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PubMed2004

Advances in applied anatomy of the eyelid and orbit.

PURPOSE OF REVIEW: The oculofacial surgeon seeking to achieve ideal outcomes and avoid complications in aesthetic, functional and reconstructive surgery must have an understanding of the three-dimensional anatomic features, as well as the dynamic aspects, of the eyelid and orbital region. Knowledge of the orbital and periorbital region may be the last frontier of human anatomy, as new information is still being uncovered every year and each clarification of anatomy leads to a new level of sophistication in the understanding and treatment of orbital and eyelid disease both medically and surgically. RECENT FINDINGS: Recently, a great deal of attention has been focused on better understanding of the vasculature, fatty tissue, fascial attachments, and lymphatic drainage of the orbital and periorbital region. This in turn has provided improved clinical understanding of orbital disease and surgical applications of these findings have been described. SUMMARY: Anatomic knowledge in ophthalmology is not yet complete but constantly evolving. Exciting new insights into treatment of ophthalmologic disease have resulted from the application of an ever greater anatomic understanding of the orbital and periorbital region.

Anatomy, Cross-Sectional↗
PubMed2003

Exophthalmos and orbital floor thickening related to maxillary sinusitis.

A 59-year-old woman presented with periocular pain and 1.5 mm of relative proptosis. A computed tomography scan demonstrated thickening of the orbital floor, and an endoscopic sinusectomy revealed chronic sinusitis. Although cases of maxillary sinus disease with orbital floor thickening have been reported, these findings are in contrast to cases of silent sinus syndrome, in which maxillary sinus disease induces thinning and inferior displacement of the orbital floor with secondary enophthalmos and hypoglobus. Thus, while chronic maxillary sinus disease may create negative antral pressure and inferior displacement of the orbital floor, whether orbital floor thinning or thickening occurs varies with each case. The present case represents a combination of maxillary sinusitis with orbital floor thickening and exophthalmos, a less common manifestation of maxillary sinus-related orbitopathy.

Chronic Disease↗
PubMed2003

Orbital xanthogranuloma: clinical and morphologic features in eight patients.

PURPOSE: To describe the clinical and morphologic features of patients with orbital xanthogranuloma (XG) with or without Erdheim-Chester disease (E-Cd). METHODS: Retrospective, noncomparative case series. A review of 8 consecutive histopathologically proven cases of orbital XG from 3 medical centers. Four male and four female patients with ages ranging from 23 to 79 years presented with 4 bilateral and 4 unilateral orbital XGs. RESULTS: Age at diagnosis, ocular and systemic manifestations, histopathologic and radiologic features, type of treatment, and prognosis were evaluated for each patient. Six of 8 patients had proptosis and 2 presented with afferent pupillary defect and severe extraocular motility limitations. Other signs and symptoms included eyelid retraction, mechanical ptosis, and chemosis. Planar xanthomas of eyelids were present in 3 individuals. CT and MRI showed infiltrating soft tissue masses within the orbit in 7 and 2 patients, respectively. Histopathology revealed proliferation of foamy histiocytes intermingled with Touton and multinucleated giant cells and lymphocytes. The absence of Birbeck granules within the histiocytic elements of the tumor, indicating that the cell of origin is a non-Langerhans histiocyte, was documented with electron microscopy in 3 cases. The most common treatment was surgical excision combined with oral corticosteroids. Two patients with E-Cd with involvement of the long bones of the upper and lower extremities and retroperitoneal region died of kidney failure within approximately 1 year of diagnosis. CONCLUSIONS: Orbital XG is a proliferative lesion of the non-Langerhans histiocytes, which may present as a solitary orbital lesion or may be associated with a systemic condition known as E-Cd with very poor prognosis.

Adult↗
PubMed2003

Use of bioglass for orbital volume augmentation in enophthalmos: a rabbit model (oryctolagus cuniculus).

