Delayed fatal intracranial hemorrhage in a pediatric patient following resection and adjuvant cranial radiotherapy for ependymoma: illustrative case

Abstract

Background: Cranial radiotherapy (CRT) is an important treatment modality for malignancies of the central nervous system. CRT has deleterious effects that are commonly classified into acute, early delayed, and late delayed. Late-delayed effects include weakening of the cerebral vasculature and the development of structurally abnormal vasculature, potentially leading to ischemic or hemorrhagic events within the brain parenchyma. Such events are not well reported in the pediatric population.

Observations: The authors present the case of a 14-year-old patient 8.2 years after CRT who experienced intracerebral hemorrhage. Autopsy demonstrated minimal pathological change without evidence of vascular malformation or aneurysm. These findings were unexpected given the degree of hemorrhage in this case. However, in the absence of other etiologies, it was believed that late-delayed radiation effect was the cause of this patient's fatal hemorrhage.

Lessons: Although not all cases of pediatric spontaneous intracerebral hemorrhage will have a determined etiology, the authors' patient's previous CRT may represent a poorly defined risk for late-delayed hemorrhage. This correlation has not been previously reported and should be considered in pediatric patients presenting with spontaneous hemorrhage in a delayed fashion after CRT. Neurosurgeons must not be dismissive of unexpected events in the remote postoperative period.

Keywords: cranial radiotherapy; death; intracerebral hemorrhage; late-delayed effects of radiotherapy; pediatric.

FIG. 1.

Preoperative axial post–gadolinium T1-weighted MRI (A) demonstrating a fourth ventricular avidly enhancing mass. Preoperative sagittal post–gadolinium T1-weighted MRI (B) demonstrating a fourth ventricular enhancing mass protruding through the foramen of Magendie. Six-month postoperative axial (C) and sagittal (D) post–gadolinium T1-weighted MRI demonstrating gross-total resection of the fourth ventricular enhancing mass.

FIG. 2.

Axial (left) and sagittal (right) post–gadolinium T1-weighted MRI performed 7.8 years post-CRT, showing stable postoperative change without vascular abnormality.

FIG. 3.

Noncontrast CT demonstrating intraparenchymal hemorrhage with extensive dissemination throughout the ventricular system occurring 8.2 years after CRT.

FIG. 4.

Chest radiograph demonstrating bilateral consolidations within the apices of the lung, consistent with neurogenic pulmonary edema occurring 8.2 years after CRT.