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4au���waaaxx>�aaa?��������������������������������������������������������������������"aaaaaaaaa��:TITLE : BILATERAL  ETHMOID SINUS MUCOPYOCELE: A CASE-REPORT

Running Title :  ETHMOID SINUS MUCOPYOCELE.

Authors :

1   Kaz1m Bozdemir, Expert MD   Ankara Ataturk Education and Research Hospital ENT Department Bilkent / Ankara / Turkey

2 - Ahmet Kutluhan, Prof MD   Ankara Ataturk Education and Research Hospital ENT Department     Bilkent / Ankara / Turkey

3  Alper Kanmaz, Expert MD   Birecik Government Hospital ENT Department Birecik/Sanl1urfa/Turkey

4 - Hasan Mervan Deer, Expert MD   Siverek Government Hospital ENT Department Siverek/Sanl1urfa/Turkey


(All authors contributed the manuscript and investigation in various aspects. We deserve all of them to assign as authors.)

CORRESPOND0NG AUTHOR :

Alper Kanmaz, Expert MD

Birecik Devlet Hastanesi
KBB Klinii
Birecik/^anl1urfa
TR 63400 TURKEY

E-mail : dr.alperkanmaz@gmail.com

Fax : 904146524775


BILATERAL ETHMOID SINUS MUCOPYOCELE: A CASE-REPORT

ABSTRACT

          Mucopyocele are epithelium-lined, slowly growing cystic lesions with sterile content. Mucopyoseles of the paranasal sinuses can manifest with ocular signs and symptoms, and mostly proptosis. Although ethmoidal mucoceles are not uncommon, bilateral involvement is rather rare. Here in we presented a case of bilateral ethmoidal mucopyosele presenting with proptosis and visual disturbance. She was a 53 years old woman who admitted with decreased visual acuity, lateral displacement of glob and double vision. Computed tomography (CT) of the paranasal sinuses revealed cystic lesions in the maxillary sinuses and anterior ethmoidal air cells bilaterally. After a marsupialization operation was performed by endoscopic sinus surgery, the symptoms of the patient recovered rapidly.

Key words: Mucopyocele, ethmoidal sinus

INTRODUCTION

          Mucoceles are epithelium-lined, slowly growing cystic lesions of the paranasal sinuses with sterile content. When the mucocele content becomes infected, the lesion is defined as mucopyocele. 1,2,3,4 The exact etiology of mucoceles is not certain 1, but possibly associated with scarring and obstruction due to chronic sinusitis or inflammatory processes. It may also be congenital or related to previous trauma or surgery1. These progressively enlarging lesions result in destruction of the walls of the sinuses, extend to the surrounding anatomical structures, and can cause serious cerebral and orbital complications due to abscess formation and rupture.5-6
          Mucopyoceles of the paranasal sinuses are commonly located in the frontal and anterior ethmoidal region and infrequently in the posterior ethmoidal, sphenoidal and maxillary area7-8 Presenting signs and symptoms can vary depending on the location, size and degree of bone involvement.1.Although ethmoidal mucoceles are not uncommon, bilateral etmoidal mucopyoceles are rare. Here in we reported a case of bilateral ethmoidal mucopyocele presenting with proptosis and visual disturbance.

CASE REPORT

          A 53 year-old-woman was admitted with decreased visual acuity, lateral displacement of globe and double vision. According to the history, her complaints began two years ago and gradually progressed. Seven months ago, she noticed a limitation in her eye movements as well as double vision. She also had headache occasionally. She had no systemic disease except for arterial hypertension. On physical examination, there was exoptalmus bilaterally. Her eye movements were restricted in both adduction and abduction, and she had  diplopia. Her visual acuity was 6/10 on the right eye and 10/10 on the left eye. Examination of the fundus of the eye revealed senile macular degeneration and grade I-II hypertensive rethinopathy. Her interpupillary distance was 85 mm (figure 1). Bilateral ocular tension was 31 mm Hg. Bilateral nasolacrimal channels were open on lacrimal lavage.
          There was edema and obliteration by a bulging cystic lesion in the middle meatus bilaterally on endoscopic nasal examination. CT scan showed cystic lesions filling the maxillary sinuses and anterior ethmoidal cells bilaterally, which caused erosion in the walls of sinuses. The lamina papyracea was defective to some extent bilaterally, and the.medial rectus muscles, optic nerves and orbital fat tissues were displaced anterolaterally on both sides. The lesions did not show any contrast enhancement (figures 2). MRI examination also showed cystic lesions in the frontoethmoid region bilaterally, which were hyperintense on T2 weight images with contrast enhancement peripherally (Figure 3).
          The patient was operated, and endoscopic endonasal sinus surgery was performed. A mucopurulant discharge was drained from the cystic lesions, and the anterior and inferior walls of the cysts were excised completely. The uncinate process and lamina papyracea were defective almost completely. The residual bony lamella of the ethmoids were excised completely. Then, the maxillary sinus ostia were enlarged. The maxillary sinus was also full of mucopurulant material. The maxillary sinuses were cleaned and irrigated with antibiotic solution.
          The patient was also treated with cefazolin and metranidazole for 14 days post-operatively, and exophtalmus and diplopia subsided. On post-operative 2nd day, the interpupillary distance was 65mm, and visual acuity was 6/10 on her right eye and 10/10 on the left side (figure 4). Histopathologic examination confirmed the cystic lesion which was lined by the respiratory epithelium. On post-operative 3th month, the control CT showed normal orbital architecture .

