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Cerebral Arteriovenous Fistula in Hereditary Hemorrhagic Telangiectasia

Cerebral Arteriovenous Fistula in Hereditary Hemorrhagic Telangiectasia Abstract To the Editor.—Recently, Boynton and Morgan1 described an infant with a positive family history of hereditary hemorrhagic telangiectasia (HHT) who died of congestive heart failure secondary to a large cerebral arteriovenous (AV) malformation. The authors believed their case was a unique instance of large cerebral AV malformation associated with HHT. However, other such cases have been reported previously2-4 and we have seen an additional example in a 46-year-old man with typical HHT, whose angiogram revealed a large AV malformation in the left frontal-parietal area and a smaller one in the right frontal pole. Cerebral vascular malformations of smaller and unspecified size have also been described in patients with HHT.5-10 In its relation to HHT, Boynton and Morgan's case was unusual only with respect to the early age at which the cerebral AV malformation manifested. In view of the high frequency of neurological symptoms in HHT, such References 1. Boynton RC, Morgan BC: Cerebral arteriovenous fistula with possible hereditary telangiectasia . Am J Dis Child 125:99-101, 1973. 2. Boczko ML: Neurological implications of hereditary hemorrhagic telangiectasis . J Nerv Ment Dis 139:525-536, 1964.Crossref 3. Chandler D: Pulmonary and cerebral arteriovenous fistula with Osler's disease . Arch Intern Med 116:277-282, 1965.Crossref 4. Czernobilsky B, Bouzarth WF: Vascular malformations in the brain in a patient with multiple telangiectasia of the intestine and lungs . J Am Med Wom Assoc 20:337-340, 1965. 5. Courville CB: Encephalic lesions in hereditary hemorrhagic telangiectasis (Rendu-Osler-Weber disease) . Bull Los Angeles Neurol Soc 22:28-35, 1957. 6. Bird RM, Jaques WE: Vascular lesion of hereditary hemorrhagic telangiectasia . N Engl J Med 260:597-599, 1959.Crossref 7. Reagan TJ, Bloom WH: The brain in hereditary hemorrhagic telangiectasia . Stroke 2:361-368, 1971.Crossref 8. Cohn HM, Rosenthal FE: Hereditary haemorrhagic teleangiectasia and its relations to other inborn vascular malformations . Acta Haematol 1:81-92, 1948.Crossref 9. Quickel KE, Whaley RJ: Subarachnoid hemorrhage in a patient with hereditary hemorrhagic telangiectasia . Neurology 17:716-719, 1967.Crossref 10. Giampalmo A, Badini A, Buffa D: Segnalazione di due casi autopsici di teleangiectasia generalizzata: L'uno con ripetute emorragie esterne (caratteristica malattia di Rendu-Osler), l'altro con emorragie pleuriche e viscerali e angioma venoso cerebrale . Pathologica 63:55-60, 1971. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png American Journal of Diseases of Children American Medical Association

Cerebral Arteriovenous Fistula in Hereditary Hemorrhagic Telangiectasia

Cerebral Arteriovenous Fistula in Hereditary Hemorrhagic Telangiectasia

Abstract

Abstract To the Editor.—Recently, Boynton and Morgan1 described an infant with a positive family history of hereditary hemorrhagic telangiectasia (HHT) who died of congestive heart failure secondary to a large cerebral arteriovenous (AV) malformation. The authors believed their case was a unique instance of large cerebral AV malformation associated with HHT. However, other such cases have been reported previously2-4 and we have seen an additional example in a 46-year-old man with...
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Publisher
American Medical Association
Copyright
Copyright © 1973 American Medical Association. All Rights Reserved.
ISSN
0002-922X
DOI
10.1001/archpedi.1973.02110190459026
Publisher site
See Article on Publisher Site

Abstract

Abstract To the Editor.—Recently, Boynton and Morgan1 described an infant with a positive family history of hereditary hemorrhagic telangiectasia (HHT) who died of congestive heart failure secondary to a large cerebral arteriovenous (AV) malformation. The authors believed their case was a unique instance of large cerebral AV malformation associated with HHT. However, other such cases have been reported previously2-4 and we have seen an additional example in a 46-year-old man with typical HHT, whose angiogram revealed a large AV malformation in the left frontal-parietal area and a smaller one in the right frontal pole. Cerebral vascular malformations of smaller and unspecified size have also been described in patients with HHT.5-10 In its relation to HHT, Boynton and Morgan's case was unusual only with respect to the early age at which the cerebral AV malformation manifested. In view of the high frequency of neurological symptoms in HHT, such References 1. Boynton RC, Morgan BC: Cerebral arteriovenous fistula with possible hereditary telangiectasia . Am J Dis Child 125:99-101, 1973. 2. Boczko ML: Neurological implications of hereditary hemorrhagic telangiectasis . J Nerv Ment Dis 139:525-536, 1964.Crossref 3. Chandler D: Pulmonary and cerebral arteriovenous fistula with Osler's disease . Arch Intern Med 116:277-282, 1965.Crossref 4. Czernobilsky B, Bouzarth WF: Vascular malformations in the brain in a patient with multiple telangiectasia of the intestine and lungs . J Am Med Wom Assoc 20:337-340, 1965. 5. Courville CB: Encephalic lesions in hereditary hemorrhagic telangiectasis (Rendu-Osler-Weber disease) . Bull Los Angeles Neurol Soc 22:28-35, 1957. 6. Bird RM, Jaques WE: Vascular lesion of hereditary hemorrhagic telangiectasia . N Engl J Med 260:597-599, 1959.Crossref 7. Reagan TJ, Bloom WH: The brain in hereditary hemorrhagic telangiectasia . Stroke 2:361-368, 1971.Crossref 8. Cohn HM, Rosenthal FE: Hereditary haemorrhagic teleangiectasia and its relations to other inborn vascular malformations . Acta Haematol 1:81-92, 1948.Crossref 9. Quickel KE, Whaley RJ: Subarachnoid hemorrhage in a patient with hereditary hemorrhagic telangiectasia . Neurology 17:716-719, 1967.Crossref 10. Giampalmo A, Badini A, Buffa D: Segnalazione di due casi autopsici di teleangiectasia generalizzata: L'uno con ripetute emorragie esterne (caratteristica malattia di Rendu-Osler), l'altro con emorragie pleuriche e viscerali e angioma venoso cerebrale . Pathologica 63:55-60, 1971.

Journal

American Journal of Diseases of ChildrenAmerican Medical Association

Published: Oct 1, 1973

References