Inferior Oblique Flap: An Approach for Improved Vascular Supply in Complex Ocular Surface Reconstruction.

This case report describes a novel surgical technique using an inferior oblique muscle flap for a patient requiring complex ocular surface reconstruction. A 63-year-old female with Stevens-Johnson syndrome and complex ocular history including multiple penetrating keratoplasties and keratoprostheses presented with recurrent keratoprosthesis implant plate necrosis. In conjunction with revision keratoplasty, a custom inferior oblique muscle flap was designed to provide improved support and vascularity to the corneal tissue bed; at 12 months postoperation, the graft remained well attached and free of graft failure. Here, we present this technique along with operative photos, describing how this technique may provide surgeons an additional option for complex ocular surface reconstruction.

Transorbital Endoscopic Approach to the Pterygopalatine Fossa: A Less Invasive Alternative to Traditional Access.

PURPOSE: The purpose of this article and accompanying video is to demonstrate a transorbital endoscopic approach for accessing the pterygopalatine fossa (PPF). This technique does not require a skin incision, avoids dissection of critical neurovascular structures, and utilizes a comparatively small osteotomy. The 2 cases presented in this article highlight the utility of a transorbital endoscopic approach for accessing an anatomic region that has traditionally required more invasive techniques to reach. METHODS: Description of surgical technique with 2 illustrative clinical cases and accompanying surgical video. RESULTS: Surgical technique: A trans-conjunctival approach is taken to the inferior orbital rim, and a subperiosteal dissection is propagated posteriorly. The bone of the posterior orbital floor is then deroofed, and the superior portion of the posterior wall of the maxillary sinus is removed, allowing access to the PPF for an incisional biopsy. CASE: A 76-year-old male with a history of left cheek squamous cell carcinoma presented with progressive V2 paresthesia and an abnormally enhancing lesion in the left PPF on MRI. CASE: A 58-year-old male with no significant medical history presented with left facial numbness (V1-V3), ptosis, an abduction deficit, and decreased hearing. Contrast-enhanced MRI demonstrated an abnormally enhanced lesion in the left PPF extending to Meckel's cave.The transorbital approach described was used to successfully obtain a diagnostic biopsy in both cases. CONCLUSIONS: These cases highlight the utility of a transorbital endoscopic approach to the PPF as a less morbid alternative to traditional access. Patient selection is key to identifying appropriate cases.

Orbital Liposarcoma: A Surveillance, Epidemiology and End Results Database Study.

PURPOSE: Orbital liposarcoma is a challenging tumor to treat due to its rarity and high rate of local recurrence, and the role of radiotherapy and chemotherapy remain unclear. Analysis of big data may improve our overall understanding of orbital disease and role of adjuvant therapies. METHODS: Data were extracted from the Surveillance, Epidemiology and End Results (SEER) Research Plus database from 1975 to 2017. All patients with a diagnosis of liposarcoma (ICD-O3 codes 8850-8858, 8869-8862, 8870, 8880, 8881) were included. Cases were divided into 4 groups by primary site: orbit, retroperitoneum, soft tissue, and other. RESULTS: A total of 16,958 patients were included. Patients with orbital involvement were younger and more likely to be female ( p < 0.05). Among orbital lesions, myxoid liposarcoma was the most common histologic subtype (6/19; 31.6%) followed by well differentiated (5/19; 26.3%). This differed from the distribution of histologic subtypes encountered elsewhere, for which well-differentiated liposarcoma was the most common (retroperitoneum 979/3,136; 31%, soft tissue 3,493/11,671; 30%, and other sites 497/2,132; 23%, p < 0.05). Dedifferentiated histologic subtype was the second most common subtype found in the retroperitoneum (946/3,136; 30%), whereas it was less common in the orbit (2/19; 11%) and soft tissue (1,396/11,671; 12%) ( p < 0.001). Patients with orbital liposarcoma had similar disease-specific mortality compared with soft-tissue location ( p = 0.825) and lower disease-specific mortality compared with retroperitoneal location ( p < 0.001). When all locations were combined, patients with well-differentiated liposarcoma had the lowest disease-specific mortality. CONCLUSIONS: Patients with orbital liposarcoma tend to be younger, female, and have a better prognosis than those with retroperitoneal disease, likely due to the lower incidence of dedifferentiated histologic subtype.

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.

