Vol.2, No.9, 525-529 (2013) Case Reports in Clinical Medicine
http://dx.doi.org/10.4236/crcm.2013.29137
Bilateral involvement in patients with granulomatous
mastitis: Surgical treatment and case report
P. Orsaria1, F. De Sanctis1, A. Esser1, L. Dori2, E. Bonanno3, G. Petrella1, O. Buonomo1
1Department of Surgery, Tor Vergata University Hospital, Rome, Italy;
2Department of Internal Medicine, Tor Vergata University Hospita l , Rome , Italy
3Department of Biopathology, Tor Vergata University Hospital, Rome, Italy
Received 18 October 2013; revised 20 November 2013; accepted 11 December 2013
Copyright © 2013 P. Orsaria et al. This is an open access article distributed under the Creative Commons Attribution License, which
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ABSTRACT
Introduction: Granulomatous mastitis (GM) is a
rare benign histopathologic lesion, associated
with tissue inflammation, architectural distor-
tions and heterogeneous parenchymal inflam-
mation upon radiological evaluation. The treat-
ment of GM is controversial, and currently, there
is no consensus about the most appropriate
therapy. Case Presentation: We presented a uni-
que, atypical GM case with a prolonged disease
course that ultimately led to a bilateral mastec-
tomy. A conservative therapeutic approach and
limited or wide surgical excisions have failed to
prevent unfavorable outcomes in both the initial
present ation and recurrent disease. Conclusions:
There’re no clear data in the literature delineat-
ing persistent recurrences of GM after conser-
vative treatment and multiple surgeries. Obtain-
ing a disease-free surgical margin might be an
import ant prognostic fa ctor for a las ting relapse-
free clinical resolution.
Keywords: Granulomatous Ma stitis; Medical
Treatment; Breast Conserving Therapy; Surgical
Margins
1. INTRODUCTION
Granulomatous mastitis (GM) is a rare benign histo-
pathologic lesion associated with tissue inflammation,
architectural distortions and heterogeneous parenchymal
inflammation upon radiologic evaluation. Although an
understanding of the etiology and association of GM
with local trauma, autoimmunity, local chemical irrita-
tions or infection is needed, it is possible that damage to
the ductal epithelium might represent the pathogenic
cause for subsequent structural changes in the affected
tissue [1]. The inflammatory infiltrate is generally con-
fined to the breast lobules and comprises histiocytes,
polymorphonuclear leukocytes, and multinucleated giant
cells of the foreign body and Langhans type. GM typi-
cally presents with sinus formation or abscesses, sug-
gesting carcinoma, but radiologic imaging is limited in
distinguishing GM from malignancy. An awareness of
this rare entity is important for the pathologist, as the
definitive diagnosis of GM is microscopically deter-
mined through Core Needle Biopsy (CNB) or excisional
biopsy of a tissue sample [2]. The treatment of GM is
controversial, and there is no consensus about the most
appropriate therapy. Patients receiving steroid therapy
should be closely observed for side effects. Furthermore,
the potential involvement of an infectious agent often
challenges the use of steroids for GM treatment. The
feasibility of surgical management represents an impor-
tant choice modality for a shorter time required with
fewer complications. The wide excision of the mass is
typically performed, but the recurrence rate (RR) is re-
portedly higher than that observed with conservative
treatment [3]. In the present study, we present a case of
GM successfully treated with radical surgery for an ag-
gressive disease with bilateral involvement and positive
microbiological findings (Table 1), which has rarely
been reported in the literature.
2. CASE REPORT
A 50-year-old Czech woman, was admitted to the hos-
pital in October 2011 for recurrent inflammatory left
breast disease, previously treated in a different hospital
using antibiotics and local excision with no clinical re-

P. Orsaria et al. / Case Reports in Clinical Medicine 2 (2013) 525-529
526
Table 1. Microbiological findings.
Microorganisms Sample Time Site
Klebsiella pneumoniae spp. pneumoni a e ; Escherichia
coli; Enterococcus faecalis Wound swab October 2011 Left breast
Stenotrophomona s maltophila Pus drainage November 2011 Left breast
Candida tropicalis Wound swab December 2011 Left breast
Klebsiella pneumoniae. spp. pneumoniae; Enterococcus
casseliflavus; Saccharomyces cerevisiae Surgical s a mple (mastectomy)February 2012 Left breast
Escherichia coli; Enterococcus faecium; Staphylococcus
epidermidis Wound swab March-April 2012 Surgical wound l e ft breast
Enterococcus caselliflavus, Klebsiella pneumoniae
spp. pneumoniae; Candida parapsilosis Surgical sample (mas t e c t omy)February 2013 R i g h t b reast
sponse. The past medical history was characterized by
one pregnancy without complication, nicotine addiction,
positive familiar history for breast cancer and the surgi-
cal removal of a cardiac recorder (placed for syncopal
episode), accidentally mobilized from the subclaveal side
to the left breast in August 2010.
