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1. Lisle JW, Bradeen HA, Kalof AN: Kaposiform hemangioendothelioma in multiple spinal levels without skin changes. Clin Orthop Relat Res; 2009 Sep;467(9):2464-71
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  • [Title] Kaposiform hemangioendothelioma in multiple spinal levels without skin changes.
  • Kaposiform hemangioendothelioma is a rare vascular tumor of childhood that is locally aggressive but has little metastatic potential and by itself is not known to be lethal.
  • It most commonly presents as a superficial or deep soft tissue mass with associated cutaneous lesions.
  • Kasabach-Merritt phenomenon, a condition characterized by profound thrombocytopenia and life-threatening hemorrhage, often is associated with kaposiform hemangioendothelioma.
  • Six cases of kaposiform hemangioendothelioma have been reported in bone, two of which were located in extracraniofacial bones.
  • We report a diagnostically challenging case of a 6-year-old girl with kaposiform hemangioendothelioma of the thoracolumbar spine without Kasabach-Merritt phenomenon or cutaneous lesions.
  • [MeSH-major] Hemangioendothelioma / pathology. Sarcoma, Kaposi / pathology. Spinal Neoplasms / pathology
  • [MeSH-minor] Angiogenesis Inhibitors / therapeutic use. Anti-Inflammatory Agents, Non-Steroidal / therapeutic use. Antigens, CD31 / analysis. Biomarkers, Tumor / analysis. Celecoxib. Child. Drug Therapy, Combination. Female. Humans. Lumbar Vertebrae / pathology. Lumbar Vertebrae / radiography. Pain / etiology. Pyrazoles / therapeutic use. Sulfonamides / therapeutic use. Syndrome. Thalidomide / therapeutic use. Thoracic Vertebrae / pathology. Thoracic Vertebrae / radiography. Treatment Outcome

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  • (PMID = 19381744.001).
  • [ISSN] 1528-1132
  • [Journal-full-title] Clinical orthopaedics and related research
  • [ISO-abbreviation] Clin. Orthop. Relat. Res.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Angiogenesis Inhibitors; 0 / Anti-Inflammatory Agents, Non-Steroidal; 0 / Antigens, CD31; 0 / Biomarkers, Tumor; 0 / Pyrazoles; 0 / Sulfonamides; 4Z8R6ORS6L / Thalidomide; JCX84Q7J1L / Celecoxib
  • [Other-IDs] NLM/ PMC2866913
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2. López Gutiérrez JC, Patrón Romero M: [Thoracic kaposiform hemangioendothelioma. Four consecutive cases with distinct outcome]. An Pediatr (Barc); 2005 Jul;63(1):72-6
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  • [Title] [Thoracic kaposiform hemangioendothelioma. Four consecutive cases with distinct outcome].
  • [Transliterated title] Hemangioendotelioma kaposiforme torácico. Cuatro casos con evolución variable.
  • INTRODUCTION: Kaposiform hemangioendothelioma (KHE) is a rare, frequently congenital, neoplasm associated with the Kasabach-Merritt phenomenon (KMP) and predilection for the trunk.
  • In addition various and concurrent therapies can be used to treat this tumor and it is difficult to predict which treatment will be successful.
  • All patients developed KMP, without skin involvement in one patient.
  • Patient 3 showed no response to steroids, interferon or vincristine therapy.
  • Patient 4 underwent incomplete removal of the tumor on her right chest wall after showing no response to antiangiogenic therapy.
  • The coagulopathy persisted and a second radical surgical procedure resolved KMP.
  • CONCLUSION: Given the variable response to pharmacological treatment and the scant possibilities of surgical resection, the management of KHE with KMP must include a multidisciplinary approach.
  • [MeSH-major] Hemangioendothelioma. Thoracic Neoplasms
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Fatal Outcome. Humans. Infant. Infant, Newborn. Interferon-alpha / therapeutic use. Recombinant Proteins. Sarcoma, Kaposi. Syndrome. Thrombocytopenia / etiology

