Bisphosphonate-induced orbital inflammation in a patient on chronic immunosuppressive therapy.

BACKGROUND: To report a case of orbital inflammation after bisphosphonate infusion in a patient who was already receiving immunosuppressive therapy. CASE PRESENTATION: A 56-year-old woman presented to the ophthalmology clinic with acute onset of right eye pain 24 h after receiving her first Zolendronic acid infusion. She has a past medical history of chronic inflammatory demyelinating polyneuropathy, Sjogren's syndrome, and systemic lupus erythematosus that have been controlled with immunosuppressive therapy for three years. Clinical ophthalmic exam and MRI studies were significant for right orbital inflammation. The patient was started on oral prednisone with rapid resolution of symptoms. CONCLUSIONS: This is the first case report of a patient receiving chronic immunosuppressive therapy to develop orbital inflammation after Zoledronic acid infusion. In addition, it demonstrates that corticosteroids can be an effective first line therapy in treating orbital inflammation in similar patients. Physicians should be aware of this rare but serious potential side effect of bisphosphonates, and have bisphosphonate-related orbital inflammation on their differential for proper initiation of treatment.

A case report of orbital inflammatory syndrome secondary to ipilimumab.

Ipilimumab is a monoclonal antibody to cytotoxic T-lymphocyte antigen-4, a negative regulator of T-cell-mediated immune response. Ipilimumab is approved by the US Food and Drug Administration for the treatment of advanced melanoma. However, its use frequently has been associated with immune-related side effects, which can be explained by its mechanism of action. More common adverse effects include dermatitis, colitis, hepatitis, and endocrinopathies, but many less common immune-related adverse effects that involve various tissues and organ systems have been reported with more widespread use of ipilimumab since its approval in 2011. A case of bilateral orbital inflammatory syndrome secondary to ipilimumab, in a patient undergoing adjuvant treatment for metastatic melanoma, is reported.

Nitrogen bisphosphonate-induced orbital inflammatory disease: gamma delta T cells--a report and review of 2 cases.

With the increased use of nitrogen bisphosphonate therapy for osteoporosis, multiple myeloma, metastatic prostate carcinoma, and Paget disease, it is now recognized that orbital inflammation is a potential sequelae of treatment. To date, 15 isolated cases of orbital inflammation exist in the literature with additional 2 cases reported herein. While the precise triggering mechanism for orbital inflammation is not definitively understood, it appears that a regulatory response from gamma delta T cells is the most likely causative factor. Nitrogen bisphosphonates are a causative agent of orbital inflammatory disease. These case reports adhered to the principles of Helsinki and the Health Insurance Portability and Accountability Act.

Orbital MRI appearance after remote retrobulbar alcohol injection.

An 88-year-old woman with a blind painful OS underwent a retrobulbar alcohol injection. Nine months following the procedure, she was evaluated in the clinic for discomfort surrounding her left brow and forehead and was found to have dysesthesia in the left V1 nerve distribution. There was no evidence of orbital inflammation on history or ocular examination. An MRI of the orbits showed intraconal abnormal signal with enhancement and enophthalmos. The chronic effect of retrobulbar alcohol injection on the orbital contents may mimic orbital inflammatory disease on the MRI, but the absence of clinical correlates should allow for appropriate diagnosis.

Bisphosphonate-associated orbital inflammation--a case report and review.

Bisphosphonates are becoming more widespread as indications for them broaden. Several ocular side effects due to bisphosphonates have been described, among the most potentially serious of which is orbital inflammation. Thirteen case reports of this side effect exist in the literature, with an additional case reported here. The most common presenting signs are lid edema, conjunctival hyperemia, and chemosis, while common symptomatology includes pain, diplopia, and blurry vision. A concomitant anterior uveitis is present in 30% of cases, and some degree of bilaterality is also seen in 30% of cases. There are two reported instances of profound visual loss, highlighting the importance of prompt recognition and treatment of this entity. Despite a paucity of rechallenge data, available reports suggest that development of orbital inflammation does not necessarily exclude patients from future bisphosphonate use.

Chronic orbital inflammation and fibrosis after retrobulbar alcohol and chlorpromazine injections in a patient with choroidal melanoma.

A 59-year-old woman developed a choroidal melanoma in her right eye and was successfully treated with plaque radiotherapy in 1995. She subsequently developed a blind, painful eye and was treated with retrobulbar alcohol and chlorpromazine injections. A transient acute severe inflammatory reaction to chlorpromazine was noted, and chronic pain persisted. MRI performed 5 months later revealed diffuse orbital enhancement suggestive of either fibrosis or tumor recurrence. Following enucleation, diffuse orbital fibrosis was confirmed. The unusual fibrosis could have been related to the orbital injections or remotely related to long-term effects of radiotherapy.

A Case Series and Review of Bisphosphonate-associated Orbital Inflammation.

