Epithelioid hemangioendothelioma of the bone: A case report with findings of bone scintigraphy

Medicine (Baltimore). 2019 May;98(19):e15546. doi: 10.1097/MD.0000000000015546.

Abstract

Introduction: Epithelioid hemangioendothelioma (EHE) of the bone is a very rare malignant vascular neoplasm, with biologic behavior between that of locally aggressive epithelioid hemangioma and malignant epithelioid angiosarcoma. We report a case of a patient with EHE who underwent bone scintigraphy, try to identify the characteristics of it, to highlight the clinical importance of whole-body bone scintigraphy and single-photon emission computed tomography/computed tomography (SPECT/ CT) in the diagnosis and treatment of EHE.

Patient concerns: A 67-year-old man with no history of trauma who reported pain in both lower limbs for 6 months, which had been worsening over the last 20 days. Anteroposterior and lateral radiographs of both lower limbs revealed numerous osteolytic lesions in the tibia and fibula bilaterally. Tc-methylene diphosphonate (MDP) bone scintigraphy demonstrated increased tracer uptake in the pelvic and bilateral lower limb bones. SPECT/CT bone imaging showed numerous osteolytic lesions cluster in the same anatomic region, with high tracer uptake in lesion margins.

Diagnosis: EHE of the bone.

Interventions: Two months after the diagnosis was confirmed, the patient was rehospitalized. Nonopioid analgesic use had not provided pain relief. Magnetic resonance imaging (MRI) of both thighs showed the bone cortex was destroyed with numerous irregular lesions, and soft-tissue was involved. A second bone scintigraphy did not show any new lesions. He was administrated with recombination human endostatin injection 15 mg ivgtt qd for 14 days, combined with apatinib mesylate tablets 500 mg po qd for 18 days.

Outcomes: He was discharged voluntarily and died 2 months later.

Conclusion: EHE of the bone is a very rare malignant vascular neoplasm with no specific radiographic imaging features. Whole-body bone scintigraphy, especially SPECT/CT bone imaging, significantly reduces ambiguous diagnoses and is recommended before treatment.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Bone Neoplasms / diagnostic imaging*
  • Bone Neoplasms / drug therapy
  • Bone Neoplasms / pathology
  • Bone and Bones / diagnostic imaging
  • Bone and Bones / pathology
  • Diagnosis, Differential
  • Fatal Outcome
  • Hemangioendothelioma, Epithelioid / diagnostic imaging*
  • Hemangioendothelioma, Epithelioid / drug therapy
  • Hemangioendothelioma, Epithelioid / pathology
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Single Photon Emission Computed Tomography Computed Tomography
  • Whole Body Imaging