Oculoplastics

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Knezevic A, Yoon MK. Differences in Wait Times for Cosmetic Blepharoplasty by ASOPRS Members. Ophthalmic Plast Reconstr Surg 2018;34(3):222-224.Abstract
PURPOSE: Adequate access to subspecialty care is of concern to patients and physicians alike. One measure of availability is the wait time for cosmetic procedures. The authors investigated geographical differences in wait times for cosmetic upper eyelid blepharoplasty of American Society of Ophthalmic Plastic and Reconstructive Surgery members across the country. METHODS: This study surveyed all 533 American Society of Ophthalmic Plastic and Reconstructive Surgery members' practices in the United States based on the publically available contact information (www.asoprs.org). Scripted telephone calls were made requesting self-referred cosmetic upper eyelid blepharoplasty. Wait times until the first available appointment and time until the first available surgery date were collected. RESULTS: Of the membership, 387 (72.6% response rate) respondents offered appointments for cosmetic upper eyelid blepharoplasty. Overall, 84.2% of respondents were male. Practice breakdown was 83.4% in private practice and 16.5% in academic practice. Median wait time until the next available appointment was 14 days (mean 21.2 days, 0-205 days; p = 0.145). Private practice wait time was shorter than academic (median 14 vs. 18 days, mean 19.7 vs. 28.9 days; p =0.004). However, there was wide variability based on region. CONCLUSIONS: Patients seeking cosmetic upper eyelid blepharoplasty have good access to care by American Society of Ophthalmic Plastic and Reconstructive Surgery members. There are variabilities based on academic versus private practice. Further study can evaluate whether similar findings exist for medically necessary functional procedures. This information may help assess the need for additional practitioners.
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Lee NG, Habib L, Hall J, Freitag SK. Simultaneous ipsilateral transconjunctival repair of upper and lower eyelid retraction in thyroid-associated ophthalmopathy. Orbit 2018;:1-6.Abstract
PURPOSE: To report a simple, highly effective technique of simultaneous transconjunctival repair of upper and lower eyelid retraction in patients with thyroid eye disease (TED). METHODS: A retrospective interventional case review was conducted on 22 eyes of 19 TED patients. The lower eyelid was recessed with placement of a tarsoconjunctival spacer graft harvested from the upper eyelid. The upper eyelid was then recessed through the conjunctival incision used to harvest the tarsal graft. A temporary tarsorrhaphy was placed for 5-7 days. The postoperative outcome was assessed by measuring the margin reflex distance of the upper eyelid (MRD1), inferior scleral show (ISS), and lagophthalmos. RESULTS: The absolute change in MRD1 ranged from 0 to 5 mm with an average of 1.86 ± 1.34 mm. The absolute change in ISS ranged from 0 to 2 mm with an average of 1.3 ± 0.49 mm. One patient had postoperative lagophthalmos and 17 of 19 had improvement in their ocular surface exposure symptoms. None of the patients' grafts were observed to undergo absorption during the postoperative course. CONCLUSIONS: This technique of harvesting a free tarsoconjunctival graft from the upper eyelid as a posterior spacer for the lower while simultaneously recessing the upper eyelid through the same incision is an effective and durable method of correcting eyelid retraction in TED.
Lefebvre DR, Freitag SK. Update on imaging of the lacrimal drainage system. Semin Ophthalmol 2012;27(5-6):175-86.Abstract
Epiphora is a common problem seen by the ophthalmologist. There are numerous etiologies of a watering eye, and the underlying diagnosis is not always clear. A variety of in-office examination techniques and procedures exist to aid with diagnosis and determination of appropriate therapy, but sometimes the diagnosis remains elusive, or an instituted therapy fails. Lacrimal imaging, particularly in these cases, can be helpful in assessing the function and anatomy of the lacrimal drainage system. This review serves to examine the literature of the last 10 years concerning imaging of the lacrimal drainage system.
