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GASTROINTESTINAL BLEEDING IN HEREDITARY HEMORRHAGIC TELANGIECTASIA: Review of the Literature and Report of a Case with Severe Recurrent Hemorrhages Necessitating Total Gastrectomy

GASTROINTESTINAL BLEEDING IN HEREDITARY HEMORRHAGIC TELANGIECTASIA: Review of the Literature and... Abstract HEREDITARY hemorrhagic telangiectasia (Rendu-Osler-Weber disease) is a hereditary vascular anomaly characterized pathologically by multiple dilatations of capillaries and venules of the skin and mucous membranes and characterized clinically by recurrent hemorrhage from these telangiectasia, most frequently manifested by epistaxis. The entity is often overlooked or the bleeding attributed to some other cause, especially a blood dyscrasia. In gastrointestinal bleeding, peptic ulcer is often suspected, and, indeed, the two conditions may be present concurrently. The condition was probably first reported by Sutton,1 in 1864, as internal hemorrhage and telangiectasia of the skin. The following year Babington 2 described a family showing a tendency to epistaxis but made no mention of the presence of telangiectasia of the skin or mucous membrane in any members of the family. Rendu,3 in 1896, reported a case of a 52-year-old man who suffered from repeated epistaxis and who had small superficial angiomata of the References 1. References 7 and 8. 2. References 10-12. 3. Sutton, H. G.: Epistaxis as an Indication of Impaired Nutrition and of Degeneration of the Vascular System , Med. Mirror 1:769, 1864. 4. Babington, B. G.: Hereditary Epistaxis , Lancet 2:362, 1865. 5. Rendu, M.: Epistaxis répétées chez un sujet porteur de petits angiomes cutanés et muqueux , Bull. et. mém. Soc. méd. hôp. Paris 13:731, 1896. 6. Osler, W.: On a Family Form of Recurrent Epistaxis, Associated with Multiple Telangiectases of the Skin and Mucous Membrane , Bull. Johns Hopkins Hosp. 12:333, 1901. 7. Weber, F. P.: A Case of Multiple Hereditary Developmental Angiomata (Telangiectases) of the Skin and Mucous Membranes Associated with Recurring Hemorrhages , Lancet 2:160, 1907. 8. Hanes, F. M.: Multiple Hereditary Telangiectases Causing Hemorrhage (Hereditary Hemorrhagic Telangiectasia) , Bull. Johns Hopkins Hosp. 20:63, 1909. 9. Goldstein, H. I.: Goldstein's Heredo-Familial Angiomatosis (Telangiectasia) with Recurring Familial Hemorrhages (Rendu-Osler-Weber Disease) , Arch. Int. Med. 48:836, 1931.Crossref 10. Goldstein, H. I.: Unusual (Benign) Hematemesis: Gastric Telangiectatic Dysplasia; Rendu-Osler-Weber Disease (Goldstein's Hematemesis) , Rev. Gastroenterol. 14:258, 1947. 11. Barrock, J. J.: Hereditary Hemorrhagic Telangiectasia: Report of a Case with Review of the Literature , Wisconsin M. J. 43:805, 1944. 12. Schwartz, S. O., and Armstrong, B. E.: Familial Hemorrhagic Telangiectasia in the Negro , New England J. Med. 239:434, 1948.Crossref 13. Kleinschmidt, W. H., and Schwartz, S. O.: Familial Hereditary Hemorrhagic Telangiectasia in the Negro: Report of a 2nd Case , Illinois M. J. 98:298, 1950. 14. Smith, J. L., and Lineback, M. I.: Hereditary Hemorrhagic Telangiectasia: Nine Cases in One Negro Family, with Special Reference to Hepatic Lesions , Am. J. Med. 17:41, 1954.Crossref 15. Fitz-Hugh, T., Jr.: The importance of Atavism in the Diagnosis of Hereditary Hemorrhagic Telangiectasia , Am. J. M. Sc. 166:884, 1923.Crossref 16. Madden, J. F.: Generalized Angiomatosis (Telangiectasia) , J. A. M. A. 102:442, 1934.Crossref 17. Figi, F. A., and Watkins, C. H.: Hereditary Hemorrhagic Telangiectasia , Ann. Otol. Rhin. & Laryng. 52:330, 1943. 