PURPOSE: To investigate the clinical and histologic response of Novabone-C/M as an osteoproductive alloplastic implant for volume augmentation in the orbit in the treatment of enophthalmos and to compare its outcome alone versus its use in combination with autogenous bone or Medpor granules. METHODS: Novabone-C/M, a bioactive silicone glass material, was implanted in the subperiosteal space of the left orbit of 12 New Zealand White rabbits. The animals were divided into 3 groups, each with 4 animals, based on the material implanted in the orbit: group 1, Novabone alone; group 2, Novabone plus Medpor granules; and group 3, Novabone plus autogenous bone fragments. All rabbits were studied clinically, radiographically, and histologically at 1-, 3-, and 6-month intervals. Animals underwent preoperative and postoperative computed tomography (CT) with 3-dimensional reconstruction, proptosis measurements, and volumetric analysis. Orbit specimens were studied histologically with mineralized bone stain (MIBS) to look for bone formation, reactivity, infection, implant resorption, and migration. RESULTS: There were no signs of significant inflammation or infection. Subcutaneous migration of the implant was seen radiographically but not clinically in groups 1 and 3. Induced proptosis averaged 2.5 mm (at 1 month) and showed regression in all groups over a 6-month period but was not statistically significant. Implant volume was markedly reduced in all groups, averaging 69% in group 1, 37% in group 2, and 59% in group 3 at 6 months. New bone formation and bone remodeling was present in all 3 groups at 3 months and only in group 2 at 6 months. The rate and amount of implant remodeling and bone formation was greatest in the Novabone/Medpor group (group 2). CONCLUSIONS: Bioglass particulate is biocompatible, easy to use in the orbit, and stimulates bone growth. Bioglass is associated with volume loss and migration over 6 months and may not provide adequate volume augmentation in the orbit when used alone for the treatment of enophthalmos. The duration and amount of bone formation may be enhanced when Novabone is used in conjunction with Medpor.

Animals↗
PubMed2004

Coralline hydroxyapatite orbital implant (bio-eye): experience with 158 patients.

PURPOSE: To assess the problems seen in 158 patients with coralline hydroxyapatite (HA) orbital implants (Bio-Eye). METHODS: A consecutive case series of 170 patients receiving coralline HA implanted by two surgeons over a 5-year period were reviewed. The authors analyzed age, type of surgery, implant size, peg system, follow-up duration, time of pegging, problems encountered, and treatment. RESULTS: Twelve patients were lost to follow-up after 5 months, leaving 158 patients who were followed from 6 to 130 months (average, 39 months). Problems in unpegged implants occurred in 36 (22.8%) patients. Discharge occurred in 18 (11.4%) patients, implant exposure in 12 (7.6%), socket discomfort in 1 (0.6%), conjunctival thinning in 3 (1.9%), chronic conjunctival swelling in 2 (1.3%), and implant infection in 3 (1.9%). Problems after pegging occurred in 68 (50.7%) of 134 patients: discharge in 27 (20.1%), pyogenic granuloma in 24 (17.9%), conjunctiva overgrowing the peg in 4 (3.0%), implant exposure around the sleeve in 5 (3.7%), clicking in 6 (4.5%), peg on an angle in 2 (1.5%), loose sleeve in 1 (0.7%), peg falling out in 18 (13.4%), popping peg in 1 (0.7%), poor transfer of movement in 3 (2.2%), pain with movement in 1 (0.7%), and implant infection in 2 (1.5%). CONCLUSIONS: The Bio-Eye orbital implant represents a porous orbital implant that is biocompatible with orbital tissues and allows fibrovascular ingrowth and improved motility when coupled to the overlying artificial eye. It is more expensive than other commercially available porous orbital implants, such as synthetic FCI3 HA, porous polyethylene (Medpor), and aluminum oxide (Bioceramic) implant. Problems encountered with its use are similar to those problems seen in patients with the synthetic FCI3 hydroxyapatite and aluminum oxide orbital implants.

