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  • The Curious Case of the Hairstylist With Hyperglobus and Hypoesthesia

    By Eve E. Moscato, MD, and Rona Z. Silkiss, MD
    Edited by Thomas A. Oetting, MD
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    This article is from June 2010 and may contain outdated material.

    Until recently, Daniel Chan* relished the busy pace of work at his salon. But lately, staying on schedule had become more of a challenge for the 51-year-old hairstylist. He had been experiencing progressive right upper lid discomfort for three months as well as diplopia when glancing down at his scissors while cutting hair. He also noticed numbness of his right upper lip over the past few months. He had been treated for presumed maxillary sinusitis with Augmentin (amoxicillin and clavulanate potassium) for three weeks without improvement of his symptoms.

    Presentation

    Mr. Chan is HIV-positive with a history of high cholesterol and periodic sinus symptoms. He had bilateral maxillary antrostomies via a Caldwell-Luc approach at age 10. Besides Augmentin, his current medications include Pravachol (pravastatin sodium) and the antiretroviral combinations Kaletra (lopinavir plus ritonavir) and Truvada (emtricitabine plus tenofovir). He also takes multivitamins.

    On examination, his BCVA was 20/25 in both eyes. His pupils were normal and reactive, and confrontation visual fields were normal, as were his intraocular pressures. Infraduction of the right eye was reduced by 20 percent; all other fields of gaze were full. The slit-lamp examination was normal, except for trace nuclear sclerosis. Further examination revealed that the right eye was displaced superiorly (hyperglobus), and Hertel exophthalmometry readings demonstrated 2 mm of proptosis of the right eye relative to the left eye (Fig. 1). The distance from the upper eyelid margin to the corneal light reflex—or margin-reflex distance one (MRD1)—was 0 to 1 mm in the right eye and 6 mm in the left. The distance from the lower eyelid margin (MRD2) was 6 mm in the right eye and 5 mm in the left. Levator function was normal bilaterally.

    A maxillofacial CT scan revealed a 3-cm mass in the right maxillary sinus. It protruded superiorly through the orbital floor and medially along the lateral wall of the right nasal fossa. Coronal views demonstrated absence of the bony orbital floor (Fig. 2).

    Differential Diagnosis

    The differential diagnosis for a sinus mass causing bony destruction is relevant. Benign lesions include inverted papilloma, dermoid, epidermoid, neurofibroma, mucocele and mucopyocele. Malignant lesions include squamous cell carcinoma, botryoid rhabdomyosarcoma, adenoid cystic carcinoma, lymphoma, schwannoma and plasmacytoma. In the absence of bony erosion, one should consider retention cysts (which arise under the mucosa), nasal polyps and chronic sinusitis.1

    In this case, the CT scan shows a dense, homogeneous lesion that is isodense with brain. This is consistent with a mucocele. Unless infected, such a lesion does not show contrast enhancement.

    Surgical Management

    Mr. Chan underwent a right endoscopic maxillary antrostomy for drainage and partial removal of the mucocele (Fig. 3; the online version of this article includes a video).

    In addition, we repaired his eroded orbital floor by placing a Medpor Titan (polyethylene/titanium mesh) implant. This was placed via a transconjunctival approach (Fig. 4). By supporting his orbital contents with an implant, we were able to avoid descent of the globe into the sinus once the mucocele was drained. Mr. Chan’s hyperglobus was corrected, as was his motility deficit in downgaze.

    Postoperatively, Mr. Chan’s diplopia resolved. The large mucocele was probably putting pressure on the inferior orbital nerve, causing right upper lip hypoesthesia. This symptom resolved after surgical drainage of the mucocele.

    We Get a Look 1

    We Get a Look 2
    We Get a Look. We noted hyperglobus, proptosis and apparent ptosis (Fig. 1). The coronal CT scan demonstrates a homogeneous, right maxillary sinus mass, protruding superiorly through the orbital floor, with a large bony defect (Fig. 2).

