Oculoplastics Publications

Park ESY, Rabinowits G, Hamnvik O-PR, Dagi LR. A case of Graves' ophthalmopathy associated with pembrolizumab (Keytruda) therapy. J AAPOS 2018;Abstract
We present the first reported case of Graves' orbitopathy induced by pembrolizumab, a new FDA-approved drug used for the treatment of multiple refractory solid tumors and classic Hodgkin lymphoma. Pembrolizumab elicits T-lymphocyte proliferation; we suspect that thyroid eye disease may result in some cases.
Wolkow N, Jakobiec FA, Yoon MK. Dermalive Facial Filler Granulomas Masquerading as Neurofibromas. Ophthal Plast Reconstr Surg 2018;Abstract
A 56-year-old woman presented with periocular nodules that were clinically suspected to be neurofibromas. Histopathologic examination of excised nodules revealed a pronounced granulomatous reaction to a foreign material that was composed of glossy polygonal palely eosinophilic fragments. These fragments were outlined in red with Masson trichrome, stained gray with the elastic stain, and were uniformly red with Gomori methenamine silver staining. The histopathologic appearance was consistent with a granulomatous reaction to Dermalive facial filler. Postoperatively the patient admitted that she had filler injections many years earlier in another country, and that nodules appeared 1 year after injection. Treatment with steroids, intralesional immunosuppressive agents and surgery had been previously attempted to eradicate the nodules. The literature pertaining to granulomatous reactions to Dermalive and related hybrid facial fillers is reviewed and treatment options are discussed. This report is the first to illustrate the unique histopathologic staining characteristics of Dermalive, which may be useful to ophthalmic pathologists in identifying this uncommon foreign material.
Mueller SK, Freitag SK, Lefebvre DR, Lee NG, Bleier BS. Revision eDCR using a superior pedicled mucosal flap. Orbit 2018;:1-6.Abstract
BACKGROUND: Endoscopic dacryocystorhinostomies (eDCRs) show patency rates between 81% and 94%. However, dacryocystorhinostomy (DCR) failure and the need for revision remain a significant challenge. One of the principal challenges in revision eDCR is the need to surgically identify the correct osteotomy site and maintain long-term patency in the setting of previously instrumented and potentially scarred tissue. At the same time, the surgeon must assume that the blood supply to the commonly described anterior and posteriorly pedicled flaps has been compromised. OBJECTIVE: The objective of the study is to describe a novel flap technique for revision eDCR. METHODS: The superior based mucosal flap is a novel technique that provides a vascularized mucosa preserving technique in revision eDCR despite previous instrumentation of the lacrimal system. This technique provides wide exposure of the revision osteotomy site while simultaneously allowing a viable mucosal flap to be replaced at the conclusion of the procedure, thereby minimizing bone exposure and cicatricial restenosis. RESULTS: The authors have utilized this technique in 13 procedures with 100% positive identification of the lacrimal sac, a 0% complication rate, and a 100% success rate after a mean follow-up of 26.93 ± 10.33 months (range 6-35 months). CONCLUSION: The eDCR using the superior pedicled mucosal flap provides excellent exposure of the maxillary bone and the lacrimal sac. This method preserves vascularity of the flap using a superiorly based pedicle which is typically inviolate during both open and endoscopic primary DCR. The mucosal flap can then be replaced, thereby minimizing bone exposure and optimizing patency.
Jakobiec FA, Ma L, Wolkow N, Cameron DJ, Maltry AC. Osseous and Adipocytic Differentiations in the Intraocular Lens and Vitreous. Am J Ophthalmol 2018;186:77-88.Abstract
PURPOSE: To analyze 3 unusual mesenchymal transformations within the eye: adipose or osseous metaplasia of the lens and adipose tissue in the vitreous cavity. DESIGN: Observational case series. METHODS: Reevaluation of clinicopathologic diagnoses and histopathologic findings in sections stained with hematoxylin-eosin, periodic acid-Schiff (PAS) reaction, and Masson trichrome method. RESULTS: The 3 cases of mesenchymal transformation occurred in microphthalmic eyes with persistent hyperplastic primary vitreous (more recently termed persistent fetal vasculature). In 1 case there was total lens replacement with lamellar bone; in another, total replacement of the crystalline lens by adipose tissue; and in a third, an anomalous pocket of adipose tissue in the central vitreous. Multifocal remnants of the lens capsule were seen in the osseous case but were absent from the adipocytic cases. The vitreous adipose tissue was surrounded by an elaborate capillary plexus with an empty, collapsed PAS-positive lens capsule in the pupillary region. Anterior pigmented neuroectodermal disorganization, dysgenesis of angle structures, and a hypoplastic or disorganized iris were also observed in the 3 cases. CONCLUSIONS: After review of the literature, it appears that lenticular osseous replacement occurs more often than adipocytic. In addition to vascularization of the lens through a capsular dehiscence, other causes are explored, including direct epithelial-mesenchymal transformations of the lens epithelium or, less likely, of the disorganized adjacent neuroectoderm. The focus of vitreous adipose tissue may represent a transformed luxated lens extruded from its capsule, which was left behind in the pupillary zone.
