British Journal of Neurosurgery
ISSN: 0268-8697 (Print) 1360-046X (Online) Journal homepage: http://www.tandfonline.com/loi/ibjn20
Cerebral infarction due to Aspergillus arteritis following glioma surgery Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch To cite this article: Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch (1992) Cerebral infarction due to Aspergillus arteritis following glioma surgery, British Journal of Neurosurgery, 6:5, 485-490, DOI: 10.3109/02688699208995040 To link to this article: http://dx.doi.org/10.3109/02688699208995040
Published online: 06 Jul 2009.
Submit your article to this journal
Article views: 11
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Citing articles: 1 View citing articles
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ibjn20 Download by: [Universite Laval]
Date: 08 April 2016, At: 12:14
British Journal of Neurosurgery (1992) 6,485-490
SHORT REPORT
Cerebral infarction due to Aspergillus arteritis following glioma surgery REWATI RAMAN SHARMA, NIHAL T. GURUSINGHE & PATRICK G. LYNCH
Downloaded by [Universite Laval] at 12:14 08 April 2016
Royal Preston Hospital, Preston, L a n a PR2 4HT, UK
Abstract Cerebral infarction due to fungal arteritis is an uncommon complication of neurosurgical operations and adjuvant immunosuppressive therapy, including long-term steroids. If unrecognized, the neurological deterioration which ensues may be mistreated by increasing the dose of steroids. A case of a 38-year-old Caucasian male who had no obvious immune deficiency or fungal infection prior to a craniotomy for cerebral tumour is described in whom perioperative aspergillus infection resulted in cerebral arteritis and extensive cerebral infarction with a fatal outcome. Long-term steroid therapy used in the management of cerebral tumours may carry an increased risk of systemic or cerebral fungal infection. The possibility of cerebral mycosis (arteritis) and dangers of its non-recognition are highlighted.
Key words: Cerebral aspergillosis, cerebral infarction, fungal arteritis, neurosurgery, postoperative complications, steroids.
Introduction Fungal arteritis is a rare disease caused by a variety of fungal infections, e.g. aspergillosis,' phycomycosis,* pythiosis: mucormycosis$ blastomycosis,5 candidosis6 and paracoccidiodomycosis.' Cerebral fungal arteritis (CFA) is usually due to opportunist pathogenic ubiquitous true hyphal fungi such as Aspergillus and Phycomy~etes.~,~ These infections are more prone to occur in patients whose resistance to infection is lowered by a co-existing condition such as diabetes mellitus, chronic pulmonary disease or malignant disease, especially leukaemia or lymphoma.8-10 In addition, patients treated with ACTH, steroids, antibiotics and cytotoxic drugs and drug abusers seem to be particularly susceptible.8-'0 Steroid therapy is used in many neurological and neurosurgical disorders and carries an increased risk of fungal infection. A literature search revealed only seven cases of cerebral
aspergillosis following neurosurgical procedures.l1-I6 We review these cases and present a case of CFA due to Aspergillus infection causing extensive cerebral arterial thrombosis and massive haemorrhagic infarction following excision of a glioma in a healthy young male who had no systemic disease or preoperative fungal infection.
