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.

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.

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.

Repeated Lacrimal Gland Biopsies.

PURPOSE: To examine the indications for repeated lacrimal gland biopsies, and the rate of detection of a new diagnosis. METHODS: A single-center, retrospective review of patients who underwent more than 1 lacrimal gland biopsy, either ipsilateral or contralateral, between 2000 and 2022. RESULTS: One hundred and twenty-three patients (80 female; 65%) had repeated lacrimal gland biopsy. The commonest diagnosis on initial biopsy was chronic nonspecific dacryoadenitis (NSD) (49/123; 40%). Indications for repeated biopsy were uncertainty in making a histopathological diagnosis (16/123; 13%), poorly-responsive or recurrent ipsilateral disease (61/123; 50%), new or continued/worsening contralateral disease (30 patients; 24%), and planned tumor resection after initial biopsy (16/123; 13%). Of the 40 patients (33%) with a different histopathological diagnosis after repeated lacrimal biopsy, 4 (10%) had lymphoma, initially reported as NSD (4/49 with NSD; 8%), and 7/40 (18%) (14% of the 49 NSD patients) were reclassified as having specific inflammations (including 2 with granulomatous polyangiitis); of the 7 having reclassification as a specific dacryoadenitis, 6/7 had ipsilateral disease failing to respond to primary treatment, and 1/7 had new onset or progression of contralateral disease. All histology after the primary biopsy of 16 patients with lacrimal gland malignancies retained the same tissue diagnosis. CONCLUSION: Repeated biopsy for lacrimal gland disease in this study revealed a diagnosis of malignancy in 20%, including lymphoma in 8% of those initially diagnosed with NSD. There was a 14% rate of diagnostic progression from "non-specific" dacryoadenitis to a more specific inflammatory disease.

Orbital Abscess Secondary to Nasolacrimal Duct Obstruction in an Extremely Preterm Infant.

Orbital abscesses are rarely encountered in children younger than 1 year. The literature is limited to isolated case reports and a few case series. Most such cases are reported in infants born at term, with the earliest reported gestational birth age at 34 weeks. Children are more prone to orbital cellulitis compared with adults due to their underdeveloped sinuses and immature immune systems, and the origin is most commonly an ethmoid sinus infection. Orbital cellulitis secondary to dacryocystitis is even less common, with only a few isolated cases reported in infants and children. Herein, the authors present a case of a large extraconal and intraconal orbital abscess secondary to nasolacrimal duct obstruction and dacryocystitis in an extremely preterm infant. We discuss the diagnosis and multidisciplinary management of this challenging case.

Acute dacryocystitis: changing practice pattern over the last three decades at a tertiary care setup.

AIM: To compare the demographics, clinical features, and changes in the management pattern of acute dacryocystitis at a tertiary care eye institute. METHODS: A retrospective review was performed of electronic medical records of all patients diagnosed with acute dacryocystitis from January 2013 to January 2023. Data retrieved include demographics, history, presenting symptoms, duration of symptoms, surgical interventions, associated systemic conditions, management, complications, and outcomes. A successful anatomical outcome was defined as patency on lacrimal irrigation, and a successful functional outcome was defined as the resolution of infection and epiphora. The data parameters obtained were compared with the historical published data of the earlier two decades from the same Institute. RESULTS: A total of 363 eyes of 349 patients were enrolled in this retrospective study. The median age was 45 years (range: 19-94 years). There were 216 (62%) females and 133 (38%) males. Surgery was performed in 320 (88%) patients. Needle aspiration or incision and drainage were performed in 102(32%) patients with lacrimal abscesses. Of the 320 patients, an endoscopic DCR was performed in 138 (43%) patients and an external DCR in 182 (57%). Of the 320 patients who underwent DCR surgery, 308 (96%) demonstrated anatomical and functional success at 1-year follow-up. CONCLUSION: There is a changing trend towards endoscopic DCR being incorporated as the primary procedure for managing acute dacryocystitis with the advantages of quicker resolution and reduced morbidity. There is a trend for choosing needle aspiration over the traditional incision and drainage in the initial management of lacrimal abscess.

[Concrements of the lacrimal apparatus]. / Konkremente des Tränenapparates.

