Presumed Lacrimal Gland Pleomorphic Adenoma With Extensive Ossification and Necrosis.

Calcification within pleomorphic adenomas of the lacrimal gland is well recognized but uncommon, being seen more readily in lacrimal gland carcinomas. Bony formation, ossification, in pleomorphic adenomas of the lacrimal glands is even rarer. Together with extensive sclerosis, or "coagulative necrosis," ossification and necrosis should alert the clinician to the risk of malignant transformation. However, both can mimic carcinomatous change, leading to misinterpretation of malignancy in an otherwise benign lacrimal gland neoplasm. We present 2 case reports of patients with clinically presumed pleomorphic adenomas of the lacrimal gland whose histopathology demonstrated lacrimal gland ossification and necrosis without features of malignancy or invasive disease.

Whole-orbit-based multiparametric assessment of disease activity of thyroid eye disease on Dixon MRI.

PURPOSE: This study aimed to explore the diagnostic value of whole-orbit-based multiparametric assessment on Dixon MRI for the evaluation of the thyroid eye disease (TED) activity. METHODS: The retrospective study enrolled patients diagnosed as TED and obtained their axial and coronal Dixon MRI scans. Multiparameters were assessed, including water fraction (WF), fat fraction (FF) of extraocular muscles (EOMs), orbital fat (OF), and lacrimal gland (LG). The thickness of OF and herniation of LG were also measured. Univariable and multivariable logistic regression was applied to construct prediction models based on single or multiple structures. Receiver operating characteristic (ROC) curve analysis was also implemented. RESULTS: Univariable logistic analysis revealed significant differences in water fraction (WF) of the superior rectus (P = 0.018), fat fraction (FF) of the medial rectus (P = 0.029), WF of OF (P = 0.004), and herniation of LG (P = 0.012) between the active and inactive TED phases. Multivariable logistic analysis and corresponding receiver operating characteristic curve (ROC) analysis of each structure attained the area under the curve (AUC) values of 0.774, 0.771, and 0.729 for EOMs, OF, and LG, respectively, while the combination of the four imaging parameters generated a final AUC of 0.909. CONCLUSIONS: Dixon MRI may be used for fine multiparametric assessment of multiple orbital structures. The whole-orbit-based model improves the diagnostic performance of TED activity evaluation.

Mucosa-associated lymphoid tissue lymphoma of the lacrimal gland: A case report and literature review.

RATIONALE: The most common subtype of primary lymphoma of the ocular adnexa is the mucosa-associated lymphoid tissue (MALT) subtype. MALT lymphoma of the lacrimal gland is relatively rare among the lacrimal gland tumors, and the early clinical symptoms are atypical, which can easily lead to misdiagnosis and missed diagnosis. Here, we report a case of MALT lymphoma of the lacrimal gland and explore its clinical manifestations, pathological characteristics, management, and pathogenesis, with the aim of helping clinicians gain an in-depth understanding of ocular adnexal MALT lymphoma. PATIENT CONCERNS: A 60-year-old man presented to our hospital with proptosis and diplopia. The right eye deviated and shifted toward the lower part of the nose. DIAGNOSIS: Orbital enhanced magnetic resonance imaging suggested a mass with a maximum cross-section of 3.2 × 2.1 cm. T1 weighted image was isointense, and the enhancement was more uniform and obvious. INTERVENTIONS: The right orbital mass was treated surgically, and the final pathology report was MALT lymphoma. After the pathological report was released, the patient was transferred to the hematology department for further diagnosis and no further treatment was given eventually. OUTCOMES: Seven months later, the patient did not complain of discomfort. Whole-body positron emission tomography-computed tomography, superficial lymph node examination and orbital magnetic resonance imaging revealed no abnormal changes. LESSONS: The clinical manifestations of MALT lymphoma are heterogeneous. Imaging examination is important for assessing the size of the tumor and its relationship with adjacent tissues. Postoperative pathological examination may provide further evidence for the evaluation of the patient's surgical efficacy and prognosis. Management of MALT lymphoma of the lacrimal gland requires a multidisciplinary approach involving ophthalmologists, hematologists, and radiotherapists.

