Complicated Subareolar Breast Abscess in Non-Lactating Woman: Case Report and Analysis

COMPLICATED SUBAREOLAR BREAST ABSCESS IN NON-LACTATING WOMAN: CASE REPORT AND ANALYSIS

Nikolaos Tasis∗, Ioannis Tsouknidas∗, Panagiota Kripouri∗∗,1, Georgios Filippou♦, Vasiliki Koumaki4 and Dimitrios Filippou♦♦ ∗Postgraduate Student, Department of Anatomy and Surgical Anatomy, Medical School, National and Kapodistrian University of Athens, Athens, Greece., ∗∗Resident in General Surgery; Postgraduate Student, Department of Anatomy and Surgical Anatomy, Medical School, National and Kapodistrian University of Athens, Athens, Greece., ♦General Surgeon., 4Department of Microbiology, Medical School, University of Athens., ♦♦General Surgeon; Director of Surgical Oncology and Laparoscopic Surgery IASO General Hospital; Research Fellow Department of Anatomy and Surgical Anatomy, Medical School, National and Kapodistrian University of Athens, Athens, Greece.

ABSTRACT Background: Non-puerperal subareolar breast abscess was first described by Zuska et al. in 1951. The disease is benign. To treat it, control of galactorrhea is advised. In this case, galactorrhea was drug-induced, but the drug could not be discontinued. We discuss our plan of treatment of Zuska’s disease in a patient with uncontrollable galactorrhea. Case summary: A fifty years old non-lactating female patient presented with a subareolar breast abscess. She underwent a needle aspiration, a Hadfield’s procedure, incision and drainage and finally resulted in a lumpectomy. We discuss her case, her comorbidities and our plan of treatment. Conclusion: We believe this case is remarkable because of the challenging treatment course of the otherwise benign disease. It suggests that in cases of continuing galactorrhea Zuska’s disease is surgically treated with the wound healing by second intention.

KEYWORDS breast abscess, Hadﬁeld’s, needle aspiration, Zuska’s disease

Case Report In December 2014, a fifty years old non-lactating female of Cau- Introduction casian origin presented in a local hospital’s emergency room A non-perpetual subareolar breast abscess is a rare benign dis- with redness, swelling and tenderness in the right breast. The ease, arduously manageable for both physician and patient. This patient was obese with a history of smoking, epilepsy, anxiety rare condition and its association with lactiferous duct fistulas disorder and galactorrhea, and had given birth and lactated were first described by Zuska et al. [1] in 1951. Therapy for twice. Clinical examination revealed swelling and tenderness this demanding condition remains controversial; it can take sev- close and under the areola. eral time, many recurrences and may even raise the need for The subareolar abscess was identified and antibiotic treat- a radical solution like a mastectomy. This is a case report of a ment of 500mg cefaclor, three times per day, was administrated female patient suffering from Zuska’s disease with an extremely as empirical treatment. A week later the patient underwent difficult therapy course. needle aspiration for diagnostic and therapeutic purposes. In the bacterial culture that followed, Staphylococcus aureus was isolated. In May 2015 the patient presented to our centre, and

asymptomatic lactiferous duct fistula was observed. Hadfield’s procedure (primary duct excision) took place under general anaesthesia and patient had a smooth postoperative recovery. However, in February 2016, a new inflammation was noted

in the same breast along with another subareolar abscess. A small incision and drainage took place.

In June 2016, there was a recurrence of the inﬂammation and the subareolar abscess. The decision for a radical solution was taken, and the patient underwent a lumpectomy. The wound healed by secondary intention and the results are satisfactory until today.

