scholarly journals Lung transplantation for idiopathic multicentric Castleman disease: potential efficacy and tolerability of a humanized anti-interleukin-6 receptor monoclonal antibody

2021 ◽  
Vol 7 (1) ◽  
Author(s):  
Yasuaki Tomioka ◽  
Shinji Otani ◽  
Shin Tanaka ◽  
Kazuhiko Shien ◽  
Ken Suzawa ◽  
...  

Abstract Background Idiopathic multicentric Castleman disease (iMCD) is a rare polyclonal lymphoproliferative disease caused by the overrepresentation of interleukin-6 (IL-6). Tocilizumab (TCZ) is a humanized monoclonal antibody that binds to the IL-6 receptor and is approved for the treatment of iMCD. The efficacy and tolerability of TCZ in patients with iMCD undergoing lung transplantation (LTx) remain unknown. Case presentation We present the case of a 48-year-old iMCD patient with end-stage lung disease (ESLD) who was successfully treated with cadaveric single-LTx. Intravenous TCZ was used to stabilize the iMCD patient every 2 weeks, except for withdrawal immediately after LTx. At 32 month post-transplant, the patient remained asymptomatic without evidence of rejection, development of de novo donor-specific antibody (DSA), and recurrent iMCD in the native lung. Conclusions Single-LTx can be a feasible treatment option for ESLD caused by iMCD. TCZ can be used safely and may be beneficial in recipients with iMCD, and TCZ in combination with usual immunosuppression can be helpful in stabilizing iMCD patients pre- and post-LTx.

Blood ◽  
2005 ◽  
Vol 106 (8) ◽  
pp. 2627-2632 ◽  
Author(s):  
Norihiro Nishimoto ◽  
Yuzuru Kanakura ◽  
Katsuyuki Aozasa ◽  
Takeshi Johkoh ◽  
Minoru Nakamura ◽  
...  

Abstract Multicentric Castleman disease (MCD) is an atypical lymphoproliferative disorder characterized by systemic lymphadenopathy and constitutional inflammatory symptoms. Dysregulated overproduction of interleukin-6 is responsible for the clinical abnormalities. This multicenter prospective study was undertaken to evaluate the safety and efficacy of a humanized anti–human interleukin-6 (IL-6) receptor monoclonal antibody (MRA) in patients with MCD. We report here results of the first 60 weeks of the study enrolling 28 patients. The initial dosing period consisted of 8 infusions of 8 mg/kg MRA administered biweekly. Adjustments in the dose and treatment interval were allowed for each patient in an extension phase after 16 weeks. Within 16 weeks, treatment with MRA consistently alleviated lymphadenopathy and all the inflammatory parameters. Hemoglobin, albumin, and total cholesterol levels, high-density lipoprotein cholesterol values, and body mass index all increased significantly. In addition, fatigue diminished. Chronic inflammatory symptoms were successfully managed over 60 weeks. In 8 (28.6%) patients, the MRA dose was decreased or the treatment interval was extended without exacerbation. Eleven (73.3%) of 15 patients who had received oral corticosteroids before study entry were able to do well on a reduced corticosteroid dose. Most adverse events were mild to moderate in severity. MRA was tolerated well and significantly alleviated chronic inflammatory symptoms and wasting in patients with MCD.


2020 ◽  
Vol 13 (11) ◽  
pp. e236283
Author(s):  
Tetsuro Aita ◽  
Sugihiro Hamaguchi ◽  
Yoko Shimotani ◽  
Yohei Nakamoto

A woman aged 45 years with a 1.5-year history of violaceous plaques on the forehead and chest presented with fever, weight loss and aggravation of the plaques. Inflammatory markers and interleukin-6 level were elevated, and superficial lymphadenopathies and splenomegaly were identified by CT scan. Immunohistochemical findings of the lymph node and the skin showed polyclonal plasmacytosis and follicular hyperplasia, leading to the diagnosis of idiopathic multicentric Castleman disease (iMCD) after human herpesvirus-8 infection was excluded. The patient was successfully treated with anti-interleukin-6 receptor antibody, tocilizumab, following relapse after prednisolone therapy.Our literature review found 11 case reports of pathologically confirmed iMCD preceded by cutaneous plasmacytosis. The median duration of asymptomatic phase with only skin lesions was 7.5 years, whereas the phase lasted only for 1.5 years in our case. iMCD can develop shortly after asymptomatic cutaneous plasmacytosis. Tocilizumab can be a treatment of choice for this type of iMCD.


Oncotarget ◽  
2015 ◽  
Vol 6 (30) ◽  
pp. 30408-30419 ◽  
Author(s):  
Frits van Rhee ◽  
Corey Casper ◽  
Peter M. Voorhees ◽  
Luis E. Fayad ◽  
Helgi van de Velde ◽  
...  

2018 ◽  
Vol 1 (2) ◽  
pp. 133-135
Author(s):  
Nambiar Vivek K. ◽  
T.S. Dhanya ◽  
Ajai Amrutha V.

Takayasu arteritis (TA) is an idiopathic inflammatory vasculitis mostly affecting young females in the second or third decades of life. Corticosteroids and conventional immunosuppressants remain the mainstay of treatment for TA, but refractory cases are dealt with biological agents. The high cost and longer duration of therapy are issues of concern. Here, we report a case of a patient with refractory TA who underwent successful treatment with tocilizumab, a humanized monoclonal antibody against interleukin-6 receptor.


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