Oculoplastics

Oculoplastics Publications

Green MB, Daly MK, Laver NMV, Lefebvre DR. Adult-onset asthma and periocular xanthogranuloma - A rare infiltrative disease of the orbit and eyelid. Am J Ophthalmol Case Rep 2021;22:101043.Abstract
Purpose: To present a case of adult onset asthma with periocular xanthogranuloma (AAPOX), and discuss existing literature on adult orbital xanthogranulomatous diseases (AOXGDs) and their treatment. Observations: A 63 year old male presented with progressive bilateral eyelid swelling with overlying yellow plaques associated with asthma. CT scan showed periorbital swelling with enlargement of the superior and lateral rectus muscles bilaterally. Biopsy demonstrated orbital xanthogranulomatous disease with increased IgG4 plasma cells. The patient was treated with intralesional triamcinolone, oral prednisone, and cyclophosphamide without significant improvement. Surgical debulking was eventually performed which improved his external symptoms until he was lost to follow up 15 months later. Conclusions and Importance: AOXGDs are a group of rare infiltrative diseases of the eyelids and orbit that can be associated with significant systemic morbidities. While they all have similar underlying histopathologic features, appreciating the clinical difference between these diseases is important in understanding patient prognosis and ensuring appropriate clinical monitoring. There is also growing research demonstrating that AAPOX, along with other AOXGDs, may represent part of a continuum of IgG4 related disease, similar to what is seen in this case. There is currently no reliably effective treatment for AOXGDs, and additional research into the management of these diseases is necessary.
Chiou CA, Reshef ER, Freitag SK. Teprotumumab for the treatment of mild compressive optic neuropathy in thyroid eye disease: A report of two cases. Am J Ophthalmol Case Rep 2021;22:101075.Abstract
Purpose: To report two cases of thyroid eye disease (TED) associated compressive optic neuropathy (CON) that resolved after treatment with teprotumumab. Observation: Two patients presented with active TED resulting in mild CON with the typical corresponding visual field (VF) defects. Both patients were initiated on intravenous (IV) corticosteroid therapy but despite treatment had persistent VF defects. Both patients were then treated with teprotumumab and demonstrated marked clinical improvement and complete resolution of TED-CON VF defects early in their infusion course. Conclusions and importance: These cases suggest that teprotumumab can be a rapid and effective treatment for TED-CON, and raises the question of whether it may be superior to IV corticosteroid therapy.
Tao JP, Aakalu VK, Freitag SK, Sobel RK, Foster JA, Wladis EJ, McCulley TJ, Yen MT. Homeopathic Agents or Vitamins in Reducing Ecchymosis after Oculofacial Surgery: A Report by the American Academy of Ophthalmology. Ophthalmology 2021;Abstract
PURPOSE: To review the published literature to determine the efficacy and safety of homeopathic agents or vitamins in reducing ecchymosis after oculofacial surgery or laser surgery. METHODS: A literature search was conducted in the PubMed database initially in December 2019 and updated in March 2020 to identify all studies in the English language literature on the use of homeopathic agents or vitamins in oculofacial procedures, including laser surgery. The search yielded 124 citations, and 11 articles met all inclusion criteria for this assessment. A panel methodologist then assigned a level of evidence rating for each study. Eleven studies met inclusion criteria; 9 were rated level I, and 2 were rated level III. RESULTS: The agents studied in the articles identified included oral or topical Arnica montana (AM), oral Melilotus extract, topical vitamin K oxide, and topical AM combined with Rhododendron tomentosum. Metrics to describe ecchymosis varied. In 7 controlled studies, perioperative AM provided no or negligible benefit versus placebo. In 2 studies, vitamin K cream was equivalent to placebo. One study of oral Melilotus extract had less ecchymosis compared with controls in paranasal and eyelid ecchymosis at postoperative day (POD) 7, but not at PODs 1 and 4. A lone cohort study of combined topical AM and R. tomentosum lacked objective metrics and adequate controls. No serious side effects from administration of homeopathic agents or vitamins were identified. CONCLUSIONS: The current literature does not support the use of AM, vitamin K oxide, R. tomentosum, or Melilotus extract for reducing ecchymosis after oculofacial surgery or pulsed dye laser surgery.