18. Fitz-Hugh, T.: Splenomegaly and Hepatic Enlargement in Hereditary Hemorrhagic Telangiectasia , Am. J. M. Sc. 181:261, 1931. 19. Baker, G. P.: Hereditary Haemorrhagic Telangiectasia with Gastrointestinal Haemorrhage and Hepato-Splenomegaly , Guy's Hosp. Rep. 102: 246, 1953. 20. Singer, K., and Wolfson, W. Q.: Hereditary Hemorrhagic Telangiectasia , New England J. Med 230:637, 1944. 21. Wells, E. B.: Hereditary Hemorrhagic Telangiectasia , Am. J. M. Sc. 211:577, 1946. 22. Plummer, K.; Burke, J. O., and Williams, J P.: Hereditary Hemorrhagic Telangiectasia with Intestinal Bleeding , Gastroenterology 12:988, 1949 23. Koch, H. J.; Escher, G. C., and Lewis, J. S.: Hormonal Management of Hereditary Hemorrhagic Telangiectasia , J. A. M. A. 149:1376, 1952.Crossref 24. Sherber, D. A.: Adrenochrome Complex in the Control of Bleeding , Am. J. Surg. 86:331, 1953Crossref 25. Garland, H. G., and Anning, S. T.: Hereditary Hemorrhagic Telangiectasia: A Genetic and Bibliographic Study , Brit. J. Dermat. 62:289, 1950Crossref 26. Gambill, E. E.: Recurring Gastrointestinal Hemorrhage in Hereditary Hemorrhagic Telangiectasia: Incidence and Report of a Case , Proc. Staff Meet., Mayo Clin. 21:157, 1946. 27. Fox, T. C.: A Case of Bilateral Telangiectases of the Trunk, with a History of Marked Epistaxis in Childhood and Recent Rectal Haemorrhage , Brit. J. Dermat. 20:145, 1908.Crossref 28. Harper, R. A. J.: Multiple Hereditary Telangiectasis with Recurring Hemorrhages , Newcastle M. J. 9:182, 1929. 29. Boston, L. N.: Gastric Hemorrhage Due to Familial Telangiectasis , Am. J. M. Sc. 180:798, 1930.Crossref 30. Hurst, A. F., and Plummer, N. S.: Hereditary Telangiectasia with Haemorrhagic Tendency , Guy's Hosp. Rep. 82:81, 1932. 31. Meikle, G. J.: A Case of Hereditary Telangiectasia , Lancet 2:863, 1933.Crossref 32. Schuster, N. H.: Familial Haemorrhagic Telangiectasia Associated with Multiple Aneurysms of the Splenic Artery , J. Path. & Bact. 44:29, 1937. 33. Renshaw, J. F.: Multiple Hemorrhagic Telangiectasia with Special Reference to Gastroscopic Appearance , Cleveland Clin. Quart. 6:226, 1939. 34. Griggs, D. E., and Baker, M. Q.: Hereditary Hemorrhagic Telangiectasia with Gastrointestinal Bleeding , Am. J. Digest. Dis. 8:344, 1941. 35. Stellar, L. I.: Hereditary Telangiectasis: Report of a Case , New England J. Med. 226:3361942. 36. Wolfsohn, H.: Hereditary Familial Telangiectasis , Lancet 2:581, 1944. 37. Rundles, R. W.: Hemorrhagic Telangiectasia with Pulmonary Artery Aneurysms: Case Report . Am. J. M. Sc. 210:76, 1945. 38. Kushlan, S. D.: Gastrointestinal Bleeding in Hereditary Hemorrhagic Telangiectasia , Gastroenterology 7:199, 1946. 39. Bell, G. I.: Hereditary Hemorrhagic Telangiectasia , Canad. M. A. J. 58:279, 1948. 40. Rumball, J. M.: Multiple Hemorrhagic Telangiectasia (Rendu-Osler-Weber Disease): Case with Visceral Involvement , Mil. Surgeon 105:53, 1949. 41. Goldman, R.; Asher, L., and Ware, E. R. R.: Hereditary Hemorrhagic Telangiectasis , Gastroenterology 12:495, 1949. 42. Brandel, E.: Recurrent Gastrointestinal Hemorrhage in Hereditary Hemorrhagic Telangiectasia (Osler) , Acta med. scandinav. 137:436. 1950. 43. Doenges, J. P.: Treatment of Hereditary Hemorrhagic Telangiectasia with Rutin: A Case Report , Bull. School Med. Univ. Maryland 38:142, 1953. 44. Dolowitz, D. A.: Hereditary Hemorrhagic Telangiectasia , Am. Surgeon 19:776, 1953. 45. Shepherd, J. A.: Angiomatous Conditions of the Gastrointestinal Tract , Brit. J. Surg. 40:409, 1953.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png A.M.A. Archives of Internal Medicine American Medical Association

