Bo’s abdominal acupuncture treatment for adult-onset Still's disease: A case report

Abstract

BACKGROUND

Adult-onset Still's disease (AOSD) is a rare autoinflammatory disease characterized by nonspecific symptoms such as fever, rash, sore throat and arthralgia. This paper reports a clinical case of AOSD successfully treated with Bo’s abdominal acupuncture (BAA).

CASE SUMMARY

We report a 20-year-old man who suffered from cold exposure, presenting with high fever, rash, sore throat, arthralgia, and elevated erythrocyte sedimentation rate, leukocytosis with neutrophilic predominance, elevated ferritin, elevated C-reactive protein, and negative rheumatoid factors. He was diagnosed with AOSD based on the Yamaguchi criteria. After treatment with traditional Chinese medicine (TCM) decoction and prednisone acetate tablets, there was some alleviation of sore throat, joint and muscle pain, and fever, but he still had persistent low-grade fever, rash, sore throat and arthralgia. He went to the TCM acupuncture outpatient department to receive BAA. Abdominal acupoints Zhongwan (CV12), Xiawan (CV10), 0.5 cm below Xiawan (CV10), Qihai (CV6), Guanyuan (CV4), bilateral Qixue (KI13), bilateral Huaroumen (ST24), bilateral Shangfengshidian (AB1) and bilateral Daheng (SP15) were selected. After 3 months treatment, all symptoms disappeared, and the laboratory examination returned to normal levels. He did not take glucocorticoids or nonsteroidal anti-inflammatory drugs afterwards, and no relapse was observed during the 3-year follow-up period.

CONCLUSION

BAA can be used as a complementary medical approach for treatment of AOSD.

Key Words: Adult-onset Still’s disease; Bo’s abdominal acupuncture; Traditional Chinese medicine; Complementary medicine; Case report

Core Tip: We report a 20-year-old man who suffered from cold exposure, presenting with high fever, rash, sore throat, arthralgia, and was diagnosed with adult-onset Still's disease based on the Yamaguchi criteria. After treatment with traditional Chinese medicine decoction and prednisone acetate tablets, there was some alleviation of symptoms. He underwent Bo’s abdominal acupuncture, while prednisone acetate tablets were reduced by 0.5 tablets per week. After 3 months treatment, all symptoms disappeared, and laboratory examination returned to normal. He did not take prednisone acetate tablets afterwards, and no relapse was observed during the 3-year follow-up.

INTRODUCTION

Adult-onset Still's disease (AOSD) is a rare systemic inflammatory disease with an unknown etiology, which is characterized by a constellation of clinical manifestations, including fever, salmon-pink rash mainly shown during fever spikes, polyarthralgia and sore throat[1,2]. The incidence of AOSD varies, with rates between 0.16 and 3.9 cases per 100000 individuals. The peak incidence of AOSD is bimodal, occurring between ages 15-25 and 36-46 years[3,4]. The Yamaguchi criteria are considered the most sensitive for diagnosing AOSD, with a sensitivity of approximately 93.5%[5]. The main criteria include joint pain lasting > 2 weeks, body temperature > 39 °C, typical rash, and white blood cell count > 10000/mm³ with a predominance of neutrophils. The secondary diagnostic criteria encompass a sore throat, abnormal liver function, lymphadenopathy and/or the presence of an enlarged spleen, and the absence of rheumatoid factor (RF) and antinuclear antibodies. To establish a diagnosis, a patient must fulfill a minimum of five criteria, including at least two primary criteria[2,6]. The initial treatment for individuals suffering from AOSD typically involves the use of corticosteroids and nonsteroidal anti-inflammatory medications (NSAIDs)[7]. However, these medications are associated with considerable side effects and may not always provide the desired therapeutic response, highlighting the need for complementary and alternative treatment options.

Acupuncture, as an essential component of traditional Chinese medicine (TCM), has a history of > 2000 years in treating human diseases. Bo’s abdominal acupuncture (BAA) is a new technique invented by Zhi-Yun Bo under the guidance of TCM theory. It is characterized by safety, painless application, effectiveness, acupuncture prescription specifications, operating standards and a wide range of indications. In addition, the location of abdominal acupuncture points is ascertained through the use of a ruler for measurement, which enhances the precision of the acupoints and ensures the reliable reproducibility of BAA. In contrast to conventional acupuncture, BAA does not necessitate the patient experiencing the deqi sensation (e.g., distention, numbness, soreness and pain). BAA has been used to treat a variety of systemic diseases through stimulating the abdominal acupoints, including cervical spondylosis, periarthritis of the shoulder, lumbar disc herniation, femoral head necrosis, knee arthritis, ankle sprains, gastrointestinal dysfunction, diabetes, insomnia, stroke, Parkinson's disease, Alzheimer's disease, and pediatric autism[8-14].

