Chronic Dacryoadenitis Research Articles

A Curious Case of IgG4-Related Disease Masquerading As an Occult Abdominal Malignancy

Introduction IgG4-related disease is an autoimmune, fibroinflammatory disorder involving multiple organ systems in the body. Diagnosis is often suggested by strong IgG4-positive stain on tissue biopsy. Patients respond well to steroids; and in cases of relapse, steroid-sparing agents like rituximab, azathioprine or cyclosporine can be used. Case Presentation Here we present the case of a 77 year-old African-American female with a past medical history significant for cerebrovascular accident, chronic hepatitis C infection status post treatment with antivirals, left chronic dacryoadenitis and chronic iron-deficiency anemia. She presented to the hematology office for a follow-up visit for chronic anemia and complained of new onset right upper quadrant pain and unexplained weight loss. Subsequent abdominal and pelvic imaging showed hypoechoic lesion in pancreatic head along with multiple epigastric, omental and gastro-hepatic nodules/lymph nodes suspicious for malignancy. This was followed by esophagogastroduodenoscopy (EGD) with endoscopic ultrasound (EUS) which revealed gastric body/antral erosions along with multiple enlarged abdominal lymph nodes and heterogeneous, hypoechoic irregular mass in the pancreatic head/neck, all of which were biopsied. While awaiting biopsy results, due to high suspicion for malignancy, PET-FDG was performed which showed hypermetabolic lesions corresponding to the EUS findings and a FDG-avid right paravertebral mid thoracic soft tissue mass favored to represent a neurogenic tumor, unrelated to abdominal disease. Laboratory work-up was significant for elevated IgG levels with particularly high titres of IgG subclass 4. Biopsy results from EUS showed no evidence of malignant cells, no clonal proliferation on flow cytometry in either the lymph nodes or pancreatic tissue. Cytology of pancreatic tissue biopsy revealed lymphoid infiltration, extensive edema and fibrosis with no malignant cells. Immunohistochemistry studies revealed strong positive stain for IgG4 suggestive of IgG4-related disease. Of note, the patient had been following up with ophthalmology for a few years for chronic inflammatory dacryoadenitis of the left eye, which can likely be attributed to IgG4-related disease. After shared-decision making with the patient regarding treatment options, treatment was initiated with a plan of rituximab for 4 cycles along with corticosteroid. As of this abstract submission, the patient received one dose of rituximab, pending assessment of response to treatment. Discussion Heterogeneous pancreatic masses on abdominal imaging should be considered malignant until otherwise proven. In cases where these masses are associated with diffuse abdominal and pelvic lymphadenopathy, definitive diagnosis is made through obtaining biopsies from the easily accessible sites. A benign, auto-immune, fibroinflammatory condition which mimics this presentation is IgG4-related disease, whose treatment is far less complicated than a metastatic carcinoma. It often involves lacrimal glands. A high index of suspicion backed by the biopsy results showing strong stain affinity to IgG4 and elevated blood IgG4 levels are required. Treatment involves initiation of steroids followed by steroid-sparing agents like azathioprine, cyclosporine or rituximab in cases of relapse after steroid induction. However, as in our case, combination therapy of simultaneous use of steroids with rituximab can be used after shared decision making with patients.

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.

Clinical, Radiological and Histopathological Features of Patients With Lacrimal Gland Enlargement.

The purpose of this study was to describe the radiologic and histopathologic features of lacrimal gland in patients presenting with lacrimal gland enlargement. We retrospectively retrieved the data of patients with lacrimal gland enlargement in Farabi Eye Hospital between 2012 and 2017. These data included demographics, the patients' facial photographs, orbital CT-scans, and histopathological findings of lacrimal gland biopsies. Forty-seven patients (15 men and 32 women) were enrolled in this study with a median age of 37.9 years (range, 15-79 years). Histopathologic diagnoses were chronic dacryoadenitis in 26 cases (55.32%), IgG4-related disease in 6 patients (12.77%), two cases of acute dacryoadenitis, two cases of non-necrotizing granulomatous inflammation, two cases of Non-Hodgkin's B-cell lymphoma, two cases of adenoid cystic carcinoma and two cases of mixed tumor (4.26% each), as well as one case of conjunctival epithelial cyst, and one case of benign lymphoid tissue and fibrofatty tissue (2.13%). In two samples (4.26%), biopsy revealed normal lacrimal glands. Interestingly, in two cases with relapsing lacrimal gland enlargement, different histopathologic diagnoses were found in biopsies taken from each lacrimal gland at different times. The average size of enlarged lacrimal glands was 19.67 mm × 7.06 mm on axial CT scan and 19.44 mm × 6.20 mm on coronal CT scan. Tissue biopsy is needed for diagnosis of lacrimal gland enlargement because it is difficult to distinguish the type of the lacrimal gland pathology based solely on clinical or radiological presentation.

