Immunoglobulin G4 (IgG4)-related dacryoadenitis and sialoadenitis (IgG4-DS) are part of a multiorgan fibroinflammatory condition of unknown etiology termed IgG4-related disease (IgG4-RD), which has been recognized as a single diagnostic entity for less than 15 years. Histopathologic examination is critical for diagnosis of IgG4-RD. CD4+ T and B cells, including IgG4-expressing plasma cells, constitute the major inflammatory cell populations in IgG4-RD and are thought to cause organ damage and tissue fibrosis. Patients with IgG4-RD who have active, untreated disease exhibit significant increase of IgG4-secreting plasmablasts in the blood. Considerable insight into the immunologic mechanisms of IgG4-RD has been achieved in the last decade using novel molecular biology approaches, including next-generation and single-cell RNA sequencing. Exploring the interactions between CD4+ T cells and B lineage cells is critical for understanding the pathophysiology of IgG4-RD. Establishment of pathogenic T cell clones and identification of antigens specific to these clones constitutes the first steps in determining the pathogenesis of the disease. Herein, the clinical features and mechanistic insights regarding pathogenesis of IgG4-RD were reviewed.
B-Lymphocytes ; Clone Cells ; Dacryocystitis ; Diagnosis ; Fibrosis ; Humans ; Immunoglobulins ; Molecular Biology ; Plasma Cells ; Sequence Analysis, RNA ; Sialadenitis ; T-Lymphocytes

PURPOSE: We report a case of acute dacryocystitis diagnosed with abscess and rupture of lacrimal sac and fistula to posterior orbit during the operation. CASE SUMMARY: A 71-year-old woman visited our clinic with edema and pain in the eyelid from three days ago. For past four months, there was viscous of the left eye and tears. The patient had severe conjunctival chemosis and hyperemia, compared with the left eyelid edema and redness. Orbital CT scan showed orbital cellulitis, which was followed by systemic antibiotics and steroid therapy. On the 4th day of therapy, orbital abscess formation was observed in orbit MRI and surgical drainage was planned. During surgery, we found rupture of the posterior part of lacrimal sac and fistula to posterior orbit. Pseudomonas aeruginosa was identified in the bacterial cultures, and after the administration of appropriate antibiotics, the disease showed improved progress, and then additional dacryocystorhinostomy was performed. CONCLUSIONS: In our case, acute dacryocystitis rarely spread in orbit, which may lead to delayed diagnosis, orbital cellulitis and abscess, resulting in serious complications of vision threat. So, we think that it is necessary to consider surgical treatment more actively in the stage of chronic dacryocysitis.
Abscess ; Aged ; Anti-Bacterial Agents ; Dacryocystitis ; Dacryocystorhinostomy ; Delayed Diagnosis ; Drainage ; Edema ; Eyelids ; Female ; Fistula ; Humans ; Hyperemia ; Magnetic Resonance Imaging ; Nasolacrimal Duct ; Orbit ; Orbital Cellulitis ; Pseudomonas aeruginosa ; Rupture ; Tears ; Tomography, X-Ray Computed