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  • (PMID = 15989875.001).
  • [ISSN] 1695-4033
  • [Journal-full-title] Anales de pediatría (Barcelona, Spain : 2003)
  • [ISO-abbreviation] An Pediatr (Barc)
  • [Language] spa
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Spain
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Interferon-alpha; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a
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3. Wilken JJ, Meier FA, Kornstein MJ: Kaposiform hemangioendothelioma of the thymus. Arch Pathol Lab Med; 2000 Oct;124(10):1542-4
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  • [Title] Kaposiform hemangioendothelioma of the thymus.
  • Kaposiform hemangioendothelioma is a rare pediatric neoplasm that presents most commonly in the soft tissues.
  • Immunoperoxidase studies on formalin-fixed, paraffin-embedded tissue demonstrated the neoplastic endothelial cells to be positive for vascular markers CD31 and CD34.
  • Although these tumors have been treated in a fashion similar to capillary hemangiomas in the past, it may be important to differentiate Kaposiform hemangioendotheliomas because of their association with Kasabach-Merritt syndrome and recent success with more aggressive chemotherapy regimens.
  • [MeSH-major] Hemangioendothelioma / pathology. Sarcoma, Kaposi / pathology. Thymus Neoplasms / pathology


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4. Saito M, Gunji Y, Kashii Y, Odaka J, Yamauchi T, Kanai N, Momoi MY: Refractory kaposiform hemangioendothelioma that expressed vascular endothelial growth factor receptor (VEGFR)-2 and VEGFR-3: a case report. J Pediatr Hematol Oncol; 2009 Mar;31(3):194-7
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  • [Title] Refractory kaposiform hemangioendothelioma that expressed vascular endothelial growth factor receptor (VEGFR)-2 and VEGFR-3: a case report.
  • This report describes the case of a 10-month-old boy who was diagnosed to have kaposiform hemangioendothelioma (KHE) with Kasabach-Merritt syndrome (KMS), which is a rare pediatric vascular tumor with a high mortality rate.
  • Although both KHE with KMS were resistant to various therapies, such as oral prednisolone, sclerotherapy, and chemotherapy, repeated radiation therapy with methylprednisolone pulse therapy did reduce the volume of KHE and improved the symptoms of KMS.
  • Unfortunately, a regrowth of KHE with KMS was observed 4 months after the cessation of treatment and the patient thereafter died from an intracranial hemorrhage and Pneumocystis carinii pneumonia, which is a complication related to repetitive radiation and steroid therapy.
  • A histopathologic examination of autopsy specimens confirmed a diagnosis of KHE and immunohistologic staining was positive for vascular endothelial growth factor receptor (VEGFR)-2 and VEGFR-3.
  • These findings may provide the rationale to further investigate the role of VEGFRs in the pathogenesis of KHE and also to elucidate its prognostic value, along with the application of inhibitors for VEGFRs for the treatment of refractory KHE.
  • [MeSH-major] Hemangioendothelioma / metabolism. Soft Tissue Neoplasms / metabolism. Vascular Endothelial Growth Factor Receptor-2 / biosynthesis. Vascular Endothelial Growth Factor Receptor-3 / biosynthesis
  • [MeSH-minor] Anti-Inflammatory Agents, Non-Steroidal / therapeutic use. Antineoplastic Combined Chemotherapy Protocols. Cyclophosphamide. Dactinomycin. Drug Resistance, Neoplasm. Guanidines / therapeutic use. Humans. Immunohistochemistry. Infant. Interferon-alpha / therapeutic use. Magnetic Resonance Imaging. Male. Oleic Acids / therapeutic use. Platelet Transfusion. Prednisolone / therapeutic use. Prognosis. Sclerosing Solutions / therapeutic use. Sclerotherapy. Thrombocytopenia / etiology. Thrombocytopenia / therapy. Vincristine

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  • (PMID = 19262246.001).
  • [ISSN] 1536-3678
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Anti-Inflammatory Agents, Non-Steroidal; 0 / Guanidines; 0 / Interferon-alpha; 0 / Oleic Acids; 0 / Sclerosing Solutions; 1CC1JFE158 / Dactinomycin; 3282-75-5 / ethanolamine oleate; 5J49Q6B70F / Vincristine; 8N3DW7272P / Cyclophosphamide; 9PHQ9Y1OLM / Prednisolone; EC 2.7.10.1 / Vascular Endothelial Growth Factor Receptor-2; EC 2.7.10.1 / Vascular Endothelial Growth Factor Receptor-3; Y25LQ0H97D / nafamostat; VAC protocol
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5. Blatt J, Stavas J, Moats-Staats B, Woosley J, Morrell DS: Treatment of childhood kaposiform hemangioendothelioma with sirolimus. Pediatr Blood Cancer; 2010 Dec 15;55(7):1396-8
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  • [Title] Treatment of childhood kaposiform hemangioendothelioma with sirolimus.
  • Limited experience suggests that it may have application to the treatment of vascular lesions.
  • We describe our experience with a 1-year-old female with a kaposiform hemangioendothelioma and Kasabach-Merritt phenomenon who had rapid and dramatic response to sirolimus (0.1 mg/kg/day).
  • This case provides further rationale for clinical trials of sirolimus in the treatment of vascular lesions.
  • [MeSH-major] Angiogenesis Inhibitors / therapeutic use. Antibiotics, Antineoplastic / therapeutic use. Hemangioendothelioma / drug therapy. Sirolimus / therapeutic use. Skin Neoplasms / drug therapy


