Vahdani, Kaveh; Rose, Geoffrey E.
To compare serum angiotensin-converting enzyme (sACE) titers in patients with newly diagnosed sarcoid-like dacryoadenitis and nongranulomatous dacryoadenitis.
Retrospective comparative case series.
Patients and Methods:
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.
Rebecca W. Liong MD Armi Salonga‐Reyes FRACP Sharon Morris FRANZCO
No abstract available
Kwan, Changyow C.; Prager, Alisa J.; Huang, Russell M.; Bryar, Paul J.; Thyparampil, Preeti J.
This is the first case of histopathologically proven blastomycosis involving the lacrimal gland. A 51-year-old woman with a history of disseminated blastomycosis involving her lungs and skin, on oral itraconazole, presented with 3 days of right upper eyelid swelling, erythema, and pain concerning for recurrent dacryoadenitis. MRI showed enlargement of the right lacrimal gland with a cystic lesion at the anterior aspect of the gland with a radiographic differential diagnosis of abscess versus cyst. After no improvement with intravenous antibiotics, orbitotomy with lacrimal gland biopsy and incision and drainage of the cystic lesion were performed. Culture and pathology of the drained fluid demonstrated an abscess with both viable and nonviable broad-based budding yeast consistent with partially treated blastomycosis. The patient’s symptoms improved after the surgery and continued itraconazole therapy.
Yu-Chieh Hung, Delia D. Wang, Louis S. M. Han, Thomas D. Betts & Robert G. Weatherhead
In systemic lupus erythematosus, ophthalmic manifestations are noted in up to one-third of patients. We describe a patient with an unusual initial presentation of this disorder.
Luke Halliday, David Curragh, Simon McRae, Dinesh Selva
Amyloidosis is a heterogeneous collection of conditions that presents with variable organ dysfunction due to extracellular aggregation of abnormally folded proteins. Systemic light chain amyloidosis is an uncommon disease, and involvement of orbital structures is particularly rare. We present a case of chronic dacryoadenitis in which λ light chain amyloid deposition was detected in the lacrimal gland 6 months after a nonspecific biopsy.
Chanjoo Ahn, Sunah Kang, Ho-Seok Sa
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 52.0 years (range, 16–76 years), 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 47.5 years), have bilateral involvement (52.0%), and have a longer symptom period (mean 15.6 months) than those with lymphoproliferative disease (60.0 years, 25.0%, and 6.7 months, 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.
Thomas L. Jenkins, Matthew M. Zhang, Neal S. Patel, Alison B. Huggins & Jacqueline R. Carrasco
Adnexal and periocular involvement in Neisseria gonorrhoeae (NG) infection is rare. This report describes the case of a patient with a delayed diagnosis of gonococcal dacryoadenitis with contiguous conjunctivitis and corneal involvement. She underwent extensive inpatient laboratory and infectious workup but rapidly progressed to corneal perforation requiring emergent penetrating keratoplasty prior to a positive culture confirming the diagnosis. To date, this is the first reported case of ophthalmologic NG infection with associated conjunctivitis, dacryoadenitis, and corneal perforation.
Akella, Sruti S.; Schlachter, Dianne M.; Black, Evan H.; Barmettler, Anne
Purpose: To review and summarize the clinical features, presentations, diagnostic modalities, and management of ophthalmic manifestations of eosinophilic granulomatosis with polyangiitis (EGPA, formerly Churg–Strauss Syndrome).
Methods: A systematic PubMed search of all English articles on EGPA with ophthalmic involvement was performed. Emphasis was placed on English-language articles, but any article with an abstract translated into English was also included. Only those cases that satisfied the American Rheumatology criteria (1990) for diagnosis were included. Data examined included epidemiology, pathogenesis, presentations, diagnostic modalities, and management.
Results: There was a wide range in ophthalmic manifestations of EGPA. In order of most frequent presentation to least frequent, these include central retinal artery or vein occlusion, ischemic optic neuropathy, conjunctival nodules, orbital myositis, proptosis, dacryoadenitis, retinal vasculitis/infarcts/edema, cranial nerve palsy, and amaurosis. The 46 qualifying cases were divided into the categories of ischemic vasculitis versus idiopathic orbital inflammation due to prognostic significance. Ischemic vasculitis cases tended to be older patients (p = 0.03), unilateral (p = 0.006), require immunosuppressive therapy beyond steroids (p = 0.015), and were less likely to show improvement on therapy (p = 0.0003).
Conclusions: Prompt diagnosis of EGPA by the ophthalmologist can decrease patient morbidity and mortality. This requires knowledge of likely ophthalmic EGPA presentations, as well as recommended workups and treatment.
Eri Ishikawa, Yasuhiro Takahashi, Ma. Regina Paula Valencia, Marian Grace Ana-Magadia, Hirohiko Kakizaki
To examine the clinicopathological characteristics of patients with thyroid eye disease (TED) who showed asymmetric lacrimal gland enlargement.
Sixteen patients were included in this retrospective, observational case series. The following data were collected: sex, age, laterality, past medical history, laboratory results for thyroid function and other autoimmune disorders, pathological results, other systemic lesions related to lacrimal gland pathology, and the clinical course after treatment.
The results of pathological examinations were consistent with non-specific dacryoadenitis in seven patients, immunoglobulin G4-related dacryoadenitis in four patients, sarcoidosis in two patients, and xanthogranuloma in one patient, respectively. The remaining two patients were proven to be extra-nodal marginal zone lymphoma of mucosa-associated lymphoid tissue type (MALT lymphoma). One patient with sarcoidosis was noted to have another related systemic lesion. After treatment or observation, the lesions were either improved or stable in almost all patients, except for the two patients with MALT lymphoma.
Patients with TED who present with asymmetric lacrimal gland enlargement need to be further evaluated. Biopsy of the lacrimal gland and full systemic work-up should be considered as these may lead to detection of other pathological entities, especially malignancies, which could help in saving patients’ lives.
Kauser Hina, Monga Seema, Khan Taskin, Haider Mehvish
We report a case of young adult female with swelling in the superolateral aspect of left orbit for two weeks which was diagnosed as tubercular dacryoadenitis with intracranial extension without any neurological features. Tubercular dacryoadenitis is very rare but still makes an important differential diagnosis of lacrimal gland swellings especially in endemic areas like India. Few cases of tubercular dacryoadenitis have been reported in the past; but not a single case with intracranial extension in young adults with short history and without neurological symptoms. Although radiological investigations are routinely done in orbital lesions, it is advisable do so even in acute cases to look for intracranial extension before the appearance of neurological features. Tubercular dacryoadenitis though rare, should be kept as a differential diagnosis of acute or chronic lacrimal gland swellings in endemic areas.