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Diagnosis of epithelioid hemangioendothelioma 8 days postpartum : is there a link with pregnancy? : A case report and review of the literature

Nicky Somers (UGent) , David Creytens (UGent) , Simon Van Belle (UGent) , Gwen Sys (UGent) and Lore Lapeire (UGent)
Author
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Abstract
Epithelioid hemangioendothelioma (EHE) is a rare and heterogeneous malignant vascular tumor. Decision making on a treatment strategy is difficult and a standard of care does not exist. EHE shows a wide age distribution but is rare in children. It can appear anywhere in the body, although lung and liver involvement are most common. There is a female predominance for visceral lesions and several case reports in which EHE developed during or after pregnancy are described in literature, hinting towards a putative role of sex hormones in the course of the disease. We present a case of a 32-year-old woman diagnosed with symptomatic pulmonary metastatic hepatic EHE (HEHE) 8 days postpartum, while the patient was completely asymptomatic before. A wait and see policy was chosen and the patient became asymptomatic in the months following the diagnosis. Although no expression of estrogen and progesterone receptors was found in the diagnostic liver biopsy specimen, we presume that the increased level of sex hormones during pregnancy may have triggered disease progression. The clinical behaviour of the disease in this case report reinforces the suspicion of female hormonal involvement in this type of malignancy and hints toward the potential role of other pregnancy-related factors, e.g. placental growth factor (PlGF), in the development of the disease.
Keywords
General Medicine, Epithelioid hemangioendothelioma, EHE, pregnancy, sex hormones, PlGF, CUTANEOUS ANGIOSARCOMAS, LIVER-TRANSPLANTATION, MONOCLONAL-ANTIBODY, GENE, HEMANGIOMA, EXPRESSION, FUSION, TUMORS

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MLA
Somers, Nicky, et al. “Diagnosis of Epithelioid Hemangioendothelioma 8 Days Postpartum : Is There a Link with Pregnancy? : A Case Report and Review of the Literature.” ACTA CLINICA BELGICA, 2020, pp. 1–6, doi:10.1080/17843286.2020.1802146.
APA
Somers, N., Creytens, D., Van Belle, S., Sys, G., & Lapeire, L. (2020). Diagnosis of epithelioid hemangioendothelioma 8 days postpartum : is there a link with pregnancy? : A case report and review of the literature. ACTA CLINICA BELGICA, 1–6. https://doi.org/10.1080/17843286.2020.1802146
Chicago author-date
Somers, Nicky, David Creytens, Simon Van Belle, Gwen Sys, and Lore Lapeire. 2020. “Diagnosis of Epithelioid Hemangioendothelioma 8 Days Postpartum : Is There a Link with Pregnancy? : A Case Report and Review of the Literature.” ACTA CLINICA BELGICA, 1–6. https://doi.org/10.1080/17843286.2020.1802146.
Chicago author-date (all authors)
Somers, Nicky, David Creytens, Simon Van Belle, Gwen Sys, and Lore Lapeire. 2020. “Diagnosis of Epithelioid Hemangioendothelioma 8 Days Postpartum : Is There a Link with Pregnancy? : A Case Report and Review of the Literature.” ACTA CLINICA BELGICA: 1–6. doi:10.1080/17843286.2020.1802146.
Vancouver
1.
Somers N, Creytens D, Van Belle S, Sys G, Lapeire L. Diagnosis of epithelioid hemangioendothelioma 8 days postpartum : is there a link with pregnancy? : A case report and review of the literature. ACTA CLINICA BELGICA. 2020;1–6.
IEEE
[1]
N. Somers, D. Creytens, S. Van Belle, G. Sys, and L. Lapeire, “Diagnosis of epithelioid hemangioendothelioma 8 days postpartum : is there a link with pregnancy? : A case report and review of the literature,” ACTA CLINICA BELGICA, pp. 1–6, 2020.
@article{8671155,
  abstract     = {Epithelioid hemangioendothelioma (EHE) is a rare and heterogeneous malignant vascular tumor. Decision making on a treatment strategy is difficult and a standard of care does not exist. EHE shows a wide age distribution but is rare in children. It can appear anywhere in the body, although lung and liver involvement are most common. There is a female predominance for visceral lesions and several case reports in which EHE developed during or after pregnancy are described in literature, hinting towards a putative role of sex hormones in the course of the disease. We present a case of a 32-year-old woman diagnosed with symptomatic pulmonary metastatic hepatic EHE (HEHE) 8 days postpartum, while the patient was completely asymptomatic before. A wait and see policy was chosen and the patient became asymptomatic in the months following the diagnosis. Although no expression of estrogen and progesterone receptors was found in the diagnostic liver biopsy specimen, we presume that the increased level of sex hormones during pregnancy may have triggered disease progression. The clinical behaviour of the disease in this case report reinforces the suspicion of female hormonal involvement in this type of malignancy and hints toward the potential role of other pregnancy-related factors, e.g. placental growth factor (PlGF), in the development of the disease.},
  author       = {Somers, Nicky and Creytens, David and Van Belle, Simon and Sys, Gwen and Lapeire, Lore},
  issn         = {1784-3286},
  journal      = {ACTA CLINICA BELGICA},
  keywords     = {General Medicine,Epithelioid hemangioendothelioma,EHE,pregnancy,sex hormones,PlGF,CUTANEOUS ANGIOSARCOMAS,LIVER-TRANSPLANTATION,MONOCLONAL-ANTIBODY,GENE,HEMANGIOMA,EXPRESSION,FUSION,TUMORS},
  language     = {eng},
  pages        = {1--6},
  title        = {Diagnosis of epithelioid hemangioendothelioma 8 days postpartum : is there a link with pregnancy? : A case report and review of the literature},
  url          = {http://dx.doi.org/10.1080/17843286.2020.1802146},
  year         = {2020},
}

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