Background <p>Infantile hemangiomas (IHs) are the most common vascular tumors in infancy, typically occurring in the skin and soft tissues. Intracranial infantile hemangiomas (IIHs) are exceedingly rare, usually extra-axial, and often associated with PHACES syndrome. Hemorrhagic intra-axial IIHs in neonates are exceptionally uncommon.</p> Case description <p>We report a newborn presenting at 7&#xa0;days of life with irritability, vomiting, and acute intracranial hypertension. Imaging revealed a hemorrhagic mass in the corpus callosum with intraventricular extension. Emergency external ventricular drainage was performed, followed by a right frontal–parietal parasagittal craniotomy. A well-circumscribed reddish-brown and vascular tumor was removed entirely. Histopathology showed capillary proliferation without atypia, and immunohistochemistry was positive for CD31, CD34, and GLUT1, consistent with infantile hemangioma.</p> Results <p>The postoperative course was uneventful. At 3&#xa0;years of follow-up, the child shows normal neurodevelopment and no evidence of recurrence on MRI.</p> Conclusion <p>Isolated intra-axial IIH with neonatal hemorrhage is an exceptional presentation. This case highlights the importance of considering IIH in the differential diagnosis of highly vascular intracranial tumors, even in the absence of cutaneous hemangiomas. Prompt surgical resection can be curative and prevent life-threatening complications.</p>

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Primary intracranial infantile hemangioma presenting with hemorrhage: histopathological and immunohistochemical confirmation of a rare lesion

  • Hamilton Matushita,
  • Daniel Dante Cardeal,
  • Vitor Nagai Yamaki,
  • Romulo Loss Mattedi

摘要

Background

Infantile hemangiomas (IHs) are the most common vascular tumors in infancy, typically occurring in the skin and soft tissues. Intracranial infantile hemangiomas (IIHs) are exceedingly rare, usually extra-axial, and often associated with PHACES syndrome. Hemorrhagic intra-axial IIHs in neonates are exceptionally uncommon.

Case description

We report a newborn presenting at 7 days of life with irritability, vomiting, and acute intracranial hypertension. Imaging revealed a hemorrhagic mass in the corpus callosum with intraventricular extension. Emergency external ventricular drainage was performed, followed by a right frontal–parietal parasagittal craniotomy. A well-circumscribed reddish-brown and vascular tumor was removed entirely. Histopathology showed capillary proliferation without atypia, and immunohistochemistry was positive for CD31, CD34, and GLUT1, consistent with infantile hemangioma.

Results

The postoperative course was uneventful. At 3 years of follow-up, the child shows normal neurodevelopment and no evidence of recurrence on MRI.

Conclusion

Isolated intra-axial IIH with neonatal hemorrhage is an exceptional presentation. This case highlights the importance of considering IIH in the differential diagnosis of highly vascular intracranial tumors, even in the absence of cutaneous hemangiomas. Prompt surgical resection can be curative and prevent life-threatening complications.