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Vocal Parameters in Individuals with Traumatic Spinal Cord Injury: A Systematic Review
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Vocal Parameters in Individuals with Traumatic Spinal Cord Injury: A Systematic Review *Felipe de Oliveira Rodrigues, *Camila de Alencar Frois, †Max Sarmet Moreira Smiderle Mello, and ‡ Laura Davison Mangilli, *yzBrasília, Brazil Summary: Objective. To analyze and assess studies on the topic and to describe the vocal parameters of patients with traumatic spinal cord injury (SCI) based on the literature. Method. Studies were identified and selected by searching for articles on the subject published in any journal, using pre-established descriptors: “spinal cord injury,” “speech,” “lang$,” “speech-lang$,” and “voice” (“traumatismos da medula espinal,” “fala,” “fona$,” “fonoaud$,” and “voz” in Brazilian Portuguese). All the phases of the study were conducted independently by the researchers and in the event of disagreement, a final decision was reached by consensus. The articles selected were critically assessed based on their objectives, treatment, and assessment criteria and methods, results and conclusions, as well their level of scientific evidence. Results. A total of 70 scientific articles were identified, eight of which were considered valid based on the inclusion criteria. Research on the contribution of speech therapy to patients with SCI is scarce, particularly regarding voice assessment and treatment. Descriptive and observational studies predominated, with a small sample. Data collection was predominated cross-sectional, which made it possible to identify evaluation and intervention techniques, but the methodologies described preclude generalizations. The results indicated that the parameters of the respiratory function and vocal production in patients with traumatic SCI were reduced lung capacity, presence of voice problems, presence of perceived voice problems, and altered activation of accessory respiratory muscle. Conclusion. The vocal parameters of the patients with traumatic SCI can be described, considering the lack of information available. Disorders related to speech therapy vary in accordance with the type and level of injury. It is important to increase scientific production in this area, particularly randomized trials, in order to establish assessment criteria and treatment techniques and improve levels of evidence. Key Words: Spinal cord injury−Breath−Voice−Speech therapy−Evaluation.
INTRODUCTION Speech therapy encompasses, health promotion, prevention, evaluation and diagnosis, patient guidance, treatment (training and rehabilitation), and improving speech-language aspects of the central and peripheral auditory systems, vestibular function, spoken and written language, voice, fluency, articulation, and the swallowing, cervical, and orofacial myofunctional systems. Speech therapists are therefore equipped to deal with communication disorders of any etiology, diagnosing and treating respiratory, voice, or speech impairments and any aspect related to human communication or the orofacial myofunctional system.1 Research conducted by the Brazilian Institute of Geography and Statistics in partnership with the Ministry of Health found that 6.2% of Brazilians have some form of disability.2 The study also shows that 1.3% of the population have a
Accepted for publication December 18, 2019. Conflicts of interest: The authors declare that there is no conflict of interest. Source of funding: This study was financed in part by the Coordena¸c ~ao de Aperfeic¸ oamento de Pessoal de Nível Superior - Brasil (CAPES) - Finance Code 001. From the *Rehabilitation Sciences Program, University of Brasília, Faculdade de Ceil^andia, Brasília, Brazil; yHospital de Apoio de Brasília, Brazil; and the zSpeech Therapy Course and Graduate Rehabilitation Sciences Program, University of Brasília, Faculdade de Ceil^ andia, Brasília, Brazil. Address correspondence and reprint requests to Laura Davison Mangilli, Faculdade de Ceil^ andia, University of Brasília, Centro Metropolitano, Conjunto A Lote 1, Brasília -DF 72220-900, Brazil. E-mail: [email protected] Journal of Voice, Vol. &&, No. &&, pp. &&−&& 0892-1997 © 2019 The Voice Foundation. Published by Elsevier Inc. All rights reserved. https://doi.org/10.1016/j.jvoice.2019.12.013