Новообразование орбиты у пациента с хроническим вирусным гепатитом С с системными проявлениями (клиническое наблюдение)

Aim. To present the case report of orbital tumor in patient with chronic hepatitis C with systemic manifestations, diagnostic and treatment approaches in such patients. Material and methods. We describe a clinical case of 48 y.o. female patient complaining of right-sided upper eyelid swelling and ptosis. According to MRI an orbital tumor was previously visualized in the location of right lacrimal gland. It was known, that the patient suffers from HCV infection since 1992. She was prescribed local and systemic immunosuppressive treatment with a positive effect, however due to hemorrhagic vasculitis development and eyelid swelling recurrence 2 weeks later she was admitted to the hospital. In diagnostic purpose the orbital tumor was surgically excised followed by morphological and immunohistochemical investigation. Results. A differential diagnosis between chronic dacryoadenitis, lymphoid tissue hyperplasia (due to longstanding antigene stimulation (MALT – tissue) and marginal lymphoma (MALT – lymphoma) was performed. Immunohistochemical staining and PET – CT failed to confirm the lymphoproliferative disease. However, in 1 year due to tumor recurrence, repeated biopsy followed by previously described analyses confirmed extranodal marginal zone B-cell lymphoma (MALT – lymphoma). Repeated PET-CT showed metabolic activity in the right orbit and in the location of paraesophageal lymph node. The patient received polychemotherapy inducting lymphoma remission. Conclusion. Current clinical case shows that differential diagnosis of orbital tumors in patients with chronic hepatitis C should include lymphoproliferative diseases. Key words: orbit neoplasm, dacryoadenitis, lymphoma, chronic viral hepatitis C.

Diseases of the Lacrimal Gland

Tear gland diseases are comparatively rare in daily ophthalmological practice. The aim of this article is on the one hand to present the necessary basic knowledge and on the other hand to draw a bow to important current publications on the subject in order to provide the reader with sufficient knowledge for daily practice after reading it.Inflammatory (acute and chronic specific dacryoadenitis, idiopathic dacryoadenitis), neoplastic (epithelial and nonepithelial) and structural (cysts etc.) are presented.Diagnostically, the importance of (open) biopsy has increased again in recent years. Only the pleomorphic adenoma of the lacrimal gland has to be removed in-toto in order to avoid a recurrence or malignant degeneration which is difficult to control curatively. Differential diagnostic aids are explained in detail.

Clinicopathologic features of biopsied lacrimal gland masses in 95 Korean patients.

To investigate the clinicopathologic features of lacrimal gland masses biopsied in a tertiary referral hospital in Korea. Records from 95 Korean patients who underwent lacrimal gland mass biopsy were retrospectively reviewed. Data included demographics, clinical presentation, imaging findings, histopathologic diagnosis, and associated systemic disease. The median age was 52.0years (range, 16-76years), and 51 patients (53.7%) were female. Thirty-three patients (34.7%) had bilateral disease. The histopathologic diagnoses were as follows: chronic dacryoadenitis (52.6%, n = 50;29 non-specific and 21 immunoglobulin G4-related disease (IgG4-RD)), lymphoproliferative disease (25.5%, n = 24; 18 lymphoma and six lymphoid hyperplasia), benign epithelial tumour (13.7%, 13 pleomorphic adenoma), malignant epithelial tumour (3.2%, three adenoid cystic carcinoma), dacryops (3.2%, n = 3), solitary fibrous tumour (1.1%, n = 1), and xanthogranulomatous inflammation (1.1%, n = 1). Patients with chronic dacryoadenitis were significantly more likely to be younger (mean 47.5years), have bilateral involvement (52.0%), and have a longer symptom period (mean 15.6months) than those with lymphoproliferative disease (60.0years, 25.0%, and 6.7months, respectively; p < 0.05, each comparison). Patients with IgG4-related dacryoadenitis were significantly more likely to have bilateral involvement (85.7%) and have associated systemic involvement (52.4%) than those with non-specific dacryoadenitis (37.9 and 0%, respectively; p < 0.05, each comparison). Sixteen patients (16.8%) had associated systemic involvement: 11 with IgG4-RD and 5 with lymphoma. Chronic dacryoadenitis and lymphoproliferative disease were the most common causes of lacrimal gland masses in our cohort. Younger patients with bilateral involvement and a longer symptom period were more likely to have chronic dacryoadenitis than lymphoproliferative disease. Associated systemic involvement was not rare in patients with IgG4-RD or lymphoma. Our results suggest that biopsy of chronic lacrimal gland masses should be performed for proper evaluation and management.

