Dysphagia 19:219–230 (2004) DOI: 10.1007/s00455-004-0016-3
Effects of Motor and Sensory Stimulation in Stroke Patients with Long-Lasting Dysphagia Mary Ha¨gg, DDS,1,2 and Bengt Larsson, MD3 1 Speech and Swallowing Centre, ENT Department, Hudiksvall Hospital, Hudiksvall, Sweden,2Centre of Research & Development, Uppsala University/County Council of, Ga¨vleborg, Sweden, and 3Department of Radiology, Hudiksvall Hospital, Hudiksvall, Sweden
Abstract. Dysphagia is a common poststroke symptom with negative effects on recovery and rehabilitation. However, the orofacial regulation therapy, developed by Castillo Morales, comprising body regulation and orofacial regulation in combination with a palatal plate application has shown promising results in stroke patients. This therapy is based not only on muscle exercises but also on an improvement of the entire sensory-motor reflex arc involved in normal deglutition, and on the knowledge that the function of face and oropharynx at deglutition is closely interrelated with the entire body posture as well as with appropriate breathing. The treatment concept is relatively unknown to caregivers, partly due to lack of scientific evaluation of treatment results. The present investigation aimed to assess the effect of motor and sensory stimulation in stroke patients with dysphagia persisting for more than six months. Seven patients were evaluated with respect to orofacial and pharyngeal motility and sensory function before and two weeks after a five-week treatment period. The evaluation comprised a swallowing capacity test, a meal observation test, clinical examination of oral motor and sensory function, a velopharyngeal closure test, and videofluoroscopy. In addition, the symptoms were scored by the patients. An overall single-blind estimation showed objective and self-assessed swallowing improvement in all seven patients. Kappa coefficients are calculated on all reliability data, both inter- and intrarater reliabilities. Sensory and motor stimulation seems to be a promising therapy in stroke patients with long-lasting and persistent oropharyngeal dysphagia. Correspondence to: Mary Ha¨gg, DDS, Speech and Swallowing Centre, ENT Department, Hudiksvall Hospital, SE 824 81, Hudiksvall, Sweden. Telephone: +46 (0) 650 92754; Fax:+46 (0) 650 92750, E-mail:
[email protected]
Key words: Dysphagia — Orofacial regulation — Palatal plate — Stroke — Deglutition — Deglutition disorders.
The so-called orofacial regulation therapy was developed by Castillo Morales [1] over a period of several decades while working with children suffering from Down’s syndrome; it has later been applied in children by other authors with encouraging results [2]. The treatment comprises body regulation, manual orofacial regulation, palatal plate application, and velopharyngeal closure training. All treatment methods except the velopharyngeal closure training are described by Castillo Morales et al. [1] and are included in the concept of orofacial regulation therapy in its wider sense. Orofacial regulation therapy is based on the recognition that the typical symptoms displayed by Down’s children, because of general hypotonic muscles, are the result of a very complex development from a primary disorder through a series of compensatory mechanisms to the final symptom complex that may involve various degrees of facial expression, breathing, swallowing impairments, speech and jaw function. Successful therapy in those areas must proceed from a detailed understanding of that development. Normal overall function depends on a complicated interplay of sensory and motor functions involving a large number of muscle groups that must achieve a proper balance. The hyoid bone is directly connected to the skull, to the mandible, and to the shoulder girdle by minor muscle chains and indirectly to the pelvis through the large muscles (Fig. 1). The concept of the treatment is based on the interdependence of the orofacial complex (orofacial muscles, mandible, and oropharynx), breathing, head control,
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b Fig. 1. The functional system according to Castillo Morales: (A) skull, (B) neck vertebral column, (C) vertebral column, (D) mandible, (E) hyoid bone, (F) shoulder girdle, (G) pelvic girdle. The chewing muscles connect the mandible to the skull. The upper muscles of the hyoid bone connect the hyoid bone to the mandible and to the skull. The lower muscles of the hyoid bone connect with the shoulder girdle. The muscles of the abdominal and thoracic cavities form a ventral connection between the shoulder and the pelvic girdles. The muscles of the neck and the vertebral column connect with the shoulder and pelvic girdles.
and body posture at deglutition—the first pattern way of motion [3]. The goal of the therapy, therefore, is to secure that balance. It attempts to first remove the compensatory mechanisms and then establish
normal constituent sensory and motor functions. The background for the hypothesis is that body and orofacial regulation have an impact on dysphagia. The second pattern way of motion explains the sensory–motor reflex arc, which is activated by sensory stimulation through the afferent path and back as an impulse in the efferent motor path. Several cranial nerves are involved in that reflex arc. The sensory part of the trigeminal nerve is responsible for sensation in the face, eyes, lips, buccal mucosa, gingiva, hard palate, and tongue, and the glossopharyngeal nerve supplies sensibility in faucial arcs, tonsils, soft palate, and the posterior third part of the tongue [4,5], which is the basis for the hypothesis that palatal plate application and orofacial regulation have an impact on swallowing dysfunctions. To reach optimal results in the treatment of swallowing and breathing control, it is necessary to recognize head, neck, and body as a functional entity [1,3]. The main treatment method comprises different forms of direct manipulation and stimulation of muscles and sensory areas, often accompanied by palatal plate application. However, it requires a therapist with not only a theoretical understanding but also a skill that can only be acquired by relatively long, hands-on training and experience. Therefore, the therapy has been relatively slow to spread and, in spite of good results, it is still regrettably little known. At the Speech and Swallowing Centre in Hudiksvall, Sweden, we have used orofacial regulation therapy on Down’s children and on adults suffering from poststroke dysphagia for about a decade. The assumption was that, even though the primary causes are different, the manifest dysfunctions in those patients are similar in nature and may be understood and treated with the resources that were initially developed in connection with other groups of patients. To our knowledge there has not been any scientific attempt to apply the orofacial regulation method in stroke patients with dysphagia. The treatment of dysphagia in children suffering from Down’s syndrome must inevitably extend over a relatively long time and require constant
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Fig. 2. (A–G) Body regulation therapy is illustrated in the seven panels. For detailed description of each procedure, see the Methods section.