Prevalence, Clinical and Imaging Characteristics of Superior Ophthalmic Vein Periphlebitis in Thyroid Eye Disease.

PURPOSE: To determine the prevalence, clinical features, and radiographic findings of superior ophthalmic vein periphlebitis (SOVP) in thyroid eye disease (TED). METHODS: Patients with a clinical diagnosis of thyroid eye disease and contrast-enhanced imaging were included. Imaging was reviewed for the presence of SOVP, and patients with SOVP were compared to those without. A random eye was determined to be the affected eye in patients without SOVP. RESULTS: A total of 212 patients met the inclusion criteria. Unilateral SOVP was identified in 4.7% of cases. There was no significant difference in age ( p = 0.22), gender ( p = 0.09), or disease duration ( p = 0.14) between patients with and without SOVP. There was a significant ( p < 0.05) difference in stage classification and clinical activity core between the groups. The affected eye in patients with SOVP had significantly ( p < 0.05) greater margin reflex distance 1, degree of relative proptosis, horizontal motility restriction, and vertical motility restriction than in patients without SOVP. There was no significant difference in horizontal strabismus ( p = 1.0), vertical strabismus ( p = 0.87), or relative intraocular pressure ( p = 0.77). On imaging, the maximal diameter of the SR and IR were found to be significantly ( p < 0.05) larger in the affected eye of patients with periphlebitis; however, there was no difference in measured diameter of the medial rectus and ( p = 0.30) or lateral rectus ( p = 0.78). CONCLUSIONS: SOVP is an under-reported imaging finding of thyroid eye disease. It is associated with significantly greater margin reflex distance 1, relative proptosis, and motility restriction on exam as well as larger superior rectus and inferior rectus diameter on imaging. These patients tend to present in the active stage of disease with greater clinical activity score.

Radiologic Features of Well-circumscribed Orbital Tumors With Histopathologic Correlation: A Multi-center Study.

PURPOSE: To delineate specific imaging characteristics of solitary fibrous tumors, schwannomas, cavernous venous malformations, and well-circumscribed orbital lymphoma. METHODS: Patients undergoing excisional biopsy of solitary fibrous tumor, schwannomas, cavernous venous malformations, or well-circumscribed orbital lymphoma with preoperative MRIs available for review were identified at 3 academic centers in the United States and Australia. An exploratory statistical analysis was performed to identify important radiologic features, which were subsequently included in a random forest model. Histopathologic correlates were evaluated in representative cases. RESULTS: A total of 91 cases were included with a mean age of 52.9 ± 17.2 years. Nearly all solitary fibrous tumors were located in the anterior or mid orbit (87.5%) and they more commonly demonstrated intralesional heterogeneity on T2-weighted imaging (45.5%) ( p < 0.01). Compared with the other tumors, schwannomas tended to be intraconal (66.7%) and were often in the mid or posterior orbit (83.4%) ( p < 0.01). Cavernous venous malformations characteristically demonstrated progressive contrast enhancement (93.9%; p < 0.01). Most lesions in all 4 groups were hypointense on T1-weighted imaging (80%-100%; p = 0.14) and only well-circumscribed orbital lymphoma tended to also be hypointense on T2 (81.8%) ( p < 0.01). Finally, cases of lymphoma had significantly lower apparent diffusion coefficient ratios (0.9 ± 0.2) ( p < 0.001), while the other 3 groups were not significantly different from one another (cavernous venous malformations: 1.8 ± 0.4; schwannomas: 1.8 ± 0.5; and solitary fibrous tumor: 1.6 ± 0.6) ( p = 0.739). CONCLUSIONS: Key features that aid in the differentiation of these 4 tumors from one another include T2 intensity and homogeneity, early contrast-enhancement pattern, and ADC ratio.

Teprotumumab for Thyroid Eye Disease-related Strabismus.

