Autoimmune Enteropathy in a Pediatric Patient: Partial Response to Tacrolimus Therapy

Steffen, Rita; Wyllie, Robert; Kay, Marsha; Kyllonen, Kay; Gramlich, Terry; Petras, Robert E.

doi:10.1177/000992289703600508

Cited by 14 publications

(7 citation statements)

References 25 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…49 In this study, one patient with pyoderma gangrenosum improved significantly with oral tacrolimus therapy (Figure 4). There has also been one report on the successful treatment of autoimmune enteropathy 50 …”

Section: Discussionmentioning

confidence: 99%

Rescue therapy with tacrolimus is effective in patients with severe and refractory inflammatory bowel disease

Baumgart

Wiedenmann

Dignaß

2003

Aliment Pharmacol Ther

119

View full text Add to dashboard Cite

Summary Background : Oral tacrolimus, approved for the prophylaxis of organ rejection in liver or kidney transplants, has been reported to be effective in anecdotal cases of refractory inflammatory bowel disease. Aim : To evaluate the usefulness of low‐dose oral tacrolimus in refractory inflammatory bowel disease. Methods: Thirty‐one adult Caucasian patients with steroid‐dependent (n = 15) or steroid‐refractory (n = 16) inflammatory bowel disease (Crohn's disease, n = 6; ulcerative colitis, n = 23; pouchitis, n = 2) were enrolled. Tacrolimus (0.1 mg/kg body weight per day) was administered orally in 30 patients and initially intravenously in one patient (0.01 mg/kg body weight per day), aiming for serum trough levels of 4–6 ng/mL. The median treatment duration was 12 months (range, 1–137 months). Results : Twenty‐eight patients (90.3%) experienced a clinical and laboratory response and 20 (64.5%) went into remission. One ulcerative colitis patient and two Crohn's disease patients did not improve. Three ulcerative colitis patients (9.7%) were colectomized at 1, 12 and 24 months after tacrolimus initiation. In 19 of 23 patients (82.6%) taking steroids, steroids were reduced or discontinued. Side‐effects included a temporary rise of creatinine (n = 3, 9.7%), tremor or paraesthesias (n = 3, 9.7%), hyperkalaemia (n = 1, 3.2%), hypertension (n = 1, 3.2%) and an opportunistic infection (n = 1, 3.2%). Conclusion : Oral tacrolimus is safe and effective in refractory inflammatory bowel disease.

show abstract

Section: Discussionmentioning

confidence: 99%

Rescue therapy with tacrolimus is effective in patients with severe and refractory inflammatory bowel disease

Baumgart

Wiedenmann

Dignaß

2003

Aliment Pharmacol Ther

119

View full text Add to dashboard Cite

show abstract

“…More than half of those patients, however, required further therapy. Early trials of Cimetidine were unsuccessful [11,15] whereas the use of corticosteroids alone seems to offer little benefit [13,14,[18][19][20][21]29,30,[32][33][34][35]. Addition of Azathioprine [2,15,16,22,24] and/or Cyclophosphamide [10,12,15,16,20,21,28,30,31] has met with mitigated success.…”

Section: Treatment and Prognosismentioning

confidence: 99%

“…However, some patients have been reported to be resistant to Cyclosporine therapy [21,24,31]. Recently, Tacrolimus has been reported in patients previously resistant to other forms of immunotherapy [34,36].…”

Section: Treatment and Prognosismentioning

confidence: 99%

Autoimmune Enteropathy

et al. 1999

View full text Add to dashboard Cite

show abstract

“…In one previous case report, MMF was used to induce remission of AE 17. Reports about tacrolimus treatment in AE are also found 7 8 18 19. The need for potent immune suppressants in addition to corticosteroids demonstrates the dependency of massive immunosuppression to treat and prevent relapse of this disease.…”

Section: Discussionmentioning

confidence: 99%

Wireless capsule endoscopy as a tool in diagnosing autoimmune enteropathy

Gram-Kampmann¹,

Lillevang

Detlefsen

et al. 2015

BMJ Case Reports

View full text Add to dashboard Cite

Autoimmune enteropathy (AE) is an immune mediated illness of the intestinal mucosa. The cause is unknown, and the diagnosis is based on typical characteristics displayed. There is no gold standard for treatment. We present two adult cases of AE and demonstrate the challenges in establishing the diagnosis. The extensive diagnostic work up excluded other more common causes of protracted diarrhoea. Wireless capsule endoscopy (WCE) displayed universal small intestinal mucosal damage with shortened villi that led to the suspicion of AE in both patients. The diagnosis was confirmed with microscopy, showing shortened villi, villous blunting and hyperplasia of crypts in both patients. In one patient, deep crypt lymphocytosis with minimal intraepithelial lymphocytosis was found as well. Both patients were successfully treated with high-dose immunosuppressant therapy to induce and maintain remission. Use of WCE as a diagnostic tool was invaluable in establishing the diagnosis of AE.

show abstract

Autoimmune Enteropathy in a Pediatric Patient: Partial Response to Tacrolimus Therapy

Cited by 14 publications

References 25 publications

Rescue therapy with tacrolimus is effective in patients with severe and refractory inflammatory bowel disease

Rescue therapy with tacrolimus is effective in patients with severe and refractory inflammatory bowel disease

Autoimmune Enteropathy

Wireless capsule endoscopy as a tool in diagnosing autoimmune enteropathy

Contact Info

Product

Resources

About