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Immunoglobulin G4-related disease (IgG4-RD) is a rare systemic fibroinflammatory disease that can affect almost any organ in the body. The most common sites of IgG4-RD include the salivary glands, orbits and lacrimal glands, pancreas, biliary tree, retroperitoneum and thyroid. Immunoglobulin G4-related lung disease (IgG4-RLD) can occur in isolation or as a component of multisystem disease. Its manifestations are heterogeneous and include lung cavitation, interstitial changes, pleural effusions and lung lesions.We describe a patient with Ig4-RLD manifesting as bronchial stenosis and left upper lobe collapse., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2025. No commercial re-use. See rights and permissions. Published by BMJ Group.)

In addition to being a drainage system, the lacrimal ducts actively defend the eye through the action of the lacrimal pump, the presence of resident flora (lacriome) and the mucosa-associated immune tissue. Infection occurs when mucosal immune functions are overwhelmed. Lacrimal infection is a vicious circle, in which infection leads to inflammation and post-inflammatory sequelae, themselves a source of occlusion and stagnation, which in turn encourages infection. Whatever the initial trigger - post-traumatic occlusion, inflammation (e.g. allergic) or infestation by a virulent germ, the management of lacrimal duct infection must take account of this vicious circle. When infection sets in, changes in the mucosa can range from submucosal inflammatory infiltrates to pseudocysts, and in the most severe cases to metaplasia, dysplasia and squamous cell carcinoma. Histological sampling is therefore imperative in the case of surgery on a chronically infected mucosa. Clinical assessment, aided if necessary by a dacryoscan, must be able to detect differential diagnoses (lacrimal sac carcinoma, dacryocystocele in children) and serious forms (gonococcal ophthalmia neonatorum, peridacryocystitis), look for signs of dacryoliths, and differentiate between acute and chronic infection. Antibiotic treatment should be adapted to the identified or presumed organism involved or used sparingly in cases of chronic infection. Dacryoliths can be removed by curettage in the case of canaliculitis with concretions, but surgery is required for larger dacryoliths (canaliculotomy, or dacryocystorhinostomy in the case of lacrimal sac dacryoliths). Antibiotic prophylaxis for lacrimal surgery is recommended only in cases with associated rhinosinusitis, inflammation of the lacrimal sac, or a history of infected mucocele or dacryocystitis., (Copyright © 2024 The Authors. Published by Elsevier Masson SAS.. All rights reserved.)

Objective: This paper aimed to describe another form of aggressive limited Granulomatosis with polyangiitis (GPA) revealed by dacryoadenitis. Methods and results: We report an unusually limited GPA in a 48-year-old man presenting with bilateral proptosis. She had never presented kidney or pulmonary manifestations, but her disease was persistently active including oto-rhino-laryngological manifestations, dacryoadenitis, and neurological manifestations unresponsive to corticosteroids and immunosuppressors. Discussion: Granulomatosis with polyangiitis (GPA) is an auto-immune inflammatory vasculitis. Involvement of lacrimal glands as the first presentation is uncommon. It is characterized by the development of granulomas. Patients with orbital mass without lacrimal gland involvement have a higher rate of systemic disease, a severe clinical course, and a higher rate of recurrences. A patient with dacryoadenitis seems to be with a good prognosis. Eye manifestations were significantly more common in patients with pachymeningitis. MPO-ANCA-positive pachymeningitis was more frequent in older female patients. PR3-ANCA-positive pachymeningitis had more severe neurological damage. Induction treatment consists of intravenous methylprednisolone (IV) associated with cyclophosphamide. Conclusion: Faced with dacryoadenitis, it is important to screen for ANCA-associated vasculitis. Abbreviations: GPA = Granulomatosis with polyangiitis, ANCA = Antineutrophil Cytoplasmic Antibodies., (#x00A9; The Authors.Romanian Society of Ophthalmology.)

A 14-year-old female presented with 2 weeks of progressive right eye pain, erythema, and proptosis. Examination demonstrated an enlarged palpable mass along the right superior lateral orbit and bilateral conjunctival petechiae. Of note, she was asymptomatic on the left side, and the petechiae were present only on the superior bulbar conjunctiva with eyelid eversion. Imaging demonstrated bilateral lacrimal gland enhancement. Testing was significant for elevated inflammatory markers, but otherwise negative workup. Biopsy of the right lacrimal gland demonstrated acute-on-chronic inflammation without evidence of lymphoproliferative disease. On repeat testing, myeloperoxidase antibody levels (MPO/p-ANCA) were elevated, indicative of an underlying immune-mediated vasculitis. This case illustrates a rare presentation of ANCA-associated vasculitis in a pediatric patient. It further demonstrates the phenomenon of initial negative serology and subsequent auto-antibody seroconversion in a patient with localized granulomatosis with polyangiitis.

