Traditional Chinese medicine treatments in a woman with a recurrent peritoneal inclusion cyst: a case report

Keywords:

• Peritoneal inclusion cysts
• recurrent
• traditional Chinese medicine
• treatments
• lifestyle management
• case report

Introduction

A peritoneal inclusion cyst (PIC) is a rare lesion defined as an aggregate mass of variably sized, fluid-filled mesothelial-lined cysts in the pelvis, upper abdomen and retroperitoneum (Lee et al. 2012). This unusual but largely benign mass has a distinct sonographic appearance, and it has also been referred to as ‘benign (multi) cystic peritoneal mesotheliomas’ and ‘inflammatory cysts of the peritoneum’ (Jones et al. 2003, Baker et al. 2014). Since the first description in 1979 by Mennemeyer and Smith, approximately 200 cases have been reported in the literature (Mennemeyer and Smith 1979, Rapisarda et al. 2018). Although the pathogenesis of PICs is not well understood, they are thought to arise secondary to intra-abdominal inflammation and subsequent cyst formation with serous fluid derived from the ovarian stroma (Lee et al. 2012). The cysts usually develop in women of reproductive age who have a history of previous pelvic surgery or pelvic inflammatory disease. In one study, a history of insult to the peritoneum was found in 70.6% of cases (Chua et al. 2020). Patients with PICs frequently present with lower abdominal pain, pelvic fullness and/or a palpable mass.

There is no established treatment for PICs. Management varies and may involve observation with serial imaging, hormones, ultrasound-guided needle aspiration with simultaneous injection of antimicrobial agents, surgical excision or complete resection. The gold standard treatment is complete resection by either laparoscopy or laparotomy. However, PICs recur after complete resection in 30–50% of patients (Singh et al. 2015, Tamai et al. 2019). As PICs mainly affect women of reproductive age, treatment is often very aggressive and has adverse effects, frequently with important repercussions on fertility (Rapisarda et al. 2018). For these reasons, many patients with PICs are more willing to rely on complementary and alternative medicine.

According to traditional Chinese medicine (TCM), in patients with deficiency of positive qi and dysfunction of the internal organs, external and internal evils interact and accumulate in the small abdomen, eventually forming a PIC. Studies have shown that the use of TCM for treatment of PICs is safe and effective (Bao 2009, He et al. 2020).

This report describes a challenging case involving a patient with a recurrent PIC who underwent TCM treatments involving an herbal enema, topical application of Si Huang San (comprising coptis, phellodendri, Scutellaria baicalensis and Rhei Radix et Rhizoma ground into a powder and mixed with honey and water to make a paste), and Fire Dragon Jar (a combination of moxibustion, ironing, scraping therapy and other comprehensive action on the meridian acupoints) along with lifestyle management. After these treatments, the severity of symptoms and size of the PIC decreased.

Case report

Chief complaints

A 43-year-old woman was admitted to the Department of Gynaecology, The Second Affiliated Hospital of Guangzhou University of Chinese Medicine on 16 October 2020, with chief persistent left lower abdominal pain.

History of present illness

The patient was a 43-year-old female undergraduate student and medical staff member from Xinjiang Uyghur. She was unmarried, nulliparous and sexually active, and she wished to remain fertile. Patient’s symptoms started two days ago with recurrent episodes of left lower abdominal pain.

History of past illness

She had undergone laparoscopic right ovarian cyst removal in 2013; appendectomy for appendicitis in 2018; radiofrequency ablation for frequent premature ventricular contractions in 2019; and open pelvic adhesiolysis, uterine fibroid removal and sigmoid surface removal of a uterine multifocal smooth muscle tumour in January 2020. Six months later, follow-up gynaecological ultrasonography showed a 97-×79-×82-mm PIC on the left side, which was treated with antibiotics. Outpatient re-examination in September 2020 showed that the pelvic encapsulated effusion on the left side had increased to 92 × 83 × 91 mm. However, it improved after inpatient antibiotic and rehydration therapy. On 9 October 2020, gynaecological ultrasonography showed a 65-×37-×58-mm PIC on the left side. On 16 October 2020, the patient presented with a two-day history of pain in the left lower abdomen with no obvious cause, and gynaecological ultrasonography indicated recurrence of the PIC (73 × 49 × 59 mm).

