Bilateral Dacryoadenitis Research Articles

Dacryoadenitis as a Rare Initial Presentation in a Patient with Suspected Crest Syndrome.

Broad spectrum of clinical features: Recognize that although CREST syndrome is primarily characterized by its hallmark features - calcinosis, Raynaud's phenomenon, esophageal dysmotility, sclerodactyly, and telangiectasia - it can also present with additional clinical features that may indicate the onset or presence of the syndrome.Diagnostic challenges and differentiation: Diagnostic challenges and differentiation: Bilateral dacryoadenitis can be challenging to diagnose due to its overlapping symptoms with other orbital or systemic conditions. The case report may highlight the importance of differentiating it from conditions like orbital cellulitis, sarcoidosis, or lymphoproliferative disorders. Advanced imaging techniques (like magnetic resonance imaging or computed tomography scans) and careful assessment of clinical history (including systemic symptoms like fever or autoimmune conditions) are crucial for accurate diagnosis.Awareness of atypical presentations and follow-up: The case report underscores the need for tailored management strategies for bilateral dacryoadenitis, from conservative treatments in mild cases to more aggressive therapies in severe ones, while also highlighting the importance of monitoring for complications and being vigilant about atypical presentations, such as those seen in patients with CREST syndrome.

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

BackgroundDacryoadenitis 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.ReportA 26-year-old Arab woman experienced chronic inflammatory dacryoadenitis after a mild SARS-CoV-2 infection, which was successfully treated with oral prednisone.ConclusionsDacryoadenitis 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.

Chronic bilateral dacryoadenitis with concurrent tattoo inflammation following COVID-19 vaccination and infection

Chronic bilateral dacryoadenitis with concurrent tattoo inflammation following COVID-19 vaccination and infection

Bilateral dacryoadenitis associated with IgG4-related disease presenting as orbital pseudo-inflammatory tumor.

Bilateral dacryoadenitis associated with IgG4-related disease presenting as orbital pseudo-inflammatory tumor.

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.

COVID-19–Related Chronic Bilateral Dacryoadenitis

Pathological features of ophthalmic aftereffects of COVID-19 are important for new insight in treating patients. To examine the expression of SARS-CoV-2 nucleocapsid protein and angiotensin-converting enzyme 2 (ACE2) in lacrimal gland tissues of a patient with COVID-19 and a patient without COVID-19. In this retrospective case-control study, the case of a 35-year-old woman with positive test results for SARS-CoV-2 who had had lacrimal gland enlargements for 6 months was analyzed. A 43-year-old woman without COVID-19 who had idiopathic chronic bilateral dacryoadenitis served as a negative control. Histopathology and immunohistochemistry with anti-SARS-CoV-2 nucleocapsid protein and ACE2 in the lacrimal glands. Both patients were Japanese women aged 35 years (case) and 43 years (control). Histopathologic findings in the patient with COVID-19 demonstrated marked inflammatory cell infiltration, lymphoid follicles, and germinal center formation in the lacrimal gland. The inflammation was mainly made up of lymphocytes and plasma cells with several polymorphonuclear leukocytes, where the lacrimal glands were atrophic. Of note, a number of lacrimal gland ducts markedly contained eosinophilic materials in the lumens, which indicated glandular damage. Immunoreactivity for SARS-CoV-2 nucleocapsid protein was noted in the inflammatory cells around the lacrimal gland ductal epithelia. In addition, strong ACE2 expression was noted in the lacrimal gland. In the patient without COVID-19, marked inflammation was noted in the lacrimal gland; however, there were no eosinophilic material deposits in the ductal lumens. SARS-CoV-2 nucleocapsid protein immunoreactivity was not observed, whereas ACE2 was expressed in the lacrimal glands. In this case-control study, expression of ACE2 indicated that the lacrimal gland could be a target organ for SARS-CoV-2 to adhere to. Chronic bilateral dacryoadenitis in the patient with COVID-19 showed SARS-CoV-2-positive inflammatory cells with glandular damage, which might be a COVID-19-associated ophthalmic aftereffect.

