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1. Agnese M, Cipolletta L, Bianco MA, Quitadamo P, Miele E, Staiano A: Blue rubber bleb nevus syndrome. Acta Paediatr; 2010 Apr;99(4):632-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Endoscopy is the gold standard technique for the diagnosis of BRBNS with GI lesions and also allows immediate therapeutic measures such as argon plasma coagulation, laser photocoagulation, sclerotherapy or band ligation.
  • In addition, pharmacological treatments based on corticosteroids, interferon alfa, vincristine or octreotide have been described for BRBNS.
  • CONCLUSION: Blue Rubber Bleb Nevus Syndrome is a congenital cutaneous and gastrointestinal haemangiomatosis.
  • The treatment is based on pharmacological or surgical therapy.


2. Al-Sebeih K, Manoukian J: Systemic steroids for the management of obstructive subglottic hemangioma. J Otolaryngol; 2000 Dec;29(6):361-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • We present 14 patients with subglottic hemangioma treated between 1984 and 1997,4 of whom had associated extralaryngeal hemangiomatous lesions (28%).
  • There were no major complications from the systemic steroid treatment.
  • One patient developed a cushingoid face that was reversed after the cessation of steroid therapy.
  • [MeSH-major] Airway Obstruction / drug therapy. Anti-Inflammatory Agents / therapeutic use. Dexamethasone / therapeutic use. Hemangioma / drug therapy. Laryngeal Neoplasms / drug therapy. Prednisolone / therapeutic use
  • [MeSH-minor] Child, Preschool. Drug Administration Schedule. Female. Humans. Infant. Male

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  • (PMID = 11770144.001).
  • [ISSN] 0381-6605
  • [Journal-full-title] The Journal of otolaryngology
  • [ISO-abbreviation] J Otolaryngol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Canada
  • [Chemical-registry-number] 0 / Anti-Inflammatory Agents; 7S5I7G3JQL / Dexamethasone; 9PHQ9Y1OLM / Prednisolone
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3. Takahashi A, Ogawa C, Kanazawa T, Watanabe H, Suzuki M, Suzuki N, Tsuchida Y, Morikawa A, Kuwano H: Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome. J Pediatr Surg; 2005 Mar;40(3):E47-50
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome.
  • The treatment of massive osteolysis with lymphangioma and/or hemangioma (Gorham-Stout syndrome) has been controversial.
  • The authors report on a patient with multiple massive osteolyses and extensive lymph-hemangiomatosis whose lesions were reduced by interferon alfa therapy.
  • There were also hemangiomas in the liver and spleen, a tumor lesion in the left lower chest wall, and hemangiomatous change on the skin surface of the left back.
  • After steroid pulse therapy, pericardial effusion disappeared.
  • During these treatments, the 7th to 10th ribs disappeared from the x-ray and scoliosis developed.
  • One month later, a cloudy fluid collection in the right lung was found on computed tomography.
  • Interferon alfa and steroid pulse therapy were started.
  • Later, the progress of osteolysis and the extension of lymph-hemangiomatosis stopped.
  • The interferon alfa therapy was stopped 14 months after it was administered.
  • At this time, computed tomography shows regression of the hemangiomatous lesion in the back.
  • The authors clinically diagnosed the patient as having Gorham-Stout syndrome with extension of lymph-hemangiomatosis.
  • Interferon alfa with or without steroid therapy should be a choice for patients with extension lesions.
  • [MeSH-major] Angiogenesis Inhibitors / therapeutic use. Interferon-alpha / therapeutic use. Osteolysis, Essential / drug therapy
  • [MeSH-minor] Adrenal Cortex Hormones / therapeutic use. Antineoplastic Agents / therapeutic use. Child, Preschool. Chylothorax / etiology. Drug Therapy, Combination. Female. Femur / pathology. Hemangioma / drug therapy. Humans. Liver Neoplasms / drug therapy. Lymphangioma / complications. Lymphangioma / drug therapy. Neoplasms, Multiple Primary / drug therapy. Picibanil / therapeutic use. Pleural Effusion / etiology. Pulmonary Atelectasis / etiology. Remission Induction. Ribs / pathology. Scoliosis / etiology. Skin Neoplasms / drug therapy. Spine / pathology. Splenic Neoplasms / drug therapy. Syndrome. Thoracic Neoplasms / drug therapy

