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LETTER TO EDITOR
Year : 2011  |  Volume : 59  |  Issue : 2  |  Page : 306-307

Resistant dermatomyositis complicated by tubercular myositis and successfully treated with rituximab


1 Department of Rheumatology, Fortis Hospital, Bangalore, Karnataka, India
2 Samarpan Health Centre, Bangalore, Karnataka, India
3 Department of Chest Medicine, Fortis Hospital, Bangalore, Karnataka, India

Date of Web Publication7-Apr-2011

Correspondence Address:
Ramesh Jois
Department of Rheumatology, Fortis Hospital, Bangalore, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.79163

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How to cite this article:
Jois R, Vasudevan N, Srinivasan P, Mehta R. Resistant dermatomyositis complicated by tubercular myositis and successfully treated with rituximab. Neurol India 2011;59:306-7

How to cite this URL:
Jois R, Vasudevan N, Srinivasan P, Mehta R. Resistant dermatomyositis complicated by tubercular myositis and successfully treated with rituximab. Neurol India [serial online] 2011 [cited 2019 Sep 16];59:306-7. Available from: http://www.neurologyindia.com/text.asp?2011/59/2/306/79163


Sir,

B-cell depleting monoclonal antibody rituximab (RTX) has been shown to be effective in patients with refractory dermatomyositis (DM). [1],[2],[3],[4] Biological therapy is associated with an increased risk of tuberculosis (TB); however, this risk is negligible with RTX. [5] We report an unusual patient of drug refractory DM complicated by tuberculous myositis and successful treatment with RTX.

A 35-year-old lady presented with severe myalgia, proximal muscle weakness, and skin rash. She was diagnosed as a case of DM based on elevated creatinine phosphokinase (CK; 13,119 U/L), electromyography features being suggestive of primary muscle disease, and muscle biopsy consistent with DM. The work-up for underlying malignancy was negative. She was initially started on prednisolone and azathioprine. Her weakness worsened over the next 2 months, although CK was normalized. Azathioprine was substituted by methotrexate but had to be discontinued as she developed anaphylaxis. She was then initiated on mycophenolate mofetil (MMF). A month later, she presented with fever, heliotrope rash, worsening of muscle weakness, and severe thigh and gluteal tenderness. Repeat CK was normal. Investigations for underlying infection were negative. Despite a course of intravenous immunoglobulin (IVIg), her myalgia worsened. She developed boggy lumps in the lower third of the left thigh and gluteal region. Magnetic resonance imaging (MRI) of the left leg revealed acute myositis and subcutaneous fluid collection in the areas mentioned [Figure 1]. The fluid from the lump showed plenty of acid-fast bacilli. A diagnosis of tuberculous myositis and abscess was considered. Mycobacterium tuberculosis was subsequently isolated from the pus culture. MMF was discontinued and prednisolone was reduced to 15 mg/day. Antituberculous therapy (ATT) resulted in a complete resolution of the abscess and muscle pains. Four months later, she had worsening of neck and limb weakness and elevation of CK, 865 U/L. An increase in the steroid dose and MMF led to the recurrence of the muscle abscess. She was given another course of IVIg with which the neck weakness improved but not limb weakness and CK increased to 4930 U/L. She was given two doses of RTX, 1 g each, 2 weeks apart while continuing ATT. She showed gradual improvement in the muscle weakness and also a progressive decline in CK values. At 4 months of the last dose of RTX, CK was 246 U/L and muscle power was normal. Seven months later, she was independent in her daily activities, and ATT was stopped, prednisolone was reduced to 7.5 mg while MMF is being continued. She has been in remission for 24 months. At the last follow-up, she was on prednisolone 2.5 mg and MMF.
Figure 1: (a) Coronal STIR image of the thigh shows a diffuse hyperintense signal of the right vastus lateralis and medialis suggestive of myositis. (b) Axial T2-weighted image of the pelvis shows turbid fluid collection lateral to the bony pelvis; signal of this fluid is less than that of the urinary bladder, suggestive of purulent nature. (c) Axial and sagittal high-frequency scan at the level of collection in (b) shows loculated thick-walled (arrow) hypoechoic fluid collection suggestive of abscess

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The co-occurrence of TB myositis with DM is extremely rare [6],[7] and must be considered as a possible cause of myositis in immunosuppressed patients, especially in high endemic areas. Treatment options for DM in this setting are limited. A significant disturbance of B-cell function is seen in DM. B-cell driven response with clonal expansion, isotype switch ultimately causing altered plasma cell maturation has been observed. [8] The B-cell functions in DM form the rationale to specifically target B-cell with RTX. An open-label study and few case reports have shown significant improvement with RTX therapy in refractory DM. [1],[2],[3],[4] A recent review of all published cases of DM treated with RTX noted a response rate of 71.4%. [1] The median time free of symptoms between two courses of RTX was 12 months. Maximal improvement in muscle strength was seen by 12 weeks. [2] Four of the six patients experienced recurrence, which improved after a second course of RTX. The average prednisolone dosage could be decreased (from 16.7 to 4 mg) and MTX could be reduced by 50%. [3] Remarkable improvements in muscle weakness and CK in our patient with RTX therapy were consistent with findings from the earlier reports. Our patient is probably the first published case of use of RTX in the setting of refractory DM and tuberculous myositis.

 
  References Top

1.Rios Fernandez R, Callejas Rubio JL, Sanchez Cano D, Saez Moreno JA, Ortego Centeno N. Rituximab in the treatment of dermatomyositis and other inflammatory myopathies. A report of 4 cases and review of the literature. Clin Exp Rheumatol 2009;27:1009-16.  Back to cited text no. 1
    
2.Levine TD. Rituximab in the treatment of dermatomyositis: An open-label pilot study. Arthritis Rheum 2005;52:601-7.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  
3.Noss EH, Hausner-Sypek DL, Weinblatt ME. Rituximab as therapy for refractory polymyositis and dermatomyositis. J Rheumatol 2006;33:1021-6.  Back to cited text no. 3
[PUBMED]  [FULLTEXT]  
4.Feist E, Dorner T, Sorensen H, Burmester GR. Longlasting remissions after treatment with rituximab for autoimmune myositis. J Rheumatol 2008;35:1230-2.  Back to cited text no. 4
    
5.Edwards JC, Szczepanski L, Szechinski J, Filipowicz-Sosnowska A, Emery P, Close DR, et al. Efficacy of B-cell targeted therapy with rituximab in patients with rheumatoid arthritis. N Engl J Med 2004;350:2572-81.  Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.Liu CH, Liu WC, Chen LW, Chen JS. Tuberculous myofasciitis in dermatomyositis. Clin Rheumatol 2008;27:S7-9.  Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.Yoshida Y, Nakayama J, Furue M, Matsuda T. Dermatomyositis with tuberculous fasciitis. Eur J Dermatol 2004;14:123-4.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.Bradshaw EM, Orihuela A, McArdel SL, Salajegheh M, Amato AA, Hafler DA, et al. A local antigen-driven humoral response is present in the inflammatory myopathies. J Immunol 2007;178:547-56.  Back to cited text no. 8
[PUBMED]  [FULLTEXT]  


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