6. Iwami D, Shimaoka S, Mochizuki I, Sakuma T: Kaposiform hemangioendothelioma of the mediastinum in a 7-month-old boy: a case report. J Pediatr Surg; 2006 Aug;41(8):1486-8
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  • [Title] Kaposiform hemangioendothelioma of the mediastinum in a 7-month-old boy: a case report.
  • Kaposiform hemangioendothelioma is an aggressive vascular proliferation that has been recognized as a separate entity from other childhood vascular tumors.
  • The authors present an unusual case of hemangioendothelioma arising in the mediastinum of a 7-month-old male infant with acute respiratory distress and Kasabach-Merritt syndrome.
  • This case had many histologic and clinical features consistent with kaposiform hemangioendothelioma.
  • [MeSH-major] Hemangioendothelioma / drug therapy. Mediastinal Neoplasms / drug therapy
  • [MeSH-minor] Airway Obstruction / etiology. Antineoplastic Agents / therapeutic use. Humans. Infant. Interferon-alpha / therapeutic use. Male. Recombinant Proteins. Respiratory Sounds / etiology

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  • (PMID = 16863862.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Interferon-alpha; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a
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7. López V, Martí N, Pereda C, Martín JM, Ramón D, Mayordomo E, Jordá E: Successful management of Kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon using vincristine and ticlopidine. Pediatr Dermatol; 2009 May-Jun;26(3):365-6
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  • [Title] Successful management of Kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon using vincristine and ticlopidine.
  • Kaposiform hemangioendothelioma is a rare locally aggressive vascular tumor that usually occurs in skin and retroperitoneum of infants and young children.
  • We present a case of a newborn with a rapid tumor growth and a life-threatening Kasabach-Merritt syndrome with a progressive remission after treatment with vincristine and ticlopidine.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / administration & dosage. Blood Coagulation Disorders / complications. Fibrinolytic Agents / administration & dosage. Hemangioendothelioma / drug therapy. Skin Neoplasms / drug therapy. Ticlopidine / administration & dosage. Vincristine / administration & dosage


8. Hauer J, Graubner U, Konstantopoulos N, Schmidt S, Pfluger T, Schmid I: Effective treatment of kaposiform hemangioendotheliomas associated with Kasabach-Merritt phenomenon using four-drug regimen. Pediatr Blood Cancer; 2007 Nov;49(6):852-4
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  • [Title] Effective treatment of kaposiform hemangioendotheliomas associated with Kasabach-Merritt phenomenon using four-drug regimen.
  • We report a case of a 6-month-old girl suffering from a kaposiform hemangioendothelioma of the chest wall, associated with Kasabach-Merritt phenomenon.
  • Despite rapid intervention with cortisone and interferon alpha the tumor led to a life-threatening clinical condition with progressive growth and consumption coagulopathy under therapy.
  • Because therapy for kaposiform hemangioendotheliomas with a single anti-angiogenic or anti-proliferative agent has not been reported to be very successful, we administered vincristine, combined with cyclophosphamide, actinomycin D, and methotrexate in a critically ill patient.
  • After six cycles of the applied four-drug regiment, the infant was in remission, which has been maintained for 5 months since stopping therapy.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Hemangioendothelioma / drug therapy. Thoracic Neoplasms / drug therapy

