Aspergilose invasiva do seio esfenoidal e paralisia do sexto nervo

Lana-Peixoto, Maria Inês de Vilhena; Lana-Peixoto, Marco Aurélio

doi:10.1590/S0004-282X1992000100019

Resumos

A aspergilose do seio esfenoidal é doença rara e pode se apresentar sob diferentes formas clínicas devido a envolvimento de. diversas estruturas anatomicamente adjacentes ao seio esfenoidal. Relatamos o caso de uma paciente com 74 anos de idade, diabética, com paralisia do sexto nervo esquerdo secundária a aspergilose do seio esfenoidal. Não havia história de cefaléia ou de queixas sugestivas de alergia respiratória. A tomografia computadorizada revelou lesão etmoídeo-esfenoidal à esquerda, com presença de imagem cálcica em seu interior e destruição óssea. A paciente foi submetida a cirurgia com retirada de material necrótico e debridamento da lesão, seguida de tratamento com anfote-ricina B e 5-fluorocitosina. Exame histológico revelou a presença de hifas sugestivas de Aspergilius sp. Após três meses de tratamento a paciente apresentou recuperação total da paresia do nervo abducente. O diagnóstico clínico pré-operatório de aspergilose do seio esfenoidal é difícil. No entanto, a presença de imagem cálcica ou de densidade metálica à radiografia simples de crânio ou à tomografia computadorizada sugere fortemente o diagnóstico. O exame hihstológico revela a presença de hifas dicotomatosas em 45,0 típicas do Aspergilius. O tratamento inclui excisão e debridamento da lesão seguida do uso de anfo-tericina B associada a 5-fluorocitosina ou rifampicina.

aspergilose; seio esfenoidal; VI nervo craniano

A case of aspergillosis of the sphenoid sinus manifested as an isolated sixth nerve palsy occurred in a 74-year old diabetic woman who had no complaints of headache or symptoms suggestive of sinusitis. A CT scan demonstrated a large mass occupying the sphenoid and ethmoid sinuses extending posteriorly to the clivus. There was a calcific density within the opacified sinus and bony erosion of the sphenoid walls and the sella turcica. The patient underwent a sublabial transseptal sphenoidotomy with removal of necrotic material and debridment of the surrounding tissue. Histologic examination revealed granulation tissue with chronic inflammatory cells and abundant dichotomously branching hyphae. Postoperatively the patient was given amphotericin B and 5-fluorocytosine. Three months later the sixth nerve palsy had completely cleared and the patient had no other complaint. Sphenoid sinus aspergillosis is a rare disease and may have variable clinical manifestations according to involvement of different structures located closely to the sinus. Our patient developed an isolated sixth nerve palsy which was at onset considered to be caused by diabetes. Computerized tomography scans disclosed abnormalities strongly indicative of invasive aspergillosis. It illustrates the need of appropriate work-up in cases of an isolated sixth nerve palsy even in patients with diabetes or other risk factors.

aspergillosis; sphenoid sinus; VI cranial nerve

Maria Inês de Vilhena Lana-Peixoto; Marco Aurélio Lana-Peixoto

Professor Adjunto de Neurologia, Faculdade de Medicina, Universidade Federal de Minas Gerais (UFMG)

RESUMO

A aspergilose do seio esfenoidal é doença rara e pode se apresentar sob diferentes formas clínicas devido a envolvimento de. diversas estruturas anatomicamente adjacentes ao seio esfenoidal. Relatamos o caso de uma paciente com 74 anos de idade, diabética, com paralisia do sexto nervo esquerdo secundária a aspergilose do seio esfenoidal. Não havia história de cefaléia ou de queixas sugestivas de alergia respiratória. A tomografia computadorizada revelou lesão etmoídeo-esfenoidal à esquerda, com presença de imagem cálcica em seu interior e destruição óssea. A paciente foi submetida a cirurgia com retirada de material necrótico e debridamento da lesão, seguida de tratamento com anfote-ricina B e 5-fluorocitosina. Exame histológico revelou a presença de hifas sugestivas de Aspergilius sp. Após três meses de tratamento a paciente apresentou recuperação total da paresia do nervo abducente. O diagnóstico clínico pré-operatório de aspergilose do seio esfenoidal é difícil. No entanto, a presença de imagem cálcica ou de densidade metálica à radiografia simples de crânio ou à tomografia computadorizada sugere fortemente o diagnóstico. O exame hihstológico revela a presença de hifas dicotomatosas em 45,0 típicas do Aspergilius. O tratamento inclui excisão e debridamento da lesão seguida do uso de anfo-tericina B associada a 5-fluorocitosina ou rifampicina.

