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SHORT COMMUNICATION ARCH SOC ESP OFTALMOL 2006; 81: 611-614 ATYPICAL FRONTAL SINUS MUCOCELE. A CASE REPORT MUCOCELE DEL SENO FRONTAL DE PRESENTACIÓN ATÍPICA. INFORME DE UN CASO CULEBRO P1, DE-BARCIA L1, SALCEDO G1, RODRÍGUEZ-REYES AA1 ABSTRACT RESUMEN Clinical case: We report a case of a 46-year-old woman who developed a tender, painful mass in the left superior eyelid over a period of about 6 months. This was a frontal mucocele with atypical clinical and histopathologic features. Discussion: Most mucoceles arise from the frontal or ethmoidal sinuses. Frontal mucoceles usually cause outward and downward displacement of the globe, and are often associated with fullness in the superonasal and medial canthal region and a palpable mass (Arch Soc Esp Oftalmol 2006; 81: 611-614). Caso clínico: Presentar un caso clínico de un mucocele de características clínicas e histopatológicas atípicas. Se informa de una paciente femenina de 46 años quien presentó una masa dolorosa en el párpado superior izquierdo de 6 meses de evolución. Discusión: Los sitios de origen más frecuentes de los mucoceles son los senos frontales seguidos de los senos etmoidales. Los mucoceles de origen frontal suelen desplazar el ojo hacia abajo y afuera; y pueden ser palpados por debajo del reborde orbitario anteromedial. Key words: Mucocele, frontal sinus, mucopyocele, computed tomography, Palabras clave: Mucocele, seno frontal, mucopiocele, tomografía computada, masa orbitaria. INTRODUCTION This paper describes the case of an adult woman who developed a frontal sinus mucocele with infrequent clinical and pathological findings. Mucocele of the paranasal sinuses are a frequent cause of orbitary problems in adults (1) because they constitute slow-growth cystic lesions caused by an obstruction of the paranasal sinus, with ensuing entrapment of the mucous-secreting epithelium. Said lesions can extend to the adjacent orbit, the nasal-pharynx area or the cranial cavity (2). The most frequently involved paranasal sinuses are the ethmoidal and frontal sinuses, and occasionally the maxillary sinus (3,4). CASE REPORT A 46-year old woman admitted to hospital in March 2005 due to a painful mass in the external third of the left upper eyelid with a 6-month evolution, accompanied by ipsilateral visual acuity (VA) reduction. The patient referred the development of Received: oct. 5, 2005. Accepted: Oct. 19, 2006. Association for avoiding blindness in Mexico. Hospital «Dr. Luis Sánchez Bulnes», Ocular Pathology Service, Oculoplastic Service, México D.F., México. 1 Ph.D. in Medicine. Correspondence: Patricia Culebro Asociación para Evitar la Ceguera en México, Hospital «Dr. Luis Sánchez Bulnes» Vicente García Torres, 46, Col. San Lucas Coyoacán, C. P. 04030 México D.F. E-mail: [email protected] CULEBRO P, et al. Fig. 1: Clinical photo. Clearly limited cystic lesion in the external third of the upper left eyelid causing ipsilateral mechanical phthosis. a similar mass 15 years before admission which was successfully managed with unspecified medical treatment. The rest of the patient’s history is not relevant to her current condition. The ophthalmological exploration revealed a VA of 0.7 in the right eye (RE) and 0.2 in the left eye (LE). The external third of the upper left eyelid there was a mass measuring 4 x 3,5 cm, which was painful and exhibited phthosis and inferior displacement of the eye on the same side (fig. 1). The patient exhibited limitation to levosupraversion of Fig. 2A: Crown TC showing a homogeneous mass with destruction of the orbitary roof, extension to the cranial cavity and displacement of the eye downwards and inwards. 