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1. Al-Tonbary Y, Fouda A: Infantile hepatic hemangioendothelioma: an 8-month old infant successfully treated with a corticosteroid. Hematol Oncol Stem Cell Ther; 2009;2(3):422-5
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  • [Title] Infantile hepatic hemangioendothelioma: an 8-month old infant successfully treated with a corticosteroid.
  • Infantile hemangioendothelioma is a rare benign vascular tumor of the liver.
  • We report a case of hepatic hemangioendothelioma in an 8- month-old female infant who presented with hepatomegaly and respiratory distress, which was successfully treated with oral prednisolone for six months.
  • [MeSH-major] Anti-Inflammatory Agents / therapeutic use. Hemangioendothelioma / drug therapy. Liver Neoplasms / drug therapy. Prednisolone / therapeutic use
  • [MeSH-minor] Female. Humans. Infant. Treatment Outcome

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  • (PMID = 20139057.001).
  • [ISSN] 1658-3876
  • [Journal-full-title] Hematology/oncology and stem cell therapy
  • [ISO-abbreviation] Hematol Oncol Stem Cell Ther
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Saudi Arabia
  • [Chemical-registry-number] 0 / Anti-Inflammatory Agents; 9PHQ9Y1OLM / Prednisolone
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2. Mehrabi A, Kashfi A, Fonouni H, Schemmer P, Schmied BM, Hallscheidt P, Schirmacher P, Weitz J, Friess H, Buchler MW, Schmidt J: Primary malignant hepatic epithelioid hemangioendothelioma: a comprehensive review of the literature with emphasis on the surgical therapy. Cancer; 2006 Nov 1;107(9):2108-21
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  • [Title] Primary malignant hepatic epithelioid hemangioendothelioma: a comprehensive review of the literature with emphasis on the surgical therapy.
  • Malignant hepatic epithelioid hemangioendothelioma (HEH) is a rare malignant tumor of vascular origin with unknown etiology and a variable natural course.
  • The authors present a comprehensive review of the literature on HEH with a focus on clinical outcome after different therapeutic strategies.
  • The reviewed parameters included demographic data, clinical manifestations, therapeutic modalities, and clinical outcome.
  • Most patients presented with multifocal tumor that involved both lobes of the liver.
  • Lung, peritoneum, lymph nodes, and bone were the most common sites of extrahepatic involvement at the time of diagnosis.
  • The most common management has been liver transplantation (LTx) (44.8% of patients), followed by no treatment (24.8% of patients), chemotherapy or radiotherapy (21% of patients), and liver resection (LRx) (9.4% of patients).
  • The 1-year and 5-year patient survival rates were 96% and 54.5%, respectively, after LTx; 39.3% and 4.5%, respectively, after no treatment, 73.3% and 30%, respectively, after chemotherapy or radiotherapy; and 100% and 75%, respectively, after LRx.
  • LRx has been the treatment of choice in patients with resectable HEH.
  • However, LTx has been proposed as the treatment of choice because of the hepatic multicentricity of HEH.
  • The role of different adjuvant therapies for patients with HEH remains to be determined.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / diagnosis. Hemangioendothelioma, Epithelioid / surgery. Liver Neoplasms / diagnosis. Liver Neoplasms / surgery
  • [MeSH-minor] Adult. Combined Modality Therapy. Diagnosis, Differential. Female. Hepatectomy. Humans. Liver Transplantation. Male. Neoplasm Metastasis. Survival Rate

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  • [Copyright] (c) 2006 American Cancer Society.
  • (PMID = 17019735.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 101
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3. Raphael C, Hudson E, Williams L, Lester JF, Savage PM: Successful treatment of metastatic hepatic epithelioid hemangioendothelioma with thalidomide: a case report. J Med Case Rep; 2010;4:413
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  • [Title] Successful treatment of metastatic hepatic epithelioid hemangioendothelioma with thalidomide: a case report.
  • INTRODUCTION: Hepatic epithelioid hemangioendothelioma is a rare malignancy arising from the vascular endothelial cells within the liver.
  • Historically, the disease is characterized as being poorly responsive to both chemotherapy and radiotherapy, with liver resection or transplantation the treatment of choice when feasible.
  • For patients with advanced disease, reports of long-term therapeutic benefits from conventional cytotoxic treatments are very limited.
  • Owing to the rarity of this malignancy, there is no structured therapeutic research, but a small number of cases have been reported to respond well to treatment with inhibitors of angiogenesis.
  • Thalidomide was originally developed as an anti-emetic but is a potent inhibitor of vascular neogenesis, and could offer potential in the treatment of hepatic epithelioid hemangioendothelioma by blocking the proliferation of the malignant vascular endothelial cells.
  • CASE PRESENTATION: We describe the case of a Caucasian British woman who presented at the age of 53 years with a hepatic mass, malignant lymphadenopathy and pulmonary metastases, which were confirmed as hepatic epithelioid hemangioendothelioma on biopsy.
  • After unproductive treatment with interferon, our patient was started on thalidomide 400 mg daily.
  • She has been successfully managed on this therapy for the past seven years, and has remained asymptomatic, with radiologically stable disease and minimal treatment-related side effects.
  • CONCLUSION: At present, there is no standard therapy for advanced hepatic epithelioid hemangioendothelioma.
  • Our case supports the role for thalidomide and potentially other inhibitors of vascular neogenesis in the treatment of patients with metastatic hepatic epithelioid hemangioendothelioma.

