Thyroid-associated orbitopathy (TAO), frequently termed Graves ophthalmopathy, is part of an autoimmune process that can affect the orbital and periorbital tissue, the thyroid gland, and, rarely, the pretibial skin or digits (thyroid acropachy), Although the use of the term thyroid ophthalmopathy is pervasive, the disease process is actually an orbitopathy in which the orbital and periocular soft tissues are primarily affected with secondary effects on the eye. Thyroid orbitopathy (TO) is most prevalent among females in the fourth and fifth decades and is usually associated with thyroid dysfunction. Thyroid-associated orbitopathy may precede, coincide, or follow the systemic complications of dysthyroidism. The ocular manifestations of thyroid-associated orbitopathy include eyelid retraction, proptosis, chemosis, periorbital edema, and altered ocular motility with significant functional, social, and cosmetic consequences. we describe a unique case of unilateral exophthalmos in a young male patient. REPORT OF A CASE: A 30 year old male patient presented with unilateral axial proptosis left eye for 6 months with eye signs of thyroid disease and history of diplopia on up gaze, increased appetite, weight loss, excessive sweating. He also gave complaints of red, gritty, photophobic and watery eyes. His visual acuity was in the right eye 20/40 improving to 20/20 and in the left eye it was 20/40 improving to 20/20. Color vision was normal. There was no RAPD present, Eye signs included Von Graffe's sign (immobility or lagging of the upper eyelid on downward rotation of the eye), Stellwag's sign (infrequent or incomplete blinking), Dalrymple sign (widened palpebral fissure), Gifford sign (Difficulty everting the upper eyelids), Pochin's sign (blinking rate is reduced), Rosenbach's sign (unusual tremor of the eyelids), Snellen's sign (Bruit heard on auscultation over the eye), Mean's sign (increased superior scleral show on upgaze), Kocher's sign (staring and frightened appearance), Mobius' sign (inability to maintain convergence of the eyes), Suker's sign (fixation becomes difficult in extreme lateral gaze). The intraocular pressure in the RE was 17 mm Hg and LE was 19 mm of Hg. There was restriction of movements in the upgaze in the left eye. There was no corneal involvement. The visual fields were normal. Serum T3, T4 were normal with elevated TSH levels, B-scan showed thickened muscle belly of inferior rectus muscle. On CT scan Proptosis of the left eye noted (the distance from the interzygomatic line to anterior aspect of globe is 28 mm), Increase in both intra and extraconal fat is noted in the left eye , Increase in the size and altered signal intensity is noted in the muscle belly of inferior rectus of left eye with sparing of the tendon. On histologic examination Fibrosis with degenerative changes in the eye muscles, Lymphocytic cell infiltration, Accumulation of mucopolysaccharides, interstitial edema with increased collagen production was seen. The patient was treated conservatively with maintenance of euthyroid