Bilateral Dacryoadenitis and Central Nervous System Involvement in a Child With Kimura Disease.

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.

Chronic Granulomatous Dacryoadenitis Associated With Pleomorphic Adenoma.

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.

Small Cell Neuroendocrine Carcinoma Presenting as Recurrent Dacryocystitis: Case Report of a Rare Entity.

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.

Epiphora and Dacryocystitis After Transcanalicular Laser-assisted Dacryocystorhinostomy due to an Undiagnosed Dacryolith.

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.

Congenital Nasolacrimal Duct Obstruction Update Study (CUP Study): Paper 4-Infantile Acute Dacryocystitis (InAD)-Presentation, Management, and Outcomes.

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.

Dacryocystitis With Skin Defects Secondary to Septorhinoplasty.

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.

Acute dacryoadenitis in a patient with SARS-CoV-2 infection.

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.

Natural killer T-cell lymphoma causing bilateral recurrent recalcitrant dacryocystitis.

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.

Aspiration Pneumonia After External Dacryocystorhinostomy Under Local Anesthesia Without Sedation: A Case Report.

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.

Ocular Glands Become Infected Secondarily to Infectious Keratitis and Play a Role in Corneal Resistance to Infection.

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.

Dacryoadenitis Associated With Juvenile Idiopathic Arthritis.

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.

Two cases of epidemic keratoconjunctivitis-associated dacryocystitis.

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.

Clinical significance of ocular manifestations in granulomatosis with polyangiitis: association with sinonasal involvement and damage.

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.

A Rare Cause for Recurrent Implant-Related Inflammation of Orbit: Dacryocystitis.

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.

Dacryocystitis and Rhinosinusitis Secondary to Sarcoidosis.

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.

[Pediatric acute dacryocystitis due to frontoethmoidal mucocele]. / Akute Dakryozystitis im Kindesalter bei frontoethmoidaler Mukozele.

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.

Orbital Inflammation as a Presenting Sign for CREST Syndrome.

A 61-year-old male was referred with a week's history of a painful and swollen left eye. Examination revealed normal visual acuities, left proptosis and global restriction of ocular ductions, and subretinal fluid at the macula. CT imaging confirmed thickening of the posterior scleral coat, with an associated choroidal effusion. Serology revealed positive antinuclear antibodies with a centromere staining pattern; subsequent rheumatology review revealed extensive telangiectasia with digital ulceration in both hands, and a diagnosis of limited cutaneous systemic sclerosis was made. Orbital inflammatory disease is often the initial presentation of systemic diseases such as sarcoidosis, granulomatosis with polyangiitis, and IgG4 disease. Limited cutaneous systemic sclerosis is rarely encountered in the context of orbital inflammation, but is a further systemic association, reminding the clinician of the diagnostic importance of peripheral symptoms and serological markers in patients presenting with orbital inflammation and scleritis.

Herpes Zoster Ophthalmicus With Orbital Findings Preceding Skin Rash.

The authors describe 2 patients who presented with orbital findings and later developed vesicular lesions that were positive for varicella zoster virus and consistent with Herpes Zoster ophthalmicus. One case is the first to involve dacryoadenitis and orbital myositis preceding disseminated Herpes Zoster. In the other case, a patient developed zoster orbital syndrome leading to elevated intraocular pressure, loss of vision, and afferent pupillary defect. Canthotomy and cantholysis were required to restore vision. In both cases, the orbital syndrome developed prior to the vesicular rash. These cases highlight the need to consider Herpes Zoster ophthalmicus in patients with orbital syndrome not responding to conventional treatment.

Dacryoadenitis as the Initial Presentation of a Natural Killer T-Cell Lymphoma.

Primary orbital natural killer T-cell lymphoma (NKTCL) is a rare condition with only a few published cases in the literature. Over 1 month, an 81-year-old man developed progressive left periocular inflammation unresponsive to treatment. Clinical examination and imaging studies demonstrated a left lacrimal gland enlargement. Bilateral anterior uveitis and erythematous nontender cutaneous lesions were also found. Biopsies of the skin and lacrimal gland on the back revealed histopathologic and immunohistochemical findings confirming Epstein-Barr virus-positive NKTCL. Metastatic work up disclosed multifocal involvement in the pancreas, stomach, and chest wall. Palliative treatment consisting of nonanthracycline-based chemotherapy and radiation was instituted, but the patient died 5 months after the onset of symptoms. This is the first example of acute dacryoadenitis, and the second of bilateral anterior uveitis, in the setting of NKTCL. Absence of naso-sinus involvement in the current case is rare in NKTCL. Despite treatment, the prognosis remains dismal. Orbital specialists should include NKTCL in the differential diagnosis of lacrimal gland/orbital masses and perform an incisional biopsy if the clinical scenario so dictates.