Hemorrhage Following Muller's Muscle Conjunctival Resection: Description and Case-Control Study.

PURPOSE: The purpose of this study was to report a series of patients with postoperative hemorrhage after Muller's muscle conjunctival resection surgery and compare risk factors and outcomes with a control population. METHODS: In this case-control study, records of patients who underwent Muller's muscle conjunctival resection over 5 years were reviewed for a history of postoperative hemorrhage occurring >24 hours after surgery. A 4:1 control population was matched for age and sex. Clinical data collected included demographics, medical history, medications, and subsequent surgery. Preoperative and 3-month postoperative marginal reflex distance 1 were measured digitally using ImageJ. The hemorrhage and control groups were compared using Fisher's exact tests for categorical variables and independent samples t tests for continuous variables. RESULTS: The hemorrhage group contained 10 patients (mean age 66.4 ± 18.5 years). The control group consisted of 40 age and sex-matched controls. Of 350 charts reviewed, there were 10 cases of postoperative hemorrhage (incidence 2.9%). Hemorrhage occurred a mean of 4.2 ± 1.3 (range 2-7) days after surgery and lasted for a mean of 29.3 ± 19.1 (range 12-72) hours. In all 10 cases, the bleeding resolved with conservative measures. There was no difference between the hemorrhage and control groups in terms of medical conditions, blood thinners, and surgical revision. Preoperative, postoperative, and change in marginal reflex distance 1 did not differ between the hemorrhage and control groups. CONCLUSIONS: Hemorrhage occurs approximately 4-5 days postoperatively in a small percentage of patients undergoing Muller's muscle conjunctival resection surgery. This investigation did not identify any consistent risk factors, and outcomes in this patient population appear no different than controls.

A Minimally Invasive Endoscopic Transnasal and Transorbital Approach to Sinonasal Tumor Resection.

A 35-year-old female with an extensive biphenotypic sinonasal sarcoma (BSNS) eroding through the left frontal sinus into the orbit required gross total resection of the tumor. We wanted to forgo an open approach and provide a less invasive alternative via combined endonasal and transorbital endoscopic techniques to allow for tumor removal through small, well-disguised incisions. We utilized three portals-nasal, transorbital, and anterior table window-to create interconnected orbit-sinonasal corridors, thus generating alternate pathways for visualization and manipulation of this extensive tumor. Laryngoscope, 134:1308-1312, 2024.

Extrascleral extension of choroidal melanoma after iodine-125 brachytherapy treatment: a case series.

BACKGROUND/OBJECTIVES: The purpose of this study is to report cases of choroidal melanoma that developed extrascleral tumour recurrence after treatment with iodine-125 brachytherapy. SUBJECTS/METHODS: In this single-institution retrospective observational case series, all instances of biopsy-confirmed orbital melanoma after known intraocular melanoma were reviewed. Tumour characteristics, clinical course, time to recurrence, cytogenetics of initial tumour and recurrence, and presence of intraocular recurrence were documented. RESULTS: Five cases of orbital melanoma following treatment with plaque radiotherapy are described. Tumour staging was Ia (1), IIa (2), and IIb (2). The primary lesion in four of the five appeared to have undergone complete regression for an average of 2 years, with the orbital melanoma developing after this interval. Recurrence of the intraocular tumour was seen in conjunction with an extrascleral component in two cases. Four cases ultimately underwent enucleation or exenteration; three had evidence of direct extension of tumour through the sclera. Four cases in this series had molecular characteristics associated with high metastatic risk (three patients with monosomy 3, one with BAP1 mutation). CONCLUSIONS: High-risk tumour biology may predispose to late appearance of extrascleral melanoma despite optimal treatment and adequate control of the intraocular tumour. Extended follow-up with detailed orbital examination and imaging is recommended for this population.

Systemic eosinophilic disease presenting as dacryoadenitis.

