Is granulomatous mastitis a localized form of hidradenitis suppurativa?

ARTICLE

Auteur(s) : Olivier Join-Lambert^1,2, Sylvie Fraitag³, Florence Ribadeau-Dumas^1,4, Anne-Sophie Leguern¹, Sylvie Behillil¹, Francisco-J Del Castillo^1,5, Paul-Henri Consigny¹, Frédéric Auquier⁶, François Eb⁶, Henri Sevestre⁶, Olivier Lortholary^1,4, Xavier Nassif^1,2, Aude Nassif¹

¹Centre d’Infectiologie, Necker-Pasteur, Institut Pasteur, Centre Médical, 25 rue du Docteur Roux, 75724 Paris Cedex 15, France
²Laboratoire de Microbiologie, Hôpital Necker-Enfants malades, 149 rue de Sèvres 75015 Paris,France
³Laboratoire d’Anatomie et de Cytologie Pathologiques, Hôpital Necker-Enfants malades, 149, rue de Sèvres 75015 Paris, France
⁴Service de Maladies Infectieuses et Tropicales, Hôpital Necker-Enfants malades, 149, rue de Sèvres 75015 Paris, France
⁵Unidad de Genética Molecular Hospital Ramon y Cajal Carretera de Colmenar, Km 9 28034-Madrid-Spain
⁶Services de Gynécologie Obstétrique, Microbiologie et Anatomie Pathologique, Centre Hospitalo-Universitaire d’Amiens, 2 place Victor Pauchet, 80054 Amiens, France

Granulomatous lobular mastitis (GM) is a rare chronic and relapsing disease simulating inflammatory carcinomatous mastitis [1]. It is usually a surgical and histological surprise showing an abscess instead of the suspected cancer. Hidradenitis suppurativa (HS) is also a chronic inflammatory skin disease affecting 1% of the population in Europe, characterized by recurrent abscesses, usually involving axillae, groins, buttocks, and rarely the breast. So far, there is no specific demonstrated etiology or causative mechanism for HS or GM. Wide surgery is the mainstay of therapy for severe cases.

We describe a patient who had typical inguinal and axillary HS for 12 years and developed a breast lesion. Histology revealed typical GM lesions.

A 35-year-old female presenting HS since age of 23 was seen at Pasteur Institute Medical Center in 2007 with 5 active HS nodules on her buttocks and groin. In the following month, 10 new HS nodules appeared in previously affected sites and one in the breast, causing nipple retraction. Mammography and ultrasound did not rule out cancer. Surgical biopsy showed a non-caseating inflammatory granuloma with lymphocytes, plasmocytes and numerous polymorphonuclear neutrophils consistent with the diagnosis of GM [2]. Bacteriological cultures isolated Proteus mirabilis and a coryneform Gram positive rod. A month later, because of a major painful enlargement of the breast, a new surgical drainage was performed, giving identical histology and microbiology results. Because of a third flare within a month, wide excision of the breast lesion was proposed. However, based on literature suggesting a role for coryneform bacteria in GM [3-5], amoxicillin and moxifloxacin were prescribed. After 6 weeks of treatment, the breast lesion was significantly reduced (figure 1), no longer oozing and most other HS affected sites (11/14) simultaneously completely healed. Antibiotic treatment was continued for 6 months until stabilization of GM and HS.

GM usually presents in young women as a painful breast nodule leading spontaneously to chronic fistulas with purulent discharges for months, potentially destructive in some cases. The diagnosis is obtained by histopathology showing lobular granulomas [2]. GM is idiopathic in most cases, but has been associated with sarcoidosis, Wegener’s disease and Churg-Strauss syndrome. For the past 12 years, owing to a prolonged biopsy culture for up to 14 days, different authors have reported the association of Corynebacterium sp to GM lesions with a beneficial antibiotherapy [3-5].

We report the first case of typical GM occurring in an HS patient. Our HS patient’s breast lesion did not differ clinically from breast HS, suggesting that HS and GM may be close entities. A careful review of the literature indeed revealed that, except for the localization of the disease, the clinical picture and natural evolution of GM lesions are similar to HS [1, 4, 5]. Interestingly, in a series of 34 GM patients, one patient developed a secondary lesion in the axillae that was compatible with a lesion of HS [5]. The link between these two entities may have been missed since GM and HS patients are seen by gynaecologists and dermatologists respectively. In addition, unlike in GM, the diagnosis of HS is clinical and biopsies are seldom performed.

Finally, in our case, the antimicrobial therapy prescribed to treat GM caused a dramatic remission of both GM and HS lesions. Even though histology can suggest an infectious process both in GM and HS, the role of bacteria of the skin flora within GM and HS lesions is debated as they could multiply in pre-existing inflammatory lesions. However, antibiotics can be helpful to treat HS flares and GM [3-6]. This case suggests that, although affecting different sites of the skin, GM and HS are two close entities.

Acknowledgements

References

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3 Ang LM, Brown H. Corynebacterium accolens isolated from breast abscess: possible association with granulomatous mastitis. J Clin Microbiol 2007; 45: 1666-8.

4 Mathelin C, Riegel P, Chenard MP, Tomasetto C, Brettes JP. Granulomatous mastitis and corynebacteria: clinical and pathologic correlations. Breast J 2005; 11: 357.

5 Taylor GB, Paviour SD, Musaad S, Jones WO, Holland DJ. A clinicopathological review of 34 cases of inflammatory breast disease showing an association between corynebacteria infection and granulomatous mastitis. Pathology 2003; 35: 109-19.

6 Leyden JJ, Revuz J. Antibiotic therapy of hidradenitis suppurativa. In: Jemec GBE, Revuz J, Leyden JJ, eds. Hidradenitis suppurativa, First ed. Berlin Heildelberg: Springer-Verlag, 2006: 120-3.