Physical examination

The whole abdomen is soft, with tenderness and rebound pain in the lower abdomen; sterilised gynaecological examination: normal vulva, unobstructed vagina, cervix often large, smooth, anterior uterus, such as 2 months of pregnancy, medium quality, poor activity and light tenderness. The left appendage touched the cystic mass, with poor activity and light tenderness, and the right appendage area was not abnormal.

Laboratory examinations

Routine laboratory tests revealed no remarkable abnormality in the patient.

Imaging examinations

Gynaecological ultrasonography indicated recurrence of the PIC (73 × 49 × 59 mm).

Final diagnosis and outcome evaluation

The PIC was diagnosed by a combination of the patient’s clinical history, ultrasound imaging and CA-125 concentration. The patient had no abnormal tumour markers; we thus diagnosed the PIC through the recurrence of pain, the lesion site on ultrasound, and the history of pelvic surgery. The patient had undergone multiple surgeries, resulting in a healthy Qi deficiency. Additionally, the patient worked in northwest China for a long time. With large temperature difference between day and night in this region, cold-dampness pathogen hindered the excretion of heat pathogen, which also led to the weakness of the healthy Qi. As a result, the patient presented with slightly darker tongue body, white and slimy fur, and sunken and tense pulse. Significantly, this cyst was caused by heat depression due to a combined pathogen of wind, cold and dampness. Therefore, we used Si Huang San, a medicine used to stop the development and expansion of the heat pathogen, and gave the patient TCM in the form of herbal enemas to discharge heat and activate blood while achieving internal and external treatment.

A numerical rating scale was used to assess the left lower abdominal pain (Fan and Wang 2019). The scale consisted of 11 items (0–10 point for each item), with higher scores indicating more severe pain. Ultrasound examination was also used to assess changes in PIC severity. TCM pattern diagnosis was performed based on the patient’s body shape, ordinary symptoms, pulse diagnosis and tongue diagnosis.

Treatment and course of symptoms

The patient underwent treatment as an inpatient for 19 days. During hospitalisation, she was administered compound Mao Dongqing (comprising Ilicis Pubescentis Radix et Caulis, Curcuma phaeocaulis, Astragali Radix and Rhei Radix et Rhizoma) as an enema to activate the blood circulation, remove blood stasis and clear the meridians; 200 mL of solution was administered and retained for 2–3 hours once daily (Yuan et al. 2013). She was also treated with a twice-daily topical application of warm Si Huang san.

From 1 to 3 November 2020, the patient was also treated with Fire Dragon Jar (Figure 1), which combines gua sha (a therapy involving the scraping of instruments dipped in a specific medium on the body surface), moxibustion and ironing. This treatment was performed on the lumbosacral region from DU1 (Changqiang) to DU3 (Yaoyangguan) and in the area around the UB31, UB32, UB33 and UB34 (Baliao) points for 15 minutes every day.

Figure 1. Fire Dragon Jar. (A) Picture of Fire Dragon Jar. (B) The nurse does Fire Dragon Jar to the patient.

Outcome and follow-up

The treatment resulted in substantial improvements in the patient’s symptoms, and she was discharged on 3 November 2020. Gynaecological ultrasonography performed on the day of discharge showed that the size of the PIC had decreased to 26 × 13 × 16 mm (Table 1). Her numerical rating scale score had also decreased from 6 to 0 (Table 2).

Table 1. Changes in size of peritoneal inclusion cyst on ultrasonography.

Date	Size (mm)	Change (mm)
2020.7.11	97 × 79×82
2020.9.24	92×83×91	5↓×4↑×9↑
2020.10.9	65×37×58	27↓×46↓×33↓
2020.10.15	73×49×59	8↑×12↑×1↑
2020.10.24	33×26×62	40↓×23↓×3↑
2020.11.3	26×13×16	7↓×13↓×46↓

Table 2. Changes in pain scores over time.