Eosinophilic disease with orbital involvement is rare. We present two patients with dacryoadenitis associated with local and systemic eosinophilia. A 32-year-old man presented with episodic dacryoadenitis, lower respiratory inflammation and peripheral eosinophilia. Lung and lacrimal gland biopsies demonstrated eosinophilic infiltrate without granuloma, necrosis, or vasculitis. He improved with oral corticosteroids and Mepolizumab, an IL-5 inhibitor. The second case involved a 33-year-old man who similarly presented with episodic dacryoadenitis, pulmonary inflammation and pain/swelling in the hands and feet. Lacrimal gland biopsy demonstrated a predominantly eosinophilic infiltrate without granuloma or vasculitis. Symptoms improved with oral corticosteroids. Although neither patient was provided a definitive diagnosis, both were determined to have an eosinophilic condition on the spectrum of eosinophilic asthma or eosinophilic granulomatosis with polyangiitis.

External carotid artery to ophthalmic artery flow associated with internal carotid artery stenosis.

PURPOSE: One of the most devastating complications of facial filler injection is sudden ischemic blindness. However, its mechanisms and predisposing factors are poorly understood. The purpose of this study was to investigate the prevalence of external carotid artery (ECA) to ophthalmic artery (OA) anterograde flow in patients with internal carotid artery (ICA) stenosis and in a control population without carotid disease. METHODS: In this cross-sectional cohort study, two groups of patients who underwent catheter cerebral angiography over a 5-year period were identified: patients with symptomatic ICA stenosis and a control group of patients with refractory epistaxis undergoing embolization. Angiograms were reviewed by an interventional neuroradiologist. The primary outcome measure was the presence of ECA to OA flow, defined as choroidal blush before filling of the circle of Willis. Secondary outcome measures included the percentage and location of ICA stenosis and ECA anastomotic branches involved. RESULTS: The study included 149 patients with ICA stenosis and 69 control patients. ECA to OA flow was more prevalent in patients with ICA stenosis (34.9%) compared to controls (2.9%) (p < .001). Logistic regression demonstrated that for each 10% increase in ICA stenosis over 70%, there was 2.8 times increased risk of ECA to OA flow (p < .001). CONCLUSIONS: ECA to ICA anterograde flow can be demonstrated in approximately 3% of control patients and in over 1/3 of patients with symptomatic carotid stenosis. This provides a plausible pathway for small filler particles to pass with blood flow from the facial to the ophthalmic circulation.

Combined Endonasal and Orbital Approach for Resection of Orbital Apical Tumors.

PURPOSE: Tumors of the medial orbital apex can be challenging to resect. Primary endonasal resection can be limited by requirements for fat retraction and poor angulation for lateral orbital dissection. Orbital resection alone can have space limitations and a limited view for dissection of the posterior aspect of the tumor. The combined transorbital transnasal approach integrates the retraction and instrumentation advantages of the transorbital approach with the optical and space advantages of the transnasal approach. METHODS: In this cross-sectional cohort study, patients who underwent surgical resection of an apical orbital tumor via a combined endoscopic and orbital approach from 2014 to 2019 were identified. Clinical history, examination findings, imaging, surgical technique, histopathology, and complications were reviewed. RESULTS: Six patients were included. Patients presented with proptosis (5), optic neuropathy (3), and motility restriction (2). Surgery involved a medial orbitotomy and endoscopic ethmoidectomy ± sphenoidotomy, medial wall removal, and periosteum opening. The lesion was prolapsed into the sinus, dissected at the apex via the orbital approach, and removed through the nasal cavity. Pathologic diagnoses were cavernous venous malformation (4), venolymphatic malformation (1), and metastatic carcinoid (1). Improvement in pain (2/2), optic neuropathy (2/3), and proptosis (5/5) were noted. Complications included ethmoid mucocele and nasal polyps. CONCLUSIONS: The combined endonasal and orbital approach involves prolapsing the lesion into the nasal cavity to aid in dissection on the lateral and posterior aspects, maximizing visibility of the tight intraconal space. This technique can be performed by a team of orbital surgeons experienced in both orbital and endonasal surgery.

Radiographic analysis of fatty infiltration of the extraocular muscles in thyroid eye disease.

