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CASE REPORT
Year : 2015  |  Volume : 32  |  Issue : 1  |  Page : 60-63  

Away from the milky way: An extremely rare case of pulmonary ectopic breast


1 Department of Surgery, Urmia University of Medical Sciences, Urmia, Iran
2 Department of Interventional Cardiology, University of Arizona Medical Center, Tucson, Arizona, United States
3 National Institute of Health Research, Tehran University of Medical Sciences, Tehran, Iran

Date of Web Publication2-Jan-2015

Correspondence Address:
Dr. Nariman Sepehrvand
National Institute of Health Research, Tehran University of Medical Sciences, No. 78, Italia Street, Keshavarz Boulevard, Tehran
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-2113.148454

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   Abstract 

Ectopic breast is defined as the presence of breast tissue outside the pectoral region. In this article, we are reporting an extremely rare case of pulmonary aberrant breast tissue. A 79-years-old Caucasian woman with the history of hypertension, diabetes mellitus and ischemic heart disease, presented to the emergency department for worsening dyspnea on exertion and pleuritic chest pain over the last few days. Both thoracic CT scan and the gross evaluation of tumor after its removal by thoracotomy were in favor of a soft tissue tumor such as pulmonary lipoma, whilst surprisingly the histological examinations revealed the mass to be ectopic breast tissue. Although it is extremely rare, this diagnosis should be considered in the evaluation of pulmonary masses.

Keywords: Ectopic breast, intra-thoracic, mass, pulmonary


How to cite this article:
Mahmodlou R, Sugumaran RK, Sepehrvand N. Away from the milky way: An extremely rare case of pulmonary ectopic breast. Lung India 2015;32:60-3

How to cite this URL:
Mahmodlou R, Sugumaran RK, Sepehrvand N. Away from the milky way: An extremely rare case of pulmonary ectopic breast. Lung India [serial online] 2015 [cited 2019 Sep 20];32:60-3. Available from: http://www.lungindia.com/text.asp?2015/32/1/60/148454


   Introduction Top


During the fifth week of gestation, ectodermal thickening is differentiated to form milk-line or mammary ridges on the ventral side, which are extended bilaterally from the axilla to the groin. [1] Normally, these ridges persist in the pectoral region allowing for the development of breast tissue and regress in non-pectoral areas. Any disruption in the embryonic regression of these ridges may result in the formation of ectopic breasts through this line. [2],[3],[4],[5],[6]

According to the prevalence rate of ectopic breast tissue among the general population (0.2-6%), [1],[7],[8],[9],[10],[11] this phenomenon may not be uncommon. However, the prevalence of ectopic breast (supernumerary and/or aberrant) outside the mammary ridges is exceptionally uncommon. The face, chest wall, back, buttocks, and thighs are just some of the various places where ectopic breast tissue (EBT) have been found. [8],[12],[13],[14],[15],[16],[17] To date, there are only three cases of EBT documented in internal organs. [17],[18],[19] We report an extremely rare case of pulmonary aberrant breast tissue.


   Case Report Top


A 79-year-old Caucasian female with a medical history significant for ischemic heart disease, hypertension, and diabetes mellitus, was found to have a pulmonary mass visualized on high resolution computed tomography (HRCT) of the chest in 2002 during a workup for dyspnea. Based on the HRCT, the mass appeared cystic in nature but the patient was lost to follow-up over the last ten years.

Recently, she presented to the emergency department for worsening dyspnea on exertion and pleuritic chest pain over the last few days. Initial chest radiography revealed a large cystic mass in her left pleural space [Figure 1].
Figure 1: Chest radiography of the patient before thoracotomy

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On physical examination, vital signs were within normal limits and pertinent findings during pulmonary auscultation revealed decreased breath sounds on the left side along with left-sided dullness with percussion. The results of the initial laboratory tests at this time, which included complete blood count (CBC), chemistry panel, and liver function, were normal. A Thoracic CT scan was requested by the surgical team and it described the mass as a well-defined concentric thick wall cavity in the left upper lobe with smooth inner surface without any calcification and with a region of interest (ROI) of -124 HU, suggesting a pulmonary lipoma [Figure 2].
Figure 2: Chest CT before surgery (note the large mass in the left lung)

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Due to size of the probable tumor and the patient's symptoms, the surgical team recommended a thoracotomy under general anesthesia in the right lateral decubitus position. The left lower lobe of the lung was collapsed due to the large size of the tumor and its compression effect. After pneumolysis, the mass was observed to occupy the entire left upper lobe. Upon visualization of numerous, enlarged lymph nodes in the mediastinum, lobectomy of the left upper lobe, lymphadenectomy, and contrapleural costal resection were performed.

