PURPOSE:: To describe a patient with Tessier cleft number 5 and 9 and review the literature on the ocular impairment and management of this extremely rare anomaly. METHODS:: Interventional case report and literature review. RESULTS:: The literature review showed that the present patient is the second case with clefts 5/9. The ophthalmic consequences of this rare association are virtually unreported. Our case demonstrates that the presence of cleft number 9 adds a cicatricial component on the upper eyelid that severely impairs the dynamics of this lid. The corneal status of the patient was successfully managed with simultaneous upper eyelid lengthening and facial reconstruction. CONCLUSION:: In order to avoid corneal perforation, simultaneous upper and lower eyelid reconstruction is mandatory in cases of cleft 5/9. The affected patients should be continuously followed in order to prevent amblyopia.
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