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Mastitis associated with Sjögren’s syndrome: a series of nine cases

  • Therapeutic Aspects in Autoimmunity
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Abstract

Sjögren’s syndrome is well known to target exocrine glands, especially lacrimal and salivary glands, which share with mammary glands anatomical, histological, and immunological features. Herein, we investigated the mammary involvement in patients with Sjögren’s syndrome and compared the histological findings with minor salivary gland involvement. We reviewed the charts of patients with Sjögren’s syndrome (followed in Montpellier University Hospital, between January 2000 and January 2015), in whom minor salivary gland and mammary tissues were available. Two expert pathologists analysed retrospectively these tissues in order to identify inflammatory patterns. Immunohistochemical stainings were performed to precise leucocyte distribution. Sixteen Sjögren’s syndrome patients with available salivary and breast tissue samples were included. All were women, with a median age of 60.1 ± 11.3 years at Sjögren’s syndrome diagnosis. Mammary biopsy was conducted because of breast symptoms in 6 patients and following imaging screening strategies for breast cancer in 10 patients. Nine patients exhibited an inflammatory breast pattern (lymphocytic infiltrates or duct ectasia), close to minor salivary gland histological findings. Immunohistochemical stainings (n = 5) revealed B and T cell infiltrates within breast tissue, with a higher proportion of T CD4+ cells, but no IgG4-secreting plasma cells were found. This is the first series to describe breast inflammatory patterns in Sjögren’s syndrome. Mastitis is in line with the classical involvement of exocrine glands in this disease. These findings are consistent with the literature data considering Sjögren’s syndrome as an “autoimmune epithelitis”.

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Acknowledgments

We owe a special thank to Dr A. Garnier (Pathological laboratory, Montpellier), Pr Jean-Emmanuel Kahn (Internal Medicine, Foch Hospital, Suresnes, France), Pr Marc Michel (Internal medicine department, Mondor Hospital, Crétéil, France), all the Pathological department laboratory technicians (Montpellier Hospital) and A Nadaradjane for their kindly help in this work.

Authors Contribution

Radjiv Goulabchand (RG) and Philippe Guilpain (PG) conceived this study. Radjiv Goulabchand (RG), Assia Hafidi (AH), and Hélène Perrochia (HP) performed the histological analysis and captures. Radjiv Goulabchand (RG), Assia Hafidi (AH), Hélène Perrochia (HP), and Philippe Guilpain (PG) conducted interpretation and analysis. Radjiv Goulabchand (RG), Hélène Perrochia (HP), Assia Hafidi (AH), and Philippe Guilpain (PG) contributed to drafting the manuscript. Philippe Guilpain, Ingrid Millet, Jacques Morel, Cédric Lukas, Sébastien Humbert, Sophie Rivière, Christian Gény, Christian Jorgensen, and Alain Le Quellec have taken part in recruiting patients and collecting data. All authors have taken part in drafting the article or revising it critically for important intellectual content and final approval of the version to be published.

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Authors and Affiliations

  1. Department of Internal Medicine, Maladies Multi-Organiques, Centre de compétence “maladies systémiques et auto-immunes rares”, St Eloi Hospital, CHRU de Montpellier, 80 Avenue Augustin Fliche, 34295, Montpellier Cedex 5, France

    Radjiv Goulabchand, Sébastien Humbert, Sophie Rivière, Alain Le Quellec & Philippe Guilpain

  2. UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France

    Radjiv Goulabchand, Assia Hafidi, Ingrid Millet, Jacques Morel, Cédric Lukas, Sébastien Humbert, Sophie Rivière, Christian Jorgensen, Alain Le Quellec, Hélène Perrochia & Philippe Guilpain

  3. Pathology Department, Gui de Chauliac Hospital, Montpellier University, 80 Avenue Augustin Fliche, 34295, Montpellier, France

    Assia Hafidi & Hélène Perrochia

  4. Medical Imaging Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France

    Ingrid Millet

  5. Rheumatology Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France

    Jacques Morel & Cédric Lukas

  6. Internal Medicine Department, Jean Minjoz Hospital, Besançon University, 3 Boulevard Fleming, 25030, Besançon, France

    Sébastien Humbert

  7. Neurology Department, Gui de Chauliac Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France

    Christian Gény

  8. Clinical Immunology and Osteoarticular Diseases Therapeutic Unit, Lapeyronie Hospital, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France

    Christian Jorgensen

  9. Inserm U1183, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295, Montpellier, France

    Christian Jorgensen & Philippe Guilpain

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  1. Radjiv Goulabchand
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  2. Assia Hafidi
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  3. Ingrid Millet
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  7. Sophie Rivière
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  8. Christian Gény
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  9. Christian Jorgensen
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  11. Hélène Perrochia
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Correspondence to Philippe Guilpain.

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Goulabchand, R., Hafidi, A., Millet, I. et al. Mastitis associated with Sjögren’s syndrome: a series of nine cases. Immunol Res 65, 218–229 (2017). https://doi.org/10.1007/s12026-016-8830-x

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