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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 2  |  Issue : 3  |  Page : 212-215

Aspergillosis of bilateral breast and chest wall in an immunocompetent male masquerading as breast cancer


Department of Pathology, Pandit Deendayal Upadhyay Government Medical College and Hospital, Rajkot, Gujarat, India

Date of Web Publication25-Oct-2013

Correspondence Address:
Jitendra G Nasit
C/4, Suryadeep Society, Near Nutan School, Behiend Chankyapuri Society, New Sama Road, Vadodara - 390 008, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-344X.120596

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  Abstract 

Fungal species are not frequently encountered in an immunocompetent host. Invasive aspergillosis typically occurs in severely immunocompromised patient. Aspergillus infection of breast and chest wall are rarely encountered in an immunocompetent as well as in immunocompromised host. Till date only 13 cases of fungal infection of breast and chest wall have been reported in the literature. This report presents a case of aspergillosis of bilateral breast and chest wall in an immunocompetent male, clinically mimicking breast cancer. Diagnosis was achieved by fine-needle aspiration cytology and subsequently Aspergillus flavus was identified on fungal culture. The patient was successfully treated with voriconazole. Prompt diagnosis by cytology and appropriate treatment is necessary to prevent adverse outcome. Here, we present this rare case of fungal infection of breast and chest wall with relevant review of the literature.

Keywords: Aspergillosis, Aspergillus flavus, breast, chest wall, immunocompetent


How to cite this article:
Nasit JG, Sojitra N, Bhalra R, Dhruva G. Aspergillosis of bilateral breast and chest wall in an immunocompetent male masquerading as breast cancer . Int J Health Allied Sci 2013;2:212-5

How to cite this URL:
Nasit JG, Sojitra N, Bhalra R, Dhruva G. Aspergillosis of bilateral breast and chest wall in an immunocompetent male masquerading as breast cancer . Int J Health Allied Sci [serial online] 2013 [cited 2019 Sep 21];2:212-5. Available from: http://www.ijhas.in/text.asp?2013/2/3/212/120596


  Introduction Top


Aspergillus species usually affect an immunocompromised host. [1],[2] They are rarely encountered in an immunocompetent host. They usually affect pulmonary system. [1],[3] Extrapulmonary manifestations are rare. [2] Aspergillus infection of breast and chest wall are very rare. [1],[4] We present an extremely rare case of aspergillosis of bilateral breast and chest wall, clinically mimicking breast cancer, in an immunocompetent male.


  Case Report Top


A 43-year-old male farmer presented with history of creeping sensation and gradually enlarging painless bilateral breast swelling and chest wall stiffness since 18 months. He complained of serous discharge from ulcerated skin over right breast. Past medical history was unremarkable. He was treated with anti-bacterial medication frequently without any definitive diagnosis. There was no history of smoking, alcohol, intravenous drug abuse, steroid intake, asthma, diabetes mellitus, or related immune deficiency. The patient had a history of open wound due to trauma in vegetable farm (fall in area where water was stagnant) over a local site before 2 years. In addition, overall hygiene of the patient was poor. Physical examination revealed a firm to hard mass measuring 7.0 Χ 6.5 cm and 6.5 Χ 6.0 cm in right and left breasts, respectively. The skin over the right breast revealed 1.2 Χ 0.9 cm healed ulcer [Figure 1]. Bilateral breast masses were fixed against underlying muscle tissue and overlying skin. Chest wall was wooden hard on touch. Peripheral lymphadenopathy was not evident. The lesion was clinically suspected to be a breast carcinoma. Hematology and biochemical parameters were normal.

Fine-needle aspiration cytology (FNAC) was done from both breasts after informed written consent. Hematoxylin-eosin stained smears showed scattered multiple septate acute branching hyphae with florid granulomatous inflammation in a necrotic and inflammatory background [Figure 2]. Cytological diagnosis of aspergillus-induced granulomatous lesion of bilateral breast and chest wall was made. However, our cytology report was not correlated with clinical diagnosis of suspected breast carcinoma. So, to reconfirm the findings and to rule out occult malignancy, ultrasound-guided tru-cut biopsy was taken. Biopsy yielded thick viscous necrotic material. Histology revealed aspergillus fungal hyphae in necrotic debris on hematoxylin-eosin, Gomori's methenamine silver, and periodic acid Schiff stains [Figure 3]. Based on these findings, a diagnosis of primary aspergillosis of bilateral breast and chest wall in an immunocompetent male was made. Aspergillus flavus was isolated after 2 weeks on fungal culture. Serum immunoglobulin E (Ig E) level was mildly elevated. Sputum examination for fungus was negative. Acid fast bacilli were not seen on Ziehl-Neelsen stain. Culture for tuberculous organism was negative. Retrospectively we investigated for any immunocompromised state through complete blood count, human immunodeficiency virus (HIV) status including CD4 and CD8 count, hepatitis B surface antigen (HBsAg), serum immunoglobulin (Ig G, Ig M and Ig A) levels, nitroblue tetrazolium dye test for chronic granulomatous disease. But none could be found positive in our case. Biochemical parameters like total protein, sugar, urea, and creatinine were unremarkable. Chest roentgenogram was unremarkable for any pulmonary lesion including ribcage infection. Ultrasound of the abdomen was negative for any organomegaly or lymphadenopathy.
Figure 1: Aspergillosis of bilateral breast and chest wall: Clinical photograph shows enlargement of right and left breast which measure 7.0 × 6.5 cm and 6.5 × 6.0 cm in size, respectively. The skin over right breast reveals 1.2 × 0.9 cm ulcer

