Meredith S. Baker

Methotrexate as an Alternative Treatment for Orbital Angiolymphoid Hyperplasia with Eosinophilia

Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon, benign disorder that presents as solitary or multiple nodules in the dermis or subcutaneous tissue. Orbital ALHE has been reported on occasion. We report a case of orbital ALHE which was refractory to systemic steroids but effectively treated with low-dose subcutaneous methotrexate. To our knowledge, this is the first reported case of methotrexate as a successful treatment for refractory ALHE.

Eyelid procedures in patients who have undergone Boston keratoprosthesis surgery.

Purpose: Artificial corneas or keratoprostheses such as the Boston keratoprosthesis (KPro) are being used more frequently to provide a clear corneal window in patients with severe corneal disease. A significant percentage of patients who undergo Boston KPro implantation require subsequent eyelid surgery. However, few articles in peer-reviewed literature evaluate the indication and outcome of eyelid procedures after Boston KPro implantation. This study examines the frequency, nature, and outcomes of oculoplastic procedures in patients with Boston KPro implantation. Methods: A retrospective chart review was conducted of all KPro-1 procedures performed at the University of Illinois at Chicago between December 2006 and September 2010 and all KPro-1 and KPro-2 procedures performed at the University of Iowa between December 2008 and October 2010. Results: One hundred and twenty eyes underwent Boston KPro-1 procedures, and 2 eyes underwent Boston KPro-2 procedures. Twenty-one (17.2%) of the 122 eyes required subsequent eyelid alterations. Chemical burn was the most common preoperative corneal diagnosis (8 of 21; 38.1%). A variety of oculoplastic procedures were performed; the most common procedure was a permanent lateral tarsorrhaphy. Seventeen (81.0%) of 21 KPro eyes that underwent oculoplastic procedures maintained the KPro at an average of 12.4 months of follow up. Conclusions: A significant number of patients with Boston KPros require subsequent eyelid surgery. With limited existing literature and increasing popularity for using Boston KPros to treat severe corneal disease, it is essential for oculoplastic and corneal surgeons to understand the need for eyelid alterations in these patients and the surgical intricacies surrounding these cases.

The Quantitated Internal Suture Browpexy: Comparison of Two Brow-lifting Techniques in Patients Undergoing Upper Blepharoplasty

Purpose: To describe and evaluate 1) the quantitated internal suture browpexy (ISB), which combines the advantages of a browpexy and “brassiere suture,” 2) the endoscopic Endotine browplasty, and 3) to compare these two techniques in patients undergoing simultaneous upper blepharoplasty. Methods: Retrospective review of patients undergoing bilateral upper blepharoplasty alone, bilateral ISB with upper blepharoplasty, and bilateral Endotine (MicroAire, Charlottesville, VA, U.S.A.) browplasty with upper blepharoplasty by one surgeon was performed. ImageJ 1.47v software (Wayne Rasband, National Institutes of Health, Bethesda, MD, U.S.A.) and standardized photographs were used to measure pre- and postoperative brow position at three positions (central, medial, lateral). Statistical analysis was performed using Sigmaplot version 12.5 for Windows (Systat Software, Inc., San Jose, CA, U.S.A.). Results: Thirty-three patients undergoing ISB with blepharoplasty, 33 undergoing Endotine browplasty with blepharoplasty, and 30 patients undergoing blepharoplasty alone were included. The 3 groups were matched for age and gender. Patients undergoing upper blepharoplasty alone showed statistically significant brow descent at all three brow positions (mean: -1.7 mm [p ⩽ 0.04]). The quantitated ISB prevented brow descent but provided minimal brow elevation (mean lateral elevation: right eye (OD) +1.3 mm [p = 0.03]; OS +0.9 mm [p = 0.08]). Endotine browplasty with upper blepharoplasty provided significant brow elevation at all brow positions, particularly laterally (OD +4.0 mm [p < 0.001]; OS +3.5 mm [p < 0.001]). There were no complications. Conclusions: Upper blepharoplasty alone is associated with brow descent; performing ISB simultaneously effectively prevents this descent. Endotine browplasty with upper blepharoplasty achieves significant brow elevation. Quantitation of the browpexy allows reproducible placement of the suture thereby producing consistent and symmetrical results.