In this case report, we present a case of abdominal acupuncture for a 20-year-old man with AOSD due to exposure to cold weather. After 3 months of abdominal acupuncture, all symptoms disappeared, and the laboratory examination returned to normal. He did not take glucocorticoids or NSAIDs afterwards, and no relapse was observed during the 3-year follow-up period.

CASE PRESENTATION

Chief complaints

On April 22, 2021, with the assistance of his family, a 20-year-old man visited the TCM acupuncture outpatient department for the treatment of fever with a body temperature of 38.5 °C, pain in the joints and muscles of the limbs, inability to walk, sore throat, red rashes on the limbs, and weight loss of 10 kg over the course of 1 month.

History of present illness

On March 20, 2021, a 20-year-old man was caught in a dust storm and suffered from cold exposure, resulting in redness, swelling, and pain in the Achilles tendon of his right foot. The next morning, the pain shifted to the Achilles tendon of his left foot, accompanied by a feeling of fatigue upon waking. On the third day, the fatigue intensified, with sore throat and shoulder and knee joint pain. In the evening, a rash appeared on the back of his hand, which disappeared during the day. He visited the hospital and was prescribed a Chinese herbal medicine Lianhua Qingwen capsules for 2 d, but the treatment was ineffective and his condition worsened. The throat pain became unbearable, making swallowing difficult, with migratory pain in the joints and muscles throughout the body, and difficulty bending the knees. By the sixth day, he was unable to walk normally, engage in activities, or swallow, and was unable to take care of himself. On the seventh night, he had a fever reaching 39 °C, which subsided the next morning, but by the evening, he was again experiencing chills and fever reaching 39 °C. He went to the fever clinic for treatment, where antibiotic therapy was administered without effect, and symptomatic treatment with NSAIDs was given.

Based on his symptoms and laboratory examination, he was diagnosed with AOSD, and treated with a TCM decoction and prednisone acetate tablets (5 mg per tablet, two tablets per day). After 1 week of treatment, intermittent fever persisted, but the sore throat and joint and muscle pain was relieved to a certain extent. The dosage of prednisone acetate tablets was increased to four tablets per day, which led to a reduction in fever, although a low-grade fever continued, with episodes of high fever occurring every 2-3 days. Although his joint pain was somewhat alleviated, he was barely able to take care of himself. Because of persistent presence of symptoms the concern of side effects of long-term use of prednisone, he began to seek acupuncture treatment. On April 22, 2021, with the assistance of his family, he went to the TCM acupuncture outpatient department to consult with Zhi-Yun Bo, the inventor of abdominal acupuncture. The main symptoms at that time were fever with a body temperature of 38.5 °C, pain in the joints and muscles of the limbs, inability to walk, sore throat, red rashes on the limbs, and weight loss of 10 kg over the course of 1 month.

History of past illness

A 20-year-old man with no significant past medical history.

Personal and family history

The patient had no remarkable personal or family history.

Physical examination

The patient had high fever, inability to walk by himself, pain in the joints and muscles of the limbs, and red rashes on the limbs.

Laboratory examinations

On March 31, 2021, laboratory investigation showed that white blood cell count was 14.96 × 10⁹/L, neutrophil count 12.57 × 10⁹/L, neutrophil percentage 84%, and platelet count 463 × 10⁹/L. Ferritin was 1500 ng/mL, C-reactive protein (CRP) 119.06 mg/L, erythrocyte sedimentation rate (ESR) 84 mm/h, lymphocyte ratio 8.1%, monocyte count 1.05 × 10⁹/L, IgA 6.37 g/L, IgG 18.1 g/L, albumin 36.6 g/L and globulin 43.3 g/L. RF, anti-streptolysin O, anti-keratin antibody, anti-perinuclear factor antibody, anti-cyclic citrullinated peptide antibody, anti-mutated citrullinated vimentin antibody and anti-rheumatoid arthritis 33 antibody were all negative (Table 1).

Table 1 Laboratory data before and after Bo’s abdominal acupuncture treatment.