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  • [Cites] Oncology. 2004;67(5-6):471-5 [15714004.001]
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  • (PMID = 21176188.001).
  • [ISSN] 1752-1947
  • [Journal-full-title] Journal of medical case reports
  • [ISO-abbreviation] J Med Case Rep
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Other-IDs] NLM/ PMC3022673
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4. Emamaullee JA, Edgar R, Toso C, Thiesen A, Bain V, Bigam D, Kneteman N, Shapiro AM: Vascular endothelial growth factor expression in hepatic epithelioid hemangioendothelioma: Implications for treatment and surgical management. Liver Transpl; 2010 Feb;16(2):191-7
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  • [Title] Vascular endothelial growth factor expression in hepatic epithelioid hemangioendothelioma: Implications for treatment and surgical management.
  • Epithelioid hemangioendothelioma (EHE) is a low-grade, malignant vascular tumor that most commonly presents within the liver.
  • Patients with hepatic EHE are often candidates for liver transplantation as the disease is usually multifocal at diagnosis.
  • Although these patients achieve excellent early outcomes post-transplant, there are very few data regarding tumor markers that can further direct chemotherapy in hepatic EHE to prevent recurrent disease.
  • The purpose of this study was to analyze the expression of the angiogenic factor vascular endothelial growth factor (VEGF) and its receptors in hepatic EHE.
  • Six patients with hepatic EHE were assessed for liver transplantation at our center.
  • Pathology specimens of primary and recurrent EHE were analyzed by hematoxylin and eosin staining and by immunofluorescence for VEGF, fetal liver kinase 1 (Flk-1), and fms-related tyrosine kinase 1 (Flt-1) expression.
  • Five patients underwent liver transplantation, and 1 patient underwent liver resection.
  • In 1 patient with recurrent hepatic EHE post-liver transplantation, a progressive increase in the VEGF fluorescence intensity and distribution was observed.
  • In conclusion, in this series, VEGF expression was observed in all hepatic EHE specimens analyzed.
  • These data suggest that anti-VEGF chemotherapeutic agents will be of use in patients with hepatic EHE, particularly as a means of reducing the tumor volume prior to resection, as a means of treating unresectable or metastatic disease, or as an adjuvant therapy in the setting of liver transplantation.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / metabolism. Hemangioendothelioma, Epithelioid / surgery. Liver Neoplasms / metabolism. Liver Neoplasms / surgery. Liver Transplantation. Vascular Endothelial Growth Factor A / metabolism
  • [MeSH-minor] Adolescent. Adult. Angiogenesis Inhibitors / therapeutic use. Biomarkers, Tumor / metabolism. Biopsy. Disease-Free Survival. Female. Humans. Immunohistochemistry. Male. Middle Aged. Neoplasm Recurrence, Local / metabolism. Neoplasm Recurrence, Local / pathology. Neovascularization, Pathologic / drug therapy. Neovascularization, Pathologic / metabolism. Neovascularization, Pathologic / pathology. Retrospective Studies. Vascular Endothelial Growth Factor Receptor-1 / metabolism. Vascular Endothelial Growth Factor Receptor-2 / metabolism

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  • (PMID = 20104492.001).
  • [ISSN] 1527-6473
  • [Journal-full-title] Liver transplantation : official publication of the American Association for the Study of Liver Diseases and the International Liver Transplantation Society
  • [ISO-abbreviation] Liver Transpl.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Angiogenesis Inhibitors; 0 / Biomarkers, Tumor; 0 / VEGFA protein, human; 0 / Vascular Endothelial Growth Factor A; EC 2.7.10.1 / FLT1 protein, human; EC 2.7.10.1 / Vascular Endothelial Growth Factor Receptor-1; EC 2.7.10.1 / Vascular Endothelial Growth Factor Receptor-2
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5. Chen CC, Kong MS, Yang CP, Hung IJ: Hepatic hemangioendothelioma in children: analysis of thirteen cases. Acta Paediatr Taiwan; 2003 Jan-Feb;44(1):8-13
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  • [Title] Hepatic hemangioendothelioma in children: analysis of thirteen cases.
  • Hepatic hemangioendothelioma (HE) is a tumor that presents in infancy and toddler.
  • The diagnosis was made by the histology specimen or various imaging studies.
  • Abdominal ultrasonography (n = 13) showed heterogeneous and hypoechoic lesions in the liver.
  • Computed tomography (n = 11) revealed central hypointensity with peripheral enhancement after contrast of the liver masses.
  • Magnetic resonance imaging studies of the hepatic masses (n = 3) showed decreased signal intensity on T1 images and high signal intensity on T2.
  • Other management included interferon, chemotherapy, embolization and/or surgery.
  • Among the other nine patients, four patients died of sepsis, hepatic failure, disseminated intravascular coagulopathy or tumor rupture with hemorrhagic shock.
  • For its management, steroid is a first-line medication.
  • Other methods of treatment were interferon, hepatic artery embolization, chemotherapy and surgery.
  • Long term follow up is needed for the evaluation of treatment response.
  • [MeSH-major] Hemangioendothelioma / diagnosis. Liver Neoplasms / diagnosis
  • [MeSH-minor] Child, Preschool. Female. Humans. Infant. Infant, Newborn. Magnetic Resonance Imaging. Male. Tomography, X-Ray Computed

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  • (PMID = 12800377.001).
  • [ISSN] 1608-8115
  • [Journal-full-title] Acta paediatrica Taiwanica = Taiwan er ke yi xue hui za zhi
  • [ISO-abbreviation] Acta Paediatr Taiwan
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] China (Republic : 1949- )
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6. Biecker E, Fischer HP, Strunk H, Sauerbruch T: Benign hepatic tumours. Z Gastroenterol; 2003 Feb;41(2):191-200
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  • [Title] Benign hepatic tumours.
  • Benign hepatic tumours include a broad spectrum of regenerative and true neoplastic processes.
  • Due to advances in imaging procedures like MRI, Cf-scan and ultrasound as well as progress in immunohistochemistry, the appropriate diagnosis is made ina high percentage of patients without laparotomy and resection.
  • Most important in clinical practice is the differential diagnosis of focal nodular hyperplasia and hepatocellular adenoma because of the risk of rupture and bleeding in the latter.
  • Cavernous haemangioma, the most common benign hepatic tumour, rarely needs treatment.
  • The diagnosis of nodular regenerative hyperplasia is often missed and patients present with secondary complications and signs of portal hypertension that necessitate treatment.
  • The main problem in angiomyolipoma is to distinguish it from malignant processes which do require treatment.
  • Because of its clinical presentation, inflammatory pseudotumour is also sometimes confused with a malignant tumour.
  • Therapeutic options are drug therapy or surgical resection.
  • [MeSH-major] Liver Diseases / diagnosis. Liver Neoplasms / diagnosis
  • [MeSH-minor] Adenoma, Liver Cell / diagnosis. Adenoma, Liver Cell / pathology. Adult. Aged. Diagnostic Imaging. Focal Nodular Hyperplasia / diagnosis. Focal Nodular Hyperplasia / pathology. Granuloma, Plasma Cell / diagnosis. Granuloma, Plasma Cell / pathology. Hemangioendothelioma / diagnosis. Hemangioendothelioma / pathology. Hemangioma, Cavernous / diagnosis. Hemangioma, Cavernous / pathology. Humans. Infant. Liver / pathology. Liver Regeneration / physiology. Middle Aged

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  • (PMID = 12650132.001).
  • [ISSN] 0044-2771
  • [Journal-full-title] Zeitschrift für Gastroenterologie
  • [ISO-abbreviation] Z Gastroenterol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 138
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7. Uchimura K, Nakamuta M, Osoegawa M, Takeaki S, Nishi H, Iwamoto H, Enjoji M, Nawata H: Hepatic epithelioid hemangioendothelioma. J Clin Gastroenterol; 2001 May-Jun;32(5):431-4
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  • [Title] Hepatic epithelioid hemangioendothelioma.
  • Epithelioid hemangioendothelioma (EHE) is a rare neoplasm of vascular origin with unpredictable malignant potential.
  • We describe two patients with primary EHE of the liver and review 34 cases previously published in Japan and compare them with those in Western countries.
  • The clinical aspects of EHE and relevant treatment results in Japanese patients were similar to those found in Western countries, the exception being the matter of difference in incidence between genders.
  • Although this tumor type has been reported to be more frequent among women, there was no significant difference in incidence between men and women.
  • Hepatic EHE generally behaves as a low-grade malignant tumor with a slow progression phenotype; however, this disease seems to be resistant to chemotherapy and to be lethal in some cases.
  • Surgical resection or liver transplantation is recommended after rapid diagnosis by radiologic examination and histologic findings, including positive staining of tumor cells for factor VIII-related antigen.
  • [MeSH-major] Hemangioendothelioma, Epithelioid. Liver Neoplasms
  • [MeSH-minor] Adult. Fatal Outcome. Humans. Male. Middle Aged. Tomography, X-Ray Computed

