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Safety and Efficacy of Bleomycin Sclerotherapy for Microcystic Lymphatic Malformation

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Abstract

Purpose

Sclerotherapy is the mainstay of treatment of macrocystic lymphatic malformation (LM), but the response using traditional sclerosants is much less beneficial in microcystic lesions. Intralesional bleomycin has been reported to be effective in microcystic LM; however, its use is limited by concerns about pulmonary fibrosis. The purpose of this study was to evaluate the safety and efficacy of bleomycin sclerotherapy in microcystic LM.

Methods

The medical records and imaging studies of all patients with microcystic or combined LM who underwent percutaneous image-guided sclerotherapy using bleomycin were retrospectively reviewed. Only patients with pre- and postprocedure imaging were included. Thirty-one patients with a mean age of 13.4 years (range 3 months–31 years) were treated. Response was graded as complete (>90 % size reduction), partial (25–90 %), or minimal/no response (<25 %). Pulmonary function tests (PFT) and chest X-rays were performed before the procedure. PFT were repeated at 6 months and 1 year postprocedure. Annual postprocedure chest X-rays were also performed.

Results

The malformations were located in the head and neck (n = 27) and trunk (n = 4). The number of procedures ranged from 1 to 4 (mean 1.7). Up to 1 U/kg of bleomycin was injected per session, with a maximum of 15 U. The mean follow-up period was 3.2 years (range 1.5–5 years). There was complete response in 38 % (n = 12), partial response in 58 % (n = 18), and no response in 3 % (n = 1). No complications were identified.

Conclusions

Preliminary indicate that sclerotherapy of microcystic LMs using bleomycin is effective and safe.

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References

  1. Burrows PE, Mitri RK, Alomari A et al (2008) Percutaneous sclerotherapy of lymphatic malformations with doxycycline. Lymphat Res Biol 6:209–216

    Article  CAS  PubMed  Google Scholar 

  2. Chaudry G, Burrows PE, Padua HM, Dillon BJ, Fishman SJ, Alomari AI (2011) Sclerotherapy of abdominal lymphatic malformations with doxycycline. J Vasc Interv Radiol 22:1431–1435

    Article  PubMed  Google Scholar 

  3. Alomari AI, Karian VE, Lord DJ, Padua HM, Burrows PE (2006) Percutaneous sclerotherapy for lymphatic malformations: a retrospective analysis of patient-evaluated improvement. J Vasc Interv Radiol 17:1639–1648

    Article  PubMed  Google Scholar 

  4. Sainsbury DC, Kessell G, Fall AJ, Hampton FJ, Guhan A, Muir T (2011) Intralesional bleomycin injection treatment for vascular birthmarks: a 5-year experience at a single United Kingdom unit. Plast Reconstr Surg 127:2031–2044

    Article  CAS  PubMed  Google Scholar 

  5. Fishman SJ (2013) Slow-flow vascular malformations. In: Mulliken JB, Burrows PE, Fishman SJ (eds) Mulliken & Young’s vascular anomalies: hemangiomas and malformations, vol 2. Oxford University Press, Oxford

    Google Scholar 

  6. Burrows PE (2013) Percutaneous treatment of slow-flow vascular malformations. In: Mulliken JB, Burrows PE, Fishman SJ (eds) Mulliken & Young’s vascular anomalies: hemangiomas and malformations, 2nd edn. Oxford University Press, New York, pp 661–710

    Google Scholar 

  7. Yamamoto T, Katayama I (2011) Vascular changes in bleomycin-induced scleroderma. Int J Rheumatol 2011:270938

    Article  PubMed Central  PubMed  Google Scholar 

  8. Ohta H, Chiba S, Ebina M, Furuse M, Nukiwa T (2012) Altered expression of tight junction molecules in alveolar septa in lung injury and fibrosis. Am J Physiol Lung Cell Mol Physiol 302:L193–L205

    Article  CAS  PubMed  Google Scholar 

  9. Hashimoto N, Phan SH, Imaizumi K et al (2010) Endothelial–mesenchymal transition in bleomycin-induced pulmonary fibrosis. Am J Respir Cell Mol Biol 43:161–172

    Article  CAS  PubMed Central  PubMed  Google Scholar 

  10. Zhang W, Chen G, Ren JG, Zhao YF (2013) Bleomycin induces endothelial mesenchymal transition through activation of mTOR pathway: a possible mechanism contributing to the sclerotherapy of venous malformations. Br J Pharmacol 170(6):1210–1220

    Article  CAS  PubMed Central  PubMed  Google Scholar 

  11. Yura J, Hashimoto T, Tsuruga N, Shibata K (1977) Bleomycin treatment for cystic hygroma in children. Nihon Geka Hokan 46:607–614