Parameters	Before BAA (March 31, 2021)	3 months after BAA (July 13, 2021)	7 months after BAA (February 10, 2022)	Normal range
White blood cells (× 109 /L)	14.96	8.47	6.86	3.5-9.5
Neutrophils (× 109 /L)	12.57	4.92	3.46	1.8-6.3
Neutrophil (%)	84	58.1	50.4	40-75
Monocytes (× 109 /L)	1.05	0.85	0.52	0.1-0.6
Lymphocytes (%)	8.1	28.7	38.6	20-50
Platelets (× 109 /L)	463	309	269	100-300
ESR (mm/h)	84	24	8	0-15
C-reactive protein (mg/L)	119.06	3.11	0.61	0-10
Albumin (g/L)	36.6	48.4	NA	40-55
Globulin (g/L)	43.3	39.7	NA	25-40
Ferritin (ng/mL)	1500	428.7	343.3	30-400
IgA (g/L)	6.37	3.59	4.43	0.82-4.53
IgG (g/L)	18.1	28.13	15.71	6.5-16
RF (IU/mL)	< 20	NA	NA	0-20
ASO (U/mL)	112	NA	NA	0-116
AKA	Negative	NA	NA	Negative
APF	Negative	NA	NA	Negative
Anti-CCP (U/mL)	< 25	NA	NA	0-25
Anti-MCV (U/mL)	3.88	NA	NA	0-20
Anti-RA33 (U/mL)	< 12.5	NA	NA	0-25

ESR: Erythrocyte sedimentation rate; RF: Rheumatoid factor; ASO: Anti-streptolysin O; AKA: Anti-keratin antibody; APF: Anti-perinuclear factor antibody; Anti-CCP: Anti-cyclic citrullinated peptide antibody; Anti-MCV: Anti-mutated citrullinated vimentin antibody; Anti-RA33: Anti-rheumatoid arthritis 33 antibody; NA: Not available.

Imaging examinations

Chest computed tomography showed no significant abnormalities.

FINAL DIAGNOSIS

The patient was diagnosed with AOSD because of sustained fever, rash (Figure 1), arthralgia, sore throat, elevated ESR, elevated leukocytosis with neutrophilic predominance, elevated ferritin, elevated CRP, and negative RF, based on the Yamaguchi criteria[5].

Figure 1 Rash occurring in the adult-onset Still’s disease. A: Rash on the hand; B: Rash on the knee; C: Rash on the ankle.

TREATMENT

Bo ZY first slapped the area around the acupoints Dazhui (GV14), Fengmen (BL12) and Feishu (BL13) on the upper back of the patient's body until the skin turned red. The patient reported a significant reduction in pain at the wrist, elbow joint, upper arm, thigh, and Achilles tendon after the slapping. The sore throat was also notably alleviated.

The patient lay in a supine position and exposed his abdomen. The acupuncturist measured and marked the acupoints on the divine turtle diagram. The determination of the acupoints' positions adhered to the guidelines outlined in the Standardized Manipulations of Acupuncture and Moxibustion, specifically Part 16, which pertains to abdominal acupuncture (Table 2). The selected acupoints are shown in Figure 2. Bo ZY checked the patient's positive reaction symptoms, such as sore throat, red rashes, high fever, and muscle pain in the four limbs. Following standard disinfection procedures, Bo employed acupuncture needles (measuring 0.22 mm in diameter and 30 mm in length, manufactured by Suzhou Hualun Medical Products Co. Ltd.) to puncture the acupoints in the sequence of Zhongwan (CV12), Xiawan (CV10), 0.5 cm below Xiawan (CV10), Qihai (CV6), Guanyuan (CV4), Qixue on the left side (left KI13), Qixue on the right side (right KI13), Huaroumen on the left side (left ST24), Shangfengshidian on the left side (left AB1), Huaroumen on the right side (right ST24), Shangfengshidian on the right side (right AB1), Daheng on the right side (right SP15), and Daheng on the left side (left SP15). The needles were inserted into the subcutaneous area of the above acupoints perpendicularly. Subsequently, the acupuncturist adjusted the depth of the needle insertion based on the degree of pain relief experienced by the patient. Following a 30 minutes period with the needles in place, the acupuncturist extracted them in the order of their insertion. The patient underwent BAA treatment for 3 months, with each individual session lasting 30 minutes. During these sessions, an electromagnetic wave lamp (model CQ-29P Chongqing Aerospace Rocket Electronic Technology Co. Ltd., Chongqing, China) was used for warming specifically on the Shenque (CV8) acupoint, three times per week.

