Qiu-Jun Wu,Hua-Nan Li,Hai-Ning Zhang,Tarazona Medina Henry David,Fei Ma,Wei Zhang,Jin-Gui Wang

1First Teaching Hospital of Tianjin University of Traditional Chinese Medicine,National Clinical Research Center for Chinese Medicine Acupuncture and Moxibustion,Tianjin,China.2Tianjin Academy of Traditional Chinese Medicine Affiliated Hospital,Tianjin,China.

Abstract Objective:A case study was performed to discuss the efficacy of traditional Chinese medicine (TCM) treatments on spasmodic torticollis.Materials and methods:A 28-year-old male was diagnosed with spasmodic torticollis due to twist and turn of neck after being frightened,who was unable to take care of himself for over 3 months.He once took an oral non-steroidal anti-inflammatory drug,but no significant improvement was seen in his symptoms.After a few months,his symptoms got worse,with onset of spasms.Therefore,he was treated at our hospital with traditional Chinese prescription and Tuina massage therapy for 3 months and followed up for 6 months.The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) and the Craniocervical Dystonia Questionnaire (CDQ-24) scores were recorded before and after treatment and during follow-up observations to evaluate the therapeutic efficacy.Results:Compared to the baseline,the TWSTRS and CDQ-24 scores decreased significantly,and there was no evident recurrence of his symptoms for at least 6 months of follow-up.Moreover,no apparent discomfort or side effects were observed during the treatment.Conclusion:Jingu abdominal massage combined with traditional Chinese prescription has a certain efficacy in the treatment of spasmodic torticollis,with almost no side effects,which can be easily accepted by patients.

Keywords:traditional Chinese prescription;spasmodic torticollis;massage therapy;case report

Highlights

Traditional Chinese medicine has a certain curative effect in the treatment of spasmodic torticollis with almost no side effects.Tuina Massage therapy is easy to be accepted by spasmodic torticollis patients.

Introduction

Spasmodic torticollis (ST) refers to an abnormal central nervous impulse leading to paroxysmal involuntary contractions of the neck muscles,so that the head and neck twists or turns to one side,resulting in hyperactivity symptoms and abnormal posture [1].The most accepted causes are related to psychogenic,extrapyramidal and neuroperipheral disorders.Although many treatments have been used for ST,none of them has shown desired effectiveness [2].According to an epidemiological survey conducted in the United States in the 1990s,the incidence of ST was around 5.7-400 per 100,000 population.The condition is more prevalent in women and among those around 50 years old [3].ST can affect the appearance,work and life routine of patients to varying degrees.In severe cases,patients even lose their ability to work and cannot look after themselves.Some patients are even accompanied by mild-to-moderate anxiety and depression.

At present,the treatments for ST mainly include oral drug therapy,botulinum toxin injection,surgery (e.g.selective peripheral neurotomy and accessory neurotomy) and physical therapy,all of which can relieve local muscular tension and spasms and improve motor symptoms to a certain extent [4-8].

These treatments,however,also have potential side effects,including drowsiness,depression,vertigo and hypotonia.Some patients treated with intramuscular botulinum toxin may experience muscle weakness,dyspnea,local bruising,dry mouth and flu-like symptoms,which though disappear without notorious side effects [9].With surgical interventions,a substantial number of patients present with neck muscle atrophy,neck and shoulder pain,neck skin numbness and other like symptoms postoperatively [10].In recent years,traditional Chinese medicine (TCM) practitioners have tried to apply non-drug therapies to treat ST.For example,Wu et al.treated several ST patients with acupuncture (5 times a week,10-week course).This study showed that acupuncture could improve neck stiffness and anxiety in the ST patient.The head and neck of the patient was no longer twisted,neck stiffness was eliminated,improving the neck mobility,and at the end of the treatment,the patient basically resumed normal lifestyle [11].Professor Wang used abdominal massage for treating ST,which had an obvious curative effect [12-13].

According to TCM theory,ST belongs to the category of "spasmodic diseases",which is caused by emotional conditions,liver Qi stagnation and meridian Qi stagnation.ST treatment aims to regulate the emotions,and to relax and loosen the tendons.Here,we report a clinical case of a ST patient who was treated with TCM.The patient exhibited rapid,significant improvement in symptoms and muscular movement.

Case report

Patient information and diagnostic assessment

This research has obtained the patient's informed consent.

