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Tongue reduction operation improves mandibular prognathism in Beckwith-Wiedemann syndrome without compromising tongue function such as articulation and taste perception.

Objectives
This study evaluated the surgical outcomes of patients with Beckwith-Wiedemann syndrome who underwent tongue-reduction surgery, and aimed to analyze whether malocclusion and mandibular prognathism caused by macroglossia could be improved.


Methods
A retrospective medical record review was performed for 11 patients with Beckwith-Wiedemann syndrome whose macroglossia was surgically treated. Demographic data, symptoms and signs, and intra- and postoperative surgical outcomes were evaluated. Surgery was performed by a single surgeon using a keyhole technique known as midline elliptical excision and anterior wedge resection. Preoperative and postoperative plain skull lateral X-rays were evaluated for prognathism improvement.


Results
Median age at the time of surgery was 35.1 months, and the ratio of males to females was 4:7. Median surgical time was 98 ± 31.45 minutes, and the median duration of postoperative ICU care was 3.81 ± 2.4 days. There were no airway complications. Two patients (18.2%) had postoperative wound dehiscence, however, there was no nerve damage, recurrence, or other complications. Among the five patients who underwent postoperative speech evaluation, all showed normal speech development, except one patient who had brain dysfunction and developmental delay. Measurement of A point - Nasion - B point angles and Sella - Nasion - B point angles (Point A is the most concave point of anterior maxilla; point B is the most concave point on mandibular symphysis) in plain X-rays showed a significant decrease in postoperative SNB angles (P < 0.001) and a significant increase in ANB angles (P < 0.011).


Conclusion
Tongue-reduction surgery is an effective and safe technique for severe forms of macroglossia associated with Beckwith-Wiedemann syndrome. In addition, it improves mandibular prognathism in young BWS patients with macroglossia.