PURPOSE: We sought to evaluate the outcomes of external dacryocystorhinostomy (DCR) and mono-canalicular intubation in patients with total obstruction of one canalicus. METHODS: Sixteen eyes of 16 patients with nasolacrimal duct obstruction and a single canaliculus obstruction who had undergone external DCR and monocanalicular intubation of the intact canaliculus were retrospectively included in the present study. The monocanalicular tube (Mini Monoka) was left in place for at least two months. Munk epiphora grading for the evaluation of epiphora and irrigation was performed both preoperatively and at 6 months postoperatively. RESULTS: Mean patient age was 46 ± 14.2 (range, 18 to 76) years. The inferior canaliculus was obstructed in nine eyes (group A) and the superior canaliculus was obstructed in seven eyes (group B), respectively. Eight eyes had chronic dacryocystitis and two of these eyes also had a history of acute dacryocystitis attack. Mean preoperative Munk scores were 3.89 in group A and 4.0 in group B. Ocular surface irritation occurred in one eye in group A. Artificial eye drops were prescribed and early tube removal was not performed. Spontaneous tube dislocation was recorded in one eye in group B. No other corneal, punctal, or canalicular complications were found. At six months, irrigation of intact canaliculus was patent in all eyes. Mucoid discharge, conjunctival hyperemia, and chronic conjunctivitis were also resolved. Postoperative Munk scores were 1.11 ± 0.9 in group A and 0.86 ± 0.9 in group B. Of note, preoperative and postoperative Munk scores were significantly different in both groups (group A, p = 0.006; group B, p = 0.017). The postoperative Munk scores were not statistically different between the two groups (p = 0.606). CONCLUSIONS: In patients with nasolacrimal duct obstruction and a total of one canaliculus obstruction, external DCR and monocanalicular intubation of the intact canaliculus is an effective surgical option.
Conjunctivitis ; Dacryocystitis ; Dacryocystorhinostomy ; Dislocations ; Eye, Artificial ; Humans ; Hyperemia ; Intubation ; Lacrimal Apparatus Diseases ; Lacrimal Duct Obstruction ; Nasolacrimal Duct ; Retrospective Studies

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.
Canaliculitis ; Child ; Dermatitis ; Disease Progression ; Enophthalmos ; Humans ; Lacrimal Apparatus Diseases ; Medical Records ; Melanoma ; Orbit ; Protons* ; Retinoblastoma ; Retrospective Studies ; Seoul

PURPOSE: We report a case of a young female patient who was diagnosed with a marginal zone B-cell lymphoma of the mucosa-associated lymphoid tissue (MALT lymphoma) of the lacrimal sac which mimicked dacrocystitis. CASE SUMMARY: A 23-year-old female suffered from epiphora for 3 years in the right eye. She had swelling and a painful lesion at the lacrimal sac 4 months prior and was referred to our hospital due to nasolacrimal duct obstruction and dacryocystitis. Lacrimal irrigation was performed with no passing and regurgitation with mucoid discharge. We performed orbital computed tomography and magnetic resonance imaging which showed a suspected tumor of the lacrimal sac in the right eye. We then performed excision and biopsy of the tumor through the skin approach. The patient was diagnosed with a MALT lymphoma with no systemic involvement. After six cycles of chemotherapy involving rituximab with cyclophosphamide, vincristine, and prednisone, the lesion of the mass and the painful symptoms decreased, which was regarded as a complete response. However, epiphora and eye discharge persisted, showing a nasolacrimal duct obstruction, so we performed endoscopic dacryocystorhinostomy with a lacrimal sac biopsy. The histological examination showed chronic inflammation, but not lymphoma. There was no recurrence of lymphoma at one year postoperatively. CONCLUSIONS: If there is an epiphora and palpable lesion in the lacrimal sac area, MALT lymphoma mimicking dacyocystitis may be suspected. After the lymphoma is first diagnosed by excision and biopsy, systemic chemotherapy with dacryocystorhinostomy could be an effective treatment.
Biopsy ; Cyclophosphamide ; Dacryocystitis ; Dacryocystorhinostomy ; Drug Therapy ; Female ; Humans ; Inflammation ; Lacrimal Apparatus Diseases ; Lymphoid Tissue ; Lymphoma ; Lymphoma, B-Cell, Marginal Zone ; Magnetic Resonance Imaging ; Nasolacrimal Duct ; Orbit ; Prednisone ; Recurrence ; Rituximab ; Skin ; Vincristine ; Young Adult

