Hunter K. L. Yuen

Ocular adnexal lymphoma associated with IgG4+ chronic sclerosing dacryoadenitis: a previously undescribed complication of IgG4-related sclerosing disease.

IgG4-related sclerosing disease is a recently recognized inflammatory lesion frequently involving pancreas, submandibular gland, lacrimal gland, and lymph node. We report 3 cases of ocular adnexal lymphoma arising in IgG4-related chronic sclerosing dacryoadenitis, a phenomenon that has not been previously reported. The patients presented with bilateral or unilateral ocular adnexal mass usually present for many years. One patient also had asymptomatic diffuse lymphadenopathy. Two patients had biopsy-proven IgG4-related chronic sclerosing dacryoadenitis before the current presentation, and 1 had systemic involvement by IgG4-related sclerosing disease as evidenced by increased IgG4+ cells in a prior nasopharyngeal biopsy. Two cases showed features of extranodal marginal zone lymphoma of mucosa-associated lymphoid-tissue type (1 with large cell transformation) and 1 follicular lymphoma. Thus, the lymphoid hyperplasia of IgG4-related sclerosing disease can provide a substrate for the emergence of lymphoma. In addition, we report 3 cases of ocular adnexal extranodal marginal zone B-cell lymphoma that show sclerosing inflammation in the background and numerous IgG4+ monotypic plasma cells. In the absence of prior biopsies or information on serum IgG4 titer, it is unclear whether these cases represent lymphoma complicating IgG4-related sclerosing disease or de novo lymphoma. Nonetheless, these cases are distinctive in that the neoplastic cells express IgG4 (light chain restricted), whereas unselected cases of ocular adnexal lymphomas do not show IgG4 expression.

Lymphadenopathy of IgG4-related sclerosing disease.

IgG4-related sclerosing disease is a recently recognized syndrome characterized by mass-forming lesions in exocrine glands or extranodal tissues due to lymphoplasmacytic infiltrates and sclerosis, a raised serum IgG4 level and increased IgG4+ plasma cells in the involved tissues. We report the morphologic features of the enlarged regional (n=3) and nonregional lymph nodes (n=3) in patients with this syndrome. The patients presented with autoimmune pancreatitis, lymphoplasmacytic sclerosing cholangitis, chronic sclerosing dacryoadenitis, or chronic sclerosing sialadenitis. The histologic features of the lymph nodes could be categorized into 3 patterns: Castleman diseaselike, follicular hyperplasia, and interfollicular expansion by immunoblasts and plasma cells. The percentage of IgG4+/IgG+ plasma cells was markedly elevated (mean 62% vs. 9.9% in 54 control lymph nodes comprising a wide variety of reactive conditions). We also report 6 cases of primary lymphadenopathy characterized by increased IgG4+/IgG+plasma cells (mean 58%). These cases share many clinical and pathologic similarities with IgG4-related sclerosing disease. In fact, 2 of these patients developed lymphoplasmacytic sclerosing cholangitis or lacrimal and submandibular gland involvement during the clinical course. These cases therefore probably represent primary lymph node manifestation of the disease. The importance of recognition of the lymphadenopathic form of IgG4-related sclerosing disease lies in the remarkable response to steroid therapy, and the potential of mistaking the disease for lymphoma either clinically or histologically.

Surgical Anatomy of the Chinese Orbit

Purpose: To describe the metric measurements and normal anatomic variants in Chinese bony orbits. Methods: Orbital osteology was studied in 194 orbits from 97 Chinese skulls. Morphologic observations and metric measurements were made to elucidate the variations of the orbital bony anatomy and to localize important fissures and foramina within the orbit. Sexual dimorphism and side asymmetry of orbital features were analyzed. Results were then compared with data from other populations. Results: The details of the orbital osteology in Chinese are described. Most orbital features show anatomic variations in terms of their existence, quantity, and location. Anatomic variants including quadruple and extrasutural posterior ethmoidal foramen are described for the first time. Side asymmetry and sexual dimorphism (male larger than female) are demonstrated in some orbital features. Compared with white and American populations, orbital measurements are generally smaller in Chinese, probably related to their smaller orbital size. Conclusions: The Chinese orbital osteology demonstrates considerable variability. The newly described anatomic variants found on the posterior ethmoidal foramen may affect surgical procedures related to the medial orbital wall. Sexual dimorphism and ethnic variations of the orbital bony anatomy may affect surgical approach to orbital diseases in different populations.