A 28-years-old male patient surnamed Zhao,who was a graduate student,reported that he had a minor accident while doing a chemical experiment during his overseas study in December 2018.After the accident,he developed neck stiffness and pain,local mild edema and other symptoms.Thus,he went to a local hospital in the country he was living.There,after a series of examinations including cervical CT and cerebral MR,the patient didn't show any abnormalities,who received oral treatment with non-steroidal anti-inflammatory drugs.However,his symptoms got worse.By February of the following year,the patient suffered from involuntary neck twitching to the right side,which gradually intensified,so he tried new treatments that could help with his condition.He did neck collar fixation at a local hospital,with no apparent improvement.Therefore,the patient returned to China in March 2019 and went to the First Teaching Hospital of the Tianjin University of Traditional Chinese Medicine.

On March 17,at the time of treatment,the patient had bilateral neck stiffness,involuntary contractions of the neck,soreness and pain of neck and shoulder muscles,right neck deviation,as well as occasional spasms and stiffness.Additionally,his neck mobility was tremendously limited,which was accompanied by intermittent transient dizziness and limb numbness.He didn't have neuropathy of lower limbs,and reported that he had occasional sweating,hypoxia and poor sleep at night.He had a red tongue with ecchymosis,thin white coating and fine pulse.

Physical examination:The patient could not control the movements of his cervical spine while sitting,making it difficult for the doctors to perform a proper cervical motion examination.He had fusiform hypertrophy of sternocleidomastoid muscle.We categorized the muscle strength of both his upper limbs as grade V.The physiological reflexes were normal.Besides,the pathological reflex test was negative.

Needle electromyography (EMG) test showed tension in the right sternocleidomastoid muscle,bilateral trapezius muscle and bilateral splenius cervicis,with small amounts of fibrillation potentials.

According to Phenomenology and classification of dystonia:A consensus update [14],we diagnosed the patient with spasmodic torticollis.

Therapeutic interventions

The treatment was conducted by a senior TCM physician at The First Teaching Hospital of the Tianjin University of Traditional Chinese Medicine,who handled the massage therapy and the TCM medication.

TCM syndrome differentiation

We diagnosed him with ST because of Ying-Wei disharmony,stagnation of meridian Qi and liver wind agitation.The treatment plan was to harmonize the Ying and Wei,loosen and relax the muscles,dredge the collaterals,stop the spasms and to disperse the liver wind.

Main treatment

The overall treatment for ST comprised using traditional Chinese prescription and Tuina therapy that included local manipulation of cervical spine and Jingu abdominal massage.As a characteristic massage technique developed in Tianjin,the Jingu abdominal massage is mainly performed on the abdomen and is used to regulate the Qi of the spleen and stomach.

Tuina massage therapy:

Local manipulation of the neck:for this specific treatment,the physician used different skills such as palpating,lifting,pressing,kneading,pushing and holding,with the emphasis on relaxing the sternocleidomastoid and trapezius muscles.This was accompanied by local acupressure at some specific points on the neck,namely Tianrong (SI17),Shuitu (ST10),Futu (LI18),Tianyou (SJ16),which potentiated the muscle relaxing effect.

Jingu abdominal massage:for this technique,the physician used different types of manipulations in the abdomen as follows:

(1) Rotatory kneading abdomen manipulation (Figure 1)

Kneading Zhongwan (RN12) with the hands:the patient lay in a supine position,while the physician stood by the patient's left side and placed his left hand in the right hand,forming a semi-round shape.Then,the physician placed both hands on the patient's abdomen,pressing and making circular movements with his hands in an anti-clockwise direction on the static point (RN12).The frequency of this manipulation was 20-30 times/min for a duration of 5 min in each session.

(2) Rubbing abdomen manipulation (Figure 2)

This manipulation could be performed either with both hands or with just one hand,depending on the case.The physician implemented the rubbing manipulation on Shenque (RN8):the patient lay in a supine position,while the physician stood by the patient's left side and pushed the patient's abdomen forward using the thenar palm area,and then pulled backward using his 2ndto 5thdigits of the hand,creating a forward-backward movement.This manipulation was performed for 5 min in each session.

