Complete excision of the nipple core combined with inverted nipple correction: a novel surgical intervention for reducing recurrence in Zuska’s disease

Introduction

Breast Zuska’s disease, also referred to as breast fistula, subareolar abscess (1-3), chronic recurrent periareolar abscess (4-6), or subareolar granulomatous mastitis (7), was initially reported by Zuska in 1951. He documented five cases and designated the condition as subareolar abscess, which is also recognized as Zuska’s disease (8-10). This disease is characterized by the existence of abscesses accompanied by squamous epithelialization of the lactiferous ducts and ductal fistulas (11-13).

Zuska’s disease predominantly manifests in the areolar region, with abscesses and granulomas serving as the primary clinical manifestations. It primarily affects non-lactating women and is not associated with breastfeeding, although a small proportion of men can also be affected. This disorder is commonly observed among unmarried women. Typically, patients exhibit prominent inverted nipples with white, pasty secretions, which readily contribute to the local accumulation of bacteria and dirt (14).

Patients generally present with inflammatory symptoms around the nipple, which may subsequently progress to suppuration and ulceration of subareolar masses, accompanied by local pain, nipple discharge, and local pus exudation, which bears resemblance to ductectasis.

Presently, there exists no internationally well-established and unified treatment protocol for Zuska’s disease (13). Nevertheless, based on relevant literature, the disease can be managed through conservative treatment or surgical intervention. Conservative medical treatment mainly encompasses the use of antibiotics and traditional Chinese medicine. However, the therapeutic efficacy of such treatment is less than optimal, with a relatively high recurrence rate. Surgery serves as the primary treatment modality for this disease, and the timing and selection of the surgical procedure are of crucial importance for its cure. Traditional incision and drainage of abscesses can temporarily evacuate the abscess contents. However, over a period of time, partially healed tissues may become re-inflamed and suppurate. Even after skin healing, skin ulceration may recur.

In this study, we present our findings on the application of core duct excision and reconstruction as a surgical modality for Zuska’s disease, a challenging condition characterized by recurrent subareolar abscesses. Our results clearly demonstrate that this method is not only safe but also highly effective in resolving the symptoms and preventing recurrence. We present this article in accordance with the STROBE reporting checklist (available at https://gs.amegroups.com/article/view/10.21037/gs-2025-1-548/rc).

Methods

A retrospective analysis was carried out on patients diagnosed with Zuska’s disease at the Maternal and Child Health Hospital of Hubei Province from January 2008 to December 2022. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. The study protocol was approved by the Institutional Review Board of Maternal and Child Health Hospital of Hubei Province (No. 2021IECLW034). Written informed consent was obtained from all participants prior to their enrollment in the study.

The inclusion criteria were as follows: (I) patients who underwent surgical operations in our hospital during the non-lactating period, presenting with inverted nipples and white, creamy secretions (Figure 1); (II) a postoperative pathological diagnosis indicating mammary inflammation accompanied by squamous metaplasia (Figure 2); (III) patients possessing complete clinicopathological and follow-up data. Patients lacking either clinicopathological or follow-up data were excluded. In total, 109 patients were included in this study.

Figure 1 Two patients with typical Zuska’s disease presented with nipple retraction, extrusion of white paste, abscess, and fistula near the nipple areola. (A) Clinical manifestations in a patient with a history of multiple surgeries. (B) Clinical presentation of a patient without surgical intervention. Pink arrows: fistula; black arrows: white paste discharge.

Figure 2 This pathological image of Zuska’s disease demonstrates squamous metaplasia of the ductal epithelium (A,B). (A) 100× HE staining image. (B) 200× HE staining image. HE, hematoxylin and eosin.