adaptation to the specific developmental phases that they are in, as well as to the specific treatment that they, being children, can be made to go through. The ability to continuously evaluate the effect of regulation therapy is an integral part of the skills required of the therapist, but that ability relies in large part on so-called ‘‘tacit knowledge’’ that is hard to make fully
explicit. Thus, although the value of the therapy will be clear to the therapist, it is relatively difficult to achieve a more intersubjective and controlled assessment. With adult poststroke patients, on the other hand, the treatment is in many ways more straightforward, typically shorter, and can be made somewhat more uniform. Thus, a controlled assessment of
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Fig. 3. (a–c) Orofacial regulation therapy of the chewing muscles. For detailed description of each procedure, see the Methods section.
the effect of orofacial regulation therapy on dysphagia is less difficult with poststroke patients, and yet it has at least some merit as an indirect evaluation of that therapy in general. The present investigation aimed at studying the effect of orofacial regulation therapy applied during five weeks in patients with a history of stroke at least half a year ago and with persistent oropharyngeal dysphagia in spite of conventional therapy. The therapeutic effect was evaluated by different objective methods before and after treatment.
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Fig. 4. (1–11,12a,b). Orofacial regulation therapy of the facial muscles and stimulation of the oral floor. For detailed description of each procedure, see the Methods section.
Methods All patients were recruited within three weeks from among those referred to the Speech and Swallowing Centre for examination and comprehensive care by specialists in geriatrics, internal medicine, neurology, and ENT diseases, and from the County Rehabilitation Centre. Patients were included in the sample if they suffered from stroke-induced oropharyngeal dysphagia that had persisted for more than six months, even after conventional therapy; they were
Fig. 5. Palatal plate. Note the knobs on the small stimulating plates (‘‘bumpers’’) and the sharp convexities on the velum arch for sensory stimulation. The patient can place the mobile cube in the middle, left, or right side of the dentoalveolar arch.
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Fig. 6. Swallowing capacity (ml/s) in six patients. Filled columns before treatment, open columns after treatment. P = history of bronchopneumonia. Table 1. Meal observation; mean severity scores (0–4) before (B) and after (A) treatment Drooling/ oral leakage
Oral preparation time
Nasal leakage
Patient
B
A
B
A
B
1 2 3 4 5 6 7 M C
3 3 0 3 3 3 0 3.0 15/5
0 1 0 1 1 1 0 0.8 4/5
0 3 3 3 0 2 3 2.8 14/5
0 2 1 2 0 1 1 1.4 7/5
0 0 0 0 3 3 0 3.0 6/2
Cough at swallowing
Hoarseness after eating
Meal duration
A
B
A
B
A
B
A
0 0 0 0 0 0 0 0.0 0/2
3 1 2 1 1 2 2 1.7 12/7
0 1 0 1 0 1 1 0.6 4/7
3 0 0 0 3 1 3 2.5 10/4
0 0 0 0 1 1 1 0.8 3/4
0 4 1 2 1 1 3 2.0 12/6
0 2 0 1 0 0 2 0.8 5/6
M/pat B–A 3.0–0.0 2.8–1.5 2.0–0.3 2.3–1.3 2.2–0.4 2.0–0.7 2.8–1.3
The mean severity scores (M) for each type of symptom are calculated (C) from only patients who had dysfunction of that type before treatment. M/patient to the right, M/variable at the foot of the table. (Kappa coefficient on intra/inter-rate reliability = 1.00). excluded if they had a history of psychiatric diseases, other neurological diseases, or any other diseases or injuries that might influence deglutition ability. In the end, seven stroke patients, one woman and six men, were included in the study (median age72 years, range-48–84 years; median duration of oropharyngeal dysphagia-1.5 years, range-6 months–4 years). All of the patients indicated that for them each meal was a psychosocial ordeal. Symptoms like drooling, spilling food down the chin, choking accompanied by coughing, difficulties to ‘‘get the swallowing started,’’ long mealtimes, and stiffness of face, tongue, and lips were annoying to all at the table. All patients felt restricted motility in lips and tongue, six of them felt a facial stiffness, four patients stated five or more symptoms, and all but one had a pathological water-drinking test [6]. Five patients had had a leftsided infarction, one patient had had left-sided bleeding, and one patient had had a right-sided infarction. Three of the patients had a poststroke history of bronchopneumonia. All had previously received conventional therapy including correct positioning and swallowing technique when eating and dietary advice. The patients
had a normal arousal state and were fed orally only. Five of the patients had expressive aphasia, four of them in a mild form. Before the start of treatment and two weeks after treatment the severity of each patient’s dysphagia problems was assessed from 57 test items divided into seven categories: a swallowing capacity test (ml/s), meal observation, oral motor performance, orofacial sensory function, a velopharyngeal closure test (VCT), videofluoroscopy, and patient self-assessment. The clinical tests were completed within two hours. Whenever feasible, the investigator was unaware of the pretreatment evaluation when scoring the treatment results and the intrarate reliability was controlled by repeating the scoring at a much later time on video recordings of the tests. On some tests a rough assessment of inter-rater reliability was performed.
