An Unusual Case of Orbital Inflammation Preceding Herpes Zoster Ophthalmicus

PURPOSE: To present a case of orbital inflammation and optic perineuritis preceding vesicular eruption in herpes zoster ophthalmicus(HZO). CASE SUMMARY: An 84-year-old woman with a history of gall bladder cancer and hypertension complained of left periorbital erythematous edema and discomfort. On examination, visual acuity was 20/25 bilaterally; no tenderness, proptosis or ophthalmoplegia was observed. Pupils were equal, round, and reactive to light without relative afferent pupillary defects. Slit-lamp examination revealed severe conjunctival injection and chemosis without keratitis or uveitis. The remainder of the ocular examination was unremarkable. Magnetic resonance imaging confirmed left-sided preseptal swelling with an enlarged left lacrimal gland, high signal intensity of the retrobulbar fat and optic nerve sheath. Systemic antibiotic therapy with steroids was started under a presumed diagnosis of idiopathic orbital inflammatory disease, but the clinical presentation was unresolved. After 2 days, vesicular lesions confined to the first division of the trigeminal nerve and pseudodendritic keratitis developed on the left side leading to a diagnosis of HZO. Treatment with acyclovir immediately resolved anterior segment inflammation and periorbital edema. While on therapy, visual acuity deteriorated to 20/125 and the pupil became dilated and unresponsive to light over a few days. All signs and symptoms of acute orbitopathy and postherpetic neuralgia had resolved 3 months later with the exception of pupil abnormality and visual acuity. CONCLUSIONS: HZO may present with symptoms and signs of orbital inflammation and optic perineuritis even in the absence of a vesicular rash. Thus, HZO should be considered in the differential diagnosis of unexplained acute orbital syndromes.

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 Bilateral Orbital Tuberculosis

PURPOSE: Periocular tuberculosis is common in the form of conjunctival tuberculosis or uveitis; however, orbital tuberculosis is very rare. We report a case of tuberculosis diagnosed based on bilateral orbital masses. CASE SUMMARY: A 64-year-old male with a history of diabetes, hypertension, and chronic obstructive pulmonary disease presented with diplopia. His best-corrected visual acuity was 1.0, and intraocular pressure was 16 mmHg in both eyes. Bilateral proptosis was noted, and inferior orbital masses were palpated in both eyes. On orbital computed tomography, irregular masses were surrounding both globes in the extraconal space. Incisional biopsy of the bilateral orbital masses was performed under local anesthesia. Pathologic examination revealed chronic granulomatous inflammation accompanied by caseous necrosis. The patient was diagnosed with bilateral orbital tuberculosis and underwent triple therapy with isoniazid, rifampin, and ethambutol for 12 months. The orbital masses completely resolved, and there was no recurrence or complication through 12 months after treatment. CONCLUSIONS: To our knowledge, bilateral orbital tuberculosis has not been reported yet. Herein, we report a case of orbital tuberculosis presenting as bilateral orbital masses.

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.

Clinical Manifestations and Treatment of Idiopathic Optic Perineuritis

PURPOSE: To investigate the clinical and radiologic manifestations of idiopathic optic perineuritis (OPN), and to evaluate the outcomes of steroid treatment for OPN. METHODS: We reviewed the medical records and radiologic findings of 10 patients (13 eyes) who were diagnosed with OPN and treated with steroid. RESULTS: The mean age was 56.5 +/- 9.3 years (range, 35-77 years) and the sex ratio was equal. The main complaint was decrease in visual acuity combined with ocular pain during extraocular eye movement in 9 patients. The median visual acuity at the first visit was 0.2 (HM-0.8) and the relative afferent papillary defect was observed in 12 eyes. Additionally, combined orbital diseases included posterior scleritis in 1 eye and myositis in 1 eye. Orbit magnetic resonance imaging (MRI) scans demonstrated intraorbital optic nerve sheath enhancement in all patients, occasionally with orbital fat involvement. All patients demonstrated improved visual acuity after high-dose oral steroid therapy (6 patients) or intravenous (IV) pulse steroid therapy (4 patients). Relapse occurred in 4 patients during steroid tapering. CONCLUSIONS: The population in this study was composed predominantly of patients with OPN in their 50's. The primary symptom of OPN was visual acuity decrease combined with ocular pain during extraocular eye movement. Radiologically, orbit MRI scans demonstrated intraorbital optic nerve sheath enhancement. The patients in this study demonstrated good responses to steroid treatment, but clinicians must be aware of the high recurrence rate during steroid tapering in this condition. A combination of clinical and radiologic findings was helpful to diagnose OPN.

