Memory B-cell subsets as a predictive marker of outcome in hypogammaglobulinemia during infancy

Moschese, Viviana; Carsetti, Rita; Graziani, S; Chini, L; Soresina, Anna Rosa; Rocca, Marianna La; Bossi, Grazia; Cesare, Silvia Di; Plebani, Alessandro

doi:10.1016/j.jaci.2007.04.002

Cited by 20 publications

(32 citation statements)

References 9 publications

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“…CD19 + 27 + memory B-cell percentages were also low in the study by Artac et al n contrast with our reports of normal values (3.4±1.4%) in one of our previous studies 9 . Moschese et al 25 . also reported a lower frequency of memory B cells in patients over the age of 24 months.…”

Section: Discussionmentioning

confidence: 91%

Does Intravenous Immunoglobulin Therapy Prolong Immunodeficiency in Transient Hypogammaglobulinemia of Infancy?

et al. 2013

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Transient hypogammaglobulinemia of infancy (THI) is characterized by recurrent infections and one or more reduced serum immunoglobulin levels. Typically, THI patients recover spontaneously, mostly within 30-40 months of age, but sometimes recovery may be delayed until 5-6 years of age. The use of intravenous immunoglobulin (IVIg) as an alternative to antibiotic prophylaxis remains contraversial also in symptomatic THI patients. In fact, some authors believe that IVIg therapy may cause a delay in the maturation of the humoral immune system because of the interference from passively transfered antibodies. The aim of this study was to investigate the effect of IVIg replacement on recovery from immunodeficiency in THI patients and determine new parameters in order to include these patients in IVIg therapy groups. In this retrospective study, 43 patients (65%) received IVIg replacement therapy while 23 patients (34.8%) showed spontaneous normalization without IVIg. The percentages of patients who had more than six times the number of febrile infections in a year decreased from 91% to 21% in the group receiving IVIg treatment. At admission, before being recruited to IVIg therapy, serum immunoglobulin G (IgG) levels and anti-hemophilus B (Hib) antibody titers were found to be significantly low in cases who were selected for IVIg replacement. The percentages of patients who did not have protective levels of anti-Hib, anti-rubella or anti-rubeola-IgG were also significantly high in IVIg cases. There was no statistically significant difference in the age at which IgG levels normalized between the IVIg and the non-IVIg group. Patients in the IVIg group and non-IVIg group reached normal IgG levels at the age of 42.9±22.0 and 40.7±19.8 months, respectively. In conclusion, IVIg infusions do not cause a delay in the maturation of the immune system in THI patients. Besides the well-established criteria, very low and non-protective specific antibody responses against previously applied vaccines are important factors to consider when selecting patients for IVIg therapy.

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Section: Discussionmentioning

confidence: 91%

Does Intravenous Immunoglobulin Therapy Prolong Immunodeficiency in Transient Hypogammaglobulinemia of Infancy?

et al. 2013

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Section: Clinical Manifestationsmentioning

confidence: 99%

“…However, one study reported transient numeric and functional abnormalities of CD4 positive T helper cells (Moschese et al 2007(Moschese et al , 2008. Other investigators have found reduced frequencies of both circulating IgM+ and "switched" (IgM-IgD-) memory B cells and an inability to produce IgG in vitro in some children with THI; a phenotype that may identify the subset of patients whose immune defects persist (Moschese et al 2007(Moschese et al , 2008. This reduction in memory B cells has also been identified in children with earlyonset common variable immunodeficiency (CVID) and selective IgA deficiency (Bukowska-Strakova et al 2009) and older patients with CVID (Carsetti et al 2005;Alachkar et al 2006).…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Transient hypogammaglobulinemia of infancy

OvadiaAdi

DalalIlan

2014

LymphoSign Journal

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Transient hypogammaglobulinemia of infancy (THI) was first described as a distinct entity by Gitlin and Janeway in 1956. Although THI has been recognized for many years, and despite significant progress in understanding the molecular basis and identifying the genes involved in the pathogenesis of many other forms of humoral immunodeficiencies, not much is known about this specific entity. This article summarizes the definition of THI, possible etiologies, clinical manifestations, treatment, and prognosis.

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“…15 Moschese et al studied 32 infants and young children by examining the circulating B cells that bore the CD27 surface marker of memory B cells, finding that putative THI subjects who achieve normal IgG levels have decreased IgM1 and switched (i.e., IgM2 ) memory B cells initially, but develop normal numbers compared to age-matched control subjects when IgG levels normalize. 16 Young children initially thought to have THI who fail to achieve normal IgG levels were found to have persistently decreased numbers of both subsets of memory B cells. 16 A subsequent report by these authors evaluated 57 infants and young children for both IgM and switched memory B cell subsets, and compared a subset to normal subjects for in vitro IgG production.…”

Section: B Cell Abnormalitiesmentioning

confidence: 99%

“…16 Young children initially thought to have THI who fail to achieve normal IgG levels were found to have persistently decreased numbers of both subsets of memory B cells. 16 A subsequent report by these authors evaluated 57 infants and young children for both IgM and switched memory B cell subsets, and compared a subset to normal subjects for in vitro IgG production. The authors reproduced the earlier finding of decreased memory B cells.…”

Section: B Cell Abnormalitiesmentioning

confidence: 99%

Transient Hypogammaglobulinemia of Infancy

McGeady

2014

Stiehm's Immune Deficiencies

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Memory B-cell subsets as a predictive marker of outcome in hypogammaglobulinemia during infancy

Cited by 20 publications

References 9 publications

Does Intravenous Immunoglobulin Therapy Prolong Immunodeficiency in Transient Hypogammaglobulinemia of Infancy?

Does Intravenous Immunoglobulin Therapy Prolong Immunodeficiency in Transient Hypogammaglobulinemia of Infancy?

Transient hypogammaglobulinemia of infancy

Transient Hypogammaglobulinemia of Infancy

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