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. Ophthal Plast Reconstr Surg 2018;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.
Wolkow N, Jakobiec FA, Dryja TP, Lefebvre DR. Mild Complications or Unusual Persistence of Porcine Collagen and Hyaluronic Acid Gel Following Periocular Filler Injections. Ophthal Plast Reconstr Surg 2018;Abstract
The purpose of this study was to describe the histopathologic appearance of dermal eyelid fillers that were unexpectedly encountered in ophthalmic plastic surgery samples from patients with mild eyelid disfigurements, and to review eyelid cases with complications that had previously been described in the literature. A retrospective histopathologic study with Alcian blue, elastic, and Masson trichrome stains of 2 cases that were submitted to the Ocular Pathology Department was conducted, and a critical review of previously published cases of the histopathologic characteristics of dermal filler material in the periocular region was also conducted. Two periocular tissue samples were found to contain dermal filler material. In one case, porcine collagen appeared as amorphous or indistinctly microfibrillar aggregates that stained light blue with the Masson trichrome method. In the other case, hyaluronic acid gel appeared as vivid blue amorphous pools of material in extracellular locules after staining with the Alcian blue method. An inflammatory response was not observed in either case. Patients who undergo facial filler procedures may, at a later time, require a surgical excisional procedure from which a specimen is generated. Previously injected dermal filler that the patient neglected to mention may be present in the pathologic sample, potentially perplexing the unsuspecting pathologist. Both ophthalmic plastic surgeons and ocular pathologists should be aware of the histopathologic features of dermal fillers. It is helpful if a surgeon who submits a specimen to the pathology service makes note of any known prior use of facial filler material or is alert to its possible presence when unfamiliar foreign material is discovered in the dermis of the eyelids.
Wolkow N, Jakobiec FA, Yoon MK. Gelatin-Based Hemostatic Agents: Histopathologic Differences. Ophthal Plast Reconstr Surg 2018;Abstract
PURPOSE: To delineate the histopathologic appearance of gelatin-based hemostatic agents, Surgiflo, Gelfoam, and Floseal, which are used by ophthalmic plastic surgeons, and which may incidentally be found as foreign materials in histopathologic tissue samples. METHODS: Histopathologic analysis was performed with hematoxylin-eosin, periodic acid-Schiff, Masson trichrome, and elastin staining on tissue samples in which gelatin-based agents were found. To better characterize these materials, similar analyses were performed on in vitro samples of commonly used gelatin-based hemostatic agents. RESULTS: Surgiflo and Gelfoam are composed of small stellate pieces of gelatin with a smooth, homogeneous quality. In tissues, they are faintly positive with periodic acid-Schiff staining, amphophilic with Masson trichrome staining, and ink-black with elastin staining. Floseal has a distinctly different morphology of large rectangular sheets, yet almost identical in vitro staining properties. DISCUSSION: While the morphology of the gelatin-based hemostatic agents is consistent under various conditions, the staining properties of these materials differ based on whether they have been in contact with human tissue. CONCLUSIONS: Gelatin-derived hemostatic agents are best identified based on their morphologic characteristics. Elastin staining highlights these materials prominently within tissue samples and may be helpful in distinguishing them from other foreign materials.