Adolescent↗
PubMed2004

Development of an orbital endoscope for use with the free electron laser.

PURPOSE: To explore the feasibility of designing, constructing, and testing an orbital endoscope for use with the free electron laser (FEL). METHODS: An experimental study with an author-designed laser delivery system through an endoscope was conducted. Two adult pig cadavers and 6 fresh human cadavers had orbital endoscopy performed to develop a method of optic nerve sheath fenestration (ONSF) with the FEL. Twelve orbits were used to develop the surgical procedure by comparing visualization media and surgical technique. In the first 7 trials, a different variable was changed; the procedure was then refined in the human cadaver experiments. An ONSF was performed with the FEL (6.45 microm, 30 Hz, 2 to 3 mJ, 250-microm spot size) through a glass hollow wave guide introduced through an endoscope in 4 human cadaver orbits. RESULTS: Visualization of the orbital structures was clearest with carbon dioxide; sodium hyaluronate did not displace the fat, and saline hydrated the orbital fat. Biopsy forceps alone did not produce a dural window in the four trials that used the forceps. A dural window was made by using the FEL through a glass hollow wave guide adapted to the Olympus HYF-XP endoscope in the second, third, fifth, and sixth human trials with the FEL. Histologic evidence of the ONSF was produced. CONCLUSIONS: A hollow wave guide capable of transmitting the FEL through an endoscope was successfully constructed. ONSF with the FEL applied through an endoscope is technically feasible. Additional studies are currently examining techniques to improve intraorbital endoscopy.

Animals↗
PubMed2004

Manifestations of fungal cellulitis of the orbit in children with neutropenia and fever.

PURPOSE: To delineate clinical manifestations of fungal orbital cellulitis in immunocompromized patients. METHODS: The charts of 7 pediatric patients with fungal orbital cellulitis treated at a tertiary children's cancer hospital were reviewed retrospectively for histologically confirmed fungal sinusitis with associated orbital cellulitis. Patients underwent CT and/or MRI of the orbits, sinuses, and brain; surgery; and therapy with antifungal medications. Main outcome measures were presenting signs and patient survival. RESULTS: Twenty-four patients with fungal sinusitis were identified, 7 of whom (4 months to 15 years of age) had documented orbital fungal cellulitis. All 7 patients presented with neutropenia and fever. Presenting symptoms included edema of the upper eyelid (n=4), headache (n=1), and facial pain (n=1). One patient was asymptomatic. Although antifungal therapy was initiated within 24 hours of presentation, disease progressed, and 5 patients eventually died of their infections. CONCLUSIONS: Because fungal orbital cellulitis can be fatal even if detected early in patients who are immunocompromised, ophthalmologists and otolaryngologists should be alert to the disease's subtle clinical manifestations.

Adolescent↗
PubMed2004

Fibrovascular ingrowth into hydroxyapatite and porous polyethylene orbital implants wrapped with acellular dermis.

PURPOSE: Acellular dermis is a frequently used wrapping material for hydroxyapatite (HA) and porous polyethylene (PP) orbital implants. In an animal model, we determined by histology the extent of fibrovascular ingrowth within orbital implants wrapped in acellular dermis at 6 and 12 weeks after surgery. METHODS: Four Yucatan minipigs were used for the study. Two minipigs had HA implants and two had PP implants. Implants were harvested at 6 or 12 weeks after surgery and were examined histologically for fibrovascular ingrowth. RESULTS: There was complete fibrovascularization of HA implants harvested at both 6 and 12 weeks after surgery. The PP implant harvested at 6 weeks had incomplete fibrovascularization, whereas the PP implant harvested at 12 weeks had complete fibrovascular ingrowth. There was no histologic evidence of inflammation seen in any of the orbital implants. On gross and histologic examination, the wraps were found to persist on the surface of all orbital implants, with little histologic evidence of inflammation localized to the acellular dermis. CONCLUSIONS: Acellular dermis wraps support fibrovascularization of both HA and PP orbital implants. Additionally, acellular dermis does not incite significant inflammation in association with HA and PP orbital implants and can persist in situ for at least 12 weeks after surgery.