(3) Pressing abdomen manipulation (Figure 3)

The patient lay in a supine position,while the physician stood by the patient's left side and placed the entire palm of his left hand on the patient's abdomen and the metacarpophalangeal joint of his index finger on the patient's Zhongwan (RN12).Then,the physician placed his right hypothenar on the metacarpophalangeal joint of his left index finger,and pressed down slowly as the patient breathed.As soon as the physician felt a strong pulsation coming from the abdominal aorta region,the downward pressing was discontinued and held for 2 min.Afterwards,the physician slowly lifted up his hands till there was no pressure in the patient's abdomen.This process took around 5 min.During the manipulation,the patient should have a heat sensation in the lower limbs.

(4) Holding abdomen manipulation (Figure 4)

Figure 1 Rotatory kneading abdomen manipulation

Figure 2 Rubbing abdomen manipulation

Figure 3 Pressing abdomen manipulation

Figure 4 Holding abdomen manipulation

Using his thumb and index finger,the physician pinched and lifted outwards the skin and the muscles of the patient at the acupuncture point Daimai (GB26).This manipulation was performed 20 times on both sides concurrently,and the patient should feel a tender sensation on both sides of the abdomen.

Complementary therapy

Since the Tuina massage alone takes effect slowly for treating ST,we also used the traditional Chinese prescription.The TCM medication aimed to adjust the Ying and Wei,release and relax the muscles,dredge the collaterals and stop the spasms.The prescription used in this specific case was Gui-Zhi-Jia-Ge-Gen-Tang (GZJGGT),which was cinnamon twig decoction plus kudzu decoction combined with Zhi-Jing-San (ZJS,spasm relieving powder).Constituents:Ram.Cinnamomi 12g,Rx.Paeoniae Alba 12g,Rz.Zingiberis Recens 10g,Rx.Puerariae 20g,Fr.Zizyphi jujube 4,Fried Rx.Glycyrrhizae 6g.Soak the above drugs for 30min,decoct them twice,300ml in total,take 150ml twice a day,30min after breakfast and dinner.Scorpio and Scolopendra are made into powder,mixed in the ratio of 1:1,1.5g a day,and added to the decoction.

Treatment regimen and Follow-up

Treatment lasted for a total of 12 weeks,during which abdominal massage was carried out 5 times a week.TCM medication was given for 3 consecutive weeks and stopped for 1 week during the 3 months of treatment.The patient did not take other drugs or therapies during this treatment.

For this study,the patient was monitored and examined before the initiation of the treatment,as well as 1,2,3,6 and 9 months after the treatment,totaling 6 times.

Outcomes

The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) [15] was used to assess this study and to describe the patient's torticollis symptoms.TWSTRS has a total score of 85,with higher scores indicating severer torticollis.Further,the Craniocervical dystonia questionnaire (CDQ-24) [16-17] was used to evaluate the patient condition,since it is specifically designed to evaluate quality of life in patients with spasmodic torticollis.Lower total scores indicate milder symptoms.

Results

TWSTRS

As shown in Table 1 (Figure 5),TWSRES scores gradually decreased after treatment with traditional Chinese prescription combined with abdominal Tuina massage.After 6 months of treatment,the incidence of neck tilt was slight in the patient (Tables 2-4).

Table 1 TWSTRS scores

Table 2 Torticollis Severity Scale

Table 3 Disability Scale

Table 4 Pain Scale

CDQ-24

The CDQ-24 scores also decreased significantly,as shown in Table 5 (Figure 6).

Table 5 CDQ-24 scores

Figure 5 TWSTRS scores

Figure 6 CDQ-24 scores

Adverse events

The patient had no adverse symptoms throughout the treatment.After completing the treatment for 3 months,the patient expressed willingness to accept follow-up and repeatedly expressed willingness to continue to receive TCM treatment for possible symptom recurrence.

Discussion

From the perspectives of symptomatology and pathogenesis,spasmodic torticollis belongs to the category of "spasmodic diseases" in TCM,which is related to obstruction in the meridians and collaterals of the body,as well as a disharmony between Ying and Wei.The severity of the disease can lead to problems such as depression and drastic changes of mood,which can aggravate the disease.Given its chronic nature,combined with the tongue and pulse characteristics,it is categorized as a syndrome of Qi-blood disharmony.Therefore,the treatment cycle is long.