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  • (PMID = 11319317.001).
  • [ISSN] 0192-0790
  • [Journal-full-title] Journal of clinical gastroenterology
  • [ISO-abbreviation] J. Clin. Gastroenterol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 7
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8. Sevinir B, Ozkan TB: Infantile hepatic hemangioendothelioma: clinical presentation and treatment. Turk J Gastroenterol; 2007 Sep;18(3):182-7
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  • [Title] Infantile hepatic hemangioendothelioma: clinical presentation and treatment.
  • BACKGROUND/AIMS: Hepatic hemangioendotheliomas are rare tumors in childhood.
  • METHODS: This retrospective analysis included eight patients with infantile hepatic hemangioendothelioma.
  • RESULTS: The median age at diagnosis was 24 days (age range: 1 to 70 days) and the female/male ratio was 5/3.
  • Four cases had single hepatic tumors while the others had multiple.
  • These lesions were located equally in the right and left hepatic lobes, and three babies had bilobar involvement.
  • Most of the multifocal hepatic tumors were associated with skin hemangiomas.
  • Treatment options were assessed individually.
  • Systemic prednisolone therapy (2 mg/kg/d) was commenced in six patients.
  • One boy with Kasabach-Merritt syndrome did not respond to this therapy.
  • Interferon-alpha (1 million units (MU)/m2/day) was started, and the daily dose of the drug was increased up to 10 MU/m2, administered 3 times per week, until clinical improvement was achieved.
  • CONCLUSIONS: The treatment approaches depend on the center's experience.
  • A multidisciplinary approach is required for the best treatment option.
  • [MeSH-major] Hemangioendothelioma / diagnosis. Hemangioendothelioma / therapy. Liver Neoplasms / diagnosis. Liver Neoplasms / therapy
  • [MeSH-minor] Abdomen. Alanine Transaminase / metabolism. Aspartate Aminotransferases / metabolism. Dilatation, Pathologic / etiology. Female. Glucocorticoids / therapeutic use. Hemangioma / diagnosis. Hepatomegaly / etiology. Humans. Immunologic Factors / therapeutic use. Infant. Infant, Newborn. Interferon-alpha / therapeutic use. Male. Neoplasms, Multiple Primary. Prednisolone / therapeutic use. Respiratory Distress Syndrome, Newborn / etiology. Retrospective Studies. Skin Neoplasms / diagnosis. Turkey / epidemiology

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  • (PMID = 17891692.001).
  • [ISSN] 2148-5607
  • [Journal-full-title] The Turkish journal of gastroenterology : the official journal of Turkish Society of Gastroenterology
  • [ISO-abbreviation] Turk J Gastroenterol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Turkey
  • [Chemical-registry-number] 0 / Glucocorticoids; 0 / Immunologic Factors; 0 / Interferon-alpha; 9PHQ9Y1OLM / Prednisolone; EC 2.6.1.1 / Aspartate Aminotransferases; EC 2.6.1.2 / Alanine Transaminase
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9. Ashraf S, Ashraf HM, Mamoon N, Luqman M: Epithelioid hemangioendothelioma of the liver. J Coll Physicians Surg Pak; 2007 May;17(5):280-2
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  • [Title] Epithelioid hemangioendothelioma of the liver.
  • Epithelioid hemangioendothelioma is an intermediate grade malignant neoplasm of vascular origin.
  • The tumor involves the liver and lungs, but other organs are affected too.
  • The key to the diagnosis is identification of cells of endothelial origin containing Factor VIII R antigen.
  • Surgical resection of isolated lesions is the treatment of choice; with unpredictable results reported for chemotherapy, radiotherapy, and resection of multiple lesions.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / diagnosis. Hemangioendothelioma, Epithelioid / surgery. Liver Neoplasms / diagnosis. Liver Neoplasms / surgery

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  • (PMID = 17553327.001).
  • [ISSN] 1022-386X
  • [Journal-full-title] Journal of the College of Physicians and Surgeons--Pakistan : JCPSP
  • [ISO-abbreviation] J Coll Physicians Surg Pak
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Pakistan
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10. Celikel C, Yumuk PF, Basaran G, Yildizeli B, Kodalli N, Ahiskali R: Epithelioid hemangioendothelioma with multiple organ involvement. APMIS; 2007 Jul;115(7):881-8
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  • [Title] Epithelioid hemangioendothelioma with multiple organ involvement.
  • Epithelioid hemangioendothelioma is a rare vascular neoplasm of uncertain malignant potential.
  • Various reports document metastatic or concurrent epithelioid hemangioendothelioma in several sites, most commonly with combined lung and liver involvement.
  • The concurrent involvement of multiple sites at presentation may cause diagnostic problems because epithelioid hemangioendothelioma can mimic other neoplastic processes.
  • Although it is a chemo-resistant disease, chemotherapy is usually advised for patients with metastatic or concurrent involvement.
  • Here we document the presentation, treatment, and outcome of two cases with concurrent involvement of the lung and liver.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / radiography. Liver Neoplasms / radiography. Lung Neoplasms / radiography
  • [MeSH-minor] Female. Humans. Male. Middle Aged. Treatment Outcome

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  • (PMID = 17614859.001).
  • [ISSN] 0903-4641
  • [Journal-full-title] APMIS : acta pathologica, microbiologica, et immunologica Scandinavica
  • [ISO-abbreviation] APMIS
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Denmark
  • [Number-of-references] 30
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11. Kalpatthi R, Germak J, Mizelle K, Yeager N: Thyroid abnormalities in infantile hepatic hemangioendothelioma. Pediatr Blood Cancer; 2007 Dec;49(7):1021-4
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  • [Title] Thyroid abnormalities in infantile hepatic hemangioendothelioma.
  • We report an infant with hepatic hemangioendothelioma (HAE) associated with compensated hypothyroidism.
  • The hepatic lesions regressed with steroid therapy and his thyroid function normalized with high doses of thyroxine supplement.
  • [MeSH-major] Hemangioendothelioma / complications. Hypothyroidism / complications. Liver Neoplasms / complications. Thyroxine / therapeutic use
  • [MeSH-minor] Child, Preschool. Dose-Response Relationship, Drug. Drug Administration Schedule. Follow-Up Studies. Heart Failure / complications. Heart Failure / drug therapy. Humans. Male. Steroids / therapeutic use. Thyroid Function Tests. Treatment Outcome