Swallowing Capacity Test (SCT) The patient was asked to swallow 150 ml of cold tap water in one sweep and as quickly as possible [6]. The subject was instructed to sit
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Fig. 7. Meal observation score (0 = normal, 4 = severe dysfunction, mean of six variables) in seven patients. Filled columns before treatment, open columns after treatment. upright, preferably on a chair at a table, then place the glass close to the lower lip, and start drinking when the ‘‘go’’ signal was given (and stop drinking in case of difficulties). The time was measured from the onset of drinking until the last swallowing was completed. Remaining water in the glass was measured. A swallowing capacity index of 10 ml/s was regarded as the lower normal limit [6].
Meal Observation Each patient was served a meal consisting of 2 dl sour milk (yogurt that is liquid but thicker than ordinary milk), one slice of hard bread, and 1.5 dl water. During the meal, the patient was recorded with a video camera on videotape and simultaneously observed by one of the authors (MH), who also filled in a questionnaire with the following parameters, each scored from 0 (normal) to 4 (severe dysfunction): length of meal (1 = >20 min, 2 = >30 min, 3 = >40 min, 4 = inability to complete the meal); oral preparation time from intake to initiation of swallowing [7] (1 = sometimes >10, 2 = often >10 s, 3 = always >10 s, 4 = complete inability to swallow); drooling/oral leakage (1 = sometimes, 2 = often, 3 = always, 4 = complete inability to keep saliva or food in the mouth); coughing when eating (1 = sometimes, 2 = often, 3 = always); leakage to the nose (1 = sometimes, 2 = often, 3 = always); and hoarseness (the patients were asked how frequently they experienced hoarseness at meals, 1 = sometimes, 2 = often, 3 = always). The intrarater reliability of the scoring was checked about a year later through a randomized blind analysis of the videotapes. In addition, inter-rater reliability was conducted by letting two assistants perform randomized scoring of the meal observation on the video recordings. Kappa coefficients were calculated on-both inter- and intrarater reliabilities [8].
Motility Function The patients were placed in an upright and slightly forward position on a chair in front of a table and instructed to perform 29 different movements divided into five sections reflecting the motor functions of head, facial muscles, lips, jaw, tongue, and soft palate
[9]. The movements relate to head control (6 variables): flexion, extension, rotation to the right and left, lateral flexion to the right and left; facial expression (4 variables) n VII facialis: close the eyes tightly, make the eyes wide open and wrinkle the forehead, pull the brows close together, wrinkle the nose; lips (7 variables) n VII facialis: pout the lips, smile with closed lips, smack as loud as possible, blow up the cheeks against pressure of a finger, suck the cheeks together; repeat ‘‘oh-eeh’’ and ‘‘pah’’ three times as quickly and rhythmically as possible (oral diadochokinesy); jaw (5 variables, assessing the functions of the four chewing muscles), n V trigeminus; n mandibularis: open and close the mouth, move the lower jaw forward, backward, and to the left and right side; tongue (8 variables) n XII hypoglossus: stretch out the tongue as much as possible, move the tongue to the left and to the right corner of the mouth, move the tongue three times alternatively to the right and to the left as quickly and rhythmically as possible, point the tip of the tongue upward and downward, lick the lips all around and the front side of the teeth in the upper and lower jaws three times; velum (1 variable) n X vagus, n V trigeminus, n VII facialis [4,5,10]: say ‘‘ah’’ for evaluation of velum lift. The different items were scored (by MH) from zero (normal) to 4 (severe dysfunction) and registered in a test protocol. The motility tests were videotaped; that enabled a blinded, randomized control about a year later of intrarater scoring reliability. In addition, inter-rater reliability was conducted by letting two assistants perform randomized scorings of the motility function on the video recordings. Kappa coefficients were calculated for both interand intrarater reliabilities [8].
Sensory Function (Oral Stereognosia and Two-Point Discrimination) The oral sensory function was examined through stereognostic tests [11]. Eight metallic objects of two different sizes (10 mm and 20 mm in diameter) and four different shapes (full circle, half circle, star, and triangle) were placed in the mouth of the patient, who was asked to match the contour felt against a picture of five different objects. The time allowed for identification was at the most 15 s.