physical disability, almost half of whom (46.8%) are severely to very severely impaired, and only 18.4% receive rehabilitation.2 Paragraph IIc, Article 2, of Federal Law 7.853/89 guarantees people with disabilities access to healthcare and stipulates the creation of a network of specialized treatment and rehabilitation centers.3 Spinal cord injury (SCI), often caused by physical trauma, results in a critical clinical condition that has major physical and psychological impacts, with irreversible neurological impairment. The annual global incidence is 10−83 cases per million people, lowering quality of life.4 Speech-language disorders can occur following spinal injury and may affect the voice and swallowing.5 Research corroborates the relationship between spinal injury and a high incidence of dysphagia; however, studies on voice disorders are incipient, with research gaps and low scientific evidence levels that demand more in-depth investigation.6−8 Spinal cord injuries affect the extrinsic muscles of the larynx, altering its movement during speech and compromising vocal resonance. This disorder requires multidisciplinary care in which the role of speech therapists is key.9 Speech therapists can provide important treatment to optimize speech and swallowing outcomes through assessment, strategic compensation techniques, dietary recommendations, patient education, and rehabilitation interventions that target communication (speech, voice, resonance) and swallowing.8 Speech therapy is efficient at improving Functional
ARTICLE IN PRESS 2 Independence Measure outcomes in patients with SCI, whose needs range from treatment for severe disability to temporary impairement.10,11 Although self-reported voice function is limited and the risk of voice problems high, people with SCI are seldom referred to a speech therapist for evaluation and treatment. This suggests limited awareness regarding the nature of the disorder and its possible vocal consequences, as well as the use of assessment methods and instruments in clinical speech therapy that are not always suited to this population.12 The number of technologies developed and incorporated in recent decades is directly related to decreased mortality and greater knowledge and information about medical technologies and care costs.13 The amount of knowledge produced on the different effects and consequences of technologies requires increasingly insightful analysis and synthesis in new forms of research to support decision making. The literature suggests that systematic reviews, meta-analysis, decision analysis, and cost-effectiveness analysis are the most effective methods to achieve this.13 Considering the above, the present study addresses verbal communication and the respective speech-language aspects of respiratory function and sound production in patients with SCI. The aim is to qualitatively assess the literature and to describe the vocal parameters of individuals with traumatic SCI. METHOD The search method was established based on the recommendations of the Cochrane Handbook.14 Studies were identified and selected by searching for articles on the subject, published in the database belonging to the regional web portal of the Virtual Health Library (BVS − http://bvsalud.org/). The following descriptors were used: “spinal cord injury,” “speech,” “lang$,” “speech-lang$,” and “voice” (“traumatismos da medula espinal,” “fala,” “fona$,” “fonoaud$,” and “voz” in Brazilian Portuguese). The Boolean operator “AND” was used to combine the descriptors. Database searches were performed independently by two researchers, with no date restrictions. Each of the articles extracted was also analyzed independently by three researchers. The titles and abstracts were read to assess their eligibility for inclusion in the study. Articles not written in English, Spanish, or Portuguese, not directly related to the topic, those with overlapping keywords, literature reviews, and letters to editors were excluded. Those deemed eligible met the following inclusion criteria: (1) original research articles; (2) describing voice and speech parameters in patients with SCI; (3) speech-language interventions targeting the speech and voice of patients with SCI. The chosen articles were critically analyzed in two stages: Stage 1 − Level of evidence and methodological quality It aimed to assess the level of evidence and methodological quality using the Oxford Centre for Evidence-based
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Medicine15 and Downs and Black checklist.16 The Oxford Centre for Evidence-based Medicine Levels of Evidence15 is based on the study design, classifying the evidence into levels 1a, 1b, 1c, 2a, 2b, 2c, 3a, 3b, 4 and 5, and then grades of recommendation (strength). Cochrane reports that the Downs and Black checklist can be used to assess the bias of individual studies included in systematic reviews.17 The checklist was developed and validated to evaluate the methodological quality of randomized and observational studies. It contains 27 questions covering five domains − reporting (nine questions), external validity (three questions), bias (seven questions), confounding/selection bias (six questions), and power (one question).17 When the criterion that characterizes quality is present, a score of 1 should be given in each of the questions and 0 if it is not. The scale was applied independently by three researchers, only to the clinical/observational studies. A single final score was produced for each article. Stage 2 − Critical analysis The articles were critically analyzed in terms of the vocal parameters exhibited by individuals with SCI, considering the following variables: the objectives; methods (number, sex, age range of participants); treatment and assessment methods and criteria; results (vocal parameters), and