In Vivo Human Lacrimal Gland Imaging Using an Ultrasound Biomicroscopy.

PURPOSE: In the present study, we introduce human lacrimal gland imaging using an ultrasound biomicroscopy (UBM) with a soft cover and show their findings. METHODS: The representative UBM findings of palpebral lobes in seven subjects (four with non-Sjögren dry eye syndrome, one with Sjögren syndrome, and two healthy subjects) were described in this study. To prolapse the palpebral lobe, the examiner pulled the temporal part of the upper eyelid in the superotemporal direction and directed the subject to look in the inferonasal direction. We scanned the palpebral lobes longitudinally and transversely using UBM. We used an Aviso UBM with a 50 MHz linear probe and ClearScan. RESULTS: In UBM of two healthy subjects, the echogenicity of the lacrimal gland was lower than that of the sclera and homogeneous. But the parenchyma of a patient with Sjögren dry eye syndrome was quite inhomogeneous compared to the healthy subjects. In two patients with dry eye syndrome, we were able to observe some lobules in the parenchyma. We could find excretory ducts running parallel at the surface of the longitudinal section in some subjects. In the longitudinal UBM scan of a subject, we observed a tubular structure at a depth of 1,500 µm that was considered a blood vessel. It ran from the superonasal to the inferotemporal direction. In a subject, we observed a large cyst beneath the conjunctiva. CONCLUSIONS: Lacrimal gland imaging using UBM has both advantages of optical coherence tomography and sonography, and could be useful for evaluating dry eye syndrome.

Radiological measurements of lacrimal gland in thyroid eye disease.

PURPOSE: Lacrimal gland enlargement is a common feature of thyroid eye disease (TED) and has been positively correlated with the clinical activity score. Although lacrimal gland volume is the preferred measure of lacrimal gland size, its calculation is not clinically translatable due to the expertise, time and advanced software required. The aim of our study is to determine whether the lacrimal gland volume in patients with TED undergoing magnetic resonance imaging (MRI) can be estimated using simpler lacrimal gland linear and area measurements. METHODS: A retrospective review of 102 orbits (51 patients) with TED who underwent orbital MRI was conducted. The maximum length, width, and area of the lacrimal gland were measured in axial and coronal sections. Lacrimal gland volume was calculated by using a manual segmentation technique on all consecutive axial slices on commercially available software, OsiriX. All quantitative measurements were correlated with the lacrimal gland volume. RESULTS: Mean age of participants was 59 ± 16 years, and 67% (n = 34) were females. With multivariate analyses, combined lacrimal gland axial and coronal areas strongly correlated with volume (r = 0.843, p < 0.01). Strong univariate predictors of volume included axial area (r = 0.704, p < 0.01) and coronal area (r = 0.722, p < 0.01), while moderate predictors included axial length (r = 0.523, p < 0.01), axial width (r = 0.521, p < 0.01), coronal length (r = 0.450, p < 0.01), and coronal width (r = 0.649, p < 0.01). CONCLUSION: In patients with thyroid eye disease, lacrimal gland volume can be estimated using axial and coronal areas, which is simpler and more time efficient than calculating volumes.

A major review of punctal stenosis: Updated anatomy, epidemiology, etiology, and clinical presentation.

We aim to provide a detailed and updated literature review on the epidemiology, etiology, clinical presentations, histopathology, and ultrastructural features of punctal stenosis. There are inconsistencies in the definition and staging of punctal stenosis. While advanced optical coherence tomography imaging techniques have revolutionized the way the punctum and vertical canaliculi are assessed or monitored following treatment, the planes of measurement to characterize punctum anatomy need to evolve further. The current criteria for diagnosing and grading punctal stenosis are inadequate and based on empirical clinical findings. There is increasing evidence of the role of lymphocytes and myofibroblasts in the pathogenesis of punctal stenosis. There is a need for a uniform assessment of punctal stenosis and a uniform reporting of severity that would help standardize the several management options available in the lacrimal armamentarium.