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Table 2. Motility dysfunction; mean severity scores (0–4) before (B) and after (A) treatment Head control 6 variables
Facial 4 variables
Lips 7 variables
Patient
B
A
B
A
B
1
*0.2 **0.2 0 0 *0.7 **0.8 1 1 *0.2 **0.2 0 0 *0.0 **0.0 0 0 *0.2 **0.2 0 0 *0.2 **0.2 0 0 *0.5 **0.5 0 0 0.3
0 0 0 0 0.3 0.2 0 0 0.2 0.2 0 0 0.0 0.0 0 0 0.2 0.2 0 0 0.2 0.2 0 0 0.2 0.2 0 0 0.2
1.0 1.0 1 1 4.0 4.0 4 4 0.0 0.0 0 0 1.0 1.0 1 1 3.5 3.5 3 3 2.3 2.1 2 3 1.5 1.6 1 2 2.2
0.0 0.0 0 0 2.7 2.8 3 3 0.0 0.0 0 0 0.0 0.0 0 0 2.7 2.5 2 2 0.0 0.0 0 0 0.0 0.0 0 0 0.9
1.7 1.5 2 1 (3.6) 3.8 4 4 0.9 0.8 1 1 1.4 1.5 1 2 3.0 3.0 3 3 3.0 3.0 3 3 2.1 2.0 2 2 2.0
2
3
4
5
6
7
M
Jaw 5 variables
Tongue 8 variables
Velum 1 variable
A
B
A
B
A
B
A
0.3 0.1 1 0 – – – – 0.3 0.2 0 0 0.7 0.6 1 1 1.4 1.2 1 1 0.3 0.2 1 1 1.6 1.4 1 1 0.8
1.6 1.4 2 2 3.6 3.4 3 3 0.0 0.0 0 0 0.2 0.2 0 0 1.2 1.2 1 1 2.4 2.2 2 2 2.0 2.0 2 2 1.8
0.8 0.6 1 1 3.6 3.4 3 3 0.0 0.0 0 0 0.2 0.2 0 0 0.4 0.2 0 0 1.8 1.6 2 1 2.0 2.0 2 2 1.5
1.6 1.8 2 2 (2.6) 2.7 3 3 0.4 0.5 1 1 1.1 1.0 1 1 0.9 0.9 1 1 3.3 3.2 3 4 2.5 2.3 2 2 1.6
0.5 0.3 1 1 – – – – 0.0 0.0 0 0 0.2 0.1 0 0 0.2 0.2 0 0 3.0 3.0 3 3 1.5 1.2 1 1 0.9
0.0 – – – – – – – 0.0 – – – 0.0 – – – 3.0 – – – 3.0 – – – 0.0 – – – 3.0
0.0 – – – – – – – 0.0 – – – 0.0 – – – 0.0 – – – 0.0 – – – 0.0 – – – 0.0
The first row (*) for each patient shows mean severity scores as assessed by the therapist, the second row (**) as assessed by the therapist at rescoring, and the third/fourth rows show randomized, blinded, overall assessments by two assistants based on video recordings. The mean severity scores (M) for the different categories of symptoms are calculated (C) from only patients who had dysfunction of that type before treatment (only the scoring of the main assessor is used). (Kappa coefficient on intra/inter-rate reliability = 0.90). Table 3. Two-point discrimination Before treatment
After treatment
Location
Right
Left
Right
Left
Lateral tongue (3 mm) Dorsal tongue (3 mm) Anterior faucial arch (3 mm) Upper lip (5 mm) Lower lip (5 mm) Cheek (15 mm)
3 4 6 5 4 4
1 3 6 4 2 2
1 5 3 3 2 2
2 0 2 0 0 0
Number of patients (out of 7) who had a dysfunction of the left or right orofacial side at the two-point discrimination test before and after treatment. The upper normal limit in parentheses.
pressure on the epithelium until a slight indentation of the area was seen. The space between the points differed depending on the area to be tested; the smallest distinguishable distance in millimeters (the upper normal limit) was for the cheeks set to 15 mm, for the lips 5 mm, for the tongue 3 mm, and for the anterior faucial arch 3 mm.
Velopharyngeal Closure Test (VCT) The ability to increase the intraoral pressure was tested by instructing the patients to inhale deeply and then exhale through a straw at a constant pace and for as long as they could against a water pressure of 12 cm. The generally accepted lower normal limit [12] is the ability to exhale against a water pressure of 5 cm for at least 5 s.
Videofluoroscopy The objects were administered randomly and each one was presented twice. Two-point discrimination [11] was tested with a pair of compasses. The two points of the compasses were placed with equal
The following parameters were analyzed by means of videofluoroscopy with low-density barium contrast medium [13]: bolus control, oral retention, epiglottic closure, retention in vallecula,
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Table 4. Dysfunction at videofluoroscopy; severity scores (0–3) before (B) and after (A) treatment 1 Patient Oral phase Impaired bolus control Oral retention Oropharyngeal phase Retention in vallecula Impaired epiglottic closure Retention in pyriform sinuses Wrong way swallowing Cough at swallowing
2
3
4
5
6
7
B
A
B
A
B
A
B
A
B
A
B
A
B
A
*2 0 *2 1
2 1 1 0
1 1 3 3
0 1 3 3
– – – –
0 0 0 0
0 1 0 0
0 1 0 0
2 1 0 0
2 0 0 0
2 2 1 0
2 2 1 0
0 1 0 0
0 0 0 0
*0 0 *0 0 *0 0 *0 0 *0 0
0 0 0 0 0 0 0 0 0 0
1 1 0 0 0 0 0 0 0 0
1 1 0 0 0 0 0 0 0 0
– – – – – – – – – –
0 0 0 0 0 0 0 0 0 0
0 0 0 0 0 0 0 0 0 0
0 0 0 0 0 0 0 0 0 0
2 1 1 1 2 1 2 1 0 0
0 0 1 0 0 0 1 0 0 0
1 1 0 0 0 0 0 0 0 0
1 1 0 0 0 0 0 0 0 0
1 0 0 0 0 0 2 1 2 0
1 0 0 0 0 0 1 1 0 0
The first row (*) for each patient shows mean severity scores as assessed by the radiologist (author BL). In addition inter-rater reliability was conducted by letting a second radiologist perform randomized scorings. Kappa coefficient on intra-rater reliability = 1.00/inter-rater reliability = 0.39.
retention in the pyriform sinus, aspiration (before, during, or after swallowing), and cough with aspiration. All variables were given a score from 0 (normal) to 3 (severe dysfunction). Scoring was performed by a radiologist (author BL) and intrarater reliability was conducted for the videofluoroscopy several weeks later. The second radiologist made a randomized and blinded rescoring on the video recordings. Kappa coefficients were calculated for both inter- and intrarater reliabilities [8].
Subjective Evaluation The impact of dysphagia on their life situation was estimated by the patients on a 100 mm VAS scale (0 = no impact, 100 = unbearable impact). Also, a history was taken regarding the frequency of bronchopulmonary complications, hoarseness in connection with meals, the patient’s mood, and hobbies.
