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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.
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