A 48-year-old woman visited our hospital because of bilateral lacrimal gland enlargement. Her serum immunoglobulin G4 (IgG4) level was high, and positron emission tomography-computed tomography showed significant positive findings in the bilateral lacrimal gland. A biopsy revealed a considerable increase in IgG4/CD138, leading to a diagnosis of IgG4-related dacryoadenitis. The disease did not respond to steroid therapy, so treatment was started with baricitinib because of exacerbation of the original atopic dermatitis and dacryoadenitis after the second dose of the coronavirus disease 2019 (COVID-19) vaccine. Baricitinib was effective for resolving both dermatitis and dacryoadenitis, and steroids were able to be discontinued. The IgG4 level also improved.

Background: Dacryoadenitis is inflammation of the lacrimal gland, mainly caused by viral infection. It can also be caused by bacterial pathogens and non-infectious processes such as auto-immune diseases and malignancy. Chronic dacryoadenitis is rarely linked to SARS-CoV-2 infection, with only five reports in the literature., Report: A 26-year-old Arab woman experienced chronic inflammatory dacryoadenitis after a mild SARS-CoV-2 infection, which was successfully treated with oral prednisone., Conclusions: Dacryoadenitis can occur due to inflammation caused by either SARS-CoV-2 exposure. The treatment typically involves the administration of steroids, with duration to be decided based on clinical response., (© 2023. BioMed Central Ltd., part of Springer Nature.)

Dacryoadenitis can be a rare presentation following COVID-19 infection or can be an immunological response post COVID-19 vaccination. Herein we report two cases of lacrimal gland involvement, one post COVID-19 infection, and the other post COVID-19 vaccination. A definitive causal relationship, however, remains uncertain.

Extranodal natural killer (NK)/T-cell lymphoma, nasal type (NNKTL) is a rare and highly aggressive non-Hodgkin lymphoma originating from NK or γδ T cells infected by Epstein-Barr virus (EBV). In the United States, NNKTL is usually noted in people of Asian or Hispanic descent. Natural killer/T-cell lymphoma, nasal type commonly involves the upper aerodigestive tract, including the nasopharynx, nasal cavity, Waldeyer's ring, and oropharynx. Extensive local destruction and invasion has been noted, especially of the paranasal sinuses, hard palate, and central nervous system; involvement of the nasolacrimal duct with dacryocystitis is yet to be reported. We report a rare case of a Hispanic man with extranodal NNKTL masquerading as persistent dacryocystitis and necrotizing sinusitis unresponsive to antibiotics and surgical intervention. An extensive background of necrosis and inflammation was noted on pathology, and additional analysis with immunohistochemistry and in situ hybridization after repeat biopsy were necessary for accurate diagnosis.

Competing Interests: Conflict of interest: None declared.

A 35-year-old female with a history of chronic extensive rhinosinusitis, previously treated twice with functional endoscopic sinus surgery, presented with recurrent dacryocystitis despite prior dacryocystorhinostomy. Histopathological specimens taken at the most recent sinus surgery demonstrated a lymphocytic inflammatory reaction without evidence of angiodestruction or necrosis. Flow cytometry was normal. Over the following 9 months, the patient developed worsening hypertelorism and bilateral recurrent acute dacryocystitis with a fistula tract to the skin. Neuroimaging revealed a hyperintense enhancing soft tissue expansion into the periorbital regions, invading the nasolacrimal canals, and obstructing the paranasal sinuses. A skin biopsy at the fistula site revealed natural killer T-cell lymphoma. Metastatic work-up disclosed lung, spleen, and bone marrow involvement. The patient underwent chemotherapy with mixed clinical response, and ultimately passed away from metastatic disease. The authors present a rare case of natural killer T-cell lymphoma involving the nasolacrimal sac, presenting as recurrent dacryocystitis and diagnosed by skin biopsy of the fistula site.

Background and Objective: Acute dacryocystitis is an atypical and rare manifestation of pediatric mononucleosis still widely underdiagnosed in clinical practice. We report this rare condition and describe challenges in its diagnosis and treatment on the basis of a presented case., Case Presentation: A 6-year-old Caucasian girl without any ophthalmic history was admitted for right preseptal cellulitis requiring intravenous antibiotic therapy. During hospitalization, she developed a fluctuating lump in the nasolacrimal region which resembled an abscess, both clinically and radiologically. There was no spontaneous purulent discharge. Serology was positive for acute mononucleosis and Epstein-Barr virus-related dacryocystitis was diagnosed. Following multidisciplinary discussion, she was treated conservatively with digital lacrimal sac massages and intravenous antibiotic therapy with an excellent outcome., Discussion: This rare form of Epstein-Barr virus is poorly documented in the literature, and thus barely known. As initial symptoms are nonspecific (rhinitis, fever, eyelid edema and erythema lack of purulent discharge, and moderate bilateral cervical lymphadenopathy), diagnosis is often difficult. Nevertheless, differentiating between dacryocystitis and abscess is crucial to select the appropriate treatment and avoid unnecessary, potentially harmful surgery. Conservative management of dacryocystitis appears to be the gold standard of treatment., Conclusion: Acute dacryocystitis in children free of ophthalmic history should raise suspicion of primary Epstein-Barr virus infection. With conservative treatment, prognosis appears to be excellent; therefore, surgery should be avoided as much as possible., (© 2022. The Author(s).)