Statistical Methods; Kappa The average of every variable, for every patient, period, and judge, has been divided into quintiles. For every judge and for these quintiles their agreement was assessed with the kappa coefficient. The kappa coefficient is in the range of 0–1 and can be interpreted as follows: 0.00–0.20, slight agreement; 0.21–0.40, fair agreement; 0.41–0.60, moderate agreement; 0.61–0.80, substantial agreement; 0.81–1.00, almost perfect agreement [8].
Treatment Method The treatment comprised body regulation, manual orofacial regulation (sensory and passive motor stimulation), palatal plate application (sensory and passive motor stimulation and active muscle exercises), and velopharyngeal closure training (active muscle exercise). All treatment methods except the velopharyngeal closure training are described by Castillo Morales et al. [1] and are
included in the concept of orofacial regulation therapy in its wider sense.
Body Regulation The body regulation was restricted to the shoulder–neck–head region. Its aim is to achieve optimal head control, equilibrium of the infrahyoidal (n XII hypoglossus) and the suprahyoidal muscles (n VII facialis, n V trigeminus, n XII hypoglossus) [4,5] and to stimulate the swallowing reflex. The therapy included seven procedures (Fig. 2A–G), each performed three times within sessions of 15 minutes. The therapist is sitting behind the patient, who is resting in supine position with a pillow under her/his knees. The patient’s muscles are stretched under pressure and vibration, and then quickly released to evoke contraction. A muscle with low tonus needs short intermittent vibration; a muscle with high tonus needs long vibration under firm pressure (the same applies to orofacial regulation). Each panel in figure 2 describes one of the seven procedures: (A) The therapist’s hands are placed under the patient’s scapular region, which is stimulated in the cranial, lateral direction during expiration of the patient. (Ba) The therapist cups her hands around the patient’s shoulders and stimulates the muscles in the caudal direction during expirations. (Bb) The procedure is repeated from each side in the contralateral direction.(C) The therapist’s hands on the upper pectoral region stimulate the muscles in the lateral, dorsal direction during expiration. (D) The therapist cups her hands around the patient’s head, which must lie safely in the therapist’s hands. Careful flexion and extension are applied and then lateral and rotating movements, followed by a slow return to the resting position. (E) With the same hand position, the therapist performs a light traction of the head in position 12, 1, and 11 o’clock and returns to the resting position. (F) With one hand on the os occipitale and the other one cupped around the patient’s chin, the therapist effects a light pressure in the dorsocranial direction followed by traction, extension, and flexion. The patient is asked to swallow, or the therapist triggers the swallowing reflex by pressing two fingers
M. Ha¨gg et al.: Dysphagia Treatment and Stroke in the cranial direction under the chin of the patient.(G) The same procedure as in movement F, but the patient’s head is (a) rotating laterally and leaning toward the shoulder after flexion. Then the therapist’s hand is placed behind the head of the patient, stretching the neck while stimulating the bottom of the mouth to trigger the swallowing reflex. Finally, (b) the flexed head is put in the midline position for retriggering the swallowing reflex. The movements in Ga and Gb are repeated for the other side.
Orofacial Regulation The orofacial regulation includes 14 different procedures, which are summarized in Figures 3 and 4. Figure 3a shows galea aponeurotica (medial m. occipitofrontalis) and 3b and 3c show the chewing muscles. The stimulation of the chewing muscles aims at optimizing jaw control (through the motor branch of n V trigeminus; n mandibularis). This stimulation has a balancing effect on the suprahyoidal muscles (through n VII facialis, n V trigeminus and, n XII hypoglossus) and thus also on the hyoid bone. The result is an enhanced release of the swallowing reflex [1]. Figure 4 (1–11) shows the facial muscles (n VII facialis) that are activated to enhance lip closure and to raise activity of the buccinator mechanism (including m. orbicularis oris, m. buccinator, m. constrictor pharyngeus superior) [14]. Figure 4(6) shows how the stimulation activates the tongue (n XII hypoglossus) to retract as a passive effect. All the movements depicted in Figure 4(1–11) give also a sensory stimulation (through n V trigeminus, afferent path) and back as a motor response (through n VII facialis, efferent path)—the sensory—motor reflex arc. Figure 4 (12 a–b) shows the muscles of the oral floor, in. digastricus, anterior abdomen (n V trigeminus), m. mylohyoideus (n V trigeminus), and m. geniohyoideus (n XII hypoglossus) [4,5]. The stimulation of these muscles will lift the hyoid bone forward and upward, encouraging the release of the swallowing reflex. The stimulation depicted in Figure 4 (12a) raises the root of the tongue, thus passively activating the receptors in the anterior faucial arch to perform the swallowing reflex and the stimulation will indirectly lift the velum. The stimulation of the muscles depicted in Figures 3 and 4 is performed through steady and calm massaging as follows (each movement is repeated three times): (3a) Galea aponeurotica—With calm and firm circular movements, the thumbs massage the area from the hairline to the top of the head and back again. The rest of the fingers are stabilizing the head. (3b) M. temporalis—With circular movements the fingers massage the temporal regions downward to processus mastoideus. This procedure is completed with a firm pressure applied on the temporal muscle which is then stretched downward/forward in a slow gliding movement. (3c) M. masseter—The massaging begins at the cheekbone and moves toward the lower jaw. It ends with pressure applied on the masseter muscle, which is then slowly stretched downward toward the angle of the jaw. [4(1)] M. occipitofrontalis—The stimulation is carried out by placing the hands on the forehead with the fingertips downward, gliding toward the eyebrows. Then the patient is asked to ‘‘look surprised,’’ look upward, wrinkle his/her forehead. [4(2)] M. corrugator superciliae—The stimulation is carried out by placing the thumbs on the muscle from the corner of the eye along the eyebrow on each side. Then the patient is asked to ‘‘look angry,’’ to frown. [4(3)] M. procerus—Here the thumbs are massaging the area from the root of the nose upward onto the lower part of the forehead. The patient is asked to ‘‘look worried.’’ M. orbicularis oculi—This region is gently stimulated by moving the index finger along the muscle from the inner corner of the eye outward, at first above and thereafter underneath the eye. Then a vibrating
