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kaposiform hemangioendothelioma

Kaposiform hemangioendothelioma is a rare tumor of childhood often associated with Kasabach-Merritt phenomenon (50%) and occasionally lymphangiomatosis.

KH is a lesion having both a vascular and lymphatic component. Its common association with Kasabach-Merritt phenomenon probably relates in part to unique architectural features that favor turbulent blood flow and platelet activation.

Immunochemistry (Endothelial cells in nodules)

- CD31+
- CD34+
- D2-40 (15920541)
- FLI1+
- GLUT1 -
- LeY-

Synopsis

- sites of platelet consumption

  • scattered "epithelioid" or glomeruloid islands featuring endothelium
  • clusters of plump alpha-smooth muscle actin-positive pericytes
  • stippled hemosiderin
  • CD61+ fibrin thrombi

Variants

- kaposiform hemangioendothelioma without Kasabach-Merritt phenomenon (15793511)

  • KHE presenting in the absence of Kasabach-Merritt phenomenon is rare, although tufted angioma frequently occurs without thrombocytopenia.

Prognosis

- alive without residual disease (45%) (15105642)
- alive with disease (36%) (15105642)
- none distant metastases (O%) (15105642)
- regional perinodal soft tissue involvement (10%) (15105642)
- death from the disease (14%) (15105642)

Differential diagnosis

- infantile hemangioma (15105642)

  • GLUT1+
  • LeY-

- Kaposi sarcoma

  • absence of HHV-8

Associations

- Milroy disease (primary hereditary lymphedema)


Return to: vascular tumors and vascular dysplasias


References

- Debelenko LV, Perez-Atayde AR, Mulliken JB, Liang MG, Archibald TH, Kozakewich HP. D2-40 immunohistochemical analysis of pediatric vascular tumors reveals positivity in kaposiform hemangioendothelioma. Mod Pathol. 2005 Nov;18(11):1454-60. PMID: 15920541

- Gruman A, Liang MG, Mulliken JB, Fishman SJ, Burrows PE, Kozakewich HP, Blei F, Frieden IJ. Kaposiform hemangioendothelioma without Kasabach-Merritt phenomenon. J Am Acad Dermatol. 2005 Apr;52(4):616-22. PMID: 15793511

- Lyons LL, North PE, Mac-Moune Lai F, Stoler MH, Folpe AL, Weiss SW. Kaposiform Hemangioendothelioma: A Study of 33 Cases Emphasizing Its Pathologic, Immunophenotypic, and Biologic Uniqueness From Juvenile Hemangioma. Am J Surg Pathol. 2004 May;28(5):559-568. PMID: 15105642