P27 Lymphoma-mimicker of IgG4-related dacryoadenitis and sialadenitis

Case report - IntroductionMajor salivary gland (sialadenitis) and lacrimal gland (dacryoadenitis) involvement can be a common feature of IgG4-related disease. There can be involvement of lacrimal and parotid gland which was previously called as Mikulicz disease and/or submandibular gland enlargement which was previously called Küttner tumour. These were previously mistakenly considered to be subcategories of Sjogren’s syndrome, but are now classified as IgG4-related disease. Here we discuss a case report of a patient who presented with bilateral dacryoadenitis and unilateral submandibular gland enlargement which initially was thought to be IgG4-related disease but turned out to be low-grade lymphoma.Case report - Case descriptionA 15-year-old boy presented with 6-month history of bilateral eyelid swelling. The swelling was more on the lateral side of the eyelids and was painless. It had come on suddenly over a couple of days. No history of weight loss, dry eyes, dry mouth, joint issues or skin rashes or any other symptoms. Physical examination revealed bilateral ptosis, no visual impairment and systemic examination revealed an enlarged right submandibular gland. He had initially been to a local hospital where he had investigations which included autoimmune screen including ANA, ENA, ANCA, dsDNA, serum ace and complement levels which were all negative. C1 esterase inhibitor was normal. Routine bloods including complete blood count, urea and electrolytes, thyroid stimulating hormone and erythrocyte sedimentation rate were within normal limits. Urine albumin to creatinine ratio was not raised. Hepatitis serology including Hepatitis B & C and HIV was negative. Ultrasound abdomen was unremarkable. CT scan of orbits showed bilateral enlarged lacrimal glands with patchy post contrast enhancement and the glands extending up to insertion of lateral rectus muscle. CT chest some enlarged axillary lymph nodes and nodes in lung query infective etiology. CT abdomen and pelvis was unremarkable. Ultrasound neck showed right submandibular node enlargement with colour doppler showing increased vascularity. Fine needle aspirate of the submandibular gland showed reactive lymphoid hyperplasia. He was given two short courses of steroids and each time the swelling rapidly responded to the steroids but recurred on cessation of the steroids. IgG subset analyses revealed elevated IgG4 levels of 1152mg/dl. The differential here was IgG4-related disease but as there was no clear tissue diagnosis a core biopsy of the right submandibular gland was done. This revealed tissue suspicious of low grade (extranodal marginal zone and mucosa-associated lymphoid tissue [MALT]) lymphoma and excision biopsy was performed for definitive diagnosis.Case report - DiscussionIgG4-related disease is an immune mediated fibroinflammatory condition which can affect a variety of organs and can present as tumour-like enlargement and/or organ dysfunction. The pathological findings in IgG4-related disease are lymphoplasmocytic infiltrates of IgG4-positive cells along with increased levels of serum IgG4 levels. Salivary and lacrimal glands can be commonly affected and present as enlargement, which is usually painless and bilateral. The combination of lacrimal gland enlargement with both parotid and submandibular gland enlargement is called IgG4-related Mikulicz disease.Apart from salivary glands, another commonly affected organ is the pancreas which can present as a pancreatic mass and painless jaundice, sclerosing cholangitis, retroperitoneal fibrosis, aortitis and periaortitis. Less commonly it can affect thyroid, kidney and lungs.Early recognition, diagnoses and treatment is important due to the fibroinflammatory nature of the disease. Malignancy is always in the differential and should be excluded. Steroids are the mainstay of treatment. If patients experience flare, rituximab can be added.Diagnosis should be confirmed with biopsy but histopathological findings are never alone diagnostic of IgG4-related disease and should be interpreted with clinical, serological and radiological findings.Case report - Key learning pointsAlthough this patient had typical presentation of IgG4-related disease with painless enlargement of salivary and lacrimal glands and elevated IgG4 serum levels, biopsy was imperative to get to the diagnoses of low-grade lymphoma and fine needle aspirate was not adequate. As mentioned above, biopsy in IgG4-related disease will confirm the diagnosis provided there are other supporting features (radiological and serological). However, it is imperative for excluding other important diseases like lymphoproliferative disorders.