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pressure is applied from the outer corner of the eye obliquely downward, upward, and then straight out to the side. The patient is asked to shut the eyes tightly. [4(5)] M. levator labii superioris alaegue nasi—With the rest of the fingers supporting the chin, the thumb massage the area from the inner corner of the eye alongside the nose and down toward the mid-upper lip. After that the patient is wrinkling his/her nose. [4(6)] M. buccinator—The stimulation is performed with a full hand from the ear toward the angle of the mouth. [4(7)] M. zygomaticus major and minor—The hand moves from the cheekbone toward the corner of the mouth, and the patient is asked to smile broadly. [4(8)] M. risorius—The cheeks are stimulated with the tips of the fingers close to the dimples toward the corners of the mouth, and the patient is asked to smile. [4(9)] M. orbicularis oris—Stimulation along the upper lip with the thumbs and the lower lip with the index fingers. Finally the corners of the mouth are extended accompanied by vibration. The patient is asked to make the lips round as if blowing, kissing. [4(10)] M. depressor anguli oris—With two fingers on the mandible bone, stimulation is carried out in an upward/inward direction toward the corners of the mouth. The patient is asked to look sour. [4(11)] M. mentalis—The thumbs stimulate the area of the chinfold and beneath, while the patient is tightening his chin and pushing up his lower lip. [4(12)] The oral floor is stimulated by the index finger and the long finger making a twisting movement in (a) upward and (b) backward/downward direction. One hand is put on the patient’s head for stabilization and is pressed downward to increase the negative intraoral pressure. The patient is asked to swallow.
Palatal Plate The palatal plate (Fig. 5) was inserted 2–3 times daily for 10–30 minutes before eating. The main plate is thin acrylic material with spring retention elements covering the entire palatal region. Four vestibular small acrylic plates (‘‘bumpers’’) with knobs in stainless steel act as stimulators for the upper lip and the buccinator mechanism [14]. For stimulation of the tip of the tongue, a mobile cube of stainless steel is attached to a dentoalveolar arch placed behind the incisors and in line with the canine teeth. For tongue base stimulation a velar arch provided with three small pointed convexities in the middle and to the sides was placed close to the Aline (the border between soft and hard palate). Three of the stroke patients had missing teeth and they received a duplicate of their maxillar denture with the same type of stimulators. The palatal plate is designed to stimulate the oral tactile receptors (passively) and the motor function (actively and passively), thus enhancing the capacity for negative intraoral pressure, the motility of the tongue, and the swallowing reflex. The negative intraoral pressure creates good lip closure and good activity in m. buccinator and velum. The plate also elicits a constant search for unfamiliar objects by the tongue. It improves the contact between tongue and palate, raises the tip of the tongue, helps the tongue to contract upward and backward, and activates m. levator anguli oris, m. zygomaticus minor and major, and m. buccinator. The swallowing improves indirectly [1,7,15]. The patients were also encouraged to do active exercises with the upper lip, tip of the tongue, tongue base, and the cheek making at least three movements against each stimulator each time. The following exercises are meant to improve labial closure and strength: Stretch the upper lip caudally and hold the extension for 1 s, stretch the lips in the ‘‘ee’’ position as far as possible, hold for 1 s, and alternately pucker the lips as tightly as possible and hold for 1 second (during each exercise the metallic knobs should be felt). Exercises which elicit and improve the buccal tension include rounding the lips tightly for ‘‘oh’’ and alternately stretching
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the lips broadly for ‘‘ee,’’ pulling the lips as far as possible alternately to the left and right side and hold for 1 s. The following exercises increase motion and strength of the tongue: With the mouth open elevate the tongue to the mobile cube, move the cube laterally, and forward, and backward; each movement as fast as possible, stretch the tongue laterally upward against the vestibular stimulators, and lick the metallic knobs toward the middle line; repeat the same movements to the other side, open the mouth and push the tongue against the front steel wire as hard as possible and hold for 1 s. The tongue base exercises include the following: Pull the tongue straight back against the back wire of stainless steel and hold it there for 1 s; try to gargle as hard as possible and then release; pretend to yawn, which also pulls the tongue base back; pronounce/k/ as hard as possible with short pauses in between [1,7].
Velopharyngeal Closure Training (VCT) The patient was instructed to train the capability for increasing intraoral pressure by performing three expirations against a water pressure of 5 cm H2O or higher three times a day before meals. The patient should sit upright with the upper end of a drinking straw (1 cm in diameter) in his mouth, its lower end reaching the bottom of the glass without pressure. The patient then had to blow bubbles as evenly as possible and for as long as possible each time.
Treatment Schedule The treatment program for all seven patients extended over five weeks. They attended the Speech and Swallowing Centre once a week for a one-hour exercise program with the orofacial regulation therapist (MH) that included all four treatment methods. In addition, body regulation and orofacial regulation were applied once a week by a physiotherapist who had received concrete, careful training in order to ‘‘calibrate’’ the treatment with the orofacial regulation therapist (MH). Training at home included palatal plate insertion, orofacial regulation [exercise in Fig. 4(6, 7, 8, 10, 11)] and velopharyngeal closure exercises 2–3 times a day. Information and instruction about the treatment methods were given to the patients, relatives, and caring staff. All home training items were recorded on videotape and sent home with the patient. Written instructions were also given and a checkup of all treatment modalities was given one week after the treatment had started. An important advantage of the therapy is that it can, in large part, be administered by the patients themselves or their relatives.