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Figure 2: Primary aspergillosis of bilateral breast and chest wall, fine needle aspiration cytology. (a-c) Cytology smears show scattered multiple septate acute branching hyphae suggest aspergillus fungal hyphae (H and E stain, ×40); (d) granulomatous inflammation with necrotic and inflammatory background (H and E stain, ×40)

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Figure 3: Primary aspergillosis of bilateral breast and chest wall, Tru-cut biopsy: Aspergillus fungal hyphae are identified by (a) H and E stain, (×40); (b) Gomori's methenamine silver stain (×40); and (c) periodic acid Schiff stain (×40)

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The patient was treated with amphotericin B (2 g) for 2 months but no clinical improvement was seen. Then voriconazole (300 mg) twice daily for 2 months followed by 200 mg twice daily for 4 months was given. There was marked clinical improvement (reduction of breast swelling and both chest and breast regions become little softer than previously wooden hard feel) on follow-up at 6 months.




  Discussion Top


Fungal infections of breast and chest wall are uncommon. [1],[4],[5] Only 13 cases of fungal infection of the breast and chest wall are reported in the literature till date. [1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11] One case of fungal infection of chest wall was described in a patient of poliomyelitis with lung aspergillosis. [1] Each single case of fungal infection of the breast was described in patients with acute myeloid leukemia; [2] diabetes mellitus; [4] postoperative status for breast carcinoma; [6] and lung transplantation. [6] Three cases were found to be associated with breast implant surgery. [7],[8],[9] Only five cases of fungal infection of breast and chest wall were reported in an immunocompetent host. [1],[3],[4],[11] To the best of our knowledge, this is the sixth case of aspergillosis of bilateral breast and chest wall in an immunocompetent patient. Review of such cases revealed that fungal infection of male breast is uncommon. All cases were diagnosed through FNAC and/or biopsy as in our case. Although all cases are immunocompetent, underlying breast malignancy, surgical procedure and chemotherapy, muscular disease like poliomyelitis, fungal infection elsewhere in the body may be the predisposing factor for fungal infection at unusual site like breast and chest region. [1],[3],[4],[11] In contrast, poor hygiene and traumatic wound over breast and chest region may be the most likely cause for fungal infection in our case.

Aspergillus species are the most ubiquitous fungi seen in soil, water, indoor air, and decaying vegetations. [1],[12] Most commonly they affect the lung and paranasal sinus, less frequently extrapulmonary sites such as brain, heart, skin, kidney and gastrointestinal tract. [7] Sometimes, they may be disseminated. [7] Cutaneous aspergillosis is a rare form of locally invasive disease, usually caused by A. fumigatus and A. flavus. [11] They may be primary, involving the site of skin injury following intravenous cannulation, trauma, occlusive dressing, burns, or surgery. Secondary skin lesions may result from widespread hematogenous seeding. [12] They may present as macules, papules, plaques or hemorrhagic bullae, pustules, subcutaneous abscess which may progress into necrotic ulcers that are covered by a heavy black eschar. [12] Aspergillus infection leads to hyphal growth, invasion of the blood vessels, hemorrhagic necrosis, infarction, and potential dissemination to any other organ in susceptible patient. [6] The major risk factors for invasive aspergillosis are prolonged neutropenia (>3 weeks) or neutrophil dysfunction (chronic granulomatous disease), corticosteroid therapy (especially prolonged high-dose therapy), transplantation (highest risk is with lung and bone marrow), hematologic malignancy (risk is higher with leukemia), cytotoxic therapy, and AIDS (risk increases with lower CD4 count). [1] However, on extensive work up, underlying immune defect was not found in our case. The presence of a foreign body such as an implant may lower the threshold for infection. [8]

