{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T14:27:33Z","timestamp":1786976853945,"version":"build-2736575974"},"reference-count":21,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2006,7,11]],"date-time":"2006-07-11T00:00:00Z","timestamp":1152576000000},"content-version":"vor","delay-in-days":4970,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J Hematol"],"published-print":{"date-parts":[[1992,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Granulocyte macrophage\u2010colony\u2010stimulating factor (GM\u2010CSF) has shown promise as a means of alleviating leukopenia associated with a wide variety of disorders. It is currently undergoing evaluation as an adjunct to bone marrow transplantation but its use in patients with metabolic disorders, such as Hurler's syndrome (HS), has not been explored. We followed bone marrow morphology in a 2\u2010year\u2010old male with HS who received up to 8 \u03bcg\/kg GM\u2010CSF per day because of failure of allogeneic bone marrow engraftment. Both premortem and postmortem bone marrow sampling revealed almost complete replacement of the marrow space by sheets of histiocytes demonstrating metachromatic cytoplasmic granules. Such cells were present in far greater numbers than are usually seen in untreated patients with HS or patients with HS undergoing successful bone marrow transplantation without GM\u2010CSF. Moreover, the in vitro culture of bone marrow from a second HS patient showed a GM\u2010CSF dose\u2010related increase in colony formation up to a dose of 250 units\/ml. Microscopic examination of these colonies showed a high percent\u2010age of histiocytes identical to those seen in the patient's bone marrow. These observations suggest that caution should be exercised when considering administration of CSFs to patients with HS and similar metabolic storage diseases. \u00a9 1992 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/ajh.2830410411","type":"journal-article","created":{"date-parts":[[2007,2,21]],"date-time":"2007-02-21T07:54:49Z","timestamp":1172044489000},"page":"280-284","source":"Crossref","is-referenced-by-count":6,"title":["Proliferation of abnormal bone marrow histiocytes, an undesired effect of granulocyte macrophage\u2010colony\u2010stimulating factor therapy in a patient with hurler's syndrome undergoing bone marrow transplantation"],"prefix":"10.1002","volume":"41","author":[{"given":"Eric","family":"Lang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael L.","family":"Cibull","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diane D.","family":"Davey","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vincent S.","family":"Gallicchio","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P.","family":"Jean Henslee\u2010Downey","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward J.","family":"Harder","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael J.","family":"Messino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2006,7,11]]},"reference":[{"key":"e_1_2_1_2_2","first-page":"99","article-title":"Human recombinant hemopoietic growth factors G\u2010CSF and GM\u2010CSF: First results of clinical trials","volume":"31","author":"Nissen\u2010Druey C","year":"1989","journal-title":"Nouv Rev Fr Hematol"},{"key":"e_1_2_1_3_2","first-page":"14","article-title":"Clinical applications of the myeloid growth factors","volume":"26","author":"Glaspy JA","year":"1989","journal-title":"Semin Hematol"},{"key":"e_1_2_1_4_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198911233212106"},{"key":"e_1_2_1_5_2","first-page":"121","article-title":"The use of recombinant human granulocyte\u2010macrophage colony\u2010stimulating factor in autologous bone marrow transplantation","volume":"338","author":"Peters WP","year":"1990","journal-title":"Prog Clin Biol Res"},{"key":"e_1_2_1_6_2","doi-asserted-by":"crossref","first-page":"245","DOI":"10.1182\/blood.V76.1.245.245","article-title":"The use of rhGM\u2010CSF for graft failure in patients after bone marrow transplantation","volume":"76","author":"Nemunaitis JJ","year":"1990","journal-title":"Blood"},{"key":"e_1_2_1_7_2","first-page":"589","article-title":"Laboratory control in predicting clinical efficacy of T\u2010cell depletion procedures used for prevention of graft\u2010versus\u2010host disease: Importance of limiting dilution analysis","volume":"3","author":"Marciniak E","year":"1988","journal-title":"Bone Marrow Transplantation"},{"key":"e_1_2_1_8_2","first-page":"123","volume-title":"Practical Surgical Pathology","author":"McAllister HA","year":"1985"},{"key":"e_1_2_1_9_2","first-page":"99","volume-title":"Syndromes of the Head and Neck","author":"Garlin RJ","year":"1990"},{"key":"e_1_2_1_10_2","doi-asserted-by":"publisher","DOI":"10.3181\/00379727-190-42837"},{"key":"e_1_2_1_11_2","doi-asserted-by":"crossref","unstructured":"KrivitW:Correction of inborn errors of metabolism by bone marrow transplant. In Hollaender A Laskin Al Rogers P (eds): \u201cBasic Biology of New Developments in Biotechnology.\u201d Basic Life Sciences 25:63\u201376 1982.","DOI":"10.1007\/978-1-4684-4460-5_5"},{"key":"e_1_2_1_12_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.3520819"},{"key":"e_1_2_1_13_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(81)91046-1"},{"key":"e_1_2_1_14_2","first-page":"1095","article-title":"Discussion of Alder's paper","volume":"801","author":"Gasser C","year":"1950","journal-title":"Schweiz Med. 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