Long Term G-CSF-Induced Remission of Ulcerative Colitis-Like Inflammatory Bowel Disease in a Patient With Glycogen Storage Disease Ib and Evaluation of Associated Neutrophil Function

被引:16
作者
Alsultan, Abdulrahman [2 ]
Sokol, Ronald J. [3 ]
Lovell, Mark A. [2 ]
Thurman, Gail [2 ]
Ambruso, Daniel R. [1 ]
机构
[1] Bonfils Blood Ctr, Denver, CO 80230 USA
[2] Univ Colorado Denver, Dept Pediat & Pathol, Aurora, CO USA
[3] Univ Colorado Denver, Childrens Hosp, Colorado Clin & Translat Sci Inst, Aurora, CO USA
关键词
glycogen storage disease Ib G CSF; neutrophil functions; ulcerative colitis; IMPAIRED METABOLIC FUNCTION; POLYMORPHONUCLEAR LEUKOCYTES; BLOOD NEUTROPHILS; 1B; DEFECTS;
D O I
10.1002/pbc.22706
中图分类号
R73 [肿瘤学];
学科分类号
100214 [肿瘤学];
摘要
We present a 23 year old female with Glycogen storage disease Ib (GSD Ib) who was diagnosed with ulcerative colitis like inflammatory bowel disease (IBD) at 7 years of age G CSF therapy reversed the IBD was required to maintain I BD remission and was well tolerated Neutrophil functions at time of diagnosis showed impaired chemotaxis but normal superoxide anion production and bactericidal activity Ulcerative colitis like IBD may also be seen in GSD Ib and is responsive to G CSF therapy Neutrophil dysfunction is variable among patients with GSD Ib Pediatr Blood Cancer 2010 55 1410-1413 (C) 2010 Wiley Liss Inc
引用
收藏
页码:1410 / 1413
页数:4
相关论文
共 16 条
[1]
INFECTIOUS AND BLEEDING COMPLICATIONS IN PATIENTS WITH GLYCOGENOSIS-IB [J].
AMBRUSO, DR ;
MCCABE, ERB ;
ANDERSON, D ;
BEAUDET, A ;
BALLAS, LM ;
BRANDT, IK ;
BROWN, B ;
COLEMAN, R ;
DUNGER, DB ;
FALLETTA, JM ;
FRIEDMAN, HS ;
HAYMOND, MW ;
KEATING, JP ;
KINNEY, TR ;
LEONARD, JV ;
MAHONEY, DH ;
MATALON, R ;
ROE, TF ;
SIMMONS, P ;
SLONIM, AE .
AMERICAN JOURNAL OF DISEASES OF CHILDREN, 1985, 139 (07) :691-697
[2]
OXIDATIVE-METABOLISM OF CORD BLOOD NEUTROPHILS - RELATIONSHIP TO CONTENT AND DE-GRANULATION OF CYTOPLASMIC GRANULES [J].
AMBRUSO, DR ;
BENTWOOD, B ;
HENSON, PM ;
JOHNSTON, RB .
PEDIATRIC RESEARCH, 1984, 18 (11) :1148-1153
[3]
RECURRENT INFECTION IN GLYCOGENOSIS TYPE-IB - ABNORMAL NEUTROPHIL MOTILITY RELATED TO IMPAIRED REDISTRIBUTION OF ADHESION SITES [J].
ANDERSON, DC ;
MACE, ML ;
BRINKLEY, BR ;
MARTIN, RR ;
SMITH, CW .
JOURNAL OF INFECTIOUS DISEASES, 1981, 143 (03) :447-459
[4]
IMPAIRED CARBOHYDRATE-METABOLISM OF POLYMORPHONUCLEAR LEUKOCYTES IN GLYCOGEN-STORAGE DISEASE-IB [J].
BASHAN, N ;
HAGAI, Y ;
POTASHNIK, R ;
MOSES, SW .
JOURNAL OF CLINICAL INVESTIGATION, 1988, 81 (05) :1317-1322
[5]
NEUTROPENIA AND IMPAIRED NEUTROPHIL MIGRATION IN TYPE-IB GLYCOGEN-STORAGE DISEASE [J].
BEAUDET, AL ;
ANDERSON, DC ;
MICHELS, VV ;
ARION, WJ ;
LANGE, AJ .
JOURNAL OF PEDIATRICS, 1980, 97 (06) :906-910
[6]
Association of glycogen storage disease 1b and Crohn disease: results of a North American survey [J].
Dieckgraefe, BK ;
Korzenik, JR ;
Husain, A ;
Dieruf, L .
EUROPEAN JOURNAL OF PEDIATRICS, 2002, 161 (Suppl 1) :S88-S92
[7]
IMPAIRED METABOLIC FUNCTION OF POLYMORPHONUCLEAR LEUKOCYTES IN GLYCOGEN-STORAGE DISEASE-IB [J].
GAHR, M ;
HEYNE, K .
EUROPEAN JOURNAL OF PEDIATRICS, 1983, 140 (04) :329-330
[8]
IMPAIRED METABOLIC FUNCTION AND SIGNALING DEFECTS IN PHAGOCYTIC-CELLS IN GLYCOGEN-STORAGE-DISEASE TYPE-1B [J].
KILPATRICK, L ;
GARTY, BZ ;
LUNDQUIST, KF ;
HUNTER, K ;
STANLEY, CA ;
BAKER, L ;
DOUGLAS, SD ;
KORCHAK, HM .
JOURNAL OF CLINICAL INVESTIGATION, 1990, 86 (01) :196-202
[9]
IMPAIRED CHEMOTAXIS AND NEUTROPHIL (POLYMORPHONUCLEAR LEUKOCYTE) FUNCTION IN GLYCOGENOSIS TYPE-IB [J].
KOVEN, NL ;
CLARK, MM ;
CODY, CS ;
STANLEY, CA ;
BAKER, L ;
DOUGLAS, SD .
PEDIATRIC RESEARCH, 1986, 20 (05) :438-442
[10]
Apoptotic neutrophils in the circulation of patients with glycogen storage disease type 1b (GSD1b) [J].
Kuijpers, TW ;
Maianski, NA ;
Tool, ATJ ;
Smit, GPA ;
Rake, JP ;
Roos, D ;
Visser, G .
BLOOD, 2003, 101 (12) :5021-5024