Lefebvre DR, Mandeville JT, Yonekawa Y, Arroyo JG, Torun N, Freitag SK. A Case Series and Review of Bisphosphonate-associated Orbital Inflammation. Ocul Immunol Inflamm 2016;24(2):134-9.Abstract
PURPOSE: To report the largest series of new cases to date of bisphosphate-associated orbital inflammation. METHODS: A retrospective case review of patients with orbital inflammation following treatment with systemic bisphosphonate. RESULTS: Six patients over an 18-month period (2 males, 4 females) with an average age of 62.2 years had onset of orbital inflammatory symptoms 1-11 days after intravenous bisphosphonate infusion or, in 1 case, 4 weeks after initiation of oral bisphosphonate therapy. Imaging revealed diffuse orbital involvement in 3 cases, isolated lateral rectus muscle involvement in 2 cases, and superior rectus-levator involvement in 1 case. Two patients' symptoms resolved spontaneously within 2 weeks, and 3 responded rapidly and completely to corticosteroid therapy. The 1 patient on oral bisphosphonate had a slower but complete response to corticosteroid treatment. CONCLUSION: Clinicians should be aware of the association between acute orbital inflammation and recent treatment with systemic bisphosphonate medication.
Lefebvre DR, Reinshagen KL, Yoon MK, Stone JH, Stagner AM. Case 39-2018: An 18-Year-Old Man with Diplopia and Proptosis of the Left Eye. N Engl J Med 2018;379(25):2452-2461.
Li Z, Cestari DM, Fortin E. Thyroid eye disease: what is new to know?. Curr Opin Ophthalmol 2018;29(6):528-534.Abstract
PURPOSE OF REVIEW: The pathophysiology of thyroid eye disease (TED) is still not fully understood. However, recently described risk factors and molecular findings have brought new insights into the mechanisms of TED and could lead to the emerging use of more targeted therapies. This article aims to review the clinical findings of TED, and the most recent advances in our understanding of the risk factors and therapeutic options for TED. RECENT FINDINGS: Smoking has been recently shown to have an impact on specific gene expression involved in several disease-related pathways, which seems to be reversible with smoking cessation. This finding further emphasizes the importance of smoking cessation in the prevention and treatment of TED. Selenium deficiency and high-serum cholesterol have been described to be potential independent risk factors for TED and their management could decrease the incidence and severity of TED. In terms of therapeutic options, immunomodulatory medications have shown some promising results for disease control in TED over the past years, but further randomized prospective studies with larger sample sizes are still needed to prove their efficacy. A new technique of P brachytherapy was shown to have quick therapeutic effects on TED without significant side effects and could be a promising therapy for selected cases of TED. SUMMARY: TED is one of the most common autoimmune inflammatory disorders of the orbit. Although its pathophysiology remains unclear, newly described genetic findings and risk factors could help in explaining its occurrence and guide future therapies. Immunosuppressant medications are increasingly used in the management of TED, but further studies are needed to confirm their effectiveness.
Lin LY, Stagner AM, Freitag SK. Minocycline-induced Conjunctival Hyperpigmentation. Ophthalmic Plast Reconstr Surg 2023;39(4):e133.
Lin LY, Azad AD, Chiou CA, Kozanno L, Stemmer-Rachamimov A, Stone J, Lee NG. Antisynthetase Syndrome Causing Necrotizing Myositis Involving Extraocular Muscles. Ophthalmic Plast Reconstr Surg 2024;Abstract
The authors describe the clinical, histologic, and serologic findings of a patient with necrotizing myositis of the extraocular muscles in the setting of antisynthetase syndrome, as well as subsequent management. This is the first case in the literature of a systemic necrotizing myositis to have associated ophthalmic findings.
