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Kasabach-Merritt Syndrome Treatment & Management

  • Author: Alexandra C Cheerva, MD, MS; Chief Editor: Hassan M Yaish, MD  more...
 
Updated: May 24, 2016
 

Approach Considerations

Management of Kasabach-Merritt syndrome (KMS) involves hastening vascular lesion regression, interfering with platelet trapping within the lesion, and supporting the patient with transfusions.

Patients with KMS are admitted to the hospital with profound thrombocytopenia and evidence of coagulopathy. They are commonly treated with drug therapy initially[15, 16, 17, 18, 19, 20, 21, 22, 23] and with surgical or interventional radiologic measures either simultaneously or subsequently.[13, 24, 25, 26, 27]

Selected patients who have KMS with absent or mild thrombocytopenia and coagulopathy may be evaluated on an outpatient basis. However, as vascular lesions grow, these infants often require inpatient evaluation and treatment because most develop significant thrombocytopenia and disseminated intravascular coagulation (DIC).

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Pharmacologic Therapy

No single therapy has been proved most effective in patients with KMS. Multiple treatments have been used in many infants. Success rates have varied. Treatments that are effective in some patients have no benefit for others. Many of these medications have serious adverse effects, especially in patients with thrombocytopenia and DIC, and should be administered only by physicians with expertise in this area. Most of them are not specifically approved by the US Food and Drug Administration (FDA) for treatment of KMS.

Corticosteroids are the drugs most commonly used and have been successful in many cases, though they do not always yield satisfactory results.[28, 26] In addition to high-dose oral steroids, pulsed or intravenous (IV) steroids have also been used.

Some success with interferon alfa-2a (3 million U/m2/day or 3 times weekly) has been reported, though the failure rate is high.[29, 30, 31] The subcutaneous injection may result in nausea, fever, or neutropenia. Interferon alfa has been associated with neurologic problems in certain patients[32, 33] ; some infants have developed spastic diplegia after receiving this agent.

Other medications, including ε-aminocaproic acid, aspirin, dipyridamole,[18] ticlopidine, pentoxifylline,[33] cryoprecipitate, and heparin, have also been used to treat KMS, with varying degrees of efficacy.

Chemotherapy, including vincristine, cyclophosphamide, and actinomycin D, has been successfully employed in some patients.[34, 17, 35, 36, 37] In particular, vincristine is increasingly often being given to treat KMS.[19, 20, 21, 23, 38, 39] Nevertheless, there have been treatment failures.

In 2008, Leaute-Labreze et al reported using propranolol in 2 infants with severe hemangiomas.[40] Potential explanations of the therapeutic effect included vasoconstriction, decreased expression of VEGF and bFGF genes, and the triggering of apoptosis of capillary endothelial cells.

A study by Su et al found that transcatheter arterial embolization with polyvinyl alcohol particles combined with drug therapy achieved good results in six infants with KMS. The patients, with lesions in the temporal region, parotid region, or submandibular region and neck, experienced shrinkage and/or lightening of the hemangioma, while the mean platelet count reached over 80,000/L in five patients; one patient achieved a platelet count of 102,000/L. Neither hemangioma rebound growth nor platelet count reduction were found at 12- to 18-month follow-up.[41]

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Surgical and Endovascular Interventions

Surgical management often is not feasible in KMS, because the lesions are too large. When the lesions are sufficiently small, surgery has resulted in a rapid normalization of the platelet count and other blood abnormalities. Complete surgical resection, when possible, is the most effective treatment of vascular lesions complicated by KMS.[42] Thrombocytopenia and DIC resolve after the lesion has been resected.

Depending on the location of the tumor, general surgery, thoracic surgery, or neurosurgery may be needed for excision or ligation of the vessels feeding the vascular lesion. Wide local excision is recommended but may be difficult. The large size and infiltrative growth pattern of the vascular lesion may cause complications (eg, hemorrhage, obstruction, and respiratory compromise). Amputation may be necessary for intractable lesions involving a limb.