GASTROINTESTINAL BLEEDING IN HEREDITARY HEMORRHAGIC TELANGIECTASIA: Review of the Literature and Report of a Case with Severe Recurrent Hemorrhages Necessitating Total Gastrectomy

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American Medical Association
Copyright
Copyright © 1955 American Medical Association. All Rights Reserved.
ISSN
0888-2479
DOI
10.1001/archinte.1955.00250070057007
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Abstract

Abstract HEREDITARY hemorrhagic telangiectasia (Rendu-Osler-Weber disease) is a hereditary vascular anomaly characterized pathologically by multiple dilatations of capillaries and venules of the skin and mucous membranes and characterized clinically by recurrent hemorrhage from these telangiectasia, most frequently manifested by epistaxis. The entity is often overlooked or the bleeding attributed to some other cause, especially a blood dyscrasia. In gastrointestinal bleeding, peptic ulcer is often suspected, and, indeed, the two conditions may be present concurrently. The condition was probably first reported by Sutton,1 in 1864, as internal hemorrhage and telangiectasia of the skin. The following year Babington 2 described a family showing a tendency to epistaxis but made no mention of the presence of telangiectasia of the skin or mucous membrane in any members of the family. Rendu,3 in 1896, reported a case of a 52-year-old man who suffered from repeated epistaxis and who had small superficial angiomata of the References 1. References 7 and 8. 2. References 10-12. 3. Sutton, H. G.: Epistaxis as an Indication of Impaired Nutrition and of Degeneration of the Vascular System , Med. Mirror 1:769, 1864. 4. Babington, B. G.: Hereditary Epistaxis , Lancet 2:362, 1865. 5. Rendu, M.: Epistaxis répétées chez un sujet porteur de petits angiomes cutanés et muqueux , Bull. et. mém. Soc. méd. hôp. Paris 13:731, 1896. 6. Osler, W.: On a Family Form of Recurrent Epistaxis, Associated with Multiple Telangiectases of the Skin and Mucous Membrane , Bull. Johns Hopkins Hosp. 12:333, 1901. 7. Weber, F. P.: A Case of Multiple Hereditary Developmental Angiomata (Telangiectases) of the Skin and Mucous Membranes Associated with Recurring Hemorrhages , Lancet 2:160, 1907. 8. Hanes, F. M.: Multiple Hereditary Telangiectases Causing Hemorrhage (Hereditary Hemorrhagic Telangiectasia) , Bull. Johns Hopkins Hosp. 20:63, 1909. 9. Goldstein, H. I.: Goldstein's Heredo-Familial Angiomatosis (Telangiectasia) with Recurring Familial Hemorrhages (Rendu-Osler-Weber Disease) , Arch. Int. Med. 48:836, 1931.Crossref 10. Goldstein, H. I.: Unusual (Benign) Hematemesis: Gastric Telangiectatic Dysplasia; Rendu-Osler-Weber Disease (Goldstein's Hematemesis) , Rev. Gastroenterol. 14:258, 1947. 11. Barrock, J. J.: Hereditary Hemorrhagic Telangiectasia: Report of a Case with Review of the Literature , Wisconsin M. J. 43:805, 1944. 12. Schwartz, S. O., and Armstrong, B. E.: Familial Hemorrhagic Telangiectasia in the Negro , New England J. Med. 239:434, 1948.Crossref 13. Kleinschmidt, W. H., and Schwartz, S. O.: Familial Hereditary Hemorrhagic Telangiectasia in the Negro: Report of a 2nd Case , Illinois M. J. 98:298, 1950. 14. Smith, J. L., and Lineback, M. I.: Hereditary Hemorrhagic Telangiectasia: Nine Cases in One Negro Family, with Special Reference to Hepatic Lesions , Am. J. Med. 17:41, 1954.Crossref 15. Fitz-Hugh, T., Jr.: The importance of Atavism in the Diagnosis of Hereditary Hemorrhagic Telangiectasia , Am. J. M. Sc. 166:884, 1923.Crossref 16. Madden, J. F.: Generalized Angiomatosis (Telangiectasia) , J. A. M. A. 102:442, 1934.Crossref 17. Figi, F. A., and Watkins, C. H.: Hereditary Hemorrhagic Telangiectasia , Ann. Otol. Rhin. & Laryng. 