New Multi-Parameters Combination of Technetium-99m-Diethylene-Triamine-Pentaacetate Orbital Single-Photon Emission Computed Tomography/Computed Tomography for the Evaluation of Graves' Orbitopathy Activity.

PURPOSE: This study investigates the new combined parameters of 99mTc-DTPA orbital single-photon emission computed tomography/computed tomography (SPECT/CT) for the evaluation of Graves' orbitopathy (GO) activity. METHODS: A retrospective analysis was performed on 41 patients. All the patients undergone the 99mTc-DTPA orbital SPECT/CT and were categorized into active and inactive group based on the standard combined by the clinical active score (CAS), magnet resonance imaging (MRI) and/or follow-up results. Quantitative parameters of lacrimal gland (LG) including the protruding degree of lacrimal gland herniation (LGH) and uptake ratios (URs) of region of interest (ROI) drawn on lacrimal gland and occipital bone. SPECT/CT reading results were based on visual analysis. Parameters were compared between the two groups and the diagnostic value on discrimination of GO activity was also evaluated. RESULTS: All parameters of SPECT/CT for active GO groups were significantly higher than those of the inactive groups (p<.05). There were notable linear positive correlations between the assumption standard and readings as well as combination models 2 and 3 (r = .794, r = .772, r = .760, respectively). ROC analysis indicated that model 2 provided the highest diagnostic performance, exhibiting an area under the curve (AUC) of .947, a sensitivity of 92.7%, and a specificity of 88.6%. CONCLUSIONS: The combined use of SPECT/CT reading results and DTPA uptake parameters of LG offers a more objective and precise evaluation of active GO. This study further recommends 99mTc-DTPA SPECT/CT might be serving as a supplementary beneficial approach for CAS in evaluating GO activity.

Diagnostic value of dynamic contrast enhancement combined with conventional MRI in differentiating benign and malignant lacrimal gland epithelial tumours.

AIM: To establish the diagnostic value of the quantitative parameters of dynamic contrast-enhanced (DCE) magnetic resonance imaging (MRI) combined with conventional MRI in differentiating of benign and malignant lacrimal gland epithelial tumours. MATERIALS AND METHODS: A retrospective analysis of primary lacrimal gland epithelial tumours confirmed by histopathology was conducted. Conventional MRI features and DCE-MRI quantitative parameters were collected and subjected to analysis. The diagnostic value was evaluated using receiver operating characteristic (ROC) curve analysis. RESULTS: A total of 53 patients were enrolled of which 29 had malignant, whereas 24 had benign tumours. Conventional MRI revealed statistically significant differences between benign and malignant tumours regarding maximum tumour diameter, posterior margin characteristic, bone destruction, and erosion. The Ktrans and Kep values obtained by DCE-MRI were higher in malignant than in benign tumours, with a statistically significant (p<0.001 and p=0.022). A type I time-signal intensity (TIC) curve was more frequent in benign tumours, whereas a type II TIC curve was prevalent in malignant tumours (p=0.001). ROC analysis showed that Ktrans had the best diagnostic value of the DCE-MRI parameters (area under the ROC curve [AUC] of 0.822, 75.9% sensitivity, and 83.3% specificity, p<0.001). The combination of conventional MRI and DCE-MRI factors had the best diagnostic value and balanced sensitivity and specificity (AUC of 0.948, 93.1% sensitivity, and 91.7% specificity, p<0.001). CONCLUSIONS: The present findings indicate that the combination of quantitative parameters of DCE-MRI and image characteristics of conventional MRI have a high diagnostic value for the diagnosis of benign and malignant lacrimal gland epithelial tumours.

Advances in imaging of the lacrimal gland in Sjögren's syndrome: A narrative review.