Surgical procedure

All surgical procedures were performed under general anesthesia. All patients in the acute phase necessitated conservative treatment with antibiotics or glucocorticoids for a duration of 3–7 days. Among the 109 cases within this cohort, 23 cases (21.1%) underwent elective surgery subsequent to 3–7 days of antibiotic therapy during the acute phase. In contrast, 86 cases (78.9%) underwent direct surgical intervention owing to the formation of recurrent chronic fistulas subsequent to repeated conservative treatments at other medical institutions. An arc-shaped incision, with a length ranging from 3 to 4 centimeters, was made adjacent to the areola, precisely above the sinus tract opening or abscess. The skin and subcutaneous adipose tissue were incised, and necrotic and pathological tissues were excised along the sinus tract in the direction of the nipple (Figure 3). The fistula was excised. The most crucial aspect of this procedure was the complete resection of all ducts posterior to the nipple and areola, leaving only the superficial layer of the superficial fascia and the underlying vascular network. At this stage, the core of the nipple was entirely removed, leaving only a 1-mm duct stump, which allowed the inverted nipple to protrude. Subsequently, the inverted nipple was corrected by intermittently suturing the superficial layer of the superficial fascia beneath the nipple root using a purse-string suture technique, thereby facilitating the protrusion of the inverted nipple. The surrounding fascial tissue flaps were mobilized and sutured to fill the space beneath the nipple to prevent collapse caused by the void (Figure 4). The surgical area was irrigated twice with hydrogen peroxide, povidone-iodine, and H₂O₂. The glandular and subcutaneous tissues were sutured with 3-0 absorbable sutures, and the skin was continuously sutured with 4-0 absorbable sutures. The resected tissues were sent for routine pathological examination post-operation. Patients were followed up through telephone calls or clinic visits at 1, 6, 12, 24, and 36 months after the operation to observe the wound healing status, the corrective effect of nipple inversion, and the incidence of recurrence.

Figure 3 Potential incision sites for Zuska’s surgery.

Figure 4 Schematic diagram of the operation: the core of the nipple is removed. The inverted nipple is corrected through this process. A tissue flap behind the nipple is formed to avert recurrence of nipple inversion.

In cases where patients present with serum prolactin levels exceeding the normal range, bromocriptine is orally administered post-operatively. The dosage ranges from 0.5 to 1.5 tablets, and the treatment duration spans 6–12 months, contingent upon the extent of prolactin elevation.

Statistical analysis

Continuous variables are presented as mean ± standard deviation, while categorical variables are summarized as number (percentage). All statistical analyses were performed with SPSS software (version 26.0).

Results

An analysis was conducted on 109 cases of breast Zuska’s disease admitted to the Department of Thyroid and Breast Surgery at Maternal and Child Health Hospital of Hubei Province from January 2008 to December 2022. All patients underwent core duct excision and reconstruction under general anesthesia. The age of the patients ranged from 13 to 54 years, with a median age of 23 years. Among them, 79 were nulliparous, 24 had given birth to one child, and 6 had given birth to two children. The mean surgical duration was 42 [27–57] minutes, and the mean duration of symptoms was 2.5 (0.3–15) months. All 109 patients presented with inverted nipples accompanied by white, pasty secretions. Thirty-three patients had a history of previous surgeries and were recurrent cases. Seventeen patients had breast masses, 63 had breast skin erythema, 62 had subcutaneous abscesses, 43 had skin ulceration, 23 had ductal fistulas, 5 had bilateral disease, 17 had not breastfed after childbirth, 37 had serum prolactin levels exceeding the normal range, 5 had a smoking history, 7 were on psychiatric medications, 3 had autoimmune diseases, 5 had essential hypertension, 2 had type 2 diabetes, and 7 had polycystic ovary syndrome (Table 1). A total of 47 patients underwent breast magnetic resonance imaging (MRI), and no malignant lesions were detected. All postoperative pathological examinations revealed benign lesions characterized by chronic mastitis and squamous metaplasia.

