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Primary Pulmonary Vascular Disease with Cor Pulmonale: Report of Three Cases in Children, One with Congenital Hypertension and Two Siblings with Allergic Vasculitis and Disorders of Skeletal Epiphyses

Primary Pulmonary Vascular Disease with Cor Pulmonale: Report of Three Cases in Children, One... Abstract In the first case here described there was holdover of fetal pulmonary arterial structure with hypertension and cor pulmonale. There were also distinctive segmental arterial lesions and foci of interstitial pneumonia. Arterial lesions outside the lungs were not found. The other two cases were brothers who died of cor pulmonale due to allergic vasculitis confined to the lungs. In addition, one had Perthes' disease and the other dysplasia epiphysalis multiplex. The first case is presented because cor pulmonale was primarily due to congenital hypertension and because there were distinctive lesions of intrapulmonary arteries. In such lesions capillaries had developed segmentally in arterial walls without apparent other damage, since smooth muscle of the media between capillaries was largely intact. Additional features of this first case were livedo reticularis, syndactyly, fibrosis around hair follicles, and congenital deformity of intramyocardial arteries. The other two cases are recorded because allergic vasculitis limited to pulmonary References 1. Civin, W. H., and Edwards, J. E.: The Postnatal Structural Changes in the Intrapulmonary Arteries and Arterioles , A. M. A. Arch. Path. 51:192, 1951. 2. Parker, F., and Weiss, W.: The Nature and Significance of the Structural Changes in the Lungs in Mitral Stenosis , Am. J. Path. 12: 573, 1936. 3. Hamman, L., and Rich, A. R.: Acute Diffuse Interstitial Fibrosis of the Lungs , Bull. Johns Hopkins Hosp. 74:177, 1944. 4. Golden, A.: Pathologic Anatomy of "Atypical Pneumonia, Etiology Undertermined," Arch. Path. 38:187, 1944. 5. Anderson, W. A. D.: Pathologic Aspects of Atypical Pneumonias (Interstitial Pneumonitis) , Dis. Chest. 15:337, 1949.Crossref 6. Alexander, W. S.: Generalized Cytomegalic Inclusion Disease in a New-Born Infant , J. Path. & Bact. 65:375, 1953. 7. Eder, H.; Hawn, C., and Thorn, G.: Report of a Case of Acute Interstitial Fibrosis of the Lungs , Bull. Johns Hopkins Hosp. 76:163, 1945. 8. Beams, A. J., and Harmos, O.: Diffuse Progressive Interstitial Fibrosis of the Lungs , Am. J. Med. 7:425, 1949. 9. Spain, D. M.: Patterns of Pulmonary Fibrosis as Related to Pulmonary Function , Ann. Int. Med. 33:1150, 1950. 10. Peabody, J. W.; Peabody, J. W., Jr.; Hayes, E. W., and Hayes, E. W., Jr.: Idiopathic Pulmonary Fibrosis: Its Occurrence in Identical Twin Sisters , Dis. Chest. 18:330, 1950. 11. Katz, H. L., and Auerbach, O.: Diffuse Interstitial Fibrosis of the Lungs (Report of a Case with Unusual Features) , Dis. Chest. 20:366, 1951. 12. Heppleston, A. G.: Chronic Diffuse Interstitial Fibrosis of the Lungs , Thorax 6:426, 1951. 13. Rubin, E. H.; Kahn, B. S., and Pecker, D.: Diffuse Interstitial Fibrosis of the Lungs , Ann. Int. Med. 36:827, 1952. 14. Callahan, W. P., Jr.; Sutherland, J. C.; Fulton, J. K., and Kline, J. R.: Acute Diffuse Interstitial Fibrosis of the Lungs , Arch. Int. Med. 90:468, 1952. 15. Hawn, C. V., and Janeway, C. A.: Histological and Serological Sequences in Experimental Hypersensitivity , J. Exper. Med. 85:571, 1947. 16. Goddard, J. W.: Granuloma, a Characteristic "Qualitative" Change in Focal Anaphylactic Inflammation , Am. J. Path. 23:943, 1947. 17. Rich, A. R., and Follis, R. H.: Studies on the Site of Sensitivity in the Arthus Phenomenon , Bull. Johns Hopkins Hosp. 66:106, 1940. 18. Fox, R. A., and Jones, L. R.: Vascular Pathology in Rabbits Following Administration of Foreign Protein , Proc. Soc. Exper. Biol. & Med. 55:294, 1944. 19. Vaubel, E.: Die Eiweissüberempfindlichkeit (Gewebshyperergie) des Bindegewebes: Experimentelle Untersuchungen zur Erzeugung des rheumatischen Gewebsschadens im Herzen und in den Gelenken , Beitr. path. Anat. 89:374, 1932. 20. Rich, A. R.: Hypersensitivity to Iodine as Cause of Polyarteritis Nodosa , Bull. Johns Hopkins Hosp. 77:43, 1945. 21. Hopps, H. C., and Wissler, R. W.: Experimental Production of Generalised Arteritis and Periarteritis (Periarteritis Nodosa) , J. Lab. & Clin. Med. 31:939, 1946. 22. More, R. H., and Kobernick, S. D.: Arteritis, Carditis, Glomerulonephritis and Bilateral Renal Cortical Necrosis Induced in Rabbits by Injection of Horse Serum or Bovine Gamma Globulin Combined with Killed Group A Streptococci or Freund's Adjuvant , A.M.A. Arch. Path. 51:361, 1951. 23. McKeown, F.: Pathology of Pulmonary Heart Disease , Brit. Heart J. 14:25, 1952. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png A.M.A. Journal of Diseases of Children American Medical Association