PURPOSE: To compare the clinical effects of the single wide-diameter bicanalicular silicone tube and the double bicanalicular silicone tube in endonasal dacryocystorhinostomy (DCR) with canalicular trephinization for canalicular obstruction. METHODS: We retrospectively reviewed the records of 121 patients with monocanalicular or common canalicular obstruction who had undergone endonasal DCR with random bicanalicular insertion of either double silicone tubes (insertion of two tubes into each canaliculus) or a single wide-diameter (0.94 mm) silicone tube. The tubes were removed at around 3 months after surgery. RESULTS: This study included 79 eyes of 61 patients in the double-tube intubation group and 68 eyes of 60 patients in the single wide-diameter tube intubation group. Anatomical success, evaluated by syringing, was achieved in 72 of the 79 eyes (91.1%) in the double-tube intubation group and 60 of the 68 eyes (88.2%) in the single wide-diameter tube intubation group. Functional success was achieved in 65 of the 79 eyes (82.3%) in the double-tube intubation group and 61 of the 68 (89.7%) eyes in the single wide-diameter tube intubation group. There were no significant differences in the success rates of surgery between the two groups. One patient in the double-tube intubation group underwent conjunctivodacryocystorhinostomy (CDCR) and two in the wide-diameter tube intubation group underwent CDCR or reintubation to treat recurrence. CONCLUSIONS: Intubation using a single wide-diameter tube during endonasal DCR is as effective as double-tube intubation for the treatment of canalicular obstruction, with a lower rate of complications such as inflammation or patient discomfort.
Canaliculitis ; Dacryocystorhinostomy ; Humans ; Inflammation ; Intubation ; Lacrimal Duct Obstruction ; Recurrence ; Retrospective Studies ; Silicon* ; Silicones*

PURPOSE: We report a case of fungal ball after Endoscopic Dacryocystorhinostomy (DCR) in a 40-year-old female patient. CASE SUMMARY: A 40-year-old female patient was admitted to our hospital for left lower eyelid tenderness and bloody discharge from the lacrimal punctum. During a planned endoscopic DCR, the sac was opened after the osteotomy, and 2 fungal balls were found in the lacrimal sac. The masses were 7 × 5 mm and, 9 × 5 mm sized, irregularly shaped, and red in color. Aspergillus fumigatus was diagnosed pathologically. Postoperative paranasal sinus magnetic resonance imaging showed no residual fungal ball. During follow-up, the patient showed patent rhinostomy opening, and there was no evidence of fungal infection on nasal endoscopic finding. CONCLUSIONS: Although Aspergillus fumigatus is a rare cause of canalicular obstruction, fungal ball development in the lacrimal sac can cause acute dacryocystitis.
Adult ; Aspergillus fumigatus ; Dacryocystitis* ; Dacryocystorhinostomy ; Eyelids ; Female ; Follow-Up Studies ; Humans ; Lacrimal Apparatus ; Magnetic Resonance Imaging ; Nasolacrimal Duct ; Osteotomy

PURPOSE: To compare characteristics in patients with and without previous dacryocystitis and satisfaction after endoscopic dacryocystorhinostomy. METHODS: We retrospectively analyzed 156 eyes of 116 patients who underwent endoscopic dacryocystorhinostomy at our Hospital from May 2011 to December 2015. The patients were grouped into those with pre-operative dacryocystitis and those without dacryocystitis. Each patient group was characterized retrospectively, followed by a telephone survey regarding post-operative satisfaction. RESULTS: Fifty-eight patients (66 eyes) had dacryocystitis, whereas 58 patients (90 eyes) did not. Patients' mean age was 64.9 years old. The mean duration of symptoms in patients with and without dacryocystitis was 11 and 20 months, respectively, showing a statistically significant difference (p < 0.05). The correlation between comorbidities and dacryocystitis was statistically insignificant (p > 0.05). Post-operative satisfaction was higher in patients with dacryocystitis, showing a statistically significant difference (p < 0.05). CONCLUSIONS: Patients with pre-operative dacryocystitis were characterized by a shorter duration of symptoms and higher post-operative satisfaction. This finding implies that post-operative satisfaction could be lower in patients without dacryocystitis, which should be noted.
Comorbidity ; Dacryocystitis* ; Dacryocystorhinostomy* ; Humans ; Retrospective Studies ; Telephone