Among the 109 surgical patients, the mean surgical duration was 42±15 minutes, and the mean blood loss was 8.7±3.6 mL. The mean scar length was 5.3±1.9 cm. Seven patients experienced postoperative nipple numbness. More crucially, no postoperative infections were noted. With a median follow-up period of 24 months (range: 18–36 months), only one case of recurrence (1%) was detected. Additionally, the postoperative cosmetic scores were as follows: 73 patients achieved scores ranging from 91 to 100, 29 patients obtained scores between 80 and 90, and 7 patients had scores below 80 (Table 2).

Discussion

Zuska’s disease is an inflammatory disorder that affects the main duct of the breast, which is classified as a benign breast condition. In comparison with other non-puerperal mastitis, Zuska’s disease is distinguishable from periductal mastitis (PDM) and granulomatous mastitis, as previously reported in the literature. Specifically, Zuska’s disease is mainly manifested by subareolar fistulas and squamous metaplasia of lactiferous ducts, while PDM and granulomatous mastitis are characterized by diffuse inflammatory infiltrates without the formation of characteristic fistulas (15).

Moreover, Zuska’s disease affects a younger population, predominantly unmarried and nulliparous women, with an average age of 23 years. The youngest patient in our hospital was only 13 years old. In contrast, the median age of patients with PDM is approximately 37 years old. Although Zuska’s disease primarily affects young women, a small proportion (4.3%) of cases have been documented in perimenopausal women (aged 45–55 years). This observation gives rise to the hypothesis that the estrogen-progesterone imbalance during the hormonal transition period may aggravate ductal epithelial metaplasia and the formation of keratin plugs.

This disease frequently manifests as inverted nipples, skin ulceration around the areola, and the formation of fistulas in the mammary ducts. The lesion area is smaller compared to that of plasma cell mastitis. The etiological factors may encompass inverted or short/malformed nipples, developmental anomalies, hyperprolactinemia, and bacterial infections.

In non-puerperal subareolar abscesses, the typically single-or double-layered cuboidal epithelial cells of the distal ducts undergo transformation into squamous epithelium. This squamous tissue generates an excessive amount of keratin, which forms a keratin plug. The keratin plug obstructs the duct, leading to ductal dilation due to the accumulation of secretions. As the duct progressively expands, the thin epithelial layer ultimately ruptures, releasing keratin that initiates an inflammatory response. The mammary tissue identifies the released keratin as a foreign substance, prompting macrophages to fuse into foreign-body macrophages in an attempt to clear this debris. Subsequently, bacterial invasion may give rise to the formation of subareolar abscesses.

Clinical manifestations mainly involve abscesses beneath the areola and skin ulceration. After anti-infective treatment and wound management, the ulcerated skin can heal. Nevertheless, a significant number of patients experience recurrent abscesses and skin ulceration, resulting in a chronic, relapsing condition with fistula formation in the areolar region. This disease rarely presents with fever, has a protracted course, and is not associated with breastfeeding. It most commonly occurs in unmarried women. The symptoms are predominantly inflammatory rather than lump-based, and the pain is not intense. Coupled with the limited health knowledge among young women, this often leads to a prolonged disease duration.

Inadequate comprehension of Zuska’s disease may result in a protracted illness and deformities due to inappropriate treatment. Conventional antibiotic treatment for abscesses and drainage (via aspiration or incision) is insufficient.

However, a consensus on the optimal treatment for this disease has yet to be achieved. Presently, the standard surgical approach mainly focuses on PDM, for instance, Hadfield’s procedure. Nevertheless, as previously deliberated, Zuska’s disease is distinguishable from PDM, and its distinct pathogenesis demands a customized therapeutic strategy. Notwithstanding, considering the pathophysiological resemblances between the conditions and the high recurrence rate typical of Zuska’s disease, we modified the Hadfield procedure—initially devised for PDM—and incorporated it with the specific pathophysiological characteristics of Zuska’s disease. This prompted us to put forward a combined approach of nipple core excision in conjunction with correction of nipple inversion for the management of this condition.

