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Pancytopenia in Mannosidosis

Pancytopenia in Mannosidosis Abstract • A case of autoimmune pancytopenia is described in a patient with mannosidosis who developed anti-platelet and anti-neutrophil antibodies and a low haptoglobin level. Bone marrow biopsy and111InCl3 bone marrow scanning demonstrated hypoplasia of the marrow with a paucity of "storage cells." Pathogenic mechanisms and implications for other inborn errors of metabolism are discussed. (Arch Intern Med 1983;143:1266-1268) References 1. Vidggoff J, Lovrien EW, Beals RK, et al: Mannosidosis in three brothers: A review of the literature. Medicine 1977;56:335-348.Crossref 2. Kistler JP, Lott IT, Kolodny EH, et al: Mannosidosis. Arch Neurol 1977;34:45-51.Crossref 3. Dickersin GR, Lott IT, Kolodny EH, et al: A light and electron microscopic study of mannosidosis. Hum Pathol 1980;11:245-256.Crossref 4. Harmon DC, Weitzman SA, Stossel T: A staphylococcal slide test for detection of antineutrophil antibodies. Blood 1980;56:64-69. 5. McMillan R, Tani P, Mason D: A method that allows shipment of whole blood for the assay of platelet-associated IgG. Blood 1979;54:1201-1202. 6. Gilbert EH, Earle JD, Goris ML, et al: The accuracy of 111InC13 as a bone marrow scanning agent. Radiology 1976;119:167-168.Crossref 7. Matoth Y, Fried K: Chronic Gaucher's disease. Isr J Med Sci 1965;1: 521-530. 8. Heaton WAL, Jacobs P: Haematological problems associated with Gaucher's disease. S Afr Med J 1974;48:1300-1302. 9. Green D, Battifora HA, Smith RT, et al: Thrombocytopenia in Gaucher's disease. Ann Intern Med 1971;74:727-731.Crossref 10. Long RG, Lake BD, Pettit JE, et al: Adult Niemann-Pick disease. Am J Med 1977;62:627-635.Crossref 11. McCabe ER, Melvin TR, O'Brien D, et al: Neutropenia in a patient with type IB glycogen storage disease: In vitro response to lithium chloride. J Pediatr 1980;97:944-946.Crossref 12. Beaudet AL, Anderson DC, Michels VV, et al: Neutropenia and impaired neutrophil migration in type IB glycogen storage disease. J Pediatr 1980;97:906-910.Crossref 13. Narisawa K, Tada K, Kuzuya T: Neutropenia in type IB glycogen storage disease. J Pediatr 1981;99:334-335.Crossref 14. Kelleher JF, Yudkoff M, Hutchinson R, et al: The pancytopenia of isovaleric acidemia. Pediatrics 1980;65:1023-1026. 15. Soriano RJ, Taitz LS, Finberg L, et al: Hyperglycinemia with ketoacidosis and leukopenia. Pediatrics 1967;39:818-821. 16. Inoue S, Krieger I, Sarniak A, et al: Inhibition of bone marrow stem cell growth in vitro by methymalonic acid: A mechanism for pancytopenia in a patient with methylmalonic acidemia. Pediatr Res 1981;15:95-98.Crossref 17. Hakozaki H, Takahashi K, Naito M, et al: Gaucher-like cells in juvenile GM1 gangliosidosis and β-thalassemia. Acta Pathol Jpn 1979;29: 303-318. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Internal Medicine American Medical Association

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Publisher
American Medical Association
Copyright
Copyright © 1983 American Medical Association. All Rights Reserved.
ISSN
0003-9926
eISSN
1538-3679
DOI
10.1001/archinte.1983.00350060198036
Publisher site
See Article on Publisher Site

Abstract

Abstract • A case of autoimmune pancytopenia is described in a patient with mannosidosis who developed anti-platelet and anti-neutrophil antibodies and a low haptoglobin level. Bone marrow biopsy and111InCl3 bone marrow scanning demonstrated hypoplasia of the marrow with a paucity of "storage cells." Pathogenic mechanisms and implications for other inborn errors of metabolism are discussed. (Arch Intern Med 1983;143:1266-1268) References 1. Vidggoff J, Lovrien EW, Beals RK, et al: Mannosidosis in three brothers: A review of the literature. Medicine 1977;56:335-348.Crossref 2. Kistler JP, Lott IT, Kolodny EH, et al: Mannosidosis. Arch Neurol 1977;34:45-51.Crossref 3. Dickersin GR, Lott IT, Kolodny EH, et al: A light and electron microscopic study of mannosidosis. Hum Pathol 1980;11:245-256.Crossref 4. Harmon DC, Weitzman SA, Stossel T: A staphylococcal slide test for detection of antineutrophil antibodies. Blood 1980;56:64-69. 5. McMillan R, Tani P, Mason D: A method that allows shipment of whole blood for the assay of platelet-associated IgG. Blood 1979;54:1201-1202. 6. Gilbert EH, Earle JD, Goris ML, et al: The accuracy of 111InC13 as a bone marrow scanning agent. Radiology 1976;119:167-168.Crossref 7. Matoth Y, Fried K: Chronic Gaucher's disease. Isr J Med Sci 1965;1: 521-530. 8. Heaton WAL, Jacobs P: Haematological problems associated with Gaucher's disease. S Afr Med J 1974;48:1300-1302. 9. Green D, Battifora HA, Smith RT, et al: Thrombocytopenia in Gaucher's disease. Ann Intern Med 1971;74:727-731.Crossref 10. Long RG, Lake BD, Pettit JE, et al: Adult Niemann-Pick disease. Am J Med 1977;62:627-635.Crossref 11. McCabe ER, Melvin TR, O'Brien D, et al: Neutropenia in a patient with type IB glycogen storage disease: In vitro response to lithium chloride. J Pediatr 1980;97:944-946.Crossref 12. Beaudet AL, Anderson DC, Michels VV, et al: Neutropenia and impaired neutrophil migration in type IB glycogen storage disease. J Pediatr 1980;97:906-910.Crossref 13. Narisawa K, Tada K, Kuzuya T: Neutropenia in type IB glycogen storage disease. J Pediatr 1981;99:334-335.Crossref 14. Kelleher JF, Yudkoff M, Hutchinson R, et al: The pancytopenia of isovaleric acidemia. Pediatrics 1980;65:1023-1026. 15. Soriano RJ, Taitz LS, Finberg L, et al: Hyperglycinemia with ketoacidosis and leukopenia. Pediatrics 1967;39:818-821. 16. Inoue S, Krieger I, Sarniak A, et al: Inhibition of bone marrow stem cell growth in vitro by methymalonic acid: A mechanism for pancytopenia in a patient with methylmalonic acidemia. Pediatr Res 1981;15:95-98.Crossref 17. Hakozaki H, Takahashi K, Naito M, et al: Gaucher-like cells in juvenile GM1 gangliosidosis and β-thalassemia. Acta Pathol Jpn 1979;29: 303-318.

Journal

Archives of Internal MedicineAmerican Medical Association

Published: Jun 1, 1983

References