Hypercalcemia related to PTH-rP revealing malignant hepatic epithelioid hemangioendothelioma

Boukhris, I.; Azzabi, S.; Kéchaou, I.; Chérif, E.; Kooli, Chékib; Romdhane, Khaled Ben; Omar, Souheil; Khalfallah, N.

doi:10.1684/abc.2015.1118

Cited by 3 publications

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“…To our surprise, our case was strongly positive for CK20 staining, which had not been seen in previous case reports. We reviewed relevant reports on the expression of CK20 in epithelioid tumors and found that epithelioid malignant mesothelioma ( 51 ) and malignant hepatic epithelioid hemangioendothelioma ( 52 ) could abnormally express CK20. Perhaps our case could serve as the first report of an anomalous EAML expression of CK20.…”

Section: Discussionmentioning

confidence: 99%

Primary renal malignant epithelioid angiomyolipoma with distant metastasis: a case report and literature review

Zhang,

Wang,

Chen

et al. 2023

Front. Oncol.

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Epithelioid angiomyolipoma (EAML) is a rare type of mesenchymal angiomyolipoma with potential malignancy in the kidney that can cause lymph node metastases, local recurrence, and distant metastases. Herein, we describe a case of EAML in the right kidney of a 51-year-old man who was admitted to the hospital with a right abdominal mass. Computed tomography revealed a heterogeneously enhanced mass with blurred margins, which was considered a malignant tumor. A radical nephrectomy was then performed. Two years later, the patient developed liver metastases from EAML and was administered sintilimab combined with bevacizumab. The patient survived after 6 months of follow-up. Histologically, the tumors showed clear boundaries and no obvious capsules. The tumor tissue mainly consisted of epithelioid tumor cells, thick-walled blood vessels, and a small amount of adipose tissue. Tumor cells with lipid vacuoles and acinar areas were large, round, polygonal, eosinophilic, or transparent in the cytoplasm. The enlarged and hyperchromatic nuclei were accompanied by distinct nucleoli and pathological mitosis. These histopathological findings resembled those of renal cell carcinoma, and immunohistochemical analysis was performed. The tumor cells were diffusely positive for HMB45, Melan-A, CK20, vimentin antibodies, and TFE3, suggesting that the tumor originated from perivascular epithelioid cells, excluding renal cell carcinoma. The Ki-67 index was 10%. These histopathological features were observed in liver mass puncture tissues. We also summarized 46 cases of EAML with distant metastasis and explored the clinicopathological features of EAML to improve the treatment of the disease. EAML is often ignored in the clinical setting, leading to metastasis and recurrence. Therefore, EAMLs require long-term follow-up, and timely detection of recurrent disease can improve the prognosis.

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Section: Discussionmentioning

confidence: 99%

Primary renal malignant epithelioid angiomyolipoma with distant metastasis: a case report and literature review

Zhang,

Wang,

Chen

et al. 2023

Front. Oncol.

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“…7 Additional paraneoplastic syndromes have also been attributed to EHE. Boukhris et al 4 describe a case of a man with hypercalcemia related to tumor production of PTH-rp who was ultimately diagnosed with EHE. Additionally, El Mahou et al 5 describe a case of a patient with a 13-year history of Cushing’s syndrome of unknown etiology despite extensive workup and hypophysectomy.…”

Section: Discussionmentioning

confidence: 99%

“…2 Although not commonly associated with a paraneoplastic syndrome, paraneoplastic syndromes in the setting of EHE have been reported. [4][5][6][7] Acute disseminated encephalomyelitis (ADEM) is an acute, autoimmune, demyelinating disease of the central nervous system known to predominantly affect the white matter tracts of the brain and spinal cord. 8 The reported incidence is between 4 and 8 cases per one million people.…”

Section: Introductionmentioning

confidence: 99%

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Hepatic epithelioid hemangioendothelioma associated with acute disseminated encephalomyelitis by a possible paraneoplastic process

et al. 2020

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Epithelioid hemangioendothelioma (EHE) is a low-grade, malignant vascular neoplasm that frequently involves the liver, lungs, bone, and soft tissue. Although not commonly associated with a paraneoplastic syndrome, paraneoplastic syndromes in the setting of EHE have been reported. Acute disseminated encephalomyelitis (ADEM) is an acute, autoimmune, demyelinating disorder of the central nervous system that most commonly occurs after an infection or vaccination. We present the case of a 23 year old female who developed the acute onset of fevers, tremors, right sided hemiplegia, global aphasia, and incontinence of urine and stool. MRI demonstrated findings consistent with a demyelinating disorder and brain biopsy confirmed the diagnosis of ADEM. The patient’s work up revealed multiple liver lesions which were biopsy proven EHE. This case report discusses the diagnosis and treatment of two concurrent rare disease processes and the possible association of the processes via a paraneoplastic syndrome.

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Kasabach‑Merritt syndrome combined with hypercalcemia: A case report

et al. 2017

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Abstract. The present case report presented the diagnosis and treatment course of an infant diagnosed with Kasabach-Merritt syndrome (KMS) combined with hypercalcemia (HC). A 35-day-old infant with swelling on the upper right arm for >1 month and thrombocytopenia for 1 day was admitted to Hunan Provincial People's Hospital (Changsha, China) and a series of treatments, including γ-globulin impact, heparin anticoagulation, platelet transfusion, supplement of cryoprecipitate and fibrinogen following heparinization and inhabitation of vascular endothelial cell proliferation by propranolol, were performed. At 2 months after the initial admission to the hospital, surgery was conducted and the hemangioma was removed through pipeline arteriosclerosis embolization when the patient was hospitalized again with symptoms of vomiting and atrophy accompanied by HC. The level of blood calcium reduced to normal following surgery. Cases of KMS combined with HC are extremely rare and the most effective way to treat such cases is surgical resection of the hemangioma.

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Hypercalcemia related to PTH-rP revealing malignant hepatic epithelioid hemangioendothelioma

Cited by 3 publications

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Primary renal malignant epithelioid angiomyolipoma with distant metastasis: a case report and literature review

Primary renal malignant epithelioid angiomyolipoma with distant metastasis: a case report and literature review

Hepatic epithelioid hemangioendothelioma associated with acute disseminated encephalomyelitis by a possible paraneoplastic process

Kasabach‑Merritt syndrome combined with hypercalcemia: A case report

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