Objective: To compare the clinical and radiological features of various etiologies of chronic diffuse lacrimal gland enlargement., Materials and Methods: We retrospectively reviewed 91 consecutive patients who underwent surgical biopsy for chronic diffuse lacrimal gland enlargement and were diagnosed with non-specific dacryoadenitis (DA) (n = 42), immunoglobulin G4-related dacryoadenitis (IgG4-RD) (n = 33), and lymphoma (n = 16). Data on patient demographics, clinical presentation, and CT imaging findings (n = 73) and MRI (n = 43) were collected. The following radiologic features of lacrimal gland enlargement were evaluated: size, unilaterality, wedge sign, angle with the orbital wall, heterogeneity, signal intensity, degree of enhancement, patterns of dynamic contrast-enhanced, and apparent diffusion coefficient value. Radiological features outside the lacrimal glands, such as extra-lacrimal orbital involvement and extra-orbital head and neck involvement, were also evaluated. The clinical and radiological findings were compared among the three diseases., Results: Compared to the DA and IgG4-RD groups, the lymphoma group was significantly older (mean 59.9 vs. 46.0 and 49.4 years, respectively; p = 0.001) and had a higher frequency of unilateral involvement (62.5% vs. 31.0% and 15.2%, respectively; p = 0.004). Compared to the IgG4-RD and lymphoma groups, the DA group had significantly smaller lacrimal glands (2.3 vs. 2.8 and 3.3 cm, respectively; p < 0.001) and a lower proportion of cases with a wedge sign (54.8% vs. 84.8% and 87.5%, respectively; p = 0.005). The IgG4-RD group showed more frequent involvement of the extra-orbital head and neck structures, including the infraorbital nerve (36.4%), paranasal sinus (72.7%), and salivary gland (58.6%) compared to the DA and lymphoma groups (4.8%-28.6%) (all p < 0.005)., Conclusion: Patient age, unilaterality, lacrimal gland size, wedge sign, and extra-orbital head and neck involvement differed significantly different between lymphoma, DA, and IgG4-RD. Our results will be useful for the differential diagnosis and proper management of chronic lacrimal gland enlargement., Competing Interests: Chong Hyun Suh, Jung Hwan Baek, and Jeong Hyun Lee who is on the editorial board of the Korean Journal of Radiology was not involved in the editorial evaluation or decision to publish this article. All remaining authors have declared no conflicts of interest., (Copyright © 2022 The Korean Society of Radiology.)

Kimura's disease (KD) is a systemic inflammatory condition characterized by lymphadenopathy and subcutaneous nodules in the head and neck region. The lesions have a distinctive histopathological pattern formed by follicular hyperplasia, eosinophilic infiltrates, fibrosis, and vessel proliferation. The disease may occur at all ages but predominates among young males with autoimmune dysfunctions. Visceral and orbital involvement is uncommon. We report a girl with KD who developed bilateral enlargement of the lacrimal glands and a lesion in the left lateral ventricle of the brain indistinguishable from a central nervous system neoplasia. A biopsy of both the lacrimal gland and the lateral ventricle was consistent with KD., Competing Interests: The authors have no financial or conflicts of interest to disclose., (Copyright © 2022 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

We describe a retrospective case report of dacryoadenitis associated with orbital inflammatory disease in a patient with confirmed SARS-CoV-2 infection.A 22-year-old previously fit and healthy male presented with 4-day history of right ocular redness, eyelid swelling and blurred vision associated with discomfort and pain in the lacrimal gland area. He was found to have right acute dacryoadenitis based on clinical examination and orbital imaging. One day after initiation of oral antibiotic and non-steroidal anti-inflammatory therapy, he developed worsening of the orbital inflammation and partial ophthalmoplegia. Oral steroids were commenced resulting in rapid resolution of symptoms within a few days and clinical stability at 2 months.The patient did not have any systemic features of COVID-19 but he was in close contact with his mother and with his partner who both had respiratory symptoms and tested positive for SARS-CoV-2 antigen (PCR testing) 4 weeks prior. PCR testing from nasopharyngeal swab was negative for SARS-CoV-2 RNA; however, the serological test was positive for IgM/IgG SARS-CoV-2 antibodies. Extensive laboratory workup including infectious and autoimmune screening and chest x-ray were unremarkable.Orbital inflammatory disease due to infectious process or immunological response may potentially occur in COVID-19 patients, although the causal relationship remains uncertain.