52:330, 1943. 18. Fitz-Hugh, T.: Splenomegaly and Hepatic Enlargement in Hereditary Hemorrhagic Telangiectasia , Am. J. M. Sc. 181:261, 1931. 19. Baker, G. P.: Hereditary Haemorrhagic Telangiectasia with Gastrointestinal Haemorrhage and Hepato-Splenomegaly , Guy's Hosp. Rep. 102: 246, 1953. 20. Singer, K., and Wolfson, W. Q.: Hereditary Hemorrhagic Telangiectasia , New England J. Med 230:637, 1944. 21. Wells, E. B.: Hereditary Hemorrhagic Telangiectasia , Am. J. M. Sc. 211:577, 1946. 22. Plummer, K.; Burke, J. O., and Williams, J P.: Hereditary Hemorrhagic Telangiectasia with Intestinal Bleeding , Gastroenterology 12:988, 1949 23. Koch, H. J.; Escher, G. C., and Lewis, J. S.: Hormonal Management of Hereditary Hemorrhagic Telangiectasia , J. A. M. A. 149:1376, 1952.Crossref 24. Sherber, D. A.: Adrenochrome Complex in the Control of Bleeding , Am. J. Surg. 86:331, 1953Crossref 25. Garland, H. G., and Anning, S. T.: Hereditary Hemorrhagic Telangiectasia: A Genetic and Bibliographic Study , Brit. J. Dermat. 62:289, 1950Crossref 26. Gambill, E. E.: Recurring Gastrointestinal Hemorrhage in Hereditary Hemorrhagic Telangiectasia: Incidence and Report of a Case , Proc. Staff Meet., Mayo Clin. 21:157, 1946. 27. Fox, T. C.: A Case of Bilateral Telangiectases of the Trunk, with a History of Marked Epistaxis in Childhood and Recent Rectal Haemorrhage , Brit. J. Dermat. 20:145, 1908.Crossref 28. Harper, R. A. J.: Multiple Hereditary Telangiectasis with Recurring Hemorrhages , Newcastle M. J. 9:182, 1929. 29. Boston, L. N.: Gastric Hemorrhage Due to Familial Telangiectasis , Am. J. M. Sc. 180:798, 1930.Crossref 30. Hurst, A. F., and Plummer, N. S.: Hereditary Telangiectasia with Haemorrhagic Tendency , Guy's Hosp. Rep. 82:81, 1932. 31. Meikle, G. J.: A Case of Hereditary Telangiectasia , Lancet 2:863, 1933.Crossref 32. Schuster, N. H.: Familial Haemorrhagic Telangiectasia Associated with Multiple Aneurysms of the Splenic Artery , J. Path. & Bact. 44:29, 1937. 33. Renshaw, J. F.: Multiple Hemorrhagic Telangiectasia with Special Reference to Gastroscopic Appearance , Cleveland Clin. Quart. 6:226, 1939. 34. Griggs, D. E., and Baker, M. Q.: Hereditary Hemorrhagic Telangiectasia with Gastrointestinal Bleeding , Am. J. Digest. Dis. 8:344, 1941. 35. Stellar, L. I.: Hereditary Telangiectasis: Report of a Case , New England J. Med. 226:3361942. 36. Wolfsohn, H.: Hereditary Familial Telangiectasis , Lancet 2:581, 1944. 37. Rundles, R. W.: Hemorrhagic Telangiectasia with Pulmonary Artery Aneurysms: Case Report . Am. J. M. Sc. 210:76, 1945. 38. Kushlan, S. D.: Gastrointestinal Bleeding in Hereditary Hemorrhagic Telangiectasia , Gastroenterology 7:199, 1946. 39. Bell, G. I.: Hereditary Hemorrhagic Telangiectasia , Canad. M. A. J. 58:279, 1948. 40. Rumball, J. M.: Multiple Hemorrhagic Telangiectasia (Rendu-Osler-Weber Disease): Case with Visceral Involvement , Mil. Surgeon 105:53, 1949. 41. Goldman, R.; Asher, L., and Ware, E. R. R.: Hereditary Hemorrhagic Telangiectasis , Gastroenterology 12:495, 1949. 42. Brandel, E.: Recurrent Gastrointestinal Hemorrhage in Hereditary Hemorrhagic Telangiectasia (Osler) , Acta med. scandinav. 137:436. 1950. 43. Doenges, J. P.: Treatment of Hereditary Hemorrhagic Telangiectasia with Rutin: A Case Report , Bull. School Med. Univ. Maryland 38:142, 1953. 44. Dolowitz, D. A.: Hereditary Hemorrhagic Telangiectasia , Am. Surgeon 19:776, 1953. 45. Shepherd, J. A.: Angiomatous Conditions of the Gastrointestinal Tract , Brit. J. Surg. 40:409, 1953.Crossref

Journal

A.M.A. Archives of Internal MedicineAmerican Medical Association

Published: Jan 1, 1955

References