Bisphosphonate-Induced Orbital Inflammation: A Case Series and Review.

INTRODUCTION: To present a series of patients with bisphosphonate induced orbital inflammation, and to review the clinical presentation, radiological features, treatment options and outcomes. METHODS: We present a multicentre, retrospective case series review of patients with a clinico-radiological diagnosis of bisphosphonate induced orbital inflammation and review all the reported cases of this complication in the literature. RESULTS: Four new patients with bisphosphonate induced orbital inflammation were added to the 25 cases in the literature. Intravenous zoledronate was the commonest precipitant (22/29, 75.9%) and inflammation occurred 1-28 (mean 3) days post-infusion. Orbital imaging identified orbital inflammation in 22/29 cases and extra-ocular muscle enlargement in 8/29. Five patients presented with reduced vision of which one - with anterior ischaemic optic neuropathy - did not resolve. The vision resolved in all except one patient, with most requiring steroid treatment. CONCLUSIONS: Bisphosphonates have a pro-inflammatory effect, which can precipitate orbital inflammation. This rare, but potentially serious complication of bisphosphonate treatment should be considered by clinicians using bisphosphonate treatment and by ophthalmologists seeing patients with orbital inflammatory disease.

Adalimumab as steroid-sparing treatment of inflammatory-stage thyroid eye disease.

PURPOSE: Steroids are often used as medical therapy for active thyroid eye disease (TED). While high-dose steroids have been shown to be effective in reducing the severity of TED symptoms, the side effects of steroids can be severe. As the pathogenesis of TED is thought to involve the upregulation of proinflammatory cytokines, including tumor necrosis factor-α (TNF-α), it has been postulated that anti-TNF agents may be used as steroid-sparing agents in the treatment of TED. This retrospective study was conducted to examine the efficacy of adalimumab, a subcutaneously administered TNF-α antagonist, in treating the inflammatory symptoms of active TED. METHODS: All patients in the inflammatory phase of TED who were treated with adalimumab at the Jules Stein Eye Institute over a 2-year period were reviewed. Data concerning visual acuity, optic nerve function, extraocular motility restriction, binocular visual fields, and proptosis were extracted from patient charts. Clinical photographs from baseline and 3-month follow-up visits were reviewed by masked orbital specialists. Each photograph was graded on the severity of conjunctival injection, chemosis, eyelid erythema, and eyelid edema on a scale from 1 to 4. An inflammatory score was calculated as the sum of these 4 elements. Groups were compared using paired t tests. RESULTS: Six of 10 patients showed a decrease in inflammatory score while on adalimumab, whereas 3 showed an increase and 1 stayed the same. One patient experienced a significant complication (hospital admission for sepsis). Eight patients received concomitant tapering steroids during the first 6 weeks of therapy as the adalimumab reached maximum efficacy. When data from all 10 subjects were analyzed together, there was no significant change in inflammatory index after 3 months of treatment with adalimumab. However, when the 5 patients with a high baseline inflammatory index (>4) were considered separately, there was a significant improvement (mean decrease of 5.2±2.7; p<0.01) after adalimumab treatment. Four of 5 patients also reported a subjective improvement in symptoms while on adalimumab. CONCLUSIONS: This study suggests that adalimumab may have a role in the treatment of active TED with prominent inflammatory symptoms. The use of adalimumab and other immunosuppressive agents in the treatment of TED may help to mitigate some of the metabolic and psychiatric side effects of pulsed steroid treatment. A future randomized controlled study will be necessary to determine the efficacy of adalimumab as a primary therapy for TED.

