Wei Kai, Ma Xiao-peng, Wang Dong-qin, Zhang Lin-lin

1 Shanghai University of Traditional Chinese Medicine, Shanghai 201203, China

2 Shanghai Research Institute of Acupuncture and Meridian, Shanghai 200030, China

Treatment of Tolosa-Hunt Syndrome with Acupuncture: A Case Report

Wei Kai1, Ma Xiao-peng2, Wang Dong-qin1, Zhang Lin-lin1

1 Shanghai University of Traditional Chinese Medicine, Shanghai 201203, China

2 Shanghai Research Institute of Acupuncture and Meridian, Shanghai 200030, China

Tolosa-Hunt Syndrome; Electroacupuncture; Acupuncture-moxibustion Therapy; Hydro-acupuncture; Medical Records

Tolosa-Hunt syndrome is a rare medical condition with special clinical manifestations, characterized by sharp severe headache as the initial symptom. The pain is usually located around the orbit or deep inside, ocular motor nerve palsy following or together with the pain[1]. We treated a case of Tolosa-Hunt syndrome with acupuncture-moxibustion treatment, and now report it as follows.

1 Case Study

General condition: A patient, male, 38 years old, worker, visited on February 13, 2012 for the first time.

Chief complaint: Droop of the right upper eyelid for 49 d.

History of present illness: The patient started to experience swelling pain and ptosis of the right eye since 49 d before the first visit with unknown causes, and was then admitted into the Neurology Department of the Jing’an Branch of Huashan Hospital Affiliated to Fudan University. By admission, the movement of the left eye was normal, the diameter of pupil was 2.5 mm, and pupil responses to light were prompt. Right eyelid swelling, obvious conjunctival injection, and chemosis were demonstrated in the right eye. The right eye was unable to move upward, downward, or internal, and the white exposure was 2 cm at external gaze. The diameter of the right pupil was 4.5 mm, and the responses to light were retarded, with diplopia but without nystagmus. Facial sensation and corneal reflex were normal. The forehead wrinkles and nasolabial folds were symmetrical. Pain, warm and tactile sensations were revealed normal. Blood sugar was at normal level; cerebrospinal fluid and liver function were found normal; digital subtraction angiography (DSA) revealed neither internal carotid artery aneurysm nor thrombosis in cavernous sinus; Magnetic Resonance Imaging (MRI) of pituitary found abnormal signal in the right cavernous sinus. He was diagnosed with non-specific inflammatory Tolosa-Hunt syndrome. He was prescribed with Dexamethasone 15 mg and Penicillin 7 200 000 U q8h, via intravenous injection. After 20-day inpatient treatment, symptoms were generally improved: pain in the right eye was gone; the right eyelid could be lifted slightly; the two pupils were equal with prompt responses to light; extortion of the right eye could reach to the external canthus, and the intortion could be partially conducted; upward and downward gazes were not complete; improvement of diplopia was not noticeable. When discharged from the hospital, the patient was prescribed with orally taken medicines: Prednisone 50 mg qd, with gradual reduction by 10 mg every week till 30 mg, and then by reduction of5 mg every week; Omeprazole 20 mg qd; Potassium Chloride Sustained-release tablets 1.0 g bid. However, the response was not good. Therefore, the patient came for acupuncture treatment.

Symptoms and signs: Difficulty opening the right eye and the eyelid can be merely lifted even when it’s opened forcefully, coupled with swollen right eyelid, diplopia of the right eye, failure in doing upward and downward movement and only slight internal movement of the right eye though the external movement was normal. The left eye was found normal. Appetite was good but sleep was poor, and discharges were normal. Tongue body was found dark red with teeth marks and thin-white coating, and his pulse was thin and wiry.

Past medical history: The patient’s health condition used to be fine, with no other diseases or similar medical history.

Diagnosis: Tolosa-Hunt syndrome.

Treatment principles: To dredge the paths of qi and blood in eye area, to expel wind and unblock meridians and collaterals, and to reinforce spleen function and tonify qi.

Acupoints: Baihui (GV 20), Shenting (GV 24); bilateral Hegu (LI 4), Zusanli (ST 36), Sanyinjiao (SP 6), Taichong (LR 3); Touwei (ST 8), Wangu (GB 12), Xinming I [Extra, locates at 0.5 cun anterior and superior to Yifeng (TE 17), and at the mid-point of the folds behind ear lobule], Xinming II (Extra, locates at 1 cun upper and 0.5 cun exterior to the end of eyebrow), Yangbai (GB 14), Cuanzhu (BL 2), and Taiyang (EX-HN 5).