Palavras chave: aspergilose, seio esfenoidal, VI nervo craniano.

SUMMARY

A case of aspergillosis of the sphenoid sinus manifested as an isolated sixth nerve palsy occurred in a 74-year old diabetic woman who had no complaints of headache or symptoms suggestive of sinusitis. A CT scan demonstrated a large mass occupying the sphenoid and ethmoid sinuses extending posteriorly to the clivus. There was a calcific density within the opacified sinus and bony erosion of the sphenoid walls and the sella turcica. The patient underwent a sublabial transseptal sphenoidotomy with removal of necrotic material and debridment of the surrounding tissue. Histologic examination revealed granulation tissue with chronic inflammatory cells and abundant dichotomously branching hyphae. Postoperatively the patient was given amphotericin B and 5-fluorocytosine. Three months later the sixth nerve palsy had completely cleared and the patient had no other complaint. Sphenoid sinus aspergillosis is a rare disease and may have variable clinical manifestations according to involvement of different structures located closely to the sinus. Our patient developed an isolated sixth nerve palsy which was at onset considered to be caused by diabetes. Computerized tomography scans disclosed abnormalities strongly indicative of invasive aspergillosis. It illustrates the need of appropriate work-up in cases of an isolated sixth nerve palsy even in patients with diabetes or other risk factors.

Key words: aspergillosis, sphenoid sinus, VI cranial nerve.

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Dra. Maria Inês de Vilhena Lana-Peixoto Departamento de Neurologia, Faculdade de Medicina, UFMG - Av. Alfredo Balena 190 - 30130 Belo Horizonte MG - Brasil.

1. Adams VF. Infection involving the ethmoid, maxillary and sphenoid sinuses and the orbit due to Aspergillus fumigatus. Arch Surg 1933, 26:999-1009.
2. Ardouin M. Urvoy M, Bourdiniere J. Le Clech G, Vallee B, Scarabin JM. Aspergillose pseudo-tumorale du sinus shénoïdal (ou l'etiologie d'une paralysie unilatérale du III). J Fr Ophtalmol 1979, 2:626-627.
3. Axelson H. Carlsoo B, Weibring J, Winblad B. Aspergillosis of the maxillary sinus. Acta Otolaryngol 1978, 86:303-308.
4. Bennett JE. Chemotherapy of systemic mieosis (First of two parts). N Engl J Med 1974, 290:30-32.
5. Fuchs HA, Evans RM, Gregg CR. Invasive aspergillosis of the sphenoid sinus manifested as a pituitary tumor. Southern Med J 1985, 78:1366-1367.
6. Fujiwara S, Fujino H, Nogami K, Nishio S, Fukui M. Aspergillosis of the sphenoid sinus with cavernous sinus syndrome. Neuroradiology 1989, 31:443-446.
7. Goldhammer Y, Smith JF, Yates RM. Mycotic intrasellar abscess. Trans Am Ophthalmol Soc 1974, 72:65-78.
8. Goldstein MF, Atlkins PC, Cogen FC, Kornstein MJ, Levine RS, Zweirman B. Allergic aspergillus sinusitis; J Allergy Clin Immuno 1985, 76:515-524.
9. Greevic, Matthews WF. Pathologic changes in acute disseminated aspergillosis: particular involvement of the central nervous system. Am J Clin Pathol 1959, 32:536-551.
10. Holt GR, Standefer JA, Brown WE, Gates GA. Infectious diseases of the sphenoid sinus. Laryngoscope 1984, 94:330-335.
11. Horton WD, Osguthorpe DJ. CT findings in sphenoid sinus aspergillosis. Otolaringol Head Neck Surg 1989, 100:606-609.
12. Jahrsdoerfer RA, Ejercits VS, Johns MME, Cantrell RM, Sydnor JB. Aspergillosis of the nose and paranasal sinus. Am J Otolaryngol 1979, 1:6-14.
13. Katzenstein A, Sale SR, Greenberger PA. Allergic aspergillus sinusitis. J Allergy Clin Immunol 1983, 72:89-93.
14. Lemoine JJ, Le Rebeller MJ, Bagot D'Arc M. Ophtalmoplegie amaurotique a bascule associée a une aspergillose du sinus sphenoidal. Bull Soc Ophtalmol Fr 1984, 84:787-790.
15. Lowe JT, Hudson WR. Rhinocerebral phycomycoses and internal carotid artery thrombosis. Arch Otolaryngol 1975, 101:100-103.
16. Mahgoub ELS. Mycological and serological studies on aspergillus flavus isolated from paranasal aspergilloma in Sudan. J Trop Med Hyg 1971, 74:162-165.
17. McGill TJ, Simpson G, Healy GB. Fulminant aspergillosis of the nose and paranasal sinuses: a new clinical entity. Laryngoscope 1980, 90:748-754.
18. McGuirt WF, Harril JA. Paranasal sinus aspergillosis. Laryngoscope 1979, 89:1563-1568.
19. Merkle D. Yarington CT, Winterbauer RH. Aspergillosis of the maxillary sinuses in otherwise healthy patients. Laryngoscope 1985, 95:776-779.
20. Miglets AW, Saunders WH, Leona A. Aspergillosis of the sphenoid sinus. Arch Otolaryngol 1978. 104:47-50.
21. Milosev B, Mahgoub ELS, Abdel Aal AO, El Hanan MA. Primary aspergilloma of the paranasal sinuses in the Sudan. Br J Surg 1969, 56:1321-137.
22. Rommett JL. Newman RK. Aspergillosis of the nose and paranasal sinuses. Laryngoscope 1982, 92:764-766.
23. Sekhar LN. Dujovny M, Rao GR. Carotid-cavernous sinus thrombosis caused by Aspergillus fumigatus: case report. J Neurosurg 1980, 52:120-125.
24. Stammberger H, Jakse R. Beaufort F. Aspergillosis of the paranasal sinuses: X-ray diagnosis, histopathology and clinical aspects. Ann Otol Rhinol Laryngol 1984, 93: 251-256.
25. Stevens MH. Primary fungal infections of the paranasal sinuses. Am J Otolaryngol 1981. 2:348-351.
26. Tveten L. Cerebral mycosis: a clinico-pathological report of four cases. Acta Neurol Scand 1965. 41:19-33.
27. Veress B. Malik OA. El Tayeb AA, El Daoud S, Mahgoub ES, El Hassan AM. Further observations on the primary paranasal aspergillus granuloma in the Sudan: a morphological study of 46 cases. Am J Trop Med Hyg 1973, 22:765-772.
28. Viollier AF. Peterson DE. DeJongh CA, Newman KA, Gray WG, Sutherland JC, Moody MA, Schimpff SC. Aspergillus sinusitis in cancer patients. Cancer 1986, 58:366-371.
29. Warder FR, Chickes PG. Hudson WR. Aspergillosis of the paranasal sinuses. Arch Otolaryngol 1975. 101:683-685.
30. Yu VL, Wagner GE, Shadomy S. Sino-orbital aspergillosis treated with combination antifungal therapy. JAMA 1980, 244:814-815.
31. Yumots E, Kitani S, Okamura H, Yanagihara N. Sino-orbital aspergillosis associated with total ophthalmoplegia. Laryngoscope 1985, 95:190-192.

