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DEFECTS OF MEMBRANOUS BONES, EXOPHTHALMOS AND DIABETES INSIPIDUS: REPORT OF A CASE WITH NECROPSY

DEFECTS OF MEMBRANOUS BONES, EXOPHTHALMOS AND DIABETES INSIPIDUS: REPORT OF A CASE WITH NECROPSY Abstract In 1919, Christian,1 under the title, "Defects in Membranous Bones, Exophthalmos and Diabetes Insipidus, an Unusual Syndrome of Dyspituitarism," reported a case presenting this syndrome and cited two similar cases previously described by Schüller.2 Three other cases have since been reported by Hand3 and one by Grosh and Stifel,4 making seven cases in all reported to date. The following case was deemed worthy of a detailed report because of the unusually widespread osseous dissolution present, because of the rarity of the condition, and finally to record a complete postmortem study of this strange disease complex. REPORT OF CASE History. —C. J., a boy, aged nine years, came to us, Feb. 15, 1923, seeking relief from excessive thirst and frequent urination.The father, mother, one sister and one brother were living and well. The mother had had no miscarriages. One brother died when five months old of cholera infantum.The patient References 1. Christian, H. A.: Contributions to Medical and Biological Research (Osler Festschrift) , New York, Paul B. Hoeber, 1919, 1:390-401. 2. Schüller, Artur: Ueber eigenartige Schadeldefekte in Jügendalter-Fortschritte der Roentgenstrahlen , 1915-1916, 23:12. 3. Hand, Alfred: Defects of Membranous Bones, Exophthalmos and Polyuria in Childhood—Is It Dyspituitarism? Am. J. M. Sc. 162:509 ( (Oct.) ) 1921.Crossref 4. Grosh, L. D., and Stifel, J. L.: Defects in Membranous Bones, Diabetes Insipidus and Exophthalmos with Report of a Case , Arch. Int. Med. 31:76 ( (Jan.) ) 1923.Crossref 5. McCrudden, F. H.: J. Biol Chem. 7:83, 1910 6. Kramer, B., and Tisdall, F. T.: Bull. Johns Hopkins Hosp. 32:44 ( (Feb.) ) 1921. 7. It would seem that for the sake of clarity Schüller's Case 1 should not be included in this syndrome. This patient did not have diabetes insipidus but dystrophia adiposo genitalis; the defect in the cranial bones was largely confined to one side (the left). Schüller's assumption was a tumor at the base of the brain ("other changes suggested pressure"), either an angioma of the dura or of the skull. 8. Rowntree, L. G.: Oxford Med. 4:185, 1921. 9. Motzfeldt, K.: Endocrinology 2:94, 1918.Crossref 10. Engelbach, W., and Tierney, J. L.: Pituitary Polyuria , Internat. Clinics 4:123, 1920. 11. Sajous, C. E. D.: Am. J. M. Sc. 167:679-696 ( (May) ) 1924.Crossref 12. Camus, J., and Roussy, G.: Compt. rend. Soc. de biol. 75:483, 1913. 13. Camus, J., and Roussy, G.: Endocrinology 4:507 ( (Oct.) -Dec.) 1920.Crossref 14. Bailey, Percival; and Bremer, Frederic: Experimental Diabetes Insipidus , Arch. Int. Med. 28:773 ( (Dec.) ) 1921.Crossref 15. Hancheet, M.: Experimental Polyuria , Am. J. M. Sc. 163:685 ( (May) ) 1922.Crossref 16. Curtis, G. M.: The Production of Experimental Diabetes Insipidus . Arch. Int. Med. 34:801 ( (Dec.) ) 1924.Crossref 17. In Hand's Case 3, which was incompletely studied, there was no polyuria, but the patient did have characteristic defects in the membranous bones. 18. While the testes were examined at necropsy and were pronounced grossly normal, they were inadvertently not included in the material saved for histologic studies. 19. It was suggested that the lung condition by causing tissue asphyxia might have induced calcium absorption and have thus played a part in the bone destruction. One of us (A. D. D.) has seen three cases at necropsy with fully as marked lung findings of a similar character in which there were no skeletal changes. 20. Berkheiser, E. J.: Multiple Myelomas in Children , Arch. Surg. 8:853881 ( (May) ) 1924.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Internal Medicine American Medical Association