conclusions of each study. RESULTS A total of 70 scientific articles were identified, eight of which were considered valid based on the inclusion criteria. Specific studies regarding alternative care for patients with SCI and/or those that did not adopt a speech therapy approach were excluded. Figure 1 illustrates the article selection process. Stage 1 − Level of evidence and methodological quality The level of evidence15 and raw score of the clinical/observational studies selected based on the Downs and Black checklist16 are described in Table 1. The checklist was not used for the study by Gordan et al10 due to your descriptive design. Stage 2 − Critical analysis The results of critical analyses are summarized in Table 2. The articles describe the vocal parameters exhibited by individuals with SCI, associated or not with speech therapy interventions. It is important to note that three of the articles assessed5,10,11 form part of a series of studies aimed at patients with SCI and the range of interventions available to them. DISCUSSION Research on the contribution of speech therapy to patients with SCI is scarce, particularly regarding voice assessment
ARTICLE IN PRESS Felipe de Oliveira Rodrigues, et al
Arcles idenfied = 70
Repeated based on overlapping keywords = 8 Arcles analyzed (tles and abstracts) = 62
Arcles selected = 13
Arcles not avaliable in their enrety = 5
3
Vocal Parameters in Individuals with Traumatic Spinal Cord Injury
Arcles excluded = 49 (literature reviews = 1; arcles that did not meet the inclusion criteria = 47)
Complete arcles analyzes = 8
FIGURE 1. Article selection process. and treatment. Studies with a small sample and that do not describe assessment and treatment methods, makes the level of evidence low. It is important to increase scientific production in this area, particularly randomized trials, in order to establish assessment criteria and treatment techniques and improve levels of evidence.17 With respect to the type of study, descriptive and observational research predominated. Data collection was crosssectional, which made it possible to identify evaluation and intervention techniques, but the methodologies described preclude generalizations. The results of this study indicated that the parameters of the respiratory function and vocal production in patients with traumatic SCI were reduced lung capacity (vital capacity − inspiratory capacity, expiratory reserve volume),7,19,21 voice
problems (voice quality, pitch, loudness, and maximum duration of speech),7 perceived voice problems (quality of the voice)7,21 and greater recruitment of accessory respiratory muscle and higher accessory muscle activation in changing from soft to loud speech.21 According to Brasil,22 healthcare involves providing services and initiatives aimed at health promotion and disease prevention, rehabilitation, and treatment. Thus, the present study aimed to summarize and critically analyze scientific production on voice assessment and training in patients with SCI. Traumatic SCI causes physical, social, and emotional damage. Its effects vary, but evidence demonstrates the occurrence of several pathologies, such as depression followed by a decline in communication and social isolation.7,23 Disorders related to speech therapy vary in accordance with the type and level of injury.5,7,11,20,24 Cervical-level injuries are the most severe and affect the respiratory system, swallowing, speech and voice, and patients with complete SCI exhibit greater impairment and a worse prognosis than those with incomplete injuries. Although less severe, thoracic spinal cord injuries require careful monitoring, since these patients achieve better safe swallowing outcomes during the acute period and are then discharged, meaning important rehabilitation and quality of life aspects may be overlooked.7,20,24 Phonation is the result of aerodynamic energy generated by airflow and converted into acoustic energy via antagonistic forces that act on the myoelasticity of the vocal cords, creating a push-pull effect of synchronized opening and closing. The myoelasticity of the vocal cords and the Bernoulli effect are the main forces involved in the vibration of the vocal cords and sound production.25,26 Reduced air flow will therefore result in less subglottic pressure, altering sound production.12 The vulnerability of patients with SCI during the acute phase is associated with the paralysis of intercostal and other expiratory muscles, which may hinder coughing and expectorating phlegm, thereby increasing the risk of pneumonia and other respiratory complications.27 In complete
TABLE 1. Evidence Level15 and Downs and Black Checklist16 Results Anderson18 Watson Gordan Brougham Nygren- Tamplin Gordan Macbean and et al10 et al5 Bonnier et al21 et al11 et al7 19 20 Hixon et al Evidence levels - OCEBM15 Reporting Methodological External Validity quality - Downs Bias and Black Confounding checklist16 variables/ Selection bias Power Total
4 3 0 0 0
4 7 0 1 0
2A -
2C 6 0 2 0
4 3 0 1 0
4 6 0 2 1
2C 7 1 2 0
4 6 0 2 0
0 3
0 8
-
0 8
0 4
0 9
0 10
0 8
