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Bilateral macrostomia as an isolated deformity

MACROSTOMIA is a congenital defect resulting from persistent lateral facial clefts. These embryonic clefts persist if the maxillary and mandibular portions of the first branchial arch fail to unite normally. Macrostomia is a rare anomaly. Blackfield and Wilde (1950) reported five cases of macrostomia in a series of 500 patients with cleft lip. This probably represents an unusually high incidence in a serie? of referral patients. FoghAndersen's report (1965) based on all facial clefts treated in Denmark over a 30-year period probably provides a better index of the fiequency of macrostomia. The total, representing all types of facial clefts, was 3,988; included were thirteen patients with macrostomia, an incidence of 0-3 per cent of the total series. The frequency of cleft lip is reported to vary fiom 1 in 500 to 1 in 1,000 live births (Fryer and Brown, 1969). From these figures, an estimated incidence of macrostomia would be from 1 in 150,000 to 1 in 300,000 live births. Fogh-Andersen stated that almost all of his thirteen patients had other associated anomalies. This has been the experience also of others writing on the subject. May (1962) felt that macrostomia received little attention in the literature because it seldom constitutes an isolated deformity; and it may be overshadowed by associated defects. Lateral facial clefts also occur on one side more often than bilaterally. It is apparent that bilateral macrostomia occurring as an isolated defect is indeed rare. The purpose of this paper is to report an infant with such a condition.