Discussion Zuska’s disease affects mainly women, with a wide age range (second to an eighth decade [2]) but with several male cases reported [3,4]. Squamous metaplasia of the cuboid epithelium of the ducts was introduced in detail as the offset of the disease by Habif et al. [5]. Metaplasia leads to hyperkeratinization, obstruction of the duct by keratin plugs, dilation and eventu- ally ruptures [6]. Keratin obstruction is considered as a foreign body setting off the inflammatory response. Bacterial invasion causes the formation of subareolar abscess, whose spontaneous drainage generates fistulas. Continual duct obstruction can lead to chronic inflammation and recurrences, raising the challenge for the physician [2]. Risk factors associated with peri- and subareolar non- Figure 1: Tumefaction and redness were apparent during clinical perpetual breast abscesses include mainly smoking, type 2 di- examination. abetes mellitus (DM), obesity and nipple piercing. Bharat et al. [7] suggest a strong connection between smoking and obesity and primary breast abscess. There was also found the relation of recurrences with smoking and at least one co-morbid condition (hypertension, diabetes, asthma or psychiatric illness). Firm rela- tivity between DM and breast abscess is encouraged by Rizzo et al. [8] as well. DM not only contributes to primary infection but also prolongs hospital stay and interferes with treatment management. Tobacco smoking and nipple piercing correlation with breast abscess are stated by Gollapalli et al. [9] along with proven high rates of recurrences for smokers. Overall, smoking seems to be the primary risk factor for primary breast abscess and recurrences, maybe due to lower β-carotene plasma levels [10] which provoke squamous metaplasia. Our patient was obese and a smoker. Resulting analysis above, considering her comor- bid conditions, such as epilepsy and anxiety disorder, she was a high-risk patient for breast abscess. Control of our patient’s Figure 2: After the second intention healing the wound left a intermittent galactorrhea could ease her treatment. However, minimal scar and a satisfactory aesthetic outcome. her galactorrhea was linked to prescribed antipsychotic drug intake. The patient received the antipsychotic under her psychi- atrist’s order who strongly advised against any changes in her treatment plan because of her previous psychiatric history. Thus, galactorrhea could not be controlled. Smoking also explains her continuous recurrences and difficult management. Following needle aspiration of our patient, Staphylococcus aureus was isolated, which is likely the most common microorganism found in primary non-perpetual subareolar breast abscesses [11]. An anaerobic microorganism or mixed flora tend to be isolated in recurring abscesses more often, while there is a high percentage of sterile culture in both primary and recurring conditions [9, 10]. Dabbas et al. [12] reinforce this claim, stating Staph aureus Figure 3: Our patient underwent needle aspiration under ultra- as the most prevalent causative organism in both lactating and sound guidance. non-lactating women. Sterile cultures percentage found higher in non-lactating women, and anaerobic bacteria were as well the second commonest culture result. Rare bacteria isolated are found in the bibliography as well, such as C. Difficile [13] and Corynebacterium kroppenstedtii [14] related with significant complications. Association of smoking and organism isolated is not supported, however, a slight increase of anaerobes and coagulase-negative Staphylococcus is noted [9]. Treatment of non-perpetual sub- or periareolar breast abscess is complicated

Figure 4: Timeline of the clinical presentation.

and controversial. Early antibiotics administration seems to im- 2. Kasales C., Han B., Chetlen A., Kaneda H., Shereef S., Non- prove abscess formation and augmentation and to prevent a puerperal Mastitis and Subareolar Abscess of the Breast. severe infection [2,10,12,15-20] especially in combination with AJR:202, February 2014, W133-W139. aspiration or incision and drainage. Empiric antibiotic treatment recommendations differ. Amoxicillin with clavulanic acid 3. Aiyappan SK, Ranga U, Veeraiyan S, Idiopathic Subareolar and erythromycin with or without metronidazole when allergy Breast Abscess in a Male Patient. Journal of Clinical and to penicillin exists [16], flucloxacillin with metronidazole [17] Diagnostic Research. 2015 Jan, Vol-9(1): TJ01. cloxacillin or first-generation cephalosporin [18, 19] are some of 4. Johnson SP, Kaoutzanis C, Schaub GA, Male Zuska’s dis- the antibiotic suggested. Aspiration with needle and drainage is ease. BMJ Case Rep. 2014. noted as first-line treatment in bibliography [2, 15, 17, 18]. Incision and drainage are chosen when dealing with larger ab- 5. Habif DV, Perzin KH, Lipton R, Lattes R. Subareolar abscess scesses (<5cm) of needle aspiration are unsuccessful [18]. How- associated with squamous metaplasia of lactiferous ducts. ever surgical management is suggested early when breast ab- Am J Surg 1970;119:523–26. scess is complicated by fistula or necrosis [20]. Surgical management is mainly proposed when recurrent breast abscesses 6. Meguid MM, Oler A, Numann PJ, Khan S. Pathogenesis- occur, more than 3 to 5 needle aspiration sessions took place or based treatment of recurring subareolar breast abscesses. infection worsened [19]. When surgical management is chosen, Surgery 1995;118:775–82. removal of the obstructed lactiferous duct is vital. Failure to accomplish total excision may lead to recurrences [10]. Several 7. Bharat A, Gao F, Aft RL, Gillanders WE, Eberlein TJ, Mar- techniques for lactiferous duct excision are described. Hadfield genthaler JA, Predictors of Primary Breast Abscesses and introduced an incision or the lower margin of the areola to al- Recurrence. World J Surg 2009; 33:2582–2586. low lifting the nipple and including any fistulas or obstructed 8. Rizzo M, Peng L, Frisch A, Jurado M, Umpierrez G, Breast lactiferous ducts underneath. [21, 22] Hadfield’s technique is Abscesses in Nonlactating Women With Diabetes: Clinical widely used [18], but other techniques with radical elliptical Features and Outcome. Am J Med S 2009; 338(2):123-126. incision [15] or transverse incision through the nipple are also described with satisfactory cosmetic and healing outcomes. Fi- 9. Gollapalli V, Liao J, Dudakovic A, Sugg SL, Scott-Conner nally, lumpectomy of radical mastectomy consists the last resort CEH, Weigel RJ, Risk Factors for Development and Recur- when recurrence follows excision of lactiferous duct or malig- rence of Primary Breast Abscesses. J Am Coll Surg 2010; nancy is revealed. In our patient, another subareolar abscess 211(1):41-47. took place after Hadfield’s excision. The need for a radical solution led to a lumpectomy with satisfactory cosmetic results and 10. Versluijs-Ossewaarde FNL, Roumen RMH, Goris RJA, Sub- recurrence-free until today. areolar Breast Abscesses: Characteristics and Results of Surgical Treatment. Br J, 2005; 11(3):179–182.

