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CASE REPORT
Ahead of print publication  

Eosinophilic mastitis: A chameleon disease in rheumatologists' domain


1 Division of Rheumatology and Clinical Immunology, Medanta - The Medicity, Gurugram, Haryana, India
2 Division of Breast Services, Medanta - The Medicity, Gurugram, Haryana, India

Correspondence Address:
Shruti Bajad,
Room No. 14, 5th Floor, Medanta - The Medicity, Gurugram, Haryana
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/injr.injr_39_19

  Abstract 


We report a rare case of eosinophilic mastitis in a young asthmatic female who presented with a breast lump and was managed conservatively with a successful outcome. Eosinophilic mastitis, characterized by eosinophilic infiltration of mammary glands, is one such entity which is frequently given a miss, and is diagnosed based on histopathological analysis. The identification of this disease is a primary step in the management of such benign disorders which can be successfully treated conservatively avoiding any surgical intervention and morbidities thereafter. Because presentation can be similar and confusing enough, we took a cue from the history of asthma, urticaria, and peripheral eosinophilia to clinch the diagnosis of eosinophilic mastitis, which was further confirmed on biopsy. To the best of our knowledge, this happens to be the youngest female patient aged 27 years who also had urticaria along with other eosinophilic manifestations, which is unreported in other cases and makes our case exceptional. She was treated with glucocorticoids and antihistaminics and is presently symptom free.

Keywords: Asthma, breast carcinoma, eosinophilic mastitis, young female



How to cite this URL:
Bajad S, Bajaj R, Tanna D, Bindroo M, Kaur K, Gupta R. Eosinophilic mastitis: A chameleon disease in rheumatologists' domain. Indian J Rheumatol [Epub ahead of print] [cited 2019 Sep 22]. Available from: http://www.indianjrheumatol.com/preprintarticle.asp?id=265809




  Introduction Top


Eosinophilic infiltration of the mammary gland, an extremely rare phenomenon, is known as eosinophilic mastitis. Sporadic cases have been reported worldwide regarding this entity. The importance of identifying this disease lies in the fact that early diagnosis and its knowledge can save the patient from unnecessary surgical intervention and its complications. Here, we discuss a very rare case of a young asthmatic female with a history of urticarial rashes who presented with a breast lump and eventually diagnosed with eosinophilic mastitis and treated successfully.


  Case Report Top


We report the case of a 27-year-old female from North India. She was a known case of childhood-onset bronchial asthma for the last 20 years. However, she was not on any medications for the same. She presented to our rheumatology outpatient department with a history of pain in the right breast associated with cheesy yellow discharge from the right nipple. The patient was nonsmoker, and her family history was unremarkable. There was a history of urticarial skin rashes on and off especially during the change of season. The rashes used to subside with intake of antihistaminics and were an important link to look for alternative etiologies rather than malignancy. There was no history of any numbness or tingling in the limbs or bladder–bowel complaints. There was no history of any drug intake including oral contraceptives. On physical examination, there was a small, tender mass in the subareolar region of the right breast. The right nipple showed cheesy yellow discharge. There were no palpable lymph nodes. Systemic examination was unremarkable.

Her routine laboratory investigations showed hemoglobin of 10.1 g/dl and total leukocyte count of 10,300/mm3 (with eosinophils of 16.5%). Absolute eosinophil count was 820/mm3. Antinuclear antibody, rheumatoid factor, and antineutrophil cytoplasmic antibody were negative. Urine examination was within normal limits with no evidence of proteinuria. Chest X-ray showed clear lung fields.

On ultrasonography (USG) examination, there were multiple thick-walled ducts seen in the lower half of the right breast extending from 6 to 8 o' clock position with associated surrounding architectural distortion and hyperreflexive surrounding parenchyma. Subsequently, USG-guided biopsy of the lesion done showed dense periductal and lobular mixed inflammatory cell infiltrates comprising neutrophils, lymphocytes, plasma cells, and sheets of eosinophils with no evidence of in situ or invasive malignancy. The biopsy findings were compatible with eosinophilic mastitis [Figure 1].
Figure 1: Acini surrounded by an inflammatory infiltrate – predominantly eosinophils, admixed with few polymorphs and lymphocytes. No atypia or malignancy was seen (H and E, ×400)

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The patient was thus started on oral steroids and antihistaminics. On repeat ultrasound done after 6 weeks, there was significant interval reduction in the previously noted ductal wall thickening with interval resolution of surrounding hyperreflective edematous changes [Figure 2]. There was complete clinical resolution observed within 6 weeks followed by which steroids were gradually tapered. The eosinophil counts also returned to normal condition.
Figure 2: At baseline and after 6 weeks of treatment. Multiple thick-walled ducts seen in the lower half of the right breast extending from 6 to 8 o' clock position with associated surrounding architectural distortion and Hyper reflective surrounding parenchyma

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  Discussion Top


Eosinophilia is usually defined as the presence of more than 500 eosinophils/mm3 in peripheral blood. Peripheral eosinophilia has been described in many conditions including parasitic infection, hypereosinophilic syndrome, eosinophilic granulomatosis, and polyangitis and allergic disorders. Focal eosinophilic infiltration which is usually accompanied by peripheral eosinophilia has been described in skin, lung, liver, and gastrointestinal tract; however, breast remains a very rare site for eosinophilic infiltration. Infiltration of the breast tissue with eosinophils describes eosinophilic mastitis.