Boal NS, Cretara EAZ, Bleier BS, Lam AC, Lefebvre DR. In vivo analysis of endocanalicular light pipe transillumination in endoscopic dacryocystorhinostomy: Anatomic considerations and cautions for the transitioning. Orbit 2021;:1-5.Abstract
Purpose: Localization of the lacrimal sac is a critical step during endoscopic dacryocystorhinostomy (endo-DCR). A "light pipe" can be used to transilluminate the lacrimal sac endonasally. We hypothesized that this may misguide the surgeon learning endo-DCR to create an osteotomy mostly posterior to the maxillary line if only the bone overlying the transillumination was to be removed, as the thinner lacrimal bone will transmit light more readily than the thicker maxillary bone of the frontal process of the maxilla that forms the anterior lacrimal sac fossa.Methods: The charts of 32 patients with primary acquired nasolacrimal duct obstruction in whom a lighted system was used during endo-DCR at Massachusetts Eye and Ear from April 2015 through October 2016 were reviewed. Patients with prior history of lacrimal surgery or trauma directly to the lacrimal sac fossa were excluded. Location of the maximal point of transillumination in relation to the maxillary line was observed and noted intraoperatively.Results: Of a total of 39 endo-DCR surgeries performed, the intraoperative transillumination point was entirely posterior to the maxillary line in 32 instances (82%).Conclusions: Use of an endocanalicular light pipe preferentially illuminates posterior to the maxillary line endonasally. The anterior lacrimal sac fossa (maxillary line and anterior as visualized endonasally) is rarely transilluminated, likely due to thicker bone in that region. Surgeons learning how to perform endo-DCR using a light pipe should be aware of this phenomenon.
Belinsky I, Creighton FX, Mahoney N, Petris CK, Callahan AB, Campbell AA, Kazim M, Lee HHB, Yoon MK, Dagi Glass LR. Teprotumumab and Hearing Loss: Case Series and Proposal for Audiologic Monitoring. Ophthalmic Plast Reconstr Surg 2021;Abstract
PURPOSE: To present a protocol for audiologic monitoring in the setting of teprotumumab treatment of thyroid eye disease, motivated by 4 cases of significant hearing loss, and review the relevant literature. METHODS: Cases of hearing loss in the setting of teprotumumab were retrospectively elicited as part of a multi-institutional focus group, including oculoplastic surgeons, a neurotologist and an endocrinologist. A literature review was performed. RESULTS: An aggregate of 4 cases of teprotumumab-associated hearing loss documented by formal audiologic testing were identified among 3 clinicians who had treated 28 patients. CONCLUSIONS: Teprotumumab may cause a spectrum of potentially irreversible hearing loss ranging from mild to severe, likely resulting from the inhibition of the insulin-like growth factor-1 and the insulin-like growth factor-1 receptor pathway. Due to the novelty of teprotumumab and the lack of a comprehensive understanding of its effect on hearing, the authors endorse prospective investigations of hearing loss in the setting of teprotumumab treatment. Until the results of such studies are available, the authors think it prudent to adopt a surveillance protocol to include an audiogram and tympanometry before, during and after infusion, and when prompted by new symptoms of hearing dysfunction.
Venkateswaran N, Sripawadkul W, Karp CL. The role of imaging technologies for ocular surface tumors. Curr Opin Ophthalmol 2021;Abstract
PURPOSE OF REVIEW: This review will discuss the utility of high-resolution anterior segment optical coherence tomography (HR-OCT), in-vivo confocal microscopy (IVCM) and ultrasound biomicroscopy (UBM) in characterizing and diagnosing various ocular surface tumors, namely ocular surface squamous neoplasia (OSSN), conjunctival lymphoma and conjunctival melanoma. The strengths and limitations of each imaging modality will be discussed along with the characteristics findings of each lesion on each imaging platform. RECENT FINDINGS: HR-OCT can consistently be utilized in the clinic setting to distinguish between epithelial ocular surface tumors such as OSSN as compared with subepithelial tumors such as conjunctival lymphoma and conjunctival melanoma given their distinctive findings. IVCM can be used as an adjunct to HR-OCT to obtain cellular and surface characteristics, whereas UBM can be used to assess tumor depth and thickness for larger and highly pigmented lesions as well as to detect intraocular invasion. SUMMARY: HR-OCT, IVCM and UBM are all helpful imaging modalities to diagnose and characterize various ocular surface tumors and can serve as valuable adjuncts to monitor treatment response and assess for recurrence ocular surface tumors.
