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Springerplus. 2014 Oct 04;3:581. doi: 10.1186/2193-1801-3-581. eCollection 2014.

A case of post-transplant adult T-cell leukemia/lymphoma presenting myelopathy similar to but distinct from human T-cell leukemia virus type I (HTLV- I)-associated myelopathy.

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Toyotaka Kawamata, Nobuhiro Ohno, Kota Sato, Masayuki Kobayashi, Norihide Jo, Koichiro Yuji, Ryuji Tanosaki, Yoshihisa Yamano, Arinobu Tojo, Kaoru Uchimaru

Affiliations

  1. Department of Hematology/Oncology, Research Hospital, The Institute of Medical Science, the University of Tokyo, 4-6-1 Shirokanedai, Minato-ku, Tokyo, 108-8639 Japan.
  2. Department of Hematology/Oncology, Research Hospital, The Institute of Medical Science, the University of Tokyo, 4-6-1 Shirokanedai, Minato-ku, Tokyo, 108-8639 Japan ; Division of Molecular Therapy, Department of The advanced Clinical Research Center, The Institute of Medical Science, the University of Tokyo, 4-6-1 Shirokanedai, Minato-ku, Tokyo, 108-8639 Japan.
  3. Department of Blood Transfusion and Cellular Therapy, National Cancer Center Hospital, 5-1-1 Tsukiji, Chuo-ku, Tokyo, 104-0045 Japan.
  4. Department of Rare Diseases Research, Institute of Medical Science, St. Marianna University Graduate School of Medicine, Sugao, Miyamae-ku, Kawasaki, Kanagawa, 216-8512 Japan.

PMID: 25332881 PMCID: PMC4197197 DOI: 10.1186/2193-1801-3-581

Abstract

INTRODUCTION: Adult T-cell leukemia/lymphoma (ATL) responds poorly to conventional chemotherapy, but allogeneic stem cell transplantation (allo-SCT) may improve disease prognosis. Herein, we report a female patient with human T-cell leukemia virus type I (HTLV-I)-associated myelopathy (HAM)-like myelopathy following allo-SCT for ATL.

CASE REPORT: She developed crural paresis 14 months after allo-SCT. Initially, she was diagnosed with central nervous system (CNS) relapse of ATL and treated with intrathecal injection and whole brain and spine irradiation. Her symptoms recurred 5 months later, when a cerebrospinal fluid (CSF) specimen showed increased CD4 + CXCR3 + CCR4+ cell numbers and levels of neopterin and CXCL10 (IP-10).

DISCUSSION: These results suggest the possible involvement of a certain immunological mechanism such as HAM in her symptoms, irrespective of the lack of anti-HTLV-I antibody in her CSF. Because a definitive diagnosis of CNS manifestation of ATL is sometimes difficult, multi-modal laboratory data are required for differential diagnosis.

Keywords: Adult T-cell leukemia/lymphoma; CXCL10 (IP-10); HTLV-I-associated myelopathy (HAM); Neopterin; Post-transplant myelopathy

References

  1. J Clin Oncol. 2007 Dec 1;25(34):5458-64 - PubMed
  2. Jpn J Ophthalmol. 1989;33(1):1-12 - PubMed
  3. Br J Haematol. 2003 Jan;120(2):304-9 - PubMed
  4. J Neurol Sci. 1998 Jun 30;158(2):226-31 - PubMed
  5. J Immunol. 2009 Feb 1;182(3):1541-7 - PubMed
  6. Proc Natl Acad Sci U S A. 1980 Dec;77(12):7415-9 - PubMed
  7. Am J Ophthalmol. 1992 Aug 15;114(2):123-9 - PubMed
  8. Am J Trop Med Hyg. 1990 Apr;42(4):365-73 - PubMed
  9. J Clin Virol. 2010 Aug;48(4):288-90 - PubMed
  10. Biol Blood Marrow Transplant. 2006 Jun;12(6):623-34 - PubMed
  11. Bone Marrow Transplant. 2001 Jan;27(1):15-20 - PubMed
  12. Bone Marrow Transplant. 2007 Jan;39(1):49-52 - PubMed
  13. Bone Marrow Transplant. 1997 Sep;20(5):365-8 - PubMed
  14. Transplantation. 1984 Nov;38(5):497-500 - PubMed
  15. Neuroradiology. 2002 Jul;44(7):559-67 - PubMed
  16. Lancet. 1986 May 3;1(8488):1031-2 - PubMed
  17. PLoS Negl Trop Dis. 2013 Oct 10;7(10):e2479 - PubMed
  18. Exp Dermatol. 2012 Nov;21(11):815-21 - PubMed
  19. J Neuroimmunol. 2001 Mar 1;114(1-2):207-12 - PubMed
  20. Blood. 1977 Sep;50(3):481-92 - PubMed
  21. J Clin Oncol. 1988 Jul;6(7):1088-97 - PubMed
  22. J Clin Invest. 2014 Aug;124(8):3431-42 - PubMed
  23. Proc Natl Acad Sci U S A. 1981 Oct;78(10):6476-80 - PubMed
  24. J Clin Oncol. 2009 Jan 20;27(3):453-9 - PubMed
  25. Br J Haematol. 2005 Aug;130(4):511-5 - PubMed
  26. Leuk Lymphoma. 2000 Aug;38(5-6):533-40 - PubMed

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