DEFECTS OF MEMBRANOUS BONES, EXOPHTHALMOS AND DIABETES INSIPIDUS: REPORT OF A CASE WITH NECROPSY

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Publisher
American Medical Association
Copyright
Copyright © 1925 American Medical Association. All Rights Reserved.
ISSN
0730-188X
DOI
10.1001/archinte.1925.00120170059005
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Abstract

Abstract In 1919, Christian,1 under the title, "Defects in Membranous Bones, Exophthalmos and Diabetes Insipidus, an Unusual Syndrome of Dyspituitarism," reported a case presenting this syndrome and cited two similar cases previously described by Schüller.2 Three other cases have since been reported by Hand3 and one by Grosh and Stifel,4 making seven cases in all reported to date. The following case was deemed worthy of a detailed report because of the unusually widespread osseous dissolution present, because of the rarity of the condition, and finally to record a complete postmortem study of this strange disease complex. REPORT OF CASE History. —C. J., a boy, aged nine years, came to us, Feb. 15, 1923, seeking relief from excessive thirst and frequent urination.The father, mother, one sister and one brother were living and well. The mother had had no miscarriages. One brother died when five months old of cholera infantum.The patient References 1. Christian, H. A.: Contributions to Medical and Biological Research (Osler Festschrift) , New York, Paul B. Hoeber, 1919, 1:390-401. 2. Schüller, Artur: Ueber eigenartige Schadeldefekte in Jügendalter-Fortschritte der Roentgenstrahlen , 1915-1916, 23:12. 3. Hand, Alfred: Defects of Membranous Bones, Exophthalmos and Polyuria in Childhood—Is It Dyspituitarism? Am. J. M. Sc. 162:509 ( (Oct.) ) 1921.Crossref 4. Grosh, L. D., and Stifel, J. L.: Defects in Membranous Bones, Diabetes Insipidus and Exophthalmos with Report of a Case , Arch. Int. Med. 31:76 ( (Jan.) ) 1923.Crossref 5. McCrudden, F. H.: J. Biol Chem. 7:83, 1910 6. Kramer, B., and Tisdall, F. T.: Bull. Johns Hopkins Hosp. 32:44 ( (Feb.) ) 1921. 7. It would seem that for the sake of clarity Schüller's Case 1 should not be included in this syndrome. This patient did not have diabetes insipidus but dystrophia adiposo genitalis; the defect in the cranial bones was largely confined to one side (the left). Schüller's assumption was a tumor at the base of the brain ("other changes suggested pressure"), either an angioma of the dura or of the skull. 8. Rowntree, L. G.: Oxford Med. 4:185, 1921. 9. Motzfeldt, K.: Endocrinology 2:94, 1918.Crossref 10. Engelbach, W., and Tierney, J. L.: Pituitary Polyuria , Internat. Clinics 4:123, 1920. 11. Sajous, C. E. D.: Am. J. M. Sc. 167:679-696 ( (May) ) 1924.Crossref 12. Camus, J., and Roussy, G.: Compt. rend. Soc. de biol. 75:483, 1913. 13. Camus, J., and Roussy, G.: Endocrinology 4:507 ( (Oct.) -Dec.) 1920.Crossref 14. Bailey, Percival; and Bremer, Frederic: Experimental Diabetes Insipidus , Arch. Int. Med. 28:773 ( (Dec.) ) 1921.Crossref 15. Hancheet, M.: Experimental Polyuria , Am. J. M. Sc. 163:685 ( (May) ) 1922.Crossref 16. Curtis, G. M.: The Production of Experimental Diabetes Insipidus . Arch. Int. Med. 34:801 ( (Dec.) ) 1924.Crossref 17. In Hand's Case 3, which was incompletely studied, there was no polyuria, but the patient did have characteristic defects in the membranous bones. 18. While the testes were examined at necropsy and were pronounced grossly normal, they were inadvertently not included in the material saved for histologic studies. 19. It was suggested that the lung condition by causing tissue asphyxia might have induced calcium absorption and have thus played a part in the bone destruction. One of us (A. D. D.) has seen three cases at necropsy with fully as marked lung findings of a similar character in which there were no skeletal changes. 20. Berkheiser, E. J.: Multiple Myelomas in Children , Arch. Surg. 8:853881 ( (May) ) 1924.Crossref

Journal

Archives of Internal MedicineAmerican Medical Association

Published: Nov 1, 1925

References