Clinical Features and Surgical Treatment Outcomes of Conjunctival Squamous Papilloma

PURPOSE: To investigate the clinical features, surgical treatments, and their outcomes in conjunctival squamous papilloma. METHODS: A retrospective chart review was conducted on 31 patients with 32 eyes that were treated for conjunctival papilloma from October 2000 to February 2015 in Seoul National University Hospital and Seoul National University Bundang Hospital. RESULTS: Among the 31 patients, 9 patients had papilloma which recurred after previous surgical excision at another hospital. Twenty-five eyes had one papilloma lesion, 2 eyes had 2 lesions, and 5 eyes had more than 3 lesions. The most affected location of papilloma was the tarsal conjunctiva. The recurrent group displayed a tendency to have multiple lesions. Surgical excision without any adjuvant therapy was performed in 13 eyes; surgical excision and cryotherapy in 15 eyes; surgical excision, cryotherapy, and topical interferon alfa-2b in 3 eyes; and surgical excision and amniotic membrane transplantation in 1 eye. The mean postoperative follow up period was 11.1 months. There were 5 cases of recurrence and the mean time of recurrence after surgical excision was 4.22 months (range, 3 days to 9 months). Among 5 cases of recurrence, 3 cases were after surgical excision only, 1 case was after surgical excision and cryotherapy, and 1 case was after surgical excision, cryotherapy, and topical interferon alfa-2b. These 5 recurred cases were retreated with surgical excision and cryotherapy, surgical excision and topical interferon alfa-2b, or surgical excision, cryotherapy, and topical interferon alfa-2b. CONCLUSIONS: Conjunctival squamous papilloma is likely to recur even though the tumor is completely removed. Therefore, long-term postoperative follow up may be necessary for recurrence.

Orbital Wall Fracture Repair: The Results of Early and Delayed Surgery

PURPOSE: To investigate the surgical results of early and delayed repair of orbital wall fracture after multiple subgrouping of patients by time between the operation and injury. METHODS: Eighty-eight eyes of 88 patients who underwent orbital wall fracture repair from January 2002 to December 2014 and who were followed up for more than 3 months postoperatively were included in this study. We divided the 88 patients into three groups: Early surgery group (surgery within 2 weeks after the injury), slightly delayed surgery group (surgery between 3 weeks and 2 months after the injury), delayed surgery group (surgery after 2 months of the injury). Preoperative and postoperative ocular motility, diplopia, and the degree of enophthalmos were analyzed retrospectively. RESULTS: The early surgery group consisted of 30 eyes; slightly delayed surgery group, 42 eyes; and delayed surgery group, 16 eyes. The mean duration between injury and surgery was 8.6 +/- 22.5 weeks in all patients, 1.5 +/- 0.5 weeks in the early surgery group, 3.5 +/- 1.3 weeks in the slightly delayed surgery group, and 35.3 +/- 44.7 weeks in the delayed surgery group. All patients were followed up for a mean of 12.9 +/- 10.8 weeks. Gaze limitation in all directions showed improvement in all groups, with the most shown in up gaze limitation. There were no significant differences in the degree of improvement between preoperative and postoperative gaze limitation among the three groups. Enophthalmos improved as well, without any significant differences among the three groups. CONCLUSIONS: Improvement in ocular motility limitation and enophthalmos after orbital wall fracture repair did not vary significantly according to the duration between the surgery and injury. Therefore, surgical repairment even for old orbital fractures may successfully treat enophthalmos or diplopia and relieve symptoms.