For example, in accordance with the standard Hadfield’s procedure, Zhang et al. reported that diverse surgical approaches, such as wide surgical excision, fistulectomy, and extended excision combined with the transfer of a random breast dermo-glandular flap (BDGF), were efficacious in the treatment of PDM (16). Additionally, Taffurelli et al. reported that eighteen female patients underwent the Hadfield surgical treatment (fistulectomy). All patients who received operative treatment exhibited no postoperative complications and were satisfied with the cosmetic outcomes (4).

In light of the age and clinical features of the aforementioned cases, it is plausible that certain patients with plasma cell mastitis or granulomatous mastitis were incorporated. Moreover, notwithstanding the adjustments made to the surgical procedure in the studies mentioned above, the recurrence rate still remains as high as around 10%. Most crucially, the limited sample size of the included cases restricts the generalizability of the surgical outcomes.

Furthermore, Naeem and colleagues reported that ultrasound-guided aspiration of breast abscesses with the appropriate utilization of antibiotics represents a superior treatment modality in comparison to incision and drainage (17). Giacalone reported that 24 patients suffering from recurrent periareolar abscesses, who had undergone surgical treatment three or more times between January 2001 and December 2008, were treated with the combined resection of the fistula, the terminal milk ducts, and the mammary gland involved in the inflammatory process. One patient was treated via excision of the nipple and areola. Six patients (25%) evaluated their treatment outcome as excellent, and 13 patients (54%) regarded their results as good. Five patients assessed their overall outcome as fair (21%), and no patients deemed their results as poor (18). Li et al. reported a retrospective analysis of patients surgically treated for subareolar abscess at a single institution from 1993 to 2005. Successful definitive treatment of retroareolar abscesses necessitates the excision of the central nipple, including the obstructed ducts. This technique attains a cure rate of 91% and an overall satisfaction rate of 95% in the cosmetic outcome of the nipple (19), although the recurrence rate remains relatively high (13).

Nevertheless, all the aforementioned studies are constrained by small sample sizes (less than 30 cases), comparatively high recurrence rates (ranging from 6% to 25%), and brief follow-up periods.

We hypothesize that the high recurrence rate can be ascribed to the fact that Hadfield’s procedure and its related techniques mainly target the fistula and only 1–2 major ducts associated with it. Conversely, Zuska’s disease may involve the majority, if not all, of the lactiferous ducts. Therefore, complete excision of all ducts posterior to the nipple is required to attain therapeutic effectiveness. Moreover, this approach does not rectify concomitant nipple inversion. The failure to address the inverted nipple can result in the accumulation of secretions within the depressed nipple, rendering patients more susceptible to recurrent infections. In conclusion, persistent nipple inversion, in combination with the presence of residual ductal tissue of considerable length posterior to the nipple, can continue to act as a focus for disease recurrence.

The present center adopts an arcuate incision along the areola. This incision optimizes the preservation of the blood supply to the nipple and areola, thereby promoting wound healing. In comparison with transverse incisions that divide the nipple and areola, this approach results in a more aesthetically appealing scar and a lower incidence of complications, such as nipple necrosis. The crux of the surgery lies in disconnecting and excising all the ducts beneath the nipple, rather than merely the diseased ones, which is of great significance for preventing recurrence. The removal of the ducts also entails the removal of some nerves, leading to a reduction in the neuroendocrine responses stimulated by the nipple (Figure 5).

Figure 5 Intraoperative and postoperative controls of Zuska’s disease. (A) Resection of the enlarged mammary duct located behind the nipple. (B) After removal of the diseased duct behind the nipple, the fistula orifice becomes visible. (C) A case of Zuska’s disease prior to operation, characterized by inverted nipple, abscess formation behind the nipple, and reddened and swollen skin. (D) Following treatment of Zuska’s disease, the nipple core is removed, thereby correcting the inverted nipple. Black arrow: diseased duct. Pink arrow: fistula orifice.

