Purpose: To report an unusual and rare case of both eyes fixed in an extreme superomedial position. Case report: A case of 48-year-old woman presented with both eyes fixed in an extreme superomedial position; the microcorneas were covered almost completely by the upper eyelids even when she opened her eyes. A forced duction test was performed to confirm there were severe restrictions in all directions. She underwent disinsertion of the superior and medial rectus muscle, inferior oblique muscle belly transposition and sclera fixation. At the 1-year follow-up, there was improved ocular alignment. Conclusion: This case may be a special form of myopic strabismus fixus. The infratemporal fossa may be a bony landmark related to the etiology of global fixation.
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