Aspergilose invasiva do seio esfenoidal e paralisia do sexto nervo

Invasive aspergillosis of the sphenoid sinus manifested as an isolated sixth nerve palsy

Datas de Publicação

Publicação nesta coleção
22 Fev 2011
Data do Fascículo
Mar 1992

This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

[1] 1. Adams VF. Infection involving the ethmoid, maxillary and sphenoid sinuses and the orbit due to Aspergillus fumigatus. Arch Surg 1933, 26:999-1009.

[2] 2. Ardouin M. Urvoy M, Bourdiniere J. Le Clech G, Vallee B, Scarabin JM. Aspergillose pseudo-tumorale du sinus shénoïdal (ou l'etiologie d'une paralysie unilatérale du III). J Fr Ophtalmol 1979, 2:626-627.

[3] 3. Axelson H. Carlsoo B, Weibring J, Winblad B. Aspergillosis of the maxillary sinus. Acta Otolaryngol 1978, 86:303-308.

[4] 4. Bennett JE. Chemotherapy of systemic mieosis (First of two parts). N Engl J Med 1974, 290:30-32.

[5] 5. Fuchs HA, Evans RM, Gregg CR. Invasive aspergillosis of the sphenoid sinus manifested as a pituitary tumor. Southern Med J 1985, 78:1366-1367.

[6] 6. Fujiwara S, Fujino H, Nogami K, Nishio S, Fukui M. Aspergillosis of the sphenoid sinus with cavernous sinus syndrome. Neuroradiology 1989, 31:443-446.

[7] 7. Goldhammer Y, Smith JF, Yates RM. Mycotic intrasellar abscess. Trans Am Ophthalmol Soc 1974, 72:65-78.