Lin LY, Stone JH, Liou VD, Stagner AM, Lee GN. Eosinophilic Angiocentric Fibrosis of the Orbit: A Clinicopathologic Review of 6 Novel Cases With Review of the Literature. Am J Ophthalmol 2023;256:9-19.Abstract
PURPOSE: To describe 6 cases and review the current state of knowledge of eosinophilic angiocentric fibrosis (EAF) involving the orbit. DESIGN: Retrospective clinicopathologic case series and review of the current literature METHODS: Clinical records and histopathologic data of orbit-involving EAF were gathered between 2004 and 2022 from a single academic institution. The patients' presenting clinical symptoms and signs, laboratory data, radiographic studies, and management documentation were collected. RESULTS: Retrospective review identified 6 novel cases, totaling 31 cases of EAF involving the orbit described as of this writing. Fourteen patients were male, and the average age of presentation was 49.8 years (range 25-78 years). Eighteen patients had concurrent sinonasal involvement, whereas 13 had primary orbital involvement. The median duration of symptoms prior to evaluation was 24 months, with nasal symptoms, proptosis, periorbital swelling, and pain being the most common presenting symptoms. The majority of patients underwent surgical debulking, as well as treatment with glucocorticoids and steroid-sparing agents, such as rituximab, with varied results. CONCLUSION: EAF involving the orbit is uncommon. The histopathologic findings include a perivascular, eosinophil-rich infiltrate and a pauci-inflammatory storiform type of fibrosis concentrated around small vessels. Orbital involvement usually results from local extension from adjacent sinuses, but primary orbital involvement has been described. Surgical debulking and immunosuppressive agents such as rituximab have been shown to stabilize disease.
Liou VD, Yoon MK. Advances in steroid sparing medical management of active thyroid eye disease. Semin Ophthalmol 2020;:1-8.Abstract
Thyroid eye disease (TED) is an autoimmune inflammatory disease of the orbit and the most common extrathyroidal manifestation of Graves disease. The release of pro-inflammatory cytokines is associated with inflammation of the ocular surface and lacrimal gland along with periorbital skin erythema and edema. Resultant tissue remodeling, fibrosis, and fat deposition can impart permanent physical changes to the ocular adnexa with effects on function and cosmesis. These changes occur in the active phase of disease, and it is during this time that steroids are often relied on to help alleviate symptoms. Due to the common and predictable side effects of long-term and high-dose steroid use, there has been a continuous effort to find alternative steroid-sparing medical management options for TED. This review highlights the various research studies that support the use of these medications.
Liou VD, Yoon MK, Maher M, Chwalisz BK. Orbital Inflammation in Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease: A Case Report and Review of the Literature. J Neuroophthalmol 2022;Abstract
BACKGROUND: To present 2 patients with myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease with unilateral orbital inflammation, optic nerve head edema, and abnormalities of the optic nerve and nerve sheath on imaging. We review the most current literature on this important and uncommon clinical phenotype. METHODS: A case report of 2 patients and a comprehensive review of the relevant literature on orbital inflammation in MOG antibody-associated disease (MOG-AD). RESULTS: Two patients presented with decreased vision and unilateral orbital inflammation. Both had optic nerve head edema and abnormalities of the optic nerve and nerve sheath on imaging. The patients were treated with immunosuppressants and had improvement of vision changes as well as their orbital inflammatory signs. MOG antibody was positive in high titers in both patients. Only 3 other cases of orbital inflammation associated with MOG antibody have been described. In all cases, orbital signs responded rapidly to intravenous methylprednisolone, but the improvement in visual acuity was variable and less robust. CONCLUSION: Orbital inflammation is a unique and underrecognized phenotype of MOG-AD with only a few reports in the literature. In patients who present with vision loss and orbital inflammation, MOG-AD should be considered in the differential.
Liou V, Yoon M. Comparative Incidence of Periocular Surgical Site Infections with Increased Surgical Mask Use during the COVID-19 Pandemic. Ocul Immunol Inflamm 2021;:1-6.Abstract
PURPOSE: To evaluate the effect of surgical mask use on infection rates for office-based periocular surgeries during the pandemic. METHODS: An Institutional Review Board-approved retrospective review of medical records identified patients who had an office-based oculofacial plastic surgery procedure during the pandemic between March and December 2020. Statistical analysis was used to compare this group to patients that underwent procedures between March and December 2019, prior to the pandemic when neither surgeon nor patient wore a surgical mask. RESULTS: The study consisted of 680 patients. Thirty-one different types of procedures were encountered. The incidence of infections in 2020 compared to 2019 was not statistically significant (1.12% (n = 3) versus 1.21% (n = 5), p = 1). All patients with infections were treated with oral antibiotics and improved without long-term complications. CONCLUSIONS: Periocular surgical site infections are uncommon, and the wearing of surgical masks by patient and surgeon during our office-based oculofacial procedures did not change the incidence of SSIs.
