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A Comparison of Surgical Techniques for Macroglossia in Beckwith-Wiedemann Syndrome
Dominic J Romeo1, Theodor Lenz1, Andrew George2
1From the Divisions of Plastic, Reconstructive, and Oral Surgery.
Background:
Tongue reduction surgery can improve symptomatic macroglossia in Beckwith-Wiedemann syndrome (BWS), but there is no consensus on the optimal tongue reduction surgical technique. This study assessed trends and outcomes of several tongue reduction techniques for treating macroglossia in BWS. Using perioperative and polysomnographic data, the authors present their institutions' rationale for increasingly adopting the peripheral resection technique.
Methods:
Medical records of patients with BWS seen between December of 2004 and June of 2024 were reviewed for molecular diagnosis, surgical history, polysomnographic findings, and perioperative complications.
Results:
A total of 103 patients underwent 114 tongue reductions at a median age of 1.3 years (interquartile range [IQR], 0.4 to 2.8 years). Use of the peripheral resection technique increased from 10% of all tongue reductions before 2019 to 76% after 2019 ( P < 0.05). Compared with the W-plasty with keyhole approach, the peripheral resection had shorter operative times (29.0 [IQR, 24.0 to 34.0 minutes] versus 35.0 minutes [IQR, 28.0 to 39.0 minutes]; P < 0.05), a higher incidence of immediate extubation (51.7% versus 25.0%), shorter intubation periods (0 [IQR, 0 to 2.02 days] versus 5.0 days [IQR, 0 to 6.0 days]; P < 0.01), shorter pediatric intensive care unit admissions (3.0 [IQR, 1.0 to 10.0 days] versus 7.0 days [IQR, 3.0 to 20.0 days]; P = 0.042), and shorter hospital stays (4.0 [IQR, 2.0 to 10.0 days] versus 11.0 days [IQR, 5.0 to 19.0 days]; P = 0.005). Improvements in polysomnography and volume of resected tissue were similar between the two approaches ( P > 0.05). The anterior wedge (22.2%) and W-plasty (15.6%) techniques had more complications compared with peripheral resection (6.6%; P = 0.351).
Conclusions:
Peripheral resection appears noninferior to the W-plasty and anterior wedge approaches in treating symptomatic macroglossia in BWS. The authors document their institutional transition toward increasingly adopting this technique.
