Sunny X Tang1, Renelle P Lim1, Saad Al-Dahmash2, Sean M Blaydon3, Raymond I Cho4, Christina H Choe5, Michael A Connor6, Vikram D Durairaj7, Lauren A Eckstein5, Brent Hayek8, Paul D Langer9, Gary J Lelli10, Ronald Mancini11, Alexander Rabinovich12, Javier Servat13, John W Shore3, Jason A Sokol14, Angelo Tsirbas15, Edward J Wladis16, Albert Y Wu17, Jerry A Shields18, Carol Shields18, Roman Shinder19. 1. Department of Ophthalmology, SUNY Downstate Medical Center, Brooklyn, New York. 2. Ocular Oncology, Wills Eye Institute, Philadelphia, Pennsylvania; King Saud University, College of Medicine, Riyadh, Saudi Arabia. 3. Texas Oculoplastic Consultants, Austin, Texas. 4. Department of Ophthalmology, Walter Reed National Military Medical Center, Bethesda, Maryland. 5. Department of Ophthalmology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania. 6. Oculoplastics and Orbital Consultants, West Palm Beach, Florida. 7. Department of Ophthalmology, Anschutz Medical Campus, University of Colorado, Denver, Colorado. 8. Department of Ophthalmology, Emory University, Atlanta, Georgia. 9. Department of Ophthalmology, Rutgers Biomedical and Health Sciences, Newark, New Jersey. 10. Department of Ophthalmology, Weill Cornell Medical College, New York, New York. 11. Department of Ophthalmology, University of Texas Southwestern Medical Center, Dallas, Texas. 12. Department of Ophthalmology, New York Eye and Ear Infirmary, New York, New York. 13. Department of Ophthalmology, Yale University School of Medicine, New Haven, Connecticut. 14. Department of Ophthalmology, University of Kansas Medical Center, Kansas City, Kansas. 15. Department of Ophthalmology, Franzco Macquarie University, Sydney, Australia. 16. Department of Ophthalmology, Albany Medical College, Albany, New York. 17. Department of Ophthalmology, Mount Sinai School of Medicine, New York, New York. 18. Ocular Oncology, Wills Eye Institute, Philadelphia, Pennsylvania. 19. Department of Ophthalmology, SUNY Downstate Medical Center, Brooklyn, New York; Texas Oculoplastic Consultants, Austin, Texas. Electronic address: shinder.roman@gmail.com.
Abstract
OBJECTIVE: Bilateral lacrimal gland (LG) disease is a unique presentation that can result from varied causes. We reviewed the diagnoses, clinical features, and outcomes of 97 patients with this entity. DESIGN: Case series. PARTICIPANTS: Ninety-seven patients with bilateral LG disease. METHODS: Retrospective review and statistical analysis using analysis of variance and the Fisher exact test. MAIN OUTCOME MEASURES: Patient demographics, clinical features, diagnostic testing, diagnosis, and treatment. RESULTS: Patient age ranging from 8 to 84 years (mean, 46 years). The predominant gender was female (77%), and race included black (49%), white (38%), and Hispanic (12%) patients. Diagnoses fell into 4 categories: inflammatory (n = 51; 53%), structural (n = 20; 21%), lymphoproliferative (n = 19; 20%), and uncommon (n = 7; 7%) entities. The most common diagnoses included idiopathic orbital inflammation (IOI; n = 29; 30%), sarcoidosis (n = 19; 20%), prolapsed LG (n = 15; 15%), lymphoma (n = 11; 11%), lymphoid hyperplasia (n = 8; 8%), and dacryops (n = 5; 5%). Inflammatory conditions were more likely in younger patients (P<0.05) and in those with pain (P<0.001) and mechanical blepharoptosis (P<0.01) at presentation, whereas lymphoma was more common in older patients (P<0.001) without active signs of inflammation at presentation. Black patients were more likely to have sarcoidosis (P<0.01). Laboratory results showed high angiotensin converting enzyme level being significantly more likely in patients with sarcoidosis (P<0.05). However, sensitivity was limited to 45%, with 25% of patients diagnosed with IOI also demonstrating positive results. Corticosteroid therapy was the treatment of choice in 38 cases, corresponding to resolution of symptoms in 29% and improvement in an additional 32%. Overall, chronic underlying disease was found in 71% of patients, among whom 26% achieved a disease-free state, whereas 3% succumbed to their underlying disease. CONCLUSIONS: The cause of bilateral lacrimal gland disease most commonly was inflammatory, followed by structural and lymphoproliferative. Patient characteristics and clinical presentations were key features distinguishing between competing possibilities. Despite local control with corticosteroids or radiotherapy, underlying disease continued in 71% of patients and led to death in 3%.
OBJECTIVE: Bilateral lacrimal gland (LG) disease is a unique presentation that can result from varied causes. We reviewed the diagnoses, clinical features, and outcomes of 97 patients with this entity. DESIGN: Case series. PARTICIPANTS: Ninety-seven patients with bilateral LG disease. METHODS: Retrospective review and statistical analysis using analysis of variance and the Fisher exact test. MAIN OUTCOME MEASURES: Patient demographics, clinical features, diagnostic testing, diagnosis, and treatment. RESULTS:Patient age ranging from 8 to 84 years (mean, 46 years). The predominant gender was female (77%), and race included black (49%), white (38%), and Hispanic (12%) patients. Diagnoses fell into 4 categories: inflammatory (n = 51; 53%), structural (n = 20; 21%), lymphoproliferative (n = 19; 20%), and uncommon (n = 7; 7%) entities. The most common diagnoses included idiopathic orbital inflammation (IOI; n = 29; 30%), sarcoidosis (n = 19; 20%), prolapsed LG (n = 15; 15%), lymphoma (n = 11; 11%), lymphoid hyperplasia (n = 8; 8%), and dacryops (n = 5; 5%). Inflammatory conditions were more likely in younger patients (P<0.05) and in those with pain (P<0.001) and mechanical blepharoptosis (P<0.01) at presentation, whereas lymphoma was more common in older patients (P<0.001) without active signs of inflammation at presentation. Black patients were more likely to have sarcoidosis (P<0.01). Laboratory results showed high angiotensin converting enzyme level being significantly more likely in patients with sarcoidosis (P<0.05). However, sensitivity was limited to 45%, with 25% of patients diagnosed with IOI also demonstrating positive results. Corticosteroid therapy was the treatment of choice in 38 cases, corresponding to resolution of symptoms in 29% and improvement in an additional 32%. Overall, chronic underlying disease was found in 71% of patients, among whom 26% achieved a disease-free state, whereas 3% succumbed to their underlying disease. CONCLUSIONS: The cause of bilateral lacrimal gland disease most commonly was inflammatory, followed by structural and lymphoproliferative. Patient characteristics and clinical presentations were key features distinguishing between competing possibilities. Despite local control with corticosteroids or radiotherapy, underlying disease continued in 71% of patients and led to death in 3%.
Authors: Ilse Mombaerts; Jurij R Bilyk; Geoffrey E Rose; Alan A McNab; Aaron Fay; Peter J Dolman; Richard C Allen; Martin H Devoto; Gerald J Harris Journal: JAMA Ophthalmol Date: 2017-07-01 Impact factor: 7.389
Authors: Min Joung Lee; Bronwyn E Hamilton; David Pettersson; Kimberly Ogle; Jennifer Murdock; Roger A Dailey; John D Ng; Eric A Steele; Rohan Verma; Stephen R Planck; Tammy M Martin; Dongseok Choi; James T Rosenbaum Journal: Sci Rep Date: 2020-12-14 Impact factor: 4.379