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  • [Copyright] 2007 Wiley-Liss, Inc
  • (PMID = 16544297.001).
  • [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 / Steroids; Q51BO43MG4 / Thyroxine
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12. Kanizaj TF, Cvrlje VC, Mrzljak A, Kardum-Skelin I, Sustercić D, Segro D, Gustin D, Kocman B: Epitheloid hemangioendothelioma in patient with liver transplantation. Coll Antropol; 2010 Mar;34(1):177-80
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  • [Title] Epitheloid hemangioendothelioma in patient with liver transplantation.
  • Malignant hepatic epithelioid hemangioendothelioma (HEH) is a rare malignant tumor of vascular origin with unknown aetiology and a variable natural course.
  • At the time of diagnosis, most patients present with multifocal tumours lesions that involve both liver lobes.
  • From the therapeutic aspect, liver resection (LRx), liver transplantation (LTx), chemotherapy, radiotherapy, and/or immunotherapy have been used in the treatment of patients with HEH.
  • However, because of the rarity of this tumor and its unpredictable natural history, it is impossible to assess the effectiveness of these respective therapies.
  • In this report, our objective was to present clinical aspects, diagnostic options, therapeutic modalities, and the clinical outcome of single patient with LTx because of this rare tumor.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / pathology. Hemangioendothelioma, Epithelioid / surgery. Liver Neoplasms / pathology. Liver Neoplasms / surgery. Liver Transplantation

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  • (PMID = 20432748.001).
  • [ISSN] 0350-6134
  • [Journal-full-title] Collegium antropologicum
  • [ISO-abbreviation] Coll Antropol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Croatia
  • [Chemical-registry-number] 0 / Antigens, CD31; 0 / Antigens, CD34; 0 / May-Grunwald Giemsa; T42P99266K / Methylene Blue; TDQ283MPCW / Eosine Yellowish-(YS)
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13. Szavay PO, Wermes C, Fuchs J, Schrappe M, Flemming P, von Schweinitz D: Effective treatment of infantile choriocarcinoma in the liver with chemotherapy and surgical resection: a case report. J Pediatr Surg; 2000 Jul;35(7):1134-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Effective treatment of infantile choriocarcinoma in the liver with chemotherapy and surgical resection: a case report.
  • Infantile choriocarcinoma of the liver is an extremely rare entity, and outcome has been fatal in almost all published cases.
  • To the authors' knowledge, this is the first report on successful treatment with preoperative chemotherapy.
  • A 10-week-old girl presented with a large liver tumor, ovarian cysts, cardiac insufficiency, progressive hemolytic anemia, and thrombocytopenia.
  • Ultrasound scan and magnetic resonance tomography (MRT) showed the typical pattern of infantile hemangioendothelioma.
  • An emergency laparotomy was performed because of increasing cardiac insufficiency with ligation of the right hepatic artery, tumor biopsy, and subtotal resection of the ovarian cysts.
  • Histology findings showed a choriocarcinoma of the liver and corpus luteum cysts of the ovaries.
  • Chemotherapy was initiated with etoposide and cisplatin.
  • When x-ray examination showed development of lung metastases, chemotherapy was intensified with etoposide, cisplatin, and ifosfamid according to the German Study Group of Extracranial Nontesticular Malignant Germ Cell Tumors in Childhood and Adolescence (MAKEI-96).
  • After the fourth course, a complete tumor resection was achieved by an extended right hemihepatectomy with adjuvant chemotherapy being administered after the operation.
  • The authors' experience shows that chemotherapy is effective for preoperative tumor reduction.
  • [MeSH-major] Choriocarcinoma / drug therapy. Choriocarcinoma / surgery. Liver Neoplasms / drug therapy. Liver Neoplasms / surgery
  • [MeSH-minor] Combined Modality Therapy. Female. Humans. Infant, Newborn. Remission Induction

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  • (PMID = 10917316.001).
  • [ISSN] 0022-3468
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] UNITED STATES
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14. Schilling G, Schuch G, Panse JP, Sterneck M, Bokemeyer C: Activity of lenalidomide in metastatic hepatic epithelioid hemangioendothelioma (HEH): A case report. J Clin Oncol; 2009 May 20;27(15_suppl):e21527

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Activity of lenalidomide in metastatic hepatic epithelioid hemangioendothelioma (HEH): A case report.
  • : e21527 Background: HEH is a rare tumor of the liver with an unpredictable malignant potential.
  • Surgical resection or liver transplantation is recommended in locally advanced disease and has been successfully performed in selected cases with extrahepatic manifestations.
  • Systemic therapy, however, is not standardized.
  • Small cohorts have been treated with interferon, chemotherapy and angiogenesis inhibitors such as thalidomide with various results.
  • CASE REPORT: A 33-year old caucasian previously healthy male was admitted to hospital with newly diagnosed suspicious lesions in liver and spleen.
  • Disease progressed during first line treatment with adriamycin and ifosfamide (2 cycles given).
  • Retrospective analysis after 9 cycles demonstrated stable disease in comparison to the recent investigation, but an overall progression of 22% according to RECIST criteria in the liver was observed.
  • Due to the excellent tolerance we increased the daily dose to 30 mg (21/28) and 6 months later a slight regression in the lung and overall stable disease in the liver was observed.
  • The patient was listed for liver transplantation and after another 4 months on lenalidomide 30 mg, he was successfully transplanted recently.
  • Because of the remaining extrahepatic lesions it is planned to continue lenalidomide therapy in combination with the immunosuppressant rapamycine.
  • CONCLUSIONS: We report the first case of successful therapy with lenalidomide in HEH.

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  • (PMID = 27963457.001).
  • [ISSN] 1527-7755
  • [Journal-full-title] Journal of clinical oncology : official journal of the American Society of Clinical Oncology
  • [ISO-abbreviation] J. Clin. Oncol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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15. Mascarenhas RC, Sanghvi AN, Friedlander L, Geyer SJ, Beasley HS, Van Thiel DH: Thalidomide inhibits the growth and progression of hepatic epithelioid hemangioendothelioma. Oncology; 2004;67(5-6):471-5
Hazardous Substances Data Bank. THALIDOMIDE .

