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Blood, 1 March 2007, Vol. 109, No. 5, pp. 2243-2249.
Prepublished online as a Blood First Edition Paper on October 26, 2006; DOI 10.1182/blood-2006-08-042820.
 Previous Article | Next Article 
Submitted August 24, 2006
Accepted October 11, 2006
Autologous hematopoietic stem cell transplantation in
refractory celiac disease with aberrant T-cells
Abdulbaqi Al-toma, Otto J Visser, Hyacintha M van Roessel, B. Mary E von Blomberg, Wieke H.M. Verbeek, Petra E.T. Scholten, Gert J Ossenkoppele, Peter C Huijgens, and Chris J.J. Mulder*
Dept of Gastroenterology, VU University Medical Center, Amsterdam, Netherlands
Dept of Hematology, VU University Medical Center, Amsterdam, Netherlands
Dept of Clinical Pathology, VU University Medical Center, Amsterdam, Netherlands
* Corresponding author; email: cjmulder{at}vumc.nl.
Autologous Hematopoietic Stem Cell Transplantation (ASCT) is an increasingly accepted treatment for refractory autoimmune diseases. Refractory Celiac Disease with aberrant T cells (RCD-type II) is unresponsive to available therapies and carries a high risk of transition into Enteropathy Associated T-cell Lymphoma (EATL). This study reports on the feasibility, safety and efficacy of ASCT in RCD type II.
Thirteen patients with RCD-II were evaluated. Seven patients [4M, 3 F, mean age 61.5 years (range 51-69 years)] were transplanted. After conditioning with fludarabine and melphalan, ASCT was performed. Patients were monitored for response, adverse effects and hematopoietic reconstitution.
All 7 patients completed the mobilization and leucopheresis procedures successfully and subsequently received conditioning and transplantation. Engraftment occurred in all patients. No major non-hematological toxicity or transplantation-related mortality was observed. There was a significant reduction in the aberrant T-cells in duodenal biopsies associated with improvement in clinical wellbeing, and normalization of hematological and biochemical markers (mean follow-up 15.5 months, range 7-30 months). One patient died 8 months post-transplant form progressive neuroceliac disease.
These preliminary results showed that high-dose chemotherapy followed by ASCT seems feasible and safe, and might result in long-term improvement of RCD II patients whose condition did not respond promptly to available drugs.
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