{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,7]],"date-time":"2025-12-07T04:25:42Z","timestamp":1765081542732,"version":"3.46.0"},"reference-count":13,"publisher":"BMJ","issue":"2","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Gut"],"published-print":{"date-parts":[[1993,2]]},"abstract":"<jats:p>The detection of an antineutrophil antibody which is highly sensitive and specific for adult primary sclerosing cholangitis using indirect immunoalkaline phosphatase has been previously described. In this study, the diagnostic potential of this method in childhood primary sclerosing cholangitis is described. A range of 72 blinded children's sera (36 boys), aged six months to 21 years (10 primary sclerosing cholangitis, eight autoimmune chronic active hepatitis, 10 alpha-1 antitrypsin deficiency, 12 extrahepatic bile duct atresia, 11 ulcerative colitis and 21 normal subjects) was assayed. Eight of the 10 primary sclerosing cholangitis patients were correctly identified. Three patients with chronic active hepatitis also showed the characteristic primary sclerosing cholangitis pattern of staining. No ulcerative colitis patients or any other patients showed this pattern of staining. All normal subjects were negative. As in adult primary sclerosing cholangitis, there is a specific antineutrophil antibody in childhood primary sclerosing cholangitis and this provides further evidence towards an autoimmune aetiology of this condition. The test may have diagnostic potential.<\/jats:p>","DOI":"10.1136\/gut.34.2.199","type":"journal-article","created":{"date-parts":[[2007,9,18]],"date-time":"2007-09-18T17:41:31Z","timestamp":1190137291000},"page":"199-202","source":"Crossref","is-referenced-by-count":23,"title":["Antineutrophil antibody: a test for autoimmune primary sclerosing cholangitis in childhood?"],"prefix":"10.1136","volume":"34","author":[{"given":"S K","family":"Lo","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"R W","family":"Chapman","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"P","family":"Cheeseman","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"C P","family":"Charlton","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J A","family":"Walker-Smith","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"G","family":"Mieli-Vergani","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"K A","family":"Fleming","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[1993,2,1]]},"reference":[{"key":"ref_1","unstructured":"The disease is being diagnosed with increasing frequency among adult patients with the advent of endoscopic retrograde cholangiography. This technique remains the gold standard in diagnosing primary sclerosing cholangitis. Primary sclerosing cholangitis has been described very rarely in childhood and this may be attributed to the limited use of endoscopic retrograde cholangiography in children. Before 1987, only five children with childhood primary sclerosing cholangitis were reported in the literature.3 5 Then, a series of 13 children with primary sclerosing cholangitis was described from King's College Hospital, London, suggesting a higher prevalence of primary sclerosing cholangitis in childhood than previously suspected.6","article-title":"The disease is being diagnosed with increasing frequency among adult patients with the advent of endoscopic retrograde cholangiography. This technique remains the gold standard in diagnosing primary sclerosing cholangitis. Primary sclerosing cholangitis has been described very rarely in childhood and this may be attributed to the limited use of endoscopic retrograde cholangiography in children. Before 1987, only five children with childhood primary sclerosing cholangitis were reported in the literature.3 5 Then, a series of 13 children with primary sclerosing cholangitis was described from King's College Hospital, London, suggesting a higher prevalence of primary sclerosing cholangitis in childhood than previously suspected.6"},{"key":"ref_2","first-page":"870","article-title":"Primary sclerosing cholangitis: a review of its clinical features, cholangiography and hepatic histology","volume-title":"Gut","volume":"21","author":"Chapman, R.W.G.; Arborgh, B.A.M.; Rhodes, J.M.; Summerfield, J.A.; Dick, R.; Scheuer, P.J.; etal","year":"1980","unstructured":"Chapman RWG, Arborgh BAM, Rhodes JM, Summerfield JA, Dick R, Scheuer PJ, etal. Primary sclerosing cholangitis: a review of its clinical features, cholangiography and hepatic histology. Gut 1980; 21: 870-7."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"200","DOI":"10.1016\/0016-5085(80)90131-6","article-title":"Clinicopathologic features of the syndrome of primary sclerosing cholangitis","volume":"79","author":"Wiesner, R.H.; La Russo, N.F.","year":"1980","unstructured":"Wiesner RH, La Russo NF. Clinicopathologic features of the syndrome of primary sclerosing cholangitis. Gastroenterology 1980; 79: 200-6.","journal-title":"Gastroenterology"},{"key":"ref_4","first-page":"433","article-title":"Sclerosing cholangitis in childhood","volume":"96","author":"Werlin, S.L.; Glicklich, M.; Jona, J.; Starshak, R.J.","year":"1980","unstructured":"Werlin SL, Glicklich M, Jona J, Starshak RJ. Sclerosing cholangitis in childhood. i Pediatr 1980; 96: 433-5.","journal-title":"i