, a spore-forming fungus of the Ascomycetes class, which is distributed worldwide, is known to be the most common fungal pathogen in paranasal sinus disease. In line with the recent increase in the use of new antibiotics, steroids, and immunosuppressants, and the advancing age of the population, the reported incidence of paranasal sinus aspergillosis

[l-21].

However, aspergillosis

has also increased

of the sphenoid

sinus

is quite rare, but it carries a poorer prognosis than

that of other sinuses because of the high risk of intracranial extension [4,9,10-12,141. Early diagnoAddress reprint surgery, University Japan. Received June

requests to: Hiroki of Tokyo Hospital, 7, 1994;

0 1995 by Elsevier Science 655 Avenue of the Americas,

accepted

Kurita, M.D., Department 7-3-l Hongo, Bunkyo-Ku, April

12, 1995.

Inc. New York,

NY 10010

of NeuroTokyo 113,

sis is very important to prevent the development of diffuse Aspergillus meningoencephalitis, which is a lethal infection [3,11]. Two cases of parasellar Aspergillus granulomas extending from the sphenoid sinus, which were clinically indistinguishable from intracranial neoplasms, are presented in this report. The criteria for diagnosing this entity and the therapy for it are reviewed.

CASEREPORTS PATIENT 1 An otherwise healthy, 69-year-old woman was referred to us in December 1991, because of headache over a 2-month period. Her medical history was unremarkable and negative results were obtained for upper respiratory tract infections. Her physical findings were normal, as were her complete blood count biochemical profile and endocrine function, and she was neurologically intact. Skull X-ray films and sellar tomography revealed destruction of the sellar floor and posterior cliinoid process, erosion of the clivus, and opacification of the sphenoid sinus. Computed tomography (CT) demonstrated a homogeneous low-density mass without contrast enhancement occupying

the sphenoid sinus and sella turcica,

which contained

calcific

densities

(Figure 1 A, B).

Magnetic resonance imaging (MRI) revealed a hy-

pointense area within a hyperintense mass on both Tr- and T,-weighted images. The mass, which was n’ot enhanced with gadolinium diethylenetriaminepentaacetic

acid (Cd-DTPA), extended to the

right cavernous sinus (Figure 2 A, B, C), and carotid angiography showed this lesion was avascular. The patient underwent exploration via the transsphenoidal approach for a presumed nonfunctional 0090-3019/95/$15.00 SSDI 009&3019(95)0020&L

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beginning of July 1992. Although she had a long history of liver cirrhosis, she had neither fever nor a history of upper respiratory tract infections. Neurologic examination on admission revealed left pt@ sis, left total ophthalmoplegia, and blindness in the left eye. Her laboratory values, including those for endocrine function, but excepting those indicating liver dysfunction, were normal. Skull X-ray films and sellar tomography revealed erosion of the sellar floor and opacification of the sphenoid sinus. CT revealed an isodense mass with irregular contrast enhancement occupying the sphenoid and left cavernous sinuses with invasion to the left orbita, and the surrounding bone structure had been destroyed (Figure 4 A, B). MRI of the lesion demonstrated isointensity on T,-weighted and slight hyperintensity on T,-weighted images with marked Cid-DTPA enhancement (Figure 5 A, B). The carotid angiographic findings were within the normal range. We presumed the lesion was a malignant neoplasm. The diagnostic surgical procedure used was transsphenoidal resection. A buttery, yellowish necrotic material, which filled the sphenoid sinus, was excised as far as possible, after which a silicone tube was inserted for continuous drainage, The material was found histologically to comprise granular tissue containing Aspergillus (Figure 6). Postoperatively, the patient was treated by local irrigation with systemic administration of antifungal drug (Fluconazole), to which she showed only a minimal response, and she died from fulminant fungal meningoencephalitis on the 23rd postoperative day. Noncontrast CT scan of Patient 1 demonstrates 0which (A)a contains low-density mass within the sphenoid sinus (s), calcifications (arrow). (B) Postcontrast CT scan of Patient 1 reveals no contrast Notice calcification (UFYOW).

enhancement.

invasive pituitary adenoma. However, brownish de bris with yellowish, purulent, mutinous fluid was found in the sphenoid and right cavernous sinuses and sella turcica, and this was totally removed. Histopathologic examination confirmed it was an Aspergiffus fungus colony (Figure 3). Antifungal agents were not used during the postoperative period. No other fungal foci were found and the patient was discharged on the 12th postoperative day. She has shown no recurrence during a 3-year follow-up

period.