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  • [Copyright] (c) 2006 Wiley-Liss, Inc.
  • (PMID = 16411198.001).
  • [ISSN] 1545-5009
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Interferon-alpha; 1CC1JFE158 / Dactinomycin; 5J49Q6B70F / Vincristine; 8N3DW7272P / Cyclophosphamide; V27W9254FZ / Cortisone; YL5FZ2Y5U1 / Methotrexate
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9. Fahrtash F, McCahon E, Arbuckle S: Successful treatment of kaposiform hemangioendothelioma and tufted angioma with vincristine. J Pediatr Hematol Oncol; 2010 Aug;32(6):506-10
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  • [Title] Successful treatment of kaposiform hemangioendothelioma and tufted angioma with vincristine.
  • BACKGROUND: Kaposiform hemangioendothelioma (KHE) and tufted angioma (TA) are rare, locally aggressive vascular tumors.
  • To date, definitive treatment for these vascular tumors has had limited success.
  • AIM: To evaluate the safety, efficacy, and role of vincristine in the treatment of KHE and TA.
  • Mean age of diagnosis was 30 months (range birth to 9 y).
  • Mean time of the follow-up was 4 years (range 4 mo to 8 y).
  • CONCLUSIONS: Vincristine is an effective treatment option for KHE/TA.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Hemangioendothelioma / drug therapy. Hemangioma / drug therapy. Vincristine / therapeutic use


10. Harper L, Michel JL, Enjolras O, Raynaud-Mounet N, Rivière JP, Heigele T, De Napoli-Cocci S: Successful management of a retroperitoneal kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon using alpha-interferon. Eur J Pediatr Surg; 2006 Oct;16(5):369-72
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  • [Title] Successful management of a retroperitoneal kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon using alpha-interferon.
  • It has been shown recently that Kasabach-Merritt phenomenon, the association of a vascular tumour and consumption coagulopathy, does not--as previously thought--complicate "classical" infantile hemangiomas but distinctive entities called kaposiform hemangioendothelioma (KHE) and tufted angioma (TA), both tumours on the same neoplastic spectrum.
  • Several therapeutic modalities, including alpha-interferon, vincristine, radiotherapy and surgery have been reported in the literature.
  • We report a case of retroperitoneal kaposiform hemangioendothelioma regression using alpha-interferon and discuss the current knowledge of this entity and its treatment.
  • [MeSH-major] Angiogenesis Inhibitors / therapeutic use. Hemangioendothelioma / drug therapy. Interferon-alpha / therapeutic use

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  • (PMID = 17160787.001).
  • [ISSN] 0939-7248
  • [Journal-full-title] European journal of pediatric surgery : official journal of Austrian Association of Pediatric Surgery ... [et al] = Zeitschrift für Kinderchirurgie
  • [ISO-abbreviation] Eur J Pediatr Surg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Angiogenesis Inhibitors; 0 / Interferon-alpha
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11. Drucker AM, Pope E, Mahant S, Weinstein M: Vincristine and corticosteroids as first-line treatment of Kasabach-Merritt syndrome in kaposiform hemangioendothelioma. J Cutan Med Surg; 2009 May-Jun;13(3):155-9
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  • [Title] Vincristine and corticosteroids as first-line treatment of Kasabach-Merritt syndrome in kaposiform hemangioendothelioma.
  • BACKGROUND: Historically, patients with the consumptive coagulopathy Kasabach-Merritt syndrome (KMS) have been treated with systemic corticosteroids as first-line therapy, but many patients do not respond.
  • Recently, there have been increasing reports of the use of the chemotherapeutic drug vincristine in these patients.
  • OBJECTIVE: To report a case of a newborn with a kaposiform hemangioendothelioma (KHE) of the right leg associated with KMS treated successfully with vincristine and oral corticosteroids.
  • RESULTS: Treatment with vincristine and corticosteroids lead to sustained shrinking of the tumor and correction of the thrombocytopenia and coagulopathy through 1 year of age.
  • We believe this is the first report in the North American literature of corticosteroids and vincristine being used concomitantly as first-line therapy for KHE with KMS.
  • CONCLUSION: Vincristine and corticosteroids should be considered first-line treatment for KMS.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / therapeutic use. Blood Coagulation Disorders / drug therapy. Glucocorticoids / therapeutic use. Hemangioendothelioma / drug therapy. Skin / pathology. Vincristine / therapeutic use. Xeroderma Pigmentosum / drug therapy
  • [MeSH-minor] Administration, Oral. Biopsy. Diagnosis, Differential. Drug Therapy, Combination. Female. Follow-Up Studies. Humans. Infant, Newborn. Syndrome


