British Journal of Neurosurgery (1992) 6, 381-384
SHORT REPORT
An ‘empty’ sphenoid mucocele
Br J Neurosurg Downloaded from informahealthcare.com by Queen's University on 01/04/15 For personal use only.
CHARLES H. G. DAVIS, MICHAEL SMALL* & VALERIE LUNDt
Departments of Neurological Surgey and *Ear, Nose and Throat Surgey , Royal Preston Hospital, Lancashire and flnstitute of L ayngology and Otology, London, UK
Abstract Sphenoid sinus mucoceles behave as mass lesions producing erosion of adjacent bone and cranial nerve palsies. The probable cause is obstruction of the ostia of the sinus. We describe a case which was ‘cured’ inadvertently by a nasal polypectomy. Key words: Mucocele, sphenoid sinus.
Introduction
Sphenoid mucoceles are rare ‘tumours’ causing enlargement of the sphenoid sinus.’-14 In this case, inadvertent drainage by nasal polypectomy cured the condition. Case report
A 65-year-old man with a 40-year history of chronic rhinitis presented with a 3-week history of diplopia in 1987. The patient had undergone nasal polypectomy in 1959 when he was noted to have a large number of nasal polypi in both nostrils. In 1967 he had a second
nasal polypectomy and at surgery it was noted that the normal anatomical landmarks in the nose, particularly the middle turbinates, were no longer present owing to previous surgery. On admission the patient was alert and general examination was essentially normal. Neurological examination revealed a partial right sixth nerve palsy; however, there were no other localizing neurological signs or signs of raised intracranial pressure. ENT examination revealed large bilateral nasal polypi filling both
nostrils and extending back to the posterior choanae. His nasopharynx was clear.
Investigations Routine blood tests and ESR were normal. Skull X-ray (Fig. l), C T (Fig. 2) and carotid angiography revealed the presence of a large round mass occupying the base of the skull and replacing the sphenoid sinus. The mass centred more to the right than left and had partially destroyed the sella turcica. The attenuation of the mass on C T suggested cystic contents, and a provisional diagnosis of sphenoid mucocele or cystic pituitary tumour was made.
First operation Under general anaesthetic large bilateral ethmoidal polypi were excised and the patient made an uneventful recovery.
Second operation Two weeks later, under general anaesthetic a
38 1
Br J Neurosurg Downloaded from informahealthcare.com by Queen's University on 01/04/15 For personal use only.
382
Charles H. G. Davis, Michael Small 6'Valerie Lund
FIG.2.Off-centre sagittal CT showing sphenoid mucocele prior to nasal polypectomy.
standard sublabial submucous transsphenoidal approach was performed. At the anterior part of the mass was a thin layer of expanded and thinned bone which was easily broached. On entering the sphenoid sinus the operator was surprised to find it completely empty. On the left side the internal carotid artery could be seen glistening and covered in a thin layer of mucosa. Palpation of the posterior part of the expanded sphenoid sinus revealed that a large part of the clivus had been eroded. The anterior part of the decompression was widened and a biopsy specimen of the lining confirmed the presence of inflammatory tissue.
FIG.3. CT,axial view, showing empty sphenoid mucocele.
Discussion This case illustrates one of the causes of this rare condition, i.e. obstruction of the sphenoid ostia due to chronic airway inflammatory disease, although an obstructive tumour may have a similar e f f e ~ t . ~ - ~Bone * ~ - ' resorption ~ with elevated mucocele levels of prostaglandin E2 and collagenase subsequently occur15 and the expanded mucocele acts as a mass. Sphenoid sinus mucoceles are rare with less than 100 described in the literature.8 Presenting symptoms include headache and ocular palsies which may be intermittent8J4 and the sixth
Br J Neurosurg Downloaded from informahealthcare.com by Queen's University on 01/04/15 For personal use only.
Sphenoid mucocele nerve is most commonly involved. The mucoceles contain sterile mucoid secretions and the normal mucosa is destroyed. In this case, unfortunately, the patient did not undergo CT between the first and second procedures. In retrospect the polypectomy was in fact enough of a decompression to empty the mucocele, thus illustrating the aetiology. In 1983 Som et al. described a sphenoid sinus pneumocele" and in 1982 Hirst et al., described a pneumosinus dilatans of the sphenoid sinus due to an optic canal meningioma.16 It may be that mucoceles, pneumoceles, and pneumosinus dilatans are all related conditions caused by permanent or intermittent obstruction of the paranasal sinuses. Address for correspondence: Mr C . H. G . Davis, Department of Neurosurgery, Royal Preston Hospital, Sharoe Green Lane, Preston PR2 4HT, UK.
References 1 Bloom DL. Mucoceles of the maxillary and sphenoid sinuses. Radiology 1965; 851102-10. 2 Close LG, O'Connor WE. Sphenoethmoidalmucoceles with intracranial extension. Otolaryngol Head Neck Surg 1983; 21:350-7.