Case report A 38-year-old right-handed previously healthy male presented with a 4-month history of episodic generalized seizures. General physical examination and neurological assessment showed normal findings with no evidence of raised intracranial pressure. Routine haematological and biochemical tests were normal. Computed axial tomographic (CT) scans of the head showed a low attenuation nonenhancing intrinsic lesion in the left fronto-
485
Downloaded by [Universite Laval] at 12:14 08 April 2016
486
Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch
temporal region with slight compression of the left lateral ventricle. The radiological diagnosis was of a low-grade astrocytoma. Cerebral angiography confirmed the C T findings of an intrinsic avascular space occupying lesion. Steroid therapy (dexamethasone, 16 mglday) was commenced a week prior to operation. A craniotomy was performed and the temporal portion of the tumour excised. The Sylvian fissure and middle cerebral artery were exposed. The histopathology of the tumour revealed a low-grade astrocytoma. No antibiotics were used postoperatively and apart from a transient dysphasia the patient made a good initial recovery. The dosage of the steroid therapy was gradually reduced but during the third postoperative week whilst the patient was awaiting radiotherapy he developed severe headaches, intermittent focal seizures involving the right arm and severe motor dysphasia. Repeat CT revealed cerebral oedema around residual tumour and the dosage of steroids (dexamethasone) was increased. The patient continued to deteriorate, becoming drowsy with the development of a left third nerve palsy and a right hemiplegia. C T now revealed extensive cerebral oedema around residual fronto-temporal tumour with areas of haemorrhage more marked in the left temporal region (Fig. 1). There was a significant shift of midline structures to the right. A further craniotomy was performed and the residual frontal portion of the tumour was excised and a partial frontal lobectomy performed. Despite aggressive therapy the patient continued to deteriorate and death occurred 2 days postoperatively. Post-mortem examination of the brain revealed a ragged cavity in the orbital region of the left frontal lobe. Most of the left temporal lobe had been excised. Sections of the brain in the coronal plane revealed a marked shift of the midline structures from left to right and massive haemorrhagic infarction of the left cerebal hemisphere within the territory of distribution of the left internal carotid and left middle cerebral arteries. The lumina of the left internal carotid artery and the proximal part of the left middle cerebral artery were occluded
FIG. 1. CT brain scan (unenhanced) showing extensive cerebral oedema around residual frontal tumour and haemorrhagic infarction in the left fronto-temporal region.
by thrombus. No residual tumour was seen. Light microscopy of the left middle cerebral artery confirmed the presence of intraluminal thrombus (Fig. 2). Fungal hyphae were seen invading the vessel wall (Fig. 3). Fungal stains showed branching septate hyphae compatible with Aspergillus, probably A. fumigatus (Fig. 4).
Discussion Opportunistic fungal infections such as candidiasis, cryptococcosis, aspergillosis and zygomycosis are now a familiar problem in many parts of the world and may account for much morbidity and mortality in immunosuppressed patients, including those with AIDS.*O However, cerebral mycosis is a rare condition and in our case it was caused by Aspergillus (probably fumigatus). The other usual human pathogenic Aspergillus species are A. niger, A . flavus and A. oxyzae. Aspergillus is ubiquitous and has branching septate hyphae of from 3 to 12 p m diameter.
Downloaded by [Universite Laval] at 12:14 08 April 2016
Cerebral infarction due to Aspergillus arteritis
FIG. 2. Left middle cerebral artery showing intraluminal thrombus and inflammation of the vessel wall. H&E.
487
X 42.
FIG. 3. Left middle cerebral artery showing virtual destruction of the tunica media with radial extension of fungus into the vessel lumen. Grocott. x 198.
Cerebral infection is usually secondary to a primary focus elsewhere in the body. Infection reaches the brain either directly from the nasal sinuses via vascular channels or is blood-borne from the lungs and gastrointestinal t r a ~ t . ~ - ~ O The , l ~ - infection l~ may also be airborne, contaminating the tissues during a
neurosurgical In our case, the possibilities are either airborne spores reaching the operative field and germinating or spread from the sphenoid sinus via the internal carotid artery. The former possibility is more likely as at autopsy the only evidence of the fungal infection was in the operative field.
Downloaded by [Universite Laval] at 12:14 08 April 2016
488
Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch
FIG.4. Photomicrograph showing branched septate hyphae characteristic of Aspergillus species. Grocott.
Fungi infecting the brain are found in three major forms with distinctive clinicomorphologic syndrome^.^ (1) Acute and chronic leptomeningitis produced by pure yeasts (blastomycosis, cryptococcosis and histoplasmosis). These small sized fungi gain access to the cerebral microcirculation from which they infect the subarachnoid space. (2) Abscesses produced by pseudohyphae (candidiasis) which occlude cerebral arterioles because of their intermediate size (larger than yeasts yet smaller than true hyphae) and result in adjacent tissue necrosis and microabscess formation. ( 3 ) Cerebral infarction produced by branched hyphal fungi (aspergillosis and phycomycosis) which mainly obstruct the large and intermediate sized cerebral arteries. The hyphae invade the vessel walls causing cerebral arterial thrombosis, associated cerebral infarction and adjacent cerebritis. The evolving haemorrhagic cerebral infarcts may convert into septic infarcts with associated cerebritis and abscesses. Some of these features were seen in the present case. The left internal carotid and left middle cerebral arteries were thrombosed and fungal hyphae were seen in the vessel wall and lumen. Owing to its variable clinical manifestations, cerebral mycosis is difficult to diagnosis in life.
