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Plasma cell mastitis in women with rheumatoid arthritis treated with TNFα antagonists: Report of 2 cases
G Model BONSOI-4299; No. of Pages 2
ARTICLE IN PRESS Joint Bone Spine xxx (2015) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the Editor Plasma cell mastitis in women with rheumatoid arthritis treated with TNF␣ antagonists: Report of 2 cases
a r t i c l e
i n f o
Keywords: Rheumatoid arthritis Plasma cell mastitis Duct ectasia of breast TNF␣ antagonists
Plasma cell mastitis, also known as duct ectasia of the breast, is an inflammatory disease. We report two cases in women with rheumatoid arthritis (RA) treated with TNF␣ antagonists.
Table 1 Reported cases of benign inflammatory breast disease in patients with inflammatory diseases. Inflammatory disease
Number of patients
Year(s) of publication
Reference
Sjögren’s syndrome Rheumatoid arthritis Erythema nodosum Sarcoidosisa Non-langerhans cell histiocytosis Granulomatosis with polyangiitis IgG4-related disease Systemic lupus erythematosus
1 1 8 7 5
2013 1991 1987 to 2014 1939 to 2010 1995 to 2010
[3] [4] [5] [6] [7]
2
1999; 2006
[8]
3 3
2005; 2009; 2010 2000; 2013; 2014
[9] [10]
a
Including 1 patient with sarcoidosis of the breast.
2. Discussion 1. Cases 1.1. Case report No. 1 In 2004, a 52-year-old woman developed recurrent abscess-like lesions in her left breast. Ultrasonography showed heterogeneous and inflammatory tissue remodeling combined with duct ectasia. A biopsy established the diagnosis of plasma cell mastitis with a typical inflammatory infiltrate surrounding the ducts, as well as abscess-like lesions. The abnormalities resolved under prednisone therapy in a daily dosage of 1 mg/kg. In 2008, during the prednisone taper, signs of seropositive RA developed. Methotrexate therapy was initiated, to little effect. In 2010, the prednisone dependency prompted the initiation of infliximab therapy (3 mg/kg every 8 weeks). The manifestations of both the RA and the plasma cell mastitis improved rapidly. At last follow-up after more than 4 years of infliximab therapy, she had had a single recurrence of plasma cell mastitis, which had responded to prednisone therapy.
1.2. Case report No. 2 This 58-year-old woman had a 10-year history of RA with secondary Sjögren’s disease. For the last 2 years she had been taking prednisone 6 mg/day and etanercept 50 mg/week. An abscess-like lesion with nipple retraction developed in the right breast. An infection was suspected and the etanercept was therefore promptly discontinued. Ultrasonography visualized duct ectasia consistent with plasma cell mastitis. The results of the biopsy ruled out carcinoma and infection and showed a non-specific inflammatory response. Re-introduction of etanercept therapy with no increase in the prednisone dosage was followed by regression of the breast abnormalities and ensured good control of the RA.
We describe two cases of plasma cell mastitis in patients with RA that were diagnosed based on the presence of duct ectasia. In both patients, breast infection with abscess formation was ruled out. TNF␣ antagonist therapy improved the clinical manifestations. Benign inflammatory breast disease manifests as breast pain and inflammatory nodules measuring 1 to 2 cm in diameter. The diagnosis rests on the histological findings and results of breast imaging studies (ultrasonography, mammography, and magnetic resonance imaging) [1]. Two variants exist, plasma cell mastitis and granulomatous mastitis. Plasma cell mastitis affects perimenopausal women and produces a lymphoplasmacytic infiltrate surrounding the ducts. Nodules develop around the areola, together with retraction of the nipple (as seen in our patient No. 2). Granulomatous mastitis affects younger women and manifests as a non-caseating inflammatory granuloma located within the lobules and not centered on the ducts [2]. The differential diagnoses are breast carcinoma and infection [3]. There have been a few reports of plasma cell mastitis in patients with inflammatory diseases including SS and RA [3–10] (Table 1). Benign inflammatory breast disease is challenging to treat. The reference standard treatment is high-dose glucocorticoid therapy (0.5–1 mg/kg/day), which is effective in controlling the flares, although relapses are common when the dose is tapered. Methotrexate produced a satisfactory response in some cases [1]. In our two patients, the plasma cell mastitis improved during TNF␣ antagonist therapy. Thus, TNF␣ antagonists may not only hold promise for treating plasma cell mastitis, but also exert a glucocorticoid-sparing effect. Disclosure of interest The authors declare that they have no competing interest.