The usual presentation of fungal infection of breast is firm, painless, non-inflamed breast lump which may be easily mistaken for breast carcinoma like in our case. [10] In our case, wooden like firm to hard feel of breast and chest region raises the possibility of connective tissue disease especially systemic sclerosis on clinical ground. However, in the absence of any other systemic compliant or abnormalities, possibility of systemic sclerosis is very unlikely. Further on cytology, granulomatous inflammation raises the possibility of tuberculous inflammation. But acid fast bacilli are not seen on the Ziehl-Neelsen stain. Also, culture for tuberculous organism is negative. Radiology findings are non-specific in our case. The diagnosis of aspergillus infection is usually made by demonstrating the septate, acute branching hyphae in the breast tissue sample by FNAC, and biopsy like in our case. [1],[10],[11] Although the patient had preceding history of trauma in vegetable farm, we speculate that Aspergillus was probably inoculated with an injury. We believe that the lesion was primary invasive aspergillosis because needle aspirates from the mass forming breast lesion contain aspergillus hyphae with marked granulomatous inflammation. Tru-cut biopsy also showed aspergillus fungal hyphae in necrotic material. These findings were supported by positive fungal culture and successful antifungal therapy. The protracted course of 2 years duration could be attributed to the irregular short-term treatment with anti-bacterial medication without definitive diagnosis of fungal infection. So, prompt diagnosis by cytology and appropriate treatment is necessary to prevent adverse outcome.

Recommended treatment for primary cutaneous aspergillosis is voriconazole, itraconazole, and amphotericin B. However, recent studies suggest that a significant portion of Aspergillus species could be resistant to amphotericin B. [12] Voriconazole has been shown to be statistically superior to amphotericin B. [12] The patient was treated with voriconazole and it was very effective and well tolerated. Aspergilloma of breast can be treated by surgical excision. [2] However, in view of extensive tissue involvement in our case, debridement was not done, as it would led to larger defect.


  Conclusion Top


Aspergillus can present with extensive breast and chest wall involvement even in an immunocompetent patient; therefore, the possibility of fungal infection should not be ignored in any slow growing long-standing breast lump in an immunocompetent status. We wish to highlight the importance of FNAC in case of fungal infection of breast when clinical and radiology findings are non-specific. Immune deficiency should be investigated in all patients with fungal infection. Prolonged anti-bacterial medications without definitive diagnosis of fungal infection lead to adverse outcome. In amphotericin B refractory case, voriconazole is a new antifungal agent found to be effective in invasive aspergillosis.

 
  References Top

1.Kumar J, Seith A, Kumar A, Madan K, Guleria R. Chest wall and mediastinal nodal aspergillosis in an immunocompetent host. Diagn Interv Radiol 2009;15:176-8.  Back to cited text no. 1
    
2.Klosta SP, Ludwig K, Kreuter M, Diallo R, Heindel W. Aspergilloma of the breast in a woman with acute myeloid leukemia. Am J Roentgenol 2002;178:1236-8.  Back to cited text no. 2
    
3.Jordan JM, Waters K, Caldwell DS. Aspergillus flavus: An unusual cause of chest wall inflammation in an immunocompetent host. J Rheumatol 1986;13:660-2.  Back to cited text no. 3
    
4.Sukhamwang N, Chaiwun B, Trakultivakorn H. Fungal infection of the breast: Two reported cases diagnosed by fine needle aspiration (FNA) cytology. Chiang Mai Med J 2009;48:65-70.  Back to cited text no. 4
    
5.Thapar VK, Deshpande A, Jain VK, Bhowate P, Madiwale C. Isolated breast mucormycosis. J Postgrad Med 2006;52:134-5.  Back to cited text no. 5
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6.Risha A, André R, Smeets A, Christiaens MR. Aspergilloma of the breast in a lung transplantation patient: A case report. Acta Chir Belg 2006;106:730-1.  Back to cited text no. 6
    
7.Williams K, Walton RL, Bunkis J. Aspergillus colonization associated with bilateral silicone mammary implants. Plas Reconstr Surg 1983;71:260-1.  Back to cited text no. 7
    
8.Tian HH, Tan SM, Tay KH. Delayed fungal infection following augmentation mammoplasty in an immunocompetent host. Singapore Med J 2007;48:256-8.  Back to cited text no. 8
    
9.Rosenblatt WB, Pollock A. Aspergillus flavus cultured from a saline filled implant. Plast Reconstr Surg 1997,99:1470-2.  Back to cited text no. 9
    
10.Farmer C, Stanley MW, Bardales RH, Korourian S, Shah H, Bradsher R, et al. Mycoses of the breast: Diagnosis by fine-needle aspiration. Diagn Cytopathol 1995;12:51-5.  Back to cited text no. 10
    
11.Govindarajan M, Verghese S, Kuruvilla S. Primary aspergillosis of the breast. Report of a case with fine needle aspiration cytology diagnosis. Acta Cytol 1993;37:234-6.  Back to cited text no. 11
    
12.Nakashima K, Yamada N, Yoshida Y, Yamamoto O. Primary Cutaneous Aspergillosis. Acta Derm Venereol 2010;90:519-20.  Back to cited text no. 12
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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