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TABLE 2. Articles Selected, with Their Respective Objectives, Methods and Conclusions Reference Country Journal Anderson18 Sweden Paraplegia
Watson and Hixon19 United States JSLHR
Gordan et al10 United States The Journal of Spinal Cord Medicine
Brougham et al5 United States The Journal of Spinal Cord Medicine
Objective
Methods
Results/Conclusion
Describe the benefits of Participants: 16 patients Respiratory and vocal parameters: not described; the Swedish (11 men) with spinal cord The tailor-made tracheostomy tubes provided modification of the injuries, requiring better adaptation, easy maintenance and low tracheostomy tube to ventilation or not; cost, in addition to improving speech and voice permit speech. Procedures: customized quality. The high-quality tube material makes it silver, silicone or silicone possible to use the same custom-made tubes for rubber tracheostomy tubes longer. for each patient. Study the effects of Participants: 3 men Respiratory and vocal parameters: (1) spirometry: abdominal trussing (24−41 years) with values were reduced based on normality as clinical treatment complete cervical spinal inspiratory capacity was the least reduced, and cord injury (C5−C6); expiratory reserve volume was the most reduced Procedure: a custom-built component of the vital capacity; (2) capacity of device was used to truss oral reading: not described (only the comparison the anterolateral with trussed and untrussed conditions) abdominal wall - the effects The results showed that abdominal trussing of trussing on lung volume increased vital capacity, presumably by and during oral reading optimizing diaphragm function. Longer utterance (average sound pressure duration for oral reading was observed with level, syllables per increased inspiratory capacity. utterance, pause duration) The findings of this study support clinical were studied. impressions that trussing can be used to improve speech in individuals with abdominal paralysis or paresis and diminished breathing function for speech. Describe a treatment Speech language pathology Respiratory and vocal parameters: not described; classification system clinicians developed a The SLP taxonomy consists of 7 intervention (taxonomy) developed taxonomy to provide a categories that address deficits - speech by speech language “system of rules and units” production for patients with artificial airway, pathologists to that would describe key motor speech and voice, swallowing, cognitiveexamine the effects of interventions in the communication, and communication - and the speech language rehabilitation of patients associated exercises and tasks that patients pathology (SLP) with SCI. perform. interventions on SCI Collaborative efforts of SCIRehab SLP rehabilitation technicians to develop the taxonomy provided outcomes. the opportunity to extensively share practice patterns and treatment methods. The clinicians identified multiple treatment inconsistencies, but also learned about differences in practice patterns. The different treatment approaches were consolidated into a practical number of taxonomic and the comprehensive SLP taxonomy for SCI rehabilitation was developed. Describe the nature and Participants: 600 patients with Respiratory and vocal parameters: not described; distribution of speech- traumatic spinal cord injury - 40% of patients received SLP treatment; of language pathology with different neurological these, 57% received intensive therapy; the activities during SCI level and injury integrity; remainder were primarily evaluated or received rehabilitation and Procedures: data was less intensive treatment. discuss predictors of collected from patient - cognitive-communication and swallowing the amount of time medical records. Speech therapy were the most common activities. spent in specific SLP language pathologists The significant variability in cognitive SLP treatment activities. entered details about treatment, which is not well explained by patient sessions times and activity and injury characteristics, sets the stage for descriptions were future research to associate treatments with considered. results.
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TABLE 2. (Continued ) Reference Country Journal
Objective
Nygren-Bonnieri et al20 Sweden Disability and Rehabilitation
Explore and describe decreased lung function in people with cervical spine injuries (CSCI).
Tamplin et al21 Australia Arch Phys Med Rehabil
Develop measurement techniques to quantitatively assess breathing patterns and respiratory muscle recruitment during singing and speech in quadriplegia.
Gordan et al11 United States The Journal of Spinal Cord Medicine
Describe associations of patient characteristics and speech-language pathology (SLP) interventions provided during inpatient SCI rehabilitation with outcomes at discharge and 1 year after injury.
Macbean et al7 Australia International Journal of SpeechLanguage Pathology
Examine changes in phonation and quality of life in the acute and sub-acute stages of recovery after cervical spinal cord injury (CSCI).