    Surgical Management 1

    Surgical Management 2
    Surgical Management. The endoscope provides a view of the maxillary sinus mucocele protruding in the middle meatus (Fig. 3). Transconjunctival approach showing bony defect in the orbital floor with protruding mucocele covered with sinus mucosa (Fig. 4).

    Discussion

    Mucoceles are benign, locally invasive paranasal masses, arising beneath the periosteum. They form secondary to obstruction of the sinus ostia. Mucus production within this mucoperiosteum- lined cavity produces gradual expansion of the mucocele with subsequent expansion of the sinus. Mucoceles can result from long-term inflammation, infection, trauma or prior surgery.

    Pressure atrophy can result in surrounding bony erosion and resorption. The most common sites of mucoceles are frontal, ethmoidal and sphenoidal sinuses. Less than 10 percent of all mucoceles arise in the maxillary sinus.1 Although mucoceles from any location can invade the orbit, invasion superiorly from the maxillary sinus is relatively uncommon. The mucocele is lined by pseudostratified columnar epithelium, and the fluid is usually sterile.

    Diagnosis is made through assessment of clinical findings, maxillofacial CT scan and histopathologic confirmation.

    Historically, an open approach with a Caldwell-Luc sinusectomy and nasoantral window has been used to drain the mucocele and remove its entire lining. In Mr. Chan’s case, an antrostomy had been formed through the anterior wall of the maxillary sinus via a sublabial route when he underwent surgery as a child. Although used as a treatment for mucoceles, this approach has also been implicated in the formation of these lesions, as observed in our case. Sinus mucosa may become entrapped after the Caldwell-Luc approach. With insufficient drainage, a mucocele may develop.2

    Advancements in drainage through endoscopic surgery have largely led to replacement of the Caldwell-Luc procedure for management of uncomplicated mucoceles.1,3–5 The endoscopic approach consists of an ethmoidectomy, middle meatal antrostomy and marsupialization, with drainage of the mucocele. This procedure has long-term favorable outcomes with little recurrence.4,5 Some endoscopic surgeons advocate use of mitomycin C to help prevent fibrosis and adhesion of the antrostomy postoperatively.6 The patient can easily be monitored in the office for closure of the antrostomy using a portable endoscope.7

    Although we describe a mucocele that caused hyperglobus and proptosis, some maxillary sinus mucoceles can cause downward displacement of the globe and enophthalmos, also known as silent sinus syndrome. These findings result from a depressed orbital floor secondary to bony erosion, although the mucocele does not invade directly, leading to a sunken appearance of the globe.

    In conclusion, the ophthalmologist should be aware of sinus processes that can lead to orbital involvement. Sinus mucoceles can cause bony erosion from the frontal, ethmoidal, sphenoidal or maxillary sinuses. Such mucoceles may lead to a variety of signs, depending on the location of invasion. These signs can include vision loss, globe displacement, apparent ptosis, decreased ocular motility, pain and facial hypoesthesia.

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    * Name of the patient is fictitious.

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    Dr. Moscato is a fellow in oculofacial plastic and reconstructive surgery at the University of California, San Francisco, and the California Pacific Medical Center.

    Dr. Silkiss is chief of the oculofacial plastic, reconstructive and orbital surgery division at CPMC.

    The authors have no financial disclosures.

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    1 Dispenza, C. et al. Acta Otorhinolaryngol Ital 2004;24:292–296.

    2 Sheth, H. G. and R. Goel. Int Ophthalmol 2007;27:365–367.

    3 Marks, S. C. et al. Otolaryngol Head Neck Surg 1997;117:18–21.

    4 Har-El, G. Laryngoscope 2001;111:2131–2134.

    5 Busuba, N. Y. and D. Kieff. Laryngoscope 2002;112:1378–1383.

    6 Gupta, M. and G. Motwani. J Laryngol Otol 2006;102:921–923.

    7 Busaba, N. Y. and S. D. Salman. Laryngoscope 1999;109:1446–1449.