Sacroiliitis is the most pathognomonic and earliest manifestation of ankylosing spondylitis.We herein report a 28-year-old female patient who presented with sacroiliitis as an initial manifestation of acute myelogenous leukemia (AML). She had a 3-month history of anemia and walking difficulty. Bone marrow findings revealed an increase of blasts with trilineage dysplasia. Although she was initially diagnosed with myelodysplastic syndrome (MDS), blasts rapidly increased and AML developed 1 month after the diagnosis of MDS with sacroiliitis. Induction chemotherapy failed to induce a complete remission of AML, but it did effectively treat the sacroiliitis. However, the sacroiliitis relapsed when the leukemia cells progressed thereafter. Oral corticosteroids helped ameliorate the sacroiliitis. She underwent bone marrow transplantation (BMT) from an HLA-identical sister during a nonremission period; however, the leukemic cells began to rapidly increase from day 30 after BMT. The close relationship between the occurrence of sacroiliitis and AML suggested that autoimmune sacroiliitis was a paraneoplastic phenomenon of AML in this patient. Although autoimmune disorders develop in a substantial number of MDS patients, they are rarely observed in de novo AML. No previous report has described sacroiliitis as the initial manifestation of de novo AML.
This is a preview of subscription content, log in to check access.
Buy single article
Instant access to the full article PDF.
Price includes VAT for USA
Subscribe to journal
Immediate online access to all issues from 2019. Subscription will auto renew annually.
This is the net price. Taxes to be calculated in checkout.
van der Linden S, Valkenburg HA, Cats A. Evaluation of diagnostic criteria for ankylosing spondylitis. A proposal for modification of New York criteria. Arthritis Rheum. 1984;27:361–368.
Yamaguchi A, Tsuchiya N, Mitsui H, et al. Association of HLA-B39 with HLA-B27-negative ankylosing spondylitis and pauciarticular juvenile rheumatoid arthritis in Japanese patients. Evidence for a role of peptide-anchoring B pocket. Arthritis Rheum. 1995;38:1672–1677.
Tanvetyanon T, Stiff P. Recurrent steroid-responsive pancreatitis associated with myelodysplastic syndrome and transformations. Leuk Lymphoma. 2005;46:151–154.
Koch B, Kranzhofer N, Pfreundschu M, Pees HW, Trumper L. First manifestations of seronegative spondylarthropathy following autologous stem cell transplantation in HLA-B27-positive patients. Bone Marrow Transplant. 2000;26:673–675.
Bakland G, Nossent H. Acute myelogenous leukaemia following etanercept therapy. Rheumatology. 2003;42:900–917.
Au WY, Hawkins BR, Cheng N, Lie AKW, Liang R, Kwong YL. Risk of haematological malignancies in HLA-B27 carriers. Br J Haematol. 2001;115:320–322.
Hamblin TJ. Immunological abnormalities in myelodysplastic syndromes. Semin Hematol. 1996;33:150–162.
Enright H, Jacob HS, Vercellotti G, Howe R, Belzer M, Miller W. Paraneoplastic autoimmune phenomena in patients with myelodysplastic syndromes: response to immunosuppressive therapy. Br J Haematol. 1995;91:403–408.
Castro M, Conn DL, Su WP, Garton JP. Rheumatic manifestations in myelodysplastic syndromes. J Rheumatol. 1991;18:721–727.
Billstrom R, Johansson H, Johansson B, Mitelman F. Immune-mediated complications in patients with myelodysplastic syndromes—clinical and cytogenetic features. Eur J Haematol. 1995;55:42–48.
Okamoto T, Okada M, Mori A, et al. Correlation between immunological abnormalities and prognosis in myelodysplastic syndrome patients. Int J Hematol. 1997;66:345–351.
Mufti GJ, Figes A, Hamblin TJ, Oscier DG, Copplestone JA. Immunological abnormalities in myelodysplastic syndromes. Serum immunoglobulins and autoantibodies. Br J Haematol. 1986;63:143–147.
Economopoulos T, Economidou J, Giannopoulos G, et al. Immune abnormalities in myelodysplastic syndromes. J Clin Pathol. 1985;38:908–911.
About this article
Cite this article
Hoshino, T., Matsushima, T., Saitoh, Y. et al. Sacroiliitis as an Initial Manifestation of Acute Myelogenous Leukemia. Int J Hematol 84, 421–424 (2006). https://doi.org/10.1532/IJH97.06106