A physical examination revealed a hard, painful, hy-
peremic mass in the upper medial portion of the left
breast with no palpable axillary lymphadenopathy. The
skin overlying the mass and the contralateral breast was
normal. The systems review was positive for low-grade
fever. A mammogram revealed an ill-defined periareolar
lesion extending into the upper quadrant with increased
density and distortion. The ultrasound showed diffuse
interstitial edema with fibro-glandular tissue consistent
with inflammation and the presence of a mixed echo-
structure nodular alteration, measuring 1.6 cm × 1.8 cm
× 0.9 cm, consistent with partially organized fluid collec-
tion. A breast MRI revealed thickening of the skin and
subcutaneous tissue of the left breast, with an abnormal
nodal enhancement pattern, lymphatic vasculature and
abscessual fluid collections. Because of the worsening
symptoms, a total body computerized tomography (CT)
was performed, which showed no damage to other or-
gans.
The results of seriated blood tests were within normal
limits, but the index of inflammation was increased.
Scant purulent fluid was collected using a fine needle
stain, and after further examination, a presumptive diag-
nosis of spontaneous infectious mastitis was made. The
wound cultures sub sequently showed positivity for Kleb-
siella pneumoniae, Escherichia coli and Enterococcus
faecalis.
As a first line of treatment, surgical drainage and tar-
geted antibiotics with corticosteroids were conservatively
administered for 2 weeks. After the completion of a
6-week course of therapy, the symptoms remained and
appeared to worsen. Thus, the patient underwent an exci-
sional biopsy of the lesion using a vacuum assisted clo-
sure therapy device. The pathology examination showed
findings consistent with GM and no evidence of carci-
noma or pathogenic organisms.
Four weeks later, the patient noticed a new growth in
the site of the previous surgical resection, and subse-
quently a second surgical drainage of the breast abscess
was performed. The microbiological examination was
positive for Stenotrophomonas maltophila, and the new
wound swab culture was positive for Candida tropicalis.
Because the symptoms worsened, despite targeted
medical therapies, a modified mastectomy was per-
formed, including all involved soft and breast glandular
tissues. The histological examination revealed multiple
foci of acute inflammation, with amorphous material
fragments attributable to the previous electromedical
device accidentally mobilized from the subclaveal side.
The microbiological examination was positive for Kleb-
siella pneumoniae, Enterococcus casseliflavus and Sac-
charomyces cerevisiae. The slow healing of the surgical
wound, with pus drain age, was observed, and the wou nd
swabs were positive for Escherichia coli, Enterococcus
faecium and Staphylococcus epidermidis. Hospitalization
was long (96 days), and the patient showed complete
resolution of the disease, without apparent recurrence.
After one year, the patient showed the same clinical
presentation in the right breast. In addition, the tissue of
right breast was painful and an eschar was observed
(Figure 1).
Basing on the findings of previous microbiological
examinations, an empirical antibiotic therapy using coli-
mycin, caspofungin and teicoplanin was administered.
After an initial improvement, the clinical presentation
worsened. An additional eschar was observed upon pal-
pation, with tenderness and diffuse swelling. In addition,
two fistulas, which discharged pus when pressuring the
breast, were also observed, and the breast was extremely
painful.
Mammography revealed an asymmetric density in-
crease with indistinct margins in the retroareolar region
and upper outer quadrant of the right breast. No micro-
calcification was evident. The breast ultrasonography
showed a loculated fluid alteration of 5 cm in the inner
quadrants, associated with hypoechoic branching in cu-

P. Orsaria et al. / Case Reports in Clinical Medicine 2 (2013) 525-529 527
Figure 1. The right breast of a GM patient with several pus-
filled fistulas and recurrence after a previous contralateral mas-
tectomy for the same breast disease. The patient presented
acute inflammatory symptoms, such as local temperature in-
crease, hyperemia, and edema in the breast.
taneous fistulas. However, the mammary tissue was ede-
matous. The breast magnetic resonance image (MRI)
revealed thickening of the mammary skin of the right
breast alongside nodular lesions and fluid collections in
different locations and of various sizes with enhancing
mass lesion (Fi gure 2).
Admission laboratory testing was significant for the
detection of leukocytosis with bandemia. The ESR and
CRP were 79 mm/h and 76.92 mg/L, respectively. Anti-
nuclear antibodies, cytoplasmic antibodies, rheumatoid
factor and anticyclic citrullinated peptide were negative.