Histopathological spectrum of ophthalmic lesions in Chhattisgarh: study from a tertiary care centre

Background: Ophthalmic pathology is a subspecialty of cellular pathology which deals with the diagnosis and characterization of eye diseases. Ophthalmic pathologists study tissues excised by ophthalmologists through evisceration and enucleation and other small biopsies from various ophthalmic surgeries. Ophthalmic lesions include a wide spectrum of lesions ranging from benign, premalignant to malignant lesions. The diagnosis of these lesions is based on the clinical as well as histopathological features. Methods: The present study was an observational prospective study conducted on a 62 ophthalmic biopsies received in Histopathology laboratory, Department of Pathology, Pt. J.N.M. Medical College and Dr. B.R.A.M. Hospital, Raipur, Chhattisgarh, India, between March 2016 and October 2016. Institutional ethics committee of Pt. J.N.M. Medical College, Raipur, India has approved this study. Informed written consent was collected from the patients or their relatives. Results: Out of 62 cases of ophthalmic biopsies, 38 (61.29%) were non-neoplastic and 24 (38.71%) were neoplastic lesions. Among non-neoplastic lesions, inflammatory lesions of lacrimal sac were more common 16 (42%) followed by conjuctival lesions 10 (26.3%). Among the neoplastic lesions, retinoblastoma was the most common malignancy 11 (45.83%). Conclusions: The most common ocular malignancy was retinoblastoma; among non-neoplastic disorder, chronic dacryoadenitis followed by benign lesions of conjunctiva. Some ocular masses composed of rhino sporangium involving conjunctiva, lid or lacrimal sac.

Adenoid cystic carcinoma of the lacrimal gland masquerading as dacryoadenitis: Case Report

Adenoid cystic carcinoma (ACC) of the lacrimal gland is a rare, life-threatening tumor often not diagnosed until advanced stages. This malignancy usually presents with nonspecific symptoms such as periocular pain and non-axial proptosis. We present a case of a 40-year-old female experiencing these symptoms, whose initial lacrimal gland biopsy was consistent with chronic dacryoadenitis. Reappearance and worsening of her symptoms after treatment led to a total dacryoadenectomy, which confirmed the presence of ACC. Close follow up of the clinical signs and imaging in this patient allowed us to suspect and finally diagnose an invasive malignancy that was masquerading as a chronic dacryoadenitis.

THE TEAR FLUID QUALITATIVE CHARACTERISTIZATION IN PATIENTS AFTER LACRIMAL GLAND EXCISION

The impact of lacrimal gland excision on tear production and qualitative tear fluid composition in patients after excision of a pathologically changed lacrimal gland (tumors, chronic dacryoadenitis).

A Survey of the Histopathology of Lacrimal Gland Lesions in a Tertiary Referral Centre