Poches palpébrales : pièges en blépharoplastie

Requests for cosmetic eyelid surgery are frequent. The surgeon must carry out a complete clinical examination in order to rule out any orbital pathologies responsible for eyelid pockets. Study of ten patients who have consulted in the oculopalpebral surgery department of La Fondation Ophtalmologique de Rothschild for cosmetic surgery of the eyelid pockets and in whom an underlying pathology was diagnosed. The clinical examination allows to look for signs of alarms, such as medical history, unilateral involvement, pain and the presence of exophthalmos. These should lead to perform orbital imaging and surgical biopsy if an orbital mass has been identified for histological analysis which will make the diagnosis. We identified four cases of orbital lymphoma, one foreign body granuloma, chronic bilateral dacryoadenitis, sarcoidosis involvement, AAPOX syndrom, cavernous hemangioma, and simple fatty hernia. These cases shows the importance of not ignoring an underlying orbital pathology before proposing cosmetic surgery for eyelid pockets.

BILATERAL ORBITAL AND FLOOR OF THE MOUTH SWELLING: A CASE REPORT OF IgG4-RELATED DISEASE

IgG4-related disease (IgG4-RD) is characterized by IgG4-positive plasma cell infiltration and fibrosis mostly in the pancreas, bile ducts, salivary glands, and orbits. A 34-year-old woman with a history of chronic rhinitis presented with bilateral asymptomatic orbital swelling of 7 years’ duration. Extra-oral examination revealed bilateral palpebral swelling and dacryoadenitis, whereas bilateral normal-colored swelling of the floor of the mouth was detected intra-orally. Serologic tests for IgG4 showed an elevated concentration of 698 mg/dL, and an incisional oral biopsy was performed. Histopathologic analysis revealed minor salivary glands with marked infiltration of lymphocytes, histiocytes, and plasma cells, with occasional lymphoid follicles. By immunohistochemistry, the histiocytes were positive for CD163, and IgG4 was positive in more than 50 plasma cells per high-power field. The final diagnosis was IgG4-RD. The patient was referred for treatment in a rheumatology service. Systemic involvement was not detected, and there was an improvement of orbital and oral lesions after oral corticosteroid therapy.

Predictive Value of Serum Angiotensin-Converting Enzyme Titers in Sarcoid-Like Dacryoadenitis.

To compare serum angiotensin-converting enzyme (sACE) titers in patients with newly diagnosed sarcoid-like dacryoadenitis and nongranulomatous dacryoadenitis. Retrospective comparative case series. Clinical details and presenting sACE titers were reviewed for patients with newly diagnosed sarcoid-like dacryoadenitis (SLD) and nongranulomatous dacryoadenitis (NGD). All sACE titers were expressed as a percentage of the upper end of normal range ("normalized"). Using Student t test, sACE titers for patients with unilateral SLD were compared with unilateral NGD and the sensitivity and specificity of sACE as a predictor for SLD calculated; this was repeated for patients with bilateral dacryoadenitis. The proportion of patients with raised sACE were compared in each group (Fisher exact test), and the mean sACE titers compared for these selected patients. For unilateral SLD the mean sACE was 97%, compared with 57% in NGD (p=0.0010), and the sensitivity of sACE titer was 42%, specificity 92%, positive predictive value 75%, and negative predictive value 73%. Area under curve from receiver operating characteristic analysis for unilateral dacryoadenitis was 0.78 (p=0.0040). In contrast, there was no significant difference for bilateral disease (SLD 148%, NGD 82%; p=0.33), and somewhat lower positive predictive value (54%), negative predictive value (71%) and area under curve (0.71; p=0.070). Although not significantly so with bilateral disease, the proportion of patients with raised sACE was consistently greater in SLD as compared to NGD-being highest with bilateral SLD (7/12 patients; 58%) and lowest in unilateral NGD (2/26; 8%). Seventeen of the 27 patients with SLD were later found to have systemic involvement: where systemic disease was present, the mean sACE levels were significantly different in unilateral (102%) and bilateral dacryoadenitis (209%; p=0.029) disease, whereas there was no difference in the absence of systemic involvement (sACE with unilateral dacryoadenitis = 86%, bilateral = 75%; p=0.72). Normalized serum titers of ACE are minimally predictive for SLD, whether unilateral or bilateral, with some false negatives and false positives in all groups. However, with unilateral dacryoadenitis, an sACE of >125% maximum range appears absolutely predictive for SLD, and >150% maximum range for bilateral disease.