Due to lymphocytic infiltration of the salivary and lacrimal glands, Sjogren's syndrome (SS), a systemic autoimmune illness that mostly affects the exocrine glands, causes dry mouth (xerostomia) and dry eyes (xerophthalmia). Additionally, SS is associated with various comorbidities such as cardiovascular diseases, infections, musculoskeletal diseases, and cancers. Among patients with SS, xerophthalmia frequently arises as a complication, leading to insufficient tear production or rapid tear evaporation, thereby causing discomfort, irritation, and a gritty sensation in the eyes. This article aims to examine recent advancements in the imaging of the lacrimal gland in Sjögren's syndrome and briefly discusses the utilization of various imaging examinations for the lacrimal gland in this particular disease.

Evaluation of Main Lacrimal Gland through Shear-wave Ultrasound Elastography in Patients with Low Schirmer Value

OBJECTIVE: To compare main lacrimal gland values through shear-wave elastography (SWE) in patients with low Schirmer value and unspecified Sjögren's syndrome (SS) with healthy controls. MATERIAL AND METHOD: Admitted to the ophthalmology department with Schirmer value <10 mm, randomly selected 46 eyes of 46 patients evaluated for Sjögren's syndrome (SS) in the rheumatology department between December 2022 and April 2023 were classified as low Schirmer group (LSG). Randomly selected 48 eyes of 48 patients at a similar age with Schirmer value >10 mm were included as controls. Main lacrimal gland SWE measurements in LSG and control groups were recorded and compared as meter/second (m/sec). RESULTS: Mean SWE values of the main lacrimal gland were measured as 2.78 ± 0.66 m/sec and 2.26 ± 0.29 m/sec in LSG and controls. SWE measurements were significantly higher in LSG patients than in controls (p<0.001). No significant correlation was found in the analysis between the Schirmer and the main lacrimal gland SWE values in LSG patients (p=0.702, r=0.058). No significant correlation was also detected between the Schirmer and main lacrimal gland SWE values in controls (p=0.097, r=0.242). No significant relationship was also found between age, gender, body mass index (BMI), and SWE values (p=0.351, p=0.493, p=0.328, respectively). CONCLUSION: Mean SWE value of the main lacrimal gland was determined as significantly higher in patients with aqueous lacrimal insufficiency without SS than in controls. We consider that SWE measurements may be an imaging method to support the diagnosis of aqueous lacrimal insufficiency and used in follow-ups of those with dry eye syndrome (DES) in the future.

Internal vascular anatomy of the human lacrimal gland: A protocol based on cadaver dissection and three-dimensional micro-computed tomography.

PURPOSE: To evaluate the feasibility of studying the vascular supply of the orbital and palpebral lobes of the human lacrimal gland using micro-computed tomography (micro-CT) and microscopic dissection. METHODS: The lacrimal gland artery of a fresh parasagittalized cadaver head (male, aged 76 years) was infused with a lead oxide-latex mixture near the occipital pole of the gland. The entire lacrimal gland was imaged using micro-CT and 3D cinematic rendering (CR) and then dissected under a surgical microscope. RESULTS: Micro-CT and CR images showed well-demarcated internal vascular branches of the lacrimal artery and their distribution within the orbital and palpebral lobes. The entire course of the artery and its branches could be visualized by CR and microscopic dissection, with the former showing better spatial orientation and finer branching. The main artery runs along the free edge of the aponeurosis of the levator palpebrae superior muscle and lies in the isthmus portion of the gland (between the orbital and palpebral lobes). The branches of the main lacrimal artery include one branch to the orbital adipose tissue just before entering the gland, two branches to the orbital lobe (medial and lateral), and two branches to the palpebral lobe (medial and lateral). The main artery terminates as palpebral and orbital lobe branches in the lateral half of the lacrimal gland. CONCLUSION: Latex and contrast-enhanced micro-CT is very well suited to visualize the vascular anatomy of the lacrimal artery within the gland. A large number of lacrimal gland examinations using the method presented here are required to demonstrate and understand the variability of the vascular anatomy of the human lacrimal gland.