    CAS  PubMed  Google Scholar 

  12. Yang Y, Sun M, Ma Q et al (2011) Bleomycin A5 sclerotherapy for cervicofacial lymphatic malformations. J Vasc Surg 53:150–155

    Article  PubMed  Google Scholar 

  13. Perkins JA, Manning SC, Tempero RM et al (2010) Lymphatic malformations: review of current treatment. Otolaryngol Head Neck Surg 142:795–803, 803.e1

  14. O’Sullivan JM, Huddart RA, Norman AR, Nicholls J, Dearnaley DP, Horwich A (2003) Predicting the risk of bleomycin lung toxicity in patients with germ-cell tumours. Ann Oncol 14:91–96

    Article  PubMed  Google Scholar 

  15. Comis RL (1992) Bleomycin pulmonary toxicity: current status and future directions. Semin Oncol 19:64–70

    CAS  PubMed  Google Scholar 

  16. Blum RH, Carter SK, Agre K (1973) A clinical review of bleomycin—a new antineoplastic agent. Cancer 31:903–914

    Article  CAS  PubMed  Google Scholar 

  17. Muir T, Kirsten M, Fourie P, Dippenaar N, Ionescu GO (2004) Intralesional bleomycin injection (IBI) treatment for haemangiomas and congenital vascular malformations. Pediatr Surg Int 19:766–773

    Article  CAS  PubMed  Google Scholar 

  18. Ionescu G, Mabeta P, Dippenaar N, Muir T, Fourie P, Shelver G (2008) Bleomycin plasma spill-over levels in paediatric patients undergoing intralesional injection for the treatment of haemangiomas. S Afr Med J 98:539–540

    CAS  PubMed  Google Scholar 

  19. Sleijfer S (2001) Bleomycin-induced pneumonitis. Chest 120:617–624

    Article  CAS  PubMed  Google Scholar 

  20. Atwa K, Abuhasna S, Shihab Z, Hashaykeh N, Hasan R (2010) Acute pulmonary toxicity following intralesional administration of bleomycin for a lymphovenous malformation. Pediatr Pulmonol 45:192–196

    Article  PubMed  Google Scholar 

  21. Mertens AC, Yasui Y, Liu Y et al (2002) Pulmonary complications in survivors of childhood and adolescent cancer. A report from the Childhood Cancer Survivor Study. Cancer 95:2431–2441

    Article  PubMed  Google Scholar 

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Conflict of interest

None of the authors have declared a conflict of interest.

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Authors and Affiliations

  1. Vascular Anomalies Center, Boston Children’s Hospital and Harvard Medical School, 300 Longwood Avenue, Boston, MA, 02115, USA

    Gulraiz Chaudry, Carlos J. Guevara, Kristy L. Rialon, Cindy Kerr, John B. Mulliken, Arin K. Greene, Steven J. Fishman, Debra Boyer & Ahmad I. Alomari

  2. Division of Vascular and Interventional Radiology, Boston Children’s Hospital and Harvard Medical School, 300 Longwood Avenue, Boston, MA, 02115, USA

    Gulraiz Chaudry, Carlos J. Guevara, Cindy Kerr & Ahmad I. Alomari

  3. Department of Surgery, Boston Children’s Hospital and Harvard Medical School, 300 Longwood Avenue, Boston, MA, 02115, USA

    Kristy L. Rialon & Steven J. Fishman

  4. Department of Plastic and Oral Surgery, Boston Children’s Hospital and Harvard Medical School, 300 Longwood Avenue, Boston, MA, 02115, USA

    John B. Mulliken & Arin K. Greene

  5. Department of Respiratory Diseases, Boston Children’s Hospital and Harvard Medical School, 300 Longwood Avenue, Boston, MA, 02115, USA

    Debra Boyer

Authors
  1. Gulraiz Chaudry
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  2. Carlos J. Guevara
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  3. Kristy L. Rialon
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  4. Cindy Kerr
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  5. John B. Mulliken
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  7. Steven J. Fishman
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  8. Debra Boyer
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  9. Ahmad I. Alomari
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Correspondence to Gulraiz Chaudry.

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Chaudry, G., Guevara, C.J., Rialon, K.L. et al. Safety and Efficacy of Bleomycin Sclerotherapy for Microcystic Lymphatic Malformation. Cardiovasc Intervent Radiol 37, 1476–1481 (2014). https://doi.org/10.1007/s00270-014-0932-z

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  • DOI: https://doi.org/10.1007/s00270-014-0932-z

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