X
396.
It may present with symptoms suggestive of meningitis, a space occupying lesion (abscess and granuloma) or as a sudden cerebrovascular event (fungal aneurysm causing subarachnoid haemorrhage and CFA haemorrhagic cerebral i n f a r ~ t i o n ) . ~ - ' ~ , ' The ~-~~ appearance of neurological symptoms in cases of an obvious extracranial fungal infection, debilitating neoplastic disorders, HIV infection, or immunosuppression may suggest cerebral m y ~ o s i s . ' ~ , * ~It- *is ~ well known that disseminated aspergillosis with involvement of the central nervous system is seen in patients debilitated by neoplastic disorders, and Meyer et aL2' found nine cases of cerebral aspergillosis out of 93 patients suffering from various systemic malignant tumours. Neurological deterioration due to superadded fungal infection following neurosurgical procedures presents a special problem. The fungal infection may go unrecognized and the neurological deterioration may be mistreated by immunosuppressive d p g s (steroids). Only seven cases of cerebral aspergillosis following neurosurgical procedures have been reported in the literature."-'6 In four patients, direct extension of Aspergillus from the paranasal sinuses was considered,13.14and in this context it is interesting to note that in one unpublished
Downloaded by [Universite Laval] at 12:14 08 April 2016
Cerebral infarction due to Aspergillus arteritis case cerebral aspergillosis occurred following surgical exploration of an infected maxillary sinus.19 In four patients, the portal of entry remained u r ~ c e r t a i n ~ ~as~in~ our ~ J ~case. J ~ It is, however, possible that fungi were introduced during the neurosurgical procedures and predisposing conditions (antibiotic therapy, diabetes mellitus, steroid therapy) accelerated the progress of infection. In our case no antibiotics were used and the patient did not have diabetes mellitus. Among the reported cases of Aspergillus infection following neurosurgical procedures (craniopharyngioma," thalamic glioma,I2 pituitary turn our^,^^ olfactory groove meningi~ma,'~ subdural haematoma,I5 internal carotid artery aneurysm16), cerebral aspergillosis presented as granuloma in three in one of which fungal abscess was a~s0ciated.l~Two patients presented with cerebral in one of which abscess was associated.16 One patient had abscess en~ephalitis,'~ and another cerebral infarction caused by va~culitis.'~ In the latter, the presence of a screw in the sphenoid sinus (transphenoidal yttrium-90 pituitary implant) fostered the Aspergillus infection and the foreign material provided an access route to the subarachnoid space. Diagnosis is made by culture on special tissue media of tissue obtained at operation or autopsy.2a The use of antifungal antibiotics (amphotericin-B, flucytosine and rifampicin) combined with aggressive surgical removal of an intracranial abscess or granuloma is the recommended treatment for cerebral aspergillosis. Nevertheless, the outcome for patients with postoperative aspergillosis remains poor. Of eight patients including the present case, only two have ~ u r v i v e d . In ~ ~cases , ~ ~ of CFA where surgery is inappropriate or contraindicated, reliance has to be placed on antifungal antibiotics and supportive therapy. Acknowledgements We gratefully acknowledge the technical contributions of Pamela Thompson, Angela Clayton and Ruth Sharples to this work. The authors also express their appreciation to
489
Pauline Ashcroft for secretarial help in the preparation of the manuscript and to the Medical Illustration Department, Royal Preston Hospital for the photographs.