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  • (PMID = 15793714.001).
  • [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 / Adrenal Cortex Hormones; 0 / Angiogenesis Inhibitors; 0 / Antineoplastic Agents; 0 / Interferon-alpha; 39325-01-4 / Picibanil
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4. Glade RS, Vinson K, Becton D, Bhutta S, Buckmiller LM: Management of complicated hemangiomas with vincristine/vinblastine: Quantitative response to therapy using MRI. Int J Pediatr Otorhinolaryngol; 2010 Nov;74(11):1221-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Management of complicated hemangiomas with vincristine/vinblastine: Quantitative response to therapy using MRI.
  • PURPOSE: To quantify the efficacy of vincristine and vinblastine in the treatment of complicated hemangiomas.
  • Only patients who received both a pre and post-treatment magnetic resonance imaging (MRI) were considered.
  • A database was created which includes patient gender, age at treatment initiation, rationale for treatment, hemangioma location, number of cycles of chemotherapy received, and complications of treatment.
  • A single pediatric radiologist calculated lesion volumes from both pre and post-treatment MRI which were compared to quantify treatment response.
  • Mean age at treatment initiation was 20 weeks (median 14, range 5-60).
  • Rationale for treatment included four patients (57%) with proptosis/orbital compromise and one patient each (14%) with heart failure, airway compression, and hemangiomatosis with rapid growth of multiple lesions.
  • Patients received a mean of 2.86 cycles of chemotherapy (median 3, range 1-5).
  • Twelve lesions were identified and analyzed for pre and post-treatment volume on MRI in the seven patients.
  • Eleven of twelve (92%) lesions decreased in size after treatment.
  • The mean volume ratio of hemangiomas at the conclusion of chemotherapy was 0.45 compared to pre-treatment size (median 0.18, range 0-2.19) Orbital compromise, airway compression, and cardiac failure either improved or resolved in all patients.
  • Three complications of treatment were seen in seven patients (42%) including bacteremia with anemia, peripheral neuropathy and motor delay.
  • All complications resolved after cessation of chemotherapy.
  • CONCLUSIONS: Treatment of complicated hemangiomas with vincristine or vinblastine can control growth and improve symptoms in the majority of patients.
  • Treatment often requires multiple cycles of chemotherapy.
  • Complications of treatment are common, but reversible.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / therapeutic use. Hemangioma / drug therapy. Hemangioma / pathology. Vincristine / therapeutic use
  • [MeSH-minor] Anemia / etiology. Bacteremia / etiology. Developmental Disabilities / etiology. Facial Neoplasms / drug therapy. Facial Neoplasms / pathology. Female. Humans. Infant. Liver Neoplasms / drug therapy. Liver Neoplasms / pathology. Magnetic Resonance Imaging. Male. Peripheral Nervous System Diseases / etiology. Retrospective Studies

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  • [Copyright] Published by Elsevier Ireland Ltd.
  • (PMID = 20884067.001).
  • [ISSN] 1872-8464
  • [Journal-full-title] International journal of pediatric otorhinolaryngology
  • [ISO-abbreviation] Int. J. Pediatr. Otorhinolaryngol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Ireland
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Phytogenic; 5J49Q6B70F / Vincristine
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5. Bonifazi E, Acquafredda A, Milano A, Montagna O, Laforgia N: Severe hypoglycemia during successful treatment of diffuse hemangiomatosis with propranolol. Pediatr Dermatol; 2010 Mar-Apr;27(2):195-6
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  • [Title] Severe hypoglycemia during successful treatment of diffuse hemangiomatosis with propranolol.
  • A 27-day-old male infant with diffuse hemangiomatosis of the skin and liver was treated with oral propranolol at a dosage of 2 mg/kg per day.
  • Although the risk appears small, increased vigilance for hypoglycemia in children on chronic propranolol treatment who have decreased caloric intake for any reason seems prudent.
  • [MeSH-major] Hemangioma / drug therapy. Hypoglycemia / chemically induced. Liver Neoplasms / drug therapy. Propranolol / adverse effects. Propranolol / therapeutic use
  • [MeSH-minor] Energy Intake. Humans. Infant, Newborn. Ketone Bodies / blood. Magnetic Resonance Imaging. Male. Skin Neoplasms / drug therapy. Treatment Outcome