PURPOSE: This article analyzes the microorganisms and antibiotics susceptibility in dacryocystitis. METHODS: In this study, patients who were diagnosed with acute and chronic dacryocystitis with nasolacrimal duct obstruction were selected and underwent endoscopic endonasal dacryocystorhinostomy. Cultures were obtained from the lacrimal sac during operation from January 2008 to January 2016, and were used to analyze the microorganisms and antibiotics susceptibility. RESULTS: The 67 patients, 9 were diagnosed with acute dacryocystitis and 58 were diagnosed with chronic dacryocystitis. Among them, 64 cases showed bacterial growth (95.5%). The most frequently detected bacteria was Staphylococcus epidermidis (S. epidermidis) (33.8%), followed by Staphylococcus aureus (S. aureus) (25.4%) and Enterobacter aerogenes (18.3%). S. epidermidis had the most powerful resistance to ciprofloxacin compared to the other bacteria (58.3%, p = 0.02). Except for S. epidermidis and S. aureus, the other bacteria responded to ciprofloxacin and gentamycin. CONCLUSIONS: As a causative microorganism of dacryocystitis, S. epidermidis is becoming more prominent, and it is thought that S. epidermidis may be resistant to quinolones (i.e., broad-spectrum antibiotics). This resistance might be increasing the percentage of present S. epidermidis when viewed as a causal pathogen in dacryocystitis.
Anti-Bacterial Agents* ; Bacteria ; Ciprofloxacin ; Dacryocystitis* ; Dacryocystorhinostomy ; Enterobacter aerogenes ; Gentamicins ; Humans ; Nasolacrimal Duct ; Quinolones ; Staphylococcus aureus ; Staphylococcus epidermidis

PURPOSE: To present a case of orbital inflammation and optic perineuritis preceding vesicular eruption in herpes zoster ophthalmicus(HZO). CASE SUMMARY: An 84-year-old woman with a history of gall bladder cancer and hypertension complained of left periorbital erythematous edema and discomfort. On examination, visual acuity was 20/25 bilaterally; no tenderness, proptosis or ophthalmoplegia was observed. Pupils were equal, round, and reactive to light without relative afferent pupillary defects. Slit-lamp examination revealed severe conjunctival injection and chemosis without keratitis or uveitis. The remainder of the ocular examination was unremarkable. Magnetic resonance imaging confirmed left-sided preseptal swelling with an enlarged left lacrimal gland, high signal intensity of the retrobulbar fat and optic nerve sheath. Systemic antibiotic therapy with steroids was started under a presumed diagnosis of idiopathic orbital inflammatory disease, but the clinical presentation was unresolved. After 2 days, vesicular lesions confined to the first division of the trigeminal nerve and pseudodendritic keratitis developed on the left side leading to a diagnosis of HZO. Treatment with acyclovir immediately resolved anterior segment inflammation and periorbital edema. While on therapy, visual acuity deteriorated to 20/125 and the pupil became dilated and unresponsive to light over a few days. All signs and symptoms of acute orbitopathy and postherpetic neuralgia had resolved 3 months later with the exception of pupil abnormality and visual acuity. CONCLUSIONS: HZO may present with symptoms and signs of orbital inflammation and optic perineuritis even in the absence of a vesicular rash. Thus, HZO should be considered in the differential diagnosis of unexplained acute orbital syndromes.
Acyclovir ; Aged, 80 and over ; Dacryocystitis ; Diagnosis ; Diagnosis, Differential ; Edema ; Exanthema ; Exophthalmos ; Female ; Gallbladder Neoplasms ; Herpes Zoster Ophthalmicus* ; Herpes Zoster* ; Humans ; Hypertension ; Inflammation* ; Keratitis ; Lacrimal Apparatus ; Magnetic Resonance Imaging ; Neuralgia, Postherpetic ; Ophthalmoplegia ; Optic Nerve ; Orbit* ; Pupil ; Pupil Disorders ; Steroids ; Trigeminal Nerve ; Uveitis ; Visual Acuity