PURPOSE: To assess and quantify teprotumumab's effect on thyroid eye disease-related strabismus by change in measured horizontal and vertical deviations and change in extraocular motility. METHODS: We reviewed a series of patients with thyroid eye disease-related strabismus treated with teprotumumab. Exclusion criteria included age under 18 years, strabismus of alternate etiology, or thyroid eye disease-related reconstructive surgery during the treatment course. Primary outcomes were absolute (prism diopters) and relative (%) differences in horizontal and vertical deviations in primary position at distance, as well as change in ductions of the more affected eye. Secondary outcomes included incidence and timing of strabismus surgery postteprotumumab. RESULTS: Thirty-one patients were included, with mean age 63 years and thyroid eye disease duration 10 months. After teprotumumab, there was 6 prism diopters (39%) mean reduction in vertical deviation ( p < 0.001), without significant change in mean horizontal deviation ( p = 0.75). Supraduction, abduction, adduction, and infraduction significantly improved in the more restricted eye ( p < 0.01, p < 0.01, p = 0.04, and p = 0.01, respectively). Thirty-five percent of patients underwent strabismus surgery posttreatment, at an average 10 months after last infusion. CONCLUSIONS: Teprotumumab produced a statistically significant reduction in vertical but not horizontal strabismus angles in primary position at distance. Extraocular motility in all 4 ductions also improved. A substantial minority of patients still required strabismus surgery following teprotumumab.

Teprotumumab for Thyroid Eye Disease-related Strabismus.

PURPOSE: To assess and quantify teprotumumab's effect on thyroid eye disease-related strabismus by change in measured horizontal and vertical deviations and change in extraocular motility. METHODS: We reviewed a series of patients with thyroid eye disease-related strabismus treated with teprotumumab. Exclusion criteria included age under 18 years, strabismus of alternate etiology, or thyroid eye disease-related reconstructive surgery during the treatment course. Primary outcomes were absolute (prism diopters) and relative (%) differences in horizontal and vertical deviations in primary position at distance, as well as change in ductions of the more affected eye. Secondary outcomes included incidence and timing of strabismus surgery postteprotumumab. RESULTS: Thirty-one patients were included, with mean age 63 years and thyroid eye disease duration 10 months. After teprotumumab, there was 6 prism diopters (39%) mean reduction in vertical deviation ( p < 0.001), without significant change in mean horizontal deviation ( p = 0.75). Supraduction, abduction, adduction, and infraduction significantly improved in the more restricted eye ( p < 0.01, p < 0.01, p = 0.04, and p = 0.01, respectively). Thirty-five percent of patients underwent strabismus surgery posttreatment, at an average 10 months after last infusion. CONCLUSIONS: Teprotumumab produced a statistically significant reduction in vertical but not horizontal strabismus angles in primary position at distance. Extraocular motility in all 4 ductions also improved. A substantial minority of patients still required strabismus surgery following teprotumumab.

Hypoglobus, Exophthalmos, and Interpupillary Distance: Measuring Globe Dystopia in Thyroid Eye Disease.

PURPOSE: Thyroid eye disease (TED) is characterized by a variety of disfiguring periocular changes. Vertical globe changes affecting the relative position of the eyelids are not well understood in patients with TED. This study seeks to determine the effect of orbital decompression on vertical globe displacement in patients with TED, without TED, and with intraconal tumor (ICT). METHODS: For this cross-sectional study, a clinical database was used to identify patients with TED. Comparison groups were drawn from separate anonymized databases. Vertical position and interpupillary distance (IPD) were measured from photographs and exophthalmos was measured via Hertel's exophthalmometer. Primary outcomes were vertical globe position at baseline and postoperatively in patients with TED and ICT. Secondary outcomes included the relationship between vertical globe position, exophthalmos, and IPD. RESULTS: Among 269 participants meeting the inclusion criteria, mean vertical globe position was significantly lower in patients with TED following lateral decompression surgery compared to controls, after accounting for race, age, and sex. While patients with ICT had a significant difference in preoperative and postoperative IPD, patients with TED did not. Medial or inferior decompression did not significantly change globe position and lateral decompression did not cause lateral canthal dystopia in patients with TED. No association between postoperative changes in exophthalmometry, IPD, and globe position was found in patients with TED. CONCLUSIONS: Patients with TED experience hypoglobus that does not improve following decompression surgery. There was no correlation between change in vertical globe position and exophthalmos or IPD among patients with TED. Surgeons should discuss the possibility of hypoglobus as a persistent finding for patients with TED undergoing decompression surgery.

Clinical and demographic predictors of buccal fat pad volume in thyroid eye disease.