Caranfa JT, Yoon MK. Septic Cavernous Sinus Thrombosis: A Review. Surv Ophthalmol 2021;Abstract
Septic cavernous sinus thrombosis (SCST) is a rare, yet severe, process typically arising from infections of the paranasal sinuses (predominately ethmoid and/or sphenoid sinusitis) and, less commonly, otogenic, odontogenic, and pharyngeal sources. Clinical symptoms of SCST arise from obstruction of venous drainage from the orbit and compression of the cranial nerves within the cavernous sinus. In the preantibiotic era, SCST was considered universally fatal (80-100%); however, with the introduction of antibiotics the overall incidence, morbidity and mortality of SCST have greatly declined. In spite of dramatic improvements, morbidity and mortality remain high, with the majority of patients experiencing neurological sequalae, highlighting the severity of the disease and the need for prompt recognition, diagnosis, and treatment. Here we review of the literature of SCST with a focus on the current recommendations and recent evidence for diagnostic and medical management of this condition.
Reshef ER, Bleier BS, Freitag SK. The Endoscopic Transnasal Approach to Orbital Tumors: A Review. Semin Ophthalmol 2021;36(4):232-240.Abstract
Historically, surgical access to orbital tumors has required a transcutaneous, transconjunctival or transcranial approach. Resection of orbital tumors is notoriously challenging due to the surrounding dense network of critical structures in a confined bony cavity. Advances in endoscopic endonasal surgery, initially used for sinonasal and skull base conditions, have allowed for expansion of its applications beyond the sinorbital interface. In the past decade, the evolution of techniques has enabled a purely endoscopic, minimally invasive approach to medially located orbital pathology with good outcomes. With experience and multidisciplinary collaboration between orbit and rhinologic surgeons, this has expanded to allow for a safe and effective transnasal approach to nearly all regions of the orbit with or without assistance from the orbital side. This review summarizes the relevant anatomy, variations of surgical approaches, and literature regarding outcomes of the endoscopic endonasal approach to orbital tumors.
Freitag SK, Yen MT. Reply. Ophthalmology 2021;128(6):e29-e30.
Hu S-L, Shi W-Q, Su T, Ge Q-M, Li Q-Y, Li B, Liang R-B, Zhu P-W, Shao Y. Surgical correction of recurrent epiblepharon in Chinese children using modified skin re-draping epicanthoplasty. Int J Ophthalmol 2021;14(2):217-222.Abstract
AIM: To evaluate the clinical efficacy of the modified skin re-draping epicanthoplasty procedure for correction of recurrent lower lid epiblepharon in Chinese children. METHODS: From 2016 to 2018, 18 children (10 males and 8 females, average age 6.2±1.7y; 30 eyes) with recurrent epiblepharon who attended Beijing Children's Hospital were included in the study. All the children had undergone lower eyelid surgery for epiblepharon. Surgical design included using an additional incision along the upper palpebral margin, to avoid vertical scarring on the upper lid. The re-draping method was used to correct recurrent epiblepharon. Follow-up ranged from 3 to 24mo. Postoperative surgical outcomes, complications, and subjective satisfaction were evaluated. RESULTS: Complete correction of cilia touching the cornea was observed in all patients during an average follow-up of 7.1mo. No "dog ears" or obvious scars were formed after surgery. All parents were satisfied with the cosmetic results and none complained. Mean astigmatism decreased from 2.39±0.79 diopter (D) preoperatively to 2.19±0.79 D at 6mo after surgery; however, the difference was not significant. Best-corrected visual acuity improved, although the change in mean visual acuity was not significant. No recurrence occurred during the follow-up period. CONCLUSION: This surgical modified skin re-draping technique is effective and highly satisfactory for correction of recurrent epiblepharon. The approach is characterized by a simple design, a straightforward procedure, inconspicuous scarring, and good postoperative appearance.