383
3 Friedman NG, Harrison S. Mucocele of the sphenoidal sinus as a cause of recurrent oculomotor nerve palsy. J Neurol Neurosurg Psychiatry 1979; 33:172-9. 4 Nugent GR, Sprinkle P, Bloor BM. Sphenoid sinus mucoceles. J Neurosurg 1970; 32:443-51. 5 Phelps PD, Toland JA. Mucocele of the sphenoidal sinus eroding the petrous temporal bone. Br J Radio1 1969; 42(503):845-7. 6 Pincus JH, Daroff RB. Sphenoid sinus mucocele: a curable cause of the ophthalmoplegic migraine syndrome. JAMA 1964; 187:459-61. 7 Proetz AW. The sphenoid sinus. BMJ 1948; 2:243-5. 8 Sellars SL, DeVilliers JC. The sphenoid sinus mucocele. J Laryngol Otol 1981; 95493-502. 9 Simms NM, Brown WE, French LA. Mucocele of the sphenoid sinus presenting as an intrasellar mass. J Neurosurg 1970; 32:708-10. 10 Simon HM, Tingwald FR. Syndrome associated with mucocele of the sphenoid sinus: report of two cases and their radiographic findings. Radiology 1955; 64:538-45. 11 Som PM, Sachdev VP, Biller HF. Sphenoid sinus pneumocele. Arch Otolaryngol 1983; 109:761-4. 12 Takahashi M, Jingu K, Nakayama T. Roentgenologic appearances of sphenoethmoidal mucocele. Neuroradi010gy 1973; 6~45-9. 13 Wyllie JW, Kern EB, Djalilian M. Isolated sphenoid sinus lesions. Laryngoscope 1973; 83: 1252-65. 14 Weaver RG, Gates GA. Mucoceles of the sphenoid sinus. Otolaryngol Head Neck Surg 1979; 87:168-73. 15 Lund VJ,Harvey W, Meghju S, Harris M. Prostaglandin synthesis in the pathogenesis of fronto-ethmoidal mucoceles. Acta Otolaryngol (Stockh) 1988; 106:145-5 1. 16 Hirst LW, Miller NR, Hodges FJ, Corbett JJ, Thompson S. Sphenoid pneumosinus dilatans. A sign of meningioma originating in the optic canal. NeuroradiO I O ~ Y 1982; 22~207-10.
SHORT REPORT
An ‘empty’ sphenoid mucocele
Br J Neurosurg Downloaded from informahealthcare.com by Queen's University on 01/04/15 For personal use only.
CHARLES H. G. DAVIS, MICHAEL SMALL* & VALERIE LUNDt
Departments of Neurological Surgey and *Ear, Nose and Throat Surgey , Royal Preston Hospital, Lancashire and flnstitute of L ayngology and Otology, London, UK
Abstract Sphenoid sinus mucoceles behave as mass lesions producing erosion of adjacent bone and cranial nerve palsies. The probable cause is obstruction of the ostia of the sinus. We describe a case which was ‘cured’ inadvertently by a nasal polypectomy. Key words: Mucocele, sphenoid sinus.
Introduction
Sphenoid mucoceles are rare ‘tumours’ causing enlargement of the sphenoid sinus.’-14 In this case, inadvertent drainage by nasal polypectomy cured the condition. Case report
A 65-year-old man with a 40-year history of chronic rhinitis presented with a 3-week history of diplopia in 1987. The patient had undergone nasal polypectomy in 1959 when he was noted to have a large number of nasal polypi in both nostrils. In 1967 he had a second
nasal polypectomy and at surgery it was noted that the normal anatomical landmarks in the nose, particularly the middle turbinates, were no longer present owing to previous surgery. On admission the patient was alert and general examination was essentially normal. Neurological examination revealed a partial right sixth nerve palsy; however, there were no other localizing neurological signs or signs of raised intracranial pressure. ENT examination revealed large bilateral nasal polypi filling both
nostrils and extending back to the posterior choanae. His nasopharynx was clear.
Investigations Routine blood tests and ESR were normal. Skull X-ray (Fig. l), C T (Fig. 2) and carotid angiography revealed the presence of a large round mass occupying the base of the skull and replacing the sphenoid sinus. The mass centred more to the right than left and had partially destroyed the sella turcica. The attenuation of the mass on C T suggested cystic contents, and a provisional diagnosis of sphenoid mucocele or cystic pituitary tumour was made.
First operation Under general anaesthetic large bilateral ethmoidal polypi were excised and the patient made an uneventful recovery.
Second operation Two weeks later, under general anaesthetic a
38 1
Br J Neurosurg Downloaded from informahealthcare.com by Queen's University on 01/04/15 For personal use only.
