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Neurologic Aspects of Hereditary Hemorrhagic Telangiectasia: Report of Two Cases

Neurologic Aspects of Hereditary Hemorrhagic Telangiectasia: Report of Two Cases Abstract • The several neurologic manifestations of hereditary hemorrhagic telangiectasia (HHT) may be caused by complications of pulmonary arteriovenous fistulae or associated central nervous system vascular malformations. The presence of skin and mucosal telangiectases should alert the clinician to the possibility of the disorder and in turn of its potential for associated neurologic disease, including cerebral hemorrhage and abscess. This report describes two cases and demonstrates that the clinical spectrum of HHT should be enlarged to include its admittedly rare, but serious, neurologic aspects. References 1. Bean WB: Congenital and hereditary lesions and birthmarks , in Vascular Spiders and Related Lesions of the Skin . Springfield, Ill, Charles C Thomas Publisher, 1958, pp 132-157. 2. Boczko ML: Neurological implications of hereditary hemorrhagic telangiectasis . J Nerv Ment Dis 139:525-536, 1964.Crossref 3. Hodgson CA, Burchell G, Good A, et al: Hereditary hemorrhagic telangiectasia and pulmonary arteriovenous fistula: Survey of a large family . N Engl J Med 261:625-636, 1959.Crossref 4. Moyer JH, Glantz G, Brest AN: Pulmonary arteriovenous fistulas: Physiologic and clinical considerations . Am J Med 32:417-435, 1962.Crossref 5. Dines DE, Arms RA, Bernatz PE, et al: Pulmonary arteriovenous fistulas . Mayo Clin Proc 49:460-465, 1974. 6. Muri JW: Arteriovenous aneurysm of the lung . Am J Surg 89:265-271, 1955.Crossref 7. LeRoux BT: Pulmonary arteriovenous fistulae . Q J Med 28:1-19, 1959. 8. Shub C, Tajik AJ, Seward JB: Detecting intrapulmonary right-to-left shunt with contract echocardiography: Observations in a patient with diffuse pulmonary arteriovenous fistulas . Mayo Clin Proc 51:81-84, 1976. 9. Hodgson CH, Kaye RL: Pulmonary arteriovenous fistula and hereditary hemorrhagic telangiectasia: A review and report of 35 cases of fistula . Dis Chest 43:449-455, 1963.Crossref 10. Sluiter-Eringa H, Orie NGM, Sluiter HG: Pulmonary arteriovenous fistula: Diagnosis and prognosis in noncomplaintant patients . Am Rev Respir Dis 100:177-188, 1969. 11. Sisel RJ, Parker BM, Bahl OP: Cerebral symptoms in pulmonary arteriovenous fistula: A result of paradoxical emboli (?) . Circulation 41:123-128, 1970.Crossref 12. Reading B: A case of congenital telangiectasis of the lung complicated by brain abscess . Tex State J Med 28:462-464, 1932. 13. Stern WE, Naffziger HC: Brain abscess associated with pulmonary angiomatous malformation . Ann Surg 138:521-531, 1953.Crossref 14. Taber RE, Ehrenhaft JL: Arteriovenous fistulae and arterial aneurysms of the pulmonary arterial tree . Arch Surg 73:567-577, 1956.Crossref 15. Dyer NH: Cerebral abscess in hereditary haemorrhagic telangiectasis: Report of two cases in a family . J Neurol Neurosurg Psychiatry 30:563-567, 1967.Crossref 16. Perret G, Nishioka H: Report on the cooperative study of intracranial aneurysms and subarachnoid hemorrhage: VI. Arteriovenous malformations . J Neurosurg 25:467-490, 1966.Crossref 17. Waller JD, Greenberg JH, Lewis CW: Hereditary hemorrhagic telangiectasia with cerebrovascular malformations . Arch Dermatol 112:49-52, 1976.Crossref 18. Cohn HM, Rosenthal FE: Hereditary haemorrhagic telangiectasia and its relations to other inborn vascular malformations . Acta Haematol 1:81-91, 1948.Crossref 19. Merry GS, Appleton DB: Spinal arterial malformation in a child with hereditary hemorrhagic telangiectasia . J Neurosurg 44:613-616, 1976.Crossref 20. Gautam HP: Pulmonary arteriovenous fistula . Int Surg 46:168-175, 1966. 21. Arnould G, Dureux JB, Tridon P, et al: Malformations vasculaires cerebrales et angiomatose de Rendu-Osler . Rev Neurol 119:230-235, 1968. 22. Schauman B, Alter M: Cerebral arteriovenous fistula in hereditary hemorrhagic telangiectasia . Am J Dis Child 126:563, 1973. 23. Boynton RC, Morgan BC: Cerebral arteriovenous fistula with possible hereditary telangiectasia . Am J Dis Child 125:99-101, 1973. 24. Courville CB: Encephalic lesions in hereditary hemorrhagic telangiectasis (Rendu-Osler-Weber Disease): Report of a case with the disclosure of microscopic telangiectasis in the leptomeninges . Bull LA Neurol Soc 22:28-35, 1957. 25. Heffner RR, Solitare GB: Hereditary hemorrhagic telangiectasis: Neuropathological observations . J Neurol Neurosurg Psychiatry 32:604-608, 1969.Crossref 26. Chandler D: Pulmonary and cerebral arteriovenous fistula with Osler's disease . Arch Intern Med 116:277-282, 1965.Crossref 27. Sochocky S: Arteriovenous fistula of the lung . Lancet 82:12-17, 1962. 28. Walton JN: Subarachnoid haemorrhage of unusual aetiology . Neurology 3:517-543, 1953.Crossref 29. Quickel KE Jr, Whaley RJ: Subarachnoid hemorrhage in a patient with hereditary hemorrhagic telangiectasia . Neurology 17:716-719, 1967.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Neurology American Medical Association