[8] 8. Goldstein MF, Atlkins PC, Cogen FC, Kornstein MJ, Levine RS, Zweirman B. Allergic aspergillus sinusitis; J Allergy Clin Immuno 1985, 76:515-524.

[9] 9. Greevic, Matthews WF. Pathologic changes in acute disseminated aspergillosis: particular involvement of the central nervous system. Am J Clin Pathol 1959, 32:536-551.

[10] 10. Holt GR, Standefer JA, Brown WE, Gates GA. Infectious diseases of the sphenoid sinus. Laryngoscope 1984, 94:330-335.

[11] 11. Horton WD, Osguthorpe DJ. CT findings in sphenoid sinus aspergillosis. Otolaringol Head Neck Surg 1989, 100:606-609.

[12] 12. Jahrsdoerfer RA, Ejercits VS, Johns MME, Cantrell RM, Sydnor JB. Aspergillosis of the nose and paranasal sinus. Am J Otolaryngol 1979, 1:6-14.

[13] 13. Katzenstein A, Sale SR, Greenberger PA. Allergic aspergillus sinusitis. J Allergy Clin Immunol 1983, 72:89-93.

[14] 14. Lemoine JJ, Le Rebeller MJ, Bagot D'Arc M. Ophtalmoplegie amaurotique a bascule associée a une aspergillose du sinus sphenoidal. Bull Soc Ophtalmol Fr 1984, 84:787-790.

[15] 15. Lowe JT, Hudson WR. Rhinocerebral phycomycoses and internal carotid artery thrombosis. Arch Otolaryngol 1975, 101:100-103.

[16] 16. Mahgoub ELS. Mycological and serological studies on aspergillus flavus isolated from paranasal aspergilloma in Sudan. J Trop Med Hyg 1971, 74:162-165.

[17] 17. McGill TJ, Simpson G, Healy GB. Fulminant aspergillosis of the nose and paranasal sinuses: a new clinical entity. Laryngoscope 1980, 90:748-754.

[18] 18. McGuirt WF, Harril JA. Paranasal sinus aspergillosis. Laryngoscope 1979, 89:1563-1568.

[19] 19. Merkle D. Yarington CT, Winterbauer RH. Aspergillosis of the maxillary sinuses in otherwise healthy patients. Laryngoscope 1985, 95:776-779.

[20] 20. Miglets AW, Saunders WH, Leona A. Aspergillosis of the sphenoid sinus. Arch Otolaryngol 1978. 104:47-50.

[21] 21. Milosev B, Mahgoub ELS, Abdel Aal AO, El Hanan MA. Primary aspergilloma of the paranasal sinuses in the Sudan. Br J Surg 1969, 56:1321-137.

[22] 22. Rommett JL. Newman RK. Aspergillosis of the nose and paranasal sinuses. Laryngoscope 1982, 92:764-766.

[23] 23. Sekhar LN. Dujovny M, Rao GR. Carotid-cavernous sinus thrombosis caused by Aspergillus fumigatus: case report. J Neurosurg 1980, 52:120-125.

[24] 24. Stammberger H, Jakse R. Beaufort F. Aspergillosis of the paranasal sinuses: X-ray diagnosis, histopathology and clinical aspects. Ann Otol Rhinol Laryngol 1984, 93: 251-256.

[25] 25. Stevens MH. Primary fungal infections of the paranasal sinuses. Am J Otolaryngol 1981. 2:348-351.

[26] 26. Tveten L. Cerebral mycosis: a clinico-pathological report of four cases. Acta Neurol Scand 1965. 41:19-33.

[27] 27. Veress B. Malik OA. El Tayeb AA, El Daoud S, Mahgoub ES, El Hassan AM. Further observations on the primary paranasal aspergillus granuloma in the Sudan: a morphological study of 46 cases. Am J Trop Med Hyg 1973, 22:765-772.

[28] 28. Viollier AF. Peterson DE. DeJongh CA, Newman KA, Gray WG, Sutherland JC, Moody MA, Schimpff SC. Aspergillus sinusitis in cancer patients. Cancer 1986, 58:366-371.

[29] 29. Warder FR, Chickes PG. Hudson WR. Aspergillosis of the paranasal sinuses. Arch Otolaryngol 1975. 101:683-685.

[30] 30. Yu VL, Wagner GE, Shadomy S. Sino-orbital aspergillosis treated with combination antifungal therapy. JAMA 1980, 244:814-815.

[31] 31. Yumots E, Kitani S, Okamura H, Yanagihara N. Sino-orbital aspergillosis associated with total ophthalmoplegia. Laryngoscope 1985, 95:190-192.