Zhang Yong-ming (張永明), Wang Jia-rong (王佳榮), Guo Fang-kai (郭方凱), Yan Yan-ning (閆彥寧), Gong Shu-hui (龔樹(shù)輝)

1 Graduate School, Hebei University of Chinese Medicine, Shijiazhuang 050011, China

2 Hebei General Hospital, Shijiazhuang 050000, China

Abstract

Keywords: Tuina; Massage; Manipulations; Atlanto-occipital Joint; Atlanto-axial Joint; Tic Disorders; Tourette Syndrome; Child

Tic disorders (TD) is a motor disorder that starts in childhood, usually presenting uncontrolled repeated movements as the primary feature, such as twisting neck, blinking eyes, twitching nose, pursing mouth, limited tics of the upper or lower limbs, coughing, clearing throat, etc. Based on the duration and severity of the disease, TD is clinically divided into three types: transient tic disorders (TTD), chronic multiple tic disorders (CMTD) and Tourette syndrome (TS)[1]. The latest study revealed that the incidence rate of TD was 1%-7% and boys run a higher risk than girls[2]. Previous research held that injuries-induced abnormal structure of the occipito-atlanto-axial joints should be the major causing factor of TD[3]. This point of view has been approved in clinic and treatments have achieved certain efficacy by targeting this factor[4-7]. This study observed the therapeutic efficacy of tuina manipulations in treating different types of TD, for seeking the relationship between TD and the occipito-atlanto-axial structural abnormalities. The report is summarized as follows.

1 Clinical Materials

1.1 Sample size estimation

The sample size was estimated using the formula for simple sample in the health statistics[8]:n=Z21-α/2π(1-π)/δ2.

In this formula,nstood for sample size,πwas population rate,δwas allowable error, andαstood for type I error rate. In this trial, the population rateπwas calculated based on clinical investigation. Whenπ=0.8,δ=0.1,α=0.05,Z=1.96 according to the table andn≈61.2 based on the formula, which meant that at least 62 patients needed to be recruited at the end of the study.

1.2 Diagnostic criteria

1.2.1 Diagnostic criteria of TD

The diagnostic criteria of TD suggested by the

Diagnostic and Statistical Manual of Mental Disorders-Fourth Edition(DSM-IV) were referred[1].

TTD: One or multiple motor or vocal tics which are uncontrolled, repeated and stereotypical, and may occur several times a day, lasting over one month but less than one year. The first onset is before 18 years old. Tics caused by drugs or internal diseases (e.g. drug-induced uncontrolled movements, Sydenham’s chorea, and viral encephalitis) were excluded.

CMTD: One or multiple motor or vocal tics which may not start at the same time but successively. The symptoms last over one year. There may occur remissions which won’t exceed three months. Tics caused by drugs or internal diseases (e.g. drug-induced uncontrolled movements, Sydenham’s chorea, and viral encephalitis) were excluded.

TS: Multiple motor or vocal tics which are uncontrolled, sudden and rapid, interfering with daily activities. The symptoms last over one year. Remission may occur but won’t exceed three months. Tics caused by drugs or internal diseases (e.g. drug-induced uncontrolled movements, Sydenham’s chorea, and viral encephalitis) were excluded.

1.2.2 Diagnostic criteria of the occipito-atlanto-axial structural abnormalities

1.3 Inclusion criteria

Conformed to the above diagnostic criteria; age <10 years old; other treatments had been terminated for at least 3 months; the guardians signed the informed consent form.

1.4 Exclusion criteria

Imaging examinations showed atlanto-axial joint displacement, atlanto-axial fusion or malformation; the Yale global tic severity scale (YGTSS) score <25 points; other extrapyramidal diseases; patients with cerebral, heart, liver, kidney or hematopoietic system diseases.

1.5 Statistical process

Data were processed using the SPSS version 21.0 statistical software. Measurement data conforming to normal distribution and homogeneity of variance were expressed as mean ± standard deviation (±s). Repeated measures data were analyzed using repeated measures ANOVA. Enumeration data were analyzed by Chi-square test.P<0.05 indicated statistical significance.

1.6 General data

This trial was approved by the Ethics Committee of Hebei General Hospital [(2019) research ethics certificate No.: (100)]. Ninety kids with TD who visited the Rehabilitation Department of Hebei General Hospital between July 2016 and January 2018, including 68 boys and 22 girls, were recruited. Their average age was (8.1±1.6) years old and the disease duration was 0.5-6.0 years, including 30 cases of TTD, 51 cases of CMTD and 9 cases of TS.

2 Treatment Methods

The patient sat astride a wooden chair of proper height facing the back of the chair, with hands grasping the back of the chair.