6. Channick RN, Olschewski H, Seeger W, Staub T, Voswinckel R, Rubin LJ: Safety and efficacy of inhaled treprostinil as add-on therapy to bosentan in pulmonary arterial hypertension. J Am Coll Cardiol; 2006 Oct 3;48(7):1433-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Safety and efficacy of inhaled treprostinil as add-on therapy to bosentan in pulmonary arterial hypertension.
  • OBJECTIVES: This study evaluated the safety and efficacy of inhaled treprostinil as add-on therapy to oral bosentan in patients with pulmonary arterial hypertension (PAH).
  • BACKGROUND: The addition of a long-acting prostacyclin analogue via the inhaled route might be a safe and effective strategy to optimize therapy in PAH patients on bosentan.
  • METHODS: Twelve patients with symptomatic PAH despite bosentan received either 30 microg of inhaled treprostinil 4 times daily (n = 6) or 45 microg 4 times daily (n = 6), via an ultrasonic nebulizer.
  • RESULTS: One patient was excluded from analysis due to the subsequent finding of pulmonary capillary hemangiomatosis.
  • [MeSH-major] Antihypertensive Agents / administration & dosage. Epoprostenol / analogs & derivatives. Hypertension, Pulmonary / drug therapy. Sulfonamides / administration & dosage
  • [MeSH-minor] Administration, Inhalation. Administration, Oral. Adult. Aged. Blood Pressure / drug effects. Female. Humans. Lung / blood supply. Male. Middle Aged. Severity of Illness Index. Treatment Outcome. Vascular Resistance / drug effects


7. Ho V, Krol A, Bhargava R, Osiovich H: Diffuse neonatal haemangiomatosis. J Paediatr Child Health; 2000 Jun;36(3):286-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Diffuse neonatal haemangiomatosis.
  • A newborn girl with severe diffuse neonatal haemangiomatosis is described.
  • [MeSH-minor] Adrenal Cortex Hormones / administration & dosage. Central Nervous System Venous Angioma / diagnosis. Central Nervous System Venous Angioma / drug therapy. Drug Therapy, Combination. Fatal Outcome. Female. Humans. Infant, Newborn. Interferon-alpha / administration & dosage. Magnetic Resonance Imaging. Recombinant Proteins

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  • (PMID = 10849236.001).
  • [ISSN] 1034-4810
  • [Journal-full-title] Journal of paediatrics and child health
  • [ISO-abbreviation] J Paediatr Child Health
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] AUSTRALIA
  • [Chemical-registry-number] 0 / Adrenal Cortex Hormones; 0 / Interferon-alpha; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a
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8. Schiavetti A, De Pasquale MD, Di Salvo S, Ventriglia F, Clerico A: Recombinant interferon alfa 2a in hepatic hemangiomatosis with congestive heart failure: a case report. Pediatr Hematol Oncol; 2003 Mar;20(2):161-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Recombinant interferon alfa 2a in hepatic hemangiomatosis with congestive heart failure: a case report.
  • The therapy with subcutaneous interferon-alfa-2a (IFN-alpha) was administered for 12 months with progressive regression of cutaneous hemangiomas, liver lesions, and cardiological alterations.
  • IFN-alpha therapy was effective without any significant adverse effects.
  • [MeSH-major] Angiogenesis Inhibitors / therapeutic use. Heart Failure / etiology. Hemangioma / drug therapy. Interferon-alpha / therapeutic use. Liver Neoplasms / drug therapy
  • [MeSH-minor] Abdominal Neoplasms / congenital. Abdominal Neoplasms / drug therapy. Digitalis Glycosides / therapeutic use. Diuretics / therapeutic use. Dyspnea / etiology. Female. Furosemide / therapeutic use. Head and Neck Neoplasms / congenital. Head and Neck Neoplasms / drug therapy. Heart Septal Defects, Atrial / complications. Heel. Hemangioma, Capillary / congenital. Hemangioma, Capillary / drug therapy. Hemangioma, Cavernous / congenital. Hemangioma, Cavernous / drug therapy. Hepatomegaly / etiology. Humans. Infant. Knee. Neoplasms, Multiple Primary / congenital. Neoplasms, Multiple Primary / drug therapy. Recombinant Proteins. Remission Induction