Lu JE, Yoon MK. The Role of Steroids for Pediatric Orbital Cellulitis - Review of the Controversy. Semin Ophthalmol 2023;:1-4.Abstract
Orbital cellulitis in the pediatric population is treated primarily with antibiotic therapy. This leaves the inflammatory component unchecked. Corticosteroid therapy has been used to accelerate recovery and decrease the long-term morbidity in other infectious conditions. Its use has also been proposed for pediatric orbital cellulitis. The aim of this manuscript is to conduct a literature review to summarize existing evidence and understand ongoing controversies. Overall, prior investigations on corticosteroid therapy for pediatric orbital cellulitis are limited by their study design and sample sizes. One of the most discussed potential benefits is that adjuvant steroid therapy for pediatric orbital cellulitis is associated with shorter hospitalization without major infectious complications. However, decreased hospitalization length is an imperfect metric, especially without standardized criteria for hospital discharge. Future studies are warranted to better guide the use of adjuvant steroid therapy and to optimize its potential in the management of pediatric orbital cellulitis.
Lu JE, Wolkow N, Lee GN, Lefebvre DR, Freitag SK, Yoon MK. Perceived change in age after functional upper blepharoplasty. Orbit 2023;:1-3.Abstract
PURPOSE: To evaluate the perceived age of patients before and after functional upper blepharoplasty. METHODS: Retrospective chart review of patients who underwent upper blepharoplasty by a single surgeon at an academic center. The inclusion criterion was having external photographs before and after blepharoplasty. Exclusion criteria included any other concurrent eyelid or facial surgery. Primary endpoint: perceived change in age after surgery as judged by the American Society of Ophthalmic Plastic & Reconstructive Surgery (ASOPRS) surgeons. RESULTS: Sixty-seven patients (14 men, 53 women) were included. Mean pre-operative age was 66.9 years (range 37.8-89.4) and mean post-operative age was 67.4 years (range 38.6-89). The mean perceived age pre-operatively was 68.9 years, and the mean perceived age post-operatively was 67.1 years, a change of 1.8 years (p = 0.0001 by two-tailed paired T-test). Inter-rater reliability of the observers was measured by intraclass correlation coefficient of 0.77 for pre-operative and 0.75 for post-operative photos. The decreased perceived age was 1.9 years for women, 1.4 years for men, 0.3 years for Asians, 1.2 years for Hispanics, and 2.1 years for whites. DISCUSSION: Functional upper blepharoplasty by an experienced ASOPRS surgeon was shown to reduce the perceived age of a patient by an average of 1.8 years.
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Ma L, Jakobiec FA, Wolkow N, Dryja TP, Borodic GE. Multiple Eyelid Cysts (Apocrine and Eccrine Hidrocystomas, Trichilemmal Cyst, and Hybrid Cyst) in a Patient With a Prolactinoma. Ophthalmic Plast Reconstr Surg 2018;34(3):e83-e85.Abstract
A 53-year-old man presented with smooth-domed, variegated cysts (polycystic disease) of all 4 eyelids, worse on the left side. Some of the cysts were clear, while others were creamy-white colored. In addition, multiple, very fine vesicopapules were noted along the eyelid margins. Histopathologic examination revealed a trichilemmal cyst, several pure apocrine hidrocystomas displaying multiple chambers, a hybrid cyst, and many small eccrine cysts of the deep dermis. The apocrine lesions, including the small ones at the eyelid margins, predominated. Smooth muscle actin sometimes positively stained outer myoepithelial cells in some of the apocrine cysts, which helped to distinguish them from eccrine cysts. Most noteworthy was the fact that the patient had been diagnosed with a prolactinoma 20 years earlier. There is only 1 previous report of multiple apocrine cysts and an antecedent prolactinoma in the dermatologic literature. This syndrome should be separated from that of Schöpf-Schulz-Passarge, which manifests multiple small eyelid apocrine cysts and other ectodermal dysplasias without any association with neoplasia, and from that of focal dermal hypoplasia (Goltz-Gorlin) syndrome with apocrine cysts but again without neoplasia.