A Case of Orbital MALT Lymphoma in a Patient with Rheumatoid Arthritis

PURPOSE: To report a case of orbital mucosa-associated lymphoid tissue (MALT) lymphoma that occurred in a patient with rheumatoid arthritis (RA). CASE SUMMARY: A 70-year-old male with alleged RA presented with a 3-month history of spontaneous and painless upper and lower eyelid swelling in the right eye. On initial examination, palpable nodules were observed at the right upper and lower eyelids and orbital computed tomography (CT) revealed a contrast-enhancing nodule just inferior to the lacrimal gland, thus an incisional biopsy was performed. The pathological report showed malignant orbital lymphoma (low grade extranodal marginal zone B cell lymphoma of MALT). Since distant metastases were not detected on whole-body CT and positron emission tomography, the patient was advised to keep previous medication and regular follow-up examinations. CONCLUSIONS: The possibility of MALT lymphoma should be considered as a differential diagnosis in patients with eyelid swelling and autoimmune diseases such as RA.

A Case Report of Eyebrow Basal Cell Carcinoma Successfully Treated with 5% Imiquimod Cream

PURPOSE: Basal cell carcinoma is the most common malignant neoplasm of the skin. Periorbital basal cell carcinoma needs to be treated for cosmetic improvement and prevention of intraorbital invasion. Although surgical excision is a standard treatment, nonsurgical treatment using imiquimod cream, a topical immunomodulator, has been attempted. The authors report the first case of periorbital basal cell carcinoma successfully treated with imiquimod cream; to the best of our knowledge, similar cases have not been published to date in the Korean ophthalmology journals. CASE SUMMARY: A 73-year-old female visited our hospital for treatment of a biopsy-proven basal cell carcinoma in the right eyebrow. A flat, elevated, 1 x 1 cm-sized mass having central indentation was observed in the centromedial area of the right eyebrow. She was treated with 5% imiquimod cream once daily at night, 5 days per week for 6 weeks. Eight weeks after the treatment with imiquimod cream was discontinued, the mass was flattened and the cilia began to grow on the mass site. Under the impression of a partial regression, an additional 4 weeks of application was recommended. The mass disappeared more and her eyebrow became more normal. At the final follow-up 31 months after treatment, her right eyebrow appeared normal. CONCLUSIONS: Imiquimod cream may be a useful medication which can be used for complex periorbital basal cell carcinomas that are difficult to reconstruct after resection or surgical candidates with poor general condition.

The Effectiveness of Topical Steroid Instillation in Patients with Recently Developed Nasolacrimal Drainage Stenosis

PURPOSE: To evaluate the effectiveness of topical steroid instillation and associated factors in patients with recently developed nasolacrimal drainage stenosis. METHODS: The medical records of 108 eyes of 56 patients who received topical steroid for nasolacrimal drainage stenosis between January 2010 and June 2013 and who developed epiphora of a three-month duration were retrospectively reviewed. Evaluations were performed at 1, 3, and 6 months after instillation of topical steroid. Subjective symptoms, tear meniscus height, and fluorescein dye disappearance test results were noted at every follow-up visit; nasolacrimal irrigation and canalicular probing were also performed as needed. RESULTS: The cumulative improvements were 42.0% at 1 month and 51.0% at 3 months after treatment, and the mean period of symptomatic improvement was 3.8 +/- 1.9 weeks (range, 3-12 weeks). At the final follow-up visit, 56 (51.9%) of 108 eyes showed improved epiphora. Final improvement was observed in 63% (12 of 19 eyes) of idiopathic nasolacrimal drainage stenosis patients, in 100% (10 of 10 eyes) of docetaxel-treated patients, and in 43% (34 of 79 eyes) of S-1-treated patients. CONCLUSIONS: Instillation of topical steroids can be effective for nasolacrimal drainage stenosis and may prevent the need for invasive surgical treatment.

Pleomorphic Adenoma of the Lacrimal Gland in a Child

PURPOSE: To report one case of pleomorphic adenoma of the lacrimal gland in a child. CASE SUMMARY: A 7-year-old boy presented with a 2-month history of an exophthalmos and mild ptosis of the left eye; there were no other ophthalmologic problems. Orbit MRI revealed a well-defined mass in the left lacrimal gland and a defect in the orbital roof due to pressure erosion. Tumor resection was performed via a lateral orbitotomy and a biopsy was requested. The biopsy revealed tumor tissue that was composed of epithelial elements with variably sized duct formation. Focal squamous metaplasia and keratin production was also observed. CONCLUSIONS: Pleomorphic adenoma of the lacrimal gland should be considered as a differential diagnosis of a lacrimal gland mass in a child.