Eosinophilic infiltration, irrespective of the cause, results in organ damage by releasing various cytotoxic granule proteins including eosinophil major basic protein and eosinophil cationic protein and other cytokines.[1] Interleukin-5 has been identified as a specific differentiation factor for eosinophils, but its role in causing tissue eosinophilia is still not described.[2] The exact pathogenesis of this entity still remains unclear though it can explain some local allergic reaction.[3] Very few such cases have been described in literature.

Our patient is a young asthmatic female who presented with a painful breast lump associated with nipple discharge. The age group in earlier cases of eosinophilic mastitis varied between 40 and 60 years; one case of a 30-year-old female was reported. Our patient is a 27-year-old female whose general health was good, and she was not on any medications. Her imaging findings were nonspecific. This patient had a history of urticaria which was not reported in earlier cases although a history of asthma and peripheral eosinophilia has been reported. The presence of urticaria suggested us regarding the presence of atopy. A review of previous cases has revealed that two patients suffered from Churg–Strauss syndrome, three patients had asthma, two had hypereosinophilic syndrome, one patient lacked both peripheral eosinophilia and systemic manifestations, one patient had only peripheral eosinophilia, and one patient reportedly had bilateral eosinophilic mastitis.[4],[5],[6],[7],[8],[9],[10],[11],[12] Then lies the importance of histopathologic examination which showed extensive infiltrate of eosinophils surrounding both mammary ducts and lobules mixed with lymphocytes and plasma cells, consistent with the diagnosis of eosinophilic mastitis. It was associated with peripheral eosinophilia though she showed no respiratory symptoms. Due to clinical and radiological resemblance with carcinoma breast, prompt identification of this condition is of utmost importance. Because this condition shows a dramatic response to steroids, the significance of this case lies in the fact that knowledge of this disease entity has led to its prompt identification, and we could save the patient from unnecessary surgical intervention and treat her successfully.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that her name and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rothenberg ME, Hogan SP. The eosinophil. Annu Rev Immunol 2006;24:147-74.  Back to cited text no. 1
    
2.
Valent P. Pathogenesis, classification, and therapy of eosinophilia and eosinophil disorders. Blood Rev 2009;23:157-65.  Back to cited text no. 2
    
3.
Laforga JB, Martínez P, Aranda FI. Benign fibroepithelial breast lesion with inflammatory eosinophilic infiltration. Breast J 2002;8:249-50.  Back to cited text no. 3
    
4.
Shiiki S. A case of hypereosinophilic syndrome with eosinophilic mastitis. J Jpn Surg Assoc 2004;65:620-4.  Back to cited text no. 4
    
5.
Komenaka IK, Schnabel FR, Cohen JA, Saqi A, Mercado C, Horowitz E. Recurrent eosinophilic mastitis. Am Surg 2003;69:620-3.  Back to cited text no. 5
    
6.
Bolca Topal N, Topal U, Gokalp G, Saraydaroglu O. Eosinophilic mastitis. JBR-BTR 2007;90:170-1.  Back to cited text no. 6
    
7.
Villalba-Nuño V, Sabaté JM, Gómez A, Vidaller A, Català I, Escobedo A, et al. Churg–Strauss syndrome involving the breast: A rare cause of eosinophilic mastitis. Eur Radiol 2002;12:646-9.  Back to cited text no. 7
    
8.
Thompson AB, Barron MM, Lapp NL. The hypereosinophilic syndrome presenting with eosinophilic mastitis. Arch Intern Med 1985;145:564-5.  Back to cited text no. 8
    
9.
Garg M, Kumar S, Neogi S. Eosinophilic mastitis masquerading as breast carcinoma. J Surg Case Rep 2012;2012:1.  Back to cited text no. 9
    
10.
Singh A, Kaur P, Sood N, Puri H, Garg B. Bilateral eosinophilic mastitis: An uncommon unheard entity. Breast Dis 2015;35:33-6.  Back to cited text no. 10
    
11.
Parakh A, Arora J, Srivastava S, Goel RK. Isolated eosinophilic infiltration of the breast. Indian J Radiol Imaging 2016;26:383-5.  Back to cited text no. 11
[PUBMED]  [Full text]  
12.
Takahashi K. A very rare case of eosinophilic mastitis. Int J Surg Case Rep 2018;49:251-4.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2]



 

 
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