PURPOSE: Fatty infiltration of the extraocular muscles has been described radiographically in patients with thyroid eye disease (TED), yet it has not been studied on a large scale nor quantified. Our purpose was to define and characterize this entity in patients with TED. METHODS: An IRB-approved cross-sectional retrospective review of medical records identified patients with a clinical diagnosis of TED and at least one CT of the orbits. A 2:1 age and sex-matched control population consisted of patients without a history nor radiographic evidence of orbital disease or systemic thyroid abnormality. The presence of fatty infiltration in each extraocular rectus muscle was defined using Hounsfield units (HU). Laterality, muscles involved, and pattern of fatty infiltration were also evaluated. Student's t-tests, Chi-square, and Fisher's exact tests were used to compare TED and control groups. RESULTS: The study population consisted of 252 patients with TED and 504 age and sex-matched controls. Fatty infiltration was significantly more prevalent in TED patients (36/252, 14.3%) compared to controls (11/504, 2.2%) (p < .001). The mean density of fat infiltration was significantly lower in TED patients (-40.4 HU) than controls (-34.8 HU) (p = .048). In TED patients, the frequency of muscle involvement was inferior rectus (61.8%), lateral rectus (19.7%), superior rectus (11.8%) and medial rectus (6.6%), which was not significantly different than controls (p > .05). Most muscles (88.2%) in the TED group exhibited a heterogeneous pattern of infiltration, which did not differ from controls (p = .34). CONCLUSIONS: This study characterizes fatty infiltration of the extraocular muscles in patients with TED.

Transorbital Debulking of Sphenoid Wing Meningioma.

OBJECTIVE: To describe the clinical features and outcomes of patients who underwent transorbital debulking of sphenoid wing meningioma. METHODS: Patients with a diagnosis of sphenoid wing meningioma who underwent transorbital debulking were included in this series. Preoperative and postoperative symptoms and examination findings, including best corrected visual acuity (BCVA) and proptosis were extracted from patient charts. All imaging studies, records of additional surgical and medical management, and complications of surgery were collated. RESULTS: Eight patients were included. The most common symptoms at presentation were blurred vision (6/8) and proptosis (6/8). The most common clinical findings at presentation were decreased visual acuity and proptosis. Mean BCVA preoperatively was 0.93 in logMARunits andmeanrelative proptosis preoperatively was 4.88 mm. All patients underwent orbitotomy with or without bone flap with decompression of hyperostotic bone and subtotal resection of soft tissue mass. Mean follow-up time was 14months. Five of eight patients experienced postoperative improvement in BCVA, for mean change of 0.32. All patients demonstrated reduction in proptosis postoperatively with a mean reduction of 3.63 mm. CONCLUSIONS: Sphenoid wing meningioma can present with decreased visual acuity and/or proptosis. It is possible to address both of these problems in selected patients with transorbital debulking, an approach that avoids the aesthetic and functional consequences of craniotomy. The aim of this technique is not surgical cure, but rather improvement in vision and disfigurement.

Solitary Extramedullary Plasmacytoma of the Lacrimal Sac With Associated Crystal-Storing Histiocytosis.

PURPOSE: To report a rare case of crystal-storing histiocytosis associated with solitary extramedullary plasmacytoma of the lacrimal sac and to review literature on the 2 entities to summarize important diagnostic, management, and prognostic considerations. METHODS: A case report of the ophthalmologic presentation, pathology workup, and oncologic management is presented. Literature search with focus on lesions occurring in ophthalmic sites and management guidelines from expert panels and working groups. RESULTS: Crystal-storing histiocytosis associated with solitary extramedullary plasmacytoma arose within the lacrimal sac of a previously healthy middle-aged woman and presented as a painless nodule with epiphora. The biopsy tissue showed sheets of crystal-filled histiocytes, interspersed with monoclonal plasma cells and rarely demonstrated plasma cell phagocytosis. Imaging and laboratory studies confirmed the localized nature. CONCLUSIONS: Crystal-storing histiocytosis is an uncommon entity in which crystals, most commonly arising from altered immunoglobulins, aggregate within histiocytes and form symptomatic mass lesions. It has been reported in ophthalmic regions in patients with a concurrent lymphoproliferative or plasma cell disorder and can rarely predate a malignancy. The current case is notable because crystal-storing histiocytosis occurs with a localized process, solitary extramedullary plasmacytoma, and presents in an unusual site, the lacrimal sac. Tissue biopsy with multimodal pathological evaluation is necessary to make the diagnosis. Ophthalmologists should recognize that crystal-storing histiocytosis is commonly associated with a hematologic malignancy and, when appropriate, refer the patient for oncologic management. Surveillance may be indicated in cases with no established etiology. Solitary extramedullary plasmacytoma should also be monitored, as a proportion of cases progress to multiple myeloma.