Purpose: To study the presentation, management, and outcomes of infantile acute dacryocystitis., Methods: Retrospective study of infants diagnosed with acute dacryocystitis over a period from June 2016 to December 2019. Data collected include demographics, clinical history, presenting features, management, complications, and outcomes. Treatment provided was intensive medical care followed by early probing under endoscopic guidance. Further interventions, where needed, were performed based on intraoperative findings during probing. Successful outcomes were defined as resolution of infection, subjective relief from epiphora, and anatomical patency determined by a normal fluorescein dye disappearance test., Results: Twenty-seven eyes of 27 infants were analyzed during the study period. The mean age of infants was 3.26 months, and males were more affected than females (male:female 15:12). The mean duration of symptoms was 4.66 weeks, with 96% (26/27) presenting with symptoms of redness, watering, discharge, and swelling. Preseptal cellulitis was seen in 74% (20/27) infants, lacrimal abscess in 67% (18/27) infants, and lacrimal fistula in 37% (10/27). Most infants had complex congenital nasolacrimal duct obstruction (62%, 13/27), of which 7 also had intranasal cysts. One infant successfully underwent endoscopic dacryocystorhinostomy. At a mean follow-up period of 7.95 months, successful outcomes were observed in 90.4% (19/21) infants. The 2 failed cases were complex congenital nasolacrimal duct obstruction with associated sac diverticula and are scheduled for an endoscopic dacryocystorhinostomy., Conclusion: Infantile acute dacryocystitis is a distinct clinical entity. The outcomes of systemic antibiotics and early probing are excellent., Competing Interests: The authors have no financial or conflicts of interest to disclose., (Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

A 14-year-old boy was referred to the ophthalmology department with a 4-day history of rapid-onset right upper lid pain, swelling and erythema starting 9 hours after his first dose of COVID-19 mRNA vaccination (BNT162b2/Comirnaty, Pfizer-BioNTech). On examination, he had significant right upper lid ptosis, oedema and erythema, with associated limitation of right eye abduction and elevation. He was found to have acute dacryoadenitis with orbital inflammatory disease on clinical and laboratory investigations. He was given tapering oral prednisone and had full resolution of symptoms within 2 weeks. This is the first known case of orbital inflammation after COVID-19 mRNA vaccination. Given the temporal association between the patient's vaccination and symptom onset, we believe it is likely that immunisation prompted the onset of disease., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2022. No commercial re-use. See rights and permissions. Published by BMJ.)

A 45-year-old female patient who had previously undergone endoscopic dacryocystorhinostomy (EN-DCR) at another surgical center was diagnosed with functional failure after DCR and underwent an uneventful transcanalicular laser-assisted DCR (TCL-DCR). After 5 months, the patient underwent endoscopic examination due to persistent epiphora and dacryocystitis attacks. Endonasal visualization revealed a large dacryolith that filled the ostium and sac and was removed en bloc mechanically with forceps. Carbonized material on the dacryolith's superior part indicated that it was perforated through-and-through by the laser beam during TCL-DCR. The patient's complaints were resolved completely following the removal of the dacryolith. The transcanalicular lacrimal procedures do not allow complete visualization of the contents of the lacrimal sac, and the laser beam can ablate even an undetected dacryolith and may result in unsuccessful DCR, although the surgical course may look completely uneventful., Competing Interests: The authors have no financial or conflicts of interest to disclose., (Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Chronic granulomatous inflammation occurs rarely alongside pleomorphic adenomas of the major salivary glands but would not appear to have been reported with lacrimal gland adenomas. We describe the clinical features, imaging and histopathology for 4 patients (3 female) who had granulomatous inflammation alongside lacrimal gland adenomas-the patients being with age 39, 44, 48, and 53 years at time of surgery. One patient had an asymptomatic lesion found on imaging, and the other 3 had symptoms for between 3 years and several decades. Conjecturally, this rare phenomenon might arise from an inflammatory response to leakage of secretions from the ductular components of the glands., (Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