PURPOSE: To understand how thyroid eye disease (TED) impacts buccal fat pad (BFP) volume. METHODS: In this cohort study, computed tomography (CT) scans and charts of adult patients with and without TED were obtained from an institutional database. The primary outcome was BFP volume in cubic centimeters. Three independent, blinded observers analyzed scans using Horos, a free, open-source medical image viewing software. Bivariate and multivariable analyses were performed. We determined sample size using an effect size based on published reports of the minimum amount of fat excision needed to notice a clinical difference. Equivalence testing against upper and lower bounds set by the same effect size was conducted to assess practical significance of the results. RESULTS: Our sample was sufficient to detect a difference as large as 1.5cc with 95% power. 72 scans were included in our study, 24 TED patients and 48 controls. Mean BFP volume was not statistically different between TED patients and controls (3.96 cc vs 4.06 cc, p = .778). Analysis of covariance adjusting for relevant patient factors (age, sex, and BMI) also failed to find a significant difference between groups. Equivalence testing was significant (p < .001) and revealed the observed difference between groups was less than any clinically meaningful difference. For an effect size of 1.5cc, the data suggests there is a 5% risk of a false negative. CONCLUSIONS: TED was not associated with a significant difference in BFP volume, suggesting that the BFP is spared from TED-related soft-tissue expansion.

Effect of external eyelid weighting on eyelid and eyebrow position in normal and ptosis patients.

PURPOSE: This study aims to characterize the physiologic response of both eyelid and eyebrow position to increasing downward forces simulated by external weights. METHODS: In this prospective observational study, both normal individuals and patients affected by ptosis were tested. External eyelid weights were placed on one upper eyelid with incrementally increasing weight from 0.2 to 2.4 g. The eyelid carrying the weight was randomly selected for normal subjects and patients with bilateral ptosis, whereas for unilateral ptosis, the ptotic eyelid was utilized. Photographs were obtained at baseline and with increasing weight until MRD1 reached 0 on the weighted side or, until 2.4 g was reached. Eyelid and brow position on the weighted and unweighted sides were digitally measured in millimeter. Primary outcome measures were change in the margin to reflex distance (MRD1) and pupil to brow distance (PTB) with weight on the weighted and unweighted sides for normal and ptosis subjects. RESULTS: The weighted eyelid MRD1 decreased linearly with increasing weight. This was true for normal and ptosis subjects. The unweighted eyelid MRD1 increased linearly with increasing weight. This was also the case for both normal and ptosis subjects. With increasing weight, PTB increased linearly on the weighted side. No significant intergroup differences were noted. CONCLUSIONS: In normal and ptosis subjects, when external weight on the eyelid is incrementally increased, the weighted eyelid MRD1 decreases, the unweighted eyelid MRD1 increases, and both brows elevate in a linear fashion.

Recent Fluctuations in Public Searches for Cosmetic Procedures as Shown by Google Trends.

PURPOSE: The goal of this study is to understand the dynamic trends of 20 cosmetic procedures in the United States using Google Trends. METHODS: This was a descriptive cross-sectional study using Google Trends to measure changes in search volumes for 20 cosmetic procedures from a 5-year period (2018 to 2023) in the United States. Outcome measures included total and weekly changes in relative search volumes and variance levels for the following 20 search terms related to cosmetic surgeries: blepharoplasty, botox, breast augmentation, breast lift, brow lift, buccal fat removal, chemical peel, dermal filler, face lift, hair removal, hair transplant, lip filler, liposuction, masseter botox, microneedling, otoplasty, photofacial, rhinoplasty, tear trough filler, and tummy tuck. Search terms were divided into 3 categories, a) those with a change of public interest over a single week of 25% or less, b) 26%-50%, and c) greater than 50%. RESULTS: There was an overall 5-year increase of public interest in all but 5 search terms: breast augmentation, breast lift, liposuction, photofacial, and tummy tuck. Within a single week, lip filler, masseter botox, buccal fat removal, and tear trough filler exhibited greater than 50% change in public interest, while public interest in microneedling, hair removal, rhinoplasty, tummy tuck, liposuction, breast lift, and augmentation were more stable with less than 26% change. CONCLUSIONS: While certain procedures may experience rapid accelerations in public interest, not all procedures will consistently increase in popularity. Physicians must recognize the shifting popularity of cosmetic procedures to provide informed cosmetic care to patients.

Orbital Vascular Malformations: Relationship Between Enophthalmos and Clinically Apparent Distensibility with Valsalva.

