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Purpose: Terrien's marginal degeneration is a rare corneal disease usually characterized by an «insidious thinning of the cornea near the limbus» (Pouliquen Y et al. 1989). Its diagnosis remains essentially clinical. Aim: To present the help of innovating imaging in an atypical cystic presentation of Terrien's marginal degeneration. Methods: An 87‐year‐old caucasian patient presented since several month a voluminous cystic lesion of the limbus, bulky, neo‐vascularized, in the inferior part of the eye, that compromised the palpebral closure. No traumatic or surgical history was found. Slit‐lamp examination of the contralateral eye also revealed a degenerative area with corneal thinning. In order to determine the etiology of this lesion, 70 Mhz ultrasound (VevoMD high‐frequency ultrasound system, Fujifilm, Toronto, Canada), anterior segment optical coherence tomography (OCT, Casia 1, Tokyo, Japan) and in vivo confocal microscopy (IVMC, Vivascope 3000, LUCID, NY, USA) were performed. Surgical removal of the lesion consisted of en bloc removal of the cystic lesion followed by limbal conjunctiva and amniotic membrane graft for analgesic purposes and to accelerate wound healing. Histology and direct immunofluorescence were performed. Results: 70 Mhz ultrasonography and OCT revealed a single non‐segmented cyst, without scleral fistula or corneal thinning. IVMC showed a single well delineated cyst with clearly visible distinct partitions with no image in favour of malignancy. No signs of malignancy or ocular pemphigoid were founded by histology. We, therefore, concluded to an atypical presentation of Terrien's marginal degeneration. Conclusions: The combination of sophisticated imaging like ultra‐high frequency 70 Mhz ultrasound, AS‐OCT and IVCM are necessary for diagnostic and therapeutic management of atypical cornea lesions. [ABSTRACT FROM AUTHOR]