A complicated case of cutaneous necrotizing vasculitis associated with monoclonal gammopathy. Case report
- Authors: Rekhtina I.G.1, Vorobyeva O.A.2, Atroshenko E.A.1, Kovrigina A.M.1, Mendeleeva L.P.1
-
Affiliations:
- National Medical Research Center for Hematology
- National Center for Clinical Morphological Diagnostics
- Issue: Vol 97, No 6 (2025): Issues of nephrology
- Pages: 522-527
- Section: Clinical notes
- URL: https://journal-vniispk.ru/0040-3660/article/view/313994
- DOI: https://doi.org/10.26442/00403660.2025.06.203260
- ID: 313994
Cite item
Full Text
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.
Keywords
Full Text
##article.viewOnOriginalSite##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, MoscowOlga 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 PetersburgElisavetta 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, MoscowAlla 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, MoscowLarisa 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, MoscowReferences
- Kristinsson SY, Tang M, Pfeiffer RM, et al. Monoclonal gammopathy of undetermined significance and risk of infections: a population-based study. Haematologica. 2012;97(6):854-8. doi: 10.3324/haematol.2011.054015
- Van de Donk NW, Palumbo A, Johnsen HE, et al. European Myeloma Network. The clinical relevance and management of monoclonal gammopathy of undetermined significance and related disorders: recommendations from the European Myeloma Network. Haematologica. 2014;99(6):984-96. doi: 10.3324/haematol.2013.100552
- Kristinsson SY, Fears TR, Gridley G, et al. Deep vein thrombosis after monoclonal gammopathy of undetermined significance and multiple myeloma. Blood. 2008;112(9):3582-6. doi: 10.1182/blood-2008-04-151076
- Fermand JP, Bridoux F, Dispenzieri A, et al. Monoclonal gammopathy of clinical significance: a novel concept with therapeutic implications. Blood. 2018;132(14):1478-85. doi: 10.1182/blood-2018-04-839480
- Leung N, Bridoux F, Batuman V, et al. The evaluation of monoclonal gammopathy of renal significance: a consensus report of the International Kidney and Monoclonal Gammopathy Research Group. Nat Rev Nephrol. 2019;15(1):45-59. doi: 10.1038/s41581-018-0077-4
- Смирнов А.В., Афанасьев Б.В., Поддубная И.В., и др. Моноклональная гаммапатия ренального значения: консенсус гематологов и нефрологов России по введению нозологии, диагностике и обоснованности клон-ориентированной терапии. Терапевтический архив. 2020;92(7):10-22 [Smirnov AV, Afanasyev BV, Poddubnaya IV, et al. Monoclonal gammopathy of renal significance: consensus of hematologists and nephrologists of Russia on the establishment of nosology, diagnostic approach and rationale for clone specific treatment. Terapevticheskii Arkhiv (Ter. Arkh.). 2020;92(7):10-22 (in Russian)]. doi: 10.26442/00403660.2020.07.000659
- Castillo JJ, Callander NS, Baljevic M, et al. The evaluation and management of monoclonal gammopathy of renal significance and monoclonal gammopathy of neurological significance. Am J Hematol. 2021;96(7):846-53. doi: 10.1002/ajh.26155
- Garderet L, Al Hariri M, Wasielica-Poslednik J, et al. Monoclonal Gammopathy of Ocular Significance (MGOS) – a Series of Corneal Manifestations and Treatment Outcomes. Blood. 2021;138(Suppl. 1):2695. doi: 10.1182/blood-2021-151460
- Lipsker D. Monoclonal gammopathy of cutaneous significance: review of a relevant concept. J Eur Acad Dermatol Venereol. 2017;31(1):45-52. doi: 10.1111/jdv.13847
- Васильев В.И., Городецкий В.Р., Логвиненко О.А., и др. Ревматические маски плазматических дискразий. Научно-практическая ревматология. 2012;50(2):35-43 [Vasilyev VI, Gorodetsky VR, Logvinenko OA, et al. Rheumatic masks of plasma cell dyscrasias. Rheumatology Science and Practice. 2012;50(2):35-43 (in Russian)]. doi: 10.14412/1995-4484-2012-1271
- Swan D, Murphy M, Elhassadi E. Cutaneous Vasculitis: An Unusual Presentation of a Biclonal Nodal Plasma Cell Dyscrasia. Case Rep Hematol. 2017;2017:8152610. doi: 10.1155/2017/8152610
- Васильев В.И., Городецкий В.Р., Седышев С.Х., и др. Клинические проявления васкулита в дебюте множественной миеломы. Научно-практическая ревматология. 2010;48(1):81-7 [Vasil'ev VI, Gorodetskiy VR, Sedyshev SKh, et al. Clinical manifestation of vasculitis at the onset of multiple myeloma. Rheumatology Science and Practice. 2010;48(1):81-7 (in Russian)]. doi: 10.14412/1995-4484-2010-1410
- Городецкий В.Р., Пробатова Н.А., Логвиненко О.А., и др. Трудности диагностики лимфоидных неоплазий у больных ревматоидным артритом. Научно-практическая ревматология. 2009;47(3):94-101 [Gorodetsky VR, Probatova NA, Logvinenko OA, et al. Difficulties of lymphoid neoplasia diagnosis in patients with rheumatoid arthritis. Rheumatology Science and Practice. 2009;47(3):94-101 (in Russian)]. doi: 10.14412/1995-4484-2009-1318
- Логвиненко О.А., Васильев В.И. Неходжскинские лимфомы при ревматических заболеваниях. Научно-практическая ревматология. 2011;49(5):71-82 [Logvinenko OA, Vasil'ev VI. Nekhodzhkinskie limfomy pri revmaticheskikh zabolevaniyah. Rheumatology Science and Practice. 2011;49(5):71-82 (in Russian)]. doi: 10.14412/1995-4484-2011
Supplementary files