Figure 2 Abdominal divine turtle diagram and selected acupoints. The acupoints marked with red dots represent the selected acupoints for treating the disease.

Table 2 Location of the acupoints selected for treating this case.

Acupoint name	Acupoint location
Zhongwan (CV12)	On the upper abdomen, 4 B-cun superior to the centre of the umbilicus, on the anterior median line
Xiawan (CV10)	On the upper abdomen, 2 B-cun superior to the centre of the umbilicus, on the anterior median line
Qihai (CV6)	On the lower abdomen, 1.5 B-cun inferior to the centre of the umbilicus, on the anterior median line
Guanyuan (CV4)	On the lower abdomen, 3 B-cun inferior to the centre of the umbilicus, on the anterior median line
Qixue (KI13)	On the lower abdomen, 3 B-cun inferior to the centre of the umbilicus, 0.5 B-cun lateral to the anterior median line
Huaroumen (ST24)	On the upper abdomen, 1 B-cun superior to the centre of the umbilicus, 2 B-cun lateral to the anterior median line
Shangfengshidian (AB1)	On the upper abdomen, 1.5 B-cun superior to the centre of the umbilicus, 2.5 B-cun lateral to the anterior median line
Daheng (SP15)	On the upper abdomen, 3.5 B-cun lateral to the centre of the umbilicus

OUTCOME AND FOLLOW-UP

After the first abdominal acupuncture treatment, sore throat disappeared, and pain in the limbs was instantly reduced by > 50%, he was able to walk and dress by himself, and there was no nocturnal fever. Subsequently, abdominal acupuncture was carried out three times a week, and the prednisone acetate tablets were reduced by 0.5 tablets per week, with continuous treatment for 3 months. All symptoms such as fever, sore throat, limb pain, and rash completely disappeared.

On July 13, 2021, laboratory tests indicated that the levels of serum ferritin, white blood cells, ESR and CRP all significantly decreased to near normal levels. On February 10, 2022, re-examination of the laboratory indicators showed that they were all within the normal range (Table 1). After a 3-year follow-up period without taking prednisone acetate tablets, the symptoms did not recur, and he was able to work and live normally.

DISCUSSION

AOSD is a systemic inflammatory condition of unknown origin. Typically, it impacts younger individuals, exhibiting two peak incidence periods, one between the ages of 15 and 25 and the other between 36 and 46 years[2]. A variety of conditions may manifest symptoms similar to AOSD, including fever, arthritis, skin rash, lymphadenopathy, increased levels of acute phase reactants, high white blood cell count, and liver enzyme abnormalities[3]. In this case, the patient was diagnosed with AOSD because of sustained fever, rash, arthralgia, sore throat, elevated ESR, elevated leukocytosis with neutrophilic predominance, elevated ferritin, elevated CRP, and negative RF, based on the Yamaguchi criteria.

In recent years, numerous evidence-based studies and basic research have confirmed that acupuncture can effectively improve the inflammatory status of a variety of inflammatory and chronic diseases. The World Health Organization also recommends the use of acupuncture for the treatment of 16 chronic inflammatory diseases. Jiang et al[15] found that acupuncture exerts anti-inflammatory effects by inhibiting the release of inflammatory cells, inflammasomes and inflammatory cytokines. Xu et al[16] summarized the current status and prospects of the effect patterns and mechanisms of acupuncture in improving chronic inflammatory responses. They found that acupoints related to target organs can regulate the microenvironment of the acupoints to initiate neural regulation. After the acupuncture information is transmitted to the central nervous system, it acts on immune cells through neuro-endocrine-immune pathways, such as the cholinergic anti-inflammatory pathway, the vagus nerve-adrenal medulla-dopamine pathway, somatic sympathetic reflexes, and the hypothalamic-pituitary-adrenal axis. It releases neurotransmitters, hormones, etc., and ultimately acts on immune cells, regulating the intracellular signaling pathways and polarization states of monocytes/macrophages, T cell subsets, and controlling the immune homeostasis of target organs and the body. Tu et al[17] conducted a randomized clinical trial comparing the effects of acupuncture vs sham acupuncture on patients with chronic sciatica due to lumbar disc herniation. The results indicated that patients treated with acupuncture experienced a significant reduction in pain and improvement in function, as measured by the visual analog scale for leg pain and the Oswestry Disability Index. The therapeutic effects in the acupuncture group were shown beginning from the second week and were sustained up to the 52^nd week, with a significant difference compared with the spontaneous remission of the disease, suggesting that acupuncture can serve as an effective therapy, alleviate symptoms, improve the quality of life, and achieve spontaneous remission of diseases.