Spors B, Seemann J, Homer N, Fay A. Lymphatic malformation with acquired Horner syndrome in an infant. J Neurointerv Surg 2018;10(3):e2.Abstract
An infant presented with right upper eyelid ptosis and was subsequently diagnosed with acquired Horner syndrome. Further evaluation revealed a right-sided cervicothoracic lymphatic malformation. At 13 weeks of age, the child underwent percutaneous intracystic sclerotherapy with a mixture of sodium tetradecyl sulphate and ethanol. Twenty-one weeks after initial treatment, ophthalmic examination showed complete resolution of the blepharoptosis and pupillary miosis. Percutaneous sclerotherapy not only effectively treated the space-occupying lymphatic malformation but also reversed the Horner syndrome that was presumably induced by neural tension (more likely) or compression.
Wolkow N, Jakobiec FA, Hatton MP. A Common Procedure With an Uncommon Pathology: Triamcinolone Acetonide Eyelid Injection. Ophthal Plast Reconstr Surg 2018;Abstract
Local corticosteroid injections are frequently employed by ophthalmologists to treat a variety of ocular, periocular, and orbital inflammatory conditions. Triamcinolone acetonide is a slowly dissolving crystalline corticosteroid that is often used for this purpose because of its prolonged anti-inflammatory effect. On occasion, previously injected corticosteroid material persists in tissues longer than anticipated, creating nodules that may masquerade as other disease conditions, or appearing incidentally in excised lesions on histopathologic examination. The histopathologic features of corticosteroid residues are unfamiliar to most ophthalmic pathologists and general pathologists. These features are described herein. Triamcinolone acetonide deposits in the skin appear as pale eosinophilic lakes of acellular frothy material on hematoxylin-eosin staining and are occasionally surrounded by a mild inflammatory reaction.
Mueller SK, Miyake MM, Lefebvre DR, Freitag SK, Bleier BS. Long-term impact of endoscopic orbital decompression on sinonasal-specific quality of life. Laryngoscope 2018;128(4):785-788.Abstract
OBJECTIVE: Endoscopic orbital decompression (EOD) is the workhorse surgical intervention for severe thyroid eye disease in Graves disease. Although EOD is a safe and effective procedure, the objective of this study is to determine the impact of orbital decompression on long-term sinonasal-pecific quality of life. METHODS: Retrospective study of 27 patients who underwent EOD by a single surgeon. The primary endpoint was change in preoperative 22-item Sinonasal Outcomes Test (SNOT-22) score at a minimum of 1 year. The secondary endpoint was to determine whether the performance of septoplasty for surgical access in patients without nasal obstruction impacted domain 1 (i.e., rhinologic domain) and total SNOT-22 scores. RESULTS: The mean follow-up was 25.7 ± 11.4 months. Domain 1 scores significantly increased at the first postoperative visit (P ≤ 0.01) and returned to baseline values between 1 and 3 months. At 1 year, significant improvements in both total score and domain 4 and 5 (psychological and sleep dysfunction, respectively) scores were seen (P < 0.01 for all scores). Septoplasty was not associated with a significant change in SNOT-22 score at 1 year (P = 0.48). CONCLUSION: Endoscopic orbital decompression is associated at 1 year with a significant improvement in sinonasal-specific quality of life, which is driven by the psychological and sleep dysfunction domains. Adjunctive septoplasty has no significant impact on SNOT-22 scores. LEVEL OF EVIDENCE: 4. Laryngoscope, 128:785-788, 2018.
Cohen LM, Elliott A, Freitag SK. Acquired Intermittent Pediatric Horner Syndrome due to Neuroblastoma. Ophthal Plast Reconstr Surg 2018;34(2):e38-e41.Abstract
A 3-month-old male developed intermittent left upper eyelid ptosis at the age of 1 month that was gradually increasing in frequency and duration. Examination revealed anisocoria and left upper and lower eyelid ptosis, consistent with a left Horner syndrome. Imaging showed a mass in the left superior posterior mediastinum, which was resected, and pathology was consistent with neuroblastoma. Eight months thereafter, the patient underwent left upper eyelid ptosis repair. Cases of infantile acquired Horner syndrome due to neuroblastoma are rare. To the authors' knowledge, there has only been one case described that presented with intermittent symptoms. The authors report the second case of intermittent acquired Horner syndrome due to neuroblastoma. This case demonstrates the importance of recognizing that Horner syndrome may present with subtle and intermittent symptoms. In a pediatric patient, one should maintain suspicion for neuroblastoma.
