The Journal of Laryngology and Otology November 1979. Vol. 93. pp. 1121-1125
Co-existing meningioma and mucocele of the frontal sinus* By ADELOLA ADELOYE (Ibadan, Nigeria) ALTHOUGH intracranial meningiomas are common, those of the frontal sinuses are very rare. New and Devine (1947) presented 33 cases of tumours of the nose and throat of neural origin seen at the Mayo Clinic from 1927 to 1947. Four of them were meningiomas—three of the frontal sinus, and the fourth occurred in the subcutaneous tissue of the root of the nose. Other case reports of frontal sinus meningiomas included the three cases of Ash and Raum (1949); two by Rao, Dinakar and Reddy (1972) and one by Majoros (1970). There was no example of a frontal sinus meningioma in the 38 cases of cranial meningiomas described from Ibadan by Odeku and Adeloye (1973). Mucoceles of the frontal sinus, on the other hand, are common. In South Africa, the blacks seem more susceptible than the whites to frontal mucoceles (Wolfowitz and Solomon, 1972). In Ibadan, Nigeria, frontal mucoceles account for a little over 80 per cent of all paranasal mucoceles encountered (Martinson, 1974). Nevertheless, the coexistence of meningioma and mucocele of the frontal sinus has not been seen in the available literature, and certainly not in our experience in Nigeria. This paper describes the first example of this diseasecomplex encountered in Nigeria.
Case report D.F., a 66-years-old Nigerian woman, presented at the University College Hospital (U.C.H.), Ibadan in July, 1977 with a two months' history of a swelling in the right orbitofrontal region, and intermittent, sometimes severe, right frontal headaches. Two years previously, she had hit her forehead rather heavily against the door of a minibus, without sustaining any obvious wound. Analgesics had relieved the pain and she remained well until May, 1977 when she noticed the swelling which brought her to our Neurosurgery Clinic. On examination, she had a huge mass in the right supra-orbital region and the adjacent part of the frontal region, extending to the midline of the forehead (Figs. 1 a and b). The mass measured 9 cm. diagonally and 6 cm. horizontally. It was hard in the frontal area, but soft and fluctuant over the supraorbital margin. The right eye was slightly proptosed, but markedly displaced laterally and inferiorly, with preservation of vision. There was no neurological deficit. A tentative diagnosis of meningioma was made, with chronic osteomyelitis as an alternative possibility. Plain skull radiographs showed destruction of the right frontal bone extending to, and involving, the superior orbital margin and the outer table of the frontal sinus. The lytic area was surrounded by some sclerosis. Right common carotid angiography showed a slight displacement of the anterior cerebral and bowing of the middle cerebral arteries, suggesting an extracerebral mass of the right frontal area. * This paper was presented at the First General/Scientific Meeting of the Nigerian Society of Oto-Rhino-Laryngology, held in Ibadan, Nigeria, on July 15,1978. 1121
FIGS. 1 A AND B
Patient before operation.
A biopsy of the cranial mass was performed on 3 August, 1977, under local anaesthesia, through a small transverse incision in the skin crease. The tumour was encountered in the subcutaneous area. Histology showed a highly cellular neoplasm composed of round and spindle-shaped cells. The round cells, which were arranged in sheets, had vesicular nuclei. The spindle-shaped cells, which had large oval nuclei, were arranged in whorls of different sizes. In places, both cell types were admixed. Collections of fibroblasts and psammoma bodies were present in several areas. One section showed osteoblastic reaction. A diagnosis of meningioma was made. On 17 August 1977, she had a frontal craniectomy, using a transcoronal incision. As soon as the scalp was reflected, a substantial part of the tumour was encountered. It was removed piecemeal, and so was the soft thin bone that covered the rest of it. The craniectomy defect was widened to expose the rest of the tumour. When the defect .was extended medially, the bluish wall of a large mucocele of the frontal sinus was seen. The mucocele was completely removed. The meningioma had destroyed a lot of the anterior wall of the sinus and the lateral part of its posterior wall, through which the meningioma extended intracranially. The dura adjacent to the tumour was excised and the defect repaired with a periosteal