Multivertebral Kaposiform hemangioendothelioma presenting as scoliosis - A case report and review of literature

Shyam, Karthik; Thippeswamy, Pushpa Bhari; Kamashi, Jayanthi; Kanakeshwar, Raja Bhaskara; Rajasekaran, S.

doi:10.1016/j.jcot.2023.102147

Journal of Clinical Orthopaedics and Trauma

2023

DOI: 10.1016/j.jcot.2023.102147

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Multivertebral Kaposiform hemangioendothelioma presenting as scoliosis - A case report and review of literature

Karthik Shyam¹,

Pushpa Bhari Thippeswamy²,

Jayanthi Kamashi³

et al.

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Cited by 2 publications

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Kaposiform hemangioendothelioma presented with raynaud phenomenon: a case report

Liu,

Gu,

Teng

et al. 2023

BMC Pediatr

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Background Kaposiform hemangioendothelioma (KHE) is a rare vascular neoplasm affecting infants or young children. KHE includes a spectrum of lesions, ranging from small and superficial tumors to large and invasive lesions with Kasabach-Merritt phenomenon (KMP). Currently, no published studies have reported a KHE presenting as thrombocytopenia and Raynaud phenomenon. Case presentation A 2-year-old boy with right hand swelling and thrombocytopenia was admitted to our hospital. His right hand turned swelling and red, even occasionally cyanotic. This condition became worse in response to cool environments and improved with warming, and platelet counts were between 50 ~ 80 × 10^9/L. Physical examination on admission revealed the swelling and frostbite-like rash of the right-hand fingers, and the skin temperature of the right hand was lower than the left. On day 3 of admission, chest CT results showed an irregular mass on the right side of the spine. The puncture biopsy demonstrated positive CD31, D2-40, and FLI1 immunohistochemical staining, but negative GLUT1 staining, confirming the diagnosis of KHE. Furthermore, endothelin-1 (ET1) expression levels significantly increased, and eNOS and A20 expression levels significantly decreased comparing with control patients. The patient received methylprednisolone and sirolimus treatments, and his condition gradually improved during the follow-up. Conclusions We reported the first case of KHE presenting with thrombocytopenia and Raynaud phenomenon. The development of Raynaud phenomenon could be associated with increased ET-1 and reduced eNOS and A20 expressions. Careful differential diagnosis of hidden KHE should be considered in children with thrombocytopenia and Raynaud phenomenon.

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Kaposiform hemangioendothelioma presented with raynaud phenomenon: a case report

Liu,

Gu,

Teng

et al. 2023

BMC Pediatr

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Diagnosis and differentiation of Kaposiform hemangioendothelioma based on ultrasound radiomics

2024

Am J Cancer Res

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Multivertebral Kaposiform hemangioendothelioma presenting as scoliosis - A case report and review of literature

Cited by 2 publications

References 23 publications

Kaposiform hemangioendothelioma presented with raynaud phenomenon: a case report

Kaposiform hemangioendothelioma presented with raynaud phenomenon: a case report

Diagnosis and differentiation of Kaposiform hemangioendothelioma based on ultrasound radiomics

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