Background: Thyroid associated orbitopathy (TAO) is defined as an immune mediated inflammatory process\naffecting the extraocular muscles, connective and adipose tissue of uncertain etiopathogenesis. TAO are classically\ndescribed in Graveâ??s disease (GD) however it may occur in euthyroid and hypothyroid patients. Those patients\nusually test positive for Thyroid Stimulating Hormone receptor antibodies (TRAb). For instance, only few cases of\nsevere Hashimotoâ??s thyroiditis (HT) associated orbitopathy with negative TRAb are reported to date.\nCase presentation: Herewith we report a rare case of a middle-aged female who presented with bilateral\nprogressive upper and lower palpebral edema and a unilateral marked proptosis associated with asthenia,\nheadache and decrease in visual acuity. Biological investigation was notable for high levels of anti-thyroid\nperoxidase antibodies (Anti-TPO) in an otherwise euthyroid patient with negative TRAb. Orbital Magnetic resonance\nimaging revealed edema of the extraocular muscles and inflammation of periorbital soft tissue. The patient received\na treatment with intravenous methylprednisolone followed by oral treatment with prednisone. This regimen was\nboth effective and safe with minimal metabolic side effects in our patient.\nConclusion: Minor ocular manifestations of HT are common; however, severe sight threatening ophtalmopathy in\nthe absence of TRAb is rare. Multiple differential diagnosis should be considered and investigated before\ndiagnosing this rare entity. Management of similar cases is currently based on reports and no clear guidelines have\nbeen elaborated, corticosteroids is the mainstream of treatment with a potential benefit of selenium\nsupplementation in mild to moderate cases.
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