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Male tuberculous mastitis

Case report

Clin Ter 2013; 164 (4):e??-??. doi: 10.7417/CT.2013.????

Male tuberculous mastitis: a rare entity


C. Cantisani1, T. Lazic2, M. Salvi1, A.G. Richetta.1, F. Frascani1, F. De Gado1, C. Mattozzi1, G. Fazia1,
S. Calvieri1
1
Department of Dermatology, Sapienza University, Rome, Italy, 2Department of Dermatology and Skin Surgery, Roger Williams
Medical Center/Boston University, Providence, Rhode Island, USA

Abstract
A 28-year old male presented to our clinic complaining of a recent
onset of a painful right breast lump with redness and nipple discharge.
Fine-needle aspiration biopsy revealed caseating granulomas, with a
culture positive for Mycobacterium tuberculosis. He was found to have
a positive PPD, but no other site of pulmonary or extra-pulmonary
tuberculosis was identified. Treatment with anti-tuberculous drugs lead
to complete clinical resolution of the breast lesion. The breast is a rare
site of extra-pulmonary tuberculosis (TB), comprising only 0.1% of
all cases. TB is re-emerging in the Western world with the increasing
prevalence of immunosuppressive disorders. Increasing immigration
rates and widespread travel are further contributing to TB globalization.
With the re-emergence of TB, atypical forms are appearing, with an
increase in the proportion of extra-pulmonary disease and a widening
of the age range at presentation. Tuberculous mastitis(TM) is found
mostly in young, multiparous women. Male TM is extremely rare,
and accounts for only 4% of all cases. This strikingly lower incidence
in males points towards a significant role of parity, pregnancy and
lactation as likely predisposing factors. Although a rare disease, TM
is an important differential diagnosis for breast cancer. A high index of
suspicion is the cornerstone for diagnosis. Awareness of this condition
is important not only for dermatologists, but for surgeons, radiologists
and pathologists, as well. Clinicians are encouraged to provide a careful
assessment of the breasts, an important organ also in men. Clin Ter
2013; 164(4):e??-??. doi: 10.7417/CT.2013.????
Key words: antituberculous drugs, breast cancer, caseating granulomas, mastitis, tuberculosis

the male breast (2). Most other diseases found in the male
breast arise from the skin and subcutaneous tissues (e.g., fat
necrosis, lipoma, epidermal inclusion cysts). Some lesions
that are common in the female breast (e.g., fibroadenomas)
do not occur in the male breast. Breast pain (mastalgia) in
males is a rare phenomenon.
Mastitis is the inflammation of the mammary gland;
there are two types: puerperal and non puerperal mastitis.
Puerperal mastitis is the inflammation of breast in connection with pregnancy, breastfeeding or weaning. It is usually
caused by blocked milk ducts or milk excess. In fact the
most prominent symptoms is tension and engorgement of
the breast. If untreated, the milk left in the breast tissue can
become infected, leading to infectious mastitis. It is relatively common; however only about 0.4-0.5% of breastfeeding
mothers develop an abscess (3).
Nonpuerperal mastitis is the inflammation of breast tissue
occurring unrelated to pregnancy and breastfeeding. This
is caused by a wide range of organisms, including gramnegative and gram-positive bacteria and mycoplasmas4.
Staphylococcus aureus is the most common etiological
organism responsible, but staphylococcus epidermidis and
streptococci are occasionally identified as well. It is important to receive treatment immediately to prevent complications, such as an abscess in the breast (5).
Nonpuerperal mastitis is also caused by Mycobacterium
tubercolosis. Tuberculous mastitis is an uncommon form
of extrapulmonary tuberculosis. It is predominant in young
women; the common age concerned between 20 to 40 years,
the period of reproductive age (6). Breast tuberculosis is
extremely rare in males.

Introduction

Benign breast disorders, classified by the ANDI system


(aberrations of normal development and involution), constitute the major workload in breast clinics in women (1). The
majority of breast lesions in men are benign. Gynecomastia
and breast cancer are the two most important diseases of

Case report

A 28-year-old man was admitted to our clinic complaining for a painful progressive swelling of the right breast.
He denied fever, chills, night sweats, weight loss, and
any other systemic symptoms. He had no past medical or

Correspondence: Dott. Carmen Cantisani, MD. Department of Oncoematology-Dermatology and Plastic Surgery. Azienda Policlinico Umberto
I, University Sapienza, Viale del Policlinico 155, 00161 Rome, Italy. Alternative address: Viale Regina Margherita 244, 00198 Rome, Italy.
Tel.:+39.347.9385719; Fax: +39.06.490243. E-mail: carmencantisanister@gmail.com or carmen.cantisani@uniroma1.it
Copyright Societ Editrice Universo (SEU)

ISSN 1972-600

e184

C. Cantisani

Fig. 1a-b. Painful right-sided gynecomastia with nipple discharge.