The solitary, encapsulated mass was surgically removed. Grossly, the mass appeared to have features of a benign soft tissue tumor (i.e. lipoma) measuring 240 × 130 × 100 mm [Figure 3].
Figure 3: Gross appearance of the pulmonary mass

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Histological examinations revealed papillary and tubular structures, similar to breast tissue. No mitosis, necrosis, or cells with atypical features were detected via histologic evaluation, suggesting the benign nature of the tumor [Figure 4].
Figure 4: Histopathology of pulmonary ectopic breast (H and E, ×100)

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Formalin-fixed, paraffin-embedded sections of the sample were stained for estrogen receptor (ER) and progesterone receptor (PR) using immunohistochemistry (IHC). For IHC investigation, we utilized FLEX monoclonal mouse anti-human antibodies: 1D5 at dilution 1:60 for ER, and PgR636 at dilution of 1:300 for PR from Dako-Denmark (A/S production svej 42 DK-2600 Glostrup, Denmark) according to the manufacturer's protocol. All slides which were stained on an automated system were negative for both ER and PR.

Post-operatively, the patient was monitored for six days and was discharged to home without any surgical or hospital complications. During a 6-month follow-up appointment, the patient denied any symptoms of shortness of breath or dyspnea on exertion. Re-expansion of the left lower lobe significantly improved the patient's condition, and she is currently undergoing regular medical follow-up.


   Discussion Top


EBT is residual tissue abnormally persisting during the embryological development against the gradual regression of mammary ridges in non-pectoral regions. [7] The axilla is the most frequently involved location for EBT (70%). The second most frequent place is the vulva. EBT may be accompanied with symptoms like discomfort or pain during pregnancy, lactation, or in the pre-menopausal period. [5],[20]

According to de-Cholnocky, approximatly 4% of EBT cases may develop into a primary ectopic breast carcinoma. [21] Therefore, the conversion of EBT to malignancy should be considered in all diagnosed cases.

Yeniay et al., reported a 71-year-old female with an abdominal aberrant breast tissue found incidentally in a piece of mesenteric biopsy during operation for a recurrent endometrial malignant tumor. [17] Another case of developed heterotropic breast tissue found in internal organs was reported by Sasaki et al. They reported a heterotopic breast epithelial inclusion of the heart incidentally found on a native heart of a 73-year-old man who received an orthotopic heart transplantation for ischemic cardiomyopathy. [18]

Sundaram in 1969 reported several pseudo-tuberculosis conditions of the lung including silicosis, flurosis, etc. One of the reported cases was of a 20-year-old woman with throat irritation and chronic cough, bilateral axillary lymph nodes, and streaky shadows on chest x-ray localized to the left upper lobe. She was treated as a case of smear-negative TB laryngitis with anti-TB therapies that resulted in no appreciable changes. The histopathologic examination of the removed axillary glands yielded the diagnosis of mature lobule of breast. Although the author proposed the diagnosis of accessory breast for the pulmonary findings in chest x-ray, no sampling was performed from the pulmonary inclusions in that case.

Our case is the only well-documented case of pulmonary ectopic breast worldwide. Although the results of ER and PR antibody studies did not add any confirmation to the diagnosis of ectopic breast, the obvious resemblance of the hisopathologic pattern of tumor to the breast tissue rejected any other possibility.

Unfortunately the concordance of ER and PR expression with the existence of breast tissue in samples was not investigated before. There are some studies around the concordance of ER and PR expression and existence of ectopic breast carcinoma. [22],[23] In the study of Shabaik et al., the results showed a sensitivity of 85.7% and specificity of 100% for ER, and a sensitivity of 80% and specificity of 100% for PR. [23] Three cases (16.6%) among the 18 different articles which investigated the ER, PR antibodies in ectopic breast cancer cases, were reported to have negative ER and PR results. [6],[20],[24]

As mentioned above, our patient did not recall any symptoms during pregnancy, lactation, or in the pre-menopausal period. This could be interpreted by the negative ER and PR status of the ectopic breast tissue. This infers that many of the cells may not contain receptors that are influenced by the alterations in hormone levels.

Generally despite the rare nature of pulmonary ectopic breast tissue, the clinicians should take into consideration this disease in evaluating the differential diagnoses of pulmonary masses.


   Acknowledgment Top


The authors would like to thank Dr. Behrooz Ilkhanizadeh and Dr. Afshin Mohammadi from Urmia University of medical Sciences for their contribution in providing the manuscript.

