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Pulmonary Leukocytoclastic Vasculitis as an Initial Presentation of Myelodysplastic Syndrome

  • Lee, Seung Hyun (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Kim, Jae Hyung (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Park, Sejin (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Won, Chang Youn (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Lee, Joo-Hyun (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Yi, Seong Yoon (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Park, Hye Kyeong (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Chang, Sun Hee (Department of Pathology, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Jung, Hoon (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Lee, Sung-Soon (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine) ;
  • Koo, Hyeon-Kyoung (Department of Internal Medicine, Inje University Ilsan Paik Hospital, Inje University College of Medicine)
  • 투고 : 2015.08.05
  • 심사 : 2016.08.10
  • 발행 : 2016.10.05

초록

Systemic vasculitis involving the lung is a rare manifestation of myelodysplastic syndrome (MDS), and secondary vasculitis is considered to have poor prognosis. A 44-year-old man presented with fever and dyspnea of 1 month duration. A chest radiograph revealed bilateral multiple wedge shaped consolidations. In addition, the results of a percutaneous needle biopsy for non-resolving pneumonia were compatible with pulmonary vasculitis. Bone marrow biopsy was performed due to the persistence of unexplained anemia and the patient was diagnosed with MDS. We reported a case of secondary vasculitis presenting as non-resolving pneumonia, later diagnosed as paraneoplastic syndrome of undiagnosed MDS. The cytopenia and vasculitis improved after a short course of glucocorticoid treatment, and there was no recurrence despite the progression of underlying MDS.

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참고문헌

  1. Dreyfus B, Vernant JP, Wechsler J, Imbert M, De Prost Y, Reyes F, et al. Refractory anaemia with an excess of myeloblasts and cutaneous vasculitis (author's transl). Nouv Rev Fr Hematol 1981;23:115-21.
  2. 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;91:403-8. https://doi.org/10.1111/j.1365-2141.1995.tb05310.x
  3. Giannouli S, Voulgarelis M, Zintzaras E, Tzioufas AG, Moutsopoulos HM. Autoimmune phenomena in myelodysplastic syndromes: a 4-yr prospective study. Rheumatology (Oxford) 2004;43:626-32. https://doi.org/10.1093/rheumatology/keh136
  4. Belizna C, Subra JF, Henrion D, Ghali A, Renier G, Royer M, et al. Prognosis of vasculitis associated myelodysplasia. Autoimmun Rev 2013;12:943-6. https://doi.org/10.1016/j.autrev.2013.01.005
  5. de Hollanda A, Beucher A, Henrion D, Ghali A, Lavigne C, Levesque H, et al. Systemic and immune manifestations in myelodysplasia: a multicenter retrospective study. Arthritis Care Res (Hoboken) 2011;63:1188-94. https://doi.org/10.1002/acr.20504
  6. Enright H, Miller W. Autoimmune phenomena in patients with myelodysplastic syndromes. Leuk Lymphoma 1997;24:483-9. https://doi.org/10.3109/10428199709055585
  7. van Rijn RS, Wittebol S, Graafland AD, Kramer MH. Immunologic phenomena as the first sign of myelodysplastic syndrome. Ned Tijdschr Geneeskd 2001;145:1529-33.
  8. Al Ustwani O, Ford LA, Sait SJ, Block AM, Barcos M, Vigil CE, et al. Myelodysplastic syndromes and autoimmune diseases: case series and review of literature. Leuk Res 2013;37:894-9. https://doi.org/10.1016/j.leukres.2013.04.007
  9. Saif MW, Hopkins JL, Gore SD. Autoimmune phenomena in patients with myelodysplastic syndromes and chronic myelomonocytic leukemia. Leuk Lymphoma 2002;43:2083-92. https://doi.org/10.1080/1042819021000016186
  10. Braun T, Fenaux P. Myelodysplastic syndromes (MDS) and autoimmune disorders (AD): cause or consequence? Best Pract Res Clin Haematol 2013;26:327-36. https://doi.org/10.1016/j.beha.2013.09.003
  11. Ma X, Does M, Raza A, Mayne ST. Myelodysplastic syndromes: incidence and survival in the United States. Cancer 2007;109:1536-42. https://doi.org/10.1002/cncr.22570
  12. Jin J, Yu M, Hu C, Ye L, Xie L, Chen F, et al. Alcohol consumption and risk of myelodysplastic syndromes: a meta-analysis of epidemiological studies. Mol Clin Oncol 2014;2:1115-20. https://doi.org/10.3892/mco.2014.376
  13. Du Y, Fryzek J, Sekeres MA, Taioli E. Smoking and alcohol intake as risk factors for myelodysplastic syndromes (MDS). Leuk Res 2010;34:1-5. https://doi.org/10.1016/j.leukres.2009.08.006
  14. Kiladjian JJ, Visentin G, Viey E, Chevret S, Eclache V, Stirnemann J, et al. Activation of cytotoxic T-cell receptor gammadelta T lymphocytes in response to specific stimulation in myelodysplastic syndromes. Haematologica 2008;93:381-9. https://doi.org/10.3324/haematol.11812
  15. Kordasti SY, Ingram W, Hayden J, Darling D, Barber L, Afzali B, et al. CD4+CD25high Foxp3+ regulatory T cells in myelodysplastic syndrome (MDS). Blood 2007;110:847-50. https://doi.org/10.1182/blood-2007-01-067546

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