Animals↗
PubMed2005

Behavior of various orbital implants under axial compression.

PURPOSE: To determine and compare the amount of force required to disrupt the integrity of various orbital implants. METHODS: Compression tests were carried out by using a servo-electrical universal testing system on orbital implants including aluminum oxide (Bioceramic implant, FCI, Issy-Les-Moulineaux, France), coralline hydroxyapatite (HA) (Bio-Eye, Integrated Orbital Implants, Inc., San Diego, CA, U.S.A.), bovine HA (Molteno M-Sphere, IOP Inc., Costa Mesa, CA, U.S.A.), synthetic HA (FCI3, FCI, Issy-Les-Moulineaux, France), Chinese HA (H + Y Comprehensive technologies, Philadelphia, PA, U.S.A.), polylactic acid (Kinsey Nash Corporation, Duluth, MN, U.S.A.), porous polyethylene (Medpor, Porex Surgical Inc., College Park, GA, U.S.A.), and polymethylmethacrylate. RESULTS: Two basic groups of implants were identified: those that eventually reach a critical compression point and collapse (coralline HA, aluminum oxide, synthetic FCI3 HA, bovine HA, Chinese HA, and polymethylmethacrylate), and those that do not collapse but gradually compress with increasing load (porous polyethylene, polylactic acid). For similar-sized implants, the critical collapse point was earliest for the FCI3 HA implant, followed by the coralline HA, aluminum oxide, and polymethylmethacrylate implants. Smaller-sized collapsible implants showed earlier critical collapse points than larger-sized implants of similar material. CONCLUSIONS: A technique was established to assess the force required to disrupt the integrity of various orbital implants that is reliable, unbiased, and repeatable with any orbital implant. Orbital implants of different materials and sizes demonstrate different degrees of integrity. It is important to use similar-sized implants when comparing the integrity of different implant materials because size influences the force required to overcome the structural integrity of the implant.

Aluminum Oxide↗
PubMed2005

Orbital invasion by esthesioneuroblastoma.

PURPOSE: Esthesioneuroblastoma is a rare malignancy arising from the olfactory mucosa in the nasal fossa, which can invade the orbit producing ophthalmic symptoms and signs. The purpose of this study was to present a case series and review the literature to assess the characteristics of orbital involvement by esthesioneuroblastoma. METHODS: Retrospective case series of all patients treated for biopsy-proven esthesioneuroblastoma at the Royal Adelaide Hospital between 1992 and 2004. RESULTS: Nine patients (5 male, 4 female) with a mean age of 50 years (range, 20 to 66 years) were reviewed. One case was classified as Kadish stage A, 1 as Kadish stage B, and 7 as Kadish stage C. Mean time from onset of symptoms to diagnosis was 17 months (range, 2 to 24 months). Radiologic orbital invasion was present in 4 cases and was associated with ophthalmic symptoms or signs in 3 of these cases. These included proptosis (2 cases), periorbital pain (1 case), decreased visual acuity (1 case), extraocular muscle restriction (1 case), and chemosis (1 case). One patient with symptoms secondary to orbital invasion was initially referred to and assessed by an ophthalmologist. All patients who had or went on to have development of orbital invasion had advanced disease at diagnosis. Treatment was surgical (9/9), with the addition of radiotherapy (6/9) and chemotherapy (2/9). Mean follow-up was 3.6 years (range, 0.5 to 8.5 years). Six of 9 patients had tumor recurrence. Two had metastasis. Five of 9 patients remained alive. CONCLUSIONS: Orbital invasion by esthesioneuroblastoma is not uncommon. It is important to be aware of this malignancy because a significant proportion of patients will present with ophthalmic signs and symptoms.