graft. Post-operatively, the patient did well. The displacement of the eye has been corrected (Figs. 2 a and b). She does not want to undergo an operation for cranioplasty, being quite content with herself and her family. Comments Meningiomas arise not from dura, but from islands of arachnoid cells which lie on the deep surface of the dura and on arachnoid granulations. These arachnoid cells may lie outside the cranium, along lines of fusion of the embryonic skull, which explains the source of extracranial and ectopic meningiomas. New and Devine (1947) claim that frontal sinus meningioma arises from embryonal arachnoid cell nests pinched off and left behind during embryonic development. The cases of meningiomas of the paranasal sinuses reported in the literature were commonly of the meningotheliomatous and psammomatous types, both with psammoma bodies. Our patient exemplified this also. The associated mucocele of the frontal sinus is most likely secondary to the meningioma. Secondary mucoceles result from obstruction of the sinus ostium by rhinitis; chronic infection; surgical procedures which encroach on the sinus; osteoma, and rarely from trauma or congenital causes (Bordley and Bosley, 1973; Martinson, 1974). In this case, the meningioma itself or the bone reaction which it produced in the bony wall of the sinus and adjacent frontal bone probably occluded the nasofrontal duct, to produce the mucocele. The age factor may be important. In the series of cases involving the paranasal sinuses reported from Ibadan (Martinson, 1974), there were five patients over 60 years, two of whom had carcinoma of the frontal sinus in association with their mucoceles. In the literature, the patients with frontal sinus meningioma were all aged below 60, their ages ranging from 14 to 52 years, and none had an associated mucocele. Our patient sustained an injury to the forehead two years before she presented at hospital. The place of trauma in the genesis of this disease-complex is difficult to evaluate. It has been stated that past trauma, as contained in the
FIGS. 2 A AND B
Patient after operation.
history of our patient, can cause intracranial meningioma. In the series of patients with meningioma described by Cushing and Eisenhardt (1938), onethird of them had a past history of relevant trauma. More recently, Walsh, Gye and Connelley (1969) suggested the same aetiological mechanism in two patients who developed meningioma, 17 and 26 years respectively after initial trauma. Injury has also been incriminated in the causation of mucoceles, particularly with frontal mucoceles. Tamari and O'Neil (1949) postulated that a submucosal injury may produce progressive elevation of the sinus mucosa, leading to the formation of a mucocele. Trauma, however, is not an important cause as it was present in only one of twelve cases of frontal mucoceles among South African blacks and in two of the 80 patients described by Martinson (1974). As most patients will associate a head injury with the subsequent development of any swelling on the head, caution is needed in the evaluation of the place of trauma in the aetiology of meningiomas and of mucoceles. Summary A rare case of coexistence of a meningioma and a mucocele of the frontal sinus is described, in a Nigerian woman of 66 years, and the possible aetiological mechanism of the disease-complex briefly discussed. REFERENCES ASH, J. E., and RAUM, M. (1949) Tumours of the nose and sinuses in Atlas of Otolaryngic Pathology, New York Armed Forces Institute of Pathology, p." 204. BORDLEY, J. E., and BOSLEY, W. R. (1973) Annals of Otology, Rhinology and Laryngology, 82, 696. CUSHING, H., and EISENHARDT, L. (1938) Meningiomas: their classification, regional behaviour, life history and surgical end results. Springfield, Illinois, Thomas, p. 71. MAJOROS, M. (1970) Laryngoscope, 80,640. MARTINSON, F. D. (1974) Ghana MedicalJournal, 13,192. NEW, G. B., and DEVINE, K. D. (1947) Archives ofOtolaryngology, 46,163. ODEKU, E. L., and ADELOYE, A. (1973) African Journal of Medical Sciences,A, 275. RAO, S. B.,DINAKAR, I., and REDDY, C. R. R. M. (1972) Journal ofNeurosurgery, 36,363. TAMARI, M. J., and O'NEIL, J. J. (1949) Journal ofLaryngology and Otology, 63,24. WALSH, J., GYE, R., and CONNELLEY, T. J. (1969) Medical Journal of Australia, 1,906. WoLFOwrrz, B. L., and SOLOMON, A. (1972) Journal of Laryngology and Otology, 96,79. Address for reprints: Adelola Adeloye, Neurosurgery Unit, Department of Surgery, University College Hospital, Ibadan, Nigeria.