382
Charles H. G. Davis, Michael Small 6'Valerie Lund
FIG.2.Off-centre sagittal CT showing sphenoid mucocele prior to nasal polypectomy.
standard sublabial submucous transsphenoidal approach was performed. At the anterior part of the mass was a thin layer of expanded and thinned bone which was easily broached. On entering the sphenoid sinus the operator was surprised to find it completely empty. On the left side the internal carotid artery could be seen glistening and covered in a thin layer of mucosa. Palpation of the posterior part of the expanded sphenoid sinus revealed that a large part of the clivus had been eroded. The anterior part of the decompression was widened and a biopsy specimen of the lining confirmed the presence of inflammatory tissue.
FIG.3. CT,axial view, showing empty sphenoid mucocele.
Discussion This case illustrates one of the causes of this rare condition, i.e. obstruction of the sphenoid ostia due to chronic airway inflammatory disease, although an obstructive tumour may have a similar e f f e ~ t . ~ - ~Bone * ~ - ' resorption ~ with elevated mucocele levels of prostaglandin E2 and collagenase subsequently occur15 and the expanded mucocele acts as a mass. Sphenoid sinus mucoceles are rare with less than 100 described in the literature.8 Presenting symptoms include headache and ocular palsies which may be intermittent8J4 and the sixth
Br J Neurosurg Downloaded from informahealthcare.com by Queen's University on 01/04/15 For personal use only.
Sphenoid mucocele nerve is most commonly involved. The mucoceles contain sterile mucoid secretions and the normal mucosa is destroyed. In this case, unfortunately, the patient did not undergo CT between the first and second procedures. In retrospect the polypectomy was in fact enough of a decompression to empty the mucocele, thus illustrating the aetiology. In 1983 Som et al. described a sphenoid sinus pneumocele" and in 1982 Hirst et al., described a pneumosinus dilatans of the sphenoid sinus due to an optic canal meningioma.16 It may be that mucoceles, pneumoceles, and pneumosinus dilatans are all related conditions caused by permanent or intermittent obstruction of the paranasal sinuses. Address for correspondence: Mr C . H. G . Davis, Department of Neurosurgery, Royal Preston Hospital, Sharoe Green Lane, Preston PR2 4HT, UK.
References 1 Bloom DL. Mucoceles of the maxillary and sphenoid sinuses. Radiology 1965; 851102-10. 2 Close LG, O'Connor WE. Sphenoethmoidalmucoceles with intracranial extension. Otolaryngol Head Neck Surg 1983; 21:350-7.
383
3 Friedman NG, Harrison S. Mucocele of the sphenoidal sinus as a cause of recurrent oculomotor nerve palsy. J Neurol Neurosurg Psychiatry 1979; 33:172-9. 4 Nugent GR, Sprinkle P, Bloor BM. Sphenoid sinus mucoceles. J Neurosurg 1970; 32:443-51. 5 Phelps PD, Toland JA. Mucocele of the sphenoidal sinus eroding the petrous temporal bone. Br J Radio1 1969; 42(503):845-7. 6 Pincus JH, Daroff RB. Sphenoid sinus mucocele: a curable cause of the ophthalmoplegic migraine syndrome. JAMA 1964; 187:459-61. 7 Proetz AW. The sphenoid sinus. BMJ 1948; 2:243-5. 8 Sellars SL, DeVilliers JC. The sphenoid sinus mucocele. J Laryngol Otol 1981; 95493-502. 9 Simms NM, Brown WE, French LA. Mucocele of the sphenoid sinus presenting as an intrasellar mass. J Neurosurg 1970; 32:708-10. 10 Simon HM, Tingwald FR. Syndrome associated with mucocele of the sphenoid sinus: report of two cases and their radiographic findings. Radiology 1955; 64:538-45. 11 Som PM, Sachdev VP, Biller HF. Sphenoid sinus pneumocele. Arch Otolaryngol 1983; 109:761-4. 12 Takahashi M, Jingu K, Nakayama T. Roentgenologic appearances of sphenoethmoidal mucocele. Neuroradi010gy 1973; 6~45-9. 13 Wyllie JW, Kern EB, Djalilian M. Isolated sphenoid sinus lesions. Laryngoscope 1973; 83: 1252-65. 14 Weaver RG, Gates GA. Mucoceles of the sphenoid sinus. Otolaryngol Head Neck Surg 1979; 87:168-73. 15 Lund VJ,Harvey W, Meghju S, Harris M. Prostaglandin synthesis in the pathogenesis of fronto-ethmoidal mucoceles. Acta Otolaryngol (Stockh) 1988; 106:145-5 1. 16 Hirst LW, Miller NR, Hodges FJ, Corbett JJ, Thompson S. Sphenoid pneumosinus dilatans. A sign of meningioma originating in the optic canal. NeuroradiO I O ~ Y 1982; 22~207-10.