12. Fernández Y, Bernabeu-Wittel M, García-Morillo JS: Kaposiform hemangioendothelioma. Eur J Intern Med; 2009 Mar;20(2):106-13
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  • [Title] Kaposiform hemangioendothelioma.
  • Kaposiform hemangioendothelioma (KHE) is a rare, locally aggressive vascular neoplasm that mainly occurs during childhood.
  • It generally originates on the skin, usually affecting deeper tissue by infiltrative growth.
  • Several factors are associated with the outcome of patients with KHE: accessibility to surgical excision, location (cutaneous versus visceral), size of tumoral mass, clinical response to interferon and glucocorticoids, and the absence of lymphangiomatosis and Kasabach-Merritt syndrome, may result in partial remissions.
  • On the other hand, bulk visceral masses lead to a 40-50% mortality rate, mainly due to progressive failure of the infiltrated organ(s), in spite of interferon, glucocorticoids, and combined chemotherapy.
  • [MeSH-major] Hemangioendothelioma / diagnosis. Hemangioendothelioma / therapy. Skin Neoplasms / diagnosis. Skin Neoplasms / therapy. Vascular Neoplasms / diagnosis. Vascular Neoplasms / therapy

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  • (PMID = 19327597.001).
  • [ISSN] 1879-0828
  • [Journal-full-title] European journal of internal medicine
  • [ISO-abbreviation] Eur. J. Intern. Med.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Netherlands
  • [Number-of-references] 87
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13. Fernandez-Pineda I, Lopez-Gutierrez JC, Ramirez G, Marquez C: Vincristine-ticlopidine-aspirin: an effective therapy in children with Kasabach-Merritt phenomenon associated with vascular tumors. Pediatr Hematol Oncol; 2010 Nov;27(8):641-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Vincristine-ticlopidine-aspirin: an effective therapy in children with Kasabach-Merritt phenomenon associated with vascular tumors.
  • Kasabach-Merritt phenomenon (KMP) is a serious coagulopathy with severe thrombocytopenia (<10,000/mm³) that occurs in the presence of an enlarging vascular tumor such as kaposiform hemangioendothelioma (KHE) and tufted angioma (TA).
  • The natural history and treatment of these lesions remain controversial.
  • The authors report a KHE case and a TA case that presented with KMP, describing their successful pharmacological management with vincristine, ticlopidine, and aspirin.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Aspirin / therapeutic use. Ticlopidine / therapeutic use. Vascular Neoplasms / drug therapy. Vincristine / therapeutic use
  • [MeSH-minor] Disseminated Intravascular Coagulation / complications. Disseminated Intravascular Coagulation / diagnosis. Disseminated Intravascular Coagulation / drug therapy. Female. Hemangioma, Capillary / complications. Hemangioma, Capillary / diagnosis. Hemangioma, Capillary / drug therapy. Humans. Infant. Kasabach-Merritt Syndrome. Treatment Outcome

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  • (PMID = 20863161.001).
  • [ISSN] 1521-0669
  • [Journal-full-title] Pediatric hematology and oncology
  • [ISO-abbreviation] Pediatr Hematol Oncol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 5J49Q6B70F / Vincristine; OM90ZUW7M1 / Ticlopidine; R16CO5Y76E / Aspirin
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14. Metry DW, Hebert AA: Benign cutaneous vascular tumors of infancy: when to worry, what to do. Arch Dermatol; 2000 Jul;136(7):905-14
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Benign cutaneous vascular tumors of infancy: when to worry, what to do.
  • To discuss 2 newly recognized entities that may be a source of diagnostic confusion with the common hemangioma, the kaposiform hemangioendothelioma and tufted angioma.
  • To discuss the risks and benefits of current treatment options, including the use of systemic corticosteroids and interferon in necessary situations.
  • CONCLUSIONS: The common hemangioma, kaposiform hemangioendothelioma, and tufted angioma, though benign histologically, may cause serious consequences for children.
  • Dermatologists should also be aware of available treatment options, including the use of systemic chemotherapy in life-threatening situations.
  • [MeSH-minor] Abnormalities, Multiple. Hemangioendothelioma / congenital. Hemangioendothelioma / diagnosis. Hemangioendothelioma / pathology. Hemangioendothelioma / therapy. Humans. Infant. Infant, Newborn. Syndrome