PATIENT 2 A 78-year-old woman had had continuous headaches since April 1992, and presented with subacute, progressive left orbital apex syndrome at the

DISCUSSION Aspergillosis of the paranasal sinus is classified either as the noninvasive type, which exhibits no bony destruction and resembles chronic or recurrent sinusitis [3,7,14], or the invasive type, which is characterized by destruction of surrounding bone structures, is more aggressive, and may show intracranial or orbital extension [3,7,1012,141. However, these types, which probably differ in character due to the location and duration of the infection or the medical status of the patient, are often indistinguishable. Of a total of 34 previously reported cases of sphenoid sinus aspergillosis, 18 (53%) had the aggressive form with parasellar granuloma extension [2,4,10-121, like that observed in our two patients. Cranial nerve symptoms due to nerve compression or invasion were observed in 17visual disturbance in 11 [2,4,10,11,16,20], external ocular move-

Parasellar Aspergilloma

Surg Neurol 1995;44:489-94

49 1

(A) Axial T,-weighted MRI of Patient 1 shows a hyI4 pointense area within a hyperintense mass in the sphenoid sinus (.S). (B) Axial T,-weighted MRI with (arrow)

Gd-DTPA of Patient 1 shows no Gd enhancement. (C) Axial T,-weighted MRI of Patient 1 shows a hypointense area (urrow) due to calcification. The remainder of the sphenoid sinus appears hyperintense.

Photomicrograph of PaEl tient 1 reveals the histopathologic findings of septate hyphae with dichotomous branching, which are characteristic of Aspergillus (Grocott stain X400).

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q 1

Kurita et al

(A) Axial T,-weighted MRI of Patient 2 reveals a Ia hyperintense mass (arrow) involving the left cavernous sinus and adjacent superior orbital fissure. (B) Axial

(A) Noncontrast CT scan of Patient 2 shows an irregular infiltrative mass (arrow) at the level of the left superior orbital fissure, extending into the orbital apex. (J3) Postcontrast CT of Patient 2 demonstrates irregular contrast enhancement.

T,-weighted MRf with Cd-DTPA of Patient 2 reveals abnormal enhancement in the left cavernous sinus and superior orbital fissure region (arrow).

ment palsy in 10 [2,4,10-12,15,16,20], trigeminal dysfunction in 5 [2,4,12,14,20] and anosmia in 1 [15]. The mean patient age was 51.2 years, but ranged from 18 to 78 years. Surprisingly, most patients had shown no associated predisposing factors [2,6,10, 12-16,18,20]. This granulomatous infection may develop even in a patient with a very benign clinical history and no evidence of debilitation secondary to a systemic disease or medical therapy and in the absence of the increased likelihood of airborne spore inhalation associated with certain occupations. However, if the host is already compromised, the infection is more serious [6,11,14]. Prompt and accurate preoperative diagnosis of

this lesion is difficult [2,3,8,10,12,14,16]. lmmunologic testing and nasal cultures have not been reliably predictive [3] and the radiologic features resemble those of malignant neoplasms [5,6,8,12-16, 201. In several recent reports, the observation of calcifications within the opacified sinus has been stated to be a useful feature for the radiologic diagnosis of aspergillosis. Concretions were observed on plain X-ray films or by tomography in about half the reported paranasal sinus aspergillosis cases [3,5,14]. Calcium accumulation within the fungus colony is detected readily using CT and appears as a hyperdense area in a nonenhanced soft-tissue

Parasellar Aspergilloma

Surg Neurol 1995;44:489 -94

493

Photomicrograph of PaEl - tient 2. The histopathologic findings are typical of aspergillosis (periodic [PAS] stain X400).

density mass, such as that in our patient 1 [3,6,7,14]. However, the reported sensitivity (70%76%) [3,21] of this criterion suggests that CT findings alone are not conclusive and the MRI findings are more specific [2,21]. Several factors, such as the calcium, ferromagnetic element, and concentrated protein contents of the fungus colony, may decrease the intensity of the “solid part” visualized on T,-weighted images and do so even more on T,weighted images [21]. This finding appears to be characteristic of aspergillosis and differs markedly from the features of bacterial sinusitis and neoplasms. With respect to the “fluid part,” however, the reported intensities are inconsistent. In exceptional cases, such as the fulminant aspergillosis that occurs in compromised patients like our patient 2, such characteristic calcification is unlikely to be observed [3], probably because of the subacute progression of the clinical course. The aim of treatment should be the complete removal of the mycotic masses as early as possible. In order to minimize cerebrospinal fluid (CSF) contamination, extradural resection via the transsphenoidal approach should be selected, despite the limited operative field. Even with the invasive type, providing intradural extension had not yet occurred,

total

excision

was

curative

without

sys-

temic use of antifungal agents [2,8,13]. However, in patients in whom total removal cannot be achieved or intradural invasion has already been confirmed, intensive systemic therapy with antifungal agents must be administered postoperatively to prevent

acid-Schiff

subsequent lethal vasculitis [4,12,14] and/or meningoenclephalitis developing [6,11,19]. Despite the reported high mortality in such cases, reaching 75%-80% [3,4], several cases have been treated successfully [ 12,201. The prognosis of patients with this disease depends

on prompt

diagnosis

and early

treatment

before intradural invasion occurs, and CT and MRI are useful diagnostic maneuvers for detecting the characteristic calcified fungus colonies of Aspeqillus. The author wishes to express his appreciation demi blichizuki for her secretarial suppoti.

to Miss Hi-

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C

APlTAL AS SUCH IS NOT EVIL; IT IS ITS WRONG USE THAT IS EVIL. CAPITAL IN SOME FORM OR OTHER WILL ALWAYS BE NEEDED. MOHANDAS

K. GANDHI (1869-1948) “HARIJAN,” JULY 28, 1940