Chronic anophthalmic socket pain treated by implant removal and dermis fat graft

Aims To report the outcome of orbital implant removal and dermis fat graft (DFG) implantation in patients with chronic anophthalmic socket pain (ASP), in whom all detectable causes of pain had been ruled out and medical management had failed. Methods Retrospective, multicentre case series. A review of all cases undergoing orbital implant replacement with DFG between 2007 and 2013 was conducted at the University of Iowa Hospitals and Clinics (UIHC), USA, and St. Erik Eye Hospital, Sweden. Inclusion criteria included (1) chronic ASP >2 years and unresponsive to treatment, (2) absence of pathological or structural cause for pain established by socket examination and orbital imaging, and (3) minimum 12-month post-surgical follow-up. Results Six cases with chronic ASP were identified, four were post-enucleation and two were eviscerated at an average age of 45 years. The incidence of chronic ASP among enucleations at UIHC over a 6-year period was 0.7%. Indications for enucleation and evisceration included tumours and glaucoma. Intractable ASP had been present for an average of 11 years and persisted despite medical management. All patients were free of pain within 3 months of implant removal and DFG placement and remained pain free at an average 24 months following surgery. Conclusions Orbital implant replacement with DFG was effective at relieving chronic ASP, and pain resolution was sustained in all cases. This surgical intervention may be a useful management option for patients in whom all detectable causes of chronic pain have been excluded and have failed medical pain management.

The hybrid lid crease approach to address lateral frontal sinus disease with orbital extension.

Objective: This study aimed to describe the hybrid lid crease approach in conjunction with functional endoscopic sinus surgery (FESS) for lateral frontal sinus disease with orbital extension. Study Design: Retrospective case review. Methods: Patients undergoing hybrid lid crease approach with FESS for frontal sinus disease were reviewed retrospectively. Surgical indications consisted of inverting papilloma with extension into the frontal sinus (n = 1) and frontal sinus mucocele (n = 2). Inclusion criteria included presence of disease in the lateral frontal sinus with extension into the orbital space and erosion of the superior orbital rim. Preoperative and postoperative parameters included complete ophthalmologic exam, endoscopic exam, and computed tomography scan. Results: We were able to access the frontal sinus and orbit in all 3 cases and address sinus pathology of the lateral frontal sinus and orbit using the lid crease approach with FESS. All patients had improvement in ophthalmologic symptoms and interval disease resolution and were satisfied with their postoperative lid crease incision. Conclusion: The lid crease approach offers direct access to the frontal sinus with minimal dissection through a well-hidden incision. In our case series of lateral frontal sinus pathology with orbital extension, the hybrid lid crease approach with FESS allowed complete eradication of disease without recurrence.

Epidemiology and Prognosis of Primary Periocular Sweat Gland Carcinomas

PURPOSE To characterize the clinical presentation, management, and outcomes of patients with sweat gland carcinoma of the periocular region. To review the pathologic classification scheme, compare the clinical behavior between subtypes, and discuss treatment recommendations for periocular sweat gland carcinomas. METHODS Retrospective study from 2 tertiary centers, which are part of the American Society of Ophthalmic Plastic and Reconstructive Surgery Oncology Database. RESULTS Twenty-two patients, of whom 13 (59%) were female, were included in the study. The median age was 69 years. The median follow-up time was 23 months. Twenty (91%) patients had lesions that were slow growing and were present for several months to years before diagnosis. Presentation was varied, and pathology included mucinous carcinoma (7; 32%), microcystic adnexal carcinoma (4; 18%), endocrine mucin-producing sweat gland carcinoma (4; 18%), eccrine carcinoma (2; 9%), apocrine carcinoma (2; 9%), poorly differential adnexal carcinoma (2; 9%), and hidroadenocarcinoma (1; 5%). Microcystic adnexal carcinoma tended to present with the largest tumor dimension (range: 25-32 mm) and mucinous carcinoma the smallest (range: 5-13 mm). Treatments included Mohs micrographic excision in 10 patients (45.5%), wide local excision in 10 patients (45.5%), and orbital exenteration in 2 patients (9%). No lesions ≤T2b (by AJCC 7th edition classification for eyelid carcinomas) had local recurrence or nodal metastasis. No patients had distant metastasis. CONCLUSIONS Sweat gland carcinomas tend to grow slowly and be present for several months to years before diagnosis. Overall, microcystic adnexal carcinoma subtype showed more aggressive clinical behavior than mucinous subtypes. Eyelid carcinomas AJCC category T2b or less were associated with better outcomes.