Adult↗
PubMedDate not supplied

Ossifying fibromyxoid tumor of the orbit.

PURPOSE: This report describes the clinical, radiographic, and histopathologic features of the first known case of ossifying fibromyxoid tumor involving the orbit. METHODS: Case report and literature review. RESULTS: An 81-year-old woman presented with a 3-month history of diplopia, pain, and right upper eyelid swelling. CT and MRI of the orbit disclosed a 2.5-cm extra-conal mass superomedially in the right orbit, with displacement of the globe inferiorly and remodeling of the orbital roof. During surgery, the tumor was noted to be firm, gritty, and extra-periosteal in location, and the bone adjacent to the tumor was irregular and abnormal in consistency. The tumor was removed intact without difficulty. Histologic, immunohistochemical, and transmission electron microscopic examination disclosed features consistent with ossifying fibromyxoid tumor. Fourteen months after removal, the tumor recurred and the patient underwent a second excisional biopsy. The tumor recurred a second time 30 months after the first surgery. The patient underwent a third surgery to remove the tumor and received adjuvant radiation therapy. She has been free of disease for more than 18 months. CONCLUSIONS: This is the first known case of ossifying fibromyxoid tumor involving the orbit. Increased cellularity, nuclear atypia, and mitotic activity have been associated with metastatic and recurrent potential. The tumor in this case had moderate cellularity, low nuclear grade, and no mitotic figures. However, the close proximity of the tumor to important structures within the orbit precluded its excision with wide margins and may have predisposed the patient to the two episodes of local recurrence.

Aged, 80 and over↗
PubMedDate not supplied

Mesenchymal chondrosarcoma of the orbit.

PURPOSE: To describe a patient with mesenchymal chondrosarcoma of the orbit, review clinical and imaging findings, and refine the differential diagnosis from other tumors of the orbit. METHODS: Interventional case report and literature review. RESULTS: A 21-year-old woman with a 6-month history of progressive proptosis presented with left-sided visual loss of recent onset. CT revealed a clearly outlined heterogeneous mass with calcified foci, whereas MRI showed an isointense signal to gray matter on T1- and T2-weighted images. The patient was diagnosed with mesenchymal chondrosarcoma of the orbit only after orbitotomy and subsequent histopathologic study. After surgery, she received adjuvant therapy with irradiation of the orbit. CONCLUSIONS: Mesenchymal chondrosarcoma is a rare malignant tumor of the orbit capable of mimicking several other lesions. Early diagnosis requires a high level of suspicion, particularly with regard to internally calcified lesions. The treatment of choice is complete surgical resection, or, when the tumor is not resectable or residual mass is present after surgery, chemotherapy and radiotherapy. Prognosis for patients with orbital mesenchymal chondrosarcoma is tentative at best, in part because of the rarity of the lesion.

Adult↗
PubMedDate not supplied

Spontaneous orbital and periocular hemorrhage in a patient with epithelioid hemangioma.

Epithelioid hemangioma, or angiolymphoid hyperplasia with eosinophilia, is a benign vascular lesion that infrequently involves the orbit. We report a case of spontaneous orbital and periocular hemorrhage in a patient with epithelioid hemangioma of the orbit. Ophthalmic examination was accompanied by CT and MRI, followed by histopathologic diagnosis. The patient presented with headache, eyelid bruising, and double vision. Ophthalmic examination showed ecchymosis, proptosis, and limited extraocular movements of the affected eye. Imaging studies showed a cystic right orbital mass. Histologic examination showed plump endothelial cells accompanied by lymphocytes, plasma cells, and eosinophils. A diagnosis of epithelioid hemangioma was rendered. To our knowledge, this is the first reported case of spontaneous orbital and periocular hemorrhage associated with epithelioid hemangioma. Epithelioid hemangioma should be considered in the differential diagnosis for patients presenting with spontaneous orbital or periocular hemorrhage.

Adult↗
PubMed2006

Orbital sequelae of rhinosinusitis after cochlear implantation in children.

OBJECTIVES: The objectives of this retrospective case review were to describe orbital complications in children after cochlear implantation, to define rhinosinusitis as a possible preoperative risk factor, and to suggest a possible pathophysiological mechanism for this previously unreported occurrence. METHODS: Records of children undergoing cochlear implantation over a 7-year period at a tertiary academic medical center were reviewed. Four children who experienced postoperative orbital sequelae were identified. We describe the demographics, clinical course, and radiologic findings in these children. RESULTS: The records of 91 children who underwent cochlear implantation were reviewed. The mean age was 6.0 years (range, 0.9-16.9 years). Forty-nine children (54%) were female and 51 (56%) were white. Four children developed postoperative orbital complications on the ipsilateral side to implantation. Orbital complications were characterized by periorbital edema and preseptal cellulitis necessitating prolonged hospitalization in all four children (mean length of stay, 3.3 days). Each child's orbital complication resolved with medical therapy that included intravenous antibiotics and nasal saline. Temporal bone images before implantation showed evidence of rhinosinusitis in all four cases. Of 76 available preoperative scans from the unaffected children, only 11 (14%) studies showed evidence of rhinosinusitis. CONCLUSIONS: Children with preoperative radiologic evidence of rhinosinusitis may be at risk of orbital sequelae after cochlear implantation. Positioning of the patient during surgery, length of surgery, and minor trauma to the lamina papyracea during drilling of the mastoid may be important etiologic factors. A careful review of medical history and computed tomography imaging before implantation may identify at-risk children.

Adolescent↗
PubMed2006

Use of calcium phosphate cement paste in orbital volume augmentation.

BACKGROUND: The development of calcium phosphate in cement paste form has made its application simplified and easy. In this study, the authors used an alpha-tricalcium phosphate/dicalcium phosphate dibasic/tetracalcium phosphate monoxide/hydroxyapatite injectable calcium phosphate cement paste to evaluate its potential use in orbital volume augmentation. METHODS: Five New Zealand white rabbits were used in this study. In each rabbit, the right orbit was directly implanted with calcium phosphate cement in its injectable paste form, and a prehardened form was placed in the left orbit. The orbital roof was approached through a subciliary skin incision and the implant was placed posterior to the globe of the eyeball to push it outward. Measurement of proptosis and intraocular pressure was monitored before and after implantation. The animals were killed after 3 months, and orbit bone-implant samples were taken for histology and microradiography. RESULTS: In both groups, proptosis was induced, 4.2 +/- 0.27 mm in the prehardened group and 3.8 +/- 0.22 mm in the injectable group. These values taken 1 week postimplantation were unchanged until the end of the experiment and were maintained without significant intraocular pressure changes. The implants were well tolerated, and no sign of infection, extrusion, or migration was noted. Histologic analysis showed good biocompatibility and osteoconductivity, and microradiography has confirmed a well-set cement with direct bone union to it. CONCLUSION: These findings therefore indicate that calcium phosphate cement implant, when used as an injectable paste or in its prehardened form, can be a safe, effective material for orbital volume augmentation.

Animals↗
PubMed2002

Computation and visualization of three-dimensional soft tissue motion in the orbit.

This work presents a method to measure the soft tissue motion in three dimensions in the orbit during gaze. It has been shown that two-dimensional (2-D) quantification of soft tissue motion in the orbit is effective in the study of orbital anatomy and motion disorders. However, soft tissue motion is a three-dimensional (3-D) phenomenon and part of the kinematics is lost in any 2-D measurement. Therefore, T1-weighted magnetic resonance (MR) imaging volume sequences are acquired during gaze and soft tissue motion is quantified using a generalization of the Lucas and Kanade optical flow algorithm to three dimensions. New techniques have been developed for visualizing the 3-D flow field as a series of color-texture mapped 2-D slices or as a combination of volume rendering for display of the anatomy and scintillation rendering for the display of the motion field. We have studied the performance of the algorithm on four-dimensional volume sequences of synthetic motion, simulated motion of a static object imaged by MR, an MR-imaged rotating object and MR-imaged motion in the human orbit during gaze. The accuracy of the analysis is sufficient to characterize motion in the orbit and scintillation rendering is an effective visualization technique for 3-D motion in the orbit.

Algorithms↗
PubMed2005

Expression of FGFR-2 and FGFR-3 in the normal human fetal orbit.

AIMS: To demonstrate the expression patterns of two fibroblast growth factor receptors (FGFR-2 and FGFR-3) in the normal human fetal orbit. METHODS: 6 microm orbital slide sections were prepared from 12 week old human fetal material obtained within established ethical guidelines. Radioactive in situ hybridisation techniques were used to demonstrate the expression patterns of FGFR-2 and FGFR-3 within these sections. Only one foetus had appropriate orbital sections taken. RESULTS: FGFR-2 was expressed within the extraocular muscles (EOMs) and the optic nerve sheath and to a lesser degree within the orbital periosteal margins and the cranial sutures. FGFR-3 was expressed a lot within the periosteal margins and cranial sutures but not within either the EOMs or the optic nerve sheath. CONCLUSIONS: FGFR-2 and FGFR-3 are differentially expressed within different orbital components. FGFR-2 gene mutations may be responsible for craniosynostotic syndromes such as Crouzon, Pfeiffer, and Apert, while those in the FGFR-3 gene may cause isolated unicoronal synostosis. EOMs may be histologically abnormal in cases of Apert, Pfeiffer, and Crouzon syndromes but not isolated unicoronal synostosis. The pattern of expression of FGFR-2 in the normal human fetal orbit may explain some of the EOM histological findings seen in some cases of Apert, Pfeiffer, and Crouzon syndromes.

Fibroblast Growth Factor 2↗
PubMed2001

A role for methotrexate in the management of non-infectious orbital inflammatory disease.

AIM: To evaluate the clinical usefulness of methotrexate for patients with non-infectious orbital inflammatory disease who fail to respond to systemic corticosteroids and/or orbital irradiation. METHODS: The medical records of patients with non-infectious orbital inflammatory disease who were treated with methotrexate at Oregon Health Sciences University between June 1993 and June 2000 were examined. Methotrexate was administered at a median maximum dose of 20 mg per week (range 15-25 mg per week) in conjunction with folate supplementation. Patients were followed with regular ophthalmic examinations, as well as serum liver enzyme levels and blood cell counts. Clinical signs of regression of the orbital inflammation, visual acuity, dosage and duration of methotrexate therapy, requirement for concurrent corticosteroid administration, and adverse drug reactions were recorded. RESULTS: The study cohort included 14 patients (24 eyes) with diagnoses including non-specific orbital inflammation (n=7), Tolosa-Hunt syndrome (n=1), thyroid orbitopathy (n=3), Wegener's granulomatosis (n=1), sarcoidosis (n=1), and Erdheim-Chester disease (n=1). In all cases, methotrexate was commenced as a corticosteroid sparing agent. 10 patients (71%) completed a 4 month therapeutic trial of methotrexate. Median duration of treatment for the nine (64%) patients who experienced clinical benefit was 25 months (range 10-47 months). Six responders were ultimately able to cease methotrexate, including the single patient who required concurrent long term corticosteroid therapy. Complications included fatigue, gastrointestinal disturbance, hair thinning and mild, reversible serum liver enzyme elevation. Two patients (14%) discontinued treatment because of adverse effects. CONCLUSION: Methotrexate is a well tolerated immunosuppressive medication which may benefit patients with recalcitrant non-infectious orbital inflammatory disease.

Adult↗