PURPOSE: To report the largest series of new cases to date of bisphosphate-associated orbital inflammation. METHODS: A retrospective case review of patients with orbital inflammation following treatment with systemic bisphosphonate. RESULTS: Six patients over an 18-month period (2 males, 4 females) with an average age of 62.2 years had onset of orbital inflammatory symptoms 1-11 days after intravenous bisphosphonate infusion or, in 1 case, 4 weeks after initiation of oral bisphosphonate therapy. Imaging revealed diffuse orbital involvement in 3 cases, isolated lateral rectus muscle involvement in 2 cases, and superior rectus-levator involvement in 1 case. Two patients' symptoms resolved spontaneously within 2 weeks, and 3 responded rapidly and completely to corticosteroid therapy. The 1 patient on oral bisphosphonate had a slower but complete response to corticosteroid treatment. CONCLUSION: Clinicians should be aware of the association between acute orbital inflammation and recent treatment with systemic bisphosphonate medication.

Orbital inflammation after sub-Tenon's anesthesia.

Two cases of orbital swelling after sub-Tenon's anesthesia are reported. The first patient presented 3 days postoperatively with proptosis and conjunctival chemosis. Computed tomography showed nonspecific inflammation of the orbital soft tissue. Signs and symptoms resolved after systemic steroids. The second patient presented with similar signs and mild pain on the fourth postoperative day. Computed tomography showed a similar diffuse orbital inflammation. The patient was treated with oral steroids and antibiotics, and all inflammation subsided within 4 weeks. Both patients had uneventful cataract surgery, were apyrexial, and were generally well. A few possible mechanisms for these episodes are infection, reaction to povidone-iodine or sub-Tenon's anesthetic, or trauma due to the sub-Tenon's cannula.

Technical modification in the intracarotid chemotherapy and osmotic blood-brain barrier disruption procedure to prevent the relapse of carboplatin-induced orbital pseudotumor.

The blood-brain barrier disruption (BBBD) procedure is an established strategy to enhance drug delivery to brain tumors. Complication rates associated with this procedure are usually low, but when complications do occur, they usually mandate discontinuation of treatment. Orbital pseudotumor is an inflammatory condition of one or more extraocular muscles that produces limitation of ocular motility. Patients usually experience sudden diplopia associated with orbital pain, conjunctival chemosis and injection, and proptosis. Imaging of the orbit shows diffuse enlargement of the extraocular muscles, exophthalmia, and, rarely, sinusal or intracranial infiltration. On pathologic examinations, the soft tissues of the orbit are infiltrated with a mixture of eosinophils, lymphocytes, and plasma cells. Many etiologies can induce this syndrome, including the intracarotid infusion of platinum molecules. As part of a phase II study, a total of 110 patients were treated for malignant brain tumors with intra-arterial carboplatin, enhanced by the BBBD procedure, at the Sherbrooke University Hospital. Here we report on three patients who developed orbital pseudotumor ipsilateral to the carotid infused a few hours to days after the procedure. After the occurrence of this syndrome in the first patient, we developed a technical modification to the procedure that enabled uninterrupted treatment in the other two patients. This modification was as follows: after the mannitol infusion, and before carboplatin, the catheter was changed for a 3.5 tracker and was repositioned just above the emergence of the ophthalmic artery.

Hyaluronidase induced orbital pseudotumor as complication of retrobulbar anesthesia.

The authors describe 5 patients who developed an orbital pseudotumor as a complication of retrobulbar anesthesia. Allergy tests revealed a hypersensitivity to hyaluronidase, suggesting an immunoallergic etiology of the pseudotumor, which needed to be differentiated from other causes of proptosis after retrobulbar injection.

Sarcoid-like granulomatous orbital inflammation induced by interferon-alpha treatment.

A 56-year-old black woman presented with bilateral orbital swelling in the lacrimal gland region 4 months after initiation of interferon-alpha and ribavirin treatment for occupationally-acquired hepatitis C infection. Histopathologic evaluation of bilateral lacrimal gland biopsy specimens revealed granulomatous inflammation. All other tests were negative for sarcoidosis including angiotensin converting enzyme level and chest x-ray. The use of interferon-alpha as an antiviral and/or chemotherapeutic agent is increasing. Therefore recognition of its possible orbital side effects is important for the oculoplastic surgeon. To the authors' knowledge, this is the first case of orbital granulomatous involvement associated with interferon-alpha therapy.

Polyarthropathy, orbital myositis and posterior scleritis: an unusual adverse reaction to influenza vaccine.

We report a patient who developed an acute symmetrical polyarthropathy, orbital myositis and posterior scleritis following inoculation with the 1993 Fluvirin (inactivated influenza vaccine: surface antigen) vaccine. We believe that this represents a hypersensitivity reaction to the vaccine as extensive investigations revealed no underlying infective, inflammatory or systemic cause. We review the current indications for vaccination against the influenza virus and the known adverse reactions to the vaccine.