Some lesions are surgically inaccessible and thus call for nonsurgical treatment modalities. Interventional radiologic procedures use polyvinyl alcohol or absolute ethanol to embolize or sclerose the vessels of the vascular lesion.[27, 43] Another technique involves injecting polyvinyl beads to stop the feeder blood supply. Endovascular treatment (eg, transfemoral arterial embolization) has yielded good results in a number of cases.[44, 45, 46, 28, 47]

Treatment with the tunable dye laser (TDL) may help patients with diffuse cutaneous vascular lesions.[48]

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Pneumatic Compression

Treatment with intermittent pneumatic compression, either alone or in combination with other therapies, has helped some patients.[49] This modality is most useful when the vascular lesion is located on an extremity.

Encircle the lesion with a blood pressure cuff, and gradually increase the pressure to midway between the systolic and diastolic blood pressures. Inflate the cuff intermittently (eg, 90 seconds of compression, then 30 seconds of rest). A cuff may be used for an extended period. Compression with surgical hose stockings may help. It is often worthwhile to attempt pneumatic compression to see if it helps before attempting other potentially more toxic treatments.

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Radiation Therapy

Radiation therapy has been used in patients with severe disease.[13, 50] It has also been combined with interferon alfa therapy, with some success.[51] Although low radiation doses have been reported to offer benefit in some cases, the long-term adverse effects of radiotherapy must be carefully weighed against the problems caused by the vascular lesion—especially in very young children, who are at risk for malignancy or decreased bone growth in the radiated field with this treatment.

Currently, because of the long-term adverse effects of radiation therapy in survivors, this mode of therapy is no longer favored for KMS, and many practitioners have abandoned it.

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Consultations

A pediatric hematology consultation is required for management of the complex hemostatic problems of KMS patients and for administration and management of many of the medications needed for the most fulminant cases. Additional consultations that may be advisable are as follows:

  • Dermatologist
  • Surgeon
  • Pediatrician
  • Interventional radiologist

Consultations with neonatal or pediatric intensive care specialists may be required, depending on the age of the patient.

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Contributor Information and Disclosures
Author

Alexandra C Cheerva, MD, MS Associate Professor of Pediatrics, University of Louisville School of Medicine; Director of Pediatric Blood and Marrow Transplantation, Section of Pediatric Hematology and Oncology, Kosair Children's Hospital

Alexandra C Cheerva, MD, MS is a member of the following medical societies: American Society for Blood and Marrow Transplantation, Children's Oncology Group, American Society of Clinical Oncology, International Pediatric Transplant Association, American Society of Pediatric Hematology/Oncology, Kentucky Medical Association

Disclosure: Nothing to disclose.

Coauthor(s)

Ashok B Raj, MD Professor, Section of Pediatric Hematology and Oncology, Department of Pediatrics, Kosair Children's Hospital, University of Louisville School of Medicine

Ashok B Raj, MD is a member of the following medical societies: American Academy of Pediatrics, American Society of Pediatric Hematology/Oncology, Kentucky Medical Association, Children's Oncology Group

Disclosure: Nothing to disclose.

Salvatore Bertolone, MD Director, Division of Pediatric Hematology/Oncology, Kosair Children's Hospital; Professor, Department of Pediatrics, University of Louisville School of Medicine

Salvatore Bertolone, MD is a member of the following medical societies: American Academy of Pediatrics, American Association for Cancer Education, American Association of Blood Banks, American Cancer Society, American Society of Hematology, American Society of Pediatric Hematology/Oncology, Kentucky Medical Association

Disclosure: Nothing to disclose.

Chief Editor

Hassan M Yaish, MD Medical Director, Intermountain Hemophilia and Thrombophilia Treatment Center; Professor of Pediatrics, University of Utah School of Medicine; Director of Hematology, Pediatric Hematologist/Oncologist, Department of Pediatrics, Primary Children's Medical Center

Hassan M Yaish, MD is a member of the following medical societies: American Academy of Pediatrics, New York Academy of Sciences, American Medical Association, American Society of Hematology, American Society of Pediatric Hematology/Oncology, Michigan State Medical Society

Disclosure: Nothing to disclose.

