Granulomatous lobular mastitis treated by a combined internal and external treatment of traditional Chinese medicine: A case report

Abstract

BACKGROUND

Granulomatous lobular mastitis (GLM) is a rare benign inflammatory disease of the breast and is classified under comedo mastitis in traditional Chinese medicine (TCM). The etiology of this disease is unknown, and it mainly occurs in women of childbearing age. The diagnosis depends on histopathological biopsy. At present, there is no systematic and standardized treatment plan for GLM. In the absence of evidence supporting an infectious etiology, affected patients might continue to receive multiple courses of antibiotics and unnecessary surgery.

CASE SUMMARY

A 37-year-old Chinese woman with a history of coronavirus disease 2019 infection presented with swelling and pain in the left breast. She also had erythema, nodules in the lower extremities, arthritis in both knees, cough, and headache. In the early stage of GLM, the mass was not significantly reduced by conservative treatment with internal application of TCM; hence, surgical treatment was carried out. The aim of postoperative treatment was to drain the pus, eliminate the necrosed tissue, and expand the muscles; fumigation and washing using TCM was applied.

CONCLUSION

Combined internal and external treatment with TCM, following the principle of “Prioritize internal treatment before ulceration and emphasize external treatment after ulceration” was effective in our patient with GLM. The prognosis was good. We believe that TCM offered valuable therapeutic benefits in this disease.

Key Words: Granulomatous mastitis; Traditional Chinese medicine; Erythema; Nodules in the lower extremities; Arthritis in both knees; Cough and headache; Case report

Core Tip: We focus on the treatment efficacy of combining traditional Chinese (TCM) and Western medicine for granulomatous lobular mastitis (GLM). GLM was treated conservatively with TCM in the early stages, but the disease symptoms did not significantly improve. Hence, surgery was required. Postoperatively, we drained the pus, removed decay, and dilated muscles through TCM-based fumigation and washing. The combination of TCM and Western medicine had a definite therapeutic effect on GLM and offered good prognosis. TCM offered valuable therapeutic benefits in this disease.

INTRODUCTION

Granulomatous lobular mastitis (GLM) is a rare chronic inflammatory breast disease characterized by aperiodic breast pain, breast lumps, and long-term recurrent episodes resulting in the formation of fistula or sinus tract[1]. While the etiology of GLM has not been fully elucidated, factors such as autoimmunity, hyperprolactinemia, hormonal imbalance, α 1-antitrypsin deficiency, smoking, and diabetes mellitus have been considered to play a role in its pathogenesis[1,2]. Therefore, there is currently no established, standardized treatment protocol for GLM.

At present, multiple therapeutic modalities are available for GLM, including hormone therapy, surgical treatment, and immunosuppressive therapy, each bearing its distinct strengths and limitations[2,3]. However, a systematic and standardized treatment protocol is yet unavailable. Traditional Chinese medicine (TCM) has been shown to have a significant therapeutic effect on GLM, especially in patients who require non-surgical treatment[4]. TCM follows the principle of “Prioritize internal treatment before ulceration, and emphasize external treatment after ulceration”, that is, conservative treatment should be adopted in the early stage of the disease; surgical debridement should be considered only if the results of conservative treatment are unsatisfactory. Moreover, postoperative TCM syndrome differentiation and dressing change can effectively promote wound healing[3-5].

CASE PRESENTATION

Chief complaints

A 37-year-old woman visited the Breast Surgery Department of Longhua Hospital on June 17, 2022, with a large, painful, ruptured, pus-filled left breast mass.

History of present illness

The patient developed a solid lump in her left breast towards the end of her coronavirus disease 2019 isolation period on April 30, 2022. A few days later, her left breast was injured by her baby's kick, accompanied by redness and swelling of the breast skin. One week later, she developed swelling and pain in both lower limbs accompanied by erythema nodules and cough. The patient received empirical cefuroxime for 2 weeks, with no relief of symptoms and subsequent headaches.

History of past illness

No special history of past illness.

Personal and family history

No specific personal and family history.

Physical examination

Physical examination showed a sunken left nipple and a hard palpable mass (12 cm × 15 cm) in the left breast and areola area, with obvious tenderness, multiple ulcerations, hypergranulated tissue, and slight pus discharge. Dark red patches were observed on both lower limbs (Figure 1).