Miositis orbitaria como complicación tras una amigdalitis por «Streptococcus pyogenes» / Orbital myositis as a complication of Streptococcus pyogenes amigdalitis

La miositis orbitaria es un trastorno inflamatorio poco común en la infancia, clasificado dentro del llamado síndrome inflamatorio de la órbita, o seudotumor orbitario, en el que se ven afectados los músculos extraoculares. Presentamos el caso de un paciente de 12 años de edad, con dolor y edema palpebral de inicio brusco, junto con limitación de la movilidad ocular. Tras la realización de pruebas de imagen y estudios de laboratorio, se diagnostica de miositis orbitaria tras una infección estreptocócica. Aun siendo una asociación poco frecuente, debemos tener en cuenta esta etiología infecciosa ante un paciente con clínica de dolor ocular brusco y limitación de la movilidad, sobre todo en casos con antecedentes de infección de las vías respiratorias altas(AU)

Orbital myositis is an uncommon inflammatory disorder in childhood, classified into orbital inflammation syndrome, or orbital pseudotumor, where extraocular muscles are affected. We report a 12 years old patient with pain, eyeld oedema and ocular motility limitation. After the scan and laboratories exam he was diagnosed of orbital myositis after streptococcal infection. Even as a rare association we should take account of this infectious etiology in a patient with symptoms of sudden eye pain and limited mobility, especially in cases with a recent history of upper respiratory tract infection(AU)

Efeitos da injeção intraorbitária de carboximetilcelulose 6.0% em coelhos: análise histológica e da mecânica ocular / Effects of intraorbital injection of 6.0% carboxymethylcellulose in rabbits: histological and ocular mechanics evaluation

OBJETIVO: Avaliar se a carboximetilcelulose 6,0% é capaz de atuar como modificadora do sistema oculomotor de coelhos por meio de forças de ação viscoelástica, a durabilidade de seu eventual efeito e reações teciduais à aplicação. MÉTODOS: Foram utilizados 25 coelhos da raça Nova Zelândia, divididos em dois grupos experimentais: um tratado com injeção peribulbar de carboximetilcelulose (CMC) 6,0 por cento e um controle que foi submetido à injeção peribulbar de 3,0 cc de soro fisiológico. No grupo tratado com CMC, variou-se o volume total injetado, obtendo-se, assim, quatro subgrupos (1,0, 1,5, 2,0 e 3,0 cc). Foram realizadas medidas da força necessária para promover movimentos tangenciais de adução, avaliação clínica e medidas da pressão intraocular antes, imediatamente após a injeção da substância e no 7º, 30º e 60º dia pós-operatório. A eutanásia dos animais foi realizada no 60º dia pós-operatório para análise histológica dos tecidos perioculares. RESULTADOS: A força média encontrada, 60 dias após a injeção da CMC 6,0 por cento, foi menor no subgrupo tratado 1,0 e maior nos subgrupos tratados 1,5, 2,0 e 3,0 relativamente à força antes da injeção. Nos subgrupos em que houve aumento da força, a análise histológica revelou processo inflamatório do tipo histiocitário com formação de fibrose e a presença da CMC nos tecidos perioculares. CONCLUSÕES: A carboximetilcelulose 6,0 por cento atuou como modificadora do sistema oculomotor de coelhos, podendo facilitar ou dificultar movimentos. Não foi possível concluir se o aumento da força deveu-se apenas ao processo inflamatório ou à soma de inflamação com um possível atrito viscoso provocado pela CMC.

PURPOSE: To determine the effects of 6.0% carboxymethylcellulose (CMC) in modifying the oculomotor system of rabbits by means of viscoelastic action forces, the durability of this effect and possible inflamatory reaction. METHODS: Twenty-five New Zealand rabbits were divided into two experimental groups: one treated with a peribulbar injection of 6.0 percent CMC and a control group submitted to peribulbar injection of 3.0 cc of physiological saline. The group submitted to peribulbar CMC injection was divided into four subgroups respectively receiving the following volumes: 1.0, 1.5, 2.0 and 3.0 cc. The force needed to promote tangential adduction dislocations, external ophthalmologic signs and intraocular pressure were evaluated. The animals were sacrificed on the 60th day after the injection for histological analysis. RESULTS: The mean force detected 60 days after the injection of 6.0 percent CMC was lower in the 1.0 treated subgroup and higher in the 1.5, 2.0 and 3.0 treated subgroups relative to the force before injection. Histological analysis revealed histiocytic infiltration with fibrosis in the subgroups in which there was an increase in force and the presence of CMC in periocular tissues. CONCLUSIONS: 6.0 percent CMC acted as a modifier of the oculomotor system of rabbits, facilitating or impairing movements. It was not possible to conclude whether the increase in force occurred as a consequence of the inflammatory process alone or of the sum of inflammation and a possible viscous attrition provoked by CMC.

