Case report: Orbital myositis triggering oxygen-responsive cluster headache.

BACKGROUND: Orbital myositis is an idiopathic, non-infectious condition, typically seen in young females and usually affecting one extraocular muscle. Orbital myositis mimicking cluster headache is a rare clinical entity, and this is the first description of a case of a secondary trigeminal autonomic cephalalgia from orbital myositis responsive to high-flow oxygen. CASE: A young woman presented with new-onset, oxygen-responsive headache, periorbital pain and autonomic features. She had associated vertical diplopia on downgaze and subtle ocular misalignment. An initial diagnosis of cluster headache was made. Initial brain MRI was unrevealing, but dedicated MRI of the orbits showed enhancement of orbital muscles. The diplopia and the imaging findings were consistent with orbital myositis. CONCLUSION: Orbital myositis mimicking cluster headache is rare, and not previously reported as an oxygen-responsive headache.

Successful "medical" orbital decompression with adjunctive rituximab for severe visual loss in IgG4-related orbital inflammatory disease with orbital myositis.

IgG4-related disease (IgG4-RD) is an inflammatory condition of unknown etiology that can cause tumefactive lesions in a number of tissues and organs, including the orbit and ocular adnexa. Diagnostic criteria for IgG4-RD, including pathology and clinical features and pathology, have been recently proposed. This study presents the first case of unilateral acute visual loss secondary to IgG4-related orbital inflammatory disease with orbital myositis that was complicated by severe compressive optic neuropathy. After initial treatment with pulsed intravenous methylprednisolone, followed by rituximab and radiotherapy, there was a marked improvement in orbital inflammation and clinical and radiological improvement in the compressive optic neuropathy. After 9 months of follow up, the orbital inflammatory disease remained in remission.

Orbital myositis in a patient with Crohn's disease: A case report of two episodes.

PURPOSE: To describe a case report of a 34 years-old patient with Crohn's Disease and two episodes of Ocular Myositis. METHODS: The research methodology employed in this study consisted of a retrospective review of the patient's complete medical history. RESULTS: Crohn's Disease is a chronic inflammatory bowel disorder known to be associated with a wide range of extraintestinal manifestations. Ocular abnormalities, such as episcleritis and uveitis, are commonly observed. However, orbital myositis is an extremely rare ocular extraintestinal manifestations characterized by acute ocular pain that worsens with eye movements and is often accompanied by diplopia. In this case report, we present the case of a 34-year-old woman with a confirmed diagnosis of Crohn's Disease, who experienced two episodes of acute orbital pain exacerbated by ocular movements and diplopia. The diagnosis was established through clinical evaluation and radiologic imaging, with confirmation after a good response to systemic corticosteroids. She responded favorably to systemic corticosteroid therapy on both episodes, and no additional treatment was required. As of now, she remains stable without any ocular sequelae. CONCLUSION: It is important to note that orbital myositis is an uncommon ocular manifestation associated with Crohn's Disease, and prompt recognition and management are crucial to achieve successful outcomes.

Adalimumab for orbital myositis in a patient with Crohn's disease who discontinued infliximab: a case report and review of the literature.

BACKGROUND: Orbital myositis is a rare extra-intestinal manifestation of inflammatory bowel disease. Seventeen cases of Crohn's disease associated orbital myositis and 3 cases of ulcerative colitis associated orbital myositis have been reported in the published literature since 1970. We report the use of adalimumab (Abbott, Canada, Inc.) for orbital myositis in a patient with Crohn's disease who discontinued infliximab (Janssen, Canada, Inc.) and review of the published literature. CASE PRESENTATION: A 35 year-old male with a 7-year history of Crohn's disease was treated with an ileocolonic resection and re-anastomosis followed by infliximab which maintained full endoscopic and clinical remission for four years. After stopping the infliximab for infusion-related reactions he presented with 3-day history of severe right eye pain, pain with ocular movement, proptosis, and conjunctival injection. He had no intestinal symptoms and endoscopic assessment revealed no active luminal disease. CT of the orbit revealed an enlarged right medial rectus muscle with tendonous involvement and a diagnosis of orbital myositis was made. Treatment with 80 mg per day prednisone with tapering dose and adalimumab, induction and maintenance, resulted in rapid resolution of the orbital myositis and ocular symptoms with no recurrences on follow-up at 10 months. CONCLUSIONS: The current case demonstrates a rare extraintestinal manifestation of Crohn's disease, orbital myositis, and its temporal relationship to the discontinuance of infliximab therapy and its successful treatment, without recurrence with tapering prednisone and adalimumab.

Atypical idiopathic orbital inflammation in a young girl.

A 7-year-old girl presented with chronic right periocular pain worst in abduction, edema, and proptosis with radiographic evidence of right medial rectus myositis. After a negative workup, she was diagnosed and treated for idiopathic orbital inflammation (IOI) with a brisk response to oral steroids. She returned 8 months later with left periocular pain worst in abduction, proptosis, and radiographic evidence of left lateral rectus myositis. She was treated for recurrent and alternating IOI with intravenous (IV) glucocorticoids with resolution. One year later, she recurred with similar left orbital signs and symptoms and was found to have a left inferior oblique myositis on MRI. She was treated with oral steroids and methotrexate with clinical resolution. Ten months later, she presented with left periocular pain worst on adduction and was found to have left medial rectus myositis on MRI. She was treated with IV steroids, IV immunoglobulin, and rituximab with resolution of all symptoms. She is currently maintained on methotrexate and oral glucocorticoids. The atypical pediatric case of IOI over a 4-year period is described in this study.

[Orbital myositis associated with ipilimumab]. / Myosite orbitaire associée à un traitement par ipilimumab.