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Thalidomide inhibits the growth and progression of hepatic epithelioid hemangioendothelioma.
  • Hepatic epithelioid hemangioendothelioma (HEH) is a rare vascular tumor of the liver with an unpredictable malignant potential.
  • Its growth can lead to hepatic failure, extrahepatic metastasis and death.
  • Surgical resection or liver transplantation is the treatment of choice if metastasis is not identified.
  • We report the case of a 52-year-old patient with HEH metastatic to the lungs who was successfully treated with oral thalidomide therapy.
  • [MeSH-major] Angiogenesis Inhibitors / therapeutic use. Antineoplastic Agents / therapeutic use. Hemangioendothelioma, Epithelioid / drug therapy. Hemangioendothelioma, Epithelioid / pathology. Liver Neoplasms / drug therapy. Liver Neoplasms / pathology. Thalidomide / therapeutic use
  • [MeSH-minor] Disease Progression. Drug Administration Schedule. Humans. Male. Middle Aged. Tomography, X-Ray Computed. Treatment Outcome

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  • [Copyright] Copyright (c) 2004 S. Karger AG, Basel
  • (PMID = 15714004.001).
  • [ISSN] 0030-2414
  • [Journal-full-title] Oncology
  • [ISO-abbreviation] Oncology
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Angiogenesis Inhibitors; 0 / Antineoplastic Agents; 4Z8R6ORS6L / Thalidomide
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16. Mehrabi A, Kashfi A, Schemmer P, Sauer P, Encke J, Fonouni H, Friess H, Weitz J, Schmidt J, Büchler MW, Kraus TW: Surgical treatment of primary hepatic epithelioid hemangioendothelioma. Transplantation; 2005 Sep 27;80(1 Suppl):S109-12
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Surgical treatment of primary hepatic epithelioid hemangioendothelioma.
  • Epithelioid hemangioendothelioma is a very rare tumor of vascular origin.
  • It can develop in different tissues such as soft tissue, lung, or liver.
  • Hepatic epithelioid hemangioendothelioma (HEH) mostly affects females.
  • The malignant potential of HEH often remains unclear in the individual patient.
  • It can range from benign hemangioma to malignant hemangioendotheliosarcoma.
  • All patients in our series with confirmed histological HEH did not show extrahepatic extension and consequently underwent surgical treatment.
  • In three patients, liver transplantation (LTx) was performed (two cadaveric and one living related).
  • No adjuvant chemotherapy was applied.
  • Early detection and surgical intervention in case of HEH can potentially offer curative treatment.
  • The treatment of first choice appears to be radical liver resection.
  • Despite the long waiting time, its often unclear dignity, and a proven progressive growth pattern, living related LTx also plays a potentially important role.
  • Long-term survival of patients with HEH is significantly higher compared to other hepatic malignancies.
  • The role of adjuvant therapy currently remains unclear.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / surgery. Liver Neoplasms / surgery
  • [MeSH-minor] Adult. Female. Humans. Middle Aged. Retrospective Studies. Survivors. Treatment Outcome

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  • (PMID = 16286886.001).
  • [ISSN] 0041-1337
  • [Journal-full-title] Transplantation
  • [ISO-abbreviation] Transplantation
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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17. Mucha K, Foroncewicz B, Zieniewicz K, Nyckowski P, Krawczyk M, Cyganek A, Paczek L: Patient with liver epithelioid hemangioendothelioma treated by transplantation: 3 years' observation. Transplant Proc; 2006 Jan-Feb;38(1):231-3
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  • [Title] Patient with liver epithelioid hemangioendothelioma treated by transplantation: 3 years' observation.
  • Epithelioid hemangioendothelioma (EHE) is a rare neoplasm of vascular origin, but unknown etiology that occurs in the liver, lungs and other organs.
  • Its hepatic form (HEHE) generally behaves as a low-grade malignant tumor with a slowly progressive phenotype.
  • Surgical resection or liver transplantation (OLT) has been recommended after diagnosis.
  • We present a 30-year-old woman with primary HEHE of the liver treated by OLT in 2002.
  • Her medical history started 3 years prior when an abdominal ultrasound examination revealed multiple focal changes in the liver.
  • The histopathological diagnosis from a needle biopsy was carcinoma cholangiogenes desmoplasticum.
  • For 2 years the patient was treated with chemotherapy combinations.
  • To explain the lack of efficacy of chemotherapy, a laparoscopic biopsy was performed and HEHE diagnosed.
  • After 3 years observation the patient presented with good liver function and no signs of tumor recurrence.
  • We concluded that immunohistochemical staining for characteristic endothelial cell markers may facilitate the correct diagnosis of HEHE.
  • After diagnosis, OLT followed by immunosuppressive therapy, consisting of basiliximab, corticoids, low doses of tacrolimus and temporary administration of rapamycin, may be safe and effective.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / surgery. Liver Neoplasms / surgery. Liver Transplantation / methods
  • [MeSH-minor] Adult. Female. Follow-Up Studies. Humans. Treatment Outcome


18. Kassam A, Mandel K: Metastatic hepatic epithelioid hemangioendothelioma in a teenage girl. J Pediatr Hematol Oncol; 2008 Jul;30(7):550-2
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  • [Title] Metastatic hepatic epithelioid hemangioendothelioma in a teenage girl.
  • SUMMARY: Hepatic epithelioid hemangioendothelioma (HEHE) is a rare malignant tumor characterized by its epithelioid structure and vascular endothelium origin.
  • The clinical course of HEHE is variable, ranging from long-term survival without treatment to a rapidly progressive course with a fatal outcome.
  • As a consequence, no standard treatment has been determined.
  • We present a case of HEHE occurring in a 13-year-old girl, in which a novel treatment approach using antiangiogenic therapy was tried and was successful in slowing the progression of the disease.
  • [MeSH-major] Hemangioendothelioma, Epithelioid / secondary. Liver Neoplasms / drug therapy. Lung Neoplasms / secondary
  • [MeSH-minor] Adolescent. Angiogenesis Inhibitors / administration & dosage. Angiogenesis Inhibitors / therapeutic use. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Carboplatin / administration & dosage. Celecoxib. Cisplatin / administration & dosage. Disease Progression. Doxorubicin / administration & dosage. Drug Therapy, Combination. Female. Humans. Interferon-alpha / therapeutic use. Liver Failure / etiology. Liver Failure / surgery. Liver Transplantation / ethics. Neovascularization, Pathologic / drug therapy. Neovascularization, Pathologic / etiology. Pyrazoles / administration & dosage. Pyrazoles / therapeutic use. Recombinant Proteins. Sulfonamides / administration & dosage. Sulfonamides / therapeutic use. Thalidomide / administration & dosage. Thalidomide / therapeutic use. Vinblastine / administration & dosage. Vinblastine / therapeutic use