Bilateral lacrimal gland disease: clinical features and outcomes.

BACKGROUND: Patients with bilateral lacrimal gland disease are a unique subset of patients where there is a paucity of literature. This presentation often represents systemic disease or malignancy and can cause diagnostic difficulties. We aim to describe the diagnoses and features of bilateral lacrimal gland disease. METHOD: Retrospective multi-centre case series involving 115 patients with bilateral lacrimal gland disease from 1995 to 2020. RESULTS: 115 patients were included. Their ages ranged from 9 to 85 (mean 47.3 years) with a female predominance (73, 63.5%). The most common category of diagnosis was inflammatory (69, 60%) followed by lymphoproliferative (23, 20%), structural (17, 14.8%) and other conditions (6, 5.2%). The five most common specific diagnoses were IgG4 related disease (20, 17.4%) and idiopathic orbital inflammatory disease (20, 17.4%), lymphoma (16, 13.9%), lacrimal gland prolapse (13, 11.3%), and sarcoidosis (11, 9.6%). Corticosteroid treatment was used most commonly (29, 25.2%) followed by observation (25, 21.7%). At last follow up, the majority of patients had complete resolution, significant improvement with mild residual disease or stable disease without further progression (104, 90.4%). CONCLUSION: Bilateral lacrimal disease may be due to a range of aetiologies, most of which are systemic. The most common are inflammatory and lymphoproliferative conditions. Due to the wide range of aetiologies of bilateral lacrimal gland disease, it is extremely difficult to accurately determine a cause based on clinical findings alone, highlighting the vital role of lacrimal gland biopsy in patients presenting with bilateral lacrimal gland disease.

Recurrence of Distensible Orbital Venous-dominant Venolymphatic Malformations After Sclerotherapy Versus Embolization With Excision.

PURPOSE: Treatment for orbital venolymphatic malformations (VLMs) commonly includes 3 major options: sclerotherapy, surgery, and embolization followed by surgical excision. Each has certain advantages, although it is not clear whether all are effective. The authors characterize the clinical course for a series of patients with distensible orbital venous-dominant VLM treated with sclerotherapy and/or embolization with excision. METHODS: In this cross-sectional cohort study, patients affected by distensible orbital venous-dominant VLM presenting to the orbital and ophthalmic plastic surgery service from 2014 to 2020 were identified. Patients were included if they presented with a moderate-flow, distensible venous-dominant malformation associated with Valsalva-related symptoms (e.g., pain, proptosis, and diplopia). RESULTS: Six cases were treated with sclerotherapy. Four underwent multiple treatments, with a mean ± SD of 3.5 ± 2.3 (range 1-7). All patients in this group failed to improve or experienced recurrence of symptoms after sclerotherapy. Twelve cases were treated with embolization and excision. Resolution of symptoms in all 12 cases was noted and maintained for a mean of 3.4 ± 2.1 years. There have been no cases of recurrence. Patients treated with sclerotherapy were more likely to experience recurrence of symptoms compared to those treated with embolization and excision (p < 0.001). CONCLUSIONS: Treatment of distensible venous-dominant moderate-flow orbital VLM with sclerotherapy may provide temporary improvement in some cases. However, in the medium to long term, recurrence was universal in this series. Embolization with excision appears to provide more definitive management, avoiding recurrence in all cases for a mean follow-up of 3 years.

The Effect of Lighting and Photograph Exposure on Perceived Attractiveness.

PURPOSE: To assess the effect of various lighting conditions and photograph exposures on perceived attractiveness. METHODS: In the first experiment, 5 variably exposed photographs were taken of 10 subjects using a consistent lighting condition (45° superior box light). In the second experiment, 10 subjects were photographed under variable lighting conditions with consistent exposure: 1) 90° overhead box light, 2) ring light, 3) 45° superior box light, 4) built-in camera flash, 5) 2 straight on box lights, each 45° from midline, and 6) natural light. Participants were instructed to maintain a neutral expression, were placed in front of a standardized blue-gray background, and were photographed during a single session. Photographs were imported into an online survey platform (Qualtrics 2020) and displayed in random order. Volunteer survey respondents were instructed to rate the subject's attractiveness on a scale of 0 to 10. Between the two experiments, a total of 22,000 scored photographs were included in the analysis. Mixed ANOVA and pairwise comparisons with Bonferroni correction were used to compare between- and within-subject ratings. RESULTS: Lighting condition had a significant impact on perceived attractiveness ( p < 0.001), with the 90° overhead box light achieving lower scores and the 45° superior box light yielding greater scores of attractiveness relative to the other conditions. Photograph exposure did not have a significant impact on subjective attractiveness ( p = 1.000). CONCLUSIONS: Our findings suggest that perceived attractiveness is enhanced when a 45° superior box light is used for illumination, and attractiveness is reduced when 90° overhead exposure is utilized. Exposure did not play a prominent role in perceived attractiveness.

