1. Ex Utero Intrapartum Treatment for Prenatally Diagnosed Cervicofacial Lymphatic Malformations.
- Author
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Gillipelli SR, Peiffer SE, Larabee SM, Ketwaroo P, Rialon KL, Bedwell J, Mehta D, Lee TC, Keswani SG, and King A
- Subjects
- Humans, Female, Pregnancy, Retrospective Studies, Infant, Newborn, Adult, Magnetic Resonance Imaging, Gestational Age, Sclerotherapy methods, Neck diagnostic imaging, Neck abnormalities, Male, Treatment Outcome, Sirolimus administration & dosage, Lymphatic Abnormalities surgery, Lymphatic Abnormalities therapy, Lymphatic Abnormalities diagnostic imaging, Lymphatic Abnormalities diagnosis, Ultrasonography, Prenatal
- Abstract
Introduction: Cervicofacial lymphatic malformations (cf-LM) may be identified on prenatal ultrasound, prompting consideration of ex utero intrapartum treatment (EXIT) to secure the fetal airway. Furthermore, the recent shift in postnatal management of cf-LM from resection alone toward a multimodal approach including sirolimus and sclerotherapy may impact the neonatal outcomes of cf-LM. This study aims to characterize the neonatal outcomes of patients with prenatally diagnosed cf-LM who underwent EXIT-to-airway., Methods: Retrospective, single-center review of all patients who underwent EXIT-to-airway for cf-LM (2011-2020) was performed. Demographics, prenatal imaging, intraoperative details, and outcomes were analyzed using descriptive statistics (median [interquartile range])., Results: Six patients with prenatally diagnosed cf-LM underwent EXIT-to-airway at a median gestational age of 36 (33.8-36.9) wk. The median volume on fetal magnetic resonance imaging was 187.5 mL (142.3-237.8) and median tracheoesophageal displacement index was 11 mL (9.25-15). All were successfully intubated on placental support with a median duration of 25 (15.25-91) d. There was one fatality at day of life 10 due to necrotizing enterocolitis totalis. Among survivors, 2 of 5 underwent tracheostomy placement, 4 of 5 underwent gastrostomy tubes placement, and all 5 received sirolimus at day of life of 9 [8-10] d. Four patients underwent debulking or excision of their cf-LM during the initial hospitalization. Patients had a median length of stay of 68 (45-129) d. One patient experi enced a pneumothorax with evidence of barotrauma following EXIT-to-airway requiring chest tube placement (duration 8 d)., Conclusions: EXIT-to-airway procedure remains a feasible strategy for mitigating neonatal hypoxia in cases of prenatally diagnosed cervicofacial lymphatic malformations. However, postnatal outcomes are variable with potential long-term aerodigestive sequelae., (Copyright © 2024 Elsevier Inc. All rights reserved.)
- Published
- 2024
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