{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,17]],"date-time":"2025-09-17T15:36:56Z","timestamp":1758123416473},"reference-count":22,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2004,10,7]],"date-time":"2004-10-07T00:00:00Z","timestamp":1097107200000},"content-version":"vor","delay-in-days":8802,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Annals of Neurology"],"published-print":{"date-parts":[[1980,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Ultrastructural study of leukocytes from 7 patients with mucopolysaccharidosis (MPS) I, II, or III demonstrated a variety of membrane\u2010bound inclusions in 34 to 68% of lymphocytes and larger mononuclear cells. The most common inclusions were membrane\u2010bound vacuoles containing faintly osmiophilic material, but a variety of clear vacuoles, dense granular inclusions, and myelin figures were also seen. Fingerprint profiles were present in small percentages in MPS II and III, indicating that they are not as specific as previously considered. Granulocytes, lymphocytes, and larger mononuclear cells from a patient with MPS VII showed many small and larger membrane\u2010bound vacuolated inclusions, some of which contained large dense granules. The presence of alterations in a readily accessible tissue such as blood indicates that ultrastructural study of leukocytes may be a useful adjunct in guiding further biochemical studies in MPS, es pecially in early or obscure cases.<\/jats:p>","DOI":"10.1002\/ana.410080320","type":"journal-article","created":{"date-parts":[[2005,1,1]],"date-time":"2005-01-01T13:15:28Z","timestamp":1104585328000},"page":"332-336","source":"Crossref","is-referenced-by-count":16,"title":["Mucopolysaccharidoses: Ultrastructure of leukocyte inclusions"],"prefix":"10.1002","volume":"8","author":[{"given":"William R.","family":"Markesbery","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Richard O.","family":"Robinson","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peggy V.","family":"Falace","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael D.","family":"Frye","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2004,10,7]]},"reference":[{"key":"e_1_2_1_2_2","first-page":"372","article-title":"Uber konstitutionell Bedingte Granulations ver\u00e4nderungen der Leukocyten","volume":"183","author":"Adler A","year":"1938","journal-title":"Dtsch Arch Klin Med"},{"key":"e_1_2_1_3_2","doi-asserted-by":"publisher","DOI":"10.1002\/ana.410040609"},{"key":"e_1_2_1_4_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(75)80968-1"},{"key":"e_1_2_1_5_2","first-page":"1","article-title":"Ultrastructure and cytochemistry of lymphocytes in the genetic mucopolysaccharidoses","volume":"93","author":"Belcher RW","year":"1972","journal-title":"Arch Pathol"},{"key":"e_1_2_1_6_2","first-page":"277","article-title":"Hepatic ultrastructure in the Hurler syndrome","volume":"48","author":"Callahan WP","year":"1966","journal-title":"Am J Pathol"},{"key":"e_1_2_1_7_2","first-page":"251","article-title":"Different clinical and biochemical phenotypes associated with \u03b2\u2010glucuronidase deficiency","volume":"10","author":"Daver BS","year":"1974","journal-title":"Birth Defects vol"},{"key":"e_1_2_1_8_2","first-page":"1095","article-title":"Contribution to discussion on Adler A: Konstitutionell Bedingte Granulationsver\u00e4nderungen der Leukocyten und Knochenver\u00e4nderungen","volume":"80","author":"Gasser C","year":"1950","journal-title":"Schweiz Med Wochenschr"},{"key":"e_1_2_1_9_2","first-page":"149","article-title":"Mucopolysaccharidosis VII: \u03b2\u2010glucuronidase deficiency","volume":"23","author":"Gehler T","year":"1974","journal-title":"Humangenetik"},{"key":"e_1_2_1_10_2","first-page":"115","article-title":"Hematologic studies in mucopolysaccharidoses and mucolipidoses","volume":"8","author":"Hansen HG","year":"1972","journal-title":"Birth Defects vol"},{"key":"e_1_2_1_11_2","doi-asserted-by":"publisher","DOI":"10.1203\/00006450-197103000-00006"},{"key":"e_1_2_1_12_2","first-page":"1578","article-title":"The site of mucopolysaccharide accumulation in Hurler's syndrome; an electron microscopic and histochemical study","volume":"15","author":"Lagunoff D","year":"1966","journal-title":"Lab Invest"},{"key":"e_1_2_1_13_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.1976.00500090036007"},{"key":"e_1_2_1_14_2","doi-asserted-by":"publisher","DOI":"10.1097\/00005792-196511000-00001"},{"key":"e_1_2_1_15_2","first-page":"1282","volume-title":"The Metabolic Basis of Inherited Disease. 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