PURPOSE: Determining the hemodynamic characteristics of an orbital vascular malformation is a critical step in management. The purpose of this study is to assess the relationship between enophthalmos and clinically apparent distensibility of orbital vascular malformations, to optimize imaging and treatment. METHODS: In this cross-sectional cohort study consecutive patients at a single institution were screened for study entry. Data extracted included age, sex, Hertel measurements, presence or absence of distensibility during the Valsalva maneuver, whether lesions were primarily venous or lymphatic based on imaging, and location of the lesion relative to the globe. Enophthalmos was defined as ≥ 2 mm difference from the opposite side. Parametric and nonparametric statistics were used, and linear regression was performed to examine factors predictive of Hertel measurement. RESULTS: Twenty-nine patients met the inclusion criteria. Relative enophthalmos ≥2 mm was significantly associated with distensibility ( p = 0.03; odds ratio = 5.33). Distensibility and venous dominant morphology were the 2 most important factors associated with enophthalmos on regression analysis. The relative position of the lesion anterior or posterior to the globe did not have a significant bearing on baseline enophthalmos. CONCLUSIONS: The presence of enophthalmos increases the likelihood that an orbital vascular malformation is distensible. This group of patients was also more likely to be characterized by venous dominant malformations. Baseline clinical enophthalmos may serve as a useful surrogate marker for distensibility and venous dominance, which may be useful in guiding the selection of appropriate imaging.

Optimizing Management of Asymmetric Ptosis: A Comparison of Three Posterior Approach Resection Algorithms.

PURPOSE: The purpose of this study was to compare the efficacy of 3 resection algorithms in the management of patients with asymmetric ptosis. METHODS: Patients undergoing bilateral Muller's muscle-conjunctival resection (MMCR) were identified. Standardized preoperative clinical photographs were examined and margin reflex distance 1 (MRD1) was measured using ImageJ. Patients presenting with ≥1 mm of asymmetry in MRD1 were included. Three groups were identified: variable (4:1 ratio, with the lower side receiving a greater resection), fixed (7 mm resection bilaterally), and tarsectomy (7 mm bilaterally + 1 mm of tarsus resected on the lower preoperative side). Postoperative MRD1 was measured from photographs obtained 3 months after surgery. The primary outcome was postoperative asymmetry. RESULTS: A total of 95 patients with a mean age of 71.0 ± 11.0 years were included. There was no significant difference in age ( p = 0.277) or length of follow-up ( p = 0.782) between the groups. Although the fixed tarsectomy group had significantly greater preoperative asymmetry ( p = 0.001), there was no significant difference in postoperative asymmetry ( p = 0.166). On multivariate analysis, preoperative asymmetry was the only significant predictor of postoperative asymmetry ( p < 0.001). Specifically, the surgical group was not a predictor of the primary outcome ( p = 0.723). CONCLUSIONS: Resection amount and technique did not predict postoperative outcomes in cases of asymmetric ptosis. This may support the hypothesis that changes in eyelid position and symmetry following MMCR is due to a dynamic system, rather than as a result of purely mechanical forces.

Orbital Involvement in Acute Adult Leukemias: Case Series and Review of Literature.

Orbital involvement in acute myeloid leukemia (AML) and acute lymphoblastic leukemia (ALL) is well-described in children but is uncommon in adults. This series reports 2 adult patients with orbital leukemic involvement and summarizes the existing literature. A 37-year-old male with recently diagnosed AML underwent induction therapy and subsequently developed a tan-pink colored sub-conjunctival lesion in the left eye. Incisional biopsy confirmed AML. A 35-year-old male with history of ALL presented with left-sided orbital mass. Fine needle aspiration biopsy confirmed ALL. Literature review of adult-onset orbital leukemia yielded 29 cases of AML and 3 cases of ALL. Orbital involvement of acute adult-onset leukemia tends to be unilateral, presents in the extraconal space and can occur at any point during systemic leukemic disease. Chemotherapy is the mainstay of treatment, often in combination with radiation and/or hematopoietic stem cell transplant.

Teprotumumab and Orbital Decompression for the Management of Proptosis in Patients With Thyroid Eye Disease.

