Prenatal diagnosis and outcome of congenital dacryocystoceles.

OBJECTIVE: To determine the incidence and present our experience with prenatal diagnosis and postnatal outcome of dacryocystocele. MATERIAL AND METHODS: All cases of congenital dacryocystocele diagnosed in our center between 2020 and 2022 were identified in our database to establish the incidence of these defects. The medical records were then reviewed for gestational age, gender, size, and side of dacryocystocele and postnatal outcome. RESULTS: A total of 26 cases with dacryocystoceles were found at a mean gestation age of 30 weeks (range, 29-33 weeks). The overall incidence was 1.35%, there was an obvious female predominance (73%), 69% of cases were unilateral and 31% were bilateral. There were no serious associated anomalies. The postnatal outcome was obtained in 88% of cases (23/26), in 39% (9 out of 23) cases the dacryocystocele was confirmed postnatally, and in 7 (77%) of these it was complicated by dacryocystitis. The spontaneous resolution was more likely in the right-sided lesions, and this was statistically significant. The treatment in cases with dacryocystitis involved massage and local antibiotics and was successful in 71% of cases. 2 cases (29%) suffer from recurrent dacryocystitis and are followed up with recurrent probing and local antibiotics. No breathing difficulties were described postnatally in our study group. CONCLUSION: The overall prenatal incidence of dacryocystocele was 1.35%. The outcome is favorable, 61% of dacryocystoceles in our study resolved spontaneously and in no case postnatal breathing complications were reported. Dacryocystitis was common in persisting cases but was usually treated successfully by massage and antibiotics. The right-sided dacryocystoceles are more likely to resolve spontaneously than left-sided, and this was the only significant factor predicting persistence.

Magnetic resonance diffusion-weighted imaging in lacrimal gland lymphoma versus inflammation: A comparative study.

PURPOSE: To determine the role of diffusion-weighted imaging (DWI) with apparent diffusion coefficient (ADC) in differentiating the inflammatory process and malignant lymphoma of the lacrimal gland. METHODS: A retrospective analysis of all subjects who underwent lacrimal gland biopsy and magnetic resonance (MR) imaging with DWI sequences during a 10-year period at the Sheba Medical Center, Israel. The lacrimal glands' ADC values were documented bilaterally by blinded observers and correlated with the final histology verified diagnoses. RESULTS: Twenty-eight patients were included, with 19 females (68%) with a mean ± SD age of 48.1 ± 25.7 years. The right orbit was involved in 14 cases (50%) and the left in 13 (46%); one patient (4%) had bilateral involvement. Seventeen cases (61%) had a final diagnosis of idiopathic inflammation or dacryoadenitis, and six cases (21%) were diagnosed with lymphoma. Additional diagnoses included pleomorphic adenoma in 3 (11%), adenoid cystic carcinoma, and solitary fibrous tumor. Lower mean ADC values were observed in the lymphoma versus inflammatory group (1.03 × 10-3 Vs. 1.45 × 10-3, P = 0.02). CONCLUSION: Restricted diffusion on MR imaging can serve as a diagnostic tool in the differentiation between inflammatory processes of the lacrimal gland and lymphoma.

Dacryoadenitis: do not forget ANCA vasculitis.

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.

Computed Tomography Signs of Sinonasal Inflammation in Patients with Acute Dacryocystitis.

BACKGROUND: Nasal and paranasal sinus abnormalities may be related to nasolacrimal duct obstructive disease but are strongly debated. Data of acute disease stage are lacking. OBJECTIVE: The purpose of this study was to determine if there are correlations between radiologic signs of sinus inflammation and acute dacryocystitis (AD). METHODS: This cross-sectional controlled study was conducted at Wenzhou, Zhejiang Province, China from February 2021 to November 2023. Forty-four consecutive patients with AD and 50 consecutive patients with orbital tumors (the control group), who completed preoperative computed tomography scans, were enrolled to evaluate the extent of their inflammatory sinonasal disease by the modified Lund-Mackay score system. RESULTS: The inflammation signs of the paranasal sinuses (total mean sinus scores, 95% CI [0.00, 2.00]; P < 0.001), namely the anterior ethmoid sinus(95% CI [0.00, 1.00]; P < 0.001), the posterior ethmoid sinus(95% CI [0.00, 0.00]; P = 0.003), the frontal sinus (95% CI [0.00, 0.00]; P = 0.02), and the ostiomeatal complex (P < 0.001) were more extensive in patients with AD when compared with the controls. The disease course was negatively correlated with the anterior ethmoid (P = 0.03) and frontal scores (P = 0.01). The symptom of eyelid swelling was positively correlated with the anterior ethmoid (P = 0.03), ostiomeatal complex (P = 0.004), and total sinus scores (P = 0.005). CONCLUSION: Inflammatory sinus disease was found to be more frequent in patients with AD, which was gradually alleviated with the prolongation of the disease course. The mutual spread of inflammation particularly in the acute course may play an important role in lacrimal duct obstructive disease.