We attempted to identify immunologic, coagulation and
chemical markers, as shown in Table 2, without evidence
of diagnostic specificity.
The clinical symptoms and personal preference, asso-
ciated with a prolonged disease course and multiple re-
currences, ultimately led to radical mastectomy. Histo-
logical staining revealed acute and chronic xanthogranu-
lomatous flogosis, consistent with GM (Figure 3). The
microbiological examination was positive for Entero-
coccus caselliflavus, Klebsiella pneumoniae and Can-
dida parapsil o si s.
At 4 days post-operation, the patient was released in
good status. Th e hospitalization lasted 8 d ays, with rapid
patient rehabilitation and no apparent recurrence.
4. DISCUSSION
In this study, we evaluated patients with recurrent and
bilateral GM, with a long history of failed multiple treat-
ment modalities and permanent cure through radical sur-
gery. The involvement was typically unilateral, as bilat-
eral involvement had rarely been reported in the litera-
ture [4]. The primary problems of GM include the high
rate of local relapse and the long duration required for
the complete resolution of the disease symptoms. All treat-
ment entities were associated with a recurrence rate of
16% - 50%, maintaining the patient under medical care
Figure 2. Post medical treatment image of right contralateral
breast recurrence after a previous left mastectomy. The MRI
shows diffuse enhancement in different areas. Mass lesions,
non-suggestive of a specific diagnosis, parenchymal inflamma-
tion, and remarkable thickening in the mammary skin of the
right breast were observed.
Table 2. Analysis of the immunological profile and coagulation
blood tests during the second hospitalization.
Silica test 0.98 Negative
APTT test Negative
Kaolin test 0.84 Negative
Russell Viper Venom test 1.02 Negative
dRVVT test Negative
Lupus Anticoagulant test Negative
Complement Factor C3 171.00 mg/dL Negative
Complement Factor C4 45.70 mg/dL Positive
Anti-cardiolipin antibodies IgM 11.68 UI/mL Negative
Anti-cardiolipin antibodies IgG 3.86 UI/mL Negative
Anti-dsDNA antibodies <12.30 U/ml Negative
ENA screen test 4.7 Negative
Anti-Jo1 antibodies Negative
Anti-SS-B antibodies Negative
Anti-PM/Scl antibodies Negative
Anti-Scl 70 antibodies Negative
Anti-Sm antibodies Negative
Anti-Sm/RNP antibodies Negative
Anti-CENP-A/B antibodies Negative
Anti-Ro (SS-A 60 kDa) antibodies Negative
CD 19+ 13%
CD 3+ 74%
CD3+CD4+ 41%
CD3+CD8+ 29%
CD16+CD56+ 8%
CD4/CD8 1.40

P. Orsaria et al. / Case Reports in Clinical Medicine 2 (2013) 525-529
528
Figure 3. Morphological examination of right contralateral
breast recurrence after a previous left mastectomy: Panel A
(magnification 2×) shows that the breast tissue was almost
completely substituted with necrotic tissue, surrounded by
heavy polymorph nucleate infiltrate extending from the skin
(arrow) to the adipose tissue (asterisk). Panel B (magnification
10×) shows that only a few islands of breast tissue were ob-
served (arrow). Panel C (higher magnification of the insert in
panel B) shows the results of an electron microscopy study
demonstrating the presence of macrophages and Panel D) mi-
croorganisms (asterisk), namely, Candida albicans, character-
ized by a multilayered wall (arrow heads) surrounding a disor-
ganized cytoplasm (asterisk).
for a prolonged period [5]. The overall trend among these
patients is a long history of disease, accompanied by an
enormous psychological burden, necessitating a reliable,
safe and rapid solution. Currently, the most commonly
applied treatment options are conservative modality
through the administration of systemic antibiotics, corti-
costeroid therapy and surgical excision.
Often, medical treatment with corticosteroids signifi-
cantly suppresses inflammation, facilitating more con-
servative surgery. However, prolonged steroid use gener-
ates local and systemic side effects and increases the
potential to develop tolerance. In a retrospective analysis,
none of the 19 patients with GM displayed a complete
clinical response to the 8-week steroid treatment, and
local excisions of the remaining lesions were performed
with good cosmetic results in all patients [6]. Further-
more, the initiation of steroids is often limited by con-
cerns related to the presence of an infectious etiology, as
described above. Thus, steroids are primarily adminis-
tered only in recurrent cases.
Although a variable percentage of cases of GM appear
aseptic, case reports of documented co-infection with
coexistent organisms have been reported in the UK,
France and Italy. Follow-up examinations, after the exci-
sion of the mass, suggest that wound infection is a fre-
quent problem [11]. However, a specific antibiotic ther-
apy might be ineffective based on the anatomical and
functional features of the breast and the disease extent.