Method: The patient first took a supine position. After standard sterilization, disposable filiform needles of 0.30 mm in diameter and 40 mm in length were used to insert into the acupoints. Lifting-thrusting and twisting needling manipulations were performed to gain needling sensations. Xinming I and Xinming II were connected to G6805-Ⅱ electroacupuncture apparatus, with continuous wave and intensity within the endurance of the patient. The needles were retained for 30 min. After removal of the needles, the patient told that the right eye could open comparatively easier. The right palpebral aperture was found enlarged. Meanwhile, acupoint injection was applied to Taiyang (EX-HN 5): a 2 mL syringe containing Vit B120.25 mg was perpendicularly inserted into the acupoint by depth of about 10 mm after routine sterilization. The needle was slowly lifted and thrust to achieve needling sensation. The medicine fluid was injected when there was no withdrawal blood. Auricular point treatment was also adopted by selecting Shenmen (TF4), Liver (CO12), Kidney (CO10), Spleen (CO13), Subcortex (AT4), Eye (LO5), Eye 1 (TG2), Eye 2 (AT11). The patient was asked to press the treated auricular points 4 times a day till auricle became hot.

Treatment process: By the second visit on February 15, the patient had comparatively bigger eye opening and swelling reduced. Therefore, the treatment protocol remained the same. By the third visit on February 17, the patient told that he could open his eyes more easily, but double vision kept unchanged. Hence, based on the former formula, Jingming (BL 2) and Qiuhou (EX-HN 7) on the right side were additionally punctured. Simultaneously, acupoint injection was used at the right Wangu (GB 12). A 5 mL syringe with 2 mLDanshen(Radix Salviae Miltiorrhizae) injection was inserted into the acupoint by depth of 10-15 mm perpendicularly after standard sterilization. The needle was slowly lifted and thrust to obtain needling sensation. The medicine fluid was injected when there was no withdrawal blood. By the fourth visit on February 20, the patient can easily open his right eye and the palpebral aperture was only slightly smaller than the left one, and diplopia was also improved. Therefore, the treatment protocol remained the same. The patient came twice a week, with symptoms gradually reduced during the whole treatment duration. After 13 times of treatment, his right eye could almost open completely, and double vision was also substantially gone and only had mild attack at esotropia gaze. The right palpebral aperture was normal, and eye lid could be lifted completely, with normal eye movement and no swelling. For work sake, the patient stopped to receive further treatment. A six-month follow-up visit was conducted and revealed no relapse.

2 Case Analysis

Tolosa-Hunt syndrome is a kind of relapsing and remitting disorder, involving damage of unilateral Ⅲ,Ⅳ, andⅣ cranial nerves individually or simultaneously, causing ophthalmoplegia and periorbital pain. It was initially reported by Tolosa and Hunt, et al, and was found responding promptly to corticosteroids. It’s rarely seen in clinic and its pathogenesis and etiology are still unclear. It’s maybe related to tumor, angiopathy, inflammation, and infectious diseases, etc.[1]and it’s pathologically involving nonspecific inflammation related to immune responses[2], hence it’s a kind of chronic nonspecific inflammation with invasion of cavernous sinus and superior orbital fissure. This disease responds well to corticosteroids, but is relapsing. Some individuals may be left with incomplete paralysis of extraocular muscles or optic atrophy, severely impairing vision.