Three reasons for the high recurrence rate of this disease have been identified. Firstly, the duct associated with the fistula may not be the sole diseased duct, and the oversight of other diseased ducts during surgery is one of the primary causes of recurrence. Secondly, the diseased duct may be interconnected with the surrounding ducts, indicating that the excision of only the visibly diseased duct fails to comprehensively resolve the problem. Thirdly, the incomplete removal of all ducts renders it challenging to completely correct inverted nipples, which may give rise to recurrent infections and epithelial squamous metaplasia in the remaining ducts, ultimately leading to recurrence (20).

Considering that all cases of this disease are characterized by inverted nipples, surgical correction of the nipple inversion should be carried out whenever feasible. The surgical procedure entails cleansing the core dominant ducts beneath the nipple and subsequently performing a purse-string suture on the tissues below the nipple (Figure 6). This suturing technique optimally prevents subsequent nipple retraction and depression. Moreover, the glandular tissue needs to be dissected from behind the nipple to fill the defect and prevent scar contraction from leading to nipple inversion again. This surgical approach achieves excellent corrective outcomes, especially for larger nipples, and seldom necessitates revision surgery.

Figure 6 The posterior image of the nipple following core papillectomy reveals that the broken end of the milk duct resembles a strawberry bulge. Only a superficial layer of superficial fascia and the blood vessels within the superficial fascia remain.

Common surgical methods employed in other hospitals include skin incision and drainage, which can remove subcutaneous necrotic tissue and abscesses, facilitating skin healing within 1–3 months. Nevertheless, the recurrence rate remains relatively high. Most patients require long-term wound care and debridement, and fistulas frequently form. Another approach is the topical application of traditional Chinese medicine, which can promote the rapid formation and rupture of abscesses, enabling wound healing. However, abscesses and ruptures tend to recur, rendering this approach ineffective for definitive treatment. Oral anti-tuberculosis drugs have also been utilized for treating this disease, but they cause substantial liver cell damage, exhibit notable side effects, require a prolonged treatment course, and have a high recurrence rate.

The crucial aspects of our surgical approach encompass strict hemostasis during the operation to preclude hematoma formation. Simultaneously, excessive debridement of the tissues beneath the nipple should be avoided to preserve the superficial vascular network of the superficial fascia, thus preventing nipple ischemia and necrosis. In cases where patients exhibit significant tissue defects subsequent to excision, any glandular flap can be employed to fill the defect, which helps prevent postoperative skin depression and maintain aesthetic appearance.

Postoperatively, drainage tubes should be placed. For larger wounds, external negative-pressure drainage balls are utilized, while for smaller wounds, sterile drainage tubes without negative-pressure devices are applied.

Although certain studies advocate staged strategies (e.g., delayed surgery subsequent to abscess drainage) to mitigate surgical risks during the acute inflammatory stage, the distinctiveness of Zuska’s disease resides in its recurrent infections and persistent engagement of the catheter system. Early radical resection can impede the continuous release of keratinous substances, preclude tissue fibrosis induced by protracted inflammation, and curtail the disease duration. Inverted papillae represent a notable anatomical predisposing factor for Zuska’s disease. This surgical approach, which integrates core resection of the papilla with subareolar tissue filling, can concurrently rectify inversion and obviate secondary surgery. Should a staged strategy (drainage followed by correction) be employed, the augmented surgical complexity attributable to inflammatory adhesions may extend the healing period.

General anesthesia was selected over local anesthesia in this study. Although local anesthesia can potentially mitigate systemic risks, the surgical scope of this research encompassed the intricate anatomical structures in the areolar region (e.g., the mammary duct system and the subdermal vascular network). The papilla-areolar area is innervated abundantly and exhibits high pain sensitivity. The surgical procedure demanded meticulous and comprehensive lesion removal, differentiation and excision of the mammary ducts, as well as precise suturing to avert nipple necrosis. The surgical process was characterized by complexity and a relatively long duration (42±15.5 minutes). General anesthesia guaranteed the immobility of the patient during the operation, thereby reducing the risks associated with the procedure. It was especially appropriate for patients with psychological anxiety or poor pain tolerance.