Blepharoptosis Repair: External Versus Posterior Approach Surgery: Why I Select One over the Other.

Ptosis surgery is performed via an anterior/external or posterior/internal approach, primarily defined by the eyelid elevator muscle surgically addressed: the levator complex anteriorly or Muller muscle posteriorly. Posterior ptosis surgery via Muller muscle conjunctival resection is an excellent first choice for cases of mild to moderate ptosis with good levator function, as it is predictable, provides a reliable cosmetic outcome, requires no patient cooperation during surgery, portends a lower rate of reoperation, and rarely leads to lagophthalmos and/or eyelid retraction postoperatively. External levator resection is preferred in patients with severe ocular surface/cicatricial conjunctival disease, shortened fornices, and lesser levator function.

Clinical Assessment and Lesion-Specific Management of Orbital Vascular Malformations.

The systematic classification of vascular disease as proposed and refined by the International Society for the Study of Vascular Anomalies (ISSVA) divides vascular pathology first into tumors and malformations. Malformations are described as simple and complex, where simple malformations contain a single vascular system and complex malformations comprised of multiple vascular systems. Arteriovenous malformations are considered in terms of inflow characteristics which are primarily responsible for the key management challenges. Management utilizing endovascular embolization and/or surgical resection is often employed; however, recurrence can occur, particularly in diffuse cases. There may be an increasing role for systemic antiangiogenic therapy in such cases. Lymphaticovenous malformations are divided into the principle components on the lymphatic and venous sides for clarity of discussion. Lymphatic malformations are described morphologically as macrocystic and microcystic, and physiologically in terms of the processes responsible for growth. In both cases, surgical options are challenging and local therapeutics intended to close large luminal spaces in the case of macrocystic and to slow biological signaling for growth in microcystic. Venous malformations are described physiologically in terms of flow and distensibility, as volume plays a critical role in the limited space of the orbital cavity. Combined embolic-surgical approaches can be effective for management. More complicated, combined lesions can be managed by dividing the lesion into principal components and treating each appropriately.

Early Experience With the Clinical Use of Teprotumumab in a Heterogenous Thyroid Eye Disease Population.

PURPOSE: To describe the clinical course in a heterogeneous series of subjects with thyroid eye disease (TED) treated with teprotumumab. METHODS: Cross-sectional cohort study including patients with clinical diagnosis of TED who was treated with teprotumumab. The entire cohort was analyzed together and subsequently in clinical subgroups based on stage and grade of disease. Primary outcome measure was change in proptosis ≥2 mm. Secondary outcome measures included change in clinical activity score (CAS), ductions, strabismic deviation, MRD1, and MRD2. Bivariate and multivariate statistics were performed. RESULTS: The study included 21 patients. Mean ± SD age was 61.5 ± 12.6 years and 71.4% were female. Reduction in proptosis ≥2 mm was achieved in 71.4% of the sample. Stage and grade were not significant predictors of outcome. Treatment with teprotumumab resulted in a 2.5 ± 1.8 mm reduction of proptosis (P < 0.001), 2.2 ± 1.4 reduction in CAS (P < 0.001), and 16.9 ± 19.3 degree improvement in extraocular motility (P < 0.001). There were no significant differences for change in CAS, proptosis, ductions, or MRD2 between different grades and stages of disease. Total strabismus and MRD1 improvement were greater in the active stage of disease (P < 0.05). Three cases of dysthyroid optic neuropathy, refractory to methylprednisolone therapy improved after initiation of teprotumumab. CONCLUSIONS: Treatment of TED with teprotumumab in a heterogeneous patient population is associated with improvement in proptosis, extraocular motility, and CAS. Patients beyond those defined in the clinical trials, including those affected by stable stage, milder grade, and vision-threatening TED may benefit from this therapy. There are, however, limits on the overall efficacy of this medication in the management of certain physical characteristics in TED including eyelid position and strabismus.