Jakobiec FA, Barrantes PC, Ma L, Mihm M. Epibulbar Proliferative Fasciitis, a Variant of Nodular Fasciitis: A Differential Diagnosis of Conditions With Focal or Diffuse Myxoid Stromas. Ophthalmic Plast Reconstr Surg 2021;Abstract
PURPOSE: To describe the clinical and pathologic features of a case of epibulbar proliferative fasciitis and to compare it with other focal or diffuse myxoid lesions. METHODS: A clinical, histopathologic, and immunohistochemical analysis was performed. The clinical history, photographic documentation, history, and referred slides were reanalyzed. Additional immunohistochemical stains were performed at our institution. RESULTS: A 68-year-old woman developed over a week a brightly vascularized and focally hemorrhagic placoid lesion on the temporal side of the OS. She had had earlier augmentation breast surgery that had been mistakenly initially reported to us to be for breast carcinoma. Hematoxylin- and eosin-stained reactions revealed microscopically a spindle cell lesion with an intact nonkeratinizing epithelium and a background myxoid stroma with prominent capillaries and a light dispersion of small T-cell lymphocytes. Most striking among the spindle cells were some widely separated large atypical cells. The atypical cells were cytokeratin positive, but an expansive panel of immunohistochemical stains for breast carcinoma was negative. The lesion was diagnosed as proliferative fasciitis and has not recurred after 1-year follow up. CONCLUSION: A rapidly evolving conjunctival lesion is unlikely to be a primary or metastatic carcinoma. In the current case, the large ganglioform or rhabdomyoblast-like cells displayed diffuse cytokeratin positivity, still consistent with a mesenchymal or connective tissue cell lineage. Cytokeratin expression has been a finding previously reported in connective tissue tumors and in lymphoma cells. While the current lesion clinically resembles a conventional nodular fasciitis, the presence of the large atypical cells can lead to the misdiagnosis of a sarcoma, which typically displays a much higher Ki-67 proliferation index in comparison with nodular/proliferative fasciitis.
Jafari A, von Sneidern M, Lehmann AE, Shen SA, Shishido S, Freitag SK, Bleier BS. Exclusively endoscopic endonasal resection of benign orbital tumors: a systematic review and meta-analysis. Int Forum Allergy Rhinol 2021;11(5):924-934.Abstract
BACKGROUND: The Cavernous Hemangioma Exclusively Endonasal Resection (CHEER) classification system was developed to standardize prospective outcome analysis following orbital cavernous hemangioma (OCH) resection. The goal of this study was to retroactively apply the CHEER system to all prior existing reports of endoscopic resection of primary benign orbital tumors (BOTs) to: (1) compare patient presentations, perioperative characteristics, and outcomes between OCH and other BOTs; and (2) determine whether the CHEER categorization regime could be expanded to other BOTs. METHODS: A systematic review of studies reporting exclusively endoscopic resections of OCH and other BOTs (eg, solitary fibrous tumor, schwannoma, and meningioma) was performed. Patient, tumor characteristics, and operative outcomes were recorded. All tumors with adequate reporting were retrospectively assigned a CHEER stage. Outcomes were compared using chi-square or Fisher's exact tests. RESULTS: Ninety-three studies met inclusion criteria, and sufficient data were available in 36 studies, comprising 105 tumors (n = 87 OCHs; n = 18 other BOTs). Baseline patient and tumor characteristics, as well as intraoperative and short-term postoperative outcomes were not significantly different between OCHs and other BOTs. Long-term outcomes (eg, visual deficits, diplopia, eye position, and recurrence) also did not differ when controlling for CHEER stage. CONCLUSION: This review represents the largest collection of outcomes data following exclusively endoscopic endonasal resection of BOTs. Short-term and long-term outcomes appear similar between OCHs and other BOTs. These results suggest that exclusively endoscopic resection of orbital tumors may be effective in a range of benign pathologies. Furthermore, these results support a broader application of the CHEER system to other benign primary orbital tumors.
