Sirolimus in blue rubber bleb naevus syndrome: A systematic review

被引:31
|
作者
Wong, Xin Lin [1 ]
Phan, Kevin [1 ,2 ]
Rodriguez Bandera, Ana I. [3 ]
Sebaratnam, Deshan F. [1 ,2 ,4 ]
机构
[1] Univ New South Wales, Sydney, NSW, Australia
[2] Sydney Childrens Hosp Network, Liverpool Hosp, Dept Dermatol, Sydney, NSW, Australia
[3] Sydney Childrens Hosp Network, Dept Dermatol, Sydney, NSW, Australia
[4] Hosp Univ La Paz, Dept Dermatol, Madrid, Spain
关键词
blue rubber bleb naevus syndrome; mTOR; sirolimus; vascular malformation; venous malformation; RECURRENT IRON-DEFICIENCY; PATIENT; MALFORMATION; RAPAMYCIN; CHILD;
D O I
10.1111/jpc.14345
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
The aim of this study is to review sirolimus as a treatment for blue rubber bleb naevus syndrome (BRBNS). A literature search of Medline, Embase, CINAHL, SCOPUS and Google Scholar was conducted for publications reporting treatment of patients with BRBNS with sirolimus. Of 46 articles identified, 17 studies reporting 23 patients met inclusion criteria. Sirolimus was well tolerated in all but one patient who required treatment cessation; 17/18 patients noted an improvement in gastrointestinal disease where this was reported; 21/22 patients noted an improvement in cutaneous disease where this was reported. Based on these results, sirolimus may be considered a first-line treatment of BRBNS depending on patient morbidity.
引用
收藏
页码:152 / 155
页数:4
相关论文
共 50 条
  • [1] Blue rubber bleb naevus syndrome: a systematic review
    Wong, X. L.
    Phan, K.
    Liu, R.
    Rodriguez Bandera, A. I.
    Sebaratnam, D. F.
    BRITISH JOURNAL OF DERMATOLOGY, 2019, 181 : 45 - 46
  • [2] Blue rubber bleb naevus syndrome - A systematic review
    Wong, X.
    Sebaratnam, D.
    AUSTRALASIAN JOURNAL OF DERMATOLOGY, 2019, 60 : 101 - 101
  • [3] Blue rubber bleb naevus syndrome
    Pei, HP
    Liao, GQ
    Yan, ZS
    EUROPEAN JOURNAL OF SURGERY, 2001, 167 (08) : 628 - 630
  • [4] Pregnancy and blue rubber bleb naevus syndrome
    Adeniji, AA
    Fairlie, FM
    Jones, TH
    Wilkinson, P
    BRITISH JOURNAL OF OBSTETRICS AND GYNAECOLOGY, 1999, 106 (12): : 1316 - 1318
  • [5] Gastrointestinal: Blue rubber bleb naevus syndrome
    Hammond, P
    Lewindon, P
    JOURNAL OF GASTROENTEROLOGY AND HEPATOLOGY, 1999, 14 (03) : 295 - 295
  • [6] Efficacy and safety of sirolimus in the treatment of blue rubber bleb naevus syndrome in paediatric patients
    Zhang, B.
    Li, L.
    Zhang, N.
    Zhao, M.
    Liu, Y.
    Wei, L.
    Ma, L.
    Xu, Z.
    CLINICAL AND EXPERIMENTAL DERMATOLOGY, 2020, 45 (01) : 79 - 85
  • [7] BLUE RUBBER BLEB NAEVUS
    WALSHE, MM
    EVANS, CD
    WARIN, RP
    BRITISH MEDICAL JOURNAL, 1966, 2 (5519): : 931 - &
  • [8] EDITORIAL COMMENT: BLUE RUBBER BLEB NAEVUS SYNDROME
    Gupta, Nitin
    Day, Andrew S.
    JOURNAL OF PAEDIATRICS AND CHILD HEALTH, 2010, 46 (1-2) : 67 - 68
  • [9] Sirolimus is a Successful Treatment for Recurrent Iron Deficiency Anaemia in Blue Rubber Bleb Naevus Syndrome
    Wang, Huaijie
    Gao, Ya
    JOURNAL OF PEDIATRIC GASTROENTEROLOGY AND NUTRITION, 2015, 61 (05): : E24 - E24
  • [10] Blue rubber bleb naevus syndrome involving the pleura
    Aussedat, P. H.
    Laroumagne, S.
    Dutau, H.
    Astoul, P.
    REVUE DES MALADIES RESPIRATOIRES, 2018, 35 (03) : 333 - 337