A Case of Lymphomatoid Papulosis of the Eyelid

PURPOSE: Lymphomatoid papulosis (LyP) is one of the primary cutaneous CD30-positive lymphoproliferative disorders. LyP of the eyelid has rarely been reported. Herein, a case of typical LyP of the medial canthal area is reported. In addition, a literature review was performed. CASE SUMMARY: A 40-year-old female presented with a skin mass in the medial canthal area of the left eye that developed 2 months earlier. Initially, a focal skin lesion developed, and even with conservative treatment at a local clinic, progressed to a mass lesion having a central ulceration and adjacent edema. After 6 weeks, the adjacent edema had gradually decreased. On ophthalmic examination, the left medial canthal lesion was a 6 x 6 mm sized elevated mass with a central crater covered by crust. The clinical impression was keratoacanthoma. The lesion was widely excised and reconstructed by a full-thickness skin graft after an incisional biopsy. Histopathologic findings showed dermal infiltration of various inflammatory cells with atypical lymphocytes showing positivity to the CD30 antigen, and LyP was diagnosed. Systemic evaluation showed no evidence of systemic lymphoma and the patient has remained free of recurrence or systemic disease after a 1-year follow-up.

A case of diffuse alveolar hemorrhage after tonsillectomy: A case report / 대한마취과학회지

Diffuse alveolar hemorrhage (DAH) is an acute, life-threatening event. The blood-gas barrier must be very thin to allow gas exchange and is therefore subjected to high mechanical stresses when the capillary pressure rises. In general anesthesia, inhaled gases cause high mechanical stresses, and thus DAH occurs under certain conditions. We describe a case of inferred postoperative DAH. A 25-year-old man had an acute episode after undergoing a tonsillectomy for snoring. During surgery, no problems occurred and no marked bleeding was observed. After removal of the endotracheal tube, however, the patient had severe cough and hemoptysis. The patient was treated with an antihemorrhagic agent and antibiotics. He recovered after 1 week. Chronic snoring likely caused the alveolar damage in this patient and intubation led to DAH. The patient presented with a benign course that regressed spontaneously with medical intervention.

Three Cases of Hyper-IgG4 Syndrome Involving Ocular Adnexa

PURPOSE: To report 3 cases of hyper-IgG4 syndrome involving ocular adnexa. CASE SUMMARY: A 66-year-old woman presented with swelling of the bilateral upper eyelids with ocular pain that began 1 year before. Nodular episcleral injection of the left eye and other generalized symptoms, such as cough, decreased hearing ability, multiple nodular lesions of the bilateral lungs and right kidney, together suggested rheumatic disease. Orbital computed tomographic images revealed diffuse swelling of the bilateral lacrimal glands. After immunostaining a surgically-biopsied specimen from the lacrimal gland for IgG4 expression, 15% of infiltrated lymphoplasmacytic cells were IgG4-positive. Similar findings were shown with biopsied specimens from the lung and kidney; therefore, the patient was diagnosed with Hyper-IgG4 syndrome. A 49-year-old woman complained of a mass in the left upper eyelid that began 4 years earlier. Orbital computed tomographic images showed a 5-mm-sized mass in the left upper eyelid. Ocular adnexal Hyper-IgG4 syndrome was confirmed by the immunostained biopsy from the left upper eyelid, showing infiltration of IgG4-positive lymphoplasmacytic cells. A 51-year-old woman presented with swelling of the bilateral lacrimal glands. Enlargement of the bilateral lacrimal glands were apparent in orbital computed tomographic images. After anti-IgG4 antibody staining of a biopsied specimen from the right lacrimal gland, dense infiltration of IgG4-positive lymphoplasmacytic cells was observed. The patient was also diagnosed with Hyper-IgG4 syndrome.