Topical cyclosporine A as a steroid-sparing agent in steroid-dependent idiopathic ocular myositis with scleritis: a case report and review of the literature.

OBJECTIVE: To report on a case of idiopathic orbital myositis with scleritis that was effectively controlled with topical 0.05% cyclosporine A and to provide a review of the literature on the treatment of ocular myositis with scleritis. METHODS: A case report. RESULTS: A 35-year-old woman presented with a longstanding history of intractable periorbital pain, redness on her left eye, and diplopia during ocular movement. Her medical history revealed that she had the same symptoms for 5 years and had used numerous prescribed medications for migraine and ocular myositis. During this period, her symptoms and signs had been lessened on systemic steroid treatment, which recurred or worsened after discontinuing or tapering the therapy. Magnetic resonance imaging scans demonstrated an isolated enlargement of the left medial rectus muscle. Laboratory examination results showed no evidence of dysthyroid ophthalmopathy or another systemic disease. Because of adverse affects of systemic corticosteroid and cyclosporine treatments, topical cyclosporine A (0.05%) and dexamethasone were administered four times daily. The patient continued to use topical 0.05% cyclosporine A for 6 months. Using only topical cyclosporine A, she currently has no recurrences of disease on the last examination after 6 months of treatment. Moreover, magnetic resonance imaging revealed a completely normal extraocular muscle configuration. CONCLUSIONS: Topical 0.05% cyclosporine A may be a safe and effective long-term treatment of ocular myositis and scleritis. It should be considered as a steroid-sparing agent, particularly in recurrent disease and in those patients who experience adverse effects of systemic medications.

[Effects of intraorbital injection of 6.0% carboxymethylcellulose in rabbits: histological and ocular mechanics evaluation]. / Efeitos da injeção intraorbitária de carboximetilcelulose 6,0% em coelhos: análise histológica e da mecânica ocular.

PURPOSE: To determine the effects of 6.0% carboxymethylcellulose (CMC) in modifying the oculomotor system of rabbits by means of viscoelastic action forces, the durability of this effect and possible inflammatory reaction. METHODS: Twenty-five New Zealand rabbits were divided into two experimental groups: one treated with a peribulbar injection of 6.0% CMC and a control group submitted to peribulbar injection of 3.0 cc of physiological saline. The group submitted to peribulbar CMC injection was divided into four subgroups respectively receiving the following volumes: 1.0, 1.5, 2.0 and 3.0 cc. The force needed to promote tangential adduction dislocations, external ophthalmologic signs and intraocular pressure were evaluated. The animals were sacrificed on the 60th day after the injection for histological analysis. RESULTS: The mean force detected 60 days after the injection of 6.0% CMC was lower in the 1.0 treated subgroup and higher in the 1.5, 2.0 and 3.0 treated subgroups relative to the force before injection. Histological analysis revealed histiocytic infiltration with fibrosis in the subgroups in which there was an increase in force and the presence of CMC in periocular tissues. CONCLUSIONS: 6.0% CMC acted as a modifier of the oculomotor system of rabbits, facilitating or impairing movements. It was not possible to conclude whether the increase in force occurred as a consequence of the inflammatory process alone or of the sum of inflammation and a possible viscous attrition provoked by CMC.