Ethical Consideration The study was approved by the Ethical Committee for Human Research at the Medical Faculty of Uppsala University, Sweden (Ups 97340), and all the patients gave written consent to participate.
Results The swallowing capacity (in ml/s) improved in six patients (Fig. 6) with a mean increase of 59%. One
M. Ha¨gg et al.: Dysphagia Treatment and Stroke
other patient was too tired to perform the test. Before treatment, the mean swallowing capacity was 5.1 ml/s (range=0.6–14.4 ml/s), and after treatment it was 9.5 ml/s (range=1.7–18.9 ml/s). The meal observation results are shown in Table 1 and Figure 7. All patients improved in three, four, or ten variables. All tested variables showed an average improvement after treatment, with a mean severity score of 2.5 before and 0.7 after treatment. The most pronounced improvement after treatment was seen with respect to drooling, cough at meals, oral preparation time, and meal time. The kappa coefficient on both intrarate and inter-rate scoring was 1.00 (Table 1). All seven patients had some orofacial motility dysfunction in some of the 31 variables tested. Improvement was seen in all patients and in most symptoms, most pronounced with respect to facial, lip, and tongue muscles; the mean severity score improved from 1.8 to 0.7 with the kappa coefficient=0.90 on both inter- and intra-rate reliabilities (Table 2). The two-point discrimination test showed a more pronounced dysfunction on the right side than the left side in all locations except for the faucial arch (Table 3). Improvement was seen in both sides of the orofacial region in all patients after the treatment period. Dysfunction of the tip of the tongue was seen in one patient before treatment but not after treatment. The oral stereognostic test was normal in four patients before treatment and in five patients after treatment. No patient could increase the intraoral pressure during the VCT before treatment. After treatment all patients could sustain intraoral pressure for a mean period of 11.8 (range=3.6–26.4 s). All but one patient reached the lower normal limit of 5 s. One patient had no dysfunction at videofluoroscopy before or after therapy, and in another patient who had no dysfunction at followup, the first examination miscarried. Of the remaining five patients, four showed oral dysfunction and four showed oropharyngeal dysfunction at videofluoroscopy prior to treatment. As shown in Table 4, the five patients with dysfunction all showed improvement in at least some variable. The kappa coefficient on intrarate reliability was 1.00 and on inter-rate reliability it was 0.39 (Table 4). All seven patients’ self-assessment according to VAS indicated improvement after treatment, with a mean severity value of 54 mm before and 37 mm after treatment (range before=27–92 mm, after=13– 72 mm). Five patients reported better mood and social activities increased in three patients.
M. Ha¨gg et al.: Dysphagia Treatment and Stroke
Discussion The basic question to be answered by this study was what effect orofacial regulation therapy may have on poststroke dysphagia patients. In order to get a reasonably clear answer, we attempted to isolate that effect by including only patients who already had received conventional treatment and could be assumed to be in a stable state of dysphagia during at least the last six months, and then applying the full resources of orofacial regulation therapy. In spite of having thus established relatively difficult conditions for the therapy, the Swallowing Capacity Test (SCT) [6] showed clear improvement in all patients after five weeks of therapy. The SCT has been demonstrated to have high intrarater, inter-rater, and test–retest reliability. It has been claimed to give reliable and valid index for assessing disordered swallowing in neurological patients and to be of value in monitoring response to therapy [6]. Dysphagia may cause a psychosocial handicap [16] with impaired life quality, which is one of the most common indications for treatment intervention. Before treatment in the present study, all patients were fed orally but each of them had some kind of dysphagia and annoyed all around the table with drooling, spilling food down the chin, coughing, difficulties getting swallowing started, long mealtimes, and stiff face, tongue, and lips. After treatment, however, all patients’ self-assessment according to VAS indicated a higher quality of life, and the meal observation showed improvements in all patients. The improved orofacial motility function, most pronounced regarding facial muscles, lips, and tongue, enhances the capacity for negative intraoral pressure at swallowing. The prerequisite for performing the VCT is the ability to increase intraoral pressure. The pretreatment inability to increase this pressure in all our patients may partly be due to weak lip closure ability and weak facial muscles but it may also be due to incomplete velum closure. All patients improved their VCT results after treatment. Facial paralysis is significantly more common in stroke patients with dysphagia than in other stroke patients [17,18]. The improved motility function of the facial muscles, lips, tongue, and velum together with the results of the SCT, meal observation, and VCT strengthens the hypothesis of the concept that palatal plate application has an impact on swallowing dysfunction and that the efficacy can increase with proper balance in the orofacial complex, optimized by body regulation and orofacial regulation [1]. The scoring for the meal observation, the motility function, the videofluoroscopy tests, etc.,
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was done by the authors themselves. However, kappa coefficients on all inter- and intrarater reliabilities indicate that results are without bias. One must recognize the great plasticity of the brain as an important factor in any recovery from damage caused by stroke [19]. Consequently, the orofacial regulation therapy may increase the recovery by focused stimulation of all sensory and motor functions involved in deglutition. The improvement in swallowing has been claimed to be an increased pharyngeal representation in the intact hemisphere — a reorganization [20]. The upper and midface, mouth, tongue, and throat all have a bilateral cortex representation [21], which can explain that there is no specific hemisphere location for dysphagia, whereas the pharynx and esophagus have an asymmetrical representation [22]. Most people have a dominant swallowing hemisphere irrespective of if they are right- or left-handed [22]. Other kinds of therapies for patients with dysphagia include adjustment of food texture, temperature and touch stimulation, and different swallowing techniques [23–26]. These therapies are mostly given shortly after the stroke and recovery can therefore be claimed to be due to spontaneous remission. Three fourths of oropharyngeal dysphagia is caused by neurological diseases. Thus, we should be more acquainted with the neurophysiology of swallowing and its disorders [27] in order to care for dysphagia patients successfully. In the future perhaps we have to evaluate dysphagia problems more objectively by the use of electromyography [27] for patient management. Conclusion In view of the clear tendency of the overall results of this pilot study, it is concluded that orofacial regulation therapy according to Castillo Morales can improve long-lasting oropharyngeal dysphagia in stroke patients. Acknowledgments. This study was supported by a grant from the Board of Rehabilitation and Technical Aids for the Disabled and The Centre for Research & Development in the County Council of Ga¨vleborg, Sweden. Many thanks go to Lita Tibbling Grahn, M.D., Ph.D, for invaluable help with the manuscript and interpretation of data, and to Reimond Dempwolf for help with editing the illustrations.