Conclusion 11. Ramakrishnan R, Trichur RV, Murugesan S, Cattamanchi S, Analysis of the microbial ﬂora in breast abscess: a retrospec- Zuska’s disease or periareolar non-perpetual breast abscess is tive cohort study conducted in the emergency department. a complicated condition with the need for careful and delicate Int Surg J. 2017; 4(7):2143-2147. management. Several risk factors and mainly smoking con- tribute to the severity of the infection. Antibiotic use and needle 12. Dabbas N, Chand M, Pallett A, Royle GT, Sainsbury R, Have aspiration with drainage are useful in early management, but the Organisms that Cause Breast Abscess Changed With failure may raise the need for invasive surgical care, lactiferous Time?– Implications for Appropriate Antibiotic Usage in duct excision or lumpectomy like in our case. In cases of uncon- Primary and Secondary Care. Br J, 2010; 16(4):412–415. trollable galactorrhea, we suggest surgical treatment of Zuska’s disease with wound healing by second intention. Careful and 13. Durojaiye O, Gaur S, Alsaffar L, Bacteraemia and breast thoughtful management, however, will result in the desired abscess: unusual extra-intestinal manifestations of Clostrid- outcome. ium difﬁcile infection. J Med Micro 2011; 60:378–380.

14. Goh Z, Tan AL, Madhukhumar P, Yong WS, Recurrent Disclosure Statement Corynebacterium kroppenstedtii Breast Abscess in a Young Asian Female. Br J,2015; 21(4):431–432. There were no financial support or relationships between the authors and any organization or professional bodies that could 15. Lannin DR, Twenty-two year experience with recurring pose any conflict of interests. subareolar abscess and lactiferous duct fistula treated by a single breast surgeon, Am J Surg,2004;188:407–410.

Competing Interests 16. Dixon JM, Breast Abscess, Br J Hosp Med (Lond). 2007 Written informed consent obtained from the patient for publica- Jun;68(6):315-20. tion of this case report and any accompanying images. 17. Agrawal A, Kissin M, Breast Abscess, Br J Hosp Med (Lond). 2007; 68(11):M198-9. References 18. Lam E, Chan T, Wiseman SM, Breast abscess: evidence 1. Zuska J., Crile G., Ayres W., Fistulas of lactiferous ducts. based management recommendations. Expert Rev Anti Am J Surg 1951;81:312–7. Infect Ther. 2014;12(7):753-62.

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22. Hadﬁeld GJ. Further experience of the operation for excision of the major duct system of the breast. Br J Surg 1968; 55:530–535.