Address for correspondence: Dr Patrick G . Lynch, Neuropathology Laboratory, Royal Preston Hospital, PO Box 202, Sharoe Green Lane, Fulwood, Preston, Lancs PR2 4HG, UK. References 1 Fernando S S , Lauer CS. Aspergtllus fumigatus infection of the optic nerve with mycotic arteritis of cerebral vessels. Histopathology 1982; 6(2):227-34. 2 Carpenter DF, Brubaker LH, Powell RD, et al. Phycomycotic thrombosis of the basilar artery. Neurology 1968; 18~807-12. 3 Tanphaichitra D. Tropical disease in the immunocompromised host: melioidosis and pythiosis. Rev Infect Dis 1989; l l ( 7 ) SuppL1629-43. 4 Martin FP, Lukeman JM, Ranson RF, et al. Mucormycosis of the central nervous system associated with thrombosis of the internal carotid artery. J Pediatr 1954; 44:437-42. 5 Houston MC, Marion JM, Curry WA. Necrotizing arteritis associated with blastomycosis. South Med J 1986; 79(4):519-20. 6 Edelson TM, McNatt EN, Porro RS. Candida meningitis with cerebral arteritis. NY J Med 1975; 75:900-4. 7 Tuder RM, el-Ibrahim R, Godoy CE, De-Brito T. Pathology of the human pulmonary paracoccidioidomycosis. Mycopathologia 1985; 92(3):179-88. 8 Scaravilli F. Parasitic and fungal infections of the nervous system. In: Hume-Adam H, Corsellis JAN, Duchen LW, eds. Greenfield's neuropathology, 4th edn. London: Edward Arnold, 1984:304-37. 9 Kirkpatrick JB. Neurologic infections due to bacteria, fungi and parasites. In: Davis RL, Robertson DM, eds. Textbook of neuropathology, 2nd edn. Baltimore: Williams & Wilkins, 1991;719-803. 0 Hay RJ. Fungal infections. Med Int (Middle Eastern Edition) 1988; 53:2203-9. 1 Visudhiphan P, Bunyaratavej S, Khantanaphar S. Cerebral aspergillosis. Report of three cases. J Neurosurg 1973; 38:472-6. 2 Shapiro K, Tabbaddor K. Cerebral aspergillosis. Surg Neurol 1975; 4:465-71. 3 Freely M, Steinberg M. Aspergillus infection complicating trans-sphenoidal Yttrium-90 pituitary implant. Report of two cases. J Neurosurg 1977; 46530-2. 14 Galassi E, Pozzati E, Poppi M, Vinci A. Cerebral aspergillosis following intracranial surgery. Case report. J Neurosurg 1978; 49:308-11. 15 Morioka T, Tashima T, Nagata S, Fukui M, Hasuo K. Cerebral aspergillosis after burr-hole surgery for chronic subdural haematoma: case report. Neurosurgery 1990; 26:332-5.
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Rewati Raman Sharma, Nihal T. Gurusinghe t3 Patrick G. Lynch
16 Piotrowski WP, Pilz P, Chuang IH. Subarachnoid haemorrhage caused by a fungal aneurysm of the vertebral artery as a complication of intracranial aneurysm clipping. J Neurosurg 1990; 73:962-4. 17 Friedman AH, Bullitt E. Fungal infections. In: Wilkins RH, Rengachary SS, eds. Neurosurgery. New York McGraw-Hill, 19852002- 10. 18 Mukoyama M, Gimple K, Posster GM. Aspergillosis of the central nervous system. Report of a brain abscess due to A. furnigatus and review of the literature. Neurology 1969; 19:967-74. 19 Lynch PG. CNS aspergillosis following surgical treatment of Aspergillus flavus infection of the left maxillary antrum. 1978. Unpublished case presented to the North of England Neurological Association, Preston, UK. 20 Linares G, McGarry PA, Baker RD. A solid solitary aspergillotic granuloma of the brain. Report of a case due to Aspergillus candidus and review of the literature. Neurology 1971;21 :177-84. 21 Wiles CM, Kocen RS, Symnon L, Scaravilli F. Aspergillus granuloma of the trigeminal ganglion. J Neurol Neurosurg Psychiatry 1981; 44:451-5.
