title: Mastitis associated with Sjögren's syndrome: a series of nine cases.
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".
authors: 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.|UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.
authors: Radjiv
authors: R
authors: Goulabchand
authors: UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Pathology Department, Gui de Chauliac Hospital, Montpellier University, 80 Avenue Augustin Fliche, 34295, Montpellier, France.
authors: Assia
authors: A
authors: Hafidi
authors: UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Medical Imaging Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France.
authors: Ingrid
authors: I
authors: Millet
authors: UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Rheumatology Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France.
authors: Jacques
authors: J
authors: Morel
authors: UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Rheumatology Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France.
authors: Cédric
authors: C
authors: Lukas
authors: 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.|UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Internal Medicine Department, Jean Minjoz Hospital, Besançon University, 3 Boulevard Fleming, 25030, Besançon, France.
authors: Sébastien
authors: S
authors: Humbert
authors: 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.|UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.
authors: Sophie
authors: S
authors: Rivière
authors: Neurology Department, Gui de Chauliac Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France.
authors: Christian
authors: C
authors: Gény
authors: UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Clinical Immunology and Osteoarticular Diseases Therapeutic Unit, Lapeyronie Hospital, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France.|Inserm U1183, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295, Montpellier, France.
authors: Christian
authors: C
authors: Jorgensen
authors: 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.|UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.
authors: Alain
authors: A
authors: Le Quellec
authors: UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.|Pathology Department, Gui de Chauliac Hospital, Montpellier University, 80 Avenue Augustin Fliche, 34295, Montpellier, France.
authors: Hélène
authors: H
authors: Perrochia
authors: 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. p-guilpain@chu-montpellier.fr.|UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France. p-guilpain@chu-montpellier.fr.|Inserm U1183, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295, Montpellier, France. p-guilpain@chu-montpellier.fr.
authors: Philippe
authors: P
authors: Guilpain
mesh_terms: D000368:Aged; D001940:Breast / Q000473:pathology; D005260:Female; D006801:Humans; D008413:Mastitis / Q000209:etiology* / Q000473:pathology; D008875:Middle Aged; D012469:Salivary Glands / Q000473:pathology; D012859:Sjogren's Syndrome / Q000150:complications* / Q000473:pathology
keywords: Epithelial glandular cell; Lymphocytic infiltrates; Mammary duct ectasia; Mastitis; Sjögren’s syndrome