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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
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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 Children – American Medical Association

Published: Aug 1, 1956

References

  • The Postnatal Structural Changes in the Intrapulmonary Arteries and Arterioles
    Civin , W. H., and Edwards, J. E.
  • The Nature and Significance of the Structural Changes in the Lungs in Mitral Stenosis
    Parker , F., and Weiss, W.
  • Acute Diffuse Interstitial Fibrosis of the Lungs
    Hamman , L., and Rich, A. R.
  • Pathologic Anatomy of "Atypical Pneumonia, Etiology Undertermined,"
    Golden , A.
  • Pathologic Aspects of Atypical Pneumonias (Interstitial Pneumonitis)
    Anderson , W. A. D.
  • Generalized Cytomegalic Inclusion Disease in a New-Born Infant
    Alexander , W. S.
  • Report of a Case of Acute Interstitial Fibrosis of the Lungs
    Eder , H.; Hawn, C., and Thorn, G.
  • Diffuse Progressive Interstitial Fibrosis of the Lungs
    Beams , A. J., and Harmos, O.
  • Patterns of Pulmonary Fibrosis as Related to Pulmonary Function
    Spain , D. M.
  • Idiopathic Pulmonary Fibrosis: Its Occurrence in Identical Twin Sisters
    Peabody , J. W.; Peabody, J. W., Jr.; Hayes, E. W., and Hayes, E. W., Jr.
  • Diffuse Interstitial Fibrosis of the Lungs (Report of a Case with Unusual Features)
    Katz , H. L., and Auerbach, O.
  • Chronic Diffuse Interstitial Fibrosis of the Lungs
    Heppleston , A. G.
  • Diffuse Interstitial Fibrosis of the Lungs
    Rubin , E. H.; Kahn, B. S., and Pecker, D.
  • Acute Diffuse Interstitial Fibrosis of the Lungs
    Callahan , W. P., Jr.; Sutherland, J. C.; Fulton, J. K., and Kline, J. R.
  • Histological and Serological Sequences in Experimental Hypersensitivity
    Hawn , C. V., and Janeway, C. A.
  • Granuloma, a Characteristic "Qualitative" Change in Focal Anaphylactic Inflammation
    Goddard , J. W.
  • Studies on the Site of Sensitivity in the Arthus Phenomenon
    Rich , A. R., and Follis, R. H.
  • Vascular Pathology in Rabbits Following Administration of Foreign Protein
    Fox , R. A., and Jones, L. R.
  • Die Eiweissüberempfindlichkeit (Gewebshyperergie) des Bindegewebes: Experimentelle Untersuchungen zur Erzeugung des rheumatischen Gewebsschadens im Herzen und in den Gelenken
    Vaubel , E.
  • Hypersensitivity to Iodine as Cause of Polyarteritis Nodosa
    Rich , A. R.
  • Experimental Production of Generalised Arteritis and Periarteritis (Periarteritis Nodosa)
    Hopps , H. C., and Wissler, R. W.
  • 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
    More , R. H., and Kobernick, S. D.
  • Pathology of Pulmonary Heart Disease
    McKeown , F.