A significant obstacle to effective treatment of autoimmune enteropathy and Inflammatory Bowel Disease (IBD) in Common Variable Immunodeficiency (CVID) is that both may be refractory to guideline-directed medical management. In this case report, utilization of immunologic testing to characterize activity along JAK/STAT signaling pathways revealed increased activity. As a result, Tofacitinib was successfully initiated to block downstream JAK1/3 signaling in a patient with disease previously refractory to anti-TNF-alpha and anti Il12/23 biologics.

Strongyloidiasis is a disease caused by Strongyloides stercoralis, is an endemic nematode in countries with a tropical and subtropical climate, sometimes the infection is asymtomatic but in other cases it can produce a hyperinfection syndrome, Which is characteristic of many more severe symptoms, such as severe respiratory distress, coughing up blood, events that can lead the person to be critically ill, the risk factors for this syndrome are: immunosuppression in general either due to HIV, transplantation, use of drugs, oncological diseases. Normally in the infection that is not so severe, anthelmintics such as albendazole can be used, but in severe cases there are studies with subcutaneous ivermectin in cases in which it cannot be administered orally or that simply has not worked.

Background: We report a rare case of a 50 year old Chinese female with cutaneous Sweet Syndrome (SS) presenting with iron deficiency anaemia who was subsequently found to have small bowel ulcers with a dense neutrophilic infiltrate on histology, with no evidence of Inflammatory Bowel Disease (IBD).

Case presentation: The 50 year old lady underwent an extensive workup including enteroscopy, blood investigations and exclusion of pertinent differentials, she was diagnosed with gut SS. After trying and failing various treatment modalities, she was successfully treated with a combination of subcutaneous adalimumab and oral Methotrexate (MTX). Gut SS is an extremely rare disease with only 2 published cases thus far and both were treated with mainly steroids. To our knowledge, this is the first reported case of this treatment combination of an anti TNF agent and MTX for recalcitrant gut and cutaneous SS.

Conclusion: A diagnosis of gut SS should be considered in the presence of gastrointestinal pathology such as ulcers, especially if histological features of inflammatory bowel disease are absent. In recalcitrant cases anti-TNF (tumor necrosis factor) agents may be an alternative treatment option.