Figure 1 Bilateral edema of the lower limbs with erythematous nodules. A: Initial diagnosis on June 17, 2022, before medication; B: Second visit on June 23, 2022, medication for 1 week; C: Third visit on June 30, 2022, 2 weeks of medication.

Laboratory examinations

B-ultrasound examination showed irregularly contoured hypoechoic mass (Figure 2A and B).

Figure 2 Granulomatous mastitis of the left breast. A and B: Ultrasound images (outer lower quadrant of the left breast: 50 mm × 24 mm, below the left nipple: 46 mm × 22 mm); C and D: Magnetic resonance imaging images, manifesting non-mass enhancement lesions in multiple areas of the left breast, with irregular shape, "grid and burr" signals at the edges, surrounding skin edema, and skin fistula formation in the inner and lower quadrants; E and F: Pathological images (hematoxylin and eosin staining; original magnification, × 200), showing squamous epithelial hyperplasia, interstitial acute and chronic inflammatory cell infiltration, local abscess and granuloma formation, with vacuoles in the center, surrounded by layers of inflammatory cells, such as neutrophils and multinucleated giant cells.

Imaging examinations

Magnetic resonance imaging showed non-mass enhancement lesions in multiple areas of the left breast with irregular shape, edema, and fistula formation in the inner and lower quadrants (Figure 2C and D).

FINAL DIAGNOSIS

Given these findings, the patient was finally diagnosed with GLM. The TCM diagnosis was acne-prone mastitis, and the syndrome differentiation was dampness-heat of liver channel.

TREATMENT

The patient subsequently received TCM treatment with the principle of soothing the liver, removing heat, regulating blood flow, and eliminating the mass. The prescription of TCM was as follows: Bupleurum (9 g), Scutellaria (9 g), Radix paeoniae rubra (9 g), Angelica (12 g), Smilax china (15 g), Chinese honeylocust spine (9 g), Qingpi (9 g), Tangerine peel (9 g), Poria with hostwood (20 g), Saposhnikovia divaricata (9 g), Rehmannia glutinosa (20 g), Fructus crataegi (12 g), Radix codonopsis (12 g), Caulis spatholobi (20 g), Luffa (18 g), Radix cyathulae (15 g), Maize silk (20 g), Pinellia (9 g), Poria (20 g), Folium isatidis (20 g), Ramulus cinnamomi (9 g). The above ingredients were decocted in 200 mL water for one daily dose, for a total of 14 doses. In addition, Chonghe ointment and Baiyu ointment made by the hospital were used for external application. A 2-mm-thick layer of ointment on gauze was applied, covering an area larger than the lump. Chonghe ointment or Baiyu ointment was alternately used for 4-8 ha day.

After 14 days of continuous TCM treatment, the left breast mass remained, but its texture softened. The cough was under control, but the patient started experiencing headaches. Therefore, we removed angelica, smilax china, Chinese honeylocust spine, qingpi, poria with hostwood, and ramulus cinnamomic from the prescription and added radix puerariae (30 g), chuanxiong rhizoma (15 g), angelica dahurica (9 g), rhizoma corydalis (30 g), malt (60 g), and cortex mori (15 g), for a total of 28 doses. After 42 days of continuous TCM treatment, the left breast mass, headache, and dark red pigmentation remained, but the symptoms of joint pain, erythema nodosum, and cough improved. Open debridement was performed on August 1, 2022. The patient was treated with TCM syndrome differentiation and dressing change according to the different post-operative stages. Two incisions appeared post-operation, dressings, drainage, and medication on the incisions led to rapid relief of headache by the 5^th post-operative day. On the 12^th post-operative day, wound suppuration had cleared and was treated with TCM fumigation. Meanwhile, infrared radiation was used to warm Yang, dissipate accumulation, promote blood circulation, and generate muscle mass. After fumigation and washing, Kangfuxin liquid gauze was used to promote wound closure, and the wound surface healed 24 days after the operation. Physical breast ultrasound examination showed that the upper outer sores of the left breast had healed (Figure 3).

Figure 3 The patient's left breast. A: On the day of surgery, multiple ulcers were seen, accompanied by a small volume of yellow-white pus overflow; B: On the 2^nd post-operative day, the two small sores are connected with a cotton thread, which can be pulled out during daily dressing changes, and after spreading the therapeutic powder, the cotton thread is dragged into the inside of the sore cavity, so that the powder is sent to the sore cavity; C: On the 12^th post-operative day, the wound cavity was fumigated and washed; D: On the 24^th post-operative day, a type of round wound measuring about 2 cm × 1 cm was observed on the medial side of the left areola.