Potential latent issues associated with the staging strategy include an elevated risk of infection during drainage, multiple bacterial colonizations, cumulative trauma from repeated surgeries, and a deterioration in the patients’ quality of life during the waiting period (e.g., inability to take a shower, incomplete wound closure lasting for weeks or months, and increased psychological anxiety). The hidden costs of the staging strategy also escalated, such as multiple surgical interventions, prolonged dressing-change procedures, and the use of antibiotics, which led to an increase in medical expenses.

Our observations also indicate that, apart from inverted and malformed nipples, hyperprolactinemia may act as a contributing factor to postoperative recurrence in Zuska’s disease. It potentially exerts its influence through regulatory effects on epithelial proliferation and inflammatory pathways. Nevertheless, the specific pathogenic mechanism remains obscure and necessitates further investigation.

For patients with hyperprolactinemia, postoperative oral administration of bromocriptine to reduce prolactin levels has been shown to diminish postoperative fluid accumulation and recurrence. Prolactin itself is a significant neuroendocrine factor with immunomodulatory functions, regulating both cellular and humoral immunity (21,22).

This research recruited nulliparous women with a mean age of 23 years. Fertility plans, breastfeeding intentions, and acceptance of functional loss were evaluated through questionnaires and interviews. The findings indicated that 84.4% of the patients had fertility plans, 79.8% attached importance to breastfeeding, yet 96.3% were prepared to sacrifice this function for the treatment of Zuska’s disease. The pathological characteristic of Zuska’s disease is ductal squamous metaplasia, necessitating the complete resection of the affected ducts to prevent recurrence, which would entirely disrupt lactation function. Non-surgical treatment is prone to recurrence, resulting in nipple ulceration, fistulas, and breast deformity. Conservative treatment is provided to a small number of patients with a strong desire to preserve function, but the recurrence rate is high, and the majority of patients choose surgery for a cure. Treatment decisions are made based on the condition and patient requirements. Surgery is recommended for recurrent or severe cases, while mild cases may be postponed. This study respected patient preferences and facilitated personalized decision-making through assessment.

Limitations

This research presents three primary limitations: geographic and demographic bias stemming from single-center recruitment, which may potentially have an impact on external validity; restricted statistical power associated with a cohort of 109 patients, thereby constraining subgroup analyses (e.g., age-tratified outcomes); inadequate mechanistic comprehension of disease recurrence, which mandates functional investigations to verify the roles of candidate genes and epigenetic regulators.

Conclusions

This research presents a novel integrated surgical methodology that combines total nipple core excision with inverted nipple correction. This approach exhibits enhanced efficacy in the management of Zuska’s disease by simultaneously addressing ductal epithelial hyperplasia and anatomical malformations. Clinical results indicated a substantial decrease in recurrence rates (from 25% to 1%) when compared with conventional treatment modalities, thereby justifying its potential for extensive clinical application.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the STROBE reporting checklist. Available at https://gs.amegroups.com/article/view/10.21037/gs-2025-1-548/rc

Data Sharing Statement: Available at https://gs.amegroups.com/article/view/10.21037/gs-2025-1-548/dss

Peer Review File: Available at https://gs.amegroups.com/article/view/10.21037/gs-2025-1-548/prf

Funding: This study was supported by the Hubei Provincial Health and Health Science and Technology Project (No. WJ2025M103).

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://gs.amegroups.com/article/view/10.21037/gs-2025-1-548/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. The study protocol was approved by the Institutional Review Board of Maternal and Child Health Hospital of Hubei Province (No. 2021IECLW034). Written informed consent was obtained from all participants prior to their enrollment in the study.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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