Introduction: The macrostomia is a facial abnormality also called transverse facial cleft or Tessier cleft number 7. Macrostomia is a rare condition with an incidence from 1/80,000 to 1/300,000 live births. The left commisure is more commonly affected. Bilateral macrostomia is extremely rare, with only 10 to 20% of all cases of macrostomia, and 50% of bilateral cases are associated with different syndromes. To date just over 20 cases of isolated bilateral macrostomia have been described in the literature.

Clinical Observation: We report the case of a patient with isolated bilateral macrostomia solved by surgery and with good aesthetic and functional results.

Comments: The treatment of bilateral macrostomia is surgical and should be done early. Although there are many techniques, the key is to recreate a new and correct commisure through a good reorientation of the orbicularis oris.

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