IgG4-related orbital disease

Abstract Case report The case is presented of a 64-year-old woman with bilateral palpebral swelling and dacryoadenitis, exophthalmos, and a history of chronic rhinitis and asthma. An increase in serum IgG4 was observed, and an incisional biopsy of lacrimal glands was performed, which showed fibrosis and a lymphoplasmacytic infiltrate with IgG4 producing cells. Discussion Orbital involvement in IgG4-related disease is frequent. Bilateral dacryoadenitis is the most common manifestation. Histopathology is essential for the diagnosis and to exclude malignancy.

Enfermedad orbitaria relacionada con Ig-G4

Case reportThe case is presented of a 64-year-old woman with bilateral palpebral swelling and dacryoadenitis, exophthalmos, and a history of chronic rhinitis and asthma. An increase in serum IgG4 was observed, and an incisional biopsy of lacrimal glands was performed, which showed fibrosis and a lymphoplasmacytic infiltrate with IgG4 producing cells. DiscussionOrbital involvement in IgG4-related disease is frequent. Bilateral dacryoadenitis is the most common manifestation. Histopathology is essential for the diagnosis and to exclude malignancy.

Orbitopathies inflammatoires. Que doit savoir l’interniste ?

Inflammatory orbitopathies relate to an inflammatory state originating within the orbit and its adnexes, except the inner ocular globe. Orbital inflammation (OI) may be either localized manifestation of a proven or like autoimmune disease, or local response from immune system against infectious, structural or tumoral antigens. We review the clinical manifestations of OI, which provide helpful clues to the diagnosis and describe the inflammatory, infectious and neoplastic conditions classically associated with OI. Autoimmune diseases are probably the most common causes of OI associated with a bilateral dacryoadenitis (e.g., sarcoidosis, granulomatosis with polyangiitis, IgG4-related disease). We focused on a major part of the IgG4-RD spectrum, the IgG4-related orbital disease which has been recently described and the idiopathic orbital inflammation syndrome that one should consider in patients 40 years of age or older with non specific inflammation OI on biopsy but without underlying local or systemic disease. An algorithm for the diagnostic approach of OI was proposed. If systemic explorations fail to diagnose an underlying disease, histopathologic control is required for distinguishing non-specific OI from other differential diagnosis, especially lymphoma. In the cases of pure myositic locations and posteriorly located tumours where biopsy could damage to the optic nerve, analysis of orbital lesions in T2W IRM sequence may be helpful to distinguish idiopathic OI (IOI) from lymphoma. When the diagnostic work-up fails, a corticosteroid trial could be used, but its beneficial effect has to be cautiously interpretated before definitively diagnosing IOI. Finally, treatments used in main infllammatory orbitopathies were also reviewed.

Bilateral Dacryoadenitis

Tuberculous dacryoadenitis occurs rarely. It should be suspected in children presenting with unremitting inflammation of the lacrimal gland despite standard antibiotic and/or anti-inflammatory treatment, and a thorough systemic evaluation should be undertaken to identify a primary infectious site. We present an adolescent girl in whom bilateral dacryoadenitis was the first sign of pulmonary tuberculosis.

Bilateral orbital myositis and dacryoadenitis after sclerosing mediastinitis

A 49-year-old male with a history of sclerosing mediastinitis presented with complaints of bilateral (left greater than right) eye pain and redness for 3 weeks. Clinically, the patient demonstrated signs of bilateral dacryoadenitis and myositis. CT confirmed enlargement of both lacrimal glands and lateral rectus muscles. Due to the relationship between sclerosing mediastinitis and orbital inflammatory pseudotumor (OIP), the patient was treated with oral prednisone with a brisk response. OIP is common in ophthalmic practice and treating physicians should be aware of the associations with systemic disease processes.

