Delayed and Long-term Remission of Refractory Hemolytic Anemia in a Child with Systemic Lupus Erythematosus Treated with Rituximab

Ju Hwan Park; Jae Wook Im; Hong Kil Jun; Hae Min Park; Seung Won Choi; Sang Kyu Park; Ji Seon Oh

doi:10.4078/jrd.2014.21.4.196

Journal List > J Rheum Dis > v.21(4) > 1064120

Go to TopGo to Top Go to BottomGo to Bottom

TOOLS

Park, Im, Jun, Park, Choi, Park, and Oh: Delayed and Long-term Remission of Refractory Hemolytic Anemia in a Child with Systemic Lupus Erythematosus Treated with Rituximab

Case Report

J Rheum Dis 2014;21(4):196-200.

Published online: 31 October 2014

DOI: https://doi.org/10.4078/jrd.2014.21.4.196

Delayed and Long-term Remission of Refractory Hemolytic Anemia in a Child with Systemic Lupus Erythematosus Treated with Rituximab

Ju Hwan Park¹, Jae Wook Im¹, Hong Kil Jun¹, Hae Min Park¹, Seung Won Choi¹, Sang Kyu Park², Ji Seon Oh¹

¹Department of Internal Medicine, Ulsan University Hospital, University of Ulsan College of Medicine, Ulsan, Korea

²Department of Pediatrics, Ulsan University Hospital, University of Ulsan College of Medicine, Ulsan, Korea

Corresponding to: Ji Seon Oh, Department of Internal Medicine, Ulsan University Hospital, University of Ulsan College of Medicine, 877 Bangeojinsunhwan-doro, Dong-gu, Ulsan 682-060, Korea. E-mail: jsoh@uuh.ulsan.kr

Received 31 May 2013 Revised 18 July 2013 Accepted 5 August 2013

This is a Free Access article, which permits unrestricted non-commerical use, distribution, and reproduction in any medium, provided the original work is properly cited.

REFERENCES

1. Lee SG, Yoo B, Kim KM, Choi HO, Oh JS, Nah SS, et al. Successful Treatment of Neuropsychiatric Syndrome with Rituximab in a Patient with Systemic Lupus Erythematosus and Dermatomyositis Overlap Syndrome. J Korean Rheum Assoc. 2008; 15:170–4.

2. Wang SW, Cheng TT. Systemic lupus erythematosus with refractory hemolytic anemia effectively treated with cyclosporin A: a case report. Lupus. 2005; 14:483–5.

3. Tokunaga M, Saito K, Nakatsuka K, Nakayamada S, Nakano K, Tsujimura S, et al. Successful treatment of intravenous cyclophosphamide pulse therapy for systemic lupus erythematosus complicated with steroid-resistant hemolytic anemia. Nihon Rinsho Meneki Gakkai Kaishi. 2003; 26:304–9.

4. Sarles HE, Levin WC. The role of splenectomy in the management of acquired autoimmune hemolytic anemia complicating systemic lupus erythematosus. Am J Med. 1959; 26:547–54.

5. Hwang KW, Ahn YS, Moon JY, Kim IY, Park YE, Kim GT, et al. Experience of Rituximab Treatment in Two Patients with Severe Systemic Lupus Erythematosus. J Korean Rheum Assoc. 2006; 13:230–5.

6. Reff ME, Carner K, Chambers KS, Chinn PC, Leonard JE, Raab R, et al. Depletion of B cells in vivo by a chimeric mouse human monoclonal antibody to CD20. Blood. 1994; 83:435–45.

7. Leandro MJ, Cambridge G, Edwards JC, Ehrenstein MR, Isenberg DA. B-cell depletion in the treatment of patients with systemic lupus erythematosus: a longitudinal analysis of 24 patients. Rheumatology (Oxford). 2005; 44:1542–5.

8. Looney RJ, Anolik JH, Campbell D, Felgar RE, Young F, Arend LJ, et al. B cell depletion as a novel treatment for systemic lupus erythematosus: a phase I/II dose-esca-lation trial of rituximab. Arthritis Rheum. 2004; 50:2580–9.

9. Leandro MJ, Edwards JC, Cambridge G, Ehrenstein MR, Isenberg DA. An open study of B lymphocyte depletion in systemic lupus erythematosus. Arthritis Rheum. 2002; 46:2673–7.