Methods
Results/Conclusion
Participants: 33 people with Respiratory and vocal parameters: note described; cervical spinal cord injury Experience related to decreased lung function (between levels C4 and C8); was grouped into three domains: limitations in Procedures: a semibreathing function, ability to cough and voice structured interview guide function. was constructed with the Breathing, the ability to cough and voice function focus on experience of were perceived as limited in most of the patients symptoms related to with CSCI. Most had adapted to the situation and decreased lung function. developed their own strategies for coping, but their limitations were not necessarily experience or referred to as a problem. Participants: six subjects Respiratory and vocal parameters: reduced lung with motor complete C5−7 capacity (vital capacity, 71% vs 102% of quadriplegia + six healthy predicted; P = 0.028); more perceived voice individuals problems (VHI score, 22.5 vs 6.5; P = 0.046), Procedure: a single 45−60greater recruitment of accessory respiratory minute assessment session muscle (loud and soft volumes) (P = 0.028); and (Questionnaire - Voice higher accessory muscle activation in changing Handicap Index (VHI); from soft to loud speech (P = 0.028); respiratory function The results showed that participants with (spirometry and respiratory quadriplegia exhibited the expected respiratory pressure meter); vocal dysfunctions and self-perceived vocal assessment (surface impairment. electromyography (EMGs), plethysmographya, audio data). Participants: 1376 patients Respiratory and vocal parameters: not described; with traumatic spinal cord Patients with SCI who also have traumatic brain injury; injury and therefore have the greatest need for Procedures: initial interventions to address cognitiveevaluation; cognitivecommunication limitations (CCLs) receive the communication treatment most SLP cognitive-communication treatment and swallowing exercises. and show the greatest improvement during Each session was rehabilitation according to the Functional documented by the speech Independence Measure (FIM), but cognitive language pathologists. functioning remained impaired at discharge. This likely explains the consistent finding that more hours of SLP treatment is associated with lower cognitive FIM scores at discharge. Participants: two patients Respiratory and vocal parameters (change was with cervical spinal cord present across measures for both cases): reduced injury + four control lung capacity (vital capacity, 80% and 40% vs 86, individuals; 41% of mean controls); more voice problems Procedure: participants (perceptual ratings of connected speech were were assessed in a quiet considered within normal limits for case one and setting, at three stages in case two speech to be mild − moderately during recovery: 1 week impaired across pitch, loudness, and voice quality after mobilization; variables, with the exception of pitch level 6 months after initial (severely impaired; maximum duration remained evaluation; 12 months after at the lower end of the normal range for case one initial evaluation (11 seconds) and in the case two was severely (spirometry; impaired (5.18 seconds); case one considered that electroglottography; the impairment of his voice rated as mild or mildcomputerized speech to-moderate, and voice function considered to be laboratory + effective and not having any substantial impact on questionnaires). his daily life, and case two considered his voice to be moderately impaired)
(Continued)
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TABLE 2. (Continued ) Reference Country Journal
Objective
Methods
Results/Conclusion In both cases change was observed over time in terms of the extent and pattern of recovery, with most deficiencies remaining relatively stable over the 12-month period. Subtle changes were observed in respiratory function, laryngeal function and phonation over time (case 1) and moderate to severe deficits in overall respiratory function and breathing patterns for speech, laryngeal function and phonation (case 2).
