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Incidence and risk factors for hypogammaglobulinemia in pediatric patients following allo-SCT.
Frangoul H, Min E, Wang W, Chandrasekhar R, Calder C, Evans M, Manes B, Bruce K, Brown V, Ho R, Domm J
(2013) Bone Marrow Transplant 48: 1456-9
MeSH Terms: Adolescent, Adult, Agammaglobulinemia, Child, Child, Preschool, Female, Hematopoietic Stem Cell Transplantation, Humans, Incidence, Infant, Infant, Newborn, Male, Prospective Studies, Risk Factors, Transplantation Conditioning, Transplantation, Autologous, Young Adult
Show Abstract · Added March 5, 2014
We evaluated the incidence and risk factors for hypogammaglobulinemia after allogeneic hematopoietic SCT (HSCT) in pediatric patients. Ig levels were measured pre-transplant, every 2 weeks until day 100 and then monthly post SCT in 185 patients undergoing myeloablative HSCT. Median age was 9 years; 142 (77%) had malignant disease and 114 (62%) received stem cells from an unrelated source. Hypogammaglobulinemia (IgG <500 mg/dL) developed in 143 (77%) of the patients at a median of 56 days (range 15-339) post SCT. The cumulative incidence of hypogammaglobulinemia at 1 year was higher among patients who developed acute GVHD (97% vs 54%, P<0.001), and for those receiving stem cells from an unrelated source (94% vs 51%, P<0.001). The cumulative incidence of TRM was significantly higher for patients with hypogammaglobulinemia (P=0.026). In multivariable analysis, lower pre-transplant IgG level (P<0.001), younger age (P=0.012), diagnosis of malignant disease (P<0.001), receiving unrelated SCT (P<0.001) and development of acute GVHD (P<0.001) were all significantly associated with higher risk of hypogammaglobulinemia post HSCT. We conclude that hypogammaglobulinemia is common, following allogeneic HSCT in pediatric patients, especially in those with malignant diseases, those who receive an unrelated transplant or patients who develop GVHD.
1 Communities
2 Members
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17 MeSH Terms
Role of A2B adenosine receptors in regulation of paracrine functions of stem cell antigen 1-positive cardiac stromal cells.
Ryzhov S, Goldstein AE, Novitskiy SV, Blackburn MR, Biaggioni I, Feoktistov I
(2012) J Pharmacol Exp Ther 341: 764-74
MeSH Terms: Adenosine, Adenosine Deaminase, Agammaglobulinemia, Animals, Antigens, Ly, Cells, Cultured, Chemokine CXCL1, Female, Interleukin-6, Male, Membrane Proteins, Mesenchymal Stem Cells, Mice, Mice, Inbred C57BL, Mice, Knockout, Mice, Transgenic, Myocardial Infarction, Myocardium, Paracrine Communication, Platelet Endothelial Cell Adhesion Molecule-1, RNA, Messenger, Receptor, Adenosine A2B, Severe Combined Immunodeficiency, Stem Cells, Up-Regulation, Vascular Endothelial Growth Factor A
Show Abstract · Added May 29, 2014
The existence of multipotent cardiac stromal cells expressing stem cell antigen (Sca)-1 has been reported, and their proangiogenic properties have been demonstrated in myocardial infarction models. In this study, we tested the hypothesis that stimulation of adenosine receptors on cardiac Sca-1(+) cells up-regulates their secretion of proangiogenic factors. We found that Sca-1 is expressed in subsets of mouse cardiac stromal CD31(-) and endothelial CD31(+) cells. The population of Sca-1(+)CD31(+) endothelial cells was significantly reduced, whereas the population of Sca-1(+)CD31(-) stromal cells was increased 1 week after myocardial infarction, indicating their relative functional importance in this pathophysiological process. An increase in adenosine levels in adenosine deaminase-deficient mice in vivo significantly augmented vascular endothelial growth factor (VEGF) production in cardiac Sca-1(+)CD31(-) stromal cells but not in Sca-1(+)CD31(+) endothelial cells. We found that mouse cardiac Sca-1(+)CD31(-) stromal cells predominantly express mRNA encoding A(2B) adenosine receptors. Stimulation of adenosine receptors significantly increased interleukin (IL)-6, CXCL1 (a mouse ortholog of human IL-8), and VEGF release from these cells. Using conditionally immortalized Sca-1(+)CD31(-) stromal cells obtained from wild-type and A(2B) receptor knockout mouse hearts, we demonstrated that A(2B) receptors are essential for adenosine-dependent up-regulation of their paracrine functions. We found that the human heart also harbors a population of stromal cells similar to the mouse cardiac Sca-1(+)CD31(-) stromal cells that increase release of IL-6, IL-8, and VEGF in response to A(2B) receptor stimulation. Thus, our study identified A(2B) adenosine receptors on cardiac stromal cells as potential targets for up-regulation of proangiogenic factors in the ischemic heart.
0 Communities
1 Members
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26 MeSH Terms
Recurrent pneumococcal arthritis as the presenting manifestation of X-linked agammaglobulinemia.
