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 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

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 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

BACKGROUND AND OBJECTIVES: To review our experience with lacrimal sac tumors in an effort to identify features, to evaluate the results of various methods of management, and to compare our data with previous studies. METHODS: We reviewed the medical records of all patients with lacrimal sac tumors who were managed in our institution between January 1990 and December 2015. The pre-operative clinical data, imaging, operation notes, and follow-up records were reviewed for each patient. RESULTS: The study group consisted of four men and six women with a mean age of 47.6 years. Most patients experienced long-standing epiphora, for a mean period of 20 months. Two of the tumors were benign, and eight of them were malignant. The benign tumors were treated with dacryocystectomy. All but one malignant tumor were treated with medial or total maxillectomy. Adjuvant radiotherapy was administered to four patients with malignant tumors. In the eight patients with malignant tumors, the mean follow-up period was 65 months. CONCLUSIONS: Important characteristics of lacrimal sac tumors include dacryocystitis, epiphora, and in some cases, a palpable medial canthal area mass. Wide en bloc resection via medial or total maxillectomy and/or postoperative radiotherapy are proper treatments for malignant lesions of the lacrimal sac.
Dacryocystitis ; Female ; Follow-Up Studies ; Humans ; Lacrimal Apparatus Diseases ; Male ; Medical Records ; Nasolacrimal Duct* ; Radiotherapy ; Radiotherapy, Adjuvant

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 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: 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

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

A dacryolith is a concretion within the lacrimal drainage system. Although it may cause intermittent epiphora without inflammation, dacryoliths are often underlying contributors to recurrent or chronic dacryocystitis. It may occur anywhere along the lacrimal drainage system, albeit most commonly in the lacrimal sac. We report an interesting case of a dacryolith presenting as a mass of inferior meatus, which was managed via endonasal endoscopic approach.
Dacryocystitis ; Dacryocystorhinostomy ; Drainage ; Inflammation ; Lacrimal Apparatus Diseases ; Lacrimal Duct Obstruction