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  • (PMID = 10890993.001).
  • [ISSN] 0003-987X
  • [Journal-full-title] Archives of dermatology
  • [ISO-abbreviation] Arch Dermatol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] UNITED STATES
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15. Rodriguez V, Lee A, Witman PM, Anderson PA: Kasabach-merritt phenomenon: case series and retrospective review of the mayo clinic experience. J Pediatr Hematol Oncol; 2009 Jul;31(7):522-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Kasabach-Merritt phenomenon (KMP) is a rare thrombocytopenic consumption coagulopathy associated with an enlarging tufted angioma or kaposiform hemangioendothelioma.
  • We report a case series of patients with KMP and discuss the treatment options and outcomes.
  • Nine patients with a diagnosis of KMP were identified through retrospective chart review-6 had "definite KMP" and 3 had "less likely KMP."
  • [MeSH-major] Disseminated Intravascular Coagulation / complications. Disseminated Intravascular Coagulation / physiopathology. Hemangioendothelioma / complications. Hemangioma / complications. Skin Neoplasms / complications
  • [MeSH-minor] Anti-Inflammatory Agents / therapeutic use. Antineoplastic Agents / therapeutic use. Female. Humans. Infant. Male. Radiotherapy. Thrombocytopenia / complications. Thrombocytopenia / drug therapy

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  • (PMID = 19564750.001).
  • [ISSN] 1536-3678
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Anti-Inflammatory Agents; 0 / Antineoplastic Agents
  • [Number-of-references] 10
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16. Haisley-Royster C, Enjolras O, Frieden IJ, Garzon M, Lee M, Oranje A, de Laat PC, Madern GC, Gonzalez F, Frangoul H, Le Moine P, Prose NS, Adams DM: Kasabach-merritt phenomenon: a retrospective study of treatment with vincristine. J Pediatr Hematol Oncol; 2002 Aug-Sep;24(6):459-62
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  • [Title] Kasabach-merritt phenomenon: a retrospective study of treatment with vincristine.
  • The purpose of this study was to assess the effectiveness of vincristine in the treatment of KMP.
  • Ten patients had biopsies of their lesions, and results included five (33.3%) kaposiform hemangioendotheliomas, three (20%) tufted angiomas, one lesion (6.7%) with features of both kaposiform hemangioendothelioma and tufted angioma, and one (6.7%) unclassified vascular tumor.
  • All 15 patients had an increase in platelet count of at least 20,000 with an average response time of 4.0 weeks after initiation of vincristine therapy.
  • Thirteen patients had an increase in fibrinogen level of 50 mg/dL with an average response time of 3.4 weeks.
  • The average duration of treatment was 21.5 (+/-12.6) weeks.
  • CONCLUSION: Vincristine presents a safe and sometimes effective treatment option in the management of KMP.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / therapeutic use. Hemangioendothelioma / drug therapy. Hemangioma, Capillary / drug therapy. Thrombocytopenia / drug therapy. Vincristine / therapeutic use

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  • [ErratumIn] J Pediatr Hematol Oncol 2002 Dec;24(9):794
  • (PMID = 12218593.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Journal Article; Multicenter Study
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Phytogenic; 5J49Q6B70F / Vincristine; 9001-32-5 / Fibrinogen
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17. Vogel AM, Alesbury JM, Burrows PE, Fishman SJ: Vascular anomalies of the female external genitalia. J Pediatr Surg; 2006 May;41(5):993-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Tumors included 20 infantile hemangiomas, 1 kaposiform hemangioendothelioma, and 1 kaposiform lymphangioendothelioma.
  • The referring diagnosis was incorrect in 56% of patients.
  • Cutaneous stains, swelling, deformity, bleeding, fluid leakage, or infection were the prominent symptoms.
  • Magnetic resonance imaging, ultrasonography, angiography, and computed tomography were used for diagnostic clarification.
  • Malformation treatment consisted of sclerotherapy, embolization, and operative resection.
  • Tumor management included observation, surgical excision, and antiangiogenic pharmacotherapy.
  • CONCLUSIONS: Vascular anomalies of the female external genitalia are uncommon, and the initial diagnosis is often inaccurate.
  • Correct diagnosis using clinical and radiographic data is feasible and leads to meaningful intervention for these frequently devastating lesions.
  • Whereas tumors may respond to excision or antiangiogenic drugs, malformations require ablation or resection.
  • [MeSH-major] Blood Vessels / abnormalities. Genital Neoplasms, Female / surgery. Genitalia, Female / blood supply. Genitalia, Female / surgery. Vascular Neoplasms / surgery

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  • (PMID = 16677899.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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18. Maguiness S, Guenther L: Kasabach-merritt syndrome. J Cutan Med Surg; 2002 Jul-Aug;6(4):335-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • There are currently no known treatment guidelines.
  • Kasabach-Merritt syndrome is associated with kaposiform hemangioendothelioma (KHE) and tufted angioma (TA); these lesions, when associated with KMS, are locally invasive, aggressive vascular tumors.
  • Treatment options include supportive care, local therapies, and drug and surgical management.
  • In most recent case reports, a multimodal approach to therapy is taken.
  • OBJECTIVE AND CONCLUSION: The objective of this article is to provide a comprehensive review of KMS and give an up-to-date summary of treatment options.
  • [MeSH-major] Hemangioendothelioma / pathology. Hemangioendothelioma / therapy