PURPOSE: To compare outcomes of patients with thyroid eye disease treated with teprotumumab or orbital decompression, or both in sequence. METHODS: Patients with thyroid eye disease and treated with decompression, teprotumumab, or both were included. Four groups were defined: decompression only, teprotumumab only, teprotumumab first with decompression later, and decompression first with teprotumumab later. The primary outcome was change in exophthalmometry. Secondary outcomes included change in extraocular muscle motility, strabismus, diplopia, and side effects. RESULTS: One hundred and thirty-nine patients were included. The mean duration for early follow-up was 1.2 months for both decompression and teprotumumab groups. The mean late follow-up was 14.4 and 8.2 months for the decompression and teprotumumab groups respectively. Mean change in exophthalmometry was significantly greater for the decompression group (3.5 mm) compared with teprotumumab (2.0 mm) at late follow-up. Improvement in total extraocular muscle restriction was significantly greater in the teprotumumab group (14.7 degrees) than in the decompression group (2.6 degrees). The teprotumumab group had a significantly higher percentage of patients with diplopia score >1 at baseline and late follow-up (p < 0.01) compared with the decompression group. Additional treatment with teprotumumab or decompression when previously treated with the opposite had similar proptosis reduction effect as that therapy alone. CONCLUSIONS: Surgical decompression has a greater proptosis reduction effect than teprotumumab, whereas teprotumumab better improves extraocular muscle motility. The addition of teprotumumab or decompression to a previous course of the opposite adds a similar effect to the supplemental treatment alone.

Outcomes of Patients With Thyroid Eye Disease Partially Treated With Teprotumumab.

PURPOSE: In response to the coronavirus (COVID-19) pandemic, teprotumumab production was temporarily halted with resources diverted toward vaccine production. Many patients who initiated treatment with teprotumumab for thyroid eye disease were forced to deviate from the standard protocol. This study investigates the response of teprotumumab when patients receive fewer than the standard 8-dose regimen. METHODS: This observational cross-sectional cohort study included patients from 15 institutions with active or minimal to no clinical activity thyroid eye disease treated with the standard teprotumumab infusion protocol. Patients were included if they had completed at least 1 teprotumumab infusion and had not yet completed all 8 planned infusions. Data were collected before teprotumumab initiation, within 3 weeks of last dose before interruption, and at the visit before teprotumumab reinitiation. The primary outcome measure was reduction in proptosis more than 2 mm. Secondary outcome measures included change in clinical activity score (CAS), extraocular motility restriction, margin reflex distance-1 (MRD1), and reported adverse events. RESULTS: The study included 74 patients. Mean age was 57.8 years, and 77% were female. There were 62 active and 12 minimal to no clinical activity patients. Patients completed an average of 4.2 teprotumumab infusions before interruption. A significant mean reduction in proptosis (-2.9 mm in active and -2.8 mm in minimal to no clinical activity patients, P < 0.01) was noted and maintained during interruption. For active patients, a 3.4-point reduction in CAS ( P < 0.01) and reduction in ocular motility restriction ( P < 0.01) were maintained during interruption. CONCLUSIONS: Patients partially treated with teprotumumab achieve significant reduction in proptosis, CAS, and extraocular muscle restriction and maintain these improvements through the period of interruption.

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.

Lacrimal hyperalgesia: a case series of post-operative painful lacrimation.

Lacrimal hyperalgesia is a rare type of periorbital neuralgia triggered by tear production. Two female patients in their mid-forties underwent orbital surgery and, several weeks following their procedures, developed pain when they produced tears. The symptom was described as a sharp, debilitating, and transient periocular pain. A possible mechanism for this lacrimal hyperalgesia is through the formation of an artificial synapse along the superolateral aspect of the orbit. Two mechanisms for this type of hyperalgesia are described herein, which include potential mechanical compression or direct disruption of the normal nerve pathways and microvascular disruption causing ischemic nerve injury. Currently, there is no accepted treatment for this aberrant neuropathic pain caused by lacrimation. Gabapentin therapy was trialed in one of these two patients, who experienced partial improvement with nightly use. In this case series, we describe the clinical and radiographic features associated with this unique type of neuralgia, emphasizing the importance of recognizing it as a complication following orbital surgery.

Orbital manifestations of B-cell acute lymphoblastic leukemia/lymphoma.

A 2-year-old boy presented with left periorbital edema, proptosis, hyperglobus and esotropia. Imaging revealed an inferotemporal orbital mass with adjacent bony erosion. Histological evaluation of an orbital biopsy revealed B-cell acute lymphoblastic leukemia/lymphoma (B-ALL/BLL). The patient was subsequently treated with chemotherapy. Although orbital involvement in acute myelogenous leukemia has been well-described, orbital manifestations of B-ALL/BLL are uncommon, with only a limited number of previous reports in the literature.