An 84-year-old man presented with a localized, firm, tender mass over the right lacrimal sac. He had a history of acute dacryocystitis in the same eye 6 months before presentation, which resolved with antibiotics followed by uneventful dacryocystorhinostomy. At repeat presentation, the patient underwent orbital imaging and excisional biopsy of the lesion. Histologic studies revealed a small cell neuroendocrine carcinoma. The patient was subsequently treated with chemotherapy and radiation. Although there are rare reports of small cell neuroendocrine carcinoma originating in the sino-orbital-lacrimal region, this is the first report of tumor presentation with acute dacryocystitis in a patient with prior dacryocystorhinostomy., Competing Interests: The authors have no financial or conflicts of interest to disclose., (Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Abstract: Septorhinoplasty is a surgical procedure frequently performed by surgeons dealing with facial aesthetic surgery and includes various risks. All surgical procedures have an increased risk of complications. Complication rate of nasal surgeries is approximately 4% to 19%. Although rhinoplasty is a nonsterile procedure, infections occur in less than 2% of all cases. This rate may decrease as the surgeon gains experience. Lacrimal ducts can be injured during nasal aesthetic surgery due to its anatomical structure. In addition to minor and temporary injuries, major injuries requiring surgical intervention may also occur. Most injuries to the lacrimal system remain asymptomatic. Postrhinoplasty dacryocystitis usually occurs secondary to soft-tissue edema that compresses the lacrimal system and typically resolves within 1 to 2 weeks. After reviewing the literature and examining possible traumatic mechanisms, we recommend that the infections in the lacrimal system should be treated with medical therapy before performing surgical interventions such as dacryocystorhinostomy and abscess drainage. Knowing the complications of surgical interventions before the study and administering some basic principles before and after surgery are highly essential. In this study, we present a relatively rare complication of septorhinoplasty known as acute postoperative dacryocystitis, which is mostly encountered in the early postoperative period., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 by Mutaz B. Habal, MD.)

Competing Interests: The authors declare no conflict of interest.

Epiphora is the first symptom of acquired nasolacrimal duct obstruction (NLDO), which may be due to various causes, including paranasal sinuses and tumours of the nasal cavity. A 28-year-old male patient presented chronic dacryocystitis with left-sided epiphora. Endoscopically, a bulging of the lateral nasal wall at the left-sided agger nasi area was noted. Imaging studies (CT and MRI) revealed a left-sided giant agger nasi mucocele. An endonasal endoscopic dacryocystorhinostomy (DCR) as well as opening of the agger nasi cell with mucocele removal and anterior ethmoidectomy was performed. Based on the literature, intranasal causes have very often occurred in patients with chronic dacryocystitis, but not reported as the cause of inflammation in the mucocele of agger nasi. Identification and recognition of this endonasal rare cause of NLDO-like agger nasi mucocele entity may facilitate timely intervention and can be treated successfully with a simultaneous endoscopic DCR procedure., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2021. No commercial re-use. See rights and permissions. Published by BMJ.)

Amid the global coronavirus disease 2019 (COVID-19) outbreak, an 89-year-old male with chronic kidney disease presented with acute dacryocystitis and a persistent dry cough. After a course of antibiotics, external dacryocystorhinostomy was performed under local anesthesia without sedation. During planned hemodialysis in the early hours after the procedure, the patient developed nausea and hematemesis followed by severe dyspnea and hypoxemia. The patient was diagnosed with aspiration pneumonia, a previously unreported complication in lacrimal surgery., Competing Interests: The authors have no financial or conflicts of interest to disclose., (Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

This report presents two cases of epidemic keratoconjunctivitis (EKC)-associated dacryocystitis. Both patients presented with preseptal edema. Orbital computed tomography (CT) scans were performed out of concern for orbital cellulitis and revealed soft tissue edema involving the preseptal aspect of the eyelids and also the lacrimal sac. Both patients were initially diagnosed with bacterial preseptal cellulitis with dacryocystitis and admitted for treatment with intravenous antibiotics. After no improvement on antibiotics, both patients were noted to have exam findings consistent with viral conjunctivitis, and one patient had a positive conjunctival swab for adenovirus. Both patients were subsequently treated with topical steroids, and symptoms improved. EKC has previously been identified as a cause of acquired nasolacrimal duct obstruction and canalicular stenosis, and acute inflammation of the lacrimal apparatus may explain this link. Dacryocystitis may also be contributory to the copious tearing seen in patients with epidemic keratoconjunctivitis.

Introduction: Contracture of dacryocyst by an implanted lacrimal silicone tube is rare. This report describes a unique case of secondary dacryocystitis and the contracture of dacryocyst caused by a lacrimal silicone tube that was placed in the lacrimal system for 10 years., Patient Concerns: A 63-year-old female was diagnosed with chronic dacryocystitis at a local hospital and underwent surgical treatment 10 years ago. In the past month, the patient complained of persistent tearing and purulent secretion from the eyes., Diagnosis: The patient was diagnosed with secondary dacryocystitis, based on clinical features and the presence of the silicone stent, granulation tissue formation, and dacryocyst contracture in the lacrimal duct, as observed by nasal endoscopy., Interventions: For treatment, the implanted silicone tube in the patient was removed, the lacrimal duct and nasal mucosa was anastomosed, and a new lacrimal silicone tube was placed again., Outcomes: Following the surgery, the patient recounted that there were no symptoms, and follow-up examinations performed over a 1-month period posttreatment revealed no recurrence of obstruction or dacryocystitis. Therefore, the surgeon removed the lacrimal drainage tube and asked the patient to return to the outpatient department regularly for examination., Conclusion: The findings, in this case, suggest that silicone tubes are safe and effective, and can be placed in the lacrimal drainage system. However, in this patient, prolonged intubation caused chronic inflammation, granulation tissue formation, and dacryocyst contracture. Our findings could inform surgeons to consider the reasonable duration of intubation for treating cases of lacrimal obstruction, in order to avoid unnecessary complications.

