First report of improvement of coeliac disease in a patient with Sjogren's syndrome treated with rituximab

Nikiphorou, Elena; Hall, Frances

doi:10.1093/rheumatology/keu161

Cited by 7 publications

(5 citation statements)

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“…Although CD20 immunostaining had essentially negative results in this patient, perivascular infiltrates of inflammatory cells have been noted in DH, with B lymphocytes particularly implicated in the early development of lesions; elimination of these cells may reduce disease burden and prevent progression of disease. Interestingly, the intestinal symptoms of celiac disease have similarly been shown to improve following rituximab therapy in 2 cases of women with celiac disease who were treated to manage concurrent Sjögren syndrome and celiac-associated arthritis . These cases, in addition to ours, suggest that rituximab may be a potential treatment for both intraintestinal and extraintestinal manifestations of celiac disease, including DH.…”

Section: Discussionsupporting

confidence: 63%

Rituximab Treatment for Recalcitrant Dermatitis Herpetiformis

et al. 2017

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ermatitis herpetiformis (DH) is an autoimmune blistering disease (AIBD) characterized by pruritic papulovesicles mostly on extensor surfaces. Conventional histologic analysis of lesional skin displays neutrophilic collections within the dermal papillae with associated subepidermal clefting. Direct immunofluorescence of nonlesional skin demonstrates granular deposits of IgA in dermal papillae and the basement membrane zone. IgA deposited in the skin is antiepidermal transglutaminase 3 (TG3). IgA serum antibodies are directed against TG3, as well as tissue transglutaminase (TG2), the latter of which is a reflection of the associated celiac disease. 1 Treatment is primarily aimed at maintaining a glutenfree diet (GFD), with initiation of dapsone therapy for refractory disease. In some cases, immune-suppressing agents are required for patients not responding to conventional therapy.We present a patient with a recalcitrant case of DH who was treated with rituximab. The patient developed clinical and serological remission with normalization of both IgA TG2 and TG3 antibody levels. He has remained symptom-free for 18 months following treatment. Report of a CaseA man in his 80s presented with a 5-year history of worsening widespread pruritic rash consisting of coalescent erythematous scaling plaques over the arms, legs, back, and buttocks with intermittent scattered vesicles (Figure 1A and B). His only medications were losartan potassium and carvedilol. Conven-tional histologic analysis demonstrated focal subepidermal blister formation with a neutrophil-predominant inflammatory infiltrate, with scattered eosinophils and lymphocytes. Collections of neutrophils in dermal papillae, papillary dermal microabscesses, were noted (Figure 2A). Immunostaining for B lymphocytes had essentially negative results (Figure 2B). Direct immunofluorescence demonstrated granular deposition of IgA in dermal papillae, diagnostic of DH (Figure 2C). Due to the patient's advanced age of presentation, repeated direct immunofluorescence was independently performed, which again confirmed DH. He denied gastrointestinal symptoms and began a GFD along with dapsone, 50 mg/d, but his pruritic rash persisted. With concern for worsening anemia, dapsone therapy was discontinued. Sulfasalazine treatment was initiated at 3 g/d but also was discontinued due to gastrointestinal symptoms. After stopping sulfasalazine, his disease worsened and he developed debilitating pruritus. The GFD was again recommended, but the patient was unable to remain adherent. He started a tapering course of prednisone from 40 to 10 mg/d along with azathioprine, titrated up to 2.5 mg/kg/d. His skin continued to worsen over the subsequent months. Given the demonstrated efficacy of rituximab in other AIBDs and the failure of conventional therapy in this patient, a trial of rituximab was initiated using 4 weekly doses (375 mg/m 2 ) while continuing prednisone, 10 mg/d, and azathioprine, 2.5 mg/kg/d. Pretreatment laboratory tests were performed, including IgG (771 mg/ dL; to convert to g...

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Section: Discussionsupporting

confidence: 63%

Rituximab Treatment for Recalcitrant Dermatitis Herpetiformis

et al. 2017

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“…Case report studies suggest that rituximab for celiac disease may be successful. 1,2 Similarly, Go et al 3 showed that refractory cases of AIHA may be treated with rituximab with varying degrees of success.…”

Section: Discussionmentioning

confidence: 99%

Rituximab treatment for dermatitis herpetiformis in the setting of type 1 diabetes mellitus, celiac disease, vitiligo, autoimmune hemolytic anemia, and autoimmune thrombocytopenia

2020

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“…CD20 is a B cell marker, and B cells are involved in the pathogenesis of CeD due to the production of various antibodies ( Figure 2 , point 8). Two case reports have shown a clinical and biological improvement in CeD in patients treated with Rituximab, but clinical trials are missing [ 125 , 126 ].…”

Section: Potential Therapeutic Targets In Celiac Diseasementioning

confidence: 99%

Emerging Pharmaceutical Therapies to Address the Inadequacy of a Gluten-Free Diet for Celiac Disease

Crepaldi,

Palo,

Maniero

et al. 2023

Pharmaceuticals

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Celiac disease (CeD) is a chronic autoimmune disorder triggered by the ingestion of gluten, affecting around 1% of the global population. It is a multifactorial disease involving both genetics and environmental factors. Nowadays, the only available treatment for CeD is a life-long gluten-free diet (GFD), which can cause a significant burden for patients, since symptoms and mucosal injury can persist despite apparent compliance with a GFD. This could also lead to psychological consequences and affect the quality of life of these patients. Thankfully, recent advances in understanding the pathogenesis of CeD and the availability of various targets have made it feasible to explore pharmaceutical treatments specific to CeD. Recently, the FDA has highlighted the unmet needs of adult patients on a GFD who experience ongoing symptoms attributed to CeD and also show persistent duodenal villous atrophy. This review will outline the limitations of a GFD, describe the targets of potential novel treatment of CeD and provide an overview of the primary clinical trials involving oral and injectable agents for a non-dietary treatment of CeD.

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First report of improvement of coeliac disease in a patient with Sjogren's syndrome treated with rituximab

Cited by 7 publications

References 1 publication

Rituximab Treatment for Recalcitrant Dermatitis Herpetiformis

Rituximab Treatment for Recalcitrant Dermatitis Herpetiformis

Rituximab treatment for dermatitis herpetiformis in the setting of type 1 diabetes mellitus, celiac disease, vitiligo, autoimmune hemolytic anemia, and autoimmune thrombocytopenia

Emerging Pharmaceutical Therapies to Address the Inadequacy of a Gluten-Free Diet for Celiac Disease

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