Purpose: To describe the histopathological distribution of biopsied lacrimal gland lesions in a tertiary referral centre in Singapore.Methods: This was a retrospective chart review. Clinical records of patients who underwent lacrimal gland biopsy at a tertiary referral centre in Singapore between 2000 to 2010, were reviewed. Data collated included patient demographics, clinical presentation, association with systemic disease and histopathological diagnosis.Results: Sixty-nine patients were studied. Median age of presentation was 50 years. Forty patients (58%) were female and the majority (84%, n = 29) were Chinese. The mean follow-up duration was 35.0 ± 34.5 months. 30.4% (n = 21) of the patients had bilateral disease. Chronic dacryoadenitis (46%, n = 32) was the most common histopathological diagnosis, followed by lymphoproliferative disorders (38%, n = 26) and pleomorphic adenoma (10%, n = 7). The diagnoses in four other patients included adenoid cystic carcinoma, lacrimal gland hypertrophy, lacrimal duct cyst and orbital vascular malformation.Conclusion: Chronic dacryoadenitis and lymphoproliferative disorders are the two commonest causes of lacrimal gland lesions in our series. Although many cases remain non-specific, about 60% have a specific inflammation that may be associated with a systemic disease. As one third of our patients with lymphoproliferative disease of the lacrimal gland had an associated systemic lymphoma, patients with such lesions should be referred for investigation of possible systemic lymphoma. The results of our study can aid in providing a more targeted approach to patient management.

Lesions mimicking lacrimal gland pleomorphic adenoma

AimTo report a series of patients with lacrimal gland lesions simulating the clinicoradiological features of lacrimal gland pleomorphic adenoma (LGPA).MethodsMulticentre retrospective, interventional case series. Clinical records of all patients with...

Orbital Necrobiotic Xanthogranuloma Associated With Systemic IgG4 Disease

Purpose: The authors describe 2 cases of orbital xanthogranulomatous disease associated with an increase in IgG4-positive plasma cells, and also examine IgG4 in other types of orbital inflammation.Methods: Immunohistochemistry for total IgG and IgG4 was performed in 18 cases of orbital inflammation, including chronic dacryoadenitis (n = 10), necrobiotic xanthogranuloma (n = 2), xanthogranuloma (n = 1), idiopathic orbital inflammation/pseudotumor (n = 4), and fungal infection (n = 1).Results: One patient presenting with necrobiotic xanthogranuloma had signs of systemic IgG4 disease. His orbital lesion showed an elevated number of IgG4 positive plasma cells (55%). An orbital xanthogranulomatous lesion in a second patient lacking systemic symptoms also contained a high percentage of IgG4-positive plasma cells (80%). Only 1 case of chronic dacryoadenitis contained prominent IgG4-positive plasma cells (mean 17/hpf).Conclusions: IgG4-positive plasma cells are relatively rare in nonsclerosing orbital inflammatory lesions. However, systemic disease IgG4 can be associated with necrobiotic xanthogranuloma of the orbit.

Processos expansivos orbitoesfenoidais: estudo anatomopatológico de 82 casos

To describe the orbito-sphenoidal expansive processes diagnosed at the Anatomo-Pathological Laboratory of the Fundação Faculdade Federal de Ciências Médicas de Porto Alegre - Complexo Hospitalar Santa Casa de Porto Alegre during a period of 15 years, evaluating their relative frequencies among other tumoral processes. We performed a statistical analysis of all orbital tumors with anatomicopathological diagnosis from January 1968 to December 1982, comparing the frequency of orbital tumors with the total number of tumors diagnosed at this Laboratory in a period of 5 years. Eighty-two cases were diagnosed of expansive processes involving the orbit in 15 years - 20.7% of the total (17 cases) affecting children (up to 14 years-old) and the remaining 79.3% (65 cases) affecting adults. Children presented more frequently optic nerve gliomas (4 of 6 cases 66.6%), retinoblastomas (4 cases - 100%) and rhabdomyosarcomas (3 of 4 cases - 75%). Less frequent diseases in children were optic nerve meningioma, neurofibroma, inflammatory pseudotumor, chronic dacryoadenitis, neuroma and chronic inflammatory process. The adult population presented more cases of basal cell carcinomas (18 cases), squamous cell carciomas (12 cases), meningiomas (10 cases), choroidal malignant melanoma (3 cases) and lacrimal gland tumors (7 cases). Tumors originated from bone or vascular structures, pseudo-tumors, and intraorbital epidermic cysts were also diagnosed, among others. Of a sum of 2,639 tumors diagnosed at this Laboratory in the time period of 5 years (1976 to 1980), there were 22 cases of orbital tumors, reaching a total of 0.8% of all cases. The anatomicopathological study of these processes is somehow important to diagnose and to establish an adequate therapy. The incidence of the expansive processes involving the orbit allows an epidemiological characterization of the different medical services responsible for the treatment of eye diseases. Despite its lower frequencies when compared to the diagnosis from other branches of Anatomical Pathology, Eye pathology has developed considerably over the past few years, contributing to more accurate diagnosis and to a better understanding of mechanisms responsible for these processes.

