Long-term Results of Unilateral Cleft Lip Repair With Multiple Infantile Hemangiomas Including the Cleft Side of the Upper Lip

Abstract

PURPOSE: Cleft lip and infantile hemangioma are relatively common congenital diseases. However, infantile hemangiomas on the cleft side, in the operative field of cleft lip, are extremely rare, and no clear guidelines have been established for their treatment. We experienced a case in which a patient with a cleft lip had infantile hemangioma on the cleft side. We performed cleft lip repair on a patient with infantile hemangioma on the cleft side of the cleft lip at 3 months after birth and have been following-up on the patient for the past 18 years. We report the results of 18-year follow-up after the surgery of this rare patient. METHODS: The male patient was diagnosed with a left unilateral complete cleft lip with alveolar cleft and a submucous cleft palate. There were multiple infantile hemangiomas throughout his body, with no notable abnormalities in the chromosomal analysis. In particular, the patient had infantile hemangiomas on the upper lip and lower lip, right ear helix, back, and left shoulder. The hemangioma on the upper lip was on the lateral segment of the cleft side. As per the general treatment guidelines, cleft repair was performed at 3 months after birth. Millard’s rotation advancement technique was used for the repair, which uses the lower small triangular flap. The patient was followed up for 18 years after surgery. RESULTS: There was no excessive bleeding during the surgery, and blood transfusion was not needed. The excised tissue was confirmed to be infantile hemangioma in the histopathologic examination. The patient did not show any abnormalities during recovery, and there was some residue of infantile hemangioma in the repair site in the upper lip vermilion. The residual infantile hemangioma in the repair site of the upper lip vermilion was involuted by 5 years old, and there was only a normal degree of scarring after cleft lip repair of the upper lip vermilion. At the same time, the infantile hemangiomas in the right ear helix, back, and left shoulder were completely involuted without any scarring. The patient has been followed up until now, and at 18 years, he has no particular problems other than the red scar on the lower lip. CONCLUSION: Infantile hemangiomas requiring lip repair for unilateral complete cleft lip are extremely rare, and there are no established surgical guidelines for this condition. In such cases, delaying lip repair until the infantile hemangioma is involuted may not be desirable for obtaining the best esthetic outcome, and it is also not desirable because it may induce psychosocial impairment in patients and caregivers. Therefore, we believe that general cleft lip repair produces good outcomes even in cases involving hemangiomas on the cleft side.