Persistent bilateral breast pain treated with traditional Kampo medicine

Abstract

A 45‐year‐old woman presented with persistent bilateral breast and axillary pain lasting for more than 3 months. Lengthy work‐up failed to identify the etiology. Physical examination was entirely normal, including breast examination. A traditional Kampo medicine, Goshaku‐san, was tried, and the symptoms began to improve gradually. This case is characterized by persistent regional pain syndrome mainly on her breast yet not accompanied by other symptoms, and we would like to propose the current case as a novel unique disease entity. We also discuss the potential benefit of Kampo medicine for the symptoms.

INTRODUCTION

Breast pain, or mastalgia, is a common presenting symptom in a primary care setting or others. Over two-thirds of women will experience breast pain in their lifetime.^1,2 Despite its commonness, breast pain is believed to be under-reported, partly due to the reluctance of both patients to disclose it and healthcare workers to ask about.³ Cyclical breast pain is more common than persistent breast pain. Cyclical breast pain is usually associated with the menstrual cycle and can be unilateral or bilateral, and it is also referred to as “physiological.”¹ On the contrary, persistent breast pain could be associated with malignancy although its incidence is low.⁴ Persistent breast pain is more likely to be unilateral and often related to breast or chest wall lesions.⁵

We here present a case of uncommon presentation of bilateral persistent breast pain not associated with structural or inflammatory problems, treated with traditional Kampo medicine.

CASE REPORT

A 45-year-old Japanese woman without significant past medical history was referred to our Kampo clinic for persistent bilateral breast and axillary pain, lasting for more than 3 months. Nine months before the initial visit, she developed amenorrhea with fatigue and visited a gynecologist a month later. Hormonal work-up was reportedly normal but she received hormone replacement therapy (HRT). Her period restarted and fatigue disappeared. However, she started to have bilateral axillary pain 1 month after HRT, and the pain spread to her bilateral breasts area several months later. She discontinued HRT but the pain persisted. She visited a breast surgeon and mammography as well as breast ultrasound examination were both normal. She then visited a general internal medicine clinic.

She denied other associated symptoms such as fever, weight loss, insomnia, anxiety, depressed mood, or strong concerns about the symptoms, including those associated with potential malignancies. She was able to eat, work, and sleep well even though she was annoyed by the pain.

Lengthy work-up did not reveal the etiology, and she was referred to us by the general internist.

She stated that the pain is not associated with her movement. She does not wake up with pain and sleeps well. She stated the pain is like “blood flow stopped there,” and sometimes radiated to her back. The pain increased while she wore the bra but removing it did not remove her pain. She denied the increased size of her breasts and stated her bra size remained C. She also stated that she has an intolerance to cold temperatures.

On physical examination, she was not in agony and appeared well. Her vital signs were normal. Physical examination was entirely normal including neurological examination. Palpation of both axillary areas failed to detect enlarged lymph nodes or any other abnormalities. Breast examination accompanied by a chaperon also failed to find any abnormalities, and palpation did not induce any pain. There were no mass lesions or inflammatory process on both breasts and chest wall. Her breasts were not excessively large nor pendulous, but we did not measure her breast size. There was no skin lesion including the area under her breasts. There were no bony or joint abnormalities on her chest wall, and we found no thrombus formation both by visual inspection and palpation.

The laboratory and imaging tests performed at previous facilities are the following. Blood tests including complete blood count, electrolytes, liver and kidney function test, erythrocyte sedimentation rate, C-reactive protein, folate, vitamin B1 level, thyroid-stimulating hormone, free thyroxine level, and other hormones such as follicular stimulating hormone, luteinizing hormone, prolactin, and estradiol were all normal. Work-up for autoimmune diseases conducted by a referred neurologist such as antinuclear antibody, anti-SS-A, B, PR3-ANCA, and MPO-ANCA were all normal. Other tests including electrocardiogram, electromyogram, magnetic resolution imaging of breast and spine, and repeated computed tomography scan of the chest were also normal.

We presumptively assessed that she is not likely to suffer from malignancy, inflammatory diseases, or endocrine abnormality and judged that she is more likely to have an idiopathic medically unexplainable illness, according to history taking, physical examination, and all other tests performed previously. Also, no apparent psychiatric illnesses were suspected. We offered one of the traditional Kampo medicine Goshaku-san 2.5 g TID orally and the symptoms improved over several weeks. She has been seen at outpatient clinic on the regular basis for several months and did not have recurrence of symptoms as of this writing.

DISCUSSION

We judged her bilateral persistent breast pain as idiopathic medically unexplained symptoms (MUS). MUS is a heterogeneous group of conditions characterized by persistent symptoms that cannot be explained by medical illnesses or injury.⁶ Somatic symptoms disorder (SSD) can present with symptoms interpreted as MUS, entailing conditions formerly known as somatization disorder, undifferentiated somatoform disorder, hypochondriasis, and pain disorder. However, the current patient did not have psychological or behavioral symptoms such as persistent thoughts about the seriousness of the symptoms, severe anxiety about the symptoms, and excessive time and energy devoted to the illness, making this diagnosis unlikely.⁷ Even though her condition was not categorized as SSD, the pain is not likely to be associated with medical conditions, such as inflammatory diseases including infections, malignancies, endocrine disorders, neurological disorders, or others, since the symptoms are persistent, bilateral, without having any abnormality on physical examination, blood tests, and imaging studies. Therefore, we would like to propose the current condition as idiopathic bilateral persistent breast pain, yet not categorized into SSD.

Goshaku-san (Ojeok-san in Korean and Wuji-san in Chinese) is a combination of traditional Chinese herbs, which is referred to as “Kampo” in Japanese. It was invented in the 12th century during the Sung dynasty of China.⁸ It is composed of the following crude drugs (ones used in Japan); Atractylodes Lancea Rhizome, Citrus Unshiu Peel, Japanese Angelica Root, Pinellia Tuber, Poria Sclerotium, Glycyrrhiza, Platycodon Root, Immature Orange, Cinnamon Bark, Magnolia Bark, Peony Root, Ginger, Cnidium Rhizome, Jujube, Angelica Dahurica Root, and Ephedra Herb. It is indicated for the relief of the following symptoms that take a chronic course, including gastroenteritis, low-back pain, neuralgia, arthralgia, menalgia, headache, oversensitivity to cold, climacteric disturbance, and common cold.⁹ Goshaku-san is often used for the treatment of bodily ache especially ones accompanied by cold sensation, but few report the effectiveness for breast pain. We were able to find only an abstract of an academic meeting, reporting the effectiveness of Goshaku-san for breast pain although the authors considered the cause of pain is mastitis.¹⁰ Further studies will be needed for the use of this medicine in persistent breast pain.

In conclusion, we present a case of rather unusual persistent, bilateral breast pain not likely to be medically explainable. This could potentially be a novel clinical entity previously unreported. Traditional Kampo medicine such as Goshaku-san could be an option in treating such illnesses.

ACKNOWLEDGEMENTS

None.

CONFLICT OF INTEREST

The other authors have stated explicitly that there are no conflicts of interest in connection with this article.

INFORMED CONSENT

The patient provided a written informed consent to present the case.