A 14-year-old female with juvenile idiopathic arthritis presented with left-sided recurrent painful periorbital swelling of 5 months duration. MRI revealed enlargement of the left lacrimal gland with extensive inflammatory change involving the orbital and periorbital soft tissues. Lacrimal gland biopsy showed evidence of chronic inflammation, and she had intralesional steroid injection at the time of biopsy. She remained asymptomatic at 6-month follow-up visit. Although juvenile idiopathic arthritis is usually related to intraocular inflammation, it may rarely be associated with dacryoadenitis.

Immunoglobulin G4-related disease (IgG4-RD) is an autoimmune disorder characterized by the infiltration of one or more organs with IgG4-positive plasma cells resulting in inflammatory lesions and fibrosis. Although the pancreas is the most commonly affected organ, involvement of extrapancreatic organs is an increasingly recognized manifestation of the disease. Patients may be asymptomatic and serum IgG4 concentrations may be elevated or normal. Treatment consists of glucocorticosteroid treatment, with excellent response. A definitive diagnosis requires histopathology with imaging playing a key role in avoiding treatment delays. This pictorial review will focus on the most current knowledge regarding IgG4-RD including its common and less common manifestations and the roles of multidetector CT, MRI and ultrasound in the diagnosis and management of suspected IgG4-RD. Knowledge of the varied imaging findings of this multi systemic disease is essential for radiologists to avoid misdiagnosis and assist with timely and effective treatment.

Objective: To present outcomes of neonatal dacryocystoceles treated through endoscopic intranasal cyst marsupialization (EICM) without general anesthesia and nasolacrimal duct (NLD) probing or irrigation., Methods: Records of eleven consecutive infants diagnosed with unilateral or bilateral congenital cysts associated with the nasolacrimal system between January 2016 and February 2019 at our institution were retrospectively reviewed., Results: Age at diagnosis ranged from 0 to 56 days, and 45.5% were male. 54.5% had dacryocystitis before surgical intervention. Patients were initially treated with a combination of massage and topical antibiotics; some received parenteral antibiotics if infected. Nearly all patients (90.9%) had one or more intranasal cysts. Of these patients, two underwent lacrimal probing and EICM in the operating room. One underwent EICM only in the operating room. Another patient initially failed bedside NLD probing and subsequently underwent bedside nasal endoscopy and awake EICM. The final six patients underwent EICM without general anesthesia or NLD probing. No complications were noted. Follow-up ranged from 7 to 906 days. Complete resolution was observed in all ten patients who underwent EICM, regardless of anesthesia., Conclusions: Neonatal dacryocystoceles and/or intranasal cysts are successfully treated at the bedside through nasal endoscopy with simple awake endoscopic intranasal cyst marsupialization. Avoidance of general anesthesia and NLD probing or irrigation would greatly simplify and decrease the cost of dacryocystocele management., (Copyright © 2019 Elsevier B.V. All rights reserved.)

Ocular glands play a critical role in eye health through the secretion of factors directly onto the ocular surface. The cornea is a normally transparent tissue necessary for visual acuity located in the anterior segment of the eye. Corneal damage can occur during microbial infection of the cornea, resulting in potentially permanent visual deficits. The involvement of ocular glands during corneal infection has been only briefly described. We hypothesized that ocular glands contribute to resistance as an arm of the eye-associated lymphoid tissue and may also be susceptible to infection secondary to microbial keratitis. Utilizing a mouse model of herpes simplex virus 1 (HSV-1) keratitis, we found that infection of corneas resulted in subsequent infection of ocular glands, including harderian glands (HGs) and extraorbital glands. Similarly, infection of corneas with Pseudomonas aeruginosa resulted in secondary infection of ocular glands. A robust immune response, characterized by increased numbers of immune cells and inflammatory mediators, occurred within ocular glands following HSV-1 keratitis. Removal of HGs altered corneal resistance to HSV-1, as measured by increased viral load, decreased corneal edema, and decreased inflammatory cell infiltration. These novel findings suggest that ocular glands are involved in microbial keratitis through their susceptibility to secondary infection and contribution to corneal resistance. IMPORTANCE Microbial keratitis accounts for up to 700,000 clinical visits annually in the United States. The involvement of ocular glands during microbial keratitis is not readily appreciated, and treatment options do not address the consequences of ocular gland dysfunction. The present study shows that ocular glands are susceptible to direct infection by common ocular pathogens, including HSV-1 and Pseudomonas aeruginosa , subsequent to microbial keratitis. Additionally, ocular glands contribute soluble factors that play a role in corneal resistance to HSV-1 and alter viral load, corneal edema, and immune cell infiltration. Further studies are needed to elucidate the mechanisms by which this occurs., (Copyright © 2019 American Society for Microbiology.)