Microbiology of acute bacterial dacryocystitis: a tertiary institutional experience in South Australia.

PURPOSE: To provide a comprehensive microbiological profile of bacterial dacryocystitis in South Australia. By identifying the specific microorganism and antibiotic susceptibility, this study intends to aid ophthalmologists in choosing appropriate empirical antibiotic therapies and development of evidence-based clinical guidelines. METHOD: A retrospective study was conducted at the Royal Adelaide Hospital (RAH) over five years (2018-2023) of patients with acute dacryocystitis. The study included 43 patients, and data encompassed demographic information, clinical presentation, microbiological analysis, management, and outcomes. Patients with chronic dacryocystitis were excluded. RESULTS: Among the 43 patients included in the study (female 28 (65%), mean age: 64 years old), the most common clinical features were pain (74%) and swelling (70%). Organisms were identified in 49% of patients, with the predominant bacteria being Staphylococcus aureus (42%), Streptococcus species (19%), and Escherichia coli (8%). Aggregatibacter species (8%), Morganella morganii (4%), Enterobacter cloaceae (4%), Hafnia alvei (4%), mixed anaerobes (4%), E coliforms (4%) and Pseudomonas aeruginosa (4%) were also identified. The most frequently prescribed empirical antibiotics were amoxicillin-clavulanic acid (50%), flucloxacillin (33%) and cefalexin (18%). CONCLUSION: The microbiological trends of acute dacryocystitis have largely remained consistent, with a predominance of Gram positive organisms. This is the most recent profile analysis of acute dacryocystitis in South Australia and will help form evidence-based clinical guidelines.

Langerhans cell histiocytosis mimicking acute dacryocystitis.

Langerhans cell histiocytosis (LCH) is a myeloid neoplasm characterized by clonal neoplastic proliferation of Langerhans-type dendritic cells associated with an inflammatory infiltrate predominantly composed of lymphocytes and eosinophils. In this article, we present an unusual case of LCH with significant swelling in the left lacrimal sac region in a 3-year-old child, clinically mimicking acute dacryocystitis. Microscopically, it showed intense inflammatory infiltrate and histiocytes with irregular nuclei. The tumor cells were positive for S-100 protein, CD1a, and CD207 (langerin). Molecular study was positive for the V600E/E2/D mutation (EXON 15). This case emphasizes the importance of careful clinical, radiographic, and microscopic evaluation, as some neoplasms may mimic common benign lesions.

AS101 regulates the Teff/Treg balance to alleviate rabbit autoimmune dacryoadenitis through modulating NFATc2.

Sjögren's syndrome (SS) dry eye can cause ocular surface inflammation and lacrimal gland (LG) damage, leading to discomfort and potential vision problems. The existing treatment options for SS dry eye are currently constrained. We investigated the possible therapeutic effect and the underlying mechanism of AS101 in autoimmune dry eye. AS101 was injected subconjunctivally into a rabbit model of autoimmune dacryoadenitis and its therapeutic effects were determined by evaluating clinical and histological scores. The expressions of effector T cells (Teff)/regulatory T cells (Treg)-related transcription factors and cytokines, inflammation mediators, and transcription factor NFATc2 were measured by quantitative real-time PCR and/or Western blot both in vivo and in vitro. Additionally, the role of NFATc2 in the immunomodulatory effects of AS101 on T cells was explored by co-culturing activated peripheral blood lymphocytes (PBLs) transfected with NFATc2 overexpression lentiviral plasmid with AS101. AS101 treatment potently ameliorated the clinical severity and reduced the inflammation of LG. Further investigation revealed that AS101 treatment led to decreased expression of Th1-related genes (T-bet and IFN-γ) and Th17-related genes (RORC, IL-17A, IL-17F, and GM-CSF) and increased expression of Treg-related gene Foxp3 in vivo and in vitro. Meanwhile, AS101 suppressed the expression of TNF-α, IL-1ß, IL-23, IL-6, MMP-2, and MMP-9. Mechanistically, AS101 downregulated the expression of NFATc2 in inflamed LGs. Overexpression of NFATc2 in activated PBLs partially blunted the effect of AS101 on Teff suppression and Treg promotion. In conclusion, AS101 is a potential regulator of Teff/Treg cell balance and could be an effective treatment agent for SS dry eye.