A Case of Orbital Mucocele Lined With Two Types of Epithelial Cells After Orbital Wall Fracture Repair

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(R) 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.

A Case of Hydranencephaly With Exotropia

PURPOSE: To report a large-angle exotropia, limited adduction, epiblepharon, high myopia and no pupillary light reflex in a patient with hydranencephaly. CASE SUMMARY: A ten-year-old girl with mental retardation presented with exotropia. The patient could fix only with the right eye and was unable to follow with either eye. The Krimsky test revealed 95 prism diopters of exotropia, and adduction was severely limited in both eyes. Pupillary light reflex was absent in both eyes. Cycloplegic refraction showed high myopia in both eyes. Slit lamp examination revealed lower lid epiblepharon and inferior corneal opacity in the right eye. No abnormal findings in the fundus examination were detected. A computed tomogram of the brain showed that the cerebral hemispheres were replaced by a cystic space filled with cerebrospinal fluid, compatible with hydranencephaly. Recession of the lateral rectus muscle and resection of the medial rectus muscle with epiblepharon repair of the lower lid were performed in both eyes. One week postoperatively, the epiblepharon was corrected, and the Krimsky test showed 16 prism diopters of left intermittent exotropia at near. CONCLUSIONS: When a combined manifestation of mental retardation, limited adduction, no pupillary light reflex and a large-angle exotropia is present, the possibility of a congenital developmental anomaly of the central nervous system including hydranencephaly should be suspected.

Lower Eyelid Epiblepharon Associated with Lower Eyelid Retraction

PURPOSE: To describe a series of patients with lower eyelid epiblepharon associated with lower eyelid retraction. METHODS: We retrospectively reviewed the medical records of patients who underwent surgery for lower eyelid retraction, epiblepharon, or thyroid-associated ophthalmopathy (TAO) between October 1999 and March 2007. Patients with both lower eyelid retraction and epiblepharon on preoperative examination were included in this study. RESULTS: Twenty-seven eyelids of 20 patients with both lower eyelid retraction and epiblepharon were enrolled. The underlying causes of lower eyelid retraction included congenital retraction (seven eyelids), congenital fibrosis of the extraocular muscles (CFEOM; seven eyelids), TAO (seven eyelids), post-operative cicatricial retraction (five eyelids), and facial nerve palsy (one eyelid). Eight of 27 eyelids were successfully corrected after the repair of retraction without the repair of epiblepharon, regardless of the cause of lower eyelid retraction. Another four eyelids with epiblepharon associated with TAO resolved after only orbital decompression. Cilia-everting sutures were additionally applied for epiblepharon in another 14 eyelids, 12 of which did not require the excision of a skin fold or the orbicularis muscles. Only one eyelid with mild retraction and epiblepharon underwent simple epiblepharon repair. Recurrence of retraction or epiblepharon developed in three eyelids during follow-up. CONCLUSIONS: In cases with both lower eyelid retraction and epiblepharon, the retraction should be repaired first, and then the epiblepharon can be corrected selectively according to the severity of the case.

A Case of Periorbital Infantile Capillary Hemangioma Treated With Propranolol

PURPOSE: To report a case of periorbital infantile capillary hemangioma treated with propranolol. CASE SUMMARY: A 6-month-old girl visited our clinic for a mass on the right upper eyelid, which had been present since birth. A dome-shaped, red-purple colored mass occupying almost the entire right upper eyelid was observed, and overlying branch-shaped telangiectases were also detected. The visual axis of the right eye was partially obscured, due to the ptotic eyelid, and her ability to fix and follow was poor in the right eye. The patient was diagnosed with infantile capillary hemagioma. Because amblyopia in her right eye was suspected, treatment with propranolol was initiated. After 2.5 months of propranolol treatment, the hemangioma decreased in size and volume dramatically, changed from red-purple to light red color, and softened. The ptosis of the right upper eyelid improved, as well as the ability of the right eye to fix and follow. No significant adverse effects (e.g. hypotension, bradycardia, hypoglycemia, bronchospasm, or elevated liver enzymes) were observed, except transient QTc prolongation during cotreatment with ibuprofen, due to an upper respiratory infection. CONCLUSIONS: Propranolol should be considered as a treatment option for periorbital infantile capillary hemangioma. Further clinical studies are required to establish the optimal guidelines and long-term outcome.