According to the TCM theory,abdominal massage is a treatment technique that can regulate the overall state of Qi and blood by stimulating the meridians running thought the abdomen,and by adjusting the visceral function,which achieves the effect of replenishing Qi and nourishing blood.TCM considers that spleen is directly related to blood flow,and spleen dysfunction can cause abnormal blood flow that can lead to certain types of diseases.Therefore,if the spleen function is affected,it will cause a disharmony between the Qi and the blood.

Meanwhile,the spleen is associated with the thoughts,so when a person has excessive thoughts,the function of his spleen will be affected,creating Qi stagnation and affecting other organs in the body.This is the reason why we used the acupuncture point Zhongwan (RN12) to regulate the spleen function during the treatment,since it has a certain therapeutic effect.

Zhongwan (RN12) is on the anterior midline of the abdomen,6 cun above the umbilicus.It is the intersection point of Conception Channel,Taiyang Small Intestine Channel of the Hand,Shaoyang Sanjiao Channel of the Hand and Yangming Stomach Channel of the Foot.The reason for performing the pressing abdomen manipulation on this point (RN12) is that it can adjust the related meridians and Qi.Besides,it can regulate the Qi of the spleen and stomach,dredge the Qi and blood of the San Jiao meridian,and regulate the patient's mood.

Important acupuncture point used in the holding abdomen manipulation is Daimai (GB26),which belongs to Shaoyang Gallbladder Channel of the Foot,and is the crossing point with Shaoyang Gallbladder Channel of the Foot and Dai Channel.It is considered the core of the Qi movement (ascending,descending,entering and exiting),and is the only transverse meridian of the human body that surrounds the waist,having the effect of constraining other longitudinal meridians of the human body.The occurrence of ST is linked to the dysfunction and the excessive restraint of this meridian [13].Therefore,by pulling and pressing Daimai (GB26) and using heavy stimulation technique,we can achieve slow and rapid spasmolysis and pain relief.

The reason why we should operate in the order of Rotatory kneading-Rubbing-Pressing-Holding abdomen manipulation is that the core idea of Jingu abdominal massage is that "Tong mai tiao qi",and the Qi is essential for the human body,so only the passage benefits,and the Qi machine is smooth,so that the blood sea can fill and reach the neck area.And the rotatory kneading method requires the movements to be coherent,the strength uniform and deep,and to do the gestures to be stable,the posture to be smooth,decisively continuous,and the palmoplantar to be slow.Can function as a grooming machine,relaxing the abdomen,so this method is used as the first step in abdominal massage.However,the rubbing abdomen manipulation method pushes the abdomen by manual manipulation to bring the peristalsis of intestine and adjust the intestinal Qi machine so that " turbid Qi drops and clear Qi rises",while it can further relax the abdominal muscles.Press the abdominal method is a core maneuver of abdominal massage,in which the whole body of Qi and blood can be regulated by pressing the pulse downward."Chong vessel" are places where blood and blood are operated and stored in the human body,and an unobstructed passage through the pulses of the pulses can propel the operation of blood and blood throughout the body,provide nutrients to five-zang-organ and six-fu-organ,and maintain normal physiological functions.Therefore,it is important in Jingu abdominal massage according to the abdominal law.And holding abdomen manipulation as the final step end manual so that the whole body meridians Qi and blood are unblocked.Because the TCM Mechanism of spastic torticollis is mainly due to the failure of Qi flow to run properly through the meridians,special attention should be paid to channel patency in the treatment,so Jingu abdominal massage need to follow a certain sequence in the treatment to strengthen the effect of Qi regulation through the meridians.

Modern medicine theorizes and considers the gut being the body's second brain [18].Studies have shown that there is an Enteric Nervous System (ENS) functioning like the human brain,which secretes numerous neuropeptides,including 5-HT,Motilin and GAS,as the brain does.Neuropeptides,like a bridge,link the ENS and the brain [19].And Tuina abdominal massage can influence them,helping treat neurological-related diseases.This has been confirmed by our previous studies [20-21].Thus,we theorize that it may be the reason the abdominal massage therapy eased symptoms of brain fatigue and improved mood and other mental symptoms.However,further research is required to confirm this theory.

Finally,since ST primarily affected the sternocleidomastoid and trapezius muscles,we used some targeted acupuncture points that relive the muscle tension,promote the movement of Qi and blood and eliminate the spasms.In the Tuina massage therapy,we used Tianrong (SI17),Shuitu (ST10),Futu (LI18) and Tianyou (SJ16) points.