10. Bussone G, Ribeiro E, Dechartres A, Viallard JF, Bonnotte B, Fain O, et al. Efficacy and safety of rituximab in adults' warm antibody autoimmune haemolytic anemia: retrospective analysis of 27 cases. Am J Hematol. 2009; 84:153–7.

11. Dierickx D, Verhoef G, Van Hoof A, Mineur P, Roest A, Triffet A, et al. Rituximab in autoimmune haemolytic anaemia and immune thrombocytopenic purpura: a Belgian retrospective multicentric study. J Intern Med. 2009; 266:484–91.

12. Kumar S, Benseler SM, Kirby-Allen M, Silverman ED. B-cell depletion for autoimmune thrombocytopenia and autoimmune hemolytic anemia in pediatric systemic lupus erythematosus. Pediatrics. 2009; 123:e159–63.

13. Oh HJ, Yun MJ, Lee ST, Lee SJ, Oh SY, Sohn I. Evans syndrome following longstanding Hashimoto's thyroiditis and successful treatment with rituximab. Korean J Hematol. 2011; 46:279–82.

14. Park CY, Chung CH. A patient with mixed type Evans syndrome: efficacy of rituximab treatment. J Korean Med Sci. 2006; 21:1115–6.

15. Lee JH, Lee KS. A Case of Autoimmune Hemolytic Anemia Treated with Rituximab in a Child. Korean J Hematol. 2006; 41:321–5.

16. Smith KG, Jones RB, Burns SM, Jayne DR. Long-term comparison of rituximab treatment for refractory systemic lupus erythematosus and vasculitis: Remission, relapse, and retreatment. Arthritis Rheum. 2006; 54:2970–82.

17. Ng KP, Cambridge G, Leandro MJ, Edwards JC, Ehrenstein M, Isenberg DA. B cell depletion therapy in systemic lupus erythematosus: long-term follow-up and predictors of response. Ann Rheum Dis. 2007; 66:1259–62.

Go to :

Figure 1.

Kidney ultrasonography reveals diffuse increased echogenecity of medulla of both kidneys, suggesting medullary nephrocalcinosis.

undefined

Figure 2.

Trends of hemoglobin and total bilirubin levels before and after rituximab treatment. The bar graph stands for daily amounts of oral steroid calculated in terms of prednisolone. Hemoglobin level increased, and total bilirubin level and the doses of oral steroid decreased after the use of rituximab. SLE, systemic lupus erythematosus; HA, hemolytic anemia.

undefined

Table 1.

Laboratory findings before and after rituximab treatment in a child with systemic lupus erythematosus and hemolytic anemia

Rituximab administration
	2008/07	2008/12	2009/04	2009/09	2009/12	2010/01	2010/02	2010/04	2010/07	2011/02	2011/09	2012/04	2012/07	2012/09	2012/12	2013/03
Hb (/dL)	7.1	10.1	5.6	8.3	6.8	12.9	10.6	8.9	9.9	11.7	13.1	13.1	13.4	14.8	13.7	13.8
CRC (%)	5.38	−	7.37	12.75	5.53	2.96	7.14	5.9	5.92	3.17	1.12	1.43	1.16	1.44	0.93	1.01
CD19 (/uL)	−	−	−	−	−	165	0.4	0.3	0.7	11.4	26.8	−	312	−	−	−
IgA (mg/dL)	−	−	−	−	−	101	−	−	46	3	2	−	0	0	2	0
IgM (mg/dL)	−	−	−	−	−	130	−	−	＜25	＜25	＜25	−	＜25	＜25	＜25	＜25
IgG (mg/dL)	−	−	−	−	−	754	−	−	579	585	557	−	376	369	355	＜300
K (mM/L)	2.3	5.1	3.9	4.5	4.8	4.3	3.8	4.2	−	4	−	4.4	4	4	4.2	4.3
tCO2 (mmol/L)	14	23	24	23	28	28	28	25	−	29	−	23	24	26	26	25
Supplementation of potassium citrate

Hb: hemoglobin, CRC: corrected reticulocyte count, CD: Cluster of Differentiation, IgA: immunoglobulin A, IgM: immunoglobulin M, IgG: immunoglobulin G, K: potassium, tCO2: total carbon dioxide.

TOOLS

Similar articles