Massive hemorrhage: A rare complication of rapidly involuting congenital hemangioma

Malki, Aysha Al; Bluwi, Shatha Al; Malloizel‐Delaunay, Julie; Mazereeuw‐Hautier, J.

doi:10.1111/pde.13474

Cited by 10 publications

(8 citation statements)

References 3 publications

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“…10,11 Our case also highlights the dearth of evidence for treatment options in CH. Despite the benign behavior of the vast majority of CH cases, 2 several reports of catastrophic CH resulting in hemorrhage, 7,16 cardiac failure, 17,18 and 5 prior reports of death are found (Table 2). 16,[19][20][21][22] In the presence of cardiac failure, the mortality rate in such cases has been reported as high as 30%.…”

Section: Discussionmentioning

confidence: 99%

Catastrophic congenital hemangioma with severe coagulopathy leading to fatal cardiac failure: Case report and review

Cohen‐Cutler

Szymanski

Bockoven

et al. 2021

Pediatric Dermatology

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Congenital hemangiomas (CH) are rare vascular tumors of the neonate, with an estimated incidence of less than 0.5%. 1 CH are present at birth and may involute to varying degrees over the first 2-12 months of age. 2 Based on the course and degree of involution, CH are subdivided into rapidly, partially, and non-involuting subtypes. [3][4][5] Complications of CH are rare and primarily related to high-flow vasculature that may initially be present before the involution phase begins. The most common sequela of this includes ulceration and bleeding, particularly in the peripartum period. 6,7 High-output congestive heart failure can rarely result from intralesional shunting in larger CH, particularly those within visceral organs. Increased blood flow can also lead to a mild coagulopathy, evidenced by hypofibrinogenemia and thrombocytopenia. 2,[8][9][10][11] These findings are usually transient and less severe than Kasabach-Merritt phenomenon (KMP), which occurs most commonly in patients with kaposiform hemangioendotheliomas (KHE) and less so with tufted angiomas (TA). 12 Typically, CH can be managed supportively and expectantly.However, in cases requiring intervention, a lack of evidence in favor of medical therapies was found. 2 Corticosteroids, propranolol, and vincristine have been used with minimal success, and only embolization and surgical resection have been shown to be definitive. 9,13,14 We present a rare, and sadly lethal, case of a neonate with a large CH and severe, overwhelming coagulopathy and multiorgan failure.Treatment options, which were carefully weighed for our patient, are

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Section: Discussionmentioning

confidence: 99%

Catastrophic congenital hemangioma with severe coagulopathy leading to fatal cardiac failure: Case report and review

Cohen‐Cutler

Szymanski

Bockoven

et al. 2021

Pediatric Dermatology

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“…RICH involutes in the irst 6-14 months of life [1]. These tumors can rarely cause complications, such as ulceration and bleeding [3]. As well, RICH associated with transient thrombocytopenia and coagulation abnormalities have been reported during the irst week of life [5].…”

Section: Discussionmentioning

confidence: 99%

“…It has been classi ied into three subtypes: rapidly involuting congenital hemangioma (RICH), non-involuting congenital hemangioma (NICH) and partially involuting congenital hemangioma (PICH) [2]. CHs rarely present complications, such as ulceration and bleeding [3]. Kasabach-Merritt syndrome (KMS) is a severe complication of some vascular tumors, which associates profound thrombocytopenia and coagulopathy that can be lifethreatening [4].…”

Section: Introductionmentioning

confidence: 99%

Rapidly involuting congenital hemangioma associated with Kasabach-Merritt Syndrome

Ballona¹,

Zevallos²,

Núñez³

2021

J Adv Pediatr Child Health

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Background: Rapidly involuting congenital hemangioma (RICH) is a rare vascular tumor that is present at birth and involutes during the first year of life. Kasabach-Merritt syndrome (KMS) is a complication of some vascular tumors such as kaposiform hemangioendothelioma and tufted angioma associated with thrombocytopenia and coagulopathy. Results: The case of a 2-month-old infant with a diagnosis of RICH with thrombocytopenia and coagulation disorder, successfully treated with surgical excision without complications or recurrence is presented. Conclusion: The association between RICH and KMS is rare. Histopathological study, immunohistochemistry and ultrasound findings are important for the diagnosis. Brief summary: This report covers the rare association between rapidly involuting congenital hemangioma and Kasabach-Merritt syndrome in a 2-months-old female infant.

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“…To the best of our knowledge, there are only six other similar cases reported in the English literature. [2][3][4][5] Severe haemorrhages seem to complicate congenital haemangiomas more than infant haemangiomas. This can be explained by the presence of larger and more superficial vessels in congenital haemangiomas making small cutaneous ulceration more susceptible to cause serious bleeding 5 .…”

mentioning

confidence: 99%

“…[2][3][4][5] Severe haemorrhages seem to complicate congenital haemangiomas more than infant haemangiomas. This can be explained by the presence of larger and more superficial vessels in congenital haemangiomas making small cutaneous ulceration more susceptible to cause serious bleeding 5 . One other risk factor for severe haemorrhages is RICH subtype.…”

mentioning

confidence: 99%

Bleeding in congenital haemangioma: A rare but serious complication

et al. 2021

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Massive hemorrhage: A rare complication of rapidly involuting congenital hemangioma

Cited by 10 publications

References 3 publications

Catastrophic congenital hemangioma with severe coagulopathy leading to fatal cardiac failure: Case report and review

Catastrophic congenital hemangioma with severe coagulopathy leading to fatal cardiac failure: Case report and review

Rapidly involuting congenital hemangioma associated with Kasabach-Merritt Syndrome

Bleeding in congenital haemangioma: A rare but serious complication

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