MacIntosh PW, Jakobiec FA, Stagner A, Rashid A, Sutula FC, Yoon MK, Fay AM. Failed Cartilaginous Grafts in the Eyelid: A Retrospective Clinicopathological Analysis of 5 Cases. Ophthal Plast Reconstr Surg 2016;32(5):347-53.Abstract

PURPOSE: To analyze the clinical and histopathologic features of 5 failed autologous cartilaginous grafts to the lower eyelids and to analyze the reasons for these failures. METHODS: In this retrospective case series, the data collected included patient ages, reasons for and duration of cartilaginous graft implants, sources of cartilaginous grafts, and clinical and histopathologic findings at time of graft removal using hematoxylin and eosin, elastic, Alcian blue, and Masson trichrome staining for analysis of tissue alterations. RESULTS: Five cartilaginous, posterior lamellar lower eyelid grafts were complicated by eyelid thickening or retraction, graft extrusion, and entropion. Histopathologic findings included segmentation of the original single implant, stripped of its perichondrium, due to "kerfing," sometimes with overlapping of the segments and scar formation between the segments. In place of the perichondrium that had been removed during the preparation the graft implants, a fibrous pseudoperichondrial capsule had formed. Pyknotic nuclei in varying degrees were typically found in the center of the grafts, despite a high degree of preservation of the extracellular matrix (collagenous, elastic, and proteoglycan components). No evidence of inflammation, cartilaginous vascularization, or necrosis was identified in any graft. CONCLUSION: Despite minimal reactive processes, kerfing (partial thickness cuts made in the graft to increase its pliancy) may be partially responsible for graft migration, deformation, and surgical failure. The consequences were graft fragmentation and overlapping of the multiple fragments. Graft migration can be exacerbated if a posterior lamellar graft is used to correct an anterior lamellar deficiency. Interference with the overall architectural integrity of the graft and its extracellular matrix appears to play no role in failure, despite removal of the perichondrium. Mild to moderate degrees of chondrocytic dropout in the absence of necrosis and inflammation are probably attributable to the thick and coarsely textured collagen of the fibrous pseudoperichondrial capsule that may impede diffusion of nutrients into the center of the graft.

McCoskey M, Neerukonda VK, Hatton MP, Wolkow N. Eccrine poroma of the eyelid. Orbit 2021;:1.Abstract
Clinical and histopathologic case of an eyelid eccrine poroma, a benign adnexal neoplasm rarely found on the periorbital skin.
Mendoza PR, Jakobiec FA, Townsend DJ. Bilateral nevus comedonicus of the eyelids. Ophthalmic Plast Reconstr Surg 2013;29(4):e95-8.Abstract
Nevus comedonicus is a rare developmental abnormality of the infundibulum of the hair follicle. It is usually unilateral and commonly presents at birth or during childhood. A rare case of late-onset, bilateral nevus comedonicus of the eyelids is reported. A 79-year-old man presented with asymptomatic but disfiguring eyelid lesions noted several months earlier. On physical examination, multiple papules resembling comedones were present bilaterally in the eyelids, canthi, temple regions, and bridge of the nose. Microscopically, there were deep invaginations of the follicular canals forming focal tunnels or pseudosinus tracts with poral openings to the surface. These variably cystic structures were lined by keratinizing and nonkeratinizing squamous epithelium, contained concentric lamellae of keratin in their lumens, and some were acutely or chronically inflamed. The diagnosis of a nevus comedonicus was made. The clinical and histopathologic characteristics, pathogenesis, differential diagnosis, and management of nevus comedonicus are briefly discussed.