612 the LE. Exophthalmometry with basis 100 gave 17 mm for both eyes. The palpebral opening was of 10 mm in the RE and 4 mm in the LE. The function of the left elevator muscle was limited. A computerized tomography was requested of both orbits, with axial and coronal sections, which showed a cystic temporal mass with bone destruction which invaded the orbit and displaced the left eye downwards, apparently depending on the frontal sinus (figs. 2A and 2B). The mass was aspired, obtaining a yellowish, dense liquid. A smear thereof showed a small amount of Gram-positive cocci in pairs and chains, accompanied by numerous polymorphonuclear leucocytes and abundant mucosity. Treatment was initiated with ampicilline, dicloxacilline and naproxene orally and sulphacetamide topically. The patient underwent a drainage of the paranasal sinus with removal of the wall. Microscopically, the preparations dyed with hematoxiline and eosin exhibited fragments covered by breathing-type epithelium. Under the epithelium, the wall was made up by dense connective tissue stroma with hemorrhage areas mixed with accumulations of inflammatory infiltrate comprised by mature leucocytes, plasmatic cells, polymorphonuclear leucocytes and numerous eosinophiles (figs. 3A and 3B). The diagnostic of frontal sinus mucocele was established. The presence of a large amount of eosinophiles as part of the inflammatory infiltrate suggested a probable etiology of the allergic or hyper-sensitivity type. Fig. 2B: CAT scan showing cystic density homogeneous mass in the left upper temporal sector. ARCH SOC ESP OFTALMOL 2006; 81: 611-614 Non-typical frontal sinus mucocele Fig. 3A: Cyst wall covered by respiratory epithelium and surrounded by connective tissue with fibrosis and chronic inflammation (Hematoxiline and eosin, original enlargement, x20). Fig. 3B: 3B: Photomicrography with largest increase showing inflammatory infiltrate rich in eosinophiles (Hemato-xiline and eosin, original enlargement, x40). DISCUSSION the respiratory epithelium, it is frequent to find squamous metaplasia changes in the covering epithelium. In this case, in addition to the elements mentioned above, a high number of eosinophiles was evidenced, as is the case in some other lesions of the upper respiratory tract related to allergic antecedents such as the so-called inflammatory or «allergic» polyps (5). To the best of our knowledge, this seems to correspond to the first reported case of frontal mucocele associated to a probable allergic or hypersensitivity etiology. Patients with Mucocele in the paranasal sinuses frequently exhibit headaches, proptosis, alterations in eye mobility or VA reduction (1). The most serious complication of periorbitary Mucocele is the loss of vision due to the compression of the ocular globe which damages the optic nerve and the posterior pole (1-4). The most frequent areas where Mucocele originate are the frontal sinuses followed by ethmoidal sinuses (3). In general, Mucocele develop more with eye displacements than with proptosis. In frontal Mucocele, the eye globe deviation frequently occurs downward and outward, whereas in ethmoidal Mucocele the displacement is more lateral. Frontal Mucocele can be palpated below the anteromedial orbitary edge. This case exhibited a downward deviation of the eye but, in contrast to the reports found in literature, the main increase in volume was found in the left upper temporal region. Histopathologically, the most frequent findings in Mucocele are the presence of a predominantly mono-nuclear inflammatory process in the sub-epithelial connective tissue, made up mainly by mature lymphocytes and plasmatic cells. In prolonged cases with chronic inflammation and irritation of BIBLIOGRAFÍA 1. González F, García A, Prieto A. Mucocele frontoetmoidal con afectación ocular. Arch Soc Esp Oftalmol 2005; 80: 301-303. 2. Lai PC, Liao SL, Jaou JR, Hou PK. Transcaruncular approach of the management of frontoethmoid mucoceles. Br J Ophthalmol 2003; 87: 699-703. 3. Ormerod LD, Weber AL, Rauch SD, Feldon SE. Ophthalmic manifestations of maxillary sinus mucoceles. Ophthalmology 1987; 94: 1013-1019. 4. Kaufman SJ. Orbital mucopyoceles. Two cases and a review. Surv Ophthalmol 1981; 25: 253-262. 5. Davidsson A, Hellquist HB. The so-called «allergic» nasal polyp. ORL J Otorhinolaryngol Relat Spec 1993; 55: 3035. ARCH SOC ESP OFTALMOL 2006; 81: 611-614 613