Step 1: With the patient’s head-neck-shoulders in the neutral position, the physician stood behind the patient, applying Tui-pushing, Na-grasping and Rou-kneading manipulations to relax the muscles of the neck and shoulders (Figure 1).

Step 2: The physician stood by the patient’s one side, flexing one elbow to fix the patient’s lower jaw and the hand supporting the occipito-temporal region of the other side, to hold the patient’s head close to the physician’s chest (Figure 2).

Step 3: Corrective manipulations were applied. Following step 2, the physician used one hand to keep the patient’s head and neck in a position reverse to that of the neck imaging (Figure 3). For the asymmetry of the lateral atlanto-dental interval, the patient was told to turn his head aside and extend his neck while looking straight forward, the physician held the C2spinous process to swing horizontally for relaxation and correction (Figure 4) at a frequency of 3 times/s, which lasted for about 2 min. When the C2spinous process can move automatically with the turning of the head, the treatment purpose was obtained. This operation can be performed repeatedly.

The manipulations were conducted once a week for 3-4 times successively, then once two weeks for 2-3 times successively, and once every three weeks for 1-2 sessions successively, for a total of 3 months.

Figure 1. Relaxing the muscles of the neck and shoulders

Figure 2. Fixing the patient’s head

Figure 3. Head-neck in the position reversing to that at the neck imaging

Figure 4. Holding C2 spinous process to swing horizontally

3 Observation of Therapeutic Efficacy

3.1 Evaluation parameters and criteria of therapeutic efficacy

3.1.1 Tics score

The YGTSS score generally rates the severity of the tics, including the number of times, frequency, intensity, complexity and the extent to which the tics are interfering. The full score is 50 points. A higher score indicates more serious tics. The patients were measured before treatment, and after 1-month and 3-month treatments. The reduction rate of the YGTSS score was taken as the criteria in evaluating the therapeutic efficacy[11]. YGTSS score reduction rate = (Pre-treatment YGTSS score – Post-treatment YGTSS score) ÷ Pre-treatment YGTSS score × 100%. Cured: YGTSS score reduction rate ≥80%.

Markedly effective: YGTSS score reduction rate ≥50% and <80%.

Effective: YGTSS score reduction rate ≥30% and <50%.

Invalid: YGTSS score reduction rate <30%.

3.1.2 X-ray examination

Neck X-ray examination was prescribed prior to treatment and after 3-month treatment, to observe the lateral and anterior atlanto-dental intervals, C2spinous process and the flexion-extension state of the occipito- atlanto-axial joints in the open-mouth and side views.

The evaluation criteria for therapeutic efficacy for occipito-atlanto-axial structural abnormalities were developed based on the X-ray examination[9].

Cured: The symptoms and body signs were gone, and the X-ray examination showed completely normal.

Markedly effective: The symptoms and body signs were substantially gone; X-ray found the lateral atlanto- dental interval difference <1 mm, the C2spinous process deviation <0.5 mm, or the lateral atlanto-dental interval <4 mm.

Effective: The symptoms and body signs were partially gone or X-ray discovered insignificant improvement.

Invalid: The symptoms and body signs remained the same, and X-ray re-examination found no improvement.

3.2 Results

3.2.1 Comparison of the YGTSS score

The YGTSS score changed significantly after 1-month and 3-month treatments compared with that before treatment (bothP<0.01); the YGTSS score after 3-month treatment was significantly different from that after 1-month treatment (P<0.01), (Table 1).

Table 1. Comparison of the YGTSS score

3.2.2 Comparison of the therapeutic efficacy for tics

The effective rate for tics was 46.6% and 86.7% respectively after 1-month and 3-month treatments, and the effective rate after 3-month treatment was significantly higher than that after 1-month treatment (P<0.01), (Table 2).

There were significant differences comparing the effective rate for tics between different types of TD after 1-month and 3-month treatments respectively (P<0.05). After 3-month treatment, the effective rate was significantly higher than that after 1-month treatment regardless of the TD type (TTD group:χ2=30.96,P=0.00; CMTD group:χ2=56.11,P=0.00; TS group:χ2=6.42,P=0.04). The details are shown in Table 3.

Table 2. Comparison of the therapeutic efficacy for tics (case)

Table 3. Comparison of the effective rate among different types of TD (case)

3.2.3 Comparison of the X-ray findings

After 3-month treatment, the incidence rates of the typical abnormal X-ray findings including the asymmetry of the lateral atlanto-dental interval, broadened anterior atlanto-dental interval, C2spinous process deviation, and occipito-atlanto-axial flexion/extension instability changed significantly compared with those before treatment (allP<0.01), (Table 4).