Further studies are needed to establish this hypothesis,
suggesting that an initial pathogenic insult might be re-
sponsible for generating a subsequent autoimmune re-
sponse, with further damage to lobular structures. In the
present study, the results of the microbiological examina-
tions, showing different germs at different times and an
inefficacy of the targeted antibiotic therapy, suggest
pathogenic colon ization rather than infection. In addition,
the presence of a local immunity deficit due to the ana-
tomical structure of the mammary gland has also been
suggested. However, in the context of a potential multi-
factorial etiology, the present study illustrates the con-
troversial nature of an optimal treatment paradigm. There
is no accepted management strategy for GM. Limited
excision alone has little benefit, although this treatment
plays an important role in the diagnostic pathway, there
is a strong tendency of recurrence. A relapse rate of 5%
to 50% was reported after surgical excision of the mass
[7]. Furthermore, there is a high rate of poor wound
healing and disfigurement after surgical intervention.
Disease recurrence and fistula formation are known com-
plications of GM, and in some cases, excision of the re-
currence alone is not an adequate treatment, unless this
procedure complies with the criteria of negative surgical
margins in terms of inflammatory tissue [1]. In the case
reported above, granulomatous deposits from the residual
breast tissue were observed after previous excisions, po-
tentially reflecting recurrence and emphasizing the ne-
cessity of mastectomy in patients with extensive struc-
tural alterations. Hladik et al. presented three patients,
with a long history of recurrences, which were success-
fully treated with mastectomy and immediate breast re-
construction. However, in one case, skin-sparing mas-
tectomy (SSM) and primary reconstruction with a trans-
verse myocutaneous gracilis flap (TMGF) were ineffec-
tive because abscesses recurred from residual breast tis-
sue [8]. In a retrospective review of cases observed over
25 years, Al-Khaffal et al. showed that regardless of the
therapeutic intervention, which included steroids, antibi-
otics, and surgical intervention, alone or in combination,
complete resolution took approximately 6 to 12 months.
Consistently, the overall outcomes were not associated
with any combination of treatment options [9]. In our
experience, the patient had previously received multiple
courses of oral and parenteral antibiotics, without im-
provement, and required bilateral mastectomy due to the
severe and recurrent nature of the disease. We are aware
of only eight other cases requiring co mplete mastectomy.

P. Orsaria et al. / Case Reports in Clinical Medicine 2 (2013) 525-529 529
[4] Ehan, Y., Vera, A., Kara, E., Ozdemir, N., Kaprac, M.,
Ozdedeli, E., Yilmaz, R., Koyuncu, A., Erhan, Y. and Oz-
bal, O. (2000) A clinicopathologic study of a rare clinical
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http://dx.doi.org/10.1054/brst.1999.0072
However, the contralateral recurrence that occurred
almost 18 months after treatment, prompt surgical ther-
apy improved the overall outcomes in the duration of
clinical resolution compared with a conservative clinical
approach. We reported an unusual presentation of IGM
which had bilateral in volvement and an unusual progres-
sion after radical treatment. Demolitive surgery repre-
sents a co nsid erab le burden fo r breas t surgery specia lists,
but new oncoplastic strategies, with the support of bio-
logical and prosthetic materials, could achieve immediate
reconstruction during a second operation [10]. Clearly
effective feedback, in the context of a multidisciplinary
team, is vital in these challenging cases. Indeed, multiple
microhistological biopsies and a complex radiological
definition of the disease extent might contribute to accu-
rate diagnosis and adequate clinical management.
[5] Kok, K.Y. and Telisinghe, P.U. (2010) Granulomatous
mastitis: Presentation, treatment and outcome in 43 pa-
tients. Surgeon, 8, 197-201.
http://dx.doi.org/10.1016/j.surge.2010.02.002
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5. COMPETING INTEREST
No benefits in any form have been received or will be
received from a commercial party related directly or in-
directly to the subject of this article. [8] Hladik, M., Schoeller, T., Ensat, F. and Wechselberger, G.
(2011) Idiopathic granulomatous mastitis: Successful treat-
ment by ma stectomy an d immediate breast reconst ruction.
Journal of Plastic, Reconstructive Aesthetic Surgery, 64,
1604-1607. http://dx.doi.org/10.1016/j.bjps.2011.07.011
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ABBREVIATIONS CT: Computerized Tomography;
ESR: Erythrocyte Sedimentation Rate;
GM: Granulomatous Mastitis; CRP: C-Reactive Protein;
CNB: Core Needle Biopsy; SSM: Skin-Sparing Mastectomy;
RR: Recurrence Rate; TMGF: Transverse Myocutaneous Gracilis flap.
MRI: Magnetic Resonance Imaging;
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