Concrements of the lacrimal apparatus, known as dacryoliths, can occur at different localizations and can cause a variety of symptoms. A common clinical sign is chronic inflammation, possibly exhibiting acute exacerbation. Based on a literature review and descriptive clinical cases with histopathological correlations, this contribution summarises the most important information concerning epidemiology, aetiopathogenesis, composition, histology, and therapy of lacrimal concrements. Furthermore, factors known to affect lacrimal lithogenesis are addressed. Concrements of the lacrimal gland cause a swelling at the lateral canthus. With only mild pain, this manifests as circumscribed conjunctival hyperaemia. Histologically, the gland tissue is characterised by acute-erosive to chronic inflammation. The concrements consist of amorphic material. Inflammatory infiltration is dominated by neutrophil granulocytes. Canalicular concrements are highly correlated with chronic canaliculitis. Besides epiphora, patients present with purulent discharge at the affected canaliculus. Actinomyces are frequently found inside these deposits and form drusen-like formations. The surrounding tissue reacts with plasma-cellular and granulocytic inflammation. Dacryoliths (concrements of the lacrimal sac) are associated with dacryocystitis, whereby acute and chronic types are common. Stones can be found in up to 18% of patients undergoing dacryocystorhinostomy or dacryoendoscopy. Preoperative diagnostic testing is challenging, as many lacrimal sac stones cannot be reliably visualised by diagnostic procedures. Recurring episodes of epiphora, mucopurulent discharge, and dacryocystitis are common indicators of dacryoliths. Lacrimal syringing is often possible and shows that total blockage is not present. Histology of the lacrimal mucosa reveals lymphocytic infiltration and submucosal fibrosis. The immediate vicinity of the dacryoliths shows acute inflammation. Therapy consists of stone extraction and improving lacrimal drainage, as the latter is recognised as the main risk factor for dacryolith formation.

Association of primary chronic dacryocystitis and meibomian gland dysfunction.

PURPOSE: To evaluate the association between meibomian gland dysfunction (MGD) and primary chronic dacryocystitis (PCD) and the effect of dacryocystorhinostomy (DCR) on tear film stability and MGD. METHODS: This prospective, interventional, non-randomized study involved 50 unilateral acquired PCD cases and 50 age-matched healthy controls. Patients with lid abnormalities, ocular trauma, previous ocular surgery, contact lens wearers, and chronic topical or systemic drug users were excluded from the study. After a detailed history, the ocular surface disease index (OSDI) was calculated. The ocular assessment included visual acuity, tear meniscus height (TMH), tear break-up time (TBUT), tear well diameter (TWD), Schirmer 1 test, meibomian gland (MG) expressibility, and meibography. PCD eyes underwent external DCR, and the tests were repeated after 8 weeks. RESULTS: The mean age of PCD cases was 42.58 ± 12.74 years, the male: female ratio was 7:19, and the mean duration of epiphora was 2.2 ± 1 years. The MG expressibility grade of ≥2 was seen in 98% (49/50) PCD eyes, which was strongly associated with PCD as compared to controls (OR = 563, P = 0.00, 95% CI = 60.71-5229.70). MG loss ≥50% was seen in 62% (31/50) of PCD eyes and none of the control eyes. Following DCR, MG loss remained unchanged, and a significant decrease occurred in OSDI scores, TWD and Schirmer 1 values, and MG expressibility grade (Z = -6.85). The mean TMH decreased from 767.60 ± 331.60 µm to 384 ± 204.29 µm ( P = 0.004) post DCR. CONCLUSIONS: PCD is strongly associated with MGD. DCR reverses the functional MG changes with improvement in the tear film stability but no effect on MG loss.

A case report of chronic dacryocystitis caused by nasal stones.

BACKGROUND: This paper reports a case of chronic dacryocystitis due to nasal stones. CASE PRESENTATION: An 84-year-old male patient was admitted to the hospital with chronic dacryocystitis of the right eye due to tearing and purulent discharge from the right eye for more than 1 month. Antibiotic treatments such as gatifloxacin eye drops were given at other hospitals but did not relieve the symptoms. A computed tomography(CT) scan of the lacrimal duct in our department showed a high-density shadow in the right lacrimal sac area, hypertrophy of the right inferior turbinate, and many nasal calculi in the nasal cavity. The patient was transferred to our otolaryngology department for further treatment, and nasal stones were removed under nasal endoscopy. Three days after surgery, the symptoms affecting the patient's right eye gradually resolved. One month after surgery, the patient underwent a follow-up examination in the ophthalmology clinic; there was no lacrimal purulent discharge from the right eye, and the lacrimal duct could be flushed smoothly. CONCLUSION: Chronic dacryocystitis is often caused by primary nasolacrimal duct obstruction. Cases of chronic dacryocystitis caused by secondary nasolacrimal duct obstruction due to nasal stones are rare in the clinic. This case can serve as a reference for the clinical diagnosis and treatment of chronic dacryocystitis.