22 Sekhar LN, Dujouny M, Rao GR. Carotid cavernous sinus thrombosis caused by Aspergillus furnigatus. J Neurosurg 1980; 52:120-5. 23 Mohandas S, Ahuja GK, Sood VP, Virmani V. Aspergillosis of the central nervous system. J Neurol Sci 1978; 38:229-33. 24 Epstein SM, Miale TD, Moosy J, Verney E, Sidansky H. Experimental intracranial aspergillosis. J Neuropathol Exp Neurology 1968; 27:473-82. 25 Pillay VKG, Wilson DM, Ing TS, Kark RM. Fungus infection in steroid-treated systemic lupus erythematoSUS. JAMA 1968; 205:261-5. 26 Symmers WStC. A case of cerebral chromoblastomycosis (cladosporiosis) occurring in Britain as a complication of polyarteritis treated with cortisone. Brain 1960; 83:37-51. 27 Meyer RD, Young LS, Armstrong D, Yu B. Aspergillosis complicating neoplastic disease. Am J Med 1973; 74:6-15. 28 Yanai Y, Wakao T, Fukamachi A, Kunimine H. Intracranial granuloma caused by Aspergillus furnigatus. Surg Neurol 1985; 23597-604.
ISSN: 0268-8697 (Print) 1360-046X (Online) Journal homepage: http://www.tandfonline.com/loi/ibjn20
Cerebral infarction due to Aspergillus arteritis following glioma surgery Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch To cite this article: Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch (1992) Cerebral infarction due to Aspergillus arteritis following glioma surgery, British Journal of Neurosurgery, 6:5, 485-490, DOI: 10.3109/02688699208995040 To link to this article: http://dx.doi.org/10.3109/02688699208995040
Published online: 06 Jul 2009.
Submit your article to this journal
Article views: 11
View related articles
Citing articles: 1 View citing articles
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ibjn20 Download by: [Universite Laval]
Date: 08 April 2016, At: 12:14
British Journal of Neurosurgery (1992) 6,485-490
SHORT REPORT
Cerebral infarction due to Aspergillus arteritis following glioma surgery REWATI RAMAN SHARMA, NIHAL T. GURUSINGHE & PATRICK G. LYNCH
Downloaded by [Universite Laval] at 12:14 08 April 2016
Royal Preston Hospital, Preston, L a n a PR2 4HT, UK
Abstract Cerebral infarction due to fungal arteritis is an uncommon complication of neurosurgical operations and adjuvant immunosuppressive therapy, including long-term steroids. If unrecognized, the neurological deterioration which ensues may be mistreated by increasing the dose of steroids. A case of a 38-year-old Caucasian male who had no obvious immune deficiency or fungal infection prior to a craniotomy for cerebral tumour is described in whom perioperative aspergillus infection resulted in cerebral arteritis and extensive cerebral infarction with a fatal outcome. Long-term steroid therapy used in the management of cerebral tumours may carry an increased risk of systemic or cerebral fungal infection. The possibility of cerebral mycosis (arteritis) and dangers of its non-recognition are highlighted.
Key words: Cerebral aspergillosis, cerebral infarction, fungal arteritis, neurosurgery, postoperative complications, steroids.
Introduction Fungal arteritis is a rare disease caused by a variety of fungal infections, e.g. aspergillosis,' phycomycosis,* pythiosis: mucormycosis$ blastomycosis,5 candidosis6 and paracoccidiodomycosis.' Cerebral fungal arteritis (CFA) is usually due to opportunist pathogenic ubiquitous true hyphal fungi such as Aspergillus and Phycomy~etes.~,~ These infections are more prone to occur in patients whose resistance to infection is lowered by a co-existing condition such as diabetes mellitus, chronic pulmonary disease or malignant disease, especially leukaemia or lymphoma.8-10 In addition, patients treated with ACTH, steroids, antibiotics and cytotoxic drugs and drug abusers seem to be particularly susceptible.8-'0 Steroid therapy is used in many neurological and neurosurgical disorders and carries an increased risk of fungal infection. A literature search revealed only seven cases of cerebral
aspergillosis following neurosurgical procedures.l1-I6 We review these cases and present a case of CFA due to Aspergillus infection causing extensive cerebral arterial thrombosis and massive haemorrhagic infarction following excision of a glioma in a healthy young male who had no systemic disease or preoperative fungal infection.