DISCUSSION

          Mucocele is a destructive cyst that arises within the paranasal sinuses 9. When these sterile cysts become infected, the lesion is defined as mucopyocele.2-4  These space-occupying lesions can increase in size as mucus retention continues and can be exacerbated by active sinusitis.3 Differential diagnosis includes thyroid eye disease, orbital pseudotumor, infection, benign or malignant tumors, encephalocele, or meningocele.3
          Mucopyoceles of the paranasal sinuses are commonly located in the frontal and ethmoidal region and infrequently in the posterior ethmoidal, sphenoidal and maxillary area.7,10 Because of their slowly growing and noninfiltrating nature, the symptoms and signs develops slowly and depends on the localization and size of the lesion.
          Mucoceles with orbital involvement generally present with a noninfiltrating mass effect resulting in globe displacement, diplopia, proptosis, eyelid swelling, palpable mass, ptosis and reduced vision.8 These symptoms may  be subtle for many years due to slowly progressive nature of the condition.11
          In general,a post-operative mucocele develops years after sinus surgery, which is due to postoperative cicatricial stenosis or infection. 10 Therefore, for prevention of mucosele, it is necessary to excise the ground lamella and ethmoid sinuses properly. 10 In our case there was no previous history of surgery or trauma, and the etiology was possibly chronic recurrent infections. Chemical mediators may also be responsible for local destruction associated with mucoceles 12
          CT and MRI help diagnose mucoceles and evaluate their relationship with the surrounding tissues.7,13 Surgical treatment is indicated for mucopyocele of the paranasal sinuses.10 Treatment includes complete removal of the mucucele and mucosal lining of the involved sinus, and obliteration of the sinus.8  However the mucosal lining of the mucocele may be adherent to dura mater or to the orbital periostium, and can increase risk of surgery .8 If the mucosa is removed incompletely and the sinus is obliterated , recurrence is highly likely.8 Recently, marsupialization with drainage (with sufficient removal of anterior and inferior walls) by endoscopic approach has been the therapeutic approach which may also prevent recurrance.8-10 Moriyama H. et all.10 suggested opening of the mucocele by an endonasal approach as the radical form of therapy. Khong JJ. et all. 8 advised that endoscopic sinus surgery should be considered as the treatment option for paranasal sinus mucocele with orbital involvement, and a direct orbital approach is not required. Marsupialization of the mucocele into the nose is effective in improving ophthalmic symptoms and signs.
          In conclusion, mucopyoceles may arise from any of the paranasal sinuses. Due to their proximity to the orbit, they may manifest with ocular signs and symptoms. It should be kept in mind: mucopyoceles are one of the most common cause of proptosis. Therefore, in suspected in cases, the required radiological investigations should be done. 








REFERENCES
1- Kelly A. Malloy, O.D. Fronto-ethmoid sinus mucocele: A case report. Optometry (2006) 77, 450-458
2- Kandoan T, Oz�er MZ, Sezgin O. Mucopyocele of the maxillary sinus: a case report. Kulak Burun Bogaz Ihtis Derg. 2007; 17 (4): 235-6
3- Palmer-Hall AM, Anderson SF. Paraocular sinus mucoceles. J Am Optom Assoc. 1997 Nov; 68 (11): 725-33
4- Matyja G, Kawczynski M, Tarnowska C. Pyocele of the posterior ethmoidal cell as the cause of visual loss. Otolaryngol Pol. 2006; 60 (2): 171-4
5- Dr.Lokman Uzun, Dr.Murat Kalayc1 ve ark. Komplike Fronto-Orbital Mukosel Olgusu. KBB ve BBC Dergisi, 12 (1): 11-15. 2004
6- Malloy KA. Fronto-ethmoid sinus mucocele: a case report. Optometry, 2006 Sep; 77 (9): 450-8
7- Tetsuzo Fujitani MD, Toshihiro Takahashi MD, Toshimichi Asai MD Optic Nerve Disturbance Caused by Frontal and Frontal-ethmoidal Mocupyoceles. Arch Otolaryngol � Vol 110, April 1984
8- JJ Khong, R Malhotra, PJ Wormald, D Selva Endoscopic sinus surgery for paranasal sinus mucocele with orbital involvement. Eye (2004) 18, 877-881
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$d�a$gd�pB9- Surayie Al-Dousary, Soliman Al-Kharashi Maxillary sinus mucopyocele in children: a case report and review of literature. International Journal of Pediatric Otorhinolaryngology 36 (1996) 53-60
10- Hiroshi Moriyama MD, Hirohiko Hesaka MD, Toshiro Tachibana MD, Yoshio Honda MD Mococoeles of Ethmoid and Sphenoid Sinus With Visual Disturbance. Arch Otolaryngol Head Neck Surg � Vol 118, Feb 1992
11- Valeri1 J.Lund, M.S, F.R.C.S. and Muriel E.Rolfe, D.O. Ophthalmic considerations in fronto-ethmoidal mucocoeles The Journal of Laryngology and Otology July 1989. Vol.103 pp.667-669
12- Jose Antonio et.all. Middle turbinate mucopyocele: a case report. Rev.Bras.Otorrinolaringol. vol.71 no.3 Sao Paulo May/June 2005
13- A.Pompili, R.Mastrostefano et.all. Mucocele of Neurosurgical Interest: Clinical Considerations on Five Cases. Acta Neurochir (Wien) (1990) 102: 114-121






Figure Legends
Figure 1 � Preoperative view of the patient showing exoptalmus and widening of the interpupillary distance
Figure 2 - Preoperative axial CT of the patient showing bilateral ethmoid mucoceles
�ArC>D�D�D�D�D�D�D�D�DZE�EUH�H�H�H�H�����������������$dda$$d��a$gd�pBFigure 3 � Preoperative axial MRI of the patient showing bilateral ethmoid mucoceles
Figure 4 � Postoperative view of the patient with normal interpupillary distance



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