Youn Joo Choi

Inflammatory pseudotumor of eyelid: a probable IgG4-related sclerosing disease clinically mimicking eyelid pilomatrixoma

BackgroundOcular adnexal IgG4-related sclerosing disease (IgG4-SD) has been categorized as a novel disease entity. It is characterized by stromal sclerosis and an infiltration of mass-forming lymphoplasmic cells containing many IgG4-positive plasma cells. Although ocular adnexal tissue involvement has been increasingly reported, a focal nodular sub-brow mass is not typical in an IgG4-SD presentation. We report a rare case of probable ocular adnexal IgG4-SD that clinically mimicked eyelid pilomatrixoma.Case presentationA 42-year-old woman presented with a nodular mass in her left sub-brow area. The initial clinical impression of her lesion was eyelid pilomatrixoma. However, the final pathologic diagnosis was IgG4–SD, but extranodal marginal zone B-cell lymphoma could not be excluded. The patient underwent testing to determine tumor malignancy and systemic IgG4-SD involvement. Laboratory testing showed normal IgG and IgG4 serum levels and imaging revealed no remarkable findings. Oral prednisolone was administered and slowly tapered to manage the possible remnant lesion and to prevent disease recurrence. Two years after initiating therapy, there was no evidence of relapse. The patient is under close surveillance for signs of recurrence, systemic involvement, and potential malignant transformation.ConclusionsWe found an unusual case of probable ocular adnexal IgG4-SD, which presented as a unilateral restricted mass involving the sub-brow area. Although the mass was surgically removed, systemic steroid treatment and long-term surveillance were initiated due to the possibility of recurrence, the potential association with systemic disease, and the potential development of extranodal mucosa-associated lymphoid tissue (MALT) lymphoma.

Intraoperatively Observed Lacrimal Obstructive Features and Surgical Outcomes in External Dacryocystorhinostomy

Purpose To analyze the features of lacrimal drainage system obstruction confirmed during external dacryocystorhinostomy surgeries and report the surgical outcomes. Methods We reviewed the medical records of a total of 769 cases who underwent external dacryocystorhinostomy for primary lacrimal drainage obstruction between 2005 and 2014. Data about detailed location and extent of obstruction were collected intraoperatively. The sites of obstruction were classified into nasolacrimal duct obstruction (NLDO), common canalicular obstruction (CCO), and canalicular obstruction. Lacrimal sac mucosa and lumen were grossly inspected, and the frequency of lacrimal sac changes, such as significant inflammation or fibrosis, was analyzed in cases of CCO or canalicular obstruction. The surgical success rate was also evaluated including effect of lacrimal sac status in the CCO and canalicular obstruction groups. Results Of 769 cases, primary NLDO with patent canaliculi was diagnosed intraoperatively in 432 cases (56.2%), CCO in 253 (32.9%), and canalicular obstruction in 84 (10.9%). Of 253 cases with CCO, 122 (48.2%) showed clear lacrimal sac lumen, and the other 131 (51.8%) showed significant inflammation or fibrosis of the lacrimal sac. In cases with canalicular obstruction, 35 of 84 (41.7%) showed a clear lacrimal sac, and the other 49 cases (58.3%) cases revealed mucosal changes of the lacrimal sac. The functional success rate was 87.5% for primary NLDO, 75.5% for CCO, and 72.6% for canalicular obstruction. In the CCO group, the functional success rate was lower in cases with significant lacrimal sac change (p = 0.044). Conclusions Even in patients with CCO or canalicular obstruction, a large number of cases have lacrimal sac changes, and those changes were associated with lower functional success rate.

Periorbital changes associated with prostaglandin analogs in Korean patients

BackgroundProstaglandin analogs (PGAs) are commonly used to treat glaucoma because of their powerful intraocular pressure lowering effect. However, various periorbital changes associated with the use of PGAs have been reported. We investigated the incidence of periorbital changes in Korean patients who were treated with PGAs, and analyzed clinical factors associated with superior sulcus deepening.MethodsThis study included 58 glaucoma patients who were treated with latanoprost, travoprost, or bimatoprost unilaterally. Face photographs were collected, and periorbital changes such as superior sulcus deepening, eyelid pigmentation, ptosis, lid retraction, dermatochalasis, and redness were evaluated by two oculoplastic specialists. For each patient, the contralateral eye served as a control. The frequency of ptosis, dermatochalasis, pigmentation, erythema, and superior sulcus deepening were analyzed. Demographic and ocular factors were compared between patients who showed superior sulcus deepening and those who did not.ResultsThirty-one patients (53.4%) showed one or more periorbital changes associated with PGAs. The most common change was superior sulcus deepening (24.1%), followed by eyelid pigmentation (19.0%), eyelid erythema (19.0%), dermatochalasis (10.3%), eyelid retraction (5.2%), and ptosis (3.4%). The age of the patient and the duration of PGA administration was significantly correlated with superior sulcus deepening (pxa0=xa00.007, pxa0=xa00.002, respectively).ConclusionsPeriorbital changes are frequently seen in patients who use PGAs, and superior sulcus deepening is the most common change in Korean patients. Long-term use of PGAs and old age were associated with superior sulcus deepening.