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  • (PMID = 11951132.001).
  • [ISSN] 1203-4754
  • [Journal-full-title] Journal of cutaneous medicine and surgery
  • [ISO-abbreviation] J Cutan Med Surg
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 28
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19. Muzaffar AR, Friedrich JB, Lu KK, Hanel DP: Infantile fibrosarcoma of the hand associated with coagulopathy. Plast Reconstr Surg; 2006 Apr 15;117(5):81e-86e
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • BACKGROUND: Large congenital neoplasms of the extremities may be associated with coagulopathies and significant hemorrhage in the neonatal period.
  • At times, the differences between coagulation derangements can be very subtle, leading to errors in diagnosis.
  • It is important to obtain accurate diagnoses of the neoplasm and the coagulopathy because the treatments of similar-appearing tumors and coagulopathies can be markedly different.
  • METHODS: The authors report the case of a newborn with a congenital tumor of the left hand that was accompanied by a coagulopathy that caused significant bleeding.
  • RESULTS: A presumption was made by the neonatal critical care physicians and hematologists that the infant had a kaposiform hemangioendothelioma along with the Kasabach-Merritt phenomenon.
  • However, steroid treatment did not reduce the size of the mass or correct the coagulopathy.
  • Only after obtaining consultation with a hand surgeon and a tissue diagnosis was it learned that the patient had an infantile fibrosarcoma that was accompanied by disseminated intravascular coagulation.
  • Limb-sparing resection of the lesion along with chemotherapy markedly improved the patient's condition.
  • CONCLUSIONS: Large congenital neoplasms presenting with attendant bleeding diatheses must be rapidly and accurately diagnosed with both a biopsy-proven tissue diagnosis and a hematologic characterization of the nature of the coagulopathy.
  • The differential diagnosis of a vascular-appearing mass in the extremity can be subtle, and presumptive diagnosis, as occurred in this case, can lead to incorrect or delayed treatment.
  • Specifically, kaposiform hemangioendothelioma must be differentiated from infantile fibrosarcoma.
  • The principles of infantile fibrosarcoma treatment are limb-sparing resection and chemotherapy.
  • [MeSH-major] Fibrosarcoma / congenital. Fibrosarcoma / diagnosis. Hand

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  • (PMID = 16641700.001).
  • [ISSN] 1529-4242
  • [Journal-full-title] Plastic and reconstructive surgery
  • [ISO-abbreviation] Plast. Reconstr. Surg.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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20. Tamai N, Hashii Y, Osuga K, Chihara T, Morii E, Aozasa K, Yoshikawa H: Kaposiform hemangioendothelioma arising in the deltoid muscle without the Kasabach-Merritt phenomenon. Skeletal Radiol; 2010 Oct;39(10):1043-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Kaposiform hemangioendothelioma arising in the deltoid muscle without the Kasabach-Merritt phenomenon.
  • Kaposiform hemangioendothelioma (KHE) is a rare tumor that occurs nearly exclusively during infancy and childhood.
  • It has features common to both capillary hemangioma and Kaposi sarcoma and for that reason many terms have been used for these tumors including "Kaposi-like infantile hemangioendothelioma" and "hemangioma with Kaposi sarcoma-like features."
  • Knowledge of the radiological findings of this uncommon tumor might be helpful for diagnosis.
  • [MeSH-major] Head and Neck Neoplasms / diagnosis
  • [MeSH-minor] Antineoplastic Agents, Hormonal / therapeutic use. Deltoid Muscle / pathology. Deltoid Muscle / ultrastructure. Diagnosis, Differential. Disseminated Intravascular Coagulation. Hemangioendothelioma / diagnosis. Hemangioendothelioma / drug therapy. Hemangioendothelioma / ultrastructure. Hemangioma, Capillary. Hemangioma, Cavernous / diagnosis. Hemangioma, Cavernous / drug therapy. Hemangioma, Cavernous / ultrastructure. Humans. Infant. Kasabach-Merritt Syndrome. Magnetic Resonance Imaging / methods. Male. Prednisolone / therapeutic use. Sarcoma, Kaposi / diagnosis. Sarcoma, Kaposi / drug therapy. Sarcoma, Kaposi / ultrastructure. Skin Neoplasms / diagnosis. Skin Neoplasms / drug therapy. Skin Neoplasms / ultrastructure. Thrombocytopenia / diagnosis. Thrombocytopenia / drug therapy. Treatment Outcome. Vascular Neoplasms / diagnosis. Vascular Neoplasms / drug therapy. Vascular Neoplasms / ultrastructure

