J Neurosurg Pediatrics 14:418–420, 2014
©AANS, 2014

Development of a de novo arteriovenous malformation after
severe traumatic brain injury
Case report
Brandon A. Miller, M.D., Ph.D.,1 David I. Bass, B.A.,1
and Joshua J. Chern, M.D., Ph.D.1,2
Department of Neurosurgery, Emory University, Atlanta; and 2Pediatric Neurosurgery Associates, Children’s
Healthcare of Atlanta, Georgia
1

Arteriovenous malformations (AVMs) are typically considered congenital lesions, although there is growing
evidence for de novo formation of these lesions as well. The authors present the case of an AVM in the same cerebral
cortex that had been affected by a severe traumatic brain injury (TBI) more than 6 years earlier. To the best of the
authors’ knowledge, this is the first report attributing the formation of an AVM directly to TBI.
(http://thejns.org/doi/abs/10.3171/2014.7.PEDS1431)

Key Words
• AVM
•
arteriovenous malformation
vascular disorders
• trauma

A

rteriovenous malformations (AVMs) are generally considered to be congenital lesions that are
discovered either incidentally or after becoming
symptomatic, most often due to hemorrhage. Several
case reports have described the de novo development of
AVMs when prior brain imaging has been performed.1,4,19
We present the unique case of a child who had sustained
a severe traumatic brain injury (TBI), underwent decompressive hemicraniectomy and cranioplasty, and later developed a de novo AVM in the injured cortex.

Case Report
History and Examination. An 11-month-old girl with
no remarkable medical history presented to an outside
hospital after falling from her parents’ porch. Initial head
CT demonstrated left-sided skull fractures and a subdural hematoma. The patient was immediately taken for clot
evacuation and decompressive hemicraniectomy and was
then transferred to our pediatric hospital. She underwent
cranioplasty 10 days after her first surgery and made a
good recovery. At 22 months of age, she underwent revision cranioplasty at our institution for a persistent skull
defect. She had recovered well from her injury with intact
speech and the ability to ambulate without assistance, albeit with persistent right hemiparesis.
Abbreviations used in this paper: AVM = arteriovenous malformation; TBI = traumatic brain injury.

418

•

traumatic brain injury

•

At 6 years of age, she developed a seizure disorder.
Magnetic resonance imaging of her brain was performed
at that time and demonstrated encephalomalacia at the
site of the prior injury with no evidence of a vascular malformation (Fig. 1 left). Her seizures were well controlled
with medication until she was 12 years old, when her seizure frequency increased to approximately two episodes
per week. In response to this change, she underwent repeat MRI, which revealed a vascular malformation within the large area of encephalomalacia in the left parietal
lobe (Fig. 1 right). Angiography demonstrated an AVM
measuring 2.5 cm in diameter, fed predominantly from
the left pericallosal artery and draining into the superior
sagittal sinus (Fig. 2). The patient had no clinical signs or
radiological evidence of hemorrhage from the AVM.
Operation. Shortly after angiography, the patient underwent elective resection of the AVM. The feeding arteries and then the draining veins were coagulated, and the
AVM was removed en bloc. Intraoperative visualization
and pathological examination of tissue revealed a typical
AVM with no unusual characteristics (Fig. 3).
Postoperative Course. There were no surgical complications, and the patient made an uneventful recovery.
Three weeks after surgery, she returned to the clinic at her
neurological baseline. Although she experienced a cluster
This article contains some figures that are displayed in color
on­line but in black-and-white in the print edition.

J Neurosurg: Pediatrics / Volume 14 / October 2014

Posttraumatic arteriovenous malformation

Fig. 3. Photomicrographs of biopsied AVM tissue demonstrating
the typical morphology of tortuous pathological vessels. H & E, original
magnification ×40 (left) and ×100 (right).
Fig. 1. Axial T2-weighted MRI studies obtained before and after
development of an AVM. Left: Image obtained when the patient presented with seizures at 6 years of age, demonstrating encephalomalacia and no vascular lesion. Right: Image obtained when the patient
was 12 years of age, demonstrating left parietal vascular malformation.

of seizures on postoperative Day 1, she has been seizure
free for 2 months since then.