Pediatr"},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1177\/000992288202100101","article-title":"Sclerosing cholangitis associated with inflammatory bowel disease","volume":"21","author":"Freese, D.; Latimer, J.S.; Gilberstadt, S.; Kane, W.; Sharp, H.L.","year":"1982","unstructured":"Freese D, Latimer JS, Gilberstadt S, Kane W, Sharp HL. Sclerosing cholangitis associated with inflammatory bowel disease. Clin Pediatr 1982; 21: 11-6.","journal-title":"Clin Pediatr"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"129","DOI":"10.1016\/0016-5085(82)90134-2","article-title":"A case of primary sclerosing cholangitis","volume":"82","author":"Spivak, W.; Grand, R.J.; Eraklis, A.","year":"1982","unstructured":"Spivak W, Grand RJ, Eraklis A. A case of primary sclerosing cholangitis. Gastroentrology 1982; 82: 129-32.","journal-title":"Gastroentrology"},{"key":"ref_7","first-page":"122635","volume-title":"Primary sclerosing cholangitis in childhood. Gastroenterology 1987; 92","author":"El-Shabrawi, M.; Wilkinson, M.L.; Portmann, B.; Mieli-Vergani, G.; Chong, S.K.; Williams, R.; Mowat, A.P.","unstructured":"El-Shabrawi M, Wilkinson ML, Portmann B, Mieli-Vergani G, Chong SK, Williams R, Mowat AP. Primary sclerosing cholangitis in childhood. Gastroenterology 1987; 92: 122635."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1370","DOI":"10.1136\/gut.33.10.1370","article-title":"Prevalence of antineutrophil antibody in primary sclerosing cholangitis and ulcerative colitis using an alkaline phosphatase method","volume":"33","author":"Lo, S.K.; Fleming, K.A.; Chapman, R.W.","year":"1992","unstructured":"Lo SK, Fleming KA, Chapman RW. Prevalence of antineutrophil antibody in primary sclerosing cholangitis and ulcerative colitis using an alkaline phosphatase method. Gut 1992; 33: 1370-5.","journal-title":"Gut"},{"key":"ref_9","first-page":"30","article-title":"Antineutrophil nuclear antibody in ulcerative colitis. Crohn's disease and primary sclerosing cholangitis. Clin Exp Immunol","volume":"76","author":"Snook, J.A.; Chapman, R.W.; Fleming, K.; Jewell, D.P.","year":"1989","unstructured":"Snook JA, Chapman RW, Fleming K, Jewell DP. Antineutrophil nuclear antibody in ulcerative colitis. Crohn's disease and primary sclerosing cholangitis. Clin Exp Immunol 1989; 76: 30-3."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1385","DOI":"10.1016\/0016-5085(91)90793-K","article-title":"Neutrophil Cytoplasmic antibodies: A link between primary sclerosing cholangitis and ulcerative colitis","volume":"100","author":"Duerr, R.H.; Targan, SR, Landers; CJ, LaRusso; NF, Lindsay; KL, Wiesner; R.H.","year":"1991","unstructured":"Duerr RH, Targan SR, Landers CJ, LaRusso NF, Lindsay KL, Wiesner RH, et al. Neutrophil Cytoplasmic antibodies: A link between primary sclerosing cholangitis and ulcerative colitis. Gastroenterology 1991; 100: 1385-91.","journal-title":"Gastroenterology"},{"key":"ref_11","first-page":"64","article-title":"Perinuclear antineutrophil cytoplasmic antibodies in primary sclerosing cholangitis","volume":"14","author":"Sharma, A.N.; Rabson, A.R.; Kaplan, M.M.","year":"1991","unstructured":"Sharma AN, Rabson AR, Kaplan MM. Perinuclear antineutrophil cytoplasmic antibodies in primary sclerosing cholangitis. Hepatology 1991; 14: 64A.","journal-title":"Hepatology"},{"key":"ref_12","first-page":"114752","article-title":"Significance and specificity of antibodies to neutrophils detected by western blotting for the serological diagnosis of primary sclerosing cholangitis","volume":"14","author":"Klein, R.; Eisenburg, J.; Weber, P.; Seibold, F.; Berg, P.A.","year":"1991","unstructured":"Klein R, Eisenburg J, Weber P, Seibold F, Berg PA. Significance and specificity of antibodies to neutrophils detected by western blotting for the serological diagnosis of primary sclerosing cholangitis. Hepatology 1991; 14: 114752.","journal-title":"Hepatology"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"198","DOI":"10.1002\/hep.1840090206","article-title":"Different immune mechanisms leading to autoimmunity in primary sclerosing cholangitis and autoimmune chronic active hepatitis of childhood","volume":"9","author":"Mieli-Vergani, G.; Lobo-Yeo, A.; McFarlane, B.M.; McFarlane, I.G.; Mowat, A.P.; Vergani, D.","year":"1989","unstructured":"Mieli-Vergani G, Lobo-Yeo A, McFarlane BM, McFarlane IG, Mowat AP, Vergani D. Different immune mechanisms leading to autoimmunity in primary sclerosing cholangitis and autoimmune chronic active hepatitis of childhood. Hepatology 1989; 9: 198-203.","journal-title":"Hepatology"}],"container-title":["Gut"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/gut.34.2.199","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,12,7]],"date-time":"2025-12-07T02:53:59Z","timestamp":1765076039000},"score":1,"resource":{"primary":{"URL":"https:\/\/gut.bmj.com\/lookup\/doi\/10.1136\/gut.34.2.199"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[1993,2]]},"references-count":13,"journal-issue":{"issue":"2","published-online":{"date-parts":[[1993,2,1]]},"published-print":{"date-parts":[[1993,2]]}},"alternative-id":["10.1136\/gut.34.2.199"],"URL":"https:\/\/doi.org\/10.1136\/gut.34.2.199","relation":{},"ISSN":["0017-5749","1468-3288"],"issn-type":[{"type":"print","value":"0017-5749"},{"type":"electronic","value":"1468-3288"}],"subject":[],"published":{"date-parts":[[1993,2]]}}}