Profound symptomatic hypogammaglobulinemia: A rare late complication after rituximab treatment for immune thrombocytopenia. Report of 3 cases and systematic review of the literature.

Lévy, Romain; Mahévas, Matthieu; Galicier, Lionel; Boutboul, David; Moroch, Julien; Loustau, Valentine; Guillaud, Constance; Languille, Laeticia; Fain, Olivier; Bierling, Philippe; Khellaf, M.; Michel, Marc; Oksenhendler, Éric; Godeau, Bertrand

doi:10.1016/j.autrev.2014.08.036

Cited by 66 publications

(43 citation statements)

References 62 publications

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“…More severe events such as neutropenia and infections were encountered in only 6% of patients. Levy et al reported a rate of hypogammaglobulinemia of 11% after administration of RTX in immune thrombopenic purpura, with some patients that developed symptomatic infectious events [41]. On the other hand, few controlled published data are available on efficacy and risks of other immunosuppressive treatments as second line therapy.…”

Section: Discussionmentioning

confidence: 98%

Efficacy and safety of rituximab in auto-immune hemolytic anemia: A meta-analysis of 21 studies

Reynaud

Durieu

Dutertre

et al. 2015

Autoimmunity Reviews

109

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Section: Discussionmentioning

confidence: 98%

Efficacy and safety of rituximab in auto-immune hemolytic anemia: A meta-analysis of 21 studies

Reynaud

Durieu

Dutertre

et al. 2015

Autoimmunity Reviews

109

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“…report in their retrospective analysis of 32 studies conducted in France between 2001 and 2014 three cases of prolonged hypogammaglobulinemia in patients who had received RTX as a second-line treatment for immune thrombocytopenia [29]. Of interest, the clinical picture was reminiscent of CVID, including recurrent infections, and lasted up to 2 years after RTX discontinuation.…”

Section: Discussionmentioning

confidence: 99%

Subcutaneous immunoglobulins for the treatment of a patient with antisynthetase syndrome and secondary chronic immunodeficiency after anti-CD20 treatment: a case report

Chérìn

Jaeger²,

Crave

et al. 2017

J Med Case Reports

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BackgroundAntisynthetase syndrome is a rare and debilitating multiorgan disease characterized by inflammatory myopathy, interstitial lung disease, cutaneous involvement, and frequent chronic inflammation of the joints. Standard treatments include corticosteroids and immunosuppressants. In some cases, treatment resistance may develop. Administration of immunoglobulins intravenously is recommended in patients with drug-resistant antisynthetase syndrome.Case presentationHere, we describe the case of a 56-year-old woman of Algerian origin. She is the first case of a patient with multidrug-resistant antisynthetase syndrome featuring pulmonary involvement and arthropathy, and chronic secondary immune deficiency with recurrent infections, after anti-CD20 treatment, in which her primary antisynthetase syndrome-related symptoms and secondary immune deficiency were treated successfully with subcutaneous administration of immunoglobulin. The administration of immunoglobulin subcutaneously was introduced at a dose of 2 g/kg per month and was well tolerated. Clinical improvement was observed within 3 months of initiation of subcutaneous administration of immunoglobulin. After 22 months of treatment, she showed a significant improvement in terms of muscle strength, pulmonary involvement, arthralgia, and immunodeficiency. Her serum creatine phosphokinase and C-reactive protein levels remained normal. Finally, she was compliant and entirely satisfied with the treatment.ConclusionsTaken together, these observations suggest that administration of immunoglobulin subcutaneously may be a useful therapeutic approach to tackle steroid-refractory antisynthetase syndrome while ensuring minimal side effects and improved treatment compliance. This treatment also allowed, in our case, for the regression of the chronic immunodeficiency secondary to rituximab treatment.

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“…IgM levels <0.4 g/l were found in 58% of the 243 patients [15]. A large review of the literature identified 21 cases of hypogammaglobulinemia after rituximab treatment out of 351 patients with idiopathic thrombocytopenic purpura (6%) [26]. The prevalence of hypogammaglobulinemia in patients receiving rituximab treatment is related to the number of treatment cycles and the cumulative rituximab dose [27,28], as well as to the immunoglobulin levels before initiation of rituximab therapy [15].…”

Section: Iatrogenic Hypogammaglobulinemiasmentioning

confidence: 99%

“…The infection frequency was not quantified in the report, and therefore, statistical evaluation of the results was not performed [68]. A recent report of three patients with idiopathic thrombocytopenic purpura who were treated with rituximab and subsequently developed symptomatic hypogammaglobulinemia also reported that IVIG maintenance therapy reduced the number of infections [26].…”

Section: Antibody Replacement Therapy In Ihgmentioning

confidence: 99%

Antibody replacement therapy in primary antibody deficiencies and iatrogenic hypogammaglobulinemia

Hoffman

Kessel

Velzen‐Blad

et al. 2015

Expert Review of Clinical Immunology

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Antibody replacement therapy has been used in the treatment of primary antibody deficiencies (PADs) for several decades, and an evidence-based guideline for its treatment is currently available. By contrast, the use of antibody replacement therapy in iatrogenic hypogammaglobulinemia (IHG), a condition that is associated with immunosuppressive medication, has hardly any evidence base and no guidelines. As IHG can be equally as severe as PAD and is much more prevalent, evidence-based guidelines are urgently needed. This review will focus on the differences and similarities between PAD and IHG and the use of antibody replacement therapy in both conditions. Suggestions for the development of evidence-based guidelines and future research are given.

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Profound symptomatic hypogammaglobulinemia: A rare late complication after rituximab treatment for immune thrombocytopenia. Report of 3 cases and systematic review of the literature.

Cited by 66 publications

References 62 publications

Efficacy and safety of rituximab in auto-immune hemolytic anemia: A meta-analysis of 21 studies

Efficacy and safety of rituximab in auto-immune hemolytic anemia: A meta-analysis of 21 studies

Subcutaneous immunoglobulins for the treatment of a patient with antisynthetase syndrome and secondary chronic immunodeficiency after anti-CD20 treatment: a case report

Antibody replacement therapy in primary antibody deficiencies and iatrogenic hypogammaglobulinemia

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