Granulomatous Dacryoadenitis in Regional Enteritis (Crohn Disease)

To evaluate the clinical and immunopathologic features of 2 patients with bilateral dacryoadenitis associated with regional enteritis. Retrospective, clinicopathologic study. Clinical records, photographs, and imaging studies were reviewed and microscopic sections of lacrimal gland biopsy samples were critically re-evaluated. The microscopic slides were stained with hematoxylin and eosin, special stains for organisms, and a range of immunohistochemical biomarkers, including CD3, CD4, CD5, CD8, CD20, CD68, CD138, CD1a, and immunoglobulins Ig G, IgG4, and IgA. Both patients were young women with a well-established diagnosis of regional enteritis. Histopathologic examination of biopsy samples disclosed moderate intraparenchymal fibrosis and lymphoplasmacytic infiltrates without lymphoid follicles. Small to medium intraparenchymal, noncaseating granulomas lacking multinucleated giant cells and, in 1 patient, CD68-positive and CD1a-negative palisading granulomas in widened interlobular fibrous septa were detected. Vasculitis and IgG4 plasma cells were not observed. Additional immunohistochemical studies revealed that CD8T lymphocytes (suppressor or cytotoxic subset) predominated over CD4-positive T lymphocytes (helper cells) surrounding the necrobiotic foci and were intermixed with the CD68-positive histiocytes in the absence of CD20 B lymphocytes. Special stains for organisms demonstrated negative results. Dacryoadenitis is the rarest form of ocular adnexal involvement in regional enteritis, which affects the orbit far more frequently than ulcerative colitis. It is a granulomatous process with the possibility of palisading necrobiotic foci. In contrast, ulcerative colitis causes an interstitial lymphocytic and nongranulomatous myositis. Sarcoidosis, Wegener granulomatosis, and pseudorheumatoid nodules must be ruled out. Treatment options entail a wide variety of agents with selection based on empirical considerations and tailored to the patient's symptoms.

Esordio di mononucleosi infettiva con edema periorbitale e palpebrale

Summary Introduction: Almost all organs can be involved in the infection by Epstein—Barr virus and consequently, the beginning symptoms of infectious mononucleosis may be different. Materials and methods: We report a case of infection by Epstein—Barr virus in a 17-year-old girl whose primary manifestation was an initially monolateral and subsequently bilateral dacryoadenitis.

Systemic immunoglobulin G4 (IgG4) disease and idiopathic orbital inflammation; removing ‘idiopathic’ from the nomenclature?

The discovery of systemic disease related to raised tissue and serum immunoglobulin G4 (IgG4) is changing diagnostic and therapeutic practice in many medical specialties. Orbital inflammation remains a diverse and heterogeneous group of disorders that can pose a diagnostic and therapeutic challenge, but with improved understanding and corresponding diagnostic advances the previously expansive group of idiopathies is reducing. The recent discovery that IgG4 has a causative role in a subtype of, what is currently termed, idiopathic orbital inflammation is encouraging. The term 'idiopathic' can now be removed from the nomenclature for another subtype of orbital inflammation. IgG4 disease should be especially considered in patients with a bilateral dacryoadenitis and systemic features (eg, lung and gastrointestinal involvement). However, reports are emerging suggesting that IgG4 may be responsible for more diverse disease subtypes. The relationship between IgG4-related disease and lymphoma remains unknown but vigilance is required.

Bilateral dacryoadenitis

Acute dacryoadenitis is an uncommon condition that involves inflammation of the lacrimal gland. In rare instances, dacryoadenitis may be bilateral. A delay in proper treatment of an otherwise simple case of dacryoadenitis may lead to significant soft tissue morbidity such as cellulitis, lacrimal gland abscess, or orbital abscess. We report the case of a 24-year-old male who presented to the emergency department with acute bilateral dacryoadenitis. The patient's symptoms did not respond to oral antibiotics and he subsequently required admission for intravenous antibiotics. During his hospitalization the patient had diagnostic testing to try to determine the etiology for his symptoms. The unique aspects of managing a case of bilateral dacryoadenitis as well as treatment recommendations are discussed in this case report.

Dacrioadenite bilateral causada pelo vírus Epstein-Barr: relato de caso

The authors present a rare case of acute bilateral dacryoadenitis in association with infectious mononucleosis. A 27 year-old woman had acute bilateral lacrimal gland enlargement with inflammatory signs. The CT scan findings and laboratorial investigations confirmed the diagnosis of infectious mononucleosis. The systemic treatment with intravenous steroids was initiated leading to complete remission of the signs and symptoms. Dacryoadenitis is an uncommon manifestation of infectious mononucleosis and may minimize other signs of the disease. Mononucleosis has to be considered in acute dacryoadenitis cases. Systemic steroids contribute to the fast regression of inflammatory condition.