surgical history, including pulmonary or extrapulmonary


tuberculosis. There was no history of smoking, alcohol, or
drug abuse. The general physical examination showed rightsided gynecomastia with firm, tender palpable glandular
tissue (Fig. 1).
The testicular examination was normal, and secondary
sexual characteristics were well established. Chest auscultation revealed coarse crackles in the right infraclavicular area.
The rest of the physical examination results were unremarkable. Laboratory examination results showed erythrocyte
sedimentation rate, 58 mm/h, while complete blood count,
liver and renal function tests, and urine microscopy were
normal. The outcome of biochemical tests and urine analysis was normal. Chest X-ray was normal. Tuberculin test
(5 Todd unit of purified protein derivative) was performed,
and 16 mm of induration was measured.
Breast ultrasonography revealed a cystic mass in the
retro-mammary region. Helical computed tomography (CT)
of the thorax showed a 6x2 cm cystic soft tissue mass on
the anterior chest wall. The ribs, pleura, and lung fields all
appeared normal. Fine-needle aspiration was performed and
drained 20 mL of purulent material. Acid-fast bacilli were
observed by ZiehlNeelsen staining.
Signs of malignancy were not present in the cytology
examination of the purulent material. Mycobacterium
tuberculosis complex colonies were isolated from the
Lowenstein Jensen culture confirmed the diagnosis of
tuberculous mastitis.
We prescribed isoniazid (300 mg/day), rifampin (600
mg/day), morphazinamide (2.5 g/d), and ethambutol (1.5
g/day) for 5 months and with complete clinical resolution
of the breast lesion by the end of the treatment.
Discussion

There has been a significant rise in the prevalence of


tuberculosis as well as an increase in its extra-pulmonary
manifestations in the past decade. Migration, drug-resistant
strains, HIV infection, chronic diseases, malignancy, tran-

splantation, and other immunosuppressive conditions have


contributed to this process. However, breast tuberculosis is
a rare form of tuberculosis (10). The incidence in western
countries varies from 0.025% to 0.1% of all surgically treated
breast disease, and 3% to 4.5% in developing countries (11).
Breast tuberculosis commonly affects women in the reproductive age group. It is uncommon in prepubescent females
and elderly women (12) and it is extremely rare in males.
Tuberculous mastitis (TM) is found mostly in young,
multiparous women. Male TM is extremely rare, and
accounts for only 4% of all cases (7). To our knowledge,
only a few cases of tuberculous mastitis in men have been
reported in the English literature since 1945 (8, 9). Data are
scant on the total number of reported cases in men, its rarity
with respect to that of females, presentation and outcomes
(8). This strikingly lower incidence in males points towards
a significant role of parity, pregnancy and lactation as likely
predisposing factors.
Differential diagnosis most often includes carcinoma of
the breast. Some literature data suggests that non inflammatory breast cancer incidence is increased within a year
following episodes of non puerperal mastitis 13. Thus, even
if tubercolousis disease of the breast is identified, adequate
tissue specimens must be examined to exclude cancer (14).
Less common diseases are traumatic fat necrosis, plasma
cell mastitis, chronic pyogenic abscess, mammary dysplasia, fibroadenomas, granulomatous mastitis, sarcoid, and
actinomycosis.
Although controversial, tuberculous mastitis can be
classified as primary or secondary disease (15, 16). Primary
involvement is tuberculosis infection confined only to the
breast and is extremely uncommon. This infection is acquired through skin abrasions or duct openings from tonsillar
tuberculosis of infants. Secondary tuberculous mastitis is
more common and occurs by retrograde lymphatic route
from axillary nodes, from the lungs via inter-mammary
nodes, or hematogenous
spread. Associated axillary lymphadenopathy may be
found in 41% of cases (9). Although frequently the breast
appears to be the only organ clinically involved, this category

e185

Male tuberculous mastitis


should probably be reserved for those rare cases of direct
inoculation of the breast by tuberculous bacilli. The breast,
skeletal muscle, and spleen are regarded as extremely resistant to tubercular infection. Our case had no pulmonary
tuberculosis and axillary lymphadenopathy. Fine-needle
aspiration of the right breast revealed caseating necrosis and
granulomatous inflammation with acid-fast bacilli.
Tuberculous mastitis generally presents in one of three
ways: painless breast mass, breast edema, or localized abscess. As many as 75 percent of patients present with the
insidious onset of a painless breast mass, with or without
axillary involvement. Less commonly, edema of the breast,
usually with extensive involvement of the axillary nodes,
may occur. Breast abscess with or without sinus tract drainage is the least common presentation. Presentation is typically
unilateral, and although the upper outer quadrant seems to be
the most frequently involved site, probably due to proximity
of the axillary nodes, any area of the breast can be involved.
Patients are otherwise generally healthy, have few or no constitutional symptoms, and usually have a positive tuberculin
skin test. Diagnosis of this disease is difficult. Clinical and
radiologic findings are often nonspecific. The accuracy of
mammography, fine-needle aspiration cytology, and excision biopsy for diagnosis of breast tuberculosis are 14%,
12%, and 60%, respectively (12-17). The overall acid-fast
bacillus positivity is 22.7% in fine-needle aspiration material (18). However, mycobacterial culture remains the gold
standard for diagnosis of tuberculosis. Moreover, culture is
not always helpful in the diagnosis of breast tuberculosis.
Polymerase chain reaction (PCR) might improve sensitivity
in some cases. Due to its rarity, no specific guidelines are
available for the treatment of tuberculous mastitis. There
is little information in the literature regarding optimum
length of therapy, but tuberculous mastitis should probably
be treated as any other form of extra-pulmonary tuberculosis, which is generally nine months of multi-drug therapy,
unless drug resistance is present. Surgical interventions are
performed only in severe joint deformation after adequate
anti-tubercular treatment. Our patient was treated successfully with isoniazid (300 mg/day), rifampin (600 mg/day),
morphazinamide (2.5 g/d), and ethambutol (1.5 g/day) for 5
months and with complete clinical resolution of the breast
lesion by the end of the treatment.
Conclusion

In summary, tuberculosis mastitis should be considered


in the differential diagnoses of a breast mass, even in male
patients. The diagnosis of breast tuberculosis in clinical
presentations remains a true challenge. It can be treated with
antitubercular drugs, while surgery is used only in cases that
fail to respond to medical treatment.

Acknowledgments

We would like to thank Associazione Romana Dermatologica.


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