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  • (PMID = 18797205.001).
  • [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 / Angiogenesis Inhibitors; 0 / Interferon-alpha; 0 / Pyrazoles; 0 / Recombinant Proteins; 0 / Sulfonamides; 4Z8R6ORS6L / Thalidomide; 5V9KLZ54CY / Vinblastine; 76543-88-9 / interferon alfa-2a; 80168379AG / Doxorubicin; BG3F62OND5 / Carboplatin; JCX84Q7J1L / Celecoxib; Q20Q21Q62J / Cisplatin
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19. Calabrò L, Di Giacomo AM, Altomonte M, Fonsatti E, Mazzei MA, Volterrani L, Miracco C, Maio M: Primary hepatic epithelioid hemangioendothelioma progressively responsive to interferon-alpha: is there room for novel anti-angiogenetic treatments? J Exp Clin Cancer Res; 2007 Mar;26(1):145-50
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Primary hepatic epithelioid hemangioendothelioma progressively responsive to interferon-alpha: is there room for novel anti-angiogenetic treatments?
  • Primary hepatic epithelioid hemangioendothelioma (HEH) is a rare, low-grade malignant neoplasm of endothelial origin, with an unpredictable clinical course and prognosis.
  • No standard therapeutic strategies are still available for HEH, due to the infrequency of the disease and to its variable natural history that limit the identification of the most effective treatment.
  • In the absence of metastatic disease, surgical resection or liver transplantation represent the treatment of choice for HEH, while several antineoplastic agents have been proposed in the presence of metastatic nonresectable disesase.
  • Herein, we describe the biological characterization and the clinical course of a primary HEH progressively responsive to treatment with intermediate doses of interferon-alpha (IFN)-alpha2a.
  • [MeSH-major] Angiogenesis Inhibitors / therapeutic use. Hemangioendothelioma, Epithelioid / drug therapy. Interferon-alpha / therapeutic use. Liver Neoplasms / drug therapy. Neovascularization, Pathologic / prevention & control
  • [MeSH-minor] Antigens, CD / analysis. Antigens, CD31 / analysis. Antigens, CD34 / analysis. Female. Humans. Immunohistochemistry. Middle Aged. Receptors, Cell Surface / analysis. Recombinant Proteins. Tomography, X-Ray Computed. Treatment Outcome

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  • (PMID = 17550144.001).
  • [ISSN] 0392-9078
  • [Journal-full-title] Journal of experimental & clinical cancer research : CR
  • [ISO-abbreviation] J. Exp. Clin. Cancer Res.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Italy
  • [Chemical-registry-number] 0 / Angiogenesis Inhibitors; 0 / Antigens, CD; 0 / Antigens, CD31; 0 / Antigens, CD34; 0 / ENG protein, human; 0 / Interferon-alpha; 0 / Receptors, Cell Surface; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a
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20. Nazir Z, Pervez S: Malignant vascular tumors of liver in neonates. J Pediatr Surg; 2006 Jan;41(1):e49-51
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  • [Title] Malignant vascular tumors of liver in neonates.
  • Malignant vascular tumors of the liver are rare in children, especially in neonates.
  • One was initially diagnosed as infantile hemangioendothelioma.
  • Rapid growth and appearance of pulmonary metastasis while on aggressive medical treatment suggested malignant transformation.
  • The other neonate presented with intractable ascites and liver biopsy showed histological features of angiosarcoma.
  • To date, surgery, chemotherapy, and radiotherapy have not improved the outcome of malignant vascular tumors of liver.
  • [MeSH-major] Hemangioendothelioma / pathology. Hemangiosarcoma / pathology. Liver Neoplasms / pathology
  • [MeSH-minor] Ascites. Diagnosis, Differential. Fatal Outcome. Female. Humans. Infant, Newborn

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  • (PMID = 16410090.001).
  • [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
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21. Haberal M, Ozcay F, Sevmis S, Karakayali H, Moray G, Torgay A, Coskun M, Demirhan B, Sarialioglu F, Arslan G: Liver transplant in an infant with bilateral cystic neuroblastoma complicated by hepatic metastases and life-threatening consumption coagulopathy. Pediatr Transplant; 2008 May;12(3):358-62
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  • [Title] Liver transplant in an infant with bilateral cystic neuroblastoma complicated by hepatic metastases and life-threatening consumption coagulopathy.
  • Here we report a patient with stage-4 bilateral cystic adrenal neuroblastomas with disseminated liver metastases and consumption coagulopathy who underwent liver transplant.
  • Our patient was initially diagnosed with infantile hepatic hemangioendothelioma and bleeding into the adrenal glands secondary to consumption coagulopathy (Kasabach-Merritt syndrome).
  • Liver transplant was performed as a life-saving procedure under this diagnosis.
  • We discuss this unique patient because of the diagnostic pitfalls of this rare disease and the successful clinical outcome after LT and subsequent chemotherapy for neuroblastoma.
  • [MeSH-major] Adrenal Gland Neoplasms / pathology. Disseminated Intravascular Coagulation / therapy. Liver Neoplasms / pathology. Liver Transplantation / methods. Neuroblastoma / pathology
  • [MeSH-minor] Adrenal Glands / pathology. Antineoplastic Agents / therapeutic use. Female. Humans. Infant. Living Donors. Neoplasm Metastasis. Treatment Outcome


22. Otte JB, de Ville de Goyet J: The contribution of transplantation to the treatment of liver tumors in children. Semin Pediatr Surg; 2005 Nov;14(4):233-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] The contribution of transplantation to the treatment of liver tumors in children.
  • Major progress has been achieved during the last decades in the treatment of malignant liver tumors in children, both in chemotherapy and surgical management.
  • Chemosensitivity varies between tumor types, and radical resection remains essential to effect a cure.
  • In tumors extensively involving a normal liver, in a diffuse or multifocal manner, radical resection cannot be accomplished with a partial hepatectomy.
  • This has been the case for some instances of advanced hepatoblastoma and epithelioid hemangioendothelioma.
  • In hepatoblastoma, current experience shows that results of primary liver transplantation with neoadjuvant chemotherapy are excellent with around an 80% 5-to-10-year disease-free survival rate.
  • Epithelioid hemangioendothelioma is very rarely seen in children and may have a more malignant behavior than in adult patients, and liver transplantation may not be the best management option.
  • In nonresectable hepatocellular carcinoma (HCC) developed on an otherwise normal liver, the results of liver transplantation are similarly poor to those obtained in adult patients, except in a few highly selected series fulfilling the Milano criteria.
  • Incidental HCC associated with chronic liver disease does not seem to impact posttransplant survival.
  • [MeSH-major] Liver Neoplasms / surgery. Liver Transplantation
  • [MeSH-minor] Carcinoma, Hepatocellular / surgery. Child. Hemangioendothelioma, Epithelioid / surgery. Hepatoblastoma / surgery. Humans