The Effect of Early Postoperative Swelling on Change in Upper Eyelid Position After External Levator Resection and Blepharoplasty.

PURPOSE: To determine if early postoperative swelling has an effect on change in the upper eyelid position after external levator resection (ELR) or blepharoplasty. METHODS: In this observational cohort study, patients >18 years old who underwent ELR or upper blepharoplasty were identified. Digital photographs in primary position were obtained at the preoperative, early postoperative week one, and late postoperative month 3 visits. Marginal reflex distance 1 (MRD1) was measured digitally. Swelling was graded on a previously validated scale from 0 to 3. Photographs were evaluated by 26 experts/professional image graders. Primary outcome measure was changed in MRD1 from preoperative to late postoperative visits. Secondary outcome measure was change in MRD1 from early to late postoperative visits. The effect of early postoperative swelling on each outcome was assessed using linear regression models. RESULTS: The sample contained 54 patients (25 ELR and 29 blepharoplasty). The model regressing change in MRD1 from preoperative to late postoperative visits with early postoperative swelling as the predictor was significant for ELR (R2 = 0.165, p = 0.044) but not blepharoplasty (R2 = 0.016, p = 0.515). However, the model controlling for preoperative MRD1 was not significant for ELR (B = 0.423, p = 0.354). The model regressing change in MRD1 from early to late postoperative follow up utilizing early postoperative swelling as the sole predictor was not significant for ELR (R2 = 0.010, p = 0.627) but was significant for blepharoplasty (R2 = 0.207, p = 0.013), with increased swelling associated with greater change in MRD1. CONCLUSIONS: Early postoperative swelling does not affect the final MRD1 outcome of ELR or upper blepharoplasty; however, blepharoplasty patients with early postoperative swelling may experience greater increase in MRD1 over follow up.

Ophthalmic Surgeries and Systemic Medical Conditions Associated With the Development of Involutional Ptosis.

PURPOSE: To investigate the associations between systemic medical conditions and types of ocular surgery and the development of aponeurotic ptosis. METHODS: In this case-control observational cohort study, the relative prevalence of involutional ptosis in a large population of adult patients was assessed. Deidentified patient data from a 10-year time period was obtained from the electronic medical records of 5 large academic medical centers. Patients were selected based on ICD9 and ICD10 codes for involutional ptosis. Control patients were age and gender matched and randomly selected from a general adult population. Systemic comorbidities were determined based on ICD9 and ICD10 codes, and prior ocular surgeries based on CPT codes. The influence of systemic comorbidities and ocular surgery was examined utilizing logistic regression analysis. RESULTS: The study cohort consisted of 8297 adult patients with involutional blepharoptosis and 13,128 matched controls. The average age was 65 years. The 3 significant risk factors for developing ptosis were ocular surgery, hyperthyroidism, and type II diabetes with odds ratios of 4.2, 2.5, and 1.45, respectively (p < 0.05). Strabismus, cornea, and glaucoma surgeries were more highly associated with developing ptosis (p < 0.05). Strabismus surgery had the greatest odds ratio of 3.37, followed by cornea surgery at 2.31, and glaucoma surgery at 1.56. CONCLUSIONS: Involutional ptosis is likely a multifactorial process. This study demonstrates that risk factors for the development of ptosis include ocular surgery, specifically strabismus, cornea, and glaucoma surgery, as well as hyperthyroidism and type II diabetes.

Punctal stenosis associated with dupilumab therapy for atopic dermatitis.

In this case series, the authors report three patients with severe atopic dermatitis who presented with epiphora and conjunctivitis while undergoing dupilumab therapy. On clinical examination, all patients were found to have punctal stenosis, with one case having progressed to punctal obstruction. An assortment of strategies was elected, including discontinuation of dupilumab, treatment of conjunctivitis, and surgical intervention with probing, punctoplasty, and silicone intubation. This report spotlights punctal stenosis as an important new side effect of dupilumab and suggests that additional cases of dupilumab-associated lacrimal drainage impairment will continue to emerge.