Estimation of Eyelid Pressure Using a Blepharo-Tensiometer in Patients with Functional Nasolacrimal Duct Obstruction

Purpose To compare the eyelid pressure between patients with functional nasolacrimal duct obstruction (FNLDO) and normal controls using blepharo-tensiometer, and to evaluate the relationship between eyelid pressure and the outcomes of silicone intubation (SI) in patients with FNLDO. Study design Prospective case-control study. Methods We enrolled 36 eyes of 36 patients with suspected FNLDO who underwent SI and 36 healthy eyes of age-matched controls. One eye of each patient with FNLDO was randomly selected for analysis. The eyelid pressure was estimated using a blepharo-tensiometer and compared between the control and FNLDO groups. The relationship between eyelid pressure and clinical variables was analyzed. The outcomes of SI were assessed at 6 months after surgery using subjective and objective criteria. Results The eyelid pressure was significantly lower in the FNLDO group than in the control group (P=0.008). In the control group, the eyelid pressure was correlated with age (P<0.001) and lower eyelid laxity (P=0.016). In the FNLDO group, the eyelid pressure was only correlated with age (P<0.001). The success rate of SI for FNLDO was 69.4% (25 of 36 eyes). The eyelid pressure was higher in the surgical success subgroup than in the failure subgroup, although the difference was not statistically significant (P=0.08). Conclusions Our results suggest that the eyelid pressure measured using a blepharo-tensiometer has a diagnostic value since it is decreased in patients with FNLDO. The role of eyelid pressure as a possible predictor of the outcomes of SI for FNLDO should be investigated in further studies. This trial is registered with KCT0002828.

Effects on Periocular Tissues after Proton Beam Radiation Therapy for Intraocular Tumors

Background To present our experience on orbital and periorbital tissue changes after proton beam radiation therapy (PBRT) in patients with intraocular tumors, apart from treatment outcomes and disease control. Methods Medical records of 6 patients with intraocular tumors who had been treated with PBRT and referred to oculoplasty clinics of two medical centers (Seoul National University Hospital and Seoul Metropolitan Government-Seoul National University Boramae Medical Center) from October 2007 to September 2014 were retrospectively reviewed. The types of adverse effects associated with PBRT, their management, and progression were analyzed. In anophthalmic patients who eventually underwent enucleation after PBRT due to disease progression, orbital volume (OV) was assessed from magnetic resonance (MR) images using the Pinnacle3 program. Results Among the six patients with PBRT history, three had uveal melanoma, and three children had retinoblastoma. Two eyes were treated with PBRT only, while the other four eyes ultimately underwent enucleation. Two eyes with PBRT only suffered from radiation dermatitis and intractable epiphora due to canaliculitis or punctal obstruction. All four anophthalmic patients showed severe enophthalmic features with periorbital hollowness. OV analysis showed that the difference between both orbits was less than 0.1 cm before enucleation, but increased to more than 2 cm3 after enucleation. Conclusion PBRT for intraocular tumors can induce various orbital and periorbital tissue changes. More specifically, when enucleation is performed after PBRT due to disease progression, significant enophthalmos and OV decrease can develop and can cause poor facial cosmesis as treatment sequelae.

Atypical Mycobacterial Infection in Anophthalmic Sockets With Porous Orbital Implant Exposure

PURPOSEnTo investigate the clinical features and risk factors of atypical mycobacterial infection in anophthalmic sockets with porous orbital implant exposure.nnnDESIGNnCase-control study.nnnMETHODSnThe medical records of all patients who had undergone surgical correction of porous orbital implant exposure were consecutively reviewed, and the patients were stratified as those with atypical mycobacterial infection (AM infection group) and others (non-AM group).nnnRESULTSnFive and 21 patients were included in the AM infection and non-AM groups, respectively. All patients of the AM infection group had a peg or motility coupling post (MCP) and showed implant exposure around it. Following up on implant exposure, 2 patients abruptly presented with severe conjunctival injection and new lesions such as erythematous nodules or eyelid masses. They underwent immediate orbital implant exchange and atypical mycobacterial infection was diagnosed. Three patients who were not suspected of having infection underwent surgery for orbital implant exposure. Results revealed erythematous eyelid nodule or recurrent exposure shortly after surgery and patients were diagnosed with atypical mycobacterial infection. In the non-AM group, 7 (33.3%) patients underwent insertion of a peg or MCP. Statistical analysis showed that the insertion of a peg or MCP was the only risk factor showing a significant difference between the 2 groups.nnnCONCLUSIONSnThe most important underlying risk factor for atypical mycobacterial infection in the anophthalmic socket is thought to be peg- or MCP-related exposure of the porous orbital implant. Surgical removal of the infected orbital implant and long-term antibiotic medication are needed for treatment.

Early results of surgical management of conjunctival dermolipoma: partial excision and free conjunctival autograft

Background and aims There is a paucity of data in the literature on the surgical management of conjunctival dermolipoma. In this study, we report outcomes of a partial mass excision technique (excision of the portion visible when eyes open) and free conjunctival autograft. Methods Medical records of 13 patients with dermolipoma who had undergone partial mass excision at Seoul National University Hospital from January 1999 to May 2014 were retrospectively reviewed. After resection of the visible part of tumour, including the overlying thick surface conjunctiva, the conjunctival defects were reconstructed with free conjunctival autograft harvested from the contralateral superior bulbar area. Results The median postoperative follow-up period was 5u2005months (range 1–84u2005months). The cosmetic surgical results were satisfactory in all 13 patients. Three patients showed thick texture on the grafted surgical area postoperatively, but there was no significant scar formation resulting in restrictive symblepharon or eye movement limitation. There were no other significant complications, such as blepharoptosis, diplopia or keratoconjunctivitissicca during the follow-up period. Conclusions Partial mass (visible part only) excision, including that of the overlying thick conjunctiva, and free conjunctival autograft from the opposite eye, is a relatively simple and effective technique for the surgical management of conjunctival dermolipoma.