Primary Pulmonary Vascular Disease with Cor Pulmonale: Report of Three Cases in Children, One with Congenital Hypertension and Two Siblings with Allergic Vasculitis and Disorders of Skeletal Epiphyses

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Publisher
American Medical Association
Copyright
Copyright © 1956 American Medical Association. All Rights Reserved.
ISSN
0096-6916
DOI
10.1001/archpedi.1956.02060030109002
Publisher site
See Article on Publisher Site

Abstract

Abstract In the first case here described there was holdover of fetal pulmonary arterial structure with hypertension and cor pulmonale. There were also distinctive segmental arterial lesions and foci of interstitial pneumonia. Arterial lesions outside the lungs were not found. The other two cases were brothers who died of cor pulmonale due to allergic vasculitis confined to the lungs. In addition, one had Perthes' disease and the other dysplasia epiphysalis multiplex. The first case is presented because cor pulmonale was primarily due to congenital hypertension and because there were distinctive lesions of intrapulmonary arteries. In such lesions capillaries had developed segmentally in arterial walls without apparent other damage, since smooth muscle of the media between capillaries was largely intact. Additional features of this first case were livedo reticularis, syndactyly, fibrosis around hair follicles, and congenital deformity of intramyocardial arteries. The other two cases are recorded because allergic vasculitis limited to pulmonary References 1. Civin, W. H., and Edwards, J. E.: The Postnatal Structural Changes in the Intrapulmonary Arteries and Arterioles , A. M. A. Arch. Path. 51:192, 1951. 2. Parker, F., and Weiss, W.: The Nature and Significance of the Structural Changes in the Lungs in Mitral Stenosis , Am. J. Path. 12: 573, 1936. 3. Hamman, L., and Rich, A. R.: Acute Diffuse Interstitial Fibrosis of the Lungs , Bull. Johns Hopkins Hosp. 74:177, 1944. 4. Golden, A.: Pathologic Anatomy of "Atypical Pneumonia, Etiology Undertermined," Arch. Path. 38:187, 1944. 5. Anderson, W. A. D.: Pathologic Aspects of Atypical Pneumonias (Interstitial Pneumonitis) , Dis. Chest. 15:337, 1949.Crossref 6. Alexander, W. S.: Generalized Cytomegalic Inclusion Disease in a New-Born Infant , J. Path. & Bact. 65:375, 1953. 7. Eder, H.; Hawn, C., and Thorn, G.: Report of a Case of Acute Interstitial Fibrosis of the Lungs , Bull. Johns Hopkins Hosp. 76:163, 1945. 8. Beams, A. J., and Harmos, O.: Diffuse Progressive Interstitial Fibrosis of the Lungs , Am. J. Med. 7:425, 1949. 9. Spain, D. M.: Patterns of Pulmonary Fibrosis as Related to Pulmonary Function , Ann. Int. Med. 33:1150, 1950. 10. Peabody, J. W.; Peabody, J. W., Jr.; Hayes, E. W., and Hayes, E. W., Jr.: Idiopathic Pulmonary Fibrosis: Its Occurrence in Identical Twin Sisters , Dis. Chest. 18:330, 1950. 11. Katz, H. L., and Auerbach, O.: Diffuse Interstitial Fibrosis of the Lungs (Report of a Case with Unusual Features) , Dis. Chest. 20:366, 1951. 12. Heppleston, A. G.: Chronic Diffuse Interstitial Fibrosis of the Lungs , Thorax 6:426, 1951. 13. Rubin, E. H.; Kahn, B. S., and Pecker, D.: Diffuse Interstitial Fibrosis of the Lungs , Ann. Int. Med. 36:827, 1952. 14. Callahan, W. P., Jr.; Sutherland, J. C.; Fulton, J. K., and Kline, J. R.: Acute Diffuse Interstitial Fibrosis of the Lungs , Arch. Int. Med. 90:468, 1952. 15. Hawn, C. V., and Janeway, C. A.: Histological and Serological Sequences in Experimental Hypersensitivity , J. Exper. Med. 85:571, 1947. 16. Goddard, J. W.: Granuloma, a Characteristic "Qualitative" Change in Focal Anaphylactic Inflammation , Am. J. Path. 23:943, 1947. 17. Rich, A. R., and Follis, R. H.: Studies on the Site of Sensitivity in the Arthus Phenomenon , Bull. Johns Hopkins Hosp. 66:106, 1940. 18. Fox, R. A., and Jones, L. R.: Vascular Pathology in Rabbits Following Administration of Foreign Protein , Proc. Soc. Exper. Biol. & Med. 55:294, 1944. 19. Vaubel, E.: Die Eiweissüberempfindlichkeit (Gewebshyperergie) des Bindegewebes: Experimentelle Untersuchungen zur Erzeugung des rheumatischen Gewebsschadens im Herzen und in den Gelenken , Beitr. path. Anat. 89:374, 1932. 20. Rich, A. R.: Hypersensitivity to Iodine as Cause of Polyarteritis Nodosa , Bull. Johns Hopkins Hosp. 77:43, 1945. 21. Hopps, H. C., and Wissler, R. W.: Experimental Production of Generalised Arteritis and Periarteritis (Periarteritis Nodosa) , J. Lab. & Clin. Med. 31:939, 1946. 22. More, R. H., and Kobernick, S. D.: Arteritis, Carditis, Glomerulonephritis and Bilateral Renal Cortical Necrosis Induced in Rabbits by Injection of Horse Serum or Bovine Gamma Globulin Combined with Killed Group A Streptococci or Freund's Adjuvant , A.M.A. Arch. Path. 51:361, 1951. 23. McKeown, F.: Pathology of Pulmonary Heart Disease , Brit. Heart J. 14:25, 1952.

Journal

A.M.A. Journal of Diseases of ChildrenAmerican Medical Association

Published: Aug 1, 1956

References