	2020.10.16	2020.10.30	2020.11.3
Left lower abdominal pain (NRS-10) score	6 (moderate pain)	4	0 (no)

The patient was discharged with an individualised care plan. She was advised in lifestyle management involving the intake of warm foods that benefit the spleen and promote the circulation of qi and blood (such as stewed pig’s feet with Astragali Radix, yam porridge and black sesame) and the avoidance of spicy and aromatic foods, raw and cold fruits, and cold foods. The patient was also taught eight pelvic exercises with abdominal breathing. This whole-body coordinated exercise regimen strengthens the pelvic ligaments, muscles and blood vessels, which accelerates blood circulation, reduces venous and pelvic stasis in the lower limbs, and promotes the absorption of local stasis (Zhong et al. 2007, Pan et al. 2015). The patient was advised to perform these exercises once in the morning and evening for about 15–20 minutes each time.

After hospital discharge, the patient was followed up via a social messaging smartphone application on 6 November, 1 December and 31 December 2020. The patient reported that she had no abdominal pain and that her condition was controlled and stable. She regained confidence regarding her physical health and mental attitude.

Discussion

The aetiology and nature of PICs are not well understood and are being continuously debated. However, most scholars believe that PICs arise secondary to intra-abdominal inflammation and subsequent cyst formation, with serous fluid derived from the ovarian stroma (Vallerie et al. 2009). One risk factor is previous intraperitoneal surgery performed 6 months to 20 years earlier by any route. The cyst may range in size from 1 to 30 cm (Vallerie et al. 2009, Singh et al. 2015). PICs occur more frequently in women of reproductive age. Although pelvic pain and a palpable abdominal mass are the most common complaints, atypical presentations such as back pain, dyspareunia, early satiety, constipation, urinary frequency, uterine bleeding and infertility have been described. However, many patients are completely asymptomatic, and up to 10% of PICs are discovered incidentally at the time of imaging or surgery (Ho-Fung et al. 2011). The treatment options for PICs range from observation to complete resection. There is no standard algorithm by which affected patients are evaluated, treated or followed up. Prior studies have suggested that the cure is only accomplished with surgical resection; however, patients are at a 50% risk of recurrence. Some scholars have suggested that the goal for such a chronic disease should not be cured but instead symptomatic relief through individualisation of treatment (Vallerie et al. 2009, Rapisarda et al. 2018). This is particularly important because PICs occur in women of reproductive age, for whom fertility considerations must be part of the discussion of any treatment.

Given the low incidence yet high recurrence rate of PICs, it is likely that recurrence reflects remnant cystic tissue that forms the basis of recurrence rather than a discreet de novo pathogenic process (Trehan and Trehan 2014). In the present case, the patient had a history of pelvic surgery. We surmise that her medical history of pelvic inflammatory disease may have led to the formation of the PIC. PICs are considered to form by a benign inflammatory process (Kanasugi et al. 2013). Our patient had been treated with antibiotics many times before, and uncontrolled inflammation may have therefore led to her recurrence. Surgery is invasive and has a high recurrence rate, and long-term administration of broad-spectrum antimicrobial agents leads to an increased risk of resistant bacteria. Patients may refuse surgery or antibiotics because of fear or other factors of their individual situation. Our patient’s lesion recurred, so she chose TCM.

In the present case, TCM was found to be effective for a woman with a PIC who chose conservative management. To the best of our knowledge, this is the first case report of a significant rehabilitation effect with TCM integrative treatments and lifestyle management in a patient with PIC. TCM has few adverse effects. This is especially helpful when the patient wishes to become pregnant. Our patient received in-patient treatment consisting of a compound Mao Dongqing enema and topical application of Si Huang san on the lower abdomen on a daily basis. The four constitutive herbs of compound Mao Dongqing are as follows. (1) Ilicis Pubescentis Radix has the function of detumescence and pain relief, activating blood circulation and dredging collaterals. It can also dilate the peripheral blood vessels, which is closely related to the concept of anti-inflammation and immunity of modern medicine. (2) Curcuma phaeocaulis contains volatile oils and curcuminoid, polysaccharides, sterols, phenolic acids and alkaloids. Modern pharmacological studies have shown that it has anti-tumour, anti-inflammatory, antithrombotic, blood lipid regulatory, analgesic, antibacterial, antiviral and other pharmacological effects. Thus, it is effective in removing coagulated blood and body fluid condensation. (3) Astragali Radix contains polysaccharides, saponins, flavonoids and other components, which improve haemodynamics by dilating vessels and improving microvascular function. It has the function of invigorating qi and promoting blood circulation. (4) Rhei Radix et Rhizoma is mainly composed of emodin, which can destroy the permeability of the bacterial cell membrane and inhibit the synthesis of protein in bacteria, thus playing a bactericidal role. Rectal drug delivery provides analgesia, relieves spasm, improves local pelvic blood circulation and promotes the absorption and regression of local inflammation (Yuan et al. 2013). Si Huang san is used to clear heat and dryness, activate blood and relieve pain in traditional medicine; additionally, it showed anti-inflammatory and analgesic effects in a clinical study (Wen and Zhang 2015). In the present case, the main symptom of abdominal pain improved during inpatient treatment.

