熟地黃苦蔘湯과 海浮散 습윤드레싱을 병행한 한방치료를 통한 샤르코 신경관절병증 환자 호전 1례

I. Introduction

Diabetic foot ulcer, also known as Charcot neuroarthropathy(CN), is a sensorial neuropathy, defined as a non-infectious and progressive invasion and destruction of bones, joints, soft tissues¹⁾. CN is a result of peripheral neuropathies from diabetes, autonomic neuropathy, infections such as leprosy and HIV, drinking alcohol, tabes dorsalis, syringomyelia, rheumatoid arthritis, multiple sclerosis, traumatic damage, or abnormal bone metabolism²⁾. CN easily occur on midfoot, causing subluxation, dislocation, fracture and deformation of surrounding.

Diabetes is the most prominent cause of CN, of which annual incident rate is about 0.1-29% worldwide. About 0.08-13% of world population is diabetic patients³⁾, For diabetic patients, CN is a long-term complication which damages foot and foot joints, resulting dislocation, instability, joint deformation, insensibility, joint alignment destruction, progressive fracture, and multiple joint dislocation. Ulceration may develop to myelitis and at worst amputation would be necessary¹⁾.

In Korean medicine, CN can be comprehended in categories of wasting-thirst(消渴) and digital gangrene(脫疽). Dongeuibogam(東醫寶鑑) mentions digital gangrene is caused by accumulated toxin due to excessive indulgence in greasy and sweet food(膏粱珍味), sexual overindulgence(房勞), drinking alcohol(酒毒), or developed as a long-term complication of wasting-thirst⁴⁾. Lingshu(靈樞) contains prognosis information of digital gangrene based on color and morphology, and mentions amputation surgery procedures when it threats patient’s life. Dongeuisoosebowon(東醫壽世保元) classifies types of wasting-thirst, its complication patterns, and treatments based on constitutional type^5-7).

There was a case report of 5 patients all with different disease or symptom, but same as Soyang(lesser yang person) type gangrene according to pattern identification(辨證), treated with same herbal prescription, Soyang type Liuweidihuang-tang(六味地黃湯)⁶⁾. However, there is no study of CN treatment with Korean medicine yet.

This study reports a case of CN as a complication of long-term diabetes improved by combined Korean medical treatment including Shudihuangkushen-tang(熟地黃苦蔘湯) and Haifu-san(海浮散) wet dressing.

II. Objects & Methods

III. Case report

Fig. 1. X-ray image of AP (Fig. 1-A), lateral (Fig. 1-B), oblique (Fig. 1-C) view of the patient’s left foot on May 20, 2019. Findings include partial amputation state of 1_st metatarsal and phalanx, bony resorption and destroyed state of 2_nd distal metatarsal bone.

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Fig. 2. MRI T2 image of sagittal (Fig. 2-A), coronal (Fig. 2-B), axial (Fig. 2-C) section view of the patient’s left foot on May 20, 2019. Findings include abscess in dorsum of the left foot, mainly 2_nd, 3_rd, 4_th proximal phalanx area, which looks similar to osteomyelitis in 1_st, 2_nd, 3_rd metatarsal, 2_nd, 3_rd proximal phalanx of toes, fracture in 3_rd metatarsal head.

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Fig. 3. Change of average blood sugar level

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Fig. 4. Lateral view X-ray image of the patient’s left foot on July 11, 2019, showing nearly complete improved state of soft tissue swelling of the diabetic foot.

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Fig. 5. Progress on the patient’s left foot. (A) May 18, (B) June 5, (C) June 10, (D) June 30, (E) July 8, (F) July 12.

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IV. Discussion

Charcot neuroarthropathy is a sensorial neuropathy involving a non-infectious and progressive invasion and destruction of bones, joints, soft tissues¹⁾. CN easily occur on midfoot area as a result of peripheral neuropathies from diabetes, autonomic neuropathy, infections such as leprosy and HIV, drinking alcohol, tabes dorsalis, syringomyelia, rheumatoid arthritis, multiple sclerosis, traumatic damage, abnormal bone metabolism²⁾, causing subluxation, dislocation, fracture and deformity of surrounding.

Main cause of CN is diabetes. About 9.3% of American population, 0.08-13% of world population have diabetes, and its annual incident rate is 0.1-29% worldwide³⁾. According to domestic statistics from National health insurance in Republic of Korea, the number of diabetic patients increased by 24.6%, from 2.02 million in 2010 to 2.52 million in 2015⁹⁾. Among diabetic patients, prevalence rate of CN varies from 0.08 to 7.55%, but it seems underestimated because of misdiagnosis, such as goat, deep vein thrombosis, soft tissue damage, rheumatoid arthritis, and infection^10-12), which delays time to get proper treatment and brings severe deformation of affected area¹⁾.

For diabetic patients, CN is a long-term complication which invades foot and foot joints, causing dislocation, instability, and joint deformation, and as sensorial neuropathy gradually proceeds, sensation becomes numb. Inappropriate treatment after loss of joint alignment or traumatic damage may cause progressive fracture or multiple joint dislocation. When ulcer develops to osteomyelitis, in severe cases, amputation would be required¹⁾. Statistically about 50% of CN patients get at least one time surgery, and ulcer or inflammation are common complications. CN, for diabetic patients, is a major risk factor for below-the-knee amputation³⁾.

Although the pathophysiology of CN is not yet fully discovered, neurotraumatic theory and neurovascular theory generally explain the mechanism of CN. According to neurotraumatic theory, as distal area becomes gradually insensitive and less aware of external stimuli, repetitive small damage causes successive reaction of infection, finally developing to CN. Neurovascular theory says that blood flow to distal extremities increases because of sympathetic nerve abnormality, resulting blockage of blood vessels, fever, swelling, bone absorption and osteolysis consequently, which leads to fracture even by small amount of load^3,13). Combination theory is suggested based on two theories above, which explains that inflammatory cytokines promote inflammation and activate osteoclasts¹⁴⁾. Osteolysis causes pain, and pain makes patients rest and apply immobilization which slows and limits inflammatory reactions. CN patients are insensitive to pain, and therefore cannot be helped by immobilization, leading to osteoclasia and osteolysis^1,3,15). As it moves to chronic stage, deformation, dislocation in instability, and bone protrusion which may lead to superficial or deep tissue ulcer and osteomyelitis¹⁶⁾.

Diagnosis of CN is made based on medical history and physical examination, referring to laboratory investigations and radiograph³⁾. Typical symptoms of CN are swelling, instability, painless sensation, but in some cases might feel somewhat pain¹⁾. Patients usually show warm, swollen, erythematous foot or ankle. Physicians have to check if neuropathy affects the other side¹⁾. Differential diagnosis of foot infections is necessary, such as cellulitis, abscess, osteomyelitis which also show edema, redness, and heat, similar to CN. CN patients do not present with systemic fever, increase of white blood cell, erythrocyte sedimentation rate, C-peptide, glucose, which implies systemic infection. If there is no external wound, it would help to diagnose neuropathy, but infection and neuropathy could be simultaneous. If edema and redness lessen as the leg is raised for two minutes, it could help to rule out infection^1,2,13).

In early stage, CN is treated conservatively, such as disinfection of ulcer area, application of total contact cast for immobilization and off-loading of the affected extremities, or pharmacological management to control bone metabolism. If conservative treatment fails, surgical treatment would be necessary such as debridement, soft tissue transplantation, exostectomy, arthrodesis, or amputation. In treating CN, debridement is the most important, which is removing dead tissue from infected area, such as callus, bone, tendon, cartilage, especially bone and tissue with osteomyelitis. The common goal of treatment methods is to obtain a stable and plantigrade shoe wearable foot, preventing recurrence of ulceration, minimizing foot deformation, and helping to progress into bone remodeling stage³⁾.

CN can be comprehended in categories of wasting-thirst and digital gangrene. Dongeuibogam mentions digital gangrene is caused by accumulated toxin due to excessive indulgence in greasy and sweet food, sexual overindulgence, drinking alcohol, or developed as a long-term complication of wasting-thirst⁴⁾. Lingshu contains information about prognosis of digital gangrene based on color and morphology, and mentions amputation surgery procedures when it worsens and threats patient’s life. Dongeuisoosebowon classifies types of wasting-thirst, its complication patterns, and treatments based on constitutional type^5-7), Soyeum(lesser yin person) type Shikso(食消), Soyang type upper, middle, lower wasting-thirst(上·中·下消), Taeyeum(greater yin person) type Iyanggeolwijiso(二陽結謂之消), and complications of wasting-thirst such as Soyeum type near brest carbuncle(乳傍近脇 發內癰), sore covered all over the body(滿身有瘡), back carbuncle(背癰), Soyang type carbuncle(癰疽), eye diseases(眼病), poisonous gall on shoulder(肩上有毒瘇), nuchal gangrene(腦疽), sore on fingertip(蛇頭瘡), lymphatic infection(連珠痰), Taeyeum type fingertip black sore disease(手指焦黑癍瘡病).

There was a case report of 5 different patients, all Soyang type gangrene based on pattern identification, treated with same herbal prescription, lesser yang person type Liuweidihuang-tang. However, there is no study of CN treatment with Korean medicine yet.

This study reports a case of CN as a complication of long-term diabetes improved by Korean medical treatment combined with herbal medicine, Shudihuangkushen-tang, Haifu-san wet dressing, acupuncture, and herbal acupuncture.

A 59-year-old male patient was diagnosed with CN on April in 2004, and had amputation surgery on distal interphalangeal joint of the left foot first toe. Ulceration gradually recurred on the left foot in 2018. After self management failed, the patient visited and started Korean medical treatment on May 18 in 2019. Edema, redness, heat was observed on the left foot, and he felt painless on the first, second, and third toe at admission. With elevated ESR level to 70mm/hr, and normal white blood cell count, systemic infection was not observed. Therefore conservative treatments were performed with herbal medicine, acupuncture, herbal acupuncture treatment, wet dressing on ulceration site, without using antibiotics and surgical treatment.

The patient's symptoms were fit to the description of sore on fingertip. Shudihuangkushen-tang was prescribed for entire treatment period, following Soyang type lower wasting-thrist treatment mentioned in Dongeuisoosebowon. From May 20 to June 9 Rehmanniae Radix Preparata from the original formula of Shudihuangkushen-tang was substituted for Rehmanniae Radix Recens to clear heat and to cool the blood(淸熱凉血), and for the latter 33 days the herbal medicine was prepared according to the original formula, using Rehmanniae Radix Preparata originally to enrich yin and tonify blood(滋陰補血)¹⁷⁾. Herbal medicine was served in decoction, three times a day.

Wet dressing was daily practiced with Haifu-san which consists of Olibanum and Myrrha. Haifu-san is reported to be effective in wound recovery, accelerating generation of granulation tissue with increased collagen content, and more effective as a combination formula than Olibanum or Myrrha alone¹⁸⁾. Haifu-san was prepared by decocting same amount of Olibanum and Myrrha for an hour into 500㎖. Haifu-san was applied after wiping out ulceration area with boric acid, hydrogen peroxide, povidone-iodine solution in order.

Taegeuk acupuncture treatment was performed two times a day. Selected meridian points are HT3, KI3, SP3, LI11, LI4, ST36 according to Soyang type Taegeuk acupuncture theory⁸⁾. Taegeuk acupuncture technique was reported to be effective in treating problems of autonomic neuropathy and circulation, which are the mechanism of CN suggested by the neurovascular theory¹⁹⁾.

Two kinds of herbal acupuncture were used, anti-inflammatory and Jahageo. From May 20 to June 3, anti-inflammatory herbal acupuncture was injected around ulceration area to clear heat and to cool the blood(淸熱凉血) and to clear heat and detoxicate(淸熱解毒)²⁰⁾, and from June 4 to July 12 Jahageo, which consists of Hominis placenta, was injected to keep the skin hydrated and strengthen skin barrier^21-22).

After treatment, edema was almost subsided, and edema improvement was also found in X-ray comparison. VAS score of edema was improved from 6 to 1. Photographs showed gradual improvement of redness and edema. The patient felt painless on the first, second, and third toe of the left foot before treatment, but since June 27, he started to feel a slight pricking pain when acupuncture needle was inserted. VAS score of pain sense was slightly improved from 0 to 1, but did not get better afterward.

Average blood sugar level decreased in the first half of treatment period, but in the latter half blood sugar level of 7AM and 3PM fluctuated, and that of 8PM increased again, higher than the first quarter. HbA1c was improved from 9.5 to 7.7%. Overall management of blood sugar level seemed to be successful, but third and fourth quarters would be better if the patient did diet control better. As edema and blood sugar level got better, the patient felt relieved and went out for dinner frequently.

In this case, after recurrence of CN, the affected area was treated without surgical treatment including debridement which is important. That means, in conclusion, conservative treatment with Korean medicine prevented further ulceration, helped to recover and ambulatory. Although improvement was confirmed by Korean medical treatment, to claim its effectiveness and generalize there are limitations as follow. Case size is only one, the treatment was not complete, and blood sugar level management was not fully satisfactory. Pain sensation recovery was positive but poor. Treatments were combined to improve symptoms, making it difficult to evaluate therapeutic effect of each treatment. Though this case would be helpful for CN patients without infections so conservative treatments can be considered as an option. Further study would be necessary if conservative Korean medical treatments practiced in this case are adequate for CN patients in need of surgical treatment.

V. Conclusion

A patient diagnosed as Charcot neuroarthropathy, and Soyang type lower wasting-thirst according to pattern identification, was conservatively treated with Korean medical treatment combined with Shudihuangkushen-tang, Haifu-san wet dressing, acupuncture, and herbal acupuncture. As a result, the following conclusions were drawn, and improvement of symptoms was confirmed