BACKGROUND: Ipilimumab is a monoclonal antibody targeting cytotoxic T-lymphocyte antigen 4 (CTLA-4) that allows increased survival and, occasionally, complete remission, in the treatment of metastatic melanoma. The most frequent adverse effects are attributed to dysimmunity. We report the case of a female patient who developed orbital myositis during treatment with ipilimumab. PATIENTS AND METHODS: A woman on ipilimumab for a heel melanoma with mediastinal metastases was referred for evaluation of painful diplopia and proptosis that began three days after the fourth infusion of ipilimumab. The clinical examination disclosed a left abductiondeficit associated with conjunctival hyperaemia and palpebral oedema. Orbital MRI disclosed enlargement of the left lateral rectus, enhancing after contrast. An extensive work-up did not find any evidence for thyroid-related eye disease, as well as other orbital inflammatory processes, orbital cellulitis or orbital metastases. Treatment with high-dose oral steroids resulted in complete clinical recovery within a few days. DISCUSSION: To our knowledge, this is the first clinical report of orbital myositis as an adverse event related to anti-CTLA-4 antibody treatment. Both timing and usual profile of adverse events support the hypothesis that orbital myositis has to be attributed there to ipilimumab. Several dysimmune toxicities were observed with ipilimumab. Ophtalmic toxicity has unusually been described. Most cases were uveitis. Whether immune-related adverse events correlate with clinical response to ipilimumab treatment remains to be determined.

Idiopathic inflammation of the orbit and contiguous structures.

Idiopathic orbital inflammation is a common cause of acute orbital signs and symptoms. It is typically confined to the bony orbit; however, it can rarely involve contiguous structures with or without lytic change raising clinical suspicion for malignancy. Three cases of idiopathic inflammation of the orbit that affected adjacent structures are reported here; 2 cases had maxillary sinus involvement, while a third had extension in the temporal fossa.

Recurrent bilateral orbital myositis: case report and review of the literature.

Orbital myositis in children is uncommon. Recurrence is a major challenge in management. An 11-year-old Malay girl who presented with bilateral orbital myositis with recurrent attacks of diplopia is reported.

A case of eosinophilic orbital myositis associated with CSS.

We report a novel case of eosinophilic orbital myositis associated with Churg-Strauss syndrome. A 56-year-old man with a 20-year history of chronic sinusitis and seasonal allergic rhinitis was admitted because of fever, swelling of cheeks and extremities, diplopia, and eosinophilia. With findings from gadolinium-enhanced FST1WI of the orbits and a muscle biopsy of the skeletal muscle, the diagnosis was made. He was treated with oral corticosteroid, and his symptoms rapidly improved.

Case Report: Orbital Myositis and Myasthenia Gravis as Symptoms of Immune Reconstitution Inflammatory Syndrome in a Patient With Human Immunodeficiency Virus Infection.

We present a case of a 37-year-old man with HIV infection who had been on antiretroviral therapy for one year. He was admitted to our hospital with red and swollen eyes, acute onset progressive exophthalmos, and intermittent diplopia endured for 7 days. His symptoms, exam, and imaging led to a diagnosis of immune reconstitution inflammatory syndrome associated orbital myositis. His symptoms improved considerably after glucocorticoid therapy. Following a reduction in the oral prednisone dose, he re-presented with left ptosis, which rapidly progressed to bilateral ptosis. Diagnostic testing led to the diagnosis of immune mediated myasthenia gravis. Treatment with pyridostigmine bromide, prednisone, and tacrolimus was initiated. One month later, the patient's symptoms improved significantly. There was a probable association between his symptoms and autoimmune immune reconstitution inflammatory syndrome. This report highlights the importance of recognizing autoimmune disorders in human immunodeficiency virus-infected patients undergoing antiretroviral therapy. Orbital myositis and myasthenia gravis in human immunodeficiency virus-infected patients correlate closely with immunity status following a marked increase in CD4+ T cell counts.

Extraocular Muscle Palsy in Patients with Chronic Orbital Myositis.

BACKGROUND: Orbital myositis (OM) is a subgroup of idiopathic orbital inflammation. It can involve single or multiple extraocular muscles and result in restriction or paresis of extraocular muscles. METHOD: We reported two unusual cases of extraocular muscle paresis in the fibrotic stage of chronic OM and reviewed the literature related to this finding. RESULTS: The first case was of a 38-year-old woman with chronic OM with large-angle right eye exotropia and right medial rectus paresis. She received a botulinum A toxin injection into the right lateral rectus muscle as a first treatment, but it was unsuccessful in correcting her deviation. Subsequently, she underwent two strabismus surgeries, which successfully resolved her diplopia in primary gaze. The second case was of a 35-year-old woman with chronic OM and left lateral rectus palsy, which was managed with a botulinum A toxin injection. CONCLUSION: OM can cause extraocular muscle palsy in the chronic fibrotic stage of the disease.

Pediatric giant cell myocarditis and orbital myositis.

Giant cell myocarditis is a rare but often fatal form of myocarditis that often requires cardiac transplantation and has been associated with autoimmune diseases. We describe a 14-year-old female who developed painful proptosis and was diagnosed clinically and histologically with orbital myositis that improved with corticosteroid therapy. Approximately 2 months later, she developed abdominal pain, vomiting, weight gain, and fatigue. She was diagnosed with congestive heart failure and cardiomyopathy, and endomyocardial biopsy revealed giant cell myocarditis. She was treated with immunosuppressive agents and has responded well, without the need for cardiac transplantation. Three previous case reports have described an association between giant cell myocarditis and orbital myositis, but we present the first pediatric case report. We conclude that if orbital myositis is diagnosed in a patient, regardless of age, cardiac function should be closely monitored to detect myocarditis, which may affect the overall outcome.

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.