Herpes Simplex Virus Dacryoadenitis Preceding Skin Vesicles.

Herpes simplex virus involvement of the orbit is rare. We report the case of an immunocompetent patient who presented with preseptal edema, erythema, decreased vision, and dacryoadenitis prior to the appearance of vesicular skin lesions and in the absence of corneal dendrites. The patient's clinical picture failed to improve on empiric therapy with intravenous antibiotics. The patient subsequently developed skin vesicles and a nucleic acid amplification swab of the ocular surface was positive for herpes simplex virus-1. The patient demonstrated rapid clinical improvement after initiation of oral antivirals. This is an unusual case of presumed herpes simplex virus dacryoadenitis preceding skin vesicles in an immunocompetent patient.

The incidence of pediatric dacryocystitis among a population-based cohort of infants with congenital nasolacrimal duct obstruction.

PURPOSE: To report the incidence, clinical characteristics, and outcomes of acute dacryocystitis among a large, population-based cohort of children born with congenital nasolacrimal duct obstruction (CNLDO) over a 10-year period. METHODS: This multicenter retrospective, population-based cohort study included all patients diagnosed with acute dacryocystitis in a cohort of patients diagnosed with CNLDO before age 5 years in Olmsted County, Minnesota, United States of America from January 1, 1995, through December 31, 2004. RESULTS: Of 1,998 patients with CNLDO, there were 70 cases (36 female [(51%)]) of acute dacryocystitis during the study, yielding an incidence rate of 243 per 100,000 children (95% CI, 170-316). Mean age at diagnosis was 9.0 months. Patients who developed dacryocystitis were significantly less likely to be born via C-section (OR = 0.29, P = 0.009). Less than half of patients with dacryocystitis were treated with oral/intravenous antibiotics (46%), but whose who were had a significantly higher odds of requiring probing (OR = 8.50, P = 0.004). Spontaneous CNLDO resolution was significantly less likely to occur in patients diagnosed with acute dacryocystitis compared with those without (OR = 2.46, P = 0.001). The median age of spontaneous resolution in the dacryocystitis group (6.0 months) was significantly older than the uncomplicated CNLDO group (P = 0.012). CONCLUSIONS: Pediatric acute dacryocystitis is an uncommon complication of CNLDO and is associated with both a lower likelihood of and older age at spontaneous resolution of CNLDO symptoms.

Medication-associated orbital inflammation: A systematic review.

We performed a comprehensive systematic review to identify medication-associated orbital inflammation and to characterize its clinico-radiological features. We reviewed English-language articles describing medication-associated orbital inflammation (i.e., orbital myositis, dacryoadenitis and orbital fat) published to June, 2023. Isolated inflammation of the intraocular structures or globe alone (i.e. uveitis, scleritis, optic neuritis and perineuritis) were excluded. In medication-associated orbital inflammation, the extraocular muscles are preferentially affected, occurring in isolation or in combination with other orbital and/or intraocular structures. Clinico-radiological manifestations may be non-specific; however, certain medications may be distinguished according to the presence of systemic prodrome, laterality, associated intraocular inflammation, and predisposition to involve certain orbital structures. Rapid identification, discontinuation of the provoking medication, and systemic corticosteroid therapy (if appropriate) typically achieves a favorable visual prognosis. As new medications become adopted by clinicians, rare adverse effects will be further delineated.Medication-associated orbital inflammation is an important diagnostic consideration in orbital inflammatory disease. A careful medication history and clinical assessment may be revealing, permitting timely discontinuation of the offending agent and initiation of appropriate management.