http://dx.doi.org/10.1016/j.jbspin.2015.12.001 1297-319X/© 2016 Published by Elsevier Masson SAS on behalf of Société française de rhumatologie.
Please cite this article in press as: Faccin M, et al. Plasma cell mastitis in women with rheumatoid arthritis treated with TNF␣ antagonists: Report of 2 cases. Joint Bone Spine (2015), http://dx.doi.org/10.1016/j.jbspin.2015.12.001
G Model BONSOI-4299; No. of Pages 2
ARTICLE IN PRESS Letter to the Editor / Joint Bone Spine xxx (2015) xxx–xxx
2
References [1] Akbulut S, Yilmaz D, Bakir S. Methotrexate in the management of idiopathic granulomatous mastitis: review of 108 published cases and report of four cases. Breast J 2011;17:661–8. [2] Faure C. Chapitre 1 – maladies non cancéreuses du sein. In: Mignotte H, editor. Maladies du sein. 2e éd. Paris: Elsevier Masson; 2011. p. 3–17. [3] Letourneux C, Diemunsch P, Korganow AS, et al. First report of granulomatous mastitis associated with Sjögren’s syndrome. World J Surg Oncol 2013;11:268. [4] Gregoir C, Hilliquin P, Acar F, et al. [Acute mastitis in rheumatoid polyarthritis with Gougerot-Sjögren syndrome treated with tiopronin (Acadione)]. Rev Rhum Mal Osteoartic 1991;58:203–6. [5] Nakamura Y, Yamaguchi M, Nakamura A, et al. Tender, red nodules and arthralgia in a young woman with mastitis. Clin Exp Dermatol 2014;39:410–2. [6] Panzacchi R, Gallo C, Fois F, et al. Primary sarcoidosis of the breast: case description and review of the literature. Pathologica 2010;102:104–7. [7] Provenzano E, Barter SJ, Wright PA, et al. Erdheim-chester disease presenting as bilateral clinically malignant breast masses. Am J Surg Pathol 2010;34: 584–8. [8] Veerysami M, Freeth M, Carmichael AR, et al. Wegener’s granulomatosis of the breast. Breast J 2006;12:268–70. [9] Ogura K, Matsumoto T, Aoki Y, et al. IgG4-related tumour-forming mastitis with histological appearances of granulomatous lobular mastitis: comparison with other types of tumour-forming mastitis. Histopathology 2010;57: 39–45.
[10] Zhang LN, Shi TY, Yang YJ, et al. An SLE patient with prolactinoma and recurrent granulomatous mastitis successfully treated with hydroxychloroquine and bromocriptine. Lupus 2014;23:417–20.
Marine Faccin a Ophélie Caillot b Jean Levêque b Aleth Perdriger a,∗ a Service de rhumatologie, hôpital Sud, 16, boulevard de Bulgarie, BP 90347, 35203 Rennes cedex 2, France b Service de gynécologie-obstétrique, hôpital Sud, 16, boulevard de Bulgarie, BP 90347, 35203 Rennes cedex 2, France ∗ Corresponding author. E-mail address: [email protected] (A. Perdriger)
Accepted 17 July 2015 Available online xxx
Please cite this article in press as: Faccin M, et al. Plasma cell mastitis in women with rheumatoid arthritis treated with TNF␣ antagonists: Report of 2 cases. Joint Bone Spine (2015), http://dx.doi.org/10.1016/j.jbspin.2015.12.001
ARTICLE IN PRESS Joint Bone Spine xxx (2015) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the Editor Plasma cell mastitis in women with rheumatoid arthritis treated with TNF␣ antagonists: Report of 2 cases
a r t i c l e
i n f o
Keywords: Rheumatoid arthritis Plasma cell mastitis Duct ectasia of breast TNF␣ antagonists
Plasma cell mastitis, also known as duct ectasia of the breast, is an inflammatory disease. We report two cases in women with rheumatoid arthritis (RA) treated with TNF␣ antagonists.