Chronic bilateral dacryoadenitis caused by SARS-CoV-2 infection: a case report.

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.

Dacryoadenitis post COVID-19 infection and immunization.

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.

Isolated tubercular dacryoadenitis in a non-immunocompromised healthy adult.

A middle-aged woman presented with a slowly enlarging mass in the right superotemporal orbit and ptosis. The patient had an elevated erythrocyte sedimentation rate, and incisional biopsy showed granulomatous inflammation with Langhan's giant cells suggestive of a tubercular etiology. Tuberculin skin test was negative. The patient responded well to anti-tuberculous therapy. Tubercular dacryoadenitis is a rare presentation of a lacrimal gland mass.

Herpes Zoster Ophthalmicus With Dacryoadenitis Complicated by Recurrent Orbital Inflammation.

Herpes zoster ophthalmicus represents a zoster infection in the first division of the trigeminal nerve and has potentially serious complications involving the ocular and orbital structures. Herpes zoster ophthalmicus occurs in approximately 10% to 20% of individuals with herpes zoster and can lead to significant morbidity, particularly in patients with multiple comorbidities. The authors present a case of herpes zoster ophthalmicus along with dacryoadenitis in a patient with uncontrolled diabetes and rheumatoid arthritis who was misdiagnosed, had delayed treatment, and experienced recurrence with orbital symptoms during follow up. Prompt recognition, initiation of therapy and careful monitoring and follow up are important for treating herpes zoster ophthalmicus and preventing recurrence and long-term sequelae.

Long-term Follow-up Results of Primary Canaliculitis Patients.

Objectives: To evaluate the demographic characteristics, clinical presentation, microbiologic profile, and treatment results of patients with primary canaliculitis. Materials and Methods: Patients diagnosed and treated for primary canaliculitis between May 2014 and May 2021 were analyzed retrospectively. Results: There were 26 patients with primary canaliculitis, including 17 females (65.4%) and 9 males (34.6%) with a mean age of 50.6±16.4 years (range: 9-80 years). Canaliculitis affected the right eye in 11 patients, the left eye in 13 patients, and bilateral involvement was seen in 2 patients. Inferior canaliculus involvement was more frequent (73%). The most common complaint was epiphora (46.1%). Five patients (19.2%) were wrongly diagnosed as chronic conjunctivitis. The time interval between the beginning of symptoms and canaliculitis diagnosis was 18.2±14.3 months (range: 1-60 months). Canaliculotomy and curettage of canalicular content with dacryolith removal were performed in 23 patients. After surgery, antibiotic irrigation of the canaliculus was added to the treatment regimen in 12 of these 23 patients. Intracanalicular antibiotic therapy was administered to the remaining 3 patients. The most cultured organism was Actinomyces (6 patients). Gemella (1 patient), Porphyromonas (1 patient), Candida parapsilosis (1 patient), Citrobacter koseri (1 patient) were also grown in culture. The follow-up time of patients was 26.2±23.7 months (range: 6-83 months). All symptoms and findings resolved in all patients in one month. In two patients, recurrence occurred at 4 and 16 months after surgical treatment. With appropriate treatment, no further recurrence was seen in either patient over 24-month follow-up. One patient presented with iatrogenic canaliculus blockage during follow-up. Conclusion: Primary canaliculitis is often overlooked and can be misdiagnosed. The most common symptom was epiphora. All patients with epiphora and chronic conjunctivitis should be examined carefully for canaliculitis.

Lacrimal Drainage Infections with Sphingomonas paucimobilis: Clinical Presentations, Complications and Outcomes.

