Clinical Characteristics of Idiopathic Orbital Inflammation Accompanied with Paranasal Sinusitis

PURPOSE: To investigate the clinical characteristics of idiopathic orbital inflammatory disease (IOI) with paranasal sinusitis. METHODS: This study is a retrospective, comparative case series of patients who were diagnosed with IOI between January 2009 and December 2016. This study included patients with available medical and radiologic data at diagnosis and who participated in follow-up for more than 12 months after treatment. The patients were divided into two groups according to accompaniment of paranasal sinusitis and were compared. RESULTS: Among 101 patients with IOI, 13 (12.9%) were identified to have paranasal sinusitis. The incidence of pain was higher in patients with sinusitis (69.2%) than in patients without sinusitis (25.0%, p = 0.003). More patients with paranasal sinusitis experienced recurrence after systemic steroid therapy (69.2%) than in the other group (34.1%, p = 0.033). Additional immunosuppressants and/or radiation therapy were needed only in 9.1% patients without sinusitis but in 38.5% patients with sinusitis (p = 0.039). CONCLUSIONS: IOI patients with sinusitis showed a significantly higher recurrence rate. More careful follow-up of patients during steroid tapering and treatment of sinusitis might be helpful to prevent recurrence of IOI.

A Case of Surgical Diagnosis and Treatment of Idiopathic Orbital Myositis with Sudden Vision Loss

PURPOSE: We report a case of idiopathic orbital myositis with sudden onset of unilateral visual loss and hypertrophy of the lateral rectus muscle at the apex of the orbit that was diagnosed using orbital biopsy and treated with intravenous corticosteroids. CASE SUMMARY: We examined a 20-year-old woman who complained of decreased visual acuity and binocular diplopia for 2 weeks. She showed 40-prism diopter left esotropia at the primary position. Her visual acuity with eyeglasses was 0.7 and 0.2 in the right and left eyes, respectively. Brain magnetic resonance imaging (MRI) revealed a round mass on the left lateral rectus muscle at the apex of the orbit with optic nerve compression. We planned orbital biopsy to exclude orbital lymphoma and other biopsy-requiring diseases. After 3 weeks, the visual acuity of her left eye decreased to 0.02. Repeated MRI revealed enlargement of all extraocular muscles on the left orbit. With biopsy showing several lymphocytes infiltrating muscle fibers, we made a diagnosis of idiopathic orbital myositis. Intravenous injection of corticosteroids was administered during hospitalization. After 2 weeks of corticosteroid therapy, the visual acuity of her left eye was improved to 0.63, the esotropia disappeared, and the enlargement of the left lateral rectus muscle was improved on orbital MRI. CONCLUSIONS: For non-specific idiopathic orbital myositis located at the apex of the orbit and enlargement of the extraocular muscle in a short period with decreased visual acuity, administration of intravenous injection of corticosteroids before orbital biopsy could help restore vision.

A Case of Idiopathic Orbital Inflammation Presenting with Isolated Myositis of the Superior Oblique Muscle

PURPOSE: To report a case of idiopathic orbital inflammation presenting with isolated myositis of the superior oblique muscle. CASE SUMMARY: A 57-year-old male presented with pain in his left eye and diplopia for three months. His past history was unremarkable except surgery for a thyroid nodule six months prior. At initial presentation, his corrected vision and intraocular pressure were normal. Biomicroscopy and fundus examination were unremarkable. Nasal side swelling of the left upper eyelid was observed, and 1.5-mm proptosis of the left eye was revealed on exophthalmometry. Two and four prism diopter right hypertropia were noted at up and right-up gaze, respectively. Axial and coronal orbital computed tomography (CT) showed enhancement of isolated superior oblique muscle hypertrophy in the left eye. Systemic work-up was negative for any inflammatory disease. A presumptive diagnosis of isolated myositis of superior oblique muscle was made, and the patient was started on 30 mg of oral prednisolone daily. Pain and diplopia disappeared and proptosis of the left eye decreased to 0.5 mm after one week of treatment. Orbital CT images obtained one month after treatment showed a significant decrease in size of the left superior oblique muscle. The steroid was tapered for two months, and the patient has shown no signs of recurrence for four months after cessation of treatment. CONCLUSIONS: Idiopathic orbital inflammation presenting with isolated myositis of the superior oblique muscle is very rare and has not been previously reported in Korea. A patient presenting with pain and diplopia was diagnosed as having isolated myositis of the superior oblique muscle based on CT scan, and good results were achieved with oral steroid therapy.

Idiopathic Orbital Inflammation Presenting as Unilateral Dacryoadenitis in a Child

PURPOSE: To report a case of idiopathic orbital inflammation presenting as unilateral acute dacryoadenitis in a child. CASE SUMMARY: A nine-year-old boy presented with painful swelling and redness in the right upper eyelid and temporal conjunctiva without systemic symptoms for five days. Acute orbital cellulitis was suspected but did not respond to systemic antibiotics. An orbital computed tomogram and magnetic resonance imaging revealed a diffusely enlarged and inflamed right lacrimal gland. The patient showed dramatic response to systemic corticosteroids, and acute dacryoadenitis due to idiopathic orbital inflammation was diagnosed. Treatment with oral corticosteroids for two months resolved the inflammation. No relapse was observed during four months of follow-up. CONCLUSIONS: Although rare, acute dacryoadenitis as a localized nonspecific orbital inflammation should be considered in the differential diagnosis of acute orbital cellulitis in pediatric patients.

Effect of Intravenous Methylprednisolone on Idiopathic Orbital Inflammation

PURPOSE: To evaluate the efficacy of intravenous methylprednisolone pulse therapy in patients with idiopathic orbital inflammation. METHODS: Fifty-two patients who received three-day pulse methylprednisolone for acute severe swelling, loss of visual acuity, limitation of eye movement, diplopia, or pain were included in the present study. The lesions were divided into five subtypes using computed tomography or magnetic resonance imaging; anterior, lacrimal, myositic, diffuse and apical. A case diagnosed as sclerotic type was excluded. The response rate, recurrence rate and cure rate were assessed. RESULTS: The total response rate to intravenous methylprednisolone from all groups was 84.6%, with 100% in the anterior subtype and 71.4% in the diffuse subtype. During the mean follow-up period of 10.5 months, 15.9% of patients experienced a recurrence after a mean of 5.7 months. Overall, the cure rate was 71.2%. Recurrence was more common in the younger patients, and three of the seven patients with recurrence had myositic inflammation. CONCLUSIONS: Intravenous methylprednisolone was effective in patients with severe idiopathic orbital inflammation. However, combining other treatment modalities in patients with diffuse or myositic subtypes and in younger patients who show relatively low response or high recurrence is recommended.