References 1.
Castillo Morales RC, Brondo JJ, Haberstock B: Darstellung der funktion der verschiadenenen elemente des orofaxialen komplexes. In: Die orofaziale Regulationstherapie, 1st ed. Richard Pflaum Verlag GmbH & Co., Mu¨nchen: 1991, pp 21–188
230
2.
3.
4. 5. 6.
7.
8. 9.
10. 11.
12.
13.
14.
M. Ha¨gg et al.: Dysphagia Treatment and Stroke
Limbrock GJ, Fischer–Brandies H, Avalle C: Castillo Morales’ orofacial therapy: treatment of 67 children with Down syndrome. Dev Med Child Neurol. 33:296–303, 1991 Anokhin PK: Systemogenesis as a general regulator of brain development. In: Himwich WA, Himwich HE (eds) The Developing Brain. New York: Elsevier 1964, pp 54–86 Chusid JK: Correlative Neuroanatomy & Functional Neurology. Los Altos, CA, LANGE Medical Publication, 1985 Heimer L: The Human Brain & Spinal Cord. New York, Spring Verlag, 1983 Nathadwarawala KM, Nicklin J, Wiles CM: A timed test of swallowing capacity for neurological patients. J Neurol Neurosurg Psychiatry 55:822–825, 1992 Logemann JA: Evaluation and Treatment of Swallowing Disorder 2nd ed. Pro-ed, Austin, TX, 1998, pp 91–95, 194, 195, 205–235 Landis JR, Koch GG: The measurement of observer agreement for categorical data. Biometrics 33(1):159–174, 1977 Hartelius L, Svensson P, Bubach A: Clinical assessment of dysarthria: Performance on a dysarthria test by normal adult subjects, and by individuals with Parkinson’s disease or with multiple sclerosis. Scand J Logop Phoniatr 18:131–141, 1993 Hardcastle W: Physiology of Speech Production: London, Academic Press, 1976 Calhoun KH, Gibson B, Hartley L, Minton J, Hokanson JA: Age-related changes in oral sensation. Laryngoscope 102:109–116, 1992 Netsell R, Hixon TA: Non-invasive method for clinically estimating subglottal air pressure. J Speech Hear Disord 43:326–330, 1978 Han TR, Paik NJ, Park JW: Quantifying swallowing function after stroke: A functional dysphagia scale based on videofluoroscopic studies. Arch Phys Med Rehabil 82:677– 682, 2001 Perkins RE, Blanton PL, Biggs N: Electromyographic analysis of the ‘‘buccinator mechanism’’ in human beings. J Dent Res 56:783–794, 1977
15.
16. 17.
18.
19. 20.
21. 22.
23. 24.
25. 26.
27.
Hoyer H, Limbrock GJ: Orofacial regulation therapy in children with Down syndrome, using the methods and appliances of Castillo Morales. ASDC J Dent Child 57:442– 444, 1990 Gustavsson B, Tibbling L: Dysphagia, an unrecognized handicap. Dysphagia 6:193–199, 1991 Barer DH: The natural history and functional consequences of dysphagia after hemispheric stroke. J Neurol Neurosurg Psychiatry 52:236–241, 1989 Nilsson H, Ekberg O, Olsson R, Hindfelt B: Dysphagia in stroke: a prospective study of quantitative aspects of swallowing in dysphagic patients. Dysphagia 13(1):32–38, 1998 Gordon C, Langton Hewer R, Wade D: Dysphagia in acute stroke. Br Med J (Clin Res Ed) 295:411–414, 1987 Hamdy S, Aziz Q, Rothwell JC, Power M, Singh KD, Nicholson D, Tallis RC, Thompson DG: Recovery of swallowing after dysphagic stroke relates to functional reorganisation in the intact motor cortex. Gastroenterology 115:1104–1112, 1998 Cohen H: Neuroscience for Rehabilitation. Philadalphia, JB, Lippincott, 1993 Hamdy S, Aziz Q, Rothwell JC, Crone R, Hughes D, Tallis RC, Thompson DG: Explaining oropharyngeal dysphagia after unilateral hemispheric stroke. Lancet 350(9079):686– 692, 1997 Davis S: Dysphagia in acute strokes. Nurs Stand 13(30):49– 54, 1999 Miller RM, Chang MW: Advances in the management of dysphagia caused by stroke. Phys Med Rehabil Clin North Am 10(4):925–941, 1999 Logemann JA: Behavioral management for oropharyngeal dysphagia. Folia Phoniatr Logop 51(4–5):199–212, 1999 Lin LC, Wang SC, Chen SH, Wang TG, Chen MY, Wu SC: Efficacy of swallowing training for residents following stroke. Adv Nurs 44(5):469–478, 2003 Ertekin C, Aydogdu I: Neurophysiology of swallowing. Clin Neurophysiol 114(12):2226–2244, 2003