Discussion

This is the first reported case of an AVM developing
within traumatized brain. Arteriovenous malformations
are generally considered to be congenital lesions, which
are discovered either incidentally or after hemorrhage.
De novo AVMs have been described in both children
and adults,4 with the majority of lesions being noted in
younger, female patients13 similar to our patient. There
are case reports of de novo AVM formation in children
associated with cavernous malformations2 or moyamoya
disease15,18 or without any previous vascular pathology.13,19
In a study of younger patients with AVMs, almost 10% of
the patients had undergone previous angiography without
evidence of vascular malformation.11 There are also multiple case reports of AVMs presenting with subdural hemorrhage, 5,17 but no reports that we are aware of attribute
an acute supratentorial subdural hemorrhage to an AVM.
The occurrence of other vascular pathologies, such
as aneurysms and dural arteriovenous fistulas, following head trauma have been well documented.3,16 To our
knowledge, only one prior report has described the development of an AVM in a child after head trauma, and that

case is substantially different from the one we pre­sent
here.7 The AVM in that patient developed within radiographically normal brain tissue distant from the site of
the injury. Furthermore, that patient had suffered a less
severe TBI, with only a small amount of traumatic subarachnoid hemorrhage. Therefore, the authors believed
that the AVM could not be definitively related to the prior
head injury.
The growing number of reports on de novo AVMs
together with reports on the growth and regression of
AVMs6 is evidence that these lesions are dynamic, rather
than static, entities. There is a small possibility that our
patient harbored an AVM from the outset of her injury
and that it went undetected until her second MRI study.
However, we do not believe this to be the case, as MRI
has been shown to be 97% sensitive in detecting unruptured AVMs.9 Our patient’s first MRI study was of good
quality and was reviewed by neurosurgeons and a pediatric neuroradiologist with knowledge of the subsequent
MRI findings. Furthermore, our patient’s medical history
was clear. Her parents described a fall and resultant head
trauma without any antecedent neurological changes, and
there were no findings on her initial posttraumatic imaging that suggested anything other than typical subdural
hemorrhage and skull fracture. Neuroimaging in the subacute and chronic phase of TBI has demonstrated hippocampal atrophy, white matter connectivity changes, and
defined areas of encephalomalacia that may be resected
for the treatment of epilepsy.8,10,20 While there are no specific guidelines on repeat imaging in patients during the
subacute or chronic phase of TBI, the present case emphasizes the fact that new or worsening symptoms remote
from a TBI still warrant radiographic investigation.

Conclusions

Fig. 2. Axial (left) and sagittal (right) angiograms obtained after a
second MRI study, demonstrating a left parietal AVM fed from branches
of the pericallosal artery.

J Neurosurg: Pediatrics / Volume 14 / October 2014

In summary, to our knowledge, this is the first report
documenting the development of an AVM in response to
severe TBI. There is a theory that de novo AVM development can be attributed to a “two-hit” phenomenon in
which trauma may serve as the “second hit.”2 Our patient
had no medical or family medical history to suggest a first
hit, leading us to ascribe the development of this AVM
solely to the severe TBI she had suffered years before the
AVM was discovered. Much research has shown that angiogenic factors, such as vascular endothelial growth factor and stromal cell–derived factor 1, increase in the days
following a TBI,12,14 but the subacute release of these fac419

B. A. Miller, D. I. Bass, and J. J. Chern
tors is unlikely to account for the protracted development
of the AVM a decade later. Thus, the underlying mechanism linking the injury to the AVM remains unclear.
Disclosure
The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this
paper.
Author contributions to the study and manuscript preparation include the following. Conception and design: Miller, Chern.
Acquisition of data: all authors. Analysis and interpretation of data:
all authors. Drafting the article: Miller, Bass. Critically revising
the article: all authors. Reviewed submitted version of manuscript:
Miller.
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Manuscript submitted January 21, 2014.
Accepted July 7, 2014.
Please include this information when citing this paper: published
online August 1, 2014; DOI: 10.3171/2014.7.PEDS1431.
Address correspondence to: Brandon A. Miller, M.D., Ph.D.,
Department of Neurosurgery, Emory University School of Medicine, 1365-B Clifton Rd., Ste. 6200, Atlanta, GA 30322. email:
brandon.miller@emory.edu.

J Neurosurg: Pediatrics / Volume 14 / October 2014