The Journal of Laryngology and Otology November 1979. Vol. 93. pp. 1127-1129
Massive epistaxis caused by cerebral arterio-venous fistula By T. A.
FRAYNE
and D. W.
BOLDT
(Adelaide)
VENOUS varicosities of the naso-ethmoid region are extremely rare. A patient who had repeated life-threatening epistaxes from such varices is reported. The underlying cause appeared to be a large intra-cerebral arterio-venous malformation. In a search of the literature we have not discovered a report of a similar association and wish to draw attention to this unusual cause of massive epistaxes.
' Case report A white female child aged 4 years presented at the Casualty Department of a pediatric hospital with a sudden onset of epistaxis two hours previously. Clinical and haematological examinations were normal and the child was sent away. Three hours later she returned following a larger bleed. She was clinically shocked due to blood loss. Five hours later a third haemorrhage occurred and required emergency resuscitation; the haemorrhage was controlled by inflation of a Brighton balloon catheter in the right nasal airway. Three attempts to remove the catheter were unsuccessful due to recurrence of bleeding. At the third attempt under general anaesthetic the blood was noted to be venous and coming from near the roof of the nose bilaterally. Calgitex gauze packing was inserted into the nose bilaterally and the pack disintegrated over the subsequent two months. Fundoscopy revealed dilated retinal veins, and prominent frontal and scalp veins were also noted. A loud bruit was heard over the cranium. The past history showed that the child first presented at the age of one day with congestive cardiac failure, for which cardiac catheterization and cine angiography had later failed to show a cause. Over the subsequent two years she was noted to have a left spastic hemiparesis, an enlarged cranium and mental retardation. Angiography was performed to define the lesion, with contrast injection into both common carotid and vertebral arteries. This demonstrated a huge arteriovenous fistula in the parietal region of the right cerebral hemisphere receiving arterial blood from both internal carotid arteries and the markedly dilated left vertebral artery. In the venous phase dilated varicosities were demonstrated around the brain-stem draining anteriorly to the back of the orbits (Fig. 1). Orbital venography demonstrated dilated tortuous veins in the medial aspect of both orbits and in the naso-ethmoid region (Fig. 2). Following disintergration of the cellulose nasal packs there was no further bleeding. Because of the patient's other clinical problems it was decided not to attempt removal of the arterio-venous fistula. Conclusion It seems likely that the varicosities derived from the arterio-venous malformation drained at least in part on to the face through orbital and naso1127
(a) Lateral: common carotid injection, the catheter having recoiled to the aorta. The arterio-venous fistula (single arrow) is supplied by branches of the right posterior (single arrow head), middle (triple arrow head) and anterior (double arrow head) cerebral arteries.
FIG. 1 Right Carotid Angiography (b) Lateral: internal carotid, late venous phase. In (b) the short arrows indicate the venous varicosities surrounding the brain stem extending anteriorly to the parasellar regions.
CLINICAL RECORDS
1129
FIG. 2 Fronto-orbital Venigram—AP Projection The dilated angular vein on the left is shown occluded (arrow) by a mid-facial tourniquet. The supra-orbital veins (triple arrow) are dilated. The dilated varicosities in the orbits medially and ethmoid complexes are shown between the large arrow heads. The double arrow head indicates a large vein draining posteriorly from the left orbit.
ethmoid veins and that rupture of one or more of the latter caused the epistaxes. In cases of massive epistaxis in children such an arterio-venous malformation must be suspected and the lesion defined by clinical and radiographic investigations. Acknowledgements We wish to thank Mr. B. Bellon, Consultant Ear, Nose and Throat Surgeon, Adelaide Children's Hospital, for allowing us to publish this paper, and Ms. Louise Rodbourn for typing the manuscript. Address for reprints: Dr. T. A. Frayne, 3 Thornton Street, Dulwich, South Australia 5065.
Massive epistaxis caused by cerebral arterio-venous fistula By T. A.
FRAYNE
and D. W.
BOLDT
(Adelaide)
VENOUS varicosities of the naso-ethmoid region are extremely rare. A patient who had repeated life-threatening epistaxes from such varices is reported. The underlying cause appeared to be a large intra-cerebral arterio-venous malformation. In a search of the literature we have not discovered a report of a similar association and wish to draw attention to this unusual cause of massive epistaxes.
' Case report A white female child aged 4 years presented at the Casualty Department of a pediatric hospital with a sudden onset of epistaxis two hours previously. Clinical and haematological examinations were normal and the child was sent away. Three hours later she returned following a larger bleed. She was clinically shocked due to blood loss. Five hours later a third haemorrhage occurred and required emergency resuscitation; the haemorrhage was controlled by inflation of a Brighton balloon catheter in the right nasal airway. Three attempts to remove the catheter were unsuccessful due to recurrence of bleeding. At the third attempt under general anaesthetic the blood was noted to be venous and coming from near the roof of the nose bilaterally. Calgitex gauze packing was inserted into the nose bilaterally and the pack disintegrated over the subsequent two months. Fundoscopy revealed dilated retinal veins, and prominent frontal and scalp veins were also noted. A loud bruit was heard over the cranium. The past history showed that the child first presented at the age of one day with congestive cardiac failure, for which cardiac catheterization and cine angiography had later failed to show a cause. Over the subsequent two years she was noted to have a left spastic hemiparesis, an enlarged cranium and mental retardation. Angiography was performed to define the lesion, with contrast injection into both common carotid and vertebral arteries. This demonstrated a huge arteriovenous fistula in the parietal region of the right cerebral hemisphere receiving arterial blood from both internal carotid arteries and the markedly dilated left vertebral artery. In the venous phase dilated varicosities were demonstrated around the brain-stem draining anteriorly to the back of the orbits (Fig. 1). Orbital venography demonstrated dilated tortuous veins in the medial aspect of both orbits and in the naso-ethmoid region (Fig. 2). Following disintergration of the cellulose nasal packs there was no further bleeding. Because of the patient's other clinical problems it was decided not to attempt removal of the arterio-venous fistula. Conclusion It seems likely that the varicosities derived from the arterio-venous malformation drained at least in part on to the face through orbital and naso1127
(a) Lateral: common carotid injection, the catheter having recoiled to the aorta. The arterio-venous fistula (single arrow) is supplied by branches of the right posterior (single arrow head), middle (triple arrow head) and anterior (double arrow head) cerebral arteries.
FIG. 1 Right Carotid Angiography (b) Lateral: internal carotid, late venous phase. In (b) the short arrows indicate the venous varicosities surrounding the brain stem extending anteriorly to the parasellar regions.
CLINICAL RECORDS
1129
FIG. 2 Fronto-orbital Venigram—AP Projection The dilated angular vein on the left is shown occluded (arrow) by a mid-facial tourniquet. The supra-orbital veins (triple arrow) are dilated. The dilated varicosities in the orbits medially and ethmoid complexes are shown between the large arrow heads. The double arrow head indicates a large vein draining posteriorly from the left orbit.
ethmoid veins and that rupture of one or more of the latter caused the epistaxes. In cases of massive epistaxis in children such an arterio-venous malformation must be suspected and the lesion defined by clinical and radiographic investigations. Acknowledgements We wish to thank Mr. B. Bellon, Consultant Ear, Nose and Throat Surgeon, Adelaide Children's Hospital, for allowing us to publish this paper, and Ms. Louise Rodbourn for typing the manuscript. Address for reprints: Dr. T. A. Frayne, 3 Thornton Street, Dulwich, South Australia 5065.