Immunopathology and histopathology of conjunctival biopsies in patients with presumed idiopathic punctal stenosis

Background/aims Numerous processes have been implicated as causes of punctal stenosis. Recent studies have highlighted inflammation in punctoplasty specimens in patients with punctal stenosis. Conjunctival biopsy has been suggested as a means to determine underlying aetiologies, although little is known regarding what conjunctival pathologies are associated with punctal stenosis. Our objective is to examine the pathological and immunological findings in conjunctival biopsy specimens in patients with presumed idiopathic punctal stenosis. Methods A retrospective chart review was performed at a single institution over a 5 year period of patients with presumed idiopathic punctal stenosis who underwent conjunctival biopsy for histopathological and/or direct immunofluorescence (DIF) examination. Patients with known aetiologies of punctal stenosis were excluded. Results 23 patients met inclusion criteria. Conjunctival biopsies (n=36) from all 23 patients underwent histological examination. 35 specimens (97.2%) showed lymphocytic infiltrates. Two patients (8.7%) had findings suggestive of sarcoidosis. Conjunctival biopsies from 18 of the 23 patients were also evaluated by DIF. Nine patients (50.0%) had fibrinogen characteristics suggestive of lichen planus. There were no complications related to the conjunctival biopsy procedures. Conclusions Conjunctiva appears to be an excellent proxy in evaluating patients with presumed idiopathic punctal stenosis. The biopsy is safe and provides ample tissue. Clinically, abnormal tissue should be targeted, if available. 11 of the 23 patients in this study demonstrated findings consistent with underlying immunological disorders, suggesting that conjunctival biopsy may play a role in identifying underlying aetiologies of punctal stenosis. Knowledge of these underlying conditions impacted treatments for several patients.

Support for the Egress Mechanism of Optic Nerve Sheath Fenestration.

Ophthal Plast Reconstr Surg, Vol. 32, No. 3, 2016 e75 A 26-year-old woman with idiopathic intracranial hypertension underwent bilateral optic nerve sheath fenestration (ONSF) for persistent papilledema and progressive visual field loss in both eyes despite maximal medical therapy. Three months later, she had persistent papilledema on the right and no papilledema on the left. An MRI of the orbits was performed. The MRI (noncontrast T2-weighted) shows a fluid collection adjacent to the area of ONSF on the left. High signal adjacent to the left ONSF (arrow), which was performed through a superiormedial eyelid crease, indicates fluid egress (Fig. 1). This finding supports the proposed egress mechanism of ONSF (reduction of pressure at the optic nerve head due to development of an open fistula or a bleb-like cyst at the area of the fenestration) rather than the scarring mechanism of ONSF (reduced transmission of increased intracranial pressure to the optic nerve head due to fibrosis or scarring in the area of the ONSF).

Keloidal scarring in hard palate grafts

Hard palate grafts are a commonly accepted treatment for lower eyelid retraction and are typically associated with low complication rates. The authors present a case of bilateral keloidal scarring within hard palate grafts requiring surgical excision. A 72-year-old woman who had undergone bilateral hard palate grafts to her lower eyelids for lower eyelid retraction was referred for the evaluation of ocular irritation. She was diagnosed with bilateral hypertrophy of the grafts with secondary entropion. The grafts were excised; the pathology was consistent with keloidal scarring. To the authors’ knowledge, there is no previously reported case of bilateral keloidal scarring in normal donor tissue, which required excision of the hard palate grafts. It is important for oculoplastic surgeons to be aware of this potential complication when considering, discussing, and consenting patients for eyelid reconstruction with hard palate grafting.

Orbital sarcomas in retinoblastoma patients: Recommendations for screening and treatment guidelines

Purpose of review Retinoblastoma is the most common primary ocular malignancy in children. Although currently retinoblastoma has an excellent survival rate in developed countries, hereditary retinoblastoma survivors as well as those with a history of radiation therapy as children are at an increased risk for second primary tumors (SPTs), and specifically, for orbital sarcomas. Despite the known increased risk for SPTs in retinoblastoma survivors and the associated morbidity and mortality, no screening or treatment guidelines exist. Recent findings Understanding of retinoblastoma tumorigenesis and genomic expression has expanded significantly, and treatment has evolved with a shift away from radiotherapy. Until the last two decades, however, radiation was the therapy of choice for patients with bilateral disease. Because both hereditary retinoblastoma and radiation are independent risk factors for the development of SPTs such as sarcomas and these SPTs are often fatal, appropriate surveillance for retinoblastoma survivors is crucial. Summary As a result of the excellent survival rates for retinoblastoma patients, it is important to: recognize the risk of sarcoma, particularly in patients with hereditary retinoblastoma and/or prior radiation therapy; establish a screening protocol, such as the one proposed, to maximize early detection; and discuss and develop treatment guidelines for high-risk patients. Future directions of research for these patients will involve the development of molecularly targeted agents and the use of proton radiotherapy.