Our patient’s symptom differentiation was that heat accumulated in the body and failed to be discharged because of cold pathogen; therefore, both internal and external treatment was performed. According to this characterisation, Fire Dragon Jar was used to treat the patient. Moxibustion improves chronic pathological conditions associated with deficiency and coldness. Directly scraping the skin and stimulating the acupoints and meridians achieves the purpose of regulating the viscera. Ironing speeds up local blood circulation. UB31, UB32, UB33 and UB34 (Baliao) were used for Fire Dragon Jar treatment; these acupoints are used to promote blood circulation and to reduce female genital and lower abdominal pain.

The size of the PIC was confirmed by ultrasonography. The therapeutic effects on PICs as evaluated by ultrasonography have already been studied (Mehta et al. 2017). In the present case, the size of the PIC was 73 × 49 × 59 mm on 15 October 2020 (Table 1). The treatments started on 16 October 2020. On 3 November 2020 (after 19 days of treatment), the last examination showed that the size of the PIC was 26 × 13 × 16 mm. After treatment, the length decreased by 47 mm, the width decreased by 36 mm and the height decreased by 43 mm (Table 1). These reductions were considered significant changes.

After the patient was discharged from the hospital, we performed two types of continuous care interventions via a social messaging smartphone application and outpatient follow-up. Case management was used to follow the whole process under the supervision of the physician, nurse and the patient’s family. According to the patient’s syndrome, she was instructed to ensure solid protection of vital qi, be careful in daily life, and perform eight pelvic exercises with abdominal breathing to restore the balance of water and fluid metabolism. This helped to avoid endogenous water accumulation, which significantly reduced the risk of disease recurrence, and the treatment effect was satisfactory.

This study has two main limitations. First, this is a case report of only a single patient; thus, more cases are needed to confirm the effect of TCM treatment. Second, which part or parts of the intervention account for the patient’s rapid improvement remains unclear. This could include, for example, effects of the TCM treatments, the natural self-limited progression of the disease, or lifestyle modifications. In our clinical opinion, this is a tolerable limitation to creating effective personalised therapies for an often incurable complex chronic medical syndrome.

In summary, this case report supports the use of TCM treatment for conservative management of PICs in women. We expect that this report can provide a basis for large systematic studies, which are needed to confirm these results.

Author contributions

The study was designed by Qing-Hua Guo and Xiao-Zhen Gong. The therapeutic Intervention was made by Jing-Ling He. The manuscript writing was performed by Wen-Fang He and Guang-Lian He. The manuscript was finalised by Qing-Hua Guo and Guang-Lian He. All authors have read and revised the manuscript critically.

Ethical approval

Ethics approval was waived as this was a case report.

Consent form

Informed written consent was obtained from the patient for publication of this report and any accompanying images.

Acknowledgements

The authors would like to thank the patient for providing informed consent for this treatment and for allowing the publication of this case report. We also thank Kelly Zammit, BVSc and Angela Morben, DVM, ELS, from Liwen Bianji (Edanz) (www.liwenbianji.cn/) for editing the English text of a draft of this manuscript.

CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).

Disclosure statement

The authors declare that they have no conflict of interest.

Additional information

Funding

This study was not specifically funded by any funding agency from the public, commercial, or non-profit sectors.