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  • (PMID = 16226698.001).
  • [ISSN] 1055-8586
  • [Journal-full-title] Seminars in pediatric surgery
  • [ISO-abbreviation] Semin. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 38
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23. Ekinci S, Karnak I, Tanyel FC, Senocak ME, Kutluk T, Büyükpamukçu M, Büyükpamukçu N: Hepatic lobectomies in children: experience of a center in the light of changing management of malignant liver tumors. Pediatr Surg Int; 2006 Mar;22(3):228-32
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Hepatic lobectomies in children: experience of a center in the light of changing management of malignant liver tumors.
  • Hepatic resection is the main treatment modality for hepatic tumors in childhood.
  • The aim of this study is to report our experience in hepatic lobectomy, which is relatively rare procedure in childhood.
  • Medical records of 25 patients who underwent hepatic lobectomy between January 1977 and June 2002 were reviewed retrospectively.
  • Age, gender, diagnosis, physical examination findings, results of preoperative laboratory investigations, radiological examination, resectability criteria, preoperative biopsies, chemotherapies, radiotherapies, postoperative pathological results, incisions, operation technique, intraoperative transfusions, drains used, antibiotic prophylaxes, and intraoperative and postoperative complications were evaluated for all patients.
  • Out of 25 patients with hepatic tumor seven patients with hepatoblastoma and four patients with hepatocellular carcinoma were given 5.7 +/- 0.3 cycles of chemotherapy before the operation.
  • Pathological examination of resected tumors revealed hepatoblastoma (n=11), mesenchymal hamartoma (n = 5), hepatocellular carcinoma (n = 4), hemangioendothelioma (n=1), malignant mesenchymal tumor (n = 1), hemangioma (n = 1), cyst adenoma (n = 1), and metastasis of cellular mesoblastic nephroma (n = 1).
  • Hepatic lobectomy is a major operation, which is feasible yielding curative results in children.
  • Safe hepatic resections with acceptable blood loss can be performed by a technique relying on good anatomic dissection and surgical control.
  • [MeSH-major] Hepatectomy / methods. Liver Neoplasms / surgery
  • [MeSH-minor] Child, Preschool. Diagnosis, Differential. Female. Follow-Up Studies. Humans. Infant. Magnetic Resonance Imaging. Male. Retrospective Studies. Tomography, X-Ray Computed. Treatment Outcome

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  • [ErratumIn] Pediatr Surg Int. 2006 Aug;22(8):695
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  • (PMID = 16395609.001).
  • [ISSN] 0179-0358
  • [Journal-full-title] Pediatric surgery international
  • [ISO-abbreviation] Pediatr. Surg. Int.
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
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24. Le Luyer B, Duquenoy A, Poinsot J, Boulloche J, Gaussin G, Le Roux P: [Use of interferon in a case of hepatic hemangioma]. Arch Pediatr; 2000 Nov;7(11):1201-4
MedlinePlus Health Information. consumer health - Liver Cancer.

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  • [Title] [Use of interferon in a case of hepatic hemangioma].
  • Hepatic hemangioendothelioma can have a fatal outcome.
  • After failure of classic therapies, and when surgery or arterial embolization are not possible, those benign tumours may benefit from interferon alpha treatment.
  • CASE REPORT: We report a case of a 14-month-old infant who presented with hepatic hemangiomatosis and cardiac failure.
  • The disease can neither be controlled by steroids nor by radiotherapy associated with digitalo-diuretic treatment.
  • CONCLUSION: Interferon alpha treatment helps to control cardiac failure and the course of hepatic hemangioma in childhood.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Hemangioendothelioma / drug therapy. Interferon-alpha / therapeutic use. Liver Neoplasms / drug therapy
  • [MeSH-minor] Female. Humans. Infant. Treatment Outcome

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  • (PMID = 11109948.001).
  • [ISSN] 0929-693X
  • [Journal-full-title] Archives de pédiatrie : organe officiel de la Sociéte française de pédiatrie
  • [ISO-abbreviation] Arch Pediatr
  • [Language] fre
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] FRANCE
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Interferon-alpha
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25. Emre S, McKenna GJ: Liver tumors in children. Pediatr Transplant; 2004 Dec;8(6):632-8
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  • [Title] Liver tumors in children.
  • Primary hepatic neoplasms in children are relatively infrequent, accounting for between 0.5 and 2.0% of all pediatric neoplasms.
  • They are clinically relevant tumors however as two thirds of them are malignant.
  • The therapy of these tumors has evolved over time and it currently involves a combination of surgery, adjuvant and neoadjuvant chemotherapy and possible transplantation.
  • [MeSH-major] Liver Neoplasms / diagnosis. Liver Neoplasms / therapy
  • [MeSH-minor] Carcinoma, Hepatocellular / diagnosis. Carcinoma, Hepatocellular / therapy. Child. Hamartoma / diagnosis. Hamartoma / therapy. Hemangioendothelioma / diagnosis. Hemangioendothelioma / therapy. Hemangiosarcoma / diagnosis. Hepatoblastoma / pathology. Hepatoblastoma / therapy. Humans. Neoplasm Staging. Sarcoma / diagnosis. Sarcoma / therapy

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  • (PMID = 15598339.001).
  • [ISSN] 1397-3142
  • [Journal-full-title] Pediatric transplantation
  • [ISO-abbreviation] Pediatr Transplant
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Denmark
  • [Number-of-references] 66
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26. Walsh R, Harrington J, Beneck D, Ozkaynak MF: Congenital infantile hepatic hemangioendothelioma type II treated with orthotopic liver transplantation. J Pediatr Hematol Oncol; 2004 Feb;26(2):121-3
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  • [Title] Congenital infantile hepatic hemangioendothelioma type II treated with orthotopic liver transplantation.
  • The authors describe a 10-week-old girl with infantile hepatic hemangioendothelioma who initially presented with difficulty feeding, hepatomegaly, and multiple hemangiomas of the skin.
  • Six weeks of steroid therapy and 2 weeks of chemotherapy failed to produce clinical improvement.
  • The patient underwent split liver transplantation.
  • A definitive diagnosis of hemangioendothelioma type II was made.
  • Imaging studies cannot differentiate between hemangioendothelioma and angiosarcoma.
  • Treatment modalities for this condition remain unclear.
  • [MeSH-major] Hemangioendothelioma / congenital. Hemangioendothelioma / surgery. Liver Neoplasms / congenital. Liver Neoplasms / surgery. Liver Transplantation
  • [MeSH-minor] Female. Humans. Infant. Tomography, X-Ray Computed. Treatment Outcome

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  • (PMID = 14767204.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Grant] United States / PHS HHS / / 1012HP000222
  • [Publication-type] Case Reports; Journal Article; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] United States
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27. Almogy G, Lieberman S, Gips M, Pappo O, Edden Y, Jurim O, Simon Slasky B, Uzieli B, Eid A: Clinical outcomes of surgical resections for primary liver sarcoma in adults: results from a single centre. Eur J Surg Oncol; 2004 May;30(4):421-7
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  • [Title] Clinical outcomes of surgical resections for primary liver sarcoma in adults: results from a single centre.
  • BACKGROUND: Primary hepatic sarcoma is a rare tumour with a poor prognosis.
  • METHODS: From 1997 to 2002 eight patients had liver resection for primary sarcoma of the liver at our institution.
  • The clinical characteristics, imaging findings, surgical procedures, adjuvant therapy and outcome were retrospectively reviewed.
  • There were two patients each with angiosarcoma (AS), leiomyosarcoma (LMS), and undifferentiated embryonal sarcoma (UES), one patient with epithelioid hemangioendothelioma (EHE) and one patient with malignant peripheral nerve sheath sarcoma (PNSS).
  • Preoperative diagnosis of a primary liver sarcoma was made in 7/8 cases, either by fine needle aspiration (n = 5) or angiography (n = 2).
  • Two patients developed complications and there was one death.
  • Systemic chemotherapy led to tumour regression in both patients with UES which enabled a second hepatic resection.
  • CONCLUSIONS: The majority of patients with primary liver sarcoma present with right upper quadrant pain, fever and a liver mass.
  • Differentiating the rare primary liver sarcoma from the much more common hepatocellular carcinoma (HCC) may aid in planning therapy.
  • Liver resection combined with adjuvant chemotherapy are the mainstays of treatment for UES in the adult.
  • [MeSH-major] Liver Neoplasms / surgery. Sarcoma / surgery
  • [MeSH-minor] Adult. Aged. Chemotherapy, Adjuvant. Diagnosis, Differential. Female. Hepatectomy / methods. Humans. Length of Stay. Liver Function Tests. Male. Middle Aged. Retrospective Studies. Tomography, X-Ray Computed. Treatment Outcome