Milman T, Jakobiec FA, Lally SE, Shields JA, Shields CL, Eagle RC. Lacrimal Gland Hamartoma (Formerly Termed Dacryoadenoma). Am J Ophthalmol 2020;217:189-197.Abstract
PURPOSE: Since the original description of "dacryadenoma" by Jakobiec and associates, the data on this unusual epibulbar lacrimal gland lesion remain sparse. The aim of this study was to characterize clinically, morphologically, and immunohistochemically this isolated epibulbar lacrimal gland lesion. DESIGN: Retrospective observational case series. METHODS: Institutional pathology records between 2000 and 2019 were searched for all cases of isolated epibulbar lacrimal gland lesions. Tissue from 3 normal lacrimal glands and 1 complex choristoma were included for comparative analysis. Clinical, histopathologic, and immunohistochemical findings were recorded. RESULTS: Four patients with isolated epibulbar lacrimal gland lesions, 2 male and 2 female, with a median age of 18 years (range, 12-57) were identified. All patients presented with recent onset of unilateral pink-to-orange, well-circumscribed subepithelial juxtaforniceal (3/4, 75%), or nasal (1/4, 25%) bulbar conjunctival nodules, which were asymptomatic (3/4, 75%) or associated with foreign body sensation (1/4, 25%). When compared with the normal lacrimal gland and complex choristoma, all isolated epibulbar lacrimal gland lesions were composed predominantly of variably dilated, branching tubular structures with pseudo-apocrine snouts, and either totally absent (2/2, 50%) or rare (2/2, 50%) ducts and rare acinar zymogen granules (3/4, 75%). CONCLUSION: Our study confirms that a subset of isolated epibulbar lacrimal gland lesions differs morphologically and immunohistochemically from normal lacrimal gland tissue and the lacrimal gland in a complex choristoma. These differences range from subtle to overt, suggesting that isolated epibulbar lacrimal gland lesions may have originated from precursor cellular elements indigenous to the conjunctiva (hamartia) and grew into disorganized lacrimal gland tissue.
Mombaerts I, Bilyk JR, Rose GE, McNab AA, Fay A, Dolman PJ, Allen RC, Devoto MH, Harris GJ, of the Society EPO. Consensus on Diagnostic Criteria of Idiopathic Orbital Inflammation Using a Modified Delphi Approach. JAMA Ophthalmol 2017;Abstract
Importance: Current practice to diagnose idiopathic orbital inflammation (IOI) is inconsistent, leading to frequent misdiagnosis of other orbital entities, including cancer. By specifying criteria, diagnosis of orbital inflammation will be improved. Objective: To define a set of criteria specific for the diagnosis of IOI. Design, Setting, and Participants: A 3-round modified Delphi process with an expert panel was conducted from June 8, 2015, to January 25, 2016. Fifty-three orbital scientist experts, identified through membership in the Orbital Society, were invited to participate in on online survey and they scored, using 5-point Likert scales, items that are eligible as diagnostic criteria from the literature and from personal experience. The items were clustered around the anatomic subtypes of IOI: idiopathic dacryoadenitis and idiopathic orbital fat inflammation (2 nonmyositic IOIs), and idiopathic orbital myositis (myositic IOI). Items with dissensus were rescored in the second round, and all items with consensus (median, ≥4; interquartile range, ≤1) were ranked by importance in the third round. Main Outcomes and Measures: Consensus on items to be included in the criteria. Results: Of the 53 experts invited to participate, a multinational panel of 35 (66%) individuals with a mean (SD) years of experience of 31 (11) years were included. Consensus was achieved on 7 of 14 clinical and radiologic items and 5 of 7 pathologic items related to diagnosis of nonmyositic IOI, and 11 of 14 clinical and radiologic items and 1 of 5 pathologic items for myositic IOI. There was agreement among panelists to focus on surgical tissue biopsy results in the diagnosis of nonmyositic IOI and on a trial with systemic corticosteroids in myositic IOI. Panelists agreed that a maximum number of 30 IgG4-positive plasma cells per high-power field in the orbital tissue is compatible with the diagnosis of IOI. Conclusions and Relevance: An international panel of experts endorsed consensus diagnostic criteria of IOI. These criteria define a level of exclusion suggested for diagnosis and include tissue biopsy for lesions not confined to the extraocular muscles. This consensus is a step toward developing guidelines for the management of IOI, which needs to be followed by validation studies of the criteria.

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