Peters TR, Brumbaugh DE, Lawton AR, Crowe JE
(2000) Clin Infect Dis 31: 1287-8
MeSH Terms: Agammaglobulinemia, Arthritis, Infectious, Child, Preschool, Genetic Linkage, Humans, Knee Joint, Male, Pneumococcal Infections, Recurrence, Streptococcus pneumoniae, X Chromosome
Show Abstract · Added August 6, 2012
Pneumococcal arthritis in children older than 24 months is unusual and can suggest underlying immunodeficiency. We report a case of recurrent pneumococcal arthritis as the presenting manifestation of X-linked agammaglobulinemia.
0 Communities
1 Members
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11 MeSH Terms
Gastrointestinal pathology in patients with common variable immunodeficiency and X-linked agammaglobulinemia.
Washington K, Stenzel TT, Buckley RH, Gottfried MR
(1996) Am J Surg Pathol 20: 1240-52
MeSH Terms: Adolescent, Adult, Agammaglobulinemia, Apoptosis, Biopsy, Child, Child, Preschool, Chronic Disease, Colon, Common Variable Immunodeficiency, Diagnosis, Differential, Digestive System, Duodenum, Female, Gastrointestinal Diseases, Graft vs Host Disease, Humans, Immunoenzyme Techniques, Infant, Intestine, Small, Male, Middle Aged, Necrosis, Retrospective Studies, X Chromosome
Show Abstract · Added April 12, 2016
Review of the medical records of 43 patients with common variable immunodeficiency (CVID) and 23 patients with X-linked agammaglobulinemia (XLAG) revealed a high incidence of chronic gastrointestinal complaints, most commonly diarrhea. Thirty-eight biopsies, four small-bowel resection specimens, and one autopsy from 10 patients with CVID and one patient with XLAG showed a wide range of abnormalities. A pattern resembling acute graft-versus-host disease, with apoptotic bodies and lymphocytes in crypts, was seen in the stomach (four patients), small bowel (three patients), and colon (three patients). Small-bowel specimens from three CVID patients with malabsorption showed mild to severe villous atrophy. Three CVID patients had Giardia in biopsies. Two cases of small bowel lymphoma associated with nodular lymphoid hyperplasia were identified in CVID patients. One patient's small bowel contained foamy histiocytes in the lamina propria, resembling Whipple's disease or chronic granulomatous disease, with numerous apoptotic bodies in crypts. Ultrastructurally, the histiocytes contained cellular debris. The patient with XLAG had recurrent fissuring necrosis of small bowel resembling Crohn's disease; a patient with CVID had colitis with features similar to ulcerative colitis. Poorly formed granulomas were seen in the stomach (one CVID patient) and the colon (two CVID patients). Lymphocyte populations were dominated by T cells; B cells were scarce except in lymphoid follicles in CVID patients with nodular lymphoid hyperplasia. Patients with CVID and XLAG manifest a spectrum of abnormalities in the gastrointestinal tract, with patterns superficially resembling graft-versus-host disease, inflammatory bowel disease, and Whipple's disease, but often lacking some of the diagnostic features of the diseases. Many of the CVID patients with chronic gastrointestinal complaints (62%) also had evidence of autoimmune phenomena, suggesting that in some patients the inflammatory process in the gastrointestinal tract has an autoimmune component.
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1 Members
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25 MeSH Terms
Persistence of Campylobacter fetus bacteremia associated with absence of opsonizing antibodies.
Neuzil KM, Wang E, Haas DW, Blaser MJ
(1994) J Clin Microbiol 32: 1718-20
MeSH Terms: Agammaglobulinemia, Antibodies, Bacterial, Bacteremia, Blood Bactericidal Activity, Campylobacter fetus, Humans, Immunization, Passive, Male, Middle Aged, Opsonin Proteins, Recurrence, Treatment Failure
Show Abstract · Added March 13, 2015
Campylobacter fetus causes systemic infections in immunocompromised hosts. We describe a case in which C. fetus bacteremia apparently relapsed after 7 years in a patient with hypogammaglobulinemia and characterize the serum resistance of the patient's C. fetus strain and the inability of the patient's serum, with and without commercial intravenous immunoglobulin, to opsonize this and another C. fetus strain effectively. The probable presence of a sequestered site of infection in bone, the intrinsic serum resistance of the C. fetus strain, and the absence of specific antibody may account for the persistent infection in this patient. These studies suggest that intravenous immunoglobulin treatment is not useful in eradicating C. fetus bacteremia.
0 Communities
1 Members
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12 MeSH Terms
Interactions among human T cell subsets.
Thomas Y, Sosman J, Irigoyen OH, Rogozinski L, Friedman SM, Chess L
(1981) Int J Immunopharmacol 3: 193-201
MeSH Terms: Agammaglobulinemia, Antibodies, Monoclonal, B-Lymphocytes, Cell Communication, Cell Differentiation, Cytotoxicity, Immunologic, Humans, Lymphocyte Cooperation, Lymphocyte Culture Test, Mixed, T-Lymphocytes, T-Lymphocytes, Regulatory
Added March 5, 2014
0 Communities
1 Members
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11 MeSH Terms