Cathepsin S (CTSS) is highly increased in Sjögren's syndrome (SS) patients tears and in tears and lacrimal glands (LG) of male non-obese diabetic (NOD) mice, a murine model of SS. To explore CTSS's utility as a therapeutic target for mitigating ocular manifestations of SS in sites where CTSS is increased in disease, the tears and the LG (systemically), the peptide-based inhibitor, Z-FL-COCHO (Z-FL), was administered to 14-15 week male NOD mice. Systemic intraperitoneal (i.p.) injection for 2 weeks significantly reduced CTSS activity in tears, LG and spleen, significantly reduced total lymphocytic infiltration into LG, reduced CD3+ and CD68+ cell abundance within lymphocytic infiltrates, and significantly increased stimulated tear secretion. Topical administration of Z-FL to a different cohort of 14-15 week male NOD mice for 6 weeks significantly reduced only tear CTSS while not affecting LG and spleen CTSS and attenuated the disease-progression related reduction of basal tear secretion, while not significantly impacting lymphocytic infiltration of the LG. These findings suggest that CTSS inhibitors administered either topically or systemically can mitigate aspects of the ocular manifestations of SS.

Tularemia is a zoonotic disease caused by Francisella tularensis , a highly virulent gram-negative coccobacillus. Oropharyngeal tularemia, one of the clinical subtypes, is the most common clinical form of the disease in Eastern Europe, including Turkey. This clinical form affects mostly the head and neck region and the most common complaints of patients are mass in the neck, sore throat, and fever. This form of tularemia may be confused with tonsillitis, pharyngitis, or cervical lymphadenitis caused by other microbial agents due to the nonspecific clinical and laboratory features. In this study, we present a patient with nasolacrimal duct obstruction and dacryocystitis caused by oropharyngeal tularemia.

Background: Pediatric acute dacryocystitis typically develops due to persistence of Hasner's membrane. Pediatric paranasal mucoceles are rare entities. In contrast, chronic dacryocystitis in cases of congenital dacryostenosis is one of the most frequent pediatric ophthalmologic issues., Methods: The case report of a 10-year-old girl suffering acute dacryocystitis is presented., Results: The area around the left lacrimal sac showed a painful swelling with edema and hyperemia. Systemic and local antibiotic therapy resulted in only slight improvement. Dacryoendoscopy detected acute dacryocystitis with significant stenosis of the nasolacrimal duct. The nasolacrimal duct was widened dacryoendoscopically and autostable bicanalicular nasolacrimal intubation was performed. Nevertheless, the lacrimal ducts were blocked subtotally and a widening of the ethmoid on the left side was shown by rhinoscopy. Magnetic resonance imaging confirmed a frontoethmoidal mucocele which was treated by marsupialization. During the 24-month follow-up there was no recurrence of acute dacryocystitis. There were no signs of sinusitis., Conclusion: Paranasal mucoceles, e. g., ethmoidal mucoceles, can compress the lacrimal pathways and cause acute lacrimal inflammation in childhood. Paranasal mucoceles should thus be excluded in cases of unclear masses in the lacrimal region.

Purpose: To describe the outcome of external dacryocystorhinostomy (DCR) as a suitable alternative to dacryocystectomy (DCT) in cases of isolated lacrimal sac rhinosporidiosis., Methods: This was a retrospective, interventional case series. Institutional review board approval was obtained for the study. The chart review of 13 patients who underwent external DCR surgery for isolated lacrimal sac rhinosporidiosis between July 2012 and May 2018 was performed. Demographic details, clinical presentation, preoperative and intraoperative findings, surgical technique used, postoperative management, surgical outcome and duration of follow up were reviewed for each patient., Results: Thirteen patients were included in the study. Nine (69.2%) were male and four (30.8%) were female. The mean age was 22.1 years (range: 8-46 years). Involvement was unilateral in all cases. The commonest presenting complaint was epiphora with discharge seen in 10 cases (76.9%). An intrasac granuloma was grossly identified intraoperatively in 12 out of the 13 patients. Mean follow up was 26.9 months (range: 1.5-68 months). Till the last follow up, all cases were symptom free and did not have any evidence of recurrence of infection., Conclusion: From this series of cases reported by the authors, external DCR with appropriate precautions to prevent recurrence appears to be a suitable alternative to conventional DCT with excellent long-term outcomes without disease recurrence., Competing Interests: None

Ultra-thin porous polyethylene has been widely preferred in orbital blow out fracture repair for many years as a safe and stable material. Delayed complications related to the implant in orbital blow out fracture repair are very rare and usually begin as a periorbital inflammation. Infections and tissue reaction to the foreign body are regarded as main causes for implant-related inflammation of which symptoms may vary among the patients and may involve subacute and chronic eyelid swelling, skin redness, pain, conjunctival injection, proptosis, and acute orbital inflammation.Presented here is a patient of recurrent periorbital inflammation who underwent orbital floor repair with ultra-thin porous polyethylene sheet which was complicated by dacryocystitis in the late follow-up period.