A case of immunoglobulin G4-related chronic sclerosing sialadenitis and dacryoadenitis associated with tuberculosis

We describe a 64-year-old woman with chronic sclerosing sialadenitis and dacryoadenitis, which developed during treatment for cervical lymph node tuberculosis. Anti-tuberculosis treatment did not improve the swelling in the lacrimal and submandibular glands, and a biopsy specimen of the lacrimal gland showed inflammation, with abundant lymphoid follicles with fibrosis and granuloma without caseous necrosis. Immunohistological examination of a repeat biopsy specimen showed abundant immunoglobulin (Ig) G4-positive plasma cell infiltration. Corticosteroid therapy improved the salivary gland swelling without reactivation of the tuberculosis. This case suggests that an abnormal immunological reaction to tuberculosis may be one of the etiological candidates for IgG4-related disease.

Monoclonal antibodies in the diagnosis of lymphoproliferative diseases of the orbit and orbital adnexae

To describe the value of monoclonal antibodies in the differential diagnosis of orbital lymphoproliferative disorders. A total of 200 sequential cases of malignant lymphoma diagnosed at St Bartholomew's Hospital as part of the ocular lymphoma service at Moorfields Eye Hospital and the Institute of Ophthalmology have been examined. Cases were acquired between January 1998 and June 2005. Each case had detailed immunophenotypic analysis using a panel of monoclonal antibodies and was classified using the WHO classification of lymphoma. These cases are discussed in relation to earlier studies of orbital lymphoma reported by our group. Lymphomas fell into three main categories. Extranodal marginal zone lymphoma was the largest group with 151 cases, arising within the orbital soft tissue, conjunctiva and lacrimal gland. Cases arising in the conjunctiva and lacrimal gland showed a higher female predominance than those arising within the deeper soft tissue. A small number of cases were associated with organ specific autoimmunity, including thyroid eye disease complicating Graves' disease. Follicular lymphoma and diffuse large B-cell lymphoma formed the next two groups, occurring with equal frequency. Many of the follicular lymphomas had evidence of disseminated disease on completion of staging. A miscellaneous group of T-cell and B-cell lymphomas formed a minority of cases during the study period. Extranodal marginal zone lymphoma is the most frequent type of primary orbital and orbital adnexal lymphoma. Its major differential diagnosis is with orbital lymphoid hyperplasia, chronic dacryoadenitis, and follicular conjunctivitis. Systemic types of lymphoma may present within the orbit or involve the orbit secondarily.

Magnetic resonance imaging of unilateral lacrimal gland lesions.

To report the findings on magnetic resonance imaging (MRI) of various benign and malignant unilateral lacrimal gland lesions. This is a retrospective noncomparative interventional case series. Thirty-one consecutive patients with a unilateral lacrimal gland lesion were analyzed. The preoperative MRI findings were correlated with the pathology results. The main outcome measures were anatomic extent, configuration, margins, angulation, internal features on T1- and T2-weighted images (with respect to extraocular muscles and cerebral gray matter), contrast enhancement of the lesion and adjacent bone change on MRI. Of the 31 patients, 21 had chronic dacryoadenitis, 3 had lymphoid tumors, and 7 had epithelial tumors including pleomorphic adenoma (3), adenoid cystic carcinoma (3), and pleomorphic adenocarcinoma (1). The results of the patients with chronic dacryoadenitis demonstrated involvement of the orbital lobe alone in 13 patients (62%), involvement of both orbital and palpebral lobes in 8 (38%), a molded configuration with ill-defined margins, sharp angles in 13 (62%), round angles in 8 (38%), lack of bone change, an isointense internal signal on T1-weighted images, a hypointense signal on T2-weighted images, and moderate contrast enhancement. The patients with lymphoid tumors demonstrated involvement of the orbital lobe, a molded configuration with ill-defined margins and sharp angles, lack of bone change, an isointense internal signal on T1-weighted images, an isointense signal on T2-weighted images, and moderate contrast enhancement. Those with epithelial tumors showed involvement of the orbital lobe, a well-circumscribed oval configuration, and round angles. Pleomorphic adenoma demonstrated smooth margins, bone expansion in two patients, and no bone change in one. Adenoid cystic carcinoma and pleomorphic adenocarcinoma showed irregular margins and bone destruction. All epithelial tumors demonstrated an isointense internal signal on T1-weighted images, a hyperintense signal on T2-weighted images, and moderate contrast enhancement. It is difficult to uniformly correlate the MRI features and histopathologic findings in lacrimal gland lesions. However, MRI seems to be useful in determining the etiology of a unilateral lacrimal gland lesion. Internal tissue features on T1- and T2-weighted images of MRI are most helpful in categorizing these lesions. Although the number of patients is small, our findings suggest that there are differences in orbital MRI findings of inflammatory lesions and lymphoid tumors as compared to benign and malignant epithelial tumors in the lacrimal gland fossa.

