Literature DB >> 27561730

Initial Experience With Propranolol Treatment of Lymphatic Anomalies: A Case Series.

June K Wu1, Ellen D Hooper2, Sherelle L Laifer-Narin3, Lynn L Simpson4, Jessica Kandel5, Carrie J Shawber6.   

Abstract

Lymphatic malformations (LMs) are congenital lymphatic lesions that impose significant and costly morbidities on affected patients. Treatment options are limited due to incomplete understanding of LM pathobiology. Expression of an activated β2-adrenergic receptor has been described in LM tissue, suggesting that this pathway may contribute to the clinical manifestations of LM. We hypothesized that propranolol, a β-adrenergic receptor antagonist, might improve symptoms of patients with LMs and lymphatic anomalies. A retrospective chart review of patients treated with propranolol as an adjunct therapy was conducted; analyses included demographic characteristics, clinical features, and response to propranolol. Three patients with cystic and noncystic LMs displayed clinical improvement at a minimum dose of 0.7 mg/kg/d, whereas symptomatic relapses were observed when propranolol doses dropped below this threshold. Two patients with Klippel-Trenaunay syndrome demonstrated partial clinical responses with reduced edema. The fetus of a mother treated with propranolol from a gestational age of 35 weeks through delivery displayed arrested growth of a cervicofacial LM. Our retrospective review suggests that propranolol improved symptoms in a subset of patients with lymphatic anomalies. Propranolol treatment may also limit the growth of congenital LMs in utero.
Copyright © 2016 by the American Academy of Pediatrics.

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Year:  2016        PMID: 27561730      PMCID: PMC5005016          DOI: 10.1542/peds.2015-4545

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  26 in total

1.  Propranolol therapy for hemorrhagic lymphangioma of the tongue.

Authors:  Nicolas Leboulanger; Catherine Garel; Isabelle Tillous Borde; Eréa-Noël Garabedian; Françoise Denoyelle
Journal:  Arch Otolaryngol Head Neck Surg       Date:  2011-05-16

2.  Ex utero intrapartum treatment (EXIT) in the management of cervical lymphatic malformation.

Authors:  Pablo Laje; William H Peranteau; Holly L Hedrick; Alan W Flake; Mark P Johnson; Julie S Moldenhauer; N Scott Adzick
Journal:  J Pediatr Surg       Date:  2014-11-10       Impact factor: 2.545

3.  Sirolimus for the treatment of complicated vascular anomalies in children.

Authors:  Adrienne M Hammill; MarySue Wentzel; Anita Gupta; Stephen Nelson; Anne Lucky; Ravi Elluru; Roshni Dasgupta; Richard G Azizkhan; Denise M Adams
Journal:  Pediatr Blood Cancer       Date:  2011-03-28       Impact factor: 3.167

4.  Propranolol as a treatment option in Gorham-Stout syndrome: a case report.

Authors:  Vered Nir; Ludmila Guralnik; Galit Livnat; Ronen Bar-Yoseph; Fahed Hakim; Anat Ilivitzki; Lea Bentur
Journal:  Pediatr Pulmonol       Date:  2013-09-02

5.  Efficacy and Safety of Sirolimus in the Treatment of Complicated Vascular Anomalies.

Authors:  Denise M Adams; Cameron C Trenor; Adrienne M Hammill; Alexander A Vinks; Manish N Patel; Gulraiz Chaudry; Mary Sue Wentzel; Paula S Mobberley-Schuman; Lisa M Campbell; Christine Brookbank; Anita Gupta; Carol Chute; Jennifer Eile; Jesse McKenna; Arnold C Merrow; Lin Fei; Lindsey Hornung; Michael Seid; A Roshni Dasgupta; Belinda H Dickie; Ravindhra G Elluru; Anne W Lucky; Brian Weiss; Richard G Azizkhan
Journal:  Pediatrics       Date:  2016-01-18       Impact factor: 7.124

6.  Prognostic factors in the treatment of lymphatic malformations.

Authors:  E Raveh; A L de Jong; G P Taylor; V Forte
Journal:  Arch Otolaryngol Head Neck Surg       Date:  1997-10

7.  Long-term propranolol therapy in pregnancy: maternal and fetal outcome.

Authors:  S C Pruyn; J P Phelan; G C Buchanan
Journal:  Am J Obstet Gynecol       Date:  1979-10-15       Impact factor: 8.661

8.  An open-label study to evaluate sildenafil for the treatment of lymphatic malformations.

Authors:  Christina Danial; Andrea L Tichy; Umar Tariq; Glenda L Swetman; Phuong Khuu; Thomas H Leung; Latanya Benjamin; Joyce Teng; Shreyas S Vasanawala; Alfred T Lane
Journal:  J Am Acad Dermatol       Date:  2014-03-20       Impact factor: 11.527

9.  Obstetric hemorrhage during an EXIT procedure for severe fetal airway obstruction.

Authors:  Alexander Butwick; Pedram Aleshi; Imad Yamout
Journal:  Can J Anaesth       Date:  2009-04-25       Impact factor: 5.063

10.  Treatment of a tongue lymphangioma with sirolimus after failure of surgical resection and propranolol.

Authors:  Canan Akyüz; Erman Ataş; Ali Varan
Journal:  Pediatr Blood Cancer       Date:  2013-11-22       Impact factor: 3.167

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  3 in total

Review 1.  Recent Progress in Lymphangioma.

Authors:  Xiaowei Liu; Cheng Cheng; Kai Chen; Yeming Wu; Zhixiang Wu
Journal:  Front Pediatr       Date:  2021-12-15       Impact factor: 3.418

2.  Clinical and genetic analyses of patients with lateralized overgrowth.

Authors:  Yoon-Myung Kim; Yena Lee; Yunha Choi; In Hee Choi; Sun Hee Heo; Jung Min Choi; Hyo-Sang Do; Ja-Hyun Jang; Mi-Sun Yum; Han-Wook Yoo; Beom Hee Lee
Journal:  BMC Med Genomics       Date:  2022-09-30       Impact factor: 3.622

3.  Successful management of extremely high-output refractory congenital chylothorax with chemical pleurodesis using 4% povidone-iodine and propranolol: a case report.

Authors:  Kathleen Borcyk; Ammar Kamil; Kristine Hagerty; Melissa Deer; Paul Tomich; Ann L Anderson Berry
Journal:  Clin Case Rep       Date:  2018-02-27
  3 in total

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