Vedolizumab in Patients With Common Variable Immune Deficiency and Gut Inflammation

Boland, Brigid S.; Riedl, Marc A.; Valasek, Mark A.; Crowe, Sheila E.; Sandborn, William J.

doi:10.1038/ajg.2017.246

Cited by 15 publications

(10 citation statements)

References 4 publications

(4 reference statements)

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“…There is some concern that the use of the anti-a4b7 integrin vedolizumab could potentially worsen enteropathy by blocking extravasation of Tregs into the gut mucosa 60 but this therapy has been used with some success. 62…”

Section: Introductionmentioning

confidence: 99%

Gastrointestinal Manifestations and Complications of Primary Immunodeficiency Disorders

Agarwal

Cunningham‐Rundles

2019

Immunology and Allergy Clinics of North America

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SUMMARY Although the hallmark of PIDs is increased susceptibility to infection, many are associated with and initially present with GI diseases, making routine evaluation of the gut necessary. A history of recurrent infections, clinical and/or histologic features atypical of the usual pattern of GI disease, or a poor response to conventional therapy should prompt further immunologic evaluation. Early diagnosis and treatment may prevent irreversible tissue damage and mortality. In most cases, treatment with replacement immunoglobulin and antibiotics does not reverse or prevent the development of GI disease; therefore, additional immunomodulatory therapies, including, in some cases, organ transplantation, are indicated.

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Section: Introductionmentioning

confidence: 99%

Gastrointestinal Manifestations and Complications of Primary Immunodeficiency Disorders

Agarwal

Cunningham‐Rundles

2019

Immunology and Allergy Clinics of North America

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“…Standard treatment with parenteral immune globulin has no apparent effect, and other methods, mostly borrowed from inflammatory bowel disease care, have been used, including mesalamine, budesonide, 6-MP, and nonabsorbable antibiotics. Recently, two of three patients with CVID and enteropathy at one center, and in a separate report of another patient [12] were reported to have had benefit from treatment with Vedolizumab, [11] a monoclonal antibody to the α 4 β 7 integrin heterodimer, licensed for use in Crohn's disease and ulcerative colitis [20]. Vedolizumab blocks the interaction of α 4 β 7 integrin with MAdCAM-1, preventing leukocytes from adhesion with endothelial surfaces, thereby reducing T cell trafficking to the gut.…”

Section: Discussionmentioning

confidence: 99%

“…Vedolizumab was approved for the treatment of adults with moderate to severe inflammatory bowel disease in 2014. In 2017, Boland et al published a study of three patients with CVID and gut inflammation, two of whom were successfully treated with vedolizumab [11]. A second report noted benefit in another case [12].…”

Section: Introductionmentioning

confidence: 99%

Vedolizumab therapy in common variable immune deficiency associated enteropathy: A case series

Sifers

Hirten

Mehandru

et al. 2020

Clinical Immunology

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A number of gastrointestinal complications occur in common variable immunodeficiency (CVID). Infections are one cause, but various forms of severe non-infectious enteropathy also lead to substantial morbidity. The presence of T cell lymphocytic infiltrates in the mucosa have suggested that vedolizumab, a humanized monoclonal antibody which binds to alpha4 beta7 integrin and inhibits the migration of effector T-lymphocytes into gastrointestinal tissues, would be an effective treatment. A previous report of 3 CVID cases suggested benefit in 2 subjects. In this study 7 CVID patients with severe enteropathy were treated with vedolizumab. Four of the 7 completed vedolizumab induction therapy but 3 subjects had acute decompensation during induction and treatment was stopped. While one subject showed improvement, 6 of the 7 patients were withdrawn from therapy. While vedolizumab may be of use in some CVID subjects, it was not ultimately found helpful in most of these patients.

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“…Therefore, it selectively impacts on gut-specific lymphocyte trafficking. 10 Boland et al 11 have recently reported three adult patients with CVID having IBD treated with vedolizumab successfully. While vedolizumab has been reported as an effective therapy alternative in adult patients with IBD, there is limited data regarding the dosage and treatment duration on pediatric cases in the literature.…”

Section: Discussionmentioning

confidence: 99%

Vedolizumab treatment in a patient with X-linked agammaglobulinemia, is it safe and efficient?

et al. 2019

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The loss of inflammatory regulation resulting from the absence of B-lymphocytes leads to a risk for autoimmune and autoinflammatory complications. There is no data on the use of Vedolizumab in patients with X-linked agammaglobulinemia (XLA) as well as children with another primary immunodeficiency (PID) diseases. A 4-year-old boy was admitted to our clinic with a history of recurrent respiratory tract infections. He was diagnosed with XLA based on extremely low immunoglobulins, very low level of B cells, genetic mutation of BTK gene, and family history. At the age of 8, he suffered from intermittent fever attacks, abdominal pain, weakness, oral aft, and weight loss. His clinical and laboratory features were consistent with inflammatory bowel disease. Histopathological examination of the biopsy material obtained from terminal ileum, colon and cecum showed Crohn's disease. Initially, he was treated with prednisolone and infliximab. Because of the lack of response, infliximab treatment was switched to adalimumab. Terminal ileum was resected to relieve obstruction complication. Although he had been treated with adalimumab, a significant improvement was not observed. Vedolizumab (Entyvio™), a humanized monoclonal antibody α4β7 integrin-receptor antagonist, was commenced. After treatment with vedolizumab, his fever and abdominal pain attacks reduced, his total daily calorie intake increased and weight gain improved. He began to walk again and continued his school education properly. No side effects were observed in 18 months. This is the first immunocompromised child treated with vedolizumab. The symptoms of the patient receded and no side effect were seen during the treatment.

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Vedolizumab in Patients With Common Variable Immune Deficiency and Gut Inflammation

Cited by 15 publications

References 4 publications

Gastrointestinal Manifestations and Complications of Primary Immunodeficiency Disorders

Gastrointestinal Manifestations and Complications of Primary Immunodeficiency Disorders

Vedolizumab therapy in common variable immune deficiency associated enteropathy: A case series

Vedolizumab treatment in a patient with X-linked agammaglobulinemia, is it safe and efficient?

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