 
   References Top

1.
Visconti G, Eltahir Y, Van Ginkel RJ, Bart J, Werker PM. Approach and management of primary ectopic breast carcinoma in the axilla: Where are we? A comprehensive historical literature review. J Plast Reconstr Aesthet Surg 2011;64:e1-11.  Back to cited text no. 1
    
2.
Caceres M, Shih J, Eckert M, Gardner R. Metaplastic carcinoma in an ectopic breast. South Med J 2002;95:462-6.  Back to cited text no. 2
    
3.
Goyal S, Puri T, Gupta R, Julka PK, Rath GK. Accessory breast tissue in axilla masquerading as breast cancer recurrence. J Cancer Res Ther 2008;4:95-6.  Back to cited text no. 3
    
4.
Lopes G, DeCesare T, Ghurani G, Vincek V, Jorda M, Glück S, et al. Primary ectopic breast cancer presenting as a vulvar mass. Clin Breast Cancer 2006;7:278-9.  Back to cited text no. 4
    
5.
Shamim M, Baddar N. Axillary ectopic carcinoma of breast. J Pak Med Assoc 2011;61:916-8.  Back to cited text no. 5
    
6.
Shin SJ, Sheikh FS, Allenby PA, Rosen PP. Invasive secretory (juvenile) carcinoma arising in ectopic breast tissue of the axilla. Arch Pathol Lab Med 2001;125:1372-4.  Back to cited text no. 6
    
7.
Dordević M, Jovanović B, Mitrović S, Dordević G. Ectopic mammary tissue in vulva. Vojnosanit Pregl 2008;65:407-9.  Back to cited text no. 7
    
8.
Grossl NA. Supernumerary breast tissue: Historical perspectives and clinical features. South Med J 2000;93:29-32.  Back to cited text no. 8
    
9.
Kawahara F, Noguchi M, Yamamichi N, Konishi F, Yagasaki R, Minami M, et al. Ectopic Breast Caner: Two case reports and review of the japanese literature. Breast Cancer 1997;4:43-8.  Back to cited text no. 9
    
10.
Routiot T, Marchal C, Verhaeghe JL, Depardieu C, Netter E, Weber B, et al. Breast carcinoma located in ectopic breast tissue: A case report and review of the literature. Oncol Rep 1998;5:413-7.  Back to cited text no. 10
    
11.
Tjalma WA, Senten LL. The management of ectopic breast cancer--case report. Eur J Gynaecol Oncol 2006;27:414-6.  Back to cited text no. 11
    
12.
Conde DM, Kashimoto E, Torresan RZ, Alvarenga M. Pseudomamma on the foot: An unusual presentation of supernumerary breast tissue. Dermatol Online J 2006;12:7.  Back to cited text no. 12
    
13.
Deaver JB, McFarland J. The breast: Its anomalies, its diseases, and their treatment. Philadelphia: P. Blakiston's Sons; 1917: 79-80.  Back to cited text no. 13
    
14.
Silverberg MA, Rahman MZ. Axillary breast tissue mistaken for suppurative hidradenitis: An avoidable error. J Emerg Med 2003;25:51-5.  Back to cited text no. 14
    
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Sultan C. Pediatric and adolescent gynecology. Evidence-based clinical practice. Preface. Endocr Dev 2004;7:9-10.  Back to cited text no. 15
    
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Yaghoobi R, Bagherani N, Mohammadpour F. Bilateral aberrant axillary breast tissue. Indian J Dermatol Venereol Leprol 2009;75:639.  Back to cited text no. 16
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17.
Yeniay L, Mulailwa K, Asgerov E, Hoşcoşkun C, Zekioğlu O. Aberrant breast in a rare site: A case report. Case Rep Med 2012;2012:943102.  Back to cited text no. 17
    
18.
Sasaki K, Parwani AV, Demetris AJ, Sasatomi E. Heterotopic breast epithelial inclusion of the heart: Report of a case. Am J Surg Pathol 2010;34:1555-9.  Back to cited text no. 18
    
19.
Sundaram V. Pseudotuberculosis conditions of lungs. Indian J Tub 1969;16:9-13.  Back to cited text no. 19
    
20.
Cheong JH, Lee BC, Lee KS. Carcinoma of the axillary breast. Yonsei Med J 1999;40:290-3.  Back to cited text no. 20
    
21.
de Cholnoky T. Supernumerary breast. Arch Surg 1939;39:926-41.  Back to cited text no. 21
    
22.
Intra M, Maggioni A, Sonzogni A, DE Cicco C, Machado LS, Sagona A, et al. A rare association of synchronous intraductal carcinoma of the breast and invasive carcinoma of ectopic breast tissue of the vulva: Case report and literature review. Int J Gynecol Cancer 2006;16 Suppl 1:428-33.  Back to cited text no. 22
    
23.
Shabaik A, Lin G, Peterson M, Hasteh F, Tipps A, Datnow B, et al. Reliability of Her2/neu, estrogen receptor, and progesterone receptor testing by immunohistochemistry on cell block of FNA and serous effusions from patients with primary and metastatic breast carcinoma. Diagn Cytopathol 2011;39:328-32.  Back to cited text no. 23
    
24.
Nakao A, Saito S, Inoue F, Notohara K, Tanaka N. Ectopic breast cancer: A case report and review of the Japanese literature. Anticancer Res 1998;18:3737-40.  Back to cited text no. 24
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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