Case report A 38-year-old right-handed previously healthy male presented with a 4-month history of episodic generalized seizures. General physical examination and neurological assessment showed normal findings with no evidence of raised intracranial pressure. Routine haematological and biochemical tests were normal. Computed axial tomographic (CT) scans of the head showed a low attenuation nonenhancing intrinsic lesion in the left fronto-
485
Downloaded by [Universite Laval] at 12:14 08 April 2016
486
Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch
temporal region with slight compression of the left lateral ventricle. The radiological diagnosis was of a low-grade astrocytoma. Cerebral angiography confirmed the C T findings of an intrinsic avascular space occupying lesion. Steroid therapy (dexamethasone, 16 mglday) was commenced a week prior to operation. A craniotomy was performed and the temporal portion of the tumour excised. The Sylvian fissure and middle cerebral artery were exposed. The histopathology of the tumour revealed a low-grade astrocytoma. No antibiotics were used postoperatively and apart from a transient dysphasia the patient made a good initial recovery. The dosage of the steroid therapy was gradually reduced but during the third postoperative week whilst the patient was awaiting radiotherapy he developed severe headaches, intermittent focal seizures involving the right arm and severe motor dysphasia. Repeat CT revealed cerebral oedema around residual tumour and the dosage of steroids (dexamethasone) was increased. The patient continued to deteriorate, becoming drowsy with the development of a left third nerve palsy and a right hemiplegia. C T now revealed extensive cerebral oedema around residual fronto-temporal tumour with areas of haemorrhage more marked in the left temporal region (Fig. 1). There was a significant shift of midline structures to the right. A further craniotomy was performed and the residual frontal portion of the tumour was excised and a partial frontal lobectomy performed. Despite aggressive therapy the patient continued to deteriorate and death occurred 2 days postoperatively. Post-mortem examination of the brain revealed a ragged cavity in the orbital region of the left frontal lobe. Most of the left temporal lobe had been excised. Sections of the brain in the coronal plane revealed a marked shift of the midline structures from left to right and massive haemorrhagic infarction of the left cerebal hemisphere within the territory of distribution of the left internal carotid and left middle cerebral arteries. The lumina of the left internal carotid artery and the proximal part of the left middle cerebral artery were occluded
FIG. 1. CT brain scan (unenhanced) showing extensive cerebral oedema around residual frontal tumour and haemorrhagic infarction in the left fronto-temporal region.
by thrombus. No residual tumour was seen. Light microscopy of the left middle cerebral artery confirmed the presence of intraluminal thrombus (Fig. 2). Fungal hyphae were seen invading the vessel wall (Fig. 3). Fungal stains showed branching septate hyphae compatible with Aspergillus, probably A. fumigatus (Fig. 4).
Discussion Opportunistic fungal infections such as candidiasis, cryptococcosis, aspergillosis and zygomycosis are now a familiar problem in many parts of the world and may account for much morbidity and mortality in immunosuppressed patients, including those with AIDS.*O However, cerebral mycosis is a rare condition and in our case it was caused by Aspergillus (probably fumigatus). The other usual human pathogenic Aspergillus species are A. niger, A . flavus and A. oxyzae. Aspergillus is ubiquitous and has branching septate hyphae of from 3 to 12 p m diameter.
Downloaded by [Universite Laval] at 12:14 08 April 2016
Cerebral infarction due to Aspergillus arteritis
FIG. 2. Left middle cerebral artery showing intraluminal thrombus and inflammation of the vessel wall. H&E.
487
X 42.
FIG. 3. Left middle cerebral artery showing virtual destruction of the tunica media with radial extension of fungus into the vessel lumen. Grocott. x 198.