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  • (PMID = 15063896.001).
  • [ISSN] 0748-7983
  • [Journal-full-title] European journal of surgical oncology : the journal of the European Society of Surgical Oncology and the British Association of Surgical Oncology
  • [ISO-abbreviation] Eur J Surg Oncol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
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28. Grabhorn E, Richter A, Fischer L, Krebs-Schmitt D, Ganschow R: Neonates with severe infantile hepatic hemangioendothelioma: limitations of liver transplantation. Pediatr Transplant; 2009 Aug;13(5):560-4
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Neonates with severe infantile hepatic hemangioendothelioma: limitations of liver transplantation.
  • IHHE as the most common vascular tumor of the liver in infancy can present with acute postnatal liver and congestive heart failure.
  • Here we discuss the benefit of LTx in cases of acute postnatal deterioration and massive extent of the hepatic tumor.
  • Two were neonates with acute postnatal decompensation and progressive liver and heart failure within days.
  • Treatment with steroids and chemotherapy was ineffective; resection surgery and interventional treatment were not considered appropriate.
  • Patients 1 and 2 had to be resuscitated during the LTx procedure because of massive bleeding and both died during the procedure.
  • Neonates with extended hepatic and extrahepatic involvement of IHHE should be evaluated carefully prior to LTx.
  • Whenever possible, alternative interventional treatment options should be considered.
  • [MeSH-major] Hemangioendothelioma / therapy. Liver Neoplasms / therapy. Liver Transplantation / methods
  • [MeSH-minor] Antineoplastic Agents / pharmacology. Female. Humans. Infant, Newborn. Male. Steroids / therapeutic use. Time Factors. Treatment Outcome


29. Albert A, Cruz O, Montaner A, Vela A, Badosa J, Castañón M, Morales L: [Congenital solid tumors. A thirteen-year review]. Cir Pediatr; 2004 Jul;17(3):133-6
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  • This is an interesting group of tumors because their type, relative incidence, natural history and response to treatment differ from those seen in older children.
  • There were 8 teratomas (3 sacrocoxigeal, 1 retroperitoneal, 1 in the CNS, 1 orbitary and two oronasal), two hepatic tumors (1 hepatoblastoma, 1 hemangioendothelioma, two CNS tumors, two giant nevus (one on a hamartoma), and one each Wilms tumor, infantile fibrosarcoma and myofibroblastic tumor.
  • Treatment was surgical resection alone in 17 cases (68%) and surgery + chemotherapy in 8 (32%) (5 neuroblastomas, one CNS tumor, one Wilms tumor and one presacral teratoma who developed a yolk sac tumor); 3 patients died (11%): one at surgery, one of tumoural airway obstruction at birth and one with craniopharyngioma.
  • Among the 14 tumors that were initially not malignant, two can be locally agressive, one was an immature teratoma, the giant nevus with hamartoma developed in situ melanoma, the other nevus had meningeal melanosis with hydrocephalus, and one mature presacral teratoma developed a yolk sac tumor.
  • CONCLUSIONS: Diagnosis of congenital tumors is performed earlier in recent years due to the wide use of prenatal ultrasound.
  • Complete surgical excision is the treatment of choice, most cases not need adjuvant chemotherapy.
  • We ought to pass this message on to our colleagues in prenatal diagnosis, so parents get reliable information.
  • [MeSH-major] Central Nervous System Neoplasms / congenital. Kidney Neoplasms / congenital. Liver Neoplasms / congenital. Neuroblastoma / congenital. Skin Neoplasms / congenital. Soft Tissue Neoplasms / congenital. Teratoma / congenital. Wilms Tumor / congenital
  • [MeSH-minor] Female. Follow-Up Studies. Humans. Infant, Newborn. Male. Neoplasm Recurrence, Local. Postoperative Complications. Pregnancy. Prenatal Diagnosis. Time Factors

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  • (PMID = 15503950.001).
  • [ISSN] 0214-1221
  • [Journal-full-title] Cirugía pediátrica : organo oficial de la Sociedad Española de Cirugía Pediátrica
  • [ISO-abbreviation] Cir Pediatr
  • [Language] spa
  • [Publication-type] Comparative Study; English Abstract; Journal Article
  • [Publication-country] Spain
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30. Nord KM, Kandel J, Lefkowitch JH, Lobritto SJ, Morel KD, North PE, Garzon MC: Multiple cutaneous infantile hemangiomas associated with hepatic angiosarcoma: case report and review of the literature. Pediatrics; 2006 Sep;118(3):e907-13
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  • [Title] Multiple cutaneous infantile hemangiomas associated with hepatic angiosarcoma: case report and review of the literature.
  • Multiple cutaneous hemangiomas can be associated with internal hemangiomas, with the liver being the most common site.
  • Here we report a case of a premature female neonate who presented with cardiac failure at birth and had typical-appearing infantile hemangiomas on the skin in association with vascular lesions in the liver.
  • Her clinical presentation was felt to be consistent with cutaneous and hepatic infantile hemangiomas.
  • After failure to respond to systemic steroids and chemotherapy, she underwent liver transplantation.
  • Histopathologic evaluation of the liver revealed a diagnosis of type 2 infantile hepatic hemangioendothelioma (regarded as synonymous with angiosarcoma) rather than benign infantile hemangioma of the liver.
  • We report this case and a review of the literature on pediatric angiosarcoma of the liver associated with cutaneous infantile hemangiomas.
  • [MeSH-major] Hemangioma / pathology. Hemangiosarcoma / pathology. Liver Neoplasms / pathology. Skin Neoplasms / pathology
  • [MeSH-minor] Female. Heart Defects, Congenital. Humans. Infant, Newborn. Liver Transplantation

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  • [ErratumIn] Pediatrics. 2007 Jun;119(6):1271
  • (PMID = 16880251.001).
  • [ISSN] 1098-4275
  • [Journal-full-title] Pediatrics
  • [ISO-abbreviation] Pediatrics
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 27
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