Bertuglia, Giuseppe

Bringhen, Sara

Bruno, Benedetto

D'Agostino, Mattia

English

Institutional Research Information System University of Turin

Br J Haematol. 2024;00:1–2.    | 1wileyonlinelibrary.com/journal/bjhI M A G E S  I N  H A E M A T O L O G YM- protein- related necrobiotic granuloma in a multiple myeloma patient treated with daratumumab, lenalidomide and dexamethasoneA 75- year- old Immunoglobulin A- light chain k (IgA- k) smouldering multiple myeloma patient started complain-ing of pain in both soles of her feet which had progressively worsened and limited her daily living activities (DLA). On physical examination, both heels and big toes were enlarged with irregular, indurated, painful, itchy nodules, some of the ulcerated; the periungual region of the right middle finger also appeared erythematosus and de- epithelised (left upper panels, photographic images). No other skin le-sions were present. Magnetic resonance imaging (MRI) of both feet showed plantar fasciitis (red arrow) with reactive oedema of the calcaneal spongy bone tissue (blue arrow), oedema of subcutaneous soft tissue (yellow arrow) and sev-eral nodules (maximum 25 mm in diameter; green arrow) on the plantar side (left middle panels, MRI). After two non- diagnostic samples, an ultrasound- guided tru- cut biopsy of the nodules was performed and histological ex-amination (lower panels from the left to the right) led to the diagnosis of reactive necrobiotic granuloma: strands of loose connective tissue with cellular areas consisting of ill- defined CD163+, CD68+ histiocyte aggregates and scattered neutrophiles with central necrosis (red arrow); absence of DOI: 10.1111/bjh.19887  This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made.© 2024 The Author(s). British Journal of Haematology published by British Society for Haematology and John Wiley & Sons Ltd.2 |   IMAGES IN HAEMATOLOGYplasma cells (CD138 reaction negative); and very low pro-liferation index (Ki67 1%). In addition to the necrobiotic granuloma, possibly related to the M- protein, a thorough work- up was repeated showing one myeloma- defining event (≥60% bone marrow plasma cells). Treatment with daratu-mumab–lenalidomide–dexamethasone (DRd) was started. Topical 0.05% clobetasol ointment, for 1 month, and urea- based topical moisturizer were also added to DRd as sug-gested by dermatologists. At 1 year through treatment, the M- protein dropped from 39 to 0.7 g/L and a very good par-tial response, as best response, was achieved. Concurrently, the skin lesions had progressively improved (right upper panels, photographic images) with the complete resolution of the pain, the itch, the ulcers and DLA recovery as well as MRI showed the resolution of the plantar fasciitis and the subcutaneous oedema with a slight reduction of the subcu-taneous nodules (right middle panels, MRI).To our knowledge, among the wide range of granulo-matous disorders, only necrobiotic xanthogranuloma has been ascribed to M- protein,1 and anti- plasma cell clone drugs have been used in some cases.2–4 On the one hand, we cannot exclude that foamy histiocytes, typically seen in necrobiotic xanthogranuloma,1 may have been present in the residual tissue not included in the tru- cut sample, but on the other hand, the clinical presentation of necro-biotic xanthogranuloma does not match to our case.1 All these elements support the theory of being an unusual and previously undescribed M- protein- related cutaneous in-volvement, successfully treated with anti- plasma cell clone regimen.AC K NOW L E D G E M E N T SWe are grateful to the patient who gave permission to write this paper. We also thank Dr. Dario Ferracane for his expert opinion in the correct interpretation of imaging studies and Dr. Enrico Amaducci for his support during the drafting of the paper. Open access publishing facilitated by Universita degli Studi di Torino, as part of the Wiley - CRUI- CARE agreement.F U N DI NG I N FOR M AT IONNo funding was used for this study.E T H IC S S TAT E M E N TThe consent form was approved by AOU Città della salute e della Scienza, University of Turin, Italy.PAT I E N T C ON SE N T S TAT E M E N TThe patient gave her informed consent to the use of her med-ical charts, MRI imaging, histology imaging and photos for medical research purpose.Giuseppe Bertuglia1,2,3 , Sara Bringhen1 , Benedetto Bruno2,3 , Giorgia Andrea Impalà4 , Mattia D'Agostino2,31SSD Clinical Trial in Oncoematologia e Mieloma Multiplo, Department of Oncology, Azienda Ospedaliero- Universitaria Città Della Salute e Della Scienza di Torino, 2Division of Hematology, Azienda Ospedaliero- Universitaria Città Della Salute e Della Scienza di Torino, 3Department of Molecular Biotechnology and Health Sciences, 4Pathology Division, Department of Medical Sciences, University of Torino, Torino, ItalyEmail: mattia.dagostino@unito.itORC I DGiuseppe Bertuglia   https://orcid.org/0009-0009-3785-9824 Sara Bringhen   https://orcid.org/0000-0003-4539-6363 Benedetto Bruno   https://orcid.org/0000-0002-7299-6770 Giorgia Andrea Impalà   https://orcid.org/0009-0005-3868-5062 Mattia D’Agostino   https://orcid.org/0000-0002-1711-6996 T W I T T E RGiuseppe Bertuglia   Bertolo_94 Mattia D’Agostino   Dagostino_MD R E F E R E N C E S 1. Claveau JS, Wetter DA, Kumar S. Cutaneous manifestations of mono-clonal gammopathy. Blood Cancer J. 2022;12(4):1–14. https:// doi. org/ 10. 1038/ s4140 8- 022- 00661 - 1 2. Martínez Fernández M, Rodríguez Prieto MA, Ruiz González I, Sánchez Sambucety P, Delgado VS. Necrobiotic xanthogranu-loma associated with myeloma. J Eur Acad Dermatology Venereol. 2004;18(3):328–31. https:// doi. org/ 10. 1111/j. 1468- 3083. 2004. 00906. x 3. Sivak- Callcott JA, Rootman J, Rasmussen SL, Nugent RA, White VA, Paridaens D, et al. Adult xanthogranulomatous disease of the orbit and ocular adnexa: new immunohistochemical findings and clinical re-view. Br J Ophthalmol. 2006;90(5):602–8. https:// doi. org/ 10. 1136/ bjo. 2005. 085894 4. Mahendran P, Wee J, Chong H, Natkunarajah J. Necrobiotic xan-thogranuloma treated with lenalidomide. Clin Exp Dermatol. 2018;43(3):345–7. https:// doi. org/ 10. 1111/ ced. 13293  13652141, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/bjh.19887 by Schweizerische Akademie Der, Wiley Online Library on [20/11/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License

M‐protein‐related necrobiotic granuloma in a multiple myeloma patient treated with daratumumab, lenalidomide and dexamethasone

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M‐protein‐related necrobiotic granuloma in a multiple myeloma patient treated with daratumumab, lenalidomide and dexamethasone

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