Up to 5%–15% of patients with ulcerative colitis (UC) require surgical treatment, with proctocolectomy with ileoanal pouch being the technique of choice. Pouchitis is its most common complication. Pouchitis includes a range of situations that remain ill-defined, ranging from classical pouchitis to Crohn's disease of the pouch.1 The increasing incidence of this complication and the risks and loss of quality of life associated with the extraction of the pouch have increased interest in finding therapeutic alternatives in this situation.2 We present the case of a patient with a pouch and chronic iron deficiency anaemia secondary to patchy inflammatory involvement of the pouch who received rescue treatment with tofacitinib after failure of treatment with TNF inhibitors.
This was a 48-year-old male patient with left-sided UC of 18 years of evolution requiring proctocolectomy with pouch due to refractory disease. Five years after surgery, there was onset of rectorrhagia and severe iron deficiency anaemia. Hence, a pouchoscopy revealed deep, friable ulcers in the pouch, causing stenosis of the access to afferent and efferent loops. Other causes of anaemia were ruled out (negative coeliac disease study, gastroscopy and capsule endoscopy without findings) and, given the characteristics of the lesions, it was decided to start treatment with oral budesonide with no clinical response, so combined treatment with infliximab (5mg/kg every eight weeks) and azathioprine was started. Despite this, the patient required high-dose intravenous iron repletion and even periodic blood transfusion, with no endoscopic improvement, so infliximab was intensified to every six weeks. However, in successive endoscopies, ulcerated stenosis of the access orifice to afferent and efferent loops persisted, so local injection of infliximab and antibiotic therapy with metronidazole 500mg/every eight hours for four weeks was administered without success. Given the patient's reluctance to have the pouch removed and the lack of therapeutic alternatives in this situation, treatment with infliximab was maintained for 10 years, with a progressive increase in the requirement for intravenous iron therapy and blood transfusions in the last two years (Fig. 1).
After another pouchoscopy in which no changes were observed, the anaemia was exacerbated with haemoglobin of 7g/dl. Resection of the pouch and end ileostomy were considered, but given the patient's reluctance to undergo further surgery, rescue treatment with tofacitinib was agreed on, following the guidelines approved for UC (10mg every 12h for eight weeks, followed by 5mg/12h), with the patient presenting with frank clinical improvement (only some isolated rectorrhagia). At five months after the start of treatment, the anaemia resolved for the first time in the last 15 months (Fig. 1). At one year of follow-up, only 1g of intravenous iron was required without transfusion support, although there was no endoscopic improvement.
Pouchitis occurs in up to 30% of patients with pouches. The therapeutic requirements of pouchitis are high, with up to 80% of patients requiring biologics and 15%–30%, surgery. However, TNF inhibitors have been shown to be effective in only half of patients with pouchitis and this, together with the desire of most patients to avoid having to have the pouch removed, requires a search for effective therapeutic alternatives. Although we do not have controlled clinical trials comparing the efficacy of tofacitinib (JAK1 and JAK3 inhibitor, approved for UC without response to conventional therapy) with other drugs, a recent meta-analysis equates it in efficacy to other biologic therapies in the treatment of UC. Additionally, high response rates to tofacitinib have been reported in patients with UC refractory to biologic treatment. In this scenario, the lack of controlled clinical trials in treating pouchitis has traditionally demanded the use of drugs that have been effective in ulcerative colitis, so the use of tofacitinib seems reasonable. To date, there are only nine cases of chronic refractory pouchitis treated with tofacitinib in the literature.3–5