Table 1 Reported cases of benign inflammatory breast disease in patients with inflammatory diseases. Inflammatory disease
Number of patients
Year(s) of publication
Reference
Sjögren’s syndrome Rheumatoid arthritis Erythema nodosum Sarcoidosisa Non-langerhans cell histiocytosis Granulomatosis with polyangiitis IgG4-related disease Systemic lupus erythematosus
1 1 8 7 5
2013 1991 1987 to 2014 1939 to 2010 1995 to 2010
[3] [4] [5] [6] [7]
2
1999; 2006
[8]
3 3
2005; 2009; 2010 2000; 2013; 2014
[9] [10]
a
Including 1 patient with sarcoidosis of the breast.
2. Discussion 1. Cases 1.1. Case report No. 1 In 2004, a 52-year-old woman developed recurrent abscess-like lesions in her left breast. Ultrasonography showed heterogeneous and inflammatory tissue remodeling combined with duct ectasia. A biopsy established the diagnosis of plasma cell mastitis with a typical inflammatory infiltrate surrounding the ducts, as well as abscess-like lesions. The abnormalities resolved under prednisone therapy in a daily dosage of 1 mg/kg. In 2008, during the prednisone taper, signs of seropositive RA developed. Methotrexate therapy was initiated, to little effect. In 2010, the prednisone dependency prompted the initiation of infliximab therapy (3 mg/kg every 8 weeks). The manifestations of both the RA and the plasma cell mastitis improved rapidly. At last follow-up after more than 4 years of infliximab therapy, she had had a single recurrence of plasma cell mastitis, which had responded to prednisone therapy.
1.2. Case report No. 2 This 58-year-old woman had a 10-year history of RA with secondary Sjögren’s disease. For the last 2 years she had been taking prednisone 6 mg/day and etanercept 50 mg/week. An abscess-like lesion with nipple retraction developed in the right breast. An infection was suspected and the etanercept was therefore promptly discontinued. Ultrasonography visualized duct ectasia consistent with plasma cell mastitis. The results of the biopsy ruled out carcinoma and infection and showed a non-specific inflammatory response. Re-introduction of etanercept therapy with no increase in the prednisone dosage was followed by regression of the breast abnormalities and ensured good control of the RA.
We describe two cases of plasma cell mastitis in patients with RA that were diagnosed based on the presence of duct ectasia. In both patients, breast infection with abscess formation was ruled out. TNF␣ antagonist therapy improved the clinical manifestations. Benign inflammatory breast disease manifests as breast pain and inflammatory nodules measuring 1 to 2 cm in diameter. The diagnosis rests on the histological findings and results of breast imaging studies (ultrasonography, mammography, and magnetic resonance imaging) [1]. Two variants exist, plasma cell mastitis and granulomatous mastitis. Plasma cell mastitis affects perimenopausal women and produces a lymphoplasmacytic infiltrate surrounding the ducts. Nodules develop around the areola, together with retraction of the nipple (as seen in our patient No. 2). Granulomatous mastitis affects younger women and manifests as a non-caseating inflammatory granuloma located within the lobules and not centered on the ducts [2]. The differential diagnoses are breast carcinoma and infection [3]. There have been a few reports of plasma cell mastitis in patients with inflammatory diseases including SS and RA [3–10] (Table 1). Benign inflammatory breast disease is challenging to treat. The reference standard treatment is high-dose glucocorticoid therapy (0.5–1 mg/kg/day), which is effective in controlling the flares, although relapses are common when the dose is tapered. Methotrexate produced a satisfactory response in some cases [1]. In our two patients, the plasma cell mastitis improved during TNF␣ antagonist therapy. Thus, TNF␣ antagonists may not only hold promise for treating plasma cell mastitis, but also exert a glucocorticoid-sparing effect. Disclosure of interest The authors declare that they have no competing interest.