SCI, all the muscles below the damage are paralyzed, including respiratory muscles, with spasticity also compromising lung function.12,27,28 As such, patients with SCI may experience breathing difficulties and problems such as dysphagia and dysphonia.10 In that case an early tracheostomy should be considered, ensuring patient survival in the acute phase and improving respiratory condition in the chronic phase.29 Tracheostomy within seven days of intubation is recognized as a good treatment option because it may improve respiratory outcomes in patients with traumatic cervical spinal cord injury and it was also associated with fewer ventilator days, lesser time to decannulation, shorter ICU stays, and lower mortality.30−32 It has been reported that 11%−77 % of patients with cervical spinal cord injury require tracheostomy depending on the level of injury31,32 and long-term mechanical ventilation was required for 6.5%.33 McCully34 found that tracheostomy is unlikely in patients with incomplete SCI, regardless of the level of injury. Decannulation of patients with SCI can be a challenge considering that decannulation protocols vary based on the patient’s unique requirements and institutional policies. However, an earlier decannulation may suggest that early tracheostomy patients are more quickly achieving respiratory independence regarding cough strength and secretion management.29,35 In clinical practice cough effectiveness and ability to tolerate tracheostomy tube capping are the most considered parameters.35 Failure to decannulate and death due to respiratory dysfunction are common in patients with a C4 complete SCI because diaphragm dysfunction continues for a long period. Conditions such smoking history, age >45 years, comorbid lung diseases, and active pneumonia can make decannulation process more difficult.32,36 However there is an extremely high possibility of decannulation in C5−7 paralysis patients who can perform a shoulder shrug shortly after injury.32 The dysphagia is common in patients with spinal injuries. Respiratory dysfunction is a major cause of morbidity and mortality in SCI, compromising the muscles of respiration and reducing vital capacity, cough efficacy, and chest wall
compliance. Lung infections are a serious concern in spinal injuries and preventing pneumonia is essential to lowering mortality and shortening hospital stays. In addition, research on respiratory function in this population is important in order to provide more effective interventions.37−39 Reduced respiratory function after cervical spine injuries can indirectly result in voice disorders.12,19−21 Complete injuries cut off motor and sensory function below the level of damage, whereas some residual function remains in incomplete SCI. Cervical injuries between C1 and C3 severely limit respiratory function, while innervation of the diaphragm is partially maintained in SCI below C3; however, the intercostal and abdominal muscles are paralyzed, reducing vital capacity.12 Additionally, although passive expiration is preserved (since it occurs largely via elastic and not muscular force), forced expiration is generally significantly compromised. Although spinal injuries do not generally compromise laryngeal motor function or the mucous membrane of the vocal folds, impaired respiratory function can indirectly affect the voice due to the paralyzed intercostal and abdominal muscles, limiting lung volumes and capacities as well as subglottic pressure control.12 Lower lung capacities and respiratory pressure and significantly greater voice impairment (Voice Handicap Index) in quadriplegics. Electromyographic data indicated significantly greater activation of the sternocleidomastoids and diaphragm during high (compared to low) vocalization in controls and of the trapezius in quadriplegic patients.21 Atypical recruitment of these muscles indicates compensation in the phonation process.19−21 Additionally, abdominal wall paralysis or paresis in individuals with cervical spine injuries, as well as neurological disorders such as muscular dystrophy. The resulting decline in inspiratory capacity decreases vital capacity, compromising the respiratory function required for speechand affecting reading and prosody.19 Experience related to decreased lung function was grouped into three domains by Nygren-Bonnieri et al20 limitations in breathing function, ability to cough, and voice function. The predominant theme of all the participant’s interviews was that although the limitations were significant
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Vocal Parameters in Individuals with Traumatic Spinal Cord Injury
in most cases compared to the situation before injury, they were not necessarily experienced or referred to as a problem. This corroborates the fact that spinal cord injuries compromise respiratory capacity, affecting voice and speech functions. Vocal changes are not caused by dysfunctions in the glottis, but rather its aerodynamics. Factors that influence dysphonia include respiratory function, limited vocal range, reduced loudness, and compensatory changes in phonation among patients with SCI, prompting patients to avoid oral communication.24 Even incomplete cervical spine injuries can cause speech and voice changes, which negatively affect quality of life in these patients. The effect on speech production differs in accordance with the nature of the injury (level and integrity) and the presence of other contributing factors, such as vocal cord paralysis.7 Comorbid traumatic SCI and traumatic brain injury (TBI) can produce a complex clinical spectrum with devastating consequences and can be a major contributor and confounder to impaired cognition40,41 posing important challenges in the rehabilitation of a dual diagnosis SCI patient. Physical, cognitive, and/or emotional impairments are expected as a result from a TBI. Neuropsychological symptoms may result from brain injury and/or may be related to nonorganic factors.42 A high frequency of TBI co-occurring