Acknowledgements

Emmanuel C Besa, MD Professor, Department of Medicine, Division of Hematologic Malignancies and Hematopoietic Stem Cell Transplantation, Kimmel Cancer Center, Jefferson Medical College of Thomas Jefferson University

Emmanuel C Besa, MD is a member of the following medical societies: American Association for Cancer Education, American College of Clinical Pharmacology, American Federation for Medical Research, American Society of Clinical Oncology, American Society of Hematology, and New York Academy of Sciences

Disclosure: Nothing to disclose.

Gary D Crouch, MD Associate Professor, Program Director of Pediatric Hematology-Oncology Fellowship, Department of Pediatrics, Uniformed Services University of the Health Sciences

Gary D Crouch, MD is a member of the following medical societies: American Academy of Pediatrics and American Society of Hematology

Disclosure: Nothing to disclose. Guy B Faguet, MD Former Professor, Department of Medicine, Section of Hematology and Oncology, Medical College of Georgia

Guy B Faguet, MD is a member of the following medical societies: American Association of Immunologists, American Society of Hematology, International Society of Hematology, New York Academy of Sciences, Southern Medical Association, and Southern Society for Clinical Investigation

Disclosure: Nothing to disclose.

Linda K Hendricks, MD Assistant Professor, Department of Internal Medicine, Section of Hematology and Oncology, Mercer University School of Medicine

Linda K Hendricks, MD is a member of the following medical societies: American Society of Hematology

Disclosure: Nothing to disclose.

Gary R Jones, MD Associate Medical Director, Clinical Development, Berlex Laboratories

Gary R Jones, MD is a member of the following medical societies: American Academy of Pediatrics, American Society of Pediatric Hematology/Oncology, and Western Society for Pediatric Research

Disclosure: Nothing to disclose.

Michael Paul Kosty, MD Associate Director, Associate Professor, Department of Internal Medicine, Divisions of Supportive Care Services and Hematology and Oncology, Ida M and Cecil H Green Cancer Center, Scripps Clinic

Michael Paul Kosty, MD is a member of the following medical societies: American College of Physicians, American Society of Hematology, and Phi Beta Kappa

Disclosure: Nothing to disclose.

Bernice R Krafchik, MBChB, FRCPC Professor Emeritus, Department of Pediatrics, Section of Dermatology, University of Toronto

Bernice R Krafchik, MBChB, FRCPC is a member of the following medical societies: American Academy of Dermatology, American Dermatological Association, Canadian Medical Association, College of Physicians and Surgeons of Ontario, Royal College of Physicians and Surgeons of Canada, and Society for Pediatric Dermatology

Disclosure: Nothing to disclose.

Sejal Kuthiala, MD Staff Physician, Department of Internal Medicine, Medical College of Georgia

Sejal Kuthiala, MD is a member of the following medical societies: American Medical Association

Disclosure: Nothing to disclose.

Ronald A Sacher, MB, BCh, MD, FRCPC Professor, Internal Medicine and Pathology, Director, Hoxworth Blood Center, University of Cincinnati Academic Health Center

Ronald A Sacher, MB, BCh, MD, FRCPC is a member of the following medical societies: American Association for the Advancement of Science, American Association of Blood Banks, American Clinical and Climatological Association, American Society for Clinical Pathology, American Society of Hematology, College of American Pathologists, International Society of Blood Transfusion, International Society on Thrombosis and Haemostasis, and Royal College of Physicians and Surgeons of Canada

Disclosure: Glaxo Smith Kline Honoraria Speaking and teaching; Talecris Honoraria Board membership

Carlos Suarez, MD Associate Professor, Department of Pediatrics, Section of Pediatric Hematology and Oncology, University of Louisville School of Medicine

Disclosure: Nothing to disclose.

Francisco Talavera, PharmD, PhD Adjunct Assistant Professor, University of Nebraska Medical Center College of Pharmacy; Editor-in-Chief, Medscape Drug Reference

Disclosure: Medscape Salary Employment

Mary L Windle, PharmD Adjunct Associate Professor, University of Nebraska Medical Center College of Pharmacy; Editor-in-Chief, Medscape Drug Reference

Disclosure: Nothing to disclose.

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Leg with a Kaposiform hemangioendothelioma, lesion associated with Kasabach-Merritt Syndrome.
Back of an arm showing the typical bruising associated with Kasabach-Merritt Syndrome.
 
 
 
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