Successful management of multilineage autoimmune cytopenia complicated with severe infection and deep vein thrombosis in a patient with Hodgkin lymphoma post-autologous hematopoietic stem cell transplantation
Wan-Yi Hsu
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorShyh-Shin Chiou
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Department of Pediatrics, School of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan
Search for more papers by this authorYu-Mei Liao
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorHsiu-Lan Shu
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Department of Nursing, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorYu-Sheng Zeng
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorCheong-Chew Wong
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorCorresponding Author
Pei-Chin Lin
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Department of Pediatrics, School of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan
Graduate Institute of Clinical Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan
Pei-Chin Lin, Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, No.100, Ziyou 1st Road., Sanmin Dist., Kaohsiung City 807, Taiwan (R.O.C.)
Tel: +886-7-3121101 ext. 5838
Fax: +886-7-3117752
E-mail: [email protected]
Search for more papers by this authorWan-Yi Hsu
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorShyh-Shin Chiou
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Department of Pediatrics, School of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan
Search for more papers by this authorYu-Mei Liao
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorHsiu-Lan Shu
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Department of Nursing, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorYu-Sheng Zeng
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorCheong-Chew Wong
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Search for more papers by this authorCorresponding Author
Pei-Chin Lin
Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
Department of Pediatrics, School of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan
Graduate Institute of Clinical Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan
Pei-Chin Lin, Division of Hematology and Oncology, Department of Pediatrics, Kaohsiung Medical University Hospital, No.100, Ziyou 1st Road., Sanmin Dist., Kaohsiung City 807, Taiwan (R.O.C.)
Tel: +886-7-3121101 ext. 5838
Fax: +886-7-3117752
E-mail: [email protected]
Search for more papers by this authorAbstract
Autoimmune cytopenia (AIHA, AITP or AIN) were uncommon paraneoplastic manifestations of HL and have been recognized in patients after HSCT with dismal outcome. We presented a case of 16-yr-old male with Hodgkin's lymphoma who developed severe AIC involving all three cell lineages after autologus bone marrow transplantation. No disease relapse was noted. Treatments with steroid, IVIG and immunosuppresants were in vain and the disease course was complicated with sepsis and deep vein thrombosis. Rituximab was administered along with broad-spectrum antibiotics and low-molecular weight heparin. The condition became stable and pancytopenia recovered after four doses of rituximab treatment. Severe multi-lineage AIC post HSCT is usually refractory to first-line treatment and difficult to manage. Second-line treatment, such as rituximab, and dedicated care for pancytopenia-induced or treatment-related complications may provide a better outcome.
References
- 1Cecinati V, Brugnoletti F, D'Angio M, et al. Autoimmune hemolytic anemia and immune thrombocytopenia as unusual presentations of childhood Hodgkin lymphoma: A case report and review of the literature. J Pediatr Hematol Oncol 2012: 34: 280–282.
- 2Costello RT, Xerri L, Bouabdallah R, Gastaut JA, Sainty D. Leukopenia, thrombocytopenia, and acute autoimmune hemolytic anemia associated with an unusual (type 2/4) Hodgkin's disease: Case report. Am J Hematol 1996: 52: 333–334.
10.1002/(SICI)1096-8652(199608)52:4<333::AID-AJH23>3.0.CO;2-D CAS PubMed Web of Science® Google Scholar
- 3Heyman MR, Walsh TJ. Autoimmune neutropenia and Hodgkin's disease. Cancer 1987: 59: 1903–1905.
10.1002/1097-0142(19870601)59:11<1903::AID-CNCR2820591110>3.0.CO;2-M CAS PubMed Web of Science® Google Scholar
- 4Lechner K, Chen YA. Paraneoplastic autoimmune cytopenias in Hodgkin lymphoma. Leuk Lymphoma 2010: 51: 469–474.
- 5Jonathan Brammer MS. Cold-agglutinin hemolytic anemia: A harbinger of recurrent lymphoma. Clin Lymphoma Myeloma Leuk 2011: 11: S132.
10.1016/j.clml.2011.05.029 Google Scholar
- 6Sanz J, Arriaga F, Montesinos P, et al. Autoimmune hemolytic anemia following allogeneic hematopoietic stem cell transplantation in adult patients. Bone Marrow Transplant 2007: 39: 555–561.
- 7Rovira J, Cid J, Gutierrez-Garcia G, et al. Fatal immune hemolytic anemia following allogeneic stem cell transplantation: Report of 2 cases and review of literature. Transfus Med Rev 2013: 27: 166–170.