Pemberton JD, Salter M, Fay A, Thuro B, Spencer H, Dajani O. Investigation of goldmann perimetry in evaluation of patients for upper eyelid blepharoplasty. Orbit 2018;37(1):48-52.Abstract
PURPOSE: To determine if preoperative Goldmann Visual Field (GVF) testing in patients with functional dermatochalasis accurately depicts the postoperative superior visual field (SVF) outcome. METHODS: A prospective cohort study was done to compare preoperative and postoperative GVF field tests in patients undergoing upper eyelid blepharoplasty for treatment of dermatochalasis. This study was conducted in accordance with the Declaration of Helsinki and approved by the University of Arkansas for Medical Sciences institutional review board. A preoperative GVF was obtained with the eyelids in the natural position (untaped) and then again with excess skin elevated (taped). One month post-blepharoplasty, another GVF was conducted with eyelids untaped. The pre- and post GVF tests were analyzed to determine if preoperative testing accurately predicts the SVF improvement post-blepharoplasty. RESULTS: Forty-six eyelids (23 patients) who underwent blepharoplasty for dermatochalasis were included. The preoperative testing underestimated 76% (35/46) of cases by a mean of 61%; and overestimated the final outcome in 24% (11/46) of cases by mean of 23%. Overall, the preoperative GVF testing underestimated the postoperative outcome by a mean of 35%. CONCLUSION: Improvement of the SVF after a blepharoplasty is typically greater than the preoperative GVF testing predicts.
Mueller SK, Freitag SK, Lefebvre DR, Bleier BS. Endoscopic DCR using bipedicled interlacing mucosal flaps. Laryngoscope 2018;128(4):794-797.Abstract
OBJECTIVE: Obstruction of the nasolacrimal duct is a relatively common condition that affects patients of all ages, races, and sexes. The surgical gold standard for complete nasolacrimal duct obstruction and dacryocystitis is dacryocystorhinostomy (DCR). The purpose of this study was to describe a novel, bipedicled interlacing mucosal sparing flap technique for endoscopic DCR (eDCR). METHODS: A posteriorly based mucosal flap over the fundus is combined with a novel, anteriorly based mucosal flap over the intraosseus portion of the nasolacrimal duct (NLD). This exposes a wide area of the maxillary bone, allowing for exposure and identification of the NLD/sac complex in a safer, more inferior position. The interlacing mucosal flaps may be replaced at the conclusion of the procedure, thereby minimizing bone exposure and maintaining excellent long-term patency. RESULTS: The authors have utilized this technique in 55 procedures with 100% positive identification of the NLD and lacrimal sac, 0% complication rate, 100% anatomical patency rate, and 96.4% success rate after a minimal follow-up of 6 months. DISCUSSION: The bipedicled interlacing flap technique for eDCR provides for safe and reproducible identification of the NLD and lacrimal sac while minimizing bone exposure and restenosis rate. CONCLUSION: The bipedicled interlacing flap technique for eDCR provides for safe and reproducible identification of the NLD and lacrimal sac while minimizing bone exposure and restenosis rate. LEVEL OF EVIDENCE: NA. Laryngoscope, 128:794-797, 2018.
Wolkow N, Jakobiec FA, Yoon MK. Intratarsal Keratinous Eyelid Cysts in Gorlin Syndrome: A Review and Reappraisal. Surv Ophthalmol 2017;Abstract
A 38-year-old woman presented with multiple bilateral recurrent eyelid cysts. Her medical history was notable for Gorlin (nevoid basal cell carcinoma) syndrome. Histopathologic and immunohistochemical examinations revealed that the lesions were intratarsal keratinous cysts. They were similar in appearance to sporadic intratarsal keratinous cysts and closely resembled odontogenic keratocysts of the jaw. Eyelid cysts occur in up to 40 percent of patients with Gorlin syndrome; however, their description has been cursory and for the most part, outside of the ophthalmic literature. Although ophthalmologists are familiar with the periocular basal cell carcinomas that occur in patients with Gorlin syndrome, up to 10 percent of patients never develop a basal cell carcinoma, but may manifest other ophthalmic findings. Awareness of these other features may contribute to the earlier diagnosis of the syndrome. We discuss the clinical and histopathologic features of intratarsal keratinous cysts in Gorlin syndrome, comparing them to sporadic intratarsal keratinous cysts, other eyelid cysts, and jaw cysts that also characterize this syndrome. We briefly review the ocular and systemic manifestations of Gorlin syndrome and recent genetic and therapeutic developments so that the eyelid cysts may be appreciated within the appropriate context of Gorlin syndrome as a whole.