Rossin EJ, Szypko C, Giese I, Hall N, Gardiner MF, Lorch A. Factors Associated With Increased Risk of Serious Ocular Injury in the Setting of Orbital Fracture. JAMA Ophthalmol 2021;139(1):77-83.Abstract
Importance: Orbital fractures are common in ocular trauma, and there is a need to develop predictive tools to estimate risk of concurrent ocular injury. Objective: To identify clinical and radiographic features that are associated with increased risk of substantial ocular injury in the setting of orbital fracture. Design, Setting, and Participants: Retrospective consecutive case series of patients who sustained orbital fractures between 2012 and 2018. Examinations were done at 1 of 2 level 1 trauma centers in the emergency or inpatient setting. A total of 430 consecutive patients (500 eyes) between 2012 and 2017 met inclusion criteria for the training sample. After building a predictive model, 88 additional consecutive patients (97 eyes) between 2017 and 2018 who met inclusion criteria were collected as a test sample. Main Outcomes and Measures: The primary outcome measure was substantial ocular injury distinct from orbital fracture. Results: The mean age of our patient population was 53.5 years (range, 16-100 years). The overall rate of substantial ocular injury was 20.4%, and the rate of injury requiring immediate ophthalmic attention was 14.4%. Five variables were found to be associated with increased risk of substantial ocular injury: blunt trauma with a foreign object (odds ratio [OR], 19.4; 95% CI, 6.3-64.1; P < .001), inability to count fingers (OR, 10.1; 95% CI, 2.8-41.1; P = .002), roof fracture (OR, 9.1; 95% CI, 2.8-30.0; P = .002), diplopia on primary gaze (OR, 6.7; 95% CI, 1.7-25.1; P = .003), and conjunctival hemorrhage or chemosis (OR, 4.2; 95% CI, 2.2-8.5; P < .001). The results were translated into a bedside tool that was tested in an independent group of eyes (n = 97) and found to be associated with substantial ocular injury with a 95% sensitivity (95% CI, 77.2-99.9), 40% specificity (95% CI, 28.9-52.0), 31.8% positive predictive value (95% CI, 27.5-36.5), and 96.8% negative predictive value (95% CI, 81.3-99.5). Conclusions and Relevance: A minority of patients with an orbital fracture had a substantial ocular injury. Certain radiographic and clinical findings were associated with substantial ocular injury. Testing of the algorithm in prospective longitudinal settings appears warranted.
Jakobiec FA, Hanbazazh M, Barrantes PC, Yoon MK. Conjunctival Implantation Cyst in the Orbicularis Oculi Muscle: Review of a Possible Origin From Displaced Stem Cells With a Differential Diagnosis. Ophthalmic Plast Reconstr Surg 2021;37(1):1-11.Abstract
PURPOSE: To document a unique case of a corneal/conjunctival epithelial inclusion cyst located in the orbicularis oculi muscle with a comprehensive review of variant conjunctival cysts and simulating conditions. METHODS: Clinicopathologic case report with detailed histopathologic and immunohistochemical evaluation for cytokeratins combined with a tabulation of mimicking lesions and relevant literature citations. RESULTS: A 59-year-old man experienced severe blunt left periorbital trauma that resulted in a limbal partial-thickness corneal wound with an associated epithelial abrasion and a full-thickness eyelid laceration extending from the superior fornix to the margin. Several months after surgical repair of the eyelid a cyst appeared in the superior pretarsal skin. Histopathologic and immunohistochemical investigations supplied data suggesting that the cyst had a high probability of a corneoscleral limbal stem cell origin. Distinctive features of the lesion are contrasted with those of allied or simulating cysts. CONCLUSIONS: Stem cells are now believed to be located at the corneoscleral limbus, in the inferior fornix, in the medial canthal region, and at the eyelid margin where transitions from conjunctival epithelium to epidermal epithelium occur. Due to their replicative, hardy and robust nature, stem cells displaced to alien environments are most likely to survive and produce cysts. The cyst's corneal-type cytologic characteristics, the absence of goblet cells, and the expression of a broad spectrum of cytokeratin biomarkers in the current case give support to the proposal that limbal stem cells in the region of the corneal laceration were displaced to the eyelid orbicularis muscle and were responsible for this most extraordinary cyst. Comparison with other epithelial cystic linings lends further evidence for this conclusion.

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