PURPOSE: To report the clinical presentations, complications, and outcomes of Sphingomonas paucimobilis-specific lacrimal drainage infections. METHODS: A retrospective chart review of all patients diagnosed with Sphingomonas paucimobilis lacrimal infections and managed at a tertiary care Dacryology Service from November 2015 to May 2022 over a 6.5-year period were recruited and analyzed. Data collected include demographic details, clinical presentation, microbiological diagnosis, antibiotic susceptibility profiles, management, complications, and outcomes. The microbiological techniques employed were aerobic and anaerobic cultures, phenotypic identification with VITEK 2R system, polymerase chain reaction, antibiotic sensitivity profile and minimal inhibitory concentration. RESULTS: Twelve Sphingomonas-specific lacrimal drainage infections of 11 patients were identified. Of these five were canaliculitis and seven were acute dacryocystitis. All the seven acute dacryocystitis presented in an advanced stage; five were with lacrimal abscess and two with orbital cellulitis. The antibiotic susceptibility profile of canaliculitis and acute dacryocystitis were comparable and the organism was sensitive to several classes of antibiotics. The outcomes of punctal dilatation and nonincisional curettage were effective for canaliculitis. Patients with acute dacryocystitis had advanced clinical stage at presentation but responded well to intensive systemic management and had excellent anatomical and functional outcomes with dacryocystorhinostomy. CONCLUSION: Sphingomonas-specific lacrimal sac infections can have aggressive clinical presentations and need early and intensive therapy. The outcomes are excellent with multimodal management.

Concrements of the Lacrimal Apparatus. / Konkremente des Tränenapparates.

Concrements of the lacrimal apparatus, known as dacryoliths, can occur at different localizations and can cause a variety of symptoms. A common clinical sign is chronic inflammation, possibly exhibiting acute exacerbation. Based on a literature review and descriptive clinical cases with histopathological correlations, this contribution summarises the most important information concerning epidemiology, aetiopathogenesis, composition, histology, and therapy of lacrimal concrements. Furthermore, factors known to affect lacrimal lithogenesis are addressed. Concrements of the lacrimal gland cause a swelling at the lateral canthus. With only mild pain, this manifests as circumscribed conjunctival hyperaemia. Histologically, the gland tissue is characterised by acute-erosive to chronic inflammation. The concrements consist of amorphic material. Inflammatory infiltration is dominated by neutrophil granulocytes. Canalicular concrements are highly correlated with chronic canaliculitis. Besides epiphora, patients present with purulent discharge at the affected canaliculus. Actinomyces are frequently found inside these deposits and form drusen-like formations. The surrounding tissue reacts with plasma-cellular and granulocytic inflammation. Dacryoliths (concrements of the lacrimal sac) are associated with dacryocystitis, whereby acute and chronic types are common. Stones can be found in up to 18% of patients undergoing dacryocystorhinostomy or dacryoendoscopy. Preoperative diagnostic testing is challenging, as many lacrimal sac stones cannot be reliably visualised by diagnostic procedures. Recurring episodes of epiphora, mucopurulent discharge, and dacryocystitis are common indicators of dacryoliths. Lacrimal syringing is often possible and shows that total blockage is not present. Histology of the lacrimal mucosa reveals lymphocytic infiltration and submucosal fibrosis. The immediate vicinity of the dacryoliths shows acute inflammation. Therapy consists of stone extraction and improving lacrimal drainage, as the latter is recognised as the main risk factor for dacryolith formation.

Systemic eosinophilic disease presenting as dacryoadenitis.

Eosinophilic disease with orbital involvement is rare. We present two patients with dacryoadenitis associated with local and systemic eosinophilia. A 32-year-old man presented with episodic dacryoadenitis, lower respiratory inflammation and peripheral eosinophilia. Lung and lacrimal gland biopsies demonstrated eosinophilic infiltrate without granuloma, necrosis, or vasculitis. He improved with oral corticosteroids and Mepolizumab, an IL-5 inhibitor. The second case involved a 33-year-old man who similarly presented with episodic dacryoadenitis, pulmonary inflammation and pain/swelling in the hands and feet. Lacrimal gland biopsy demonstrated a predominantly eosinophilic infiltrate without granuloma or vasculitis. Symptoms improved with oral corticosteroids. Although neither patient was provided a definitive diagnosis, both were determined to have an eosinophilic condition on the spectrum of eosinophilic asthma or eosinophilic granulomatosis with polyangiitis.

An Unusual Case of Extranodal Natural Killer/T-Cell Lymphoma, Nasal Type Masquerading as Dacryocystitis and Sinusitis.

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.