Surgical Repair of Canalicular Defects and Congenital Eyelid Colobomas Associated with Tessier No. 3 Cleft

PURPOSE: To report a single case of surgical repair of the canalicular defects and congenital eyelid colobomas associated with Tessier No. 3 craniofacial cleft. CASE SUMMARY: A one-month-old girl presented with eyelid colobomas and discharges from the eyes. The patient was diagnosed with a Tessier No. 3 craniofacial cleft with bilateral lower eyelid colobomas medial to the puncta. At the age of 55 months, examination under general anesthesia revealed mid-canalicular obstructions in both lower canaliculi. After pentagonal excision of eyelid colobomas in the left upper and both lower eyelids, both ends of the canaliculi were found at the cut edge of the lower eyelids. After the repair of canalicular defects and bilateral nasolacrimal duct silicone tube intubation, the primary closure of the eyelid defect was performed layer by layer. Although there was no subjective improvement of epiphora in the left eye, a subjective improvement of epiphora in the right eye was achieved, and tear meniscus height in the right eye was halved. Additionally, the eyelid colobomas were cosmetically well repaired at postoperative 6 weeks. The patient still had mild tearing symptoms, but did not complain any longer of discharge at postoperative 4 months. CONCLUSIONS: Tessier No. 3 craniofacial cleft with eyelid colobomas can be associated with canalicular defects and nasolacrimal duct obstructions. Surgical repair of the canalicular defects associated with eyelid colobomas should be considered to achieve a functional recovery of the lacrimal drainage system.

Management of Dermoid Tumor in the Medial Canthal Area

Dermoid tumors in the medial canthal area are rare, but when present they commonly adhere to the lacrimal canaliculi. Three patients presented with a mass in the medial canthal area. The authors performed excisional biopsies, and the masses were diagnosed as dermoid tumors. In two patients, canalicular lacerations were found after mass excision, which suggested that the masses had been firmly adherent to the lacrimal canaliculi. The lacerated canaliculi were repaired after bicanalicular silicone intubation. In the remaining patient, lacrimal silicone intubation was performed at the beginning of surgery, and the mass was successfully dissected from the canaliculi, leaving them intact. Excision of dermoid tumors in the medial canthal area requires careful dissection to avoid canalicular laceration. Bicanalicular silicone intubation at the beginning of surgery is helpful for the identification of the canaliculi and for the prevention of canalicular laceration during dermoid tumor excision.

A Case of Eyelid Schwannoma

PURPOSE: To report a rare case of solitary eyelid schwannoma. CASE SUMMARY: A 60-year-old female visited our clinic with a small cyst-like mass of the left upper eyelid, which had developed 3 years earlier. A 3x3 mm-sized, nonpigmented, cyst-like mass was located in the lateral area of the left upper eyelid margin. We performed an excisional biopsy under local anesthesia. The tumor was well encapsulated, easily dissected, and could be completely removed. Histopathologic examination showed cellular areas of spindle cells with Verocay bodies forming palisades and less cellular myxoid areas. Immunohistochemical analysis for S-100 protein showed a strongly positive reaction. Based on the histopathologic features, a diagnosis of benign Schwannoma was made. CONCLUSIONS: Schwannoma should be also considered in the differential diagnosis of a well-circumscribed mass lesion in the eyelid.