Regarding the treatment of ST,Gui-Zhi-Jia-Ge-Gen-Tang (GZJGGT),which was cinnamon twig decoction plus kudzu decoction combined with Zhi-Jing-San (ZJS,spasm relieving powder) constituted by Ram.Cinnamomi,Rx.Paeoniae Alba,Rz.Zingiberis Recens,Rx.Puerariae,Fr.Zizyphi jujube,Fried Rx.Glycyrrhizae;Scorpio and Scolopendra,had the functions of relieving the muscle tension and alleviating pain.The spasm relieving powder is composed of scorpio and scolopendra,both of which act on the liver meridian and have the effect of stopping spasms and alleviating pain by dispersing wind and relieving convulsion.The obstruction of meridians and collaterals can cause spasmodic diseases by external pathogens such as wind,leading to the stagnation of Qi and blood,strong and acute neck pain,muscle contractures and other related muscular diseases.

The pathogenesis of ST is linked to the liver,spleen and kidney.TCM considers that the liver controls the tendons,so obstruction of liver meridian Qi can cause acute and local muscle tension,discomfort and spasms.The treatment of ST with spasm relieving powder can dredge collaterals,while the cinnamon twig decoction plus kudzu decoction can get rid of pathogens in the body,help nourish muscles,and harmonize Qi and blood.

Through this study,we found that the TWSTRS and CDQ-24 scores of the patient decreased significantly after Tuina massage and TCM medication,and the patient's neck tilt,pain,disability and quality of life were all significantly improved.Moreover,the symptoms did not reappear in the patient during 6 months of follow-up,showing the significant therapeutic efficacy of TCM on ST.Although modern research has not yet clarified the mechanism of TCM treatment for this disease,existing studies have shown that the occurrence of ST is associated with gene mutation [22],which further affects the basal ganglia in the striatum [23] and the deep cerebral cortex of the globus pallidus [24].

For spasmodic torticollis,drug therapy is the primary treatment both in China and abroad.However,a great number of patients in China choose TCM treatment,including acupuncture,Tuina massage and Chinese prescriptions.In terms of pharmaceutical choice,benzodiazepines,anticholinergics and dopamine are mostly used.Despite certain curative effects,their lasting effects are undesirable.Some reports pointed out that there were some serious side effects,and specially for refractory torticollis,surgery is the best option [25].

Botulinum toxin injection is currently the most effective way to treat ST,which has been recommended in the ST treatment guidelines [26].However,the efficacy of the drug is reduced or even ineffective in some patients after repeated use.Besides,it is a biological product,which needs to be used cautiously [27],since there will be adverse reactions such as allergies and local paralysis when using it.In China,several scholars have treated ST patients by TCM,which achieved clear curative effects.For example,Professor Han used traditional Chinese drug to treat ST patients [28],Professor Wang used Tuina massage combined with traditional Chinese prescription [12],and Professor Wu chose acupuncture treatment [29].Hence,there is no special treatment for ST and different therapeutic plans should be selected according to the patients' own symptoms.TCM has clear advantages in this respect.TCM treatment requires syndrome differentiation and the treatment regimen may vary from person to person.It also causes fewer side effects and less pain for patients,which can be easily accepted.

Limitations

Although the clinical symptoms of the patient improved in this study,the evaluation scales were only used as the clinical indicators,which lacked objective evidence.Besides,the observation time was short,and a long-term follow-up was lacking.

This study had a small sample size and lacked a randomized controlled trial,which could not clarify the efficacy of TCM treatment.Additionally,to attain a high clinical effect,it is necessary for doctors to practice Tuina abdominal massage for a long time,and get familiar with the standards and theories of human meridian Qi and blood circulation.The treatment environment needs to be relatively private and quiet,and the cooperation between doctors and patients is necessary,so there are still some inevitable interferences in clinical treatment.Hence,in the later stage of this research,we hope to summarize the clinical standards for treating ST by TCM,and further explore the internal mechanism,in order to better guide the clinical treatment.

Conclusion

Tuina massage therapy combined with traditional Chinese prescription can effectively improve the neck symptoms and quality of life of patients with spasmodic torticollis.These treatments work by relaxing local muscles in the neck and regulating the movement of Qi and blood.However,randomized controlled trials with large samples are still lacking,so the effectiveness of this treatment option remains to be seen,as well as their mechanisms of action remains to be further elucidated.