e183Male tuberculous mastitis

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

1Department of Dermatology, Sapienza University, Rome, Italy, 2Department of Dermatology and Skin Surgery, Roger Williams Medical Center/Boston University, Providence, Rhode Island, USA

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

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

Introduction

Benign breast disorders, classified by the ANDI system (aberrations of normal development and involution), consti-tute 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

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 gra-nulomas, 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 connec-tion 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 relative-ly 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 gram-negative 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 impor-tant to receive treatment immediately to prevent complica-tions, 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.

Case report

A 28-year-old man was admitted to our clinic complai-ning 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

e184 C. Cantisani

surgical history, including pulmonary or extrapulmonary tuberculosis. There was no history of smoking, alcohol, or drug abuse. The general physical examination showed right-sided gynecomastia with firm, tender palpable glandular tissue (Fig. 1).

The testicular examination was normal, and secondary sexual characteristics were well established. Chest ausculta-tion revealed coarse crackles in the right infraclavicular area. The rest of the physical examination results were unremar-kable. 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 analy-sis 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 repro-ductive 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 inflam-matory 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 dyspla-sia, 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 acqui-red 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

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

e185Male 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 re-sistant 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 ab-scess. 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 draina-ge 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 con-stitutional 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 exci-sion 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 mate-rial (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 tuberculo-sis, 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 succes-sfully 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 Derma-tologica.

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