This disease belongs to the scope of strabism in Chinese medicine, and is believed to be caused by invasion of meridians and collaterals by pathogens leading to disharmony between qi and blood, and imbalance between relaxation and tension of tendons and vessels due to malnutrition[3-4]. In this cases, thepatients is fond of foods hot in nature, leading to internal heat damaging spleen and stomach, and the impaired spleen and stomach function would cause insufficient defensive yang energy and meridians and collaterals short of qi and blood. Under the circumstance, external pathogens would invade the meridians and collaterals in eye area, resulting in blocked vessels and collaterals and malnutrition of tendons and vessels. The disease was then consequently onset. Correspondingly, acupuncture-moxibustion treatment should target to unblock the path of qi and blood, to expel wind and dredge collaterals, and to tonify spleen and supplement qi. For calming mind initially, Baihui (GV 20) and Shenting (GV 24) were selected for acupuncture treatment. In local area, Xinming I, Xinming II, Yangbai (GB 14), Cuanzhu (BL 2), Taiyang (EX-HN 5) and Touwei (ST 8) were selected to expel wind and dredge collaterals, improve the circulation of qi and blood around the eyes, and supply nutrition to the upper eyelid. Jingming (BL 2) and Qiuhou (EX-HN 7) were used directly to promote the circulation of qi and blood in extraocular muscles, and help the recovery of functions. Of the selected points, Xinming I and Xinming II are two extra points. The stimulation of electroacupuncture strengthens their function of activating blood circulation and improving eyesight. For distal acupoint selection, Hegu (LI 4) and Taichong (LR 3), so called “Kai-OpeningSi Guan(four gates)”, were used together to activate circulation of qi and blood, dredge meridians and collaterals, clear heat and dampness. As the Lower He-Sea point of the Stomach Meridian, Zusanli (ST 36) works to dredge meridians and collaterals, promote the function of spleen and stomach, tonify qi in middle energizer, and nourish tendons and muscles. As the crossing point of the three foot yin meridians, Sanyinjiao (SP 6) works to tonify spleen and stomach, reinforce liver and kidney, harmonize qi and blood, soothe mind, and regulate qi and blood generally with the help of Hegu (LI 4) and Taichong (LR 3). Meanwhile, acupoint injection to Taiyang (EX-HN 5) with Vit B12nourishes the nerves around the eyes. Acupoint injection ofDanshen(Radix Salviae Miltiorrhizae) injection to Wangu (GB 12) combines the function ofDanshen(Radix Salviae Miltiorrhizae) to unblock stasis and activate the circulation of blood, and the function of Wangu (GB 12) to improve the eyesight. What’s more, acupoint injection is also featured by long-term stimulation of medicines to acupoints. Auricular points assist to regulate qi and blood, as well as to restore the functions of internal organs (liver, spleen, and kidney) during the intervals of acupuncture treatment. Integrative use of acupoints and treatment methods not only eliminates inflammation and restores the function of ocular muscles, but also modulates general qi and blood, balances yin and yang, harmonizes internal organs, and unblocks meridians and collaterals, and finally drives away disease.

Because it’s rarely encountered in clinic, there is short of clinical researches on acupuncture-moxibustion treatment for Tolosa-Hunt syndrome, and it’s usually seen as case reports currently. According to literatures abroad[5-6], acupuncture-moxibustion treatment is effective in eliminating orbital pain in Tolosa-Hunt syndrome. Domestic literature also reported a case of Tolosa-Hunt syndrome treated with puncturing the eight orbital points[7], and 3 cases treated with acupuncture together with heat-sensitive moxibustion[8], both achieving content results. The present case was treated with acupuncture plus acupoint injection and auricular point sticking, and significant effect was achieved in treating ophthalmoplegia in Tolosa-Hunt syndrome, without any adverse effects. The treatment is worth practicing, and its action mechanism expects further researches.

[1] Fu Y, Liu JR, Chen SD. Clinical analysis of 15 cases of Tolosa-Hunt syndrome. Cuzhong Yu Shenjing Jibing, 2005, 12(4): 236-237.

[2] Gladstone JP. An approach to the patient with painful ophthalmoplegia, with a focus on Tolosa-Hunt syndrome. Curr Pain Headache Rep, 2007, 11(4): 317-325.

[3] Zhang FS, Qian WZ. Clinical analysis of 9 cases of Tolosa-Hunt syndrome. Shenjing Jibing Yu Jingshen Weisheng, 2010, 10(4): 383-384.

[4] Yan AM. Treatment of ophthalmoplegia by traditional Chinese medicine: case series. Zhongxiyi Jiehe Yanke Zazhi, 1996, 14(1): 44.

[5] Nepp J, Grdser S, Flarrer S, Spacek A, Mudrich C, Stockenhuber D, Wedrich A. Tolosa Hunt Syndromeintractable pain treatment with acupuncture? Acupunct Electrother Res, 2000, 5(3-4): 155-63.

[6] Nepp J, Jandrasits K, Schauersberger J, Schild G, Wedrich A, Sabine GL, Spacek A. Is acupuncture a useful tool for paintreatment in ophthalmology? Acupunct Electrother Res, 2002, 7(3-4): 171-182.

[7] Liu HH, Sun ZR. Acupuncture for Tolosa-Hunt syndrome: a case report. Zhenjiu Linchuang Zazhi, 2009, 25(11): 25.

[8] Li JJ, Kang MF. Three cases of Tolosa-Hunt syndrome treated with acupuncture plus heat-sensitive moxibustion. Jiangxi Zhongyiyao Zazhi, 2011, 42(11): 38-40.

Translator: Hong Jue

R246.8

B

Date: September 28, 2012

Author: Wei Kai, master degree candidate. E-mail: pyweikai87@yahoo.cn

Ma Xiao-peng, M.D., researcher. E-mail: pengpengma@163.com