with traumatic SCI of 39.6% was reported40 but previous estimates have been ranging wide from 10% to 74%.42 Potential co-contributors include concomitant brain injury, psychological or somatic comorbidities, decentralized cardiovascular control, and sleep apnea.41,42 Noncompliance, inability to learn, maladaptive reactions to SCI, and poor motivation are neuropsychological symptoms of a missed TBI diagnosis that may be perceived during rehabilitation. It is possible to expect other cognitive symptoms like difficulties with concentration, attention, memory, word finding/speech, perception, information processing, and executive functions. Emotional symptoms may include irritability, liability, anxiety, and depression.42 The highest incidence rate of dual diagnosis reported are for those between 15 and 24 years old. Motor vehicle collisions were most likely to yield a comorbid TBI diagnosis, followed by falls. Patients with cervical and thoracic injuries showed a very similar frequency of comorbid TBI.40 A study conducted in the United States on the taxonomy of speech-language pathology in people with SCI demonstrated that speech disorders were more prevalent in the respiratory and laryngeal subsystems of speech production.5,10,11 Respiratory dysfunctions include shallow inspiration and/or shorter, less controlled expiration, whereas laryngeal injuries may manifest themselves in prosodic disorders related to speech rate, duration, stress, and/or accent patterns. Additionally, vocal cord paralysis can be caused by damage to the peripheral nerve of the superior and/or recurrent laryngeal nerve.10
7
Based on a previously developed taxonomy for stroke rehabilitation, speech language pathology clinicians developed a taxonomy to provide a “system of rules and units” that would describe key interventions in the rehabilitation of patients with SCI. Time is recorded for each category, and supplementary information focuses on cueing needs and family involvement that help describe and guide intervention selection.10 The patients could receive intensive therapy (more than five sessions during rehabilitation) or punctual interventions (one to five sessions). At the intensive treatment, the patients received an average of 16.1 hours of SLP, and significant differences were observed in the time spent on each activity among neurological injury groups. The cognitive-communication and swallowing therapy were the most common activities. The patients with motor levels of injury at C1−C4 spent more time in swallowing therapy, whereas those with low tetraplegia and paraplegia focused the greatest percentage of time on cognitive-communicative work.5 Associations between patient characteristics and speechlanguage pathology (SLP) interventions provided during inpatient SCI rehabilitation with outcomes at discharge and 1 year after injury. SLP treatment factors explain a small amount of variation in Functional Independence Measure, participation and mood. Variation explained by treatment factors for cognitive outcomes at discharge increased when the patient group was more homogeneous. More time in SLP cognitive communication interventions had a negative relationship, while longer length of stay was positive.11 Based on the results of this review, research is still incipient and as a result, there are no specific assessment and treatment methods that target communication in this population. In this case, the future research and targeted clinical practice should be based on prior knowledge of speech-language pathology (SLP) in general and closing the gap in knowledge regarding SLP directly related to this population. Methods must be established for this specific intervention considering evidence-based practice to ensure better rehabilitation results in patients with SCI. Dysphagia is generally treated during the acute period of injury, with better established evaluation and treatment procedures. Given the severity of SCI and its traumatic impact on those affected, voice and speech become secondary complaints, but remain a vital part of the rehabilitation and inclusion process, contributing to providing patients with a new perspective on life. Speech is the most important form of human communication and social inclusion. As such, even when speech disorders go unnoticed or ignored by patients, proper care should be provided at all levels to ensure their health and quality of life.43 Several studies highlight the need for respiratory training in patients with SCI as a foundation for speech therapy. This is based on the idea that increasing respiratory capacity improves expectoration, thereby reducing cases of bronchoaspiration and enhancing aerodynamics to ensure better quality vocalization and clearer, more fluent speech. It is
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important to investigate which patients might benefit from this therapeutic approach, considering complete high-level injuries and the likelihood that the muscles in question will not be reactivated. In these cases, voice modulation and resonance should be considered as a means of compensating for the compromised airflow.44 Atypical muscle activation was observed during loud speech, indicating spontaneous compensation and raising the question of whether this pathway might be a therapeutic alternative or harmful behavior.21 In this respect, speech assessments using well-defined controlled methods are important in this population in order to provide medical staff with valuable information on patient prognosis and how best to meet their needs. Objective assessment and treatment techniques are needed and should be based on research that specifically targets this pathology.
4.
5.
6.
7.
8.
9.
CONCLUSION The vocal parameters of the patients with traumatic SCI can be described, considering the lack of information available. Disorders related to speech therapy vary in accordance with the type and level of injury. Individuals with SCI exhibit stomatognathic system disorders such as ineffