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Paul A Brannan - One of the best experts on this subject based on the ideXlab platform.

  • A review of sclerosing idiopathic Orbital Inflammation.
    Current opinion in ophthalmology, 2007
    Co-Authors: Paul A Brannan
    Abstract:

    Purpose of review Sclerosing idiopathic Orbital Inflammation is a rare, distinct subset of Orbital Inflammation. No consistent therapeutic regimen has been described and the majority of reports on this disease are case reports or small case series. This review looks at historical and current treatment modalities in an attempt to define the most current, effective management strategy for this disease. Recent findings Recently, the largest review to date of these patients was published, detailing clinical findings and responses to the various interventions. Two other case reports of patients who responded completely to therapy appeared in the literature. Summary Sclerosing idiopathic Orbital Inflammation is a rare disease with an unknown pathogenesis and poor prognosis. Current treatments are disappointing and often have little beneficial effect. There are anecdotal reports of success using combinations of immunosuppression, radiotherapy, and surgery, but a consistent, effective treatment course is lacking. Younger patients and those with a shorter duration of disease seem to have the best prognosis. Further research into the immunologic basis of the disease and a large, controlled study comparing the various sclerosing idiopathic Orbital Inflammation treatments are needed to determine the most effective treatment regimen.

  • Sclerosing idiopathic Orbital Inflammation.
    Journal of pediatric ophthalmology and strabismus, 2006
    Co-Authors: Paul A Brannan, Robert C. Kersten, Dwight R. Kulwin
    Abstract:

    A 5-year-old girl referred for Orbital cellulitis was found to have a right Orbital mass. Computed tomography revealed a mass occupying the inferotemporal orbit, extending into the maxillary sinus. Biopsy yielded a diagnosis of sclerosing idiopathic Orbital Inflammation. She was successfully treated with prednisone.

Geoffrey E Rose - One of the best experts on this subject based on the ideXlab platform.

  • Orbital Inflammation: biopsy first
    Survey of ophthalmology, 2016
    Co-Authors: Ilse Mombaerts, Geoffrey E Rose, James A. Garrity
    Abstract:

    Orbital Inflammation is a response of the immune system and not a diagnosis in itself. Exposing the underlying disease introduces a labyrynthine challenge owing to the broad array of possible causes ranging from infectious, structural, autoimmune, idiopathic to neoplastic origin. In this regard, and despite its unkown etiology, idiopathic Orbital Inflammation (IOI) intrinsically is a genuine diagnostic entity. Where clinical and radiological findings of an Orbital inflammatory mass are inconclusive, pathological examination of the tissue biopsy-obtained by minimally invasive approach and local anesthesia-is advocated to work towards a diagnosis in a most timely and effective manner. A corticosteroid response can be observed in most Orbital disorders with lymphocytic components, including IOI, and, accordingly, constitutes a paradoxical and weak tool to identify the diagnosis in Orbital Inflammation.

  • idiopathic Orbital Inflammation a new dimension with the discovery of immunoglobulin g4 related disease
    Current Opinion in Ophthalmology, 2012
    Co-Authors: Antonella Berrybrincat, Geoffrey E Rose
    Abstract:

    Purpose of reviewTo review idiopathic Orbital Inflammation, with particular reference to the evolving entity of ocular adnexal immunoglobulin G4 (IgG4) disease.Recent findingsEmerging evidence suggests that sclerosing idiopathic Orbital Inflammation can be an IgG4-related disease and discovery of th

  • Intraocular malignant melanomas presenting with Orbital Inflammation
    Eye, 1993
    Co-Authors: Geoffrey E Rose, H Bing Hoh, Richard A. Harrad, John L. Hungerford
    Abstract:

    Symptoms and signs of Orbital Inflammation may be prominent features in the presentation of choroidal malignant melanoma, even when the tumour is solely intraocular. Three cases with this clinical presentation are described.

Bulent Yazici - One of the best experts on this subject based on the ideXlab platform.

  • Rituximab Therapy for Recalcitrant Idiopathic Sclerosing Orbital Inflammation.
    Ophthalmic plastic and reconstructive surgery, 2020
    Co-Authors: Bulent Yazici, Sukru Cekic, Ulviye Yalcinkaya, S Sebnem Kilic
    Abstract:

    Three patients (3 female patients; aged 7, 35, and 61 years) who had recalcitrant idiopathic sclerosing Orbital Inflammation were treated with rituximab. The disease was bilateral in 1 patient (4 orbits in total): diffuse in 2 and localized in 2 orbits. It caused optic neuropathy in 1 orbit of each patient. Conventional immunotherapy and tumor debulking surgery were unsuccessful in controlling the disease. After rituximab infusions (375 mg/m/week for 4 weeks), all patients improved symptomatically. Radiologically, the local lesions resolved completely and diffuse lesions partially. Two patients with recurrent Inflammation during follow up (78, 58, and 51 months) responded well to immediate, short-term steroid treatments. Short-term rituximab therapy can induce effective remissions in patients with refractory idiopathic sclerosing Orbital Inflammation. Early and local lesions may respond better to treatment than diffuse lesions. Nevertheless, inflammatory exacerbations can occur during late follow up.

  • AB1016 Rituximab Treatment in Patients with Idiopathic Sclerosing Orbital Inflammation
    Annals of the Rheumatic Diseases, 2015
    Co-Authors: S Sebnem Kilic, Bulent Yazici
    Abstract:

    Background Idiopathic sclerosing Orbital Inflammation (ISOI) is a rare disease characterized by fibrous pseudotumor formation, inflammatory changes and poor response to corticosteroids, immunosupressive therapy or radiotherapy. Objectives In this report, 2 patients with ISOI who were refractory to conventional immunosupressive therapy and were treated with rituximab are presented. Methods Case 1: Ten-year-old girl presented with proptosis, ocular motility restriction, lagophthalmus and optic neuropathy in the right eye. Orbita MRI showed a diffuse, infiltrative mass in the superior and retrobulbar orbit. After tumor debulking and histological examination, the diagnosis of ISOI was made. The treatments with prednisolone, cyclophosphamide, methotrexate and/or azothiopurin failed in achieving a complete remission. After the treatment with rituximab (375 mg/m 2 x4), all inflammatory signs disaapeared dramatically and did not reccur during follow up. Case 2: A 36-year-old female presented with painful proptosis, restrictive myopathy and blepharoptosis in her right eye. Orbita MRI showed an infiltrative mass in the upper orbit, encasing the superior rectus muscle and lacrimal gland. Biopsy showed dense fibrous tissue with a mixed cellular infiltrate consistent with ISOI. Systemic evaluation did not find any accompanying abnormality. The disease initially responded well to the oral prednisolon andmethotrexate treatment, but then recurred with a mass regrowth associated with inflammatory symptoms and optic neuropathy. After a tumor debulking surgery, the patient received rituximab (375 mg/m 2 x4) was commenced. Clinical symptoms completely regressed after this treatment and did not recur during follow up. Results In both patients, following the treatment with rituximab (375 mg/m 2 , 4 cure) the lesions have regressed apparently. Recurrence has not been observed for 6 months. Conclusions Idiopathic sclerosing Orbital Inflammation is characterized by aggressive, chronic, insidious sclerosis and poor response to corticosteroids and immunosuppressive drugs. The presented 2 cases support that rituximab alone may provide a complete clinical remission in patients refractory to the conventional immunosuppressive drugs. References Galvez-Ruiz A, Chaudhry I, Al Katan H. Idiopathic sclerosing Orbital Inflammation: presentation of an unusual case with isolated bilateral optic nerve involvement. J Neuroophthalmol. 2013;33:308-10. Li Y, Lip G, Chong V, Yuan J, Ding Z. Idiopathic Orbital Inflammation syndrome with retro-Orbital involvement: a retrospective study of eight patients. PLoS One. 2013;8:e57126. Pemberton JD, Fay A.Idiopathic sclerosing Orbital Inflammation: a review of demographics, clinical presentation, imaging, pathology, treatment, and outcome. Ophthal Plast Reconstr Surg. 2012;28:79-83. Disclosure of Interest None declared

Zhongxiang Ding - One of the best experts on this subject based on the ideXlab platform.

  • Idiopathic Orbital Inflammation syndrome with retro-Orbital involvement: a retrospective study of eight patients.
    PloS one, 2013
    Co-Authors: G. Lip, Vincent F. H. Chong, Jianhua Yuan, Zhongxiang Ding
    Abstract:

