JKOS Journal of the Korean Ophthalmological Society J Korean Ophthalmol Soc 0378-6471 2092-9374 Korean Ophthalmological Society 10.3341/jkos.2010.51.7.998 jkos-51-998 Original Article A Case of Orbital Mucocele Lined With Two Types of Epithelial Cells After Orbital Wall Fracture Repair KimMijin MD 1 LeeMin Joung MD 1 KimChan-Joong MD 1 KimNam Ju MD 1 2 ChoungHo-Kyung MD 1 3 KhwargSang In MD 1 Department of Ophthalmology, Seoul National University College of Medicine, Seoul, Korea Department of Ophthalmology, Seoul National University Bundang Hospital, Seongnam, Korea Department of Ophthalmology, Seoul National University Boramae Hospital, Seoul, Korea Address reprint requests to Sang In Khwarg, MD, Department of Ophthalmology, Seoul National University College of Medicine #28 Yongon-dong, Chongno-gu, Seoul 110-744, Korea Tel: 82-2-2072-2879, Fax: 82-2-741-3187, E-mail: khwarg@snu.ac.kr 08 2010 31 08 2010 51 7 998 1002 25022010 04052010 Copyright © 2010 Korean Ophthalmological Society 2010 This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Abstract Purpose

The authors report a case of orbital mucocele lined with two types of histological epithelial cells developed after repair of the orbital wall fractures.

 Case summary

A 53-year-old man presented with proptosis of the left eye for two years. The patient had a history of left orbital inferomedial wall fracture repair ten years earlier at a different hospital. Examination revealed 4 mm proptosis and superior globe displacement of the left eye. Restriction of left ocular movements on elevation, depression and adduction were observed. A computed tomography scan demonstrated a large, non-enhancing, cystic tumor in the left inferior orbit with the inferior and medial wall displaced toward the paranasal sinus. An orbital cystic tumor was excised with the removal of previously-inserted orbital implant via a transconjunctival and transcaruncular approach. The inferior, and medial orbital walls were reconstructed using a MEDPOR® TITAN TM implant. The initial pathologic diagnosis was epidermal cyst. Histopathologic re-review revealed an orbital cyst lined with both stratified squamous epithelium and pseudostratified ciliated columnar epithelium, thus diagnosis was changed to orbital mucocele. Proptosis and restriction in ocular motility improved postoperatively.

 Conclusions

Mucocele formation should be considered in patients in whom a cyst developed after orbital fracture repair. J Korean Ophthalmol Soc 2010;51(7):998-1002

 Implant Mucocele Orbital fracture repair References Mauriello JA Jr Fiore PM Kotch M Dacryocystitis. Late complication of orbital floor fracture repair with implant Ophthalmology 19879424850 Brown AE Banks P Late extrusion of alloplastic orbital floor implants Br J Oral Maxillofac Surg 1993311547 Rubin PA Bilyk JR Shore JW Orbital reconstruction using porous polyethylene sheets Ophthalmology 19941011697708 Neves RB Yeatts RP Martin TJ Pneumo-orbital cyst after orbital fracture repair Am J Ophthalmol 199812587980 Tahhan M Alkhardaji F Durrani OM Price NJ Intraorbital epithelial cyst formation: a rare complication of silastic implantation Arch Ophthalmol 200212017689 Glavas I Lissauer B Hornblass A Chronic subperiosteal hematic cyst formation twelve years after orbital fracture repair with alloplastic orbital floor implant Orbit 200524479 Tan CS Ang LP Choo CT Orbital cysts lined with both stratified squamous and columnar epithelia: a late complication of silicone implants Ophthal Plast Reconstr Surg 200622398400 Kwun YK Kim YD Two cases of mucocele after orbital fracture repair J Korean Ophthalmol Soc 2009506127 Kang SJ Kwak IH Hematic cyst formation after repair of blow-out fracture Korean J Ophthalmol 199610602 Lee SB Park KS Kim YD Orbital cyst after repair of blow-out fracture J Korean Ophthalmol Soc 1999402737 James CR Lyness R Wright JE Respiratory epithelium lined cysts presenting in the orbit without associated mucocele formation Br J Ophthalmol 19867038790 Mee JJ McNab AA McKelvie P Respiratory epithelial orbital cysts Clin Experiment Ophthalmol 20023035660 Wang TJ Liao SL Jou JR Lin LL Clinical manifestations and management of orbital mucoceles: the role of ophthalmologists Jpn J Ophthalmol 20054923945 Figures

Preoperative appearance of the patient showing superior displacement (A) and exophthalmos (B) of the left eye.

Preoperative Orbital CT. (A) Axial view demonstrates large, non-enhancing soft tissue mass with relative low density occupying inferolateral extraconal portion of the left orbit. (B) Coronal view showed haziness of the maxillary sinus and a large, non-enhancing, cystic tumor in the left inferior orbit with the inferior and medial wall displaced toward the paranasal sinus. Previously implanted silastic sheet (arrowhead) and polyethylene sheets (arrow) are observed.

Histopathology of the cyst. (A) Low magnification view shows an unilocular cyst which had even wall thickness of 1 mm. (B) There are multiple needle-shaped spaces consistent with cholesterol clefts and inflammatory cells in the adjacent soft tissue of the cyst. (C) Stratified squamous epithelium lining the cystic cavity. (D) Pseudostratified ciliated columnar epithelium in the other area of the cyst wall (H&E staining, magnification ×40 (A, B), ×400 (C, D)).

Eleven months postoperative appearance of the patient showing improved superior displacement (A) and exophthalmos (B) of the left eye.