Sarcoidosis of lacrimal sac is a very uncommon disease. When the patient has symptoms of lacrimal sac diseases, it is necessary to take into account the histological evaluation, to demonstrate the involvement of the lacrimal sac by this pathology. In patients with sarcoidosis history, the treatment is surgical and it is suggested to do an external dacryocystorhinostomy, in order to take a biopsy, to get a diagnosis and to establish the incidence of this pathology. The case is presented of a 44 year-old female, with a history of sarcoidosis lymph nodes, who presented with a right chronic dacryocystitis of one year onset., (Copyright © 2018 Sociedad Española de Oftalmología. Publicado por Elsevier España, S.L.U. All rights reserved.)

Extranodal natural killer (NK-)/T cell lymphoma, nasal type (ENKL), 1 is a rare disease that often mimics rheumatological and infectious conditions and can therefore be difficult to diagnose. The authors present a case of a 55-year-old Vietnamese woman who was misdiagnosed with severe atrophic rhinitis and chronic sinus osteitis. Over a period of 8 months from initial referral, she underwent multiple biopsies and was treated with various antimicrobial regimens until the histopathological diagnosis of ENKL was finally made. Her presentation was complicated by bacterial dacrocystitis, preseptal cellulitis and a retrobulbar extraconal phlegmon requiring surgical drainage. She also subsequently developed a naso-oral fistula on treatment. This case highlights the importance of repeated biopsies, in particular from non-necrotic regions of the sinonasal tract when a patient does not respond to therapy and clinical suspicion of neoplastic pathology remains. This is the first case of ENKL to describe significant orbital complication., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2019. No commercial re-use. See rights and permissions. Published by BMJ.)

Ocular involvement is present in 50-60% of granulomatosis with polyangiitis (GPA) patients and can affect any part of the ocular globe. The present study describes ophthalmologic manifestations, association with systemic symptoms, disease activity and damage in GPA. A cross-sectional study was conducted including patients with GPA who underwent rheumatologic and ophthalmologic evaluation. Demographics, comorbidities, ophthalmologic symptoms, serologic markers, radiographic studies, disease activity and damage were assessed. Descriptive statistics, correlation, univariable logistic regression analyses, Student's t, Mann-Whitney U, Chi-square and Fisher's exact tests were performed. Fifty patients were included, 60% female, the median age was 56 years, disease duration 72.5 months. Nineteen (38%) patients had ocular manifestations at GPA diagnosis, scleritis being the most frequent; 27 (54%) patients presented ocular involvement during follow-up, repeated scleritis and dacryocystitis being the most common manifestations. Concomitant ophthalmic and sinonasal involvement was present in 12 (24%). Ocular and ENT damage occurred in 58% and 70%, respectively. Epiphora and blurred vision were the most frequent symptoms; scleromalacia and conjunctival hyperemia (27%) the most frequent clinical abnormalities. Ocular involvement at diagnosis was associated with concomitant ocular and sinonasal involvement at follow-up (OR 4.72, 95% CI 1.17-19.01, p = 0.01). Ocular involvement at follow-up was associated with age at GPA diagnosis (OR 0.94, 95% CI 0.90-0.99, p = 0.03), VDI (OR 1.29, 95% CI 1.03-1.61, p = 0.02), and ENT damage (OR 5.27, 95% CI 1.37-20.13, p = 0.01). In GPA, ocular involvement is frequent, therefore, non-ophthalmologist clinicians should be aware of this manifestation to reduce the risk of visual morbidity and organ damage.

Sarcoidosis rarely develops in the lacrimal sac. Lacrimal sac sarcoidosis may be associated with sarcoidosis-related rhinosinusitis. The anatomical and histological relationship between the lacrimal drainage system and nasal mucosa has been indicated as a predisposing factor for this concomitance. However, the characteristics of this condition are not fully understood. Herein, the authors report a case where dacryocystitis and rhinosinusitis developed secondary to sarcoidosis. Pathological examination of the lacrimal sac wall revealed noncaseating epithelioid granulomas in the subepithelial layer, which contains the lacrimal-drainage-associated lymphoid tissue. The lacrimal-drainage-associated lymphoid tissue comprises the main immune mechanism of the lacrimal system and forms a functional unit with the lacrimal gland, conjunctiva, and nasal mucosa to maintain ocular surface integrity through lymphocyte recirculation. Because sarcoidosis is an autoimmune disorder, this lymphocyte recirculation may be associated with the concurrent nasal lesions in the authors' case.