Retrospective radiological analysis to establish anatomical variation in angulation between medial canthus and the middle turbinate, for optimal positioning of Lester Jones Lacrimal bypass tubes in treatment of Epiphora.

PURPOSE: To determine the optimum angle for placement of Lester Jones lacrimal bypass tube using fixed radiological markers on CT scan head with axial and coronal cuts, as well as analysing the anatomical variation and range of angulation between individuals within our local population. METHODS: A retrospective radiological study conducted on a randomly selected sample of 384 adult patients in a UK Teaching Hospital. The angle between the medial canthus and the middle turbinate was measured on CT scans of the head using fixed radiological anatomical landmarks and analysed using the IMPAX software. Patients with orbital or nasal fractures, as well as those with history of surgical procedures involving the facial bones, were excluded. The accuracy of our measurements was validated using three dimensional (3D) CT head reconstruction technology. RESULTS: Analysis of the results showed a range of angulation between 28-45 degrees, with a mean angle of 36.99 ± 4.78 SD. There was no significant correlation found when comparing the different age groups using the One Way ANOVA test. Furthermore, a non-significant correlation was found between males and females when their mean angles were compared using the independent t-test. CONCLUSION: Our study showed that the ideal angle for insertion of Lester Jones tube would be between 30-45 degrees, with a mean of 37 degrees. No significant correlation was found between the age of the patient and the ideal angle of insertion of Lester Jones tube. Moreover, no significant difference was found in the angle measurements between males and females.

An Unusual Case of Lacrimal Gland Dacryops in a 10-Year-Old Child.

Lacrimal gland dacryops are an unusual type of benign epithelial neoplasm. Most of the literature refers to dacryops of the lacrimal palpebral lobe, with orbital lobe involvement less frequent. The authors report a case of dacryops in a 10-year-old child involving the orbital lobe of the lacrimal gland. [J Pediatr Ophthalmol Strabismus. 2023;60(6):e79-e82.].

Isolated lacrimal gland enlargement as a presenting sign of relapse of B-cell acute lymphoblastic leukemia in an adult.

A 25-year-old woman with a history of acute lymphoblastic leukemia with Philadelphia chromosome (BCR 22q11.2-ABL1 (9q34)) presented with progressively worsening swelling and pain in her left upper eyelid of 3 months' duration. Magnetic resonance imaging showed a heterogeneously enhancing mass lesion in the left lacrimal gland. An incisional biopsy was performed; histopathology showed atypical lymphocytic cells, and flow cytometry revealed a similar immunophenotype to her previously diagnosed cancer. Her presentation was consistent with recurrence of acute lymphoblastic leukemia in the lacrimal gland, which can rarely be involved in relapse of acute lymphoblastic leukemia.

Clinical characteristics, radiologic features, and histopathology of biopsied lacrimal gland tumors.

Herein, we described the clinicopathologic and radiologic features of biopsied lacrimal gland tumors. A retrospective case series of 79 patients treated between 2004 and 2021 was reviewed. The median age was 48.9 years (range 18.3-88.3 years), with 51.9% females. The histopathologic diagnoses were as follows: immunoglobulin G4-related disease (IgG4-RD) = 23, reactive lymphoid hyperplasia = 14, lymphoma = 14, nonspecific inflammation = 10, adenoid cystic carcinoma (ACC) = 9, and pleomorphic adenoma = 9. The proportion of histopathologic diagnoses did not differ significantly over the range of symptom durations (≤ 1 month, > 1-3 months, > 3 months). Patients with ACC had significantly shorter symptom duration and more frequent proptosis than those with pleomorphic adenoma (p = 0.040 and p = 0.009, respectively). Patients with IgG4-RD were older (median 54.3 years) than those with nonspecific inflammation (36.2 years; p = 0.046). Patients with ACC were more likely to present with diplopia than those with lymphoma (p < 0.001). The superior wedge sign increased the likelihood of ACC compared with that of non-epithelial non-malignant lacrimal gland tumors (relative risk ratio = 13.44, p = 0.002). The overall survival of patients with ACC and lymphoma did not differ significantly. Although these patients present with a short symptom duration, urgent orbital imaging, tissue biopsy, and prompt treatment should be performed in patients with lacrimal gland tumors.

