A complicated case of cutaneous necrotizing vasculitis associated with monoclonal gammopathy. Case report

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Abstract

Diagnosing diseases related to monoclonal gammopathy (MG) with clinical significance is challenging. In real-world practice, proving the connection between organ damage and paraprotein or plasma cell dyscrasia is difficult or even impossible. Only an atypical course of the disease and non-compliance with the established criteria may suggest the contribution of plasma cell neoplasia in pathogenesis. Rheumatic diseases associated with monoclonal secretion do not belong to MG with clinical significance. Therefore, in the absence of hematological malignancy, clone reduction therapy is not indicated. We present the first clinical case of a patient with cutaneous necrotizing vasculitis and MG, who received clone reduction therapy with no hematological malignancy. The rationale for the choice of such treatment approach was the atypical course of vasculitis, as well as the unstable effect after methylprednisolone therapy. Therapy with lenalidomide, cyclophosphamide, and dexamethasone was highly effective; the regression of clinical symptoms was associated with a decrease in paraprotein to trace amounts. However, the lack of evidence of the relationship of vasculitis with plasma cell dyscrasia does not support considering the described case as one of the forms of MG with clinical significance. Further observation and resumption of monoclonal secretion, coinciding with the recurrence of vasculitis, may additionally support the relationship between these two conditions. This case was discussed at the Council of Experts with hematologists, rheumatologists, and pathologists.

About the authors

Irina G. Rekhtina

National Medical Research Center for Hematology

Author for correspondence.
Email: rekhtina.i@blood.ru
ORCID iD: 0000-0001-5440-4340

д-р мед. наук, зав. отд-нием гематологии и химиотерапии плазмоклеточных дискразий 

Russian Federation, Moscow

Olga A. Vorobyeva

National Center for Clinical Morphological Diagnostics

Email: rekhtina.i@blood.ru
ORCID iD: 0000-0002-6946-6816

канд. мед. наук, зав. отд-нием патологии почки и сложного морфологического диагноза 

Russian Federation, Saint Petersburg

Elisavetta A. Atroshenko

National Medical Research Center for Hematology

Email: rekhtina.i@blood.ru
ORCID iD: 0009-0005-2588-5612

врач-гематолог клинико-диагностического отд-ния химиотерапии с дневным стационаром 

Russian Federation, Moscow

Alla M. Kovrigina

National Medical Research Center for Hematology

Email: rekhtina.i@blood.ru
ORCID iD: 0000-0002-1082-8659

д-р биол. наук, проф., зав. патологоанатомическим отд-нием 

Russian Federation, Moscow

Larisa P. Mendeleeva

National Medical Research Center for Hematology

Email: rekhtina.i@blood.ru
ORCID iD: 0000-0002-4966-8146

д-р мед. наук, проф., рук. управления по научной и образовательной работе, зав. отд. химиотерапии парапротеинемических гемобластозов 

Russian Federation, Moscow

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Supplementary files

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2. Fig. 1. Rashes on legs and face.

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3. Fig. 2. Light microscopy of the skin: a – a focus of necrotizing vasculitis in the deep dermis layer at the level of skin appendages, with perivascular and transmural infiltration, karyorexis, fibrinoid wall necrosis and thrombosis. Hematoxylin and eosin staining; magnification ×100; b – the same site. Fibrinoid necrosis of vessel wall, infiltration of vessel wall with cariorexis, fibrin thrombus in vessel lumen. Hematoxylin and eosin staining; magnification ×400.

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4. Fig. 3. Light microscopy of the skin: a – vascular thrombosis in the deep layer of the dermis at the border with subcutaneous fatty tissue. Hematoxylin and eosin staining; magnification ×100; b – the same vessel. Fibrin-erythrocytic thrombus in the lumen of the vessel. Masson trichrome staining; magnification ×400.

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