Characterization and localization of the supraorbital and frontal exits of the supraorbital nerve in Chinese: an anatomic study.

Purpose: To elucidate the characteristics and location of the supraorbital and frontal exits of the supraorbital nerve in Chinese skulls and to compare these findings with other ethnic populations. Methods: The anatomy of the supraorbital nerve exits was studied in 97 adult Chinese skulls (194 orbits). The characteristics and location of the supraorbital nerve exits were determined. Analyses were performed to compare differences between sexes and sides. Data were then compared with those of the other populations. Results: Supraorbital exits were found in all 194 orbits. Among these, 89 (45.9%) were foramina. Frontal exits were found in 69 of the 194 (35.6%) orbits in which 18 were foramina (9.3%). The location of the supraorbital exits showed a skewed distribution toward the medial aspect of the superior orbital rim. The median distance between the supraorbital exits and the facial midline was 24.56 mm. Eighty percent of the supraorbital exits lay between 20.77 mm and 30.52 mm from the facial midline. The mean frontal exit was 7.02 mm medial to the supraorbital exits. Compared with other populations, a higher proportion of Chinese have foramina above the superior orbital rim as the exits for the supraorbital neurovascular bundle. The location of the supraorbital nerve exit is highly variable in all studied populations. Conclusions: In a significant proportion of Chinese, the supraorbital nerve exits are located toward the medial aspect of and above the superior orbital rim. Extra care should be taken during dissection around this region to avoid damage to the supraorbital nerve.

Limited nasal septoplasty by ophthalmologists during endonasal dacryocystorhinostomy: is it safe?

Purpose: Significant deviated nasal septum (DNS) may preclude the performance of endonasal dacryocystorhinostomy (DCR). The purpose of this study was to investigate the outcomes of limited nasal septoplasty in tackling signficant DNS performed by trained ophthalmologists during endonasal DCR. Methods: In this retrospective interventional case series, the records of all patients requiring limited nasal septoplasty for significant DNS during endonasal DCR at 2 tertiary ophthalmic centers in Hong Kong during January 2006 to December 2007 were reviewed. Surgical outcomes, demographic factors, and intraoperative and postoperative details were recorded and analyzed. Results: A total of 25 septoplasties were performed in 25 patients (total of 28 DCR). After the nasal septoplasty, all nasal cavities had better exposure of the surgical site allowing completion of the endonasal DCR. At the latest follow-up, the anatomical and functional success rates of the endonasal DCR were both 96.4% (27/28). In one patient, septoplasty was complicated by asymptomatic mucosal adhesion between the lateral nasal wall and the nasal septum. Conclusions: In the hands of trained ophthalmic lacrimal surgeons, limited nasal septoplasty is an effective and safe procedure during endonasal DCR, allowing better exposure of the surgical field in patients with significant DNS.

Complication of IgG4-Related Chronic Sclerosing Dacryoadenitis by Lymphoma

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The use of postoperative topical mitomycin C in the treatment of recurrent conjunctival papilloma.

Purpose. To describe the use of postoperative topical mitomycin C (MMC) in the treatment of recurrent conjunctival papilloma. Methods. Case report. Results. We report a 26-year-old man with recurrent conjunctival papilloma despite repeated surgical excision, cauterization, and cryotherapy. He was then treated with excision by cryotherapy, followed by a 2-week course of topical MMC eyedrops prescribed at postoperative day 7 (0.02 mg/mL, four times daily). No recurrence was observed 24 months postoperatively, and no complication was observed during the follow-up period. Conclusion. Postoperative topical MMC may be a useful adjunct in the management of recurrent conjunctival papilloma.

Clinical characteristics and surgical outcomes of phacoemulsification in true exfoliation syndrome.

