J Korean Med Sci.  2010 Sep;25(9):1364-1367. 10.3346/jkms.2010.25.9.1364.

A Case of Multicentric Castleman's Disease Having Lung Lesion Successfully Treated with Humanized Anti-interleukin-6 Receptor Antibody, Tocilizumab

• Higuchi T
• Nakanishi
• Takada K
• Matsumoto
• Okada M
• Horikoshi
• Suzuki K

• KMID: 1785918
• DOI: http://doi.org/10.3346/jkms.2010.25.9.1364

Abstract

This report presents the case of a patient demonstrating multicentric Castleman's disease (MCD) with a lung lesion that was successfully treated with an anti-interleukin-6 receptor antibody, tocilizumab in combination with corticosteroid and tacrolimus. A 43-yr-old female with abnormal shadows on a chest X-ray was referred to the hospital for further examination. She was diagnosed as having MCD based on the characteristic pathology of inguinal lymph node, lung lesions, laboratory data, and undifferentiated arthritis. Corticosteroid and rituximab therapy did not fully ameliorate the symptoms; thus, the therapeutic regimen was changed to include tocilizumab, oral corticosteroid and tacrolimus. This regimen resulted in clinical remission and the dose of tocilizumab and corticosteroid could be tapered. Tocilizumab in combination with corticosteroid and tacrolimus may therefore be a beneficial treatment regimen for lung lesions associated with MCD.

Keyword

Giant Lymph Node Hyperplasia; Biological Products; Immunosuppressive Agents

MeSH Terms

Adrenal Cortex Hormones/therapeutic use
Adult
Antibodies, Monoclonal/*therapeutic use
Drug Therapy, Combination
Female
Giant Lymph Node Hyperplasia/*diagnosis/drug therapy/radiography
Humans
Immunosuppressive Agents/therapeutic use
Lung Diseases, Interstitial/*drug therapy/pathology
Lymph Nodes/pathology
Receptors, Interleukin-6/antagonists & inhibitors
Tacrolimus/therapeutic use
Tomography, X-Ray Computed

Figure

• 

  Fig. 1 Chest CT findings (A) just before the second regimen, of tocilizumab in combination with corticosteroid and tacrolimus: centrilobular nodules, thin walled cysts, the thickening of the bronchovascular bundles and ground-grass opacities were noted; (B) Thirteen months after the continuation of the second regimen; (C) after twenty three months: most of the lesions had alleviated.

• 

  Fig. 2 The specimen obtained from the inguinal lymph node. Microscopic examination of the lymph node showed typical features of plasma cell type Castleman's disease (A, H&E stain, ×40; B, H&E stain, ×200).

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  Fig. 3 Clinical course of the patient. mPSL, methylprednisolone; RTX, rituximab 375 mg/m2; tocilizumab,tocilizumab 6 mg/kg. The dose of mPSL could be gradually tapered from 16 mg/day to 4 mg/day.

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