BAA is a new technique invented by Bo ZY. Based on the theories of viscera and meridians, Bo proposed the Shenque (CV8) regulation system theory. It is believed that during the embryonic stage, nutrients are delivered to the fetus through the umbilical cord. After birth, the original channels for transporting qi and blood still remain. Bo ZY discovered a system in the superficial layer of the abdominal wall that affects the entire body. Acupoints related to various parts of the body can be found in the abdomen. When these abdominal acupoints are connected, they form the divine turtle diagram centered on the navel, which exhibits a homologous relationship with the body in a similar form. Therefore, it has become mainly used for treating chronic and difficult diseases throughout the body by needling abdominal acupoints. By following the abdominal acupuncture operation standards and stimulating the corresponding acupoints in the superficial layer of the abdominal wall, rapid therapeutic effects can be achieved. In clinical practice, many patients vividly describe that after the acupuncture, there is no needle sensation in the abdomen, but the pain disappears as if pressing an electric button[13,18].

BAA as a complementary and alternative therapy has been widely used in the treatment of systemic diseases, such as knee osteoarthritis, shoulder–hand syndrome, fatigue, complex regional pain syndrome, neck pain, post-stroke motor dysfunction, Parkinson’s disease, post-stroke depression, cervical spondylosis, insomnia, and sequelae of cerebrovascular disease[19-27]. It has the characteristics of simple operation, standardized formulation, accurate acupoint location, safety, low pain, efficient, wide applicability, easy acceptance, and few side effects.

Bo ZY considered AOSD to be a type of hypersensitivity immune response disorder, which has a relationship with pulmonary infection, thus manifesting symptoms such as fever and rash. Therefore, the basic principle of treatment is to detoxify and clear heat and improve lung function. The treatment methods include patting the skin of areas such as the Dazhui (GV14), Fengmen (BL12), and Feishu (BL13) acupoints on the back, as well as abdominal acupuncture prescriptions such as the detoxification and heat-clearing formula which includes Zhongwan (CV12), Xiawan (CV10), 0.5 cm below Xiawan (CV10), Shangfengshidian on the left side (left AB1) and Shangfengshidian on the right side (right AB1). The acupoints Qihai (CV6), Guanyuan (CV4) and Qixue (KI13) were used to strengthen the body's resistance and dispel pathogenic factors, playing a role in reducing fever. AOSD was considered a rare autoimmune disease characterized by pain in multiple joints throughout the body. Bo associated it with spleen deficiency. In TCM, it is said that the spleen governs the muscles and limbs, and when the spleen is deficient, the limbs and muscles do not receive proper nourishment. In treatment, the acupoints Daheng (SP15) were chosen to regulate the spleen, eliminate dampness, and lubricate the joints, thus enhancing immunity. When combined with Shangfengshidian (AB1) and Xiafengshidian (AB4), arthritis throughout the body can be treated.

After 3 months of abdominal acupuncture, three times a week for 30 minutes each session, high fever, rash, sore throat, arthralgia and fatigue completely disappeared, and the laboratory test results returned to normal. After a 3-year follow-up, there was no recurrence. This case is the first clinical report of using abdominal acupuncture to treat AOSD, and the therapeutic effect of abdominal acupuncture was significant. Although the mechanism of abdominal acupuncture in treating AOSD is not yet fully understood, based on the significant therapeutic effects of acupuncture in regulating chronic inflammatory diseases, the mechanism may be related to its involvement in modulating immune-inflammatory responses.

CONCLUSION

AOSD is an uncommon and complex systemic inflammatory disease characterized by a range of clinical manifestations, which can lead to a delayed diagnosis. It can cause damage to multiple organs such as the skin, joints, muscles, and internal organs, leading to a widespread inflammatory response that severely affects normal life and mobility. Long-term use of corticosteroid drugs can have significant side effects. This case is the first reported instance where acupuncture was used to cure AOSD. Although the regulatory mechanism was not clear, from the perspective of TCM that seeks to treat the root cause, it improved the patient's symptoms, normalized the indicators, and the therapeutic effect was definite, with minimal side effects and a high level of patient acceptance. It is a complementary therapy worth promoting and applying in the clinical treatment of this disease.

ACKNOWLEDGEMENTS

The authors extend their appreciation to the patient for his support.