Orbital Leiomyoma: A Case Report

PURPOSE: To report a case of orbital leiomyoma presenting with proptosis. CASE SUMMARY: A 69-year-old woman presented with a 3-month history of proptosis in her left eye. Intraocular pressure was 17 mmHg in her right eye and 23 mmHg in her left eye. There was a left hypotropia on upgaze. A fundus examination showed retinal folds in the superotemporal area in her left eye. Computed tomography revealed a 2.6 cm-sized well-defined enhancing solid mass in the superotemporal extraconal space of the left orbit, pushing her left eye forward. Lateral orbitotomy, tumor removal, and biopsy were performed. Pathological findings showed a fascicular pattern of benign spindle cells with mild cellular pleomorphism and hyaline degeneration, without mitotic figures. Immunohistochemical stain was positive with smooth muscle actin (SMA), which was compatible with orbital leiomyoma. CONCLUSIONS: Orbital leiomyoma is rare, but it should be considered as a differential diagnosis of orbital tumor when the presenting symptom is proptosis and limitation of ocular movement.

Canaliculitis Associated With SmartPlugtrade mark Punctal Plug Insertion: Clinical Features and Management

PURPOSE: To report the clinical features and treatment of canaliculitis associated with SmartPlug punctal plug insertion. METHODS: Case selection criteria included patients with canaliculitis, who were managed at Seoul National University Hospital from January 2006 to October 2008, presenting with a history of punctal plug insertion. The operation reports were reviewed to identify patients in whom SmartPlug was discovered during the operation. Six patients (8 eyes) were identified, and a retrospective chart review was performed for all the patients. RESULTS: The mean age of the patients was 34.3+/-8.6 years, and there were 1 men and 5 women. Common symptoms were mucous discharge (6 eyes) and conjunctival injection (2 eyes). The mean time from insertion of the plug to onset of symptoms was 27.0+/-27.0 months (range 4 to 77 months). All patients underwent surgical removal of the punctal plug by one-snip punctoplasty, canalicular retrograde compression using 2 cotton-tipped applications (2 eyes), or canalicular curettage (6 eyes). All patients had resolution of symptoms after the procedure. CONCLUSIONS: Canaliculitis should be considered when there is conjunctival discharge or injection in patients with SmartPlug. One-snip punctoplasty and retrograde compression of canaliculus can be attempted preferentially as a minimally-invasive treatment option.

Two Cases of Bilateral Endogenous Klebsiella pneumoniae Endophthalmitis in Primary Klebsiella pneumoniae Liver Abscess Patients

PURPOSE: To report 2 cases of bilateral endogenous endophthalmitis in patients with Klebsiella pneumoniae primary liver abscess. CASE SUMMARY: A 78-year-old woman and a 76-year-old woman presented with bilateral blurred vision for 2 to 3 days. The women had fever, cough, and chills for 5 to 10 days. Abdominal computed tomography revealed liver abscess and ophthalmologic examination results suggested bilateral endophthalmitis. In both patients, Klebsiella pneumoniae was identified from the liver abscess aspirate. In one patient, Klebsiella pneumoniae was also identified from a vitreous sample. Both patients lost their vision despite intravitreal and intravenous antibiotics injection.

Two Cases of Orbital Castleman's Disease

PURPOSE:To report two cases of multicentric Castleman's disease involving the orbital area. CASE SUMMARY: A 52-year-old man presented with discomfort of the right eye on eyeball movement that began 4~5 months ago. He had been diagnosed as Castleman's disease upon the biopsy of his neck lymph nodes 2 years ago. Proptosis was found in his right eye, and orbital computed tomography revealed an enlarged mass in the inferior orbit compared to the former image. An incisional biopsy confirmed Castleman's disease. A 57-year-old man presented with left eyelid swelling that began 3 months ago. Orbital and chest computed tomography revealed a mass in the left superotemporal orbit and the enlarged lymph nodes of the mediastinal area. Castleman's disease was confirmed by biopsy of the lymph nodes in the mediastinal area. CONCLUSIONS: Steroid therapy was prescribed for the 52-year-old man. The 57-year-old man was placed under close observation.