TY - JOUR
T1 - Propranolol for the treatment of lymphatic malformations in a neonate – a case report and review of literature
AU - Liviskie, Caren J.
AU - Brennan, Colleen C.
AU - McPherson, Christopher C.
AU - Vesoulis, Zachary A.
N1 - Publisher Copyright:
© Pediatric Pharmacy Association. All rights reserved. For permissions, [email protected].
PY - 2020
Y1 - 2020
N2 - Lymphatic malformations in neonates often manifest as a chylothorax, and although rare, morbidity and mortality can be significant. First-line treatment with medium-chain triglyceride–enriched formulas, or enteric rest with total parenteral nutrition, are not always successful. We describe the case of a premature neonate with trisomy 21 who presented with bilateral pleural effusions and a pericardial effusion that worsened with the initiation of enteral nutrition. Clinical improvement was not seen until the initiation of treatment with oral propranolol at a maximum dosage of 0.5 mg/kg/day divided every 8 hours with extubation 8 days after propranolol initiation. Two case reports have described the use of propranolol in similar patients receiving 2 mg/kg/day; however, our experience is the first to report treatment success at a much lower dose. A review of the literature for alternative medication treatments uncovered numerous case reports and series documenting variable results with incongruent definitions of treatment success in a diverse patient population. The rarity of this disease state makes accrual of patients difficult and more robust treatment data unlikely. Therefore, selection of the optimal adjunctive treatment must be based on individual patient and disease state characteristics as well as safety and efficacy profile of the medication.
AB - Lymphatic malformations in neonates often manifest as a chylothorax, and although rare, morbidity and mortality can be significant. First-line treatment with medium-chain triglyceride–enriched formulas, or enteric rest with total parenteral nutrition, are not always successful. We describe the case of a premature neonate with trisomy 21 who presented with bilateral pleural effusions and a pericardial effusion that worsened with the initiation of enteral nutrition. Clinical improvement was not seen until the initiation of treatment with oral propranolol at a maximum dosage of 0.5 mg/kg/day divided every 8 hours with extubation 8 days after propranolol initiation. Two case reports have described the use of propranolol in similar patients receiving 2 mg/kg/day; however, our experience is the first to report treatment success at a much lower dose. A review of the literature for alternative medication treatments uncovered numerous case reports and series documenting variable results with incongruent definitions of treatment success in a diverse patient population. The rarity of this disease state makes accrual of patients difficult and more robust treatment data unlikely. Therefore, selection of the optimal adjunctive treatment must be based on individual patient and disease state characteristics as well as safety and efficacy profile of the medication.
KW - Chylothorax
KW - Chylous effusion
KW - Lymphangiogenesis
KW - Newborn
KW - Propranolol
UR - http://www.scopus.com/inward/record.url?scp=85079875904&partnerID=8YFLogxK
U2 - 10.5863/1551-6776-25.2.155
DO - 10.5863/1551-6776-25.2.155
M3 - Article
C2 - 32071591
AN - SCOPUS:85079875904
SN - 1551-6776
VL - 25
SP - 155
EP - 162
JO - Journal of Pediatric Pharmacology and Therapeutics
JF - Journal of Pediatric Pharmacology and Therapeutics
IS - 2
ER -