3.2.4 Comparison of the therapeutic efficacy based on the X-ray findings

After 3-month treatment, the abnormal neck X-ray findings showed improvement to different extent. Of the 90 patients, 20 were cured, 22 showed markedly effective, 22 showed effective, and the cured and markedly effective rate (cured rate + markedly effective rate) was 75.6%.

Table 4. Comparison of the typical X-ray findings regarding the occipito-atlanto-axial structure (case)

4 Discussion

Occipito-atlanto-axis is a structure consisting of occipital bone, atlas, axis, joint capsule and adjacent ligaments. Ligaments and fasciae are important tissues linking and restricting atlanto-axial vertebrae and occipital bone. Kids’ cervical muscles are relatively thin and weak and lack mature protective and repair function. In this condition, the stability of occipito- atlanto-axis mainly depends on the integrity of ligaments in this area[12]. Ligaments are mainly composed of collagen fibers and elastic fibers. The occipito-cervical ligament is a limiting ligament, mostly made of collagen fibers, with a small amount of elastic fibers at the edge, thus presenting a biomechanical characteristic of high stiffness but low elasticity[12]. Children usually run a high risk of head-neck injuries[13]. Occipito-cervical ligament injuries can be easily caused by fall, collision and whiplash injury, etc. and adhesions or scar tissues may form during the repair of ligament injuries[14].

TTD and CMTD may be associated with the abnormal structure of occipito-atlanto-axis which stimulates the superior sympathetic ganglion resulting in constant excitement and hyperactive dopamine system. Moreover, long-term occipito-atlanto-axial deformation may lead to the mechanical and functional disorders of the cervical vertebrae or even the whole spine, presenting various discomforts affecting the torso or limbs. Excessive proprioception enters and stimulates sympathetic nervous system, causing symptoms such as shaking head, shrugging shoulder, blinking eyes, pursing mouth, twitching nose, snorting and clearing throat. TS may be caused by constant stimulation to the superior cervical sympathetic ganglion that activates sympathetic nervous system and leads to hyperactive dopamine system which acts on the limbic system that controls reproductive function, presenting involuntary sexual behaviors[15].

The previous study found that manipulations adjusting the occipito-atlanto-axial structure produced satisfactory result in treating TTD[16]. Manipulations can rebalance the tension of the occipito-cervical ligament, improve the surrounding muscle spindles and joint mechanoreceptors, reduce the noxious stimulation to occipito-atlanto-axial sensory nerves, block the hyper- excitement of sympathetic nerves, rebalance the dopamine and endocrine systems, and consequently get the uncontrolled movements under control. In this trial, manipulations were applied to different types of TD. Respectively after 1-month and 3-month treatments, the YGTSS score showed significant improvement compared with that before treatment; there were significant differences comparing the effective rate in different types of TD after 1-month and 3-month treatments (P<0.05). After 3-month treatment, the effective rate was significantly higher than that after 1-month treatment in the three types of TD; the effective rate for tics was respectively 100%, 80.4% and 77.7% in TTD, MCTD and TS, suggesting that the efficacy was more significant for TTD and CMTD compared with TS, and the efficacy for TTD was most significant.

Regarding the radiographic parameters, the abnormal cervical X-ray features all showed improvement to different extent after 3-month treatment, and the cured and markedly effective rate was 75.6%.

The imaging examinations showed that the improvement in some structures was insignificant, which may be because the repair of ligament requires a long process going through swelling, scar repair, scar softening and finally the restoration of elasticity. Although the anatomical displacement was not completely corrected according to the imaging examination, the manipulation therapy was still effective, since it can regulate the nervous reflex, humor, and endocrine system besides correcting the abnormal structures[17], which also indicates that it should be a complex mechanism that involves multiple factors in manipulation treatment of TD.

TD has always been considered as an extrapyramidal disease of unclear causes and mainly treated with drugs. The results of this study has confirmed the efficacy of tuina manipulations for different types of TD and provided novel idea for treating TD from the perspective of correcting the abnormal occipito-atlanto- axial structure, which is worthy of further research.

Received: 24 October 2019/Accepted: 18 December 2019

Conflict of Interest

The authors declare that there is no conflict of interest.

Acknowledgments

This work was supported by Scientific Research Project of Health Commission of Hebei Province (河北省衛(wèi)生健康委員會(huì)科研項(xiàng)目, No. 20190327); Science and Technology Plan Project of Hebei Province (河北省科技計(jì)劃項(xiàng)目, No. 14K57715D).

Statement of Informed Consent

Informed consent was obtained from the guardians of all individual participants.