Chronic Dacryoadenitis Misdiagnosed as Eyelid Edema and Allergic Conjunctivitis

Purpose: To report the case of a 53-year-old woman with a 2-year history of episodic upper eyelid swelling and nonspecific complaints, who was diagnosed as having allergic conjunctivitis. Methods: A complete ocular examination, orbital computerized tomographic (CT) scans followed by complete physical and systemic examinations. Results: The results of physical and systemic examinations were unremarkable for systemic lymphoma and a primary focus of cancer. The results of the ocular examination were normal. CT scans demonstrated well-defined lesions bilaterally with a homogeneous internal structure in the lacrimal gland fossa, which suggested a diagnosis of chronic dacryoadenitis. The differential diagnosis included lymphoma and orbital metastases. The patient refused a biopsy and was started on a tapering dose of 60 mg oral prednisolone daily. The follow-up CT scans 1 month after cessation of 6-week oral corticosteroid treatment showed near complete resolution of the orbital lesions. Conclusion: This case demonstrates that orbital inflammation can be misdiagnosed as refractory allergic conjunctivitis.

Epithelial tumors of the lacrimal gland

The clinical features of 30 primary epithelial lacrimal gland tumors encountered during a 30-year-period are discussed. Fourteen patients (46.6%) had pleomorphic adenomas, twelve (40%) had adenoid cystic carcinoma, two (6.7%) had pleomorphic adenocarcinoma and two (6.7%) had mucoepidermoid carcinoma. Patients with malignant tumors usually had a short history of a few months with pain while pleomorphic adenoma cases had a longer history without pain. Pleomorphic adenomas generally caused enlargement of bone but there was bone erosion in two cases. Malignant tumors were radiologically normal or there was bone expansion or destructive changes. Chronic dacryoadenitis was found to closely masquerade both the benign and malignant lacrimal gland tumors. An en bloc excisional biopsy is necessary in pleomorphic adenomas while an extraperiosteal approach should not be used in malignant tumors.

Chronic bilateral dacryo-adenitis in identical twins: a possible incomplete form of Sjögren syndrome

We report an unusual case of chronic bilateral dacryo-adenitis in 10-year-old identical twin sisters. Both girls presented with bilateral lacrimal gland enlargement and developed moderate xerophthalmia and keratitis. Both the lacrimal and minor salivary gland biopsies showed a non-granulomatous inflammatory infiltration of mono-nuclear cells. All granulomatous diseases and neoplasms could therefore be ruled out and only Sjögren syndrome and very few other forms of chronic dacryo-adenitis remained as possible diagnoses. Both patients and their parents were evaluated for auto-antibodies. Very low titres of smooth muscle antibodies were found in one, antinuclear antibodies in two and anti-dsDNA antibodies in all four members of the family. Even though the titres of antinuclear and anti-dsDNA antibodies increased in one of the sisters, both patients did not develop any sign or symptom of a systemic connective tissue disease. During the 6 years' follow up, both patients showed persistent tarsal gland enlargement but no other symptoms apart from a moderate xerophthalmia and occasional mild keratitis.

Bilateral Lacrimal Duct Cyst

Reports of lacrimal gland cysts have been rare, and mostly of small, unilateral cysts of the palpebral lobes. An unusual case of bilateral, large acquired cysts of the orbital lobe of the lacrimal glands is presented. Pathologic examination showed chronic inflammation and fibrosis of the lacrimal gland structures. We suggest that prolapse of the lacrimal glands with obstruction of the lacrimal ducts, combined with chronic dacryoadenitis, led to cyst formation.