    Background The aim of this retrospective study was to document the clinical findings and radiological features of idiopathic Orbital Inflammation syndrome with retro-Orbital involvement. Methods We searched for ophthalmological patients who received Orbital imaging at Zhejiang Provincial People's Hospital between October 2003 and April 2010. Seventy-three patients were diagnosed with idiopathic Orbital Inflammation syndrome based on clinicoradiological features, with pathological confirmation of nonspecific inflammatory conditions in 47 patients. Eight patients (11%) had MRI or CT evidence of retro-Orbital involvement. All 8 patients were diagnosed with idiopathic Orbital Inflammation syndrome after biopsy of the Orbital lesion. MR images were obtained for all 8 patients; 3 patients also had a contrast-enhanced CT scan. Results Seven out of 8 patients with retro-Orbital involvement also had Orbital apex lesions. Of the 65 patients without retro-Orbital involvement, 19 had Orbital apex lesions. The difference in the number of patients with Orbital apex lesions between the two populations was significant (Fisher exact test P = .002). In all 8 patients with retro-Orbital involvement, the Inflammation spread through the superior Orbital fissure. The retro-Orbital lesions were isointense to grey matter on T1-weighted images, hypointense on T2-weighted images, and displayed uniform contrast enhancement; on contrast-enhanced CT scans, they were hyperdense relative to the contralateral mirror area and had radiological contours that were similar to those seen on MR images. The diffuse Inflammation with marked sclerosis and hyalinization that we observed in the patients with retro-Orbital involvement is consistent with the diagnosis of the sclerosing subtype of idiopathic Orbital Inflammation syndrome. All 8 patients also complained of mild to moderate periOrbital pain (headache). Conclusions In patients with idiopathic Orbital Inflammation syndrome, it is important to perform MRI and CT scans to identify possible retro-Orbital involvement. Retro-Orbital involvement is more frequent when the lesion is present in the Orbital apex.

James A. Garrity - One of the best experts on this subject based on the ideXlab platform.

  • Not a Tumor-Nonspecific Orbital Inflammation
    Journal of neurological surgery. Part B Skull base, 2021
    Co-Authors: James A. Garrity
    Abstract:

    Objective This study was aimed to illustrate the features and complexities of nonspecific Orbital Inflammation via discussion of two representative cases. Design Present study is a retrospective case review. Setting The study was conducted at a tertiary care medical center. Participants Two patients with nonspecific Orbital Inflammation were participants of this retrospective study. Main Outcome Measures Outcome of the study was disease-free patients and off all medications. Results At follow-up, both patients are disease free and off all medications. Conclusion Surgery plays a diagnostic and therapeutic role. While the clinical subtype is important for differential diagnosis and symptomatic treatment, the histologic subtype is similarly important. For inflammatory dacryoadenitis, surgery can be therapeutic. For extensive granulomatosis with polyangiitis, debulking surgery may allow better penetration of medications, especially rituximab.

  • Orbital Inflammation: biopsy first
    Survey of ophthalmology, 2016
    Co-Authors: Ilse Mombaerts, Geoffrey E Rose, James A. Garrity
    Abstract:

    Orbital Inflammation is a response of the immune system and not a diagnosis in itself. Exposing the underlying disease introduces a labyrynthine challenge owing to the broad array of possible causes ranging from infectious, structural, autoimmune, idiopathic to neoplastic origin. In this regard, and despite its unkown etiology, idiopathic Orbital Inflammation (IOI) intrinsically is a genuine diagnostic entity. Where clinical and radiological findings of an Orbital inflammatory mass are inconclusive, pathological examination of the tissue biopsy-obtained by minimally invasive approach and local anesthesia-is advocated to work towards a diagnosis in a most timely and effective manner. A corticosteroid response can be observed in most Orbital disorders with lymphocytic components, including IOI, and, accordingly, constitutes a paradoxical and weak tool to identify the diagnosis in Orbital Inflammation.

  • Treatment of recalcitrant idiopathic Orbital Inflammation (chronic Orbital myositis) with infliximab.
    American journal of ophthalmology, 2004
    Co-Authors: James A. Garrity, Austin W. Coleman, Eric L. Matteson, Eric Eggenberger, David M. Waitzman
    Abstract:

    Purpose To report results of treatment with a monoclonal antibody (infliximab) directed against tumor necrosis factor α in seven patients with chronic and difficult-to-control idiopathic Orbital Inflammation (Orbital myositis). Design Observational case series. Methods Retrospective data were collected from seven patients who had idiopathic Orbital Inflammation and who were evaluated at three medical centers. All patients were treated with infliximab after the failure of traditional therapy, which included corticosteroids, radiotherapy, or anti-inflammatory chemotherapeutic agents. Results All seven patients had a favorable response to treatment with infliximab. One patient with Behcet disease required supplemental oral corticosteroids. Pain, swelling, and need for concomitant corticosteroids were the primary measures of treatment success. Symptoms of comorbid disease in four patients also improved (Crohn disease in two, Behcet disease in one, and psoriasis in one). There were no untoward effects of treatment after a mean follow-up of 15.7 months (range, 4 to 31 months). Conclusions Treatment with infliximab appears to offer another therapeutic option in cases of recalcitrant or recurrent idiopathic Orbital Inflammation in which conventional treatment fails.