http://dx.doi.org/10.1016/j.jbspin.2015.12.001 1297-319X/© 2016 Published by Elsevier Masson SAS on behalf of Société française de rhumatologie.
Please cite this article in press as: Faccin M, et al. Plasma cell mastitis in women with rheumatoid arthritis treated with TNF␣ antagonists: Report of 2 cases. Joint Bone Spine (2015), http://dx.doi.org/10.1016/j.jbspin.2015.12.001
G Model BONSOI-4299; No. of Pages 2
ARTICLE IN PRESS Letter to the Editor / Joint Bone Spine xxx (2015) xxx–xxx
2
References [1] Akbulut S, Yilmaz D, Bakir S. Methotrexate in the management of idiopathic granulomatous mastitis: review of 108 published cases and report of four cases. Breast J 2011;17:661–8. [2] Faure C. Chapitre 1 – maladies non cancéreuses du sein. In: Mignotte H, editor. Maladies du sein. 2e éd. Paris: Elsevier Masson; 2011. p. 3–17. [3] Letourneux C, Diemunsch P, Korganow AS, et al. First report of granulomatous mastitis associated with Sjögren’s syndrome. World J Surg Oncol 2013;11:268. [4] Gregoir C, Hilliquin P, Acar F, et al. [Acute mastitis in rheumatoid polyarthritis with Gougerot-Sjögren syndrome treated with tiopronin (Acadione)]. Rev Rhum Mal Osteoartic 1991;58:203–6. [5] Nakamura Y, Yamaguchi M, Nakamura A, et al. Tender, red nodules and arthralgia in a young woman with mastitis. Clin Exp Dermatol 2014;39:410–2. [6] Panzacchi R, Gallo C, Fois F, et al. Primary sarcoidosis of the breast: case description and review of the literature. Pathologica 2010;102:104–7. [7] Provenzano E, Barter SJ, Wright PA, et al. Erdheim-chester disease presenting as bilateral clinically malignant breast masses. Am J Surg Pathol 2010;34: 584–8. [8] Veerysami M, Freeth M, Carmichael AR, et al. Wegener’s granulomatosis of the breast. Breast J 2006;12:268–70. [9] Ogura K, Matsumoto T, Aoki Y, et al. IgG4-related tumour-forming mastitis with histological appearances of granulomatous lobular mastitis: comparison with other types of tumour-forming mastitis. Histopathology 2010;57: 39–45.
[10] Zhang LN, Shi TY, Yang YJ, et al. An SLE patient with prolactinoma and recurrent granulomatous mastitis successfully treated with hydroxychloroquine and bromocriptine. Lupus 2014;23:417–20.
Marine Faccin a Ophélie Caillot b Jean Levêque b Aleth Perdriger a,∗ a Service de rhumatologie, hôpital Sud, 16, boulevard de Bulgarie, BP 90347, 35203 Rennes cedex 2, France b Service de gynécologie-obstétrique, hôpital Sud, 16, boulevard de Bulgarie, BP 90347, 35203 Rennes cedex 2, France ∗ Corresponding author. E-mail address: [email protected] (A. Perdriger)
Accepted 17 July 2015 Available online xxx
Please cite this article in press as: Faccin M, et al. Plasma cell mastitis in women with rheumatoid arthritis treated with TNF␣ antagonists: Report of 2 cases. Joint Bone Spine (2015), http://dx.doi.org/10.1016/j.jbspin.2015.12.001