- 8Dimou M, Angelopoulou MK, Pangalis GA, et al. Autoimmune hemolytic anemia and autoimmune thrombocytopenia at diagnosis and during follow-up of Hodgkin lymphoma. Leuk Lymphoma 2012: 53: 1481–1487.
- 9Varoczy L, Gergely L, Zeher M, Szegedi G, Illes A. Malignant lymphoma-associated autoimmune diseases–a descriptive epidemiological study. Rheumatol Int 2002: 22: 233–237.
- 10Poppema S, Potters M, Emmens R, Visser L, van den Berg A. Immune reactions in classical Hodgkin's lymphoma. Semin Hematol 1999: 36: 253–259.
- 11Skinnider BF, Mak TW. The role of cytokines in classical Hodgkin lymphoma. Blood 2002: 99: 4283–4297.
- 12Saeed M, Rubinger M, Ahsanuddin A, et al. If at first you don't succeed: Try, try again. Am J Hematol 2012: 87: 295–297.
- 13Keung YK, Cobos E, Bolanos-Meade J, Issarachai S, Brideau A, Morgan D. Evans syndrome after autologous bone marrow transplant for recurrent Hodgkin's disease. Bone Marrow Transplant 1997: 20: 1099–1101.
- 14Stern M, Buser AS, Lohri A, Tichelli A, Nissen-Druey C. Autoimmunity and malignancy in hematology–more than an association. Crit Rev Oncol Hematol 2007: 63: 100–110.
- 15Michel M. Classification and therapeutic approaches in autoimmune hemolytic anemia: An update. Expert Rev Hematol 2011: 4: 607–618.
- 16Cooper N. A review of the management of childhood immune thrombocytopenia: How can we provide an evidence-based approach? Br J Haematol 2014: 165: 756–767.
- 17Bluml S, McKeever K, Ettinger R, Smolen J, Herbst R. B-cell targeted therapeutics in clinical development. Arthritis Res Ther 2013: 15(Suppl 1): S4.
- 18Pescovitz MD. Rituximab, an anti-cd20 monoclonal antibody: History and mechanism of action. Am J Transplant 2006: 6: 859–866.
- 19Nagel A, Hertl M, Eming R. B-cell-directed therapy for inflammatory skin diseases. J Invest Dermatol 2009: 129: 289–301.
- 20Dungarwalla M, Marsh JC, Tooze JA, et al. Lack of clinical efficacy of rituximab in the treatment of autoimmune neutropenia and pure red cell aplasia: Implications for their pathophysiology. Ann Hematol 2007: 86: 191–197.
- 21Faurschou M, Hasselbalch HC, Nielsen OJ. Sustained remission of platelet counts following monoclonal anti-CD20 antibody therapy in two cases of idiopathic autoimmune thrombocytopenia and neutropenia. Eur J Haematol 2001: 66: 408–411.
- 22Rabusin M, Snowden JA, Veys P, et al. Long-term outcomes of hematopoietic stem cell transplantation for severe treatment-resistant autoimmune cytopenia in children. Biol Blood Marrow Transplant 2013: 19: 666–669.
- 23Waespe N, Zeilhofer U, Gungor T. Treatment-refractory multi-lineage autoimmune cytopenia after unrelated cord blood transplantation: Remission after combined bortezomib and vincristine treatment. Pediatr Blood Cancer 2014: 61: 2112–2114.
- 24Yusuf HR, Hooper WC, Grosse SD, Parker CS, Boulet SL, Ortel TL. Risk of venous thromboembolism occurrence among adults with selected autoimmune diseases: A study among a U.S. cohort of commercial insurance enrollees. Thromb Res 2015: 135: 50–57.
- 25Norgaard M. Thrombosis in patients with primary chronic immune thrombocytopenia. Thromb Res 2012: 130(Suppl 1): S74–S75.
- 26Escher R. Extensive venous thrombosis following administration of high-dose glucocorticosteroids and tranexamic acid in relapsed Evans syndrome. Blood Coagul Fibrinolysis 2008: 19: 741–742.
- 27Iroh Tam PY, Richardson M, Grewal S. Fatal case of bilateral internal jugular vein thrombosis following IVIg infusion in an adolescent girl treated for ITP. Am J Hematol 2008: 83: 323–325.
Citing Literature