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  • (PMID = 20309545.001).
  • [ISSN] 1432-2161
  • [Journal-full-title] Skeletal radiology
  • [ISO-abbreviation] Skeletal Radiol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Hormonal; 9PHQ9Y1OLM / Prednisolone; Kaposiform Hemangioendothelioma
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21. Minard-Colin V, Orbach D, Martelli H, Bodemer C, Oberlin O: [Soft tissue tumors in neonates]. Arch Pediatr; 2009 Jul;16(7):1039-48

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Soft tissue tumors in neonates].
  • Soft tissue tumors account for approximately 25% of neonatal tumors and are most often benign (more than 2/3 of cases).
  • Kaposiform hemangioendothelioma (KH) is a rare vascular tumor with locally aggressive behavior.
  • Malignant soft tissue tumors are, after neuroblastoma, the second cause of cancer in neonates.
  • Chemotherapy is indicated when initial surgical removal cannot be accomplished without unacceptable morbidity.
  • Treatment is based on age-adapted chemotherapy and surgery.
  • [MeSH-major] Soft Tissue Neoplasms / congenital
  • [MeSH-minor] Fibrosarcoma / congenital. Fibrosarcoma / diagnosis. Fibrosarcoma / genetics. Fibrosarcoma / therapy. Gene Fusion / genetics. Gene Rearrangement / genetics. Hemangioendothelioma / congenital. Hemangioendothelioma / diagnosis. Hemangioendothelioma / therapy. Hemangioma / congenital. Hemangioma / diagnosis. Hemangioma / therapy. Humans. Infant, Newborn. Prognosis. Proto-Oncogene Proteins c-ets / genetics. Receptor, trkC / genetics. Repressor Proteins / genetics. Rhabdomyosarcoma / congenital. Rhabdomyosarcoma / diagnosis. Rhabdomyosarcoma / therapy

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  • (PMID = 19398311.001).
  • [ISSN] 1769-664X
  • [Journal-full-title] Archives de pédiatrie : organe officiel de la Sociéte française de pédiatrie
  • [ISO-abbreviation] Arch Pediatr
  • [Language] fre
  • [Publication-type] English Abstract; Journal Article; Review
  • [Publication-country] France
  • [Chemical-registry-number] 0 / ETS translocation variant 6 protein; 0 / Proto-Oncogene Proteins c-ets; 0 / Repressor Proteins; EC 2.7.10.1 / Receptor, trkC
  • [Number-of-references] 37
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22. Deb G: Kaposiform hemangioendothelioma and therapy with interferon-alpha. Med Pediatr Oncol; 2003 Dec;41(6):593
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  • [Title] Kaposiform hemangioendothelioma and therapy with interferon-alpha.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Hemangioendothelioma / drug therapy. Interferon-alpha / therapeutic use. Sarcoma, Kaposi / drug therapy
  • [MeSH-minor] Biopsy. Humans. Infant. Prognosis. Steroids / therapeutic use. Vascular Endothelial Growth Factor A / blood

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  • [CommentOn] Med Pediatr Oncol. 2002 Jun;38(6):424-7 [11984805.001]
  • (PMID = 14595731.001).
  • [ISSN] 0098-1532
  • [Journal-full-title] Medical and pediatric oncology
  • [ISO-abbreviation] Med. Pediatr. Oncol.
  • [Language] eng
  • [Publication-type] Comment; Letter
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Interferon-alpha; 0 / Steroids; 0 / Vascular Endothelial Growth Factor A
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23. Chung MT, Chen CH, Chiu CH, Yang CP, Hsueh C, Jaing TH: Successful nonoperative therapy for Kaposiform hemangioendothelioma involving the neck: report of 1 case. Otolaryngol Head Neck Surg; 2003 Nov;129(5):605-7
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  • [Title] Successful nonoperative therapy for Kaposiform hemangioendothelioma involving the neck: report of 1 case.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Head and Neck Neoplasms / drug therapy. Head and Neck Neoplasms / pathology. Hemangioendothelioma / drug therapy. Hemangioendothelioma / pathology. Interferons / therapeutic use. Sarcoma, Kaposi / pathology






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