Cerebral infection is usually secondary to a primary focus elsewhere in the body. Infection reaches the brain either directly from the nasal sinuses via vascular channels or is blood-borne from the lungs and gastrointestinal t r a ~ t . ~ - ~ O The , l ~ - infection l~ may also be airborne, contaminating the tissues during a
neurosurgical In our case, the possibilities are either airborne spores reaching the operative field and germinating or spread from the sphenoid sinus via the internal carotid artery. The former possibility is more likely as at autopsy the only evidence of the fungal infection was in the operative field.
Downloaded by [Universite Laval] at 12:14 08 April 2016
488
Rewati Raman Sharma, Nihal T. Gurusinghe & Patrick G. Lynch
FIG.4. Photomicrograph showing branched septate hyphae characteristic of Aspergillus species. Grocott.
Fungi infecting the brain are found in three major forms with distinctive clinicomorphologic syndrome^.^ (1) Acute and chronic leptomeningitis produced by pure yeasts (blastomycosis, cryptococcosis and histoplasmosis). These small sized fungi gain access to the cerebral microcirculation from which they infect the subarachnoid space. (2) Abscesses produced by pseudohyphae (candidiasis) which occlude cerebral arterioles because of their intermediate size (larger than yeasts yet smaller than true hyphae) and result in adjacent tissue necrosis and microabscess formation. ( 3 ) Cerebral infarction produced by branched hyphal fungi (aspergillosis and phycomycosis) which mainly obstruct the large and intermediate sized cerebral arteries. The hyphae invade the vessel walls causing cerebral arterial thrombosis, associated cerebral infarction and adjacent cerebritis. The evolving haemorrhagic cerebral infarcts may convert into septic infarcts with associated cerebritis and abscesses. Some of these features were seen in the present case. The left internal carotid and left middle cerebral arteries were thrombosed and fungal hyphae were seen in the vessel wall and lumen. Owing to its variable clinical manifestations, cerebral mycosis is difficult to diagnosis in life.
X
396.
It may present with symptoms suggestive of meningitis, a space occupying lesion (abscess and granuloma) or as a sudden cerebrovascular event (fungal aneurysm causing subarachnoid haemorrhage and CFA haemorrhagic cerebral i n f a r ~ t i o n ) . ~ - ' ~ , ' The ~-~~ appearance of neurological symptoms in cases of an obvious extracranial fungal infection, debilitating neoplastic disorders, HIV infection, or immunosuppression may suggest cerebral m y ~ o s i s . ' ~ , * ~It- *is ~ well known that disseminated aspergillosis with involvement of the central nervous system is seen in patients debilitated by neoplastic disorders, and Meyer et aL2' found nine cases of cerebral aspergillosis out of 93 patients suffering from various systemic malignant tumours. Neurological deterioration due to superadded fungal infection following neurosurgical procedures presents a special problem. The fungal infection may go unrecognized and the neurological deterioration may be mistreated by immunosuppressive d p g s (steroids). Only seven cases of cerebral aspergillosis following neurosurgical procedures have been reported in the literature."-'6 In four patients, direct extension of Aspergillus from the paranasal sinuses was considered,13.14and in this context it is interesting to note that in one unpublished
Downloaded by [Universite Laval] at 12:14 08 April 2016
Cerebral infarction due to Aspergillus arteritis case cerebral aspergillosis occurred following surgical exploration of an infected maxillary sinus.19 In four patients, the portal of entry remained u r ~ c e r t a i n ~ ~as~in~ our ~ J ~case. J ~ It is, however, possible that fungi were introduced during the neurosurgical procedures and predisposing conditions (antibiotic therapy, diabetes mellitus, steroid therapy) accelerated the progress of infection. In our case no antibiotics were used and the patient did not have diabetes mellitus. Among the reported cases of Aspergillus infection following neurosurgical procedures (craniopharyngioma," thalamic glioma,I2 pituitary turn our^,^^ olfactory groove meningi~ma,'~ subdural haematoma,I5 internal carotid artery aneurysm16), cerebral aspergillosis presented as granuloma in three in one of which fungal abscess was a~s0ciated.l~Two patients presented with cerebral in one of which abscess was associated.16 One patient had abscess en~ephalitis,'~ and another cerebral infarction caused by va~culitis.'~ In the latter, the presence of a screw in the sphenoid sinus (transphenoidal yttrium-90 pituitary implant) fostered the Aspergillus infection and the foreign material provided an access route to the subarachnoid space. Diagnosis is made by culture on special tissue media of tissue obtained at operation or autopsy.2a The use of antifungal antibiotics (amphotericin-B, flucytosine and rifampicin) combined with aggressive surgical removal of an intracranial abscess or granuloma is the recommended treatment for cerebral aspergillosis. Nevertheless, the outcome for patients with postoperative aspergillosis remains poor. Of eight patients including the present case, only two have ~ u r v i v e d . In ~ ~cases , ~ ~ of CFA where surgery is inappropriate or contraindicated, reliance has to be placed on antifungal antibiotics and supportive therapy. Acknowledgements We gratefully acknowledge the technical contributions of Pamela Thompson, Angela Clayton and Ruth Sharples to this work. The authors also express their appreciation to
489
Pauline Ashcroft for secretarial help in the preparation of the manuscript and to the Medical Illustration Department, Royal Preston Hospital for the photographs.
