J Korean Neurosurg Soc.  2012 Jul;52(1):48-51. 10.3340/jkns.2012.52.1.48.

Destructive Radiologic Development of Intravascular Papillary Endothelial Hyperplasia on Skull Bone

Affiliations
  • 1Department of Neurosurgery, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital and Medical School, Gwangju, Korea. jung-ty@chonnam.ac.kr
  • 2Department of Pediatrics, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital and Medical School, Gwangju, Korea.
  • 3Department of Radiology, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital and Medical School, Gwangju, Korea.

Abstract

Intravascular papillary endothelial hyperplasia (IPEH) is a rare vascular benign lesion that rarely involves the central nervous system with or without skull invasion. We report a rare case of IPEH on the skull bone, which displayed destructive radiologic development associated with hemorrhage. A 14-year-old male presented with an incidentally detected a small enhancing, left frontal osteolytic lesion. Previously, he underwent operation and received adjuvant chemoradiation therapy for cerebellar medulloblastoma. Follow-up magnetic resonance imaging revealed a left frontal bone lesion, which expanded to an approximately 2 cm-sized well-circumscribed osteolytic lesion associated with hemorrhage for 20 months. Frontal craniectomy and cranioplasty were performed. Destructive change was detected on the inner table and diploic space of the skull. The mass had a cystic feature with hemorrhagic content without dural attachment. Pathologic examination showed the capsule consisted of parallel collagen lamellae representing a vascular wall, vascular lumen, which was pathognomonic for IPEH. Immunohistochemical staining revealed that the capsule was positive for CD34 and factor VIII, which favor the final diagnosis of IPEH. This was the first case of intracalvarial IPEH.

Keyword

Destructive; Papillary endothelial hyperplasia; Radiologic; Skull

MeSH Terms

Adolescent
Central Nervous System
Collagen
Factor VIII
Follow-Up Studies
Frontal Bone
Hemorrhage
Humans
Hyperplasia
Magnetic Resonance Imaging
Male
Medulloblastoma
Skull
Collagen
Factor VIII

Figure

  • Fig. 1 Radiologic findings with time. A : Two years post-operatively, dilated or prominent vascular structures (arrow) were detected around the left frontal bone marrow. B : Six months later, follow-up MRI reveals a well defined enhanced nodular lesion. C : Another 7 months later, growth of the rim enhanced cystic mass was evident. D : On a gradient echo image, newly developed internal hemorrhage is observed in the cystic mass. E : Computed tomography reveals a well-circumscribed, expansile osteolytic mass located on the inner table and diploic space of the frontal bone.

  • Fig. 2 Operative findings. A : The of the skull shows destructive change of the inner table with diploic space. B : The mass shows a necrotic and hemorrhagic morphology with lining of the thin capsule.

  • Fig. 3 Pathologic findings. A : Pathologic findings at the periphery of the tumor show a capsule made of parallel lamellae of collagen representing a vascular wall, vascular lumen, and papillary projection into the lumen. B : The pathologic result is factor VIII positive.


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