Purpose To review the clinical characteristics and surgical outcomes of patients with true exfoliation syndrome of the lens capsule having phacoemulsification cataract surgery. Setting Hong Kong Eye Hospital, Kowloon, Hong Kong. Design Case series. Methods Eyes of patients with true exfoliation syndrome diagnosed clinically or intraoperatively had standard phacoemulsification and intraocular lens (IOL) implantation. The anterior capsules removed during capsulorhexis were sent for histological confirmation. Results Twenty‐four eyes of 18 patients were included. All eyes were confirmed to have true exfoliation syndrome by histological examination of the anterior lens capsule. Seven eyes (29.2%) had preexisting chronic glaucoma; 5 (20.8%) had laser iridotomy before the diagnosis. During phacoemulsification, all eyes had a continuous curvilinear capsulorhexis without complications. Trypan blue staining was used during capsulorhexis in 11 eyes (45.8%). Posterior capsule rupture occurred during lens chopping in 1 eye (4.2%). No loose zonular fibers or radial extension of capsulorhexis occurred. Capsular bag IOL implantation was successfully performed in all cases except the 1 with a posterior capsule rupture, in which an anterior chamber IOL was implanted. Conclusions Phacoemulsification was safely performed in eyes with true exfoliation syndrome. Trypan blue staining can be used to facilitate capsulorhexis creation. Ultrasound biomicroscopy or anterior segment optical coherence tomography may be useful in preoperative assessment of such cases. The disease may be associated with glaucoma and laser iridotomy. Financial Disclosure No author has a financial or proprietary interest in any material or method mentioned.

Use of sodium hyaluronate and indocyanine green for conjunctival cyst excision

Ductal adenocarcinoma of the lacrimal gland was fi rst documented by Katz et al. in 1996. After that only six cases were reported. Differential diagnosis may include adenocarcinoma not otherwise specifi ed (NOS), mucoepidermoid carcinoma, and adenocarcinoma ex pleomorphic adenoma. Comedo-type necrosis is a characteristic feature of primary ductal adenocarcinoma corresponding to salivary duct carcinoma, but it can be minimally present in adenocarcinoma NOS. Squamoid or intermediate tumor cell components are evident in mucoepidermoid carcinoma. In the present case, no squamoid or intermediate tumor cell components were found and mucinous epithelial features were negligible. There was no evidence of pleomorphic adenoma. As all these possible entities were ruled out, the case was fi nally diagnosed as primary ductal adenocarcinoma. Comparison of the CT and MRI scans of the present case showed that the tumor expanded over 2 years and compressed the orbital tissue displacing the globe inferomedially but did not infi ltrate the adjacent bone, keeping the demarcation border intact. However, this kind of tumor is known to be as aggressive as salivary ductal carcinoma, and exenteration is recommended. Among the six reported cases, all patients who underwent en bloc removal of the tumor without exenteration experienced local recurrence, whereas those who underwent orbital exenteration with or without radiotherapy had no recurrence for a specifi ed period. In order to determine the effi cacy of this treatment regimen, further follow-up, especially the study of more cases, is required. This report shows that a lacrimal gland tumor can spawn a variety of other, rare adenocarcinomas.

Retained presumed intraocular cotton fiber after cataract operation: Long-term follow-up with in vivo confocal microscopy

PURPOSE: To evaluate the long‐term clinical outcomes in eyes with retained presumed intraocular cotton fibers after cataract surgery. SETTING: Hong Kong Eye Hospital, Hong Kong, The Peoples Republic of China. METHODS: A retrospective review of 19 eyes with retained presumed intraocular cotton fibers after cataract surgery was performed. Outcome measures were fiber‐related complications. In vivo confocal microscopy was performed in eyes with entrapped cotton fibers at the wound site. RESULTS: The duration of retained presumed cotton fibers ranged from 5 to 110 months (mean 42.3 months). No complications were noted in any of these eyes, including endophthalmitis, persistent uveitis, or corneal endothelial cell loss. In vivo confocal microscopy in eyes with entrapped fibers at the wound site showed normal corneal endothelium morphology with no keratocyte activation or inflammatory response. CONCLUSIONS: Retained presumed fibers after cataract operations are more common than anticipated. In contrast to other organic foreign bodies, these retained fibers appear to be well tolerated. Conservative treatment can be adopted for these fibers as these pose minimal toxicity to the eye.