Punctal Plug Granuloma After Blepharoplasty.

This case report discusses a diagnosis of foreign body granuloma resulting from a punctal plug in a patient aged 66 years with chronic dry eye and a history of blepharoplasty.

New insights into lacrimal gland anatomy using 7T MRI and electron microscopy: Relevance for lacrimal gland targeted therapies and bioengineering.

PURPOSE: To study the tissue architecture, isthmus (connection between two lobes) of the lacrimal gland using preclinical 7T MRI in combination with histology and electron microscopy. METHODS: Ten lacrimal glands from Caucasian body donors (mean age 78.7 years) were studied using 7T-MRI (N = 5; scanned at 75-µm intervals), histology, and electron microscopy (N = 5) and 3D cinematic rendering (CR) techniques. RESULTS: 3D CR images showed uniform-sized lobules (widest lobule diameter, 1.68 ± 0.19 mm in orbital lobe, 1.68 ± 0.17 mm in palpebral lobe) in both lobes, separated by septae (size, 0.29 ± 0.09 mm). The internal framework of the gland resembled a honeycoomb pattern. In CR and histology, the isthmus contained glandular acini, large blood vessels, nerves, and no more than two ducts having a tortuous course towards the conjunctival surface. On assigning a color display to the rendered lacrimal gland, all glands showed a blood vessel originating from the main lacrimal artery just 5 mm beyond the hilum and making it course to the palpebral lobe via isthmus. The distance between the conjunctiva and the central substance of the orbital and palpebral lobe was 9.4 ± 0.2 mm and 2.8 ± 0.7 mm, respectively. Electron microscopy of the palpebral lobe revealed compact subepithelial layer in the overlying conjunctiva, followed by loosely scattered collagen bundles that contained the gland lobules. CONCLUSION: 3D-CR can be used to study the lacrimal gland microstructure, help fabricate a 3D scaffold for lacrimal gland bioprinting, and serve as guide for transconjunctival lacrimal gland targeted therapies i.e., 2.9 & 9 mm long needle to reach the orbital and palpebral lobe center, respectively in normal-size glands.

Normal dimensions of the lacrimal gland on magnetic resonance imaging in Indian adult population: a retrospective study.

Introduction: the lacrimal gland size is affected by a variety of pathologic conditions like inflammatory, infections, neoplastic, autoimmune and granulomatous disorders. Earlier, the dimensions of the gland were estimated by extracting lacrimal glands from cadavers, later ultrasonography and computed tomography studies were used, but had limited soft tissue differentiation. The aim of this study was to retrospectively evaluate Magnetic Resonance Imaging (MRI) data from normal orbits and determine lacrimal gland dimensions. Methods: five hundred and twelve (512) consecutive MRI brain contrast scans (of 240 females, of 272 males; age range 40±20 years) for non-orbital diseases were evaluated retrospectively. The mean axial length (AL), axial width (AW), coronal length (CL), and coronal width (CW) of each lacrimal gland were measured separately. Results: five hundred and twelve (512) MRI brain contrast scans of 272 men and 240 women, with mean age of 40 ± 20 years were included. Right and left LG dimensions were similar, mean AL (13.2±1.35 mm versus 13.11±1.24 mm), mean AW [3.5±0.99 mm versus 3.3±0.82 mm], mean CL [16.3±2.5 mm versus 16.10±2.4 mm], and mean CW (4.15±0.89 mm versus 4.11±0.85 mm). The AL of both lacrimal glands and the CL and CW of right but not left lacrimal glands were significantly lower in women than in men. Age showed significant correlations with the AL and CL of both LGs. Conclusion: in this study, evaluation of normal morphometric parameters of the lacrimal gland in Indian population was established. LG dimensions are smaller in women than men, as well as correlating with side and age.