Address for correspondence: Dr Patrick G . Lynch, Neuropathology Laboratory, Royal Preston Hospital, PO Box 202, Sharoe Green Lane, Fulwood, Preston, Lancs PR2 4HG, UK. References 1 Fernando S S , Lauer CS. Aspergtllus fumigatus infection of the optic nerve with mycotic arteritis of cerebral vessels. Histopathology 1982; 6(2):227-34. 2 Carpenter DF, Brubaker LH, Powell RD, et al. Phycomycotic thrombosis of the basilar artery. Neurology 1968; 18~807-12. 3 Tanphaichitra D. Tropical disease in the immunocompromised host: melioidosis and pythiosis. Rev Infect Dis 1989; l l ( 7 ) SuppL1629-43. 4 Martin FP, Lukeman JM, Ranson RF, et al. Mucormycosis of the central nervous system associated with thrombosis of the internal carotid artery. J Pediatr 1954; 44:437-42. 5 Houston MC, Marion JM, Curry WA. Necrotizing arteritis associated with blastomycosis. South Med J 1986; 79(4):519-20. 6 Edelson TM, McNatt EN, Porro RS. Candida meningitis with cerebral arteritis. NY J Med 1975; 75:900-4. 7 Tuder RM, el-Ibrahim R, Godoy CE, De-Brito T. Pathology of the human pulmonary paracoccidioidomycosis. Mycopathologia 1985; 92(3):179-88. 8 Scaravilli F. Parasitic and fungal infections of the nervous system. In: Hume-Adam H, Corsellis JAN, Duchen LW, eds. Greenfield's neuropathology, 4th edn. London: Edward Arnold, 1984:304-37. 9 Kirkpatrick JB. Neurologic infections due to bacteria, fungi and parasites. In: Davis RL, Robertson DM, eds. Textbook of neuropathology, 2nd edn. Baltimore: Williams & Wilkins, 1991;719-803. 0 Hay RJ. Fungal infections. Med Int (Middle Eastern Edition) 1988; 53:2203-9. 1 Visudhiphan P, Bunyaratavej S, Khantanaphar S. Cerebral aspergillosis. Report of three cases. J Neurosurg 1973; 38:472-6. 2 Shapiro K, Tabbaddor K. Cerebral aspergillosis. Surg Neurol 1975; 4:465-71. 3 Freely M, Steinberg M. Aspergillus infection complicating trans-sphenoidal Yttrium-90 pituitary implant. Report of two cases. J Neurosurg 1977; 46530-2. 14 Galassi E, Pozzati E, Poppi M, Vinci A. Cerebral aspergillosis following intracranial surgery. Case report. J Neurosurg 1978; 49:308-11. 15 Morioka T, Tashima T, Nagata S, Fukui M, Hasuo K. Cerebral aspergillosis after burr-hole surgery for chronic subdural haematoma: case report. Neurosurgery 1990; 26:332-5.
Downloaded by [Universite Laval] at 12:14 08 April 2016
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Rewati Raman Sharma, Nihal T. Gurusinghe t3 Patrick G. Lynch
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