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  • (PMID = 12554528.001).
  • [ISSN] 0888-0018
  • [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] 0 / Angiogenesis Inhibitors; 0 / Digitalis Glycosides; 0 / Diuretics; 0 / Interferon-alpha; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a; 7LXU5N7ZO5 / Furosemide
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10. Isaacs H Jr: Fetal and neonatal hepatic tumors. J Pediatr Surg; 2007 Nov;42(11):1797-803
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • The purpose of this review is to focus on the fetus and neonate in an attempt to determine the various ways liver tumors differ clinically and pathologically from those found in the older child and adult and to show that certain types of tumors have a better prognosis than others.
  • There were 76 (65%) examples of solitary (unifocal) hemangiomas and 41 (35%) of multifocal (which included the entity diffuse hemangiomatosis) with 86% and 71% survival rates, respectively.
  • There was some relationship between histologic type and prognosis.
  • Fifteen of 32 hepatoblastoma patients received surgical resection with or without chemotherapy, resulting in 7 (47%) of 15 cures.
  • Pre- or postoperative chemotherapy is reserved for those patients with unresectable tumors or metastatic disease.

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  • (PMID = 18022426.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 24
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11. Yu DC, Grabowski MJ, Kozakewich HP, Perez-Atayde AR, Voss SD, Shamberger RC, Weldon CB: Primary lung tumors in children and adolescents: a 90-year experience. J Pediatr Surg; 2010 Jun;45(6):1090-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • A wide range of histopathologic tumor types occurs.
  • This study aims to determine the incidence of different primary lung tumors in children and to contribute data leading to the development of evidence-based treatment models.
  • RESULTS: Forty patients were identified (23 boys, 17 girls) with a mean age of 9.6 years (range, 3 months to 19 years).
  • Fourteen distinct histopathologic tumor types were identified.
  • The most common tumor types were carcinoid (8), inflammatory myofibroblastic tumor (7), and pleuropulmonary blastoma (6).
  • Rare pediatric lung tumors including small cell carcinoma, adenocarcinoma, and pulmonary capillary hemangiomatosis were also seen.
  • Chemotherapy was used in 23% (9) and radiation in 20% (8) of the patients.
  • Of the 33 survivors, 28 had follow-up with a median duration of 29.5 months (mean, 63.2 months; range, 1-471 months).
  • The tumor spectrum involves many types not seen in adults, and unlike adults, patients rarely have a history of exposure to external predisposing factors.
  • Although complete resection remains the standard for treatment of most tumors, addition of adjuvant therapy is dependent on both tumor stage and histopathologic type.
  • [MeSH-minor] Adolescent. Age Distribution. Child. Child, Preschool. Diagnosis, Differential. Female. Follow-Up Studies. Humans. Infant. Male. Morbidity / trends. Prognosis. Retrospective Studies. Sex Distribution. Survival Rate / trends. Time Factors. United States / epidemiology. Young Adult

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  • [Copyright] Copyright 2010 Elsevier Inc. All rights reserved.
  • (PMID = 20620301.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article
  • [Publication-country] United States
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12. 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.
  • OBJECTIVES: To discuss the current knowledge regarding complicated hemangiomas (cervicofacial, periorbital, lumbosacral, and parotid), including the associated syndromes of diffuse neonatal hemangiomatosis and PHACES (posterior fossa malformations, most commonly of the Dandy-Walker variant; hemangiomas [especially large, plaquelike, facial lesions]; arterial anomalies; cardiac anomalies and coarctation of the aorta; eye abnormalities; and sternal cleft and/or supraumbilical raphe).
  • To discuss the risks and benefits of current treatment options, including the use of systemic corticosteroids and interferon in necessary situations.
  • 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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