OUTCOME AND FOLLOW-UP

The patient was followed-up on September 24, 2022. There was no discomfort in the left breast and the ulcerated cavity had healed (Figure 4).

Figure 4 Images of the patient's breasts on September 24. A: The surgical wound has been completely healed and the surgical scar is not obvious; B: The shape of the patient's both side of breasts is basically symmetrical after surgery. This combination internal and external traditional Chinese medicine treatment of granulomatous lobular mastitis has a protective effect on the patient's breast appearance.

DISCUSSION

In TCM theory, the nipple belongs to the liver meridian and the breast belongs to the stomach meridian[6]. Liver Qi thrives in a state of relaxation and free-flowing and resists being constrained or suppressed. However, women are prone to depression, which depresses the liver and causes liver fire to build up over time[5,6]. Moreover, when liver Qi is not flowing smoothly, it can easily invade the spleen, causing the spleen to lose its function of transportation and transformation. Finally body fluids accumulate into phlegm, which makes the disease protracted and difficult to cure[7]. Chinese herbal medicine is usually used as a formulation guided by TCM theory, combining multiple herbs in the prescription[6-8]. The prescription used in this study is based on radix codonopsis and poria to invigorate the spleen and dissipate phlegm, and tangerine peel and Pinellia to dry and dampen the phlegm. Smilax china and Chinese honeylocust spine can reduce the swelling and discharge of pus[8]. Fructus crataegi can reduce fat turbidity and blood circulation detumescence. Folium isatidis and rehmannia glutinosa can clear heat and detoxify. Qingpi and luffa are used to regulate qi and dredge meridians. Angelica and caulis spatholobi activate blood circulation and remove blood stasis[8,9]. In addition, caulis spatholobi combined with radix cyathulae can also relieve pain in the lower limbs, and maize silk can remove moisture and dredge the mammary ducts[10]. Ramulus cinnamomi and saposhnikovia divaricata can relieve the surface and disperse pathogenic factors, warm meridians, and relieve cough caused by wind and cold[11]. In addition, poria with hostwood can help with good sleep and can calm the mind, especially for women with anxiety and sleep problems because of prolonged pain during the illness[8,10]. In external treatment, Baiyu ointment was effective to clear heat and swelling and Chonghe ointment could promote blood circulation and remove blood stasis. They were used alternately to increase the curative effect[11,12]. At the second visit, the patient developed headaches, which was thought to be caused by blood stasis and blocking collaterals[3,10]. TCM for regulating qi and promoting blood circulation was added, such as with pueraria, chuanxiong, angelica dahurica, and rhizoma corydalis[12-14]. The patient still had cough, so cortex mori was added to relieve cough and asthma, and finally stir-baked malt was added to regulate qi, strengthen the spleen, and eliminate swelling[10,12].

Based on internal treatment, external treatment was given to improve the treatment efficacy. After surgery, the external treatment was adjusted to steam fumigation and washing with TCM every day. TCM fumigation and washing can benefit qi and promote blood circulation with the help of medicinal force and heat, promote wound healing, and shorten the disease course. The fumigation and washing prescription of TCM (raw astragalus 30 g, angelica 9 g, salvia miltiorrhiza 30 g, ramulus cinnamomi 9 g, dandelion 30 g, radix curcumae 15 g, catechu 12 g, lycopus lucidus 15 g) is heated by a machine to form steam and act on the wound surface. The specific scheme is as follows: once a day for 30 minutes, and generally 12 days for a treatment cycle. The length of the cycle can be adjusted according to the size of the ulcer cavity. During fumigation and washing, the steam nozzle should be kept a proper distance from the wound surface to avoid scalding. Infrared radiation is also used to promote wound blood circulation and muscle relaxation[13,14].

Most patients with GLM are women of childbearing age[2]. Considering the breast aesthetics and psychological factors, conservative and less invasive treatment methods are often used in the early stage[4-6]. In the early stage of GLM, oral TCM combined with external application of plaster may completely relieve the swelling. If the condition does not improve, surgical intervention may be required. Our patient was treated according to the above-mentioned protocol and showed good recovery.

CONCLUSION

A combined method of internal and external treatment with TCM, following the principle of “Prioritize internal treatment before ulceration and emphasize external treatment after ulceration” was effective for GLM. The final prognosis was good, and TCM appears to offer good benefits in the treatment of this disease.