Successful Treatment With Cyclophosphamide of Life-threatening Diffuse Hemangiomatosis Involving the Liver

Hurvitz, Sara A.; Hurvitz, Carole H.; Sloninsky, Liliana; Sanford, Margaret C.

doi:10.1097/00043426-200011000-00012

Cited by 51 publications

(46 citation statements)

References 39 publications

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“…mTOR is regulated by phosphoinositide-3-kinase (PI3K) [67]. The mTOR inhibitor sirolimus has been shown to inhibit lymphangiogenesis, and it has been successfully used as a treatment option for vascular anomalies in children [28,[68][69][70].…”

Section: Role Of Mtor Inhibitorsmentioning

confidence: 99%

Clinical Review: Management of Adult Kasabach-Merritt Syndrome Associated with Hemangiomas

Master¹,

Kallam²,

Eo³

et al. 2017

J Blood Disord Transfus

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Kasabach-Merritt syndrome (KMS) consists of a clinical trial of capillary hemangioma, thrombocytopenia and disseminated intravascular coagulation. KMS occurs most commonly in the pediatric population, and its occurrence in adults is rare. Specific guidelines or randomized clinical trials guiding clinical management of KMS in adult patients are lacking. This manuscript provides a comprehensive review of KMS and discusses recent advances in the medical management of KMS. We also propose a systematic therapeutic approach which would serve as a guide in the management of adult patients with KMS caused by hemangiomas.

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Section: Role Of Mtor Inhibitorsmentioning

confidence: 99%

Clinical Review: Management of Adult Kasabach-Merritt Syndrome Associated with Hemangiomas

Master¹,

Kallam²,

Eo³

et al. 2017

J Blood Disord Transfus

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“…8 The mortality rate in patients with DNH is extremely high, up to 95%. 6,9 The main reason for death is congestive heart failure. 6,8,9 Severe hypothyroidism can be associated with massive hepatic hemangioma.…”

Section: Discussionmentioning

confidence: 99%

“…6,9 The main reason for death is congestive heart failure. 6,8,9 Severe hypothyroidism can be associated with massive hepatic hemangioma. 10 The first line of therapy is systemic corticosteroids.…”

Section: Discussionmentioning

confidence: 99%

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Diffuse Neonatal Hemangiomatosis Treatment With Cyclophosphamide

Vlahovic

Simić²,

Djokic³

et al. 2009

Journal of Pediatric Hematology/Oncology

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We present 3-month-old male infant with diffuse neonatal hemangiomatosis. There were 63 cutaneous hemangiomas over the scalp, face, trunk, and extremities. Computed tomography scan revealed the presence of hemangiomas in the liver and kidneys; laryngobronchoscopy identified the presence of hemangioma in tracheobronchial tree. The child had symptoms of heart failure therefore digitals and diuretics were administrated. Thyroid functions were normal. Treatment with corticosteroids, in dose of 3 mg/kg/d intravenously, was initiated. As there was no significant clinical improvement, cyclophosphamide was administrated. He received 4 courses, 10 days apart. Each course consisted of 10 mg/kg/d of cyclophosphamide and 10 mg/kg/d of mesna for 4 consecutive days. After 4 cycles of cyclophosphamide, the liver was notably decreased in size and the cardiac failure was resolved. Magnetic resonance imaging of the abdomen revealed the marked decrease in size of the liver hemangioma. After 3 years of follow-up the child is well developed, fully recovered, without cardiologic or respiratory problems.

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“…In addition, there are some reports of cyclophosphamide use in the pediatric hematology-oncology literature for the treatment of lifethreatening diffuse neonatal hemangiomatosis. 69 Laser therapy is another treatment modality for IH. The pulsed dye laser (PDL) has been used to treat IH, with varying results.…”

Section: Treatmentmentioning

confidence: 99%

Infantile hemangiomas: from pathogenesis to clinical features

Rosenblatt¹,

Mathes²,

Rosbe

2012

RRN

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Infantile hemangiomas (IH) are benign vascular tumors consisting of a collection of immature cells, including progenitor stem cells and disorganized blood vessels. They are the most common benign tumors in childhood. Recently, there have been significant, exciting advancements in the understanding of the pathogenesis and treatment of infantile hemangiomas, which are discussed in this review. The decision to initiate treatment for IH is based on many factors, including size and location, functional compromise, psychosocial implications, and risks and benefits of the proposed therapy. For most families of children with hemangiomas, education about the natural history of IH and reassurance are often the only "treatment" required. A minority of patients with large, complex lesions or lesions that cause functional compromise require early intervention. These patients and families benefit from a multidisciplinary approach to care in vascular birthmark centers. Ongoing multi-institutional clinical trials will provide further important data on the efficacy and safety of hemangioma treatments.

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Successful Treatment With Cyclophosphamide of Life-threatening Diffuse Hemangiomatosis Involving the Liver

Cited by 51 publications

References 39 publications

Clinical Review: Management of Adult Kasabach-Merritt Syndrome Associated with Hemangiomas

Clinical Review: Management of Adult Kasabach-Merritt Syndrome Associated with Hemangiomas

Diffuse Neonatal Hemangiomatosis Treatment With Cyclophosphamide

Infantile hemangiomas: from pathogenesis to clinical features

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