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.

Diagnostic Significance and Usefulness in Digital Infrared Thermal Imaging (DITI) of Patients with Nonspecific Orbital Inflammation

PURPOSE: The present study assessed the diagnostic significance of digital infrared thermal imaging (DITI) in determining the inflammatory state of patients with nonspecific orbital inflammation. METHODS: The present study included 11 patients diagnosed with nonspecific orbital inflammation between December 2009 and March 2011 and who were followed-up for more than 6 months. All patients were all diagnosed based on computed tomography (CT), magnetic resonance image (MRI) and tissue biopsy. The grade of eyelid swelling was classified as 5 and severe compared with the temperature of location. The temperature of the upper eyelid, caruncle, medial conjunctiva, lateral conjunctiva, lower eyelid, cornea and lateral orbit were measured with DITI. RESULTS: When comparing the normal eye with the diseased eye in patients with nonspecific orbital inflammation, the temperature of the upper eyelid and cornea were statistically significant (p = 0.003, p = 0.038, respectively, Mann-Whitney test). The correlation between the grade of eyelid swelling and the temperature of location was most highly related in the temperature of the upper eyelid (Spearman's correlation coefficient, r = 0.55, p = 0.008) and cornea (Spearman's correlation coefficient, r = 0.45, p = 0.037). CONCLUSIONS: DITI may aid in evaluating the inflammatory state of nonspecific orbital inflammation. In particular, the temperature of the upper eyelid and cornea can be very useful indicator. Future studies, including larger study population are necessary in order to confirm DITI as a diagnostic tool which can assess the results of medical treatment by comparing temperature before and after treatment.

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.

Tratamiento quirúrgico del estrabismo en la enfermedad de graves / Surgical treatment of strabismus in graves disease

El estrabismo en la Oftalmopatía (Orbitopatía) de Graves se origina en la fibrosis post inflamatoria de uno o varios músculos, en uno o ambos ojos. Esto provoca un desalineamiento de los ejes visuales con diplopia que puede llegar a ser invalidante. Los músculos más frecuentemente comprometidos son los Rectos Inferiores y Medios. Algunos pacientes compensan su diplopia con posiciones anómalas de la cabeza. La cirugía se plantea en la etapa inactiva de la inflamación orbitaria, con ángulos estables por al menos seis meses; con los objetivos de eliminar la diplopia al menos en mirada al frente y en posición de lectura, eliminar las posiciones viciosas y mejorar la estética. La técnica quirúrgica, en la mayoría de los casos, consiste en el retroceso de todos los músculos responsables del estrabismo; idealmente con técnica ajustable, la que permite dosificar mejor la cirugía. La cirugía del estrabismo debe hacerse después de la descompresión orbitaria (si corresponde) y antes de la eventual cirugía de párpados. Los resultados en general son satisfactorios, pero con limitaciones

Strabismus in Graves Ophthalmopathy is caused by postinflammatory retraction in one or more muscles of one or both eyes. The most frequently involved muscles are inferior and medial rectus. This event provokes the loss of visual axes alignment, and, eventually, invalidant diplopia. Some patients compensate their diplopia with anomalous head positions. Surgery is indicated in the inactive phase of the orbital inflammatory process, with the goals of eliminate diplopia and abnormal head positions and to restore aesthetics. Surgical technique, in the majority of cases, consists in recessing all muscles involved in the strabismus; ideally with an adjustable technique that grants a better dosing of surgery. Strabismus surgery must be done after orbital decompression (if indicated) and before the eventual lid surgery. Results in general are satisfactory, but with some limitations in most cases

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.