Neurologic Aspects of Hereditary Hemorrhagic Telangiectasia: Report of Two Cases

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Publisher
American Medical Association
Copyright
Copyright © 1977 American Medical Association. All Rights Reserved.
ISSN
0003-9942
eISSN
1538-3687
DOI
10.1001/archneur.1977.00500140055011
Publisher site
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Abstract

Abstract • The several neurologic manifestations of hereditary hemorrhagic telangiectasia (HHT) may be caused by complications of pulmonary arteriovenous fistulae or associated central nervous system vascular malformations. The presence of skin and mucosal telangiectases should alert the clinician to the possibility of the disorder and in turn of its potential for associated neurologic disease, including cerebral hemorrhage and abscess. This report describes two cases and demonstrates that the clinical spectrum of HHT should be enlarged to include its admittedly rare, but serious, neurologic aspects. References 1. Bean WB: Congenital and hereditary lesions and birthmarks , in Vascular Spiders and Related Lesions of the Skin . Springfield, Ill, Charles C Thomas Publisher, 1958, pp 132-157. 2. Boczko ML: Neurological implications of hereditary hemorrhagic telangiectasis . J Nerv Ment Dis 139:525-536, 1964.Crossref 3. Hodgson CA, Burchell G, Good A, et al: Hereditary hemorrhagic telangiectasia and pulmonary arteriovenous fistula: Survey of a large family . N Engl J Med 261:625-636, 1959.Crossref 4. Moyer JH, Glantz G, Brest AN: Pulmonary arteriovenous fistulas: Physiologic and clinical considerations . Am J Med 32:417-435, 1962.Crossref 5. Dines DE, Arms RA, Bernatz PE, et al: Pulmonary arteriovenous fistulas . Mayo Clin Proc 49:460-465, 1974. 6. Muri JW: Arteriovenous aneurysm of the lung . Am J Surg 89:265-271, 1955.Crossref 7. LeRoux BT: Pulmonary arteriovenous fistulae . Q J Med 28:1-19, 1959. 8. Shub C, Tajik AJ, Seward JB: Detecting intrapulmonary right-to-left shunt with contract echocardiography: Observations in a patient with diffuse pulmonary arteriovenous fistulas . Mayo Clin Proc 51:81-84, 1976. 9. Hodgson CH, Kaye RL: Pulmonary arteriovenous fistula and hereditary hemorrhagic telangiectasia: A review and report of 35 cases of fistula . Dis Chest 43:449-455, 1963.Crossref 10. Sluiter-Eringa H, Orie NGM, Sluiter HG: Pulmonary arteriovenous fistula: Diagnosis and prognosis in noncomplaintant patients . Am Rev Respir Dis 100:177-188, 1969. 11. Sisel RJ, Parker BM, Bahl OP: Cerebral symptoms in pulmonary arteriovenous fistula: A result of paradoxical emboli (?) . Circulation 41:123-128, 1970.Crossref 12. Reading B: A case of congenital telangiectasis of the lung complicated by brain abscess . Tex State J Med 28:462-464, 1932. 13. Stern WE, Naffziger HC: Brain abscess associated with pulmonary angiomatous malformation . Ann Surg 138:521-531, 1953.Crossref 14. Taber RE, Ehrenhaft JL: Arteriovenous fistulae and arterial aneurysms of the pulmonary arterial tree . Arch Surg 73:567-577, 1956.Crossref 15. Dyer NH: Cerebral abscess in hereditary haemorrhagic telangiectasis: Report of two cases in a family . J Neurol Neurosurg Psychiatry 30:563-567, 1967.Crossref 16. Perret G, Nishioka H: Report on the cooperative study of intracranial aneurysms and subarachnoid hemorrhage: VI. Arteriovenous malformations . J Neurosurg 25:467-490, 1966.Crossref 17. Waller JD, Greenberg JH, Lewis CW: Hereditary hemorrhagic telangiectasia with cerebrovascular malformations . Arch Dermatol 112:49-52, 1976.Crossref 18. Cohn HM, Rosenthal FE: Hereditary haemorrhagic telangiectasia and its relations to other inborn vascular malformations . Acta Haematol 1:81-91, 1948.Crossref 19. Merry GS, Appleton DB: Spinal arterial malformation in a child with hereditary hemorrhagic telangiectasia . J Neurosurg 44:613-616, 1976.Crossref 20. Gautam HP: Pulmonary arteriovenous fistula . Int Surg 46:168-175, 1966. 21. Arnould G, Dureux JB, Tridon P, et al: Malformations vasculaires cerebrales et angiomatose de Rendu-Osler . Rev Neurol 119:230-235, 1968. 22. Schauman B, Alter M: Cerebral arteriovenous fistula in hereditary hemorrhagic telangiectasia . Am J Dis Child 126:563, 1973. 23. Boynton RC, Morgan BC: Cerebral arteriovenous fistula with possible hereditary telangiectasia . Am J Dis Child 125:99-101, 1973. 24. Courville CB: Encephalic lesions in hereditary hemorrhagic telangiectasis (Rendu-Osler-Weber Disease): Report of a case with the disclosure of microscopic telangiectasis in the leptomeninges . Bull LA Neurol Soc 22:28-35, 1957. 25. Heffner RR, Solitare GB: Hereditary hemorrhagic telangiectasis: Neuropathological observations . J Neurol Neurosurg Psychiatry 32:604-608, 1969.Crossref 26. Chandler D: Pulmonary and cerebral arteriovenous fistula with Osler's disease . Arch Intern Med 116:277-282, 1965.Crossref 27. Sochocky S: Arteriovenous fistula of the lung . Lancet 82:12-17, 1962. 28. Walton JN: Subarachnoid haemorrhage of unusual aetiology . Neurology 3:517-543, 1953.Crossref 29. Quickel KE Jr, Whaley RJ: Subarachnoid hemorrhage in a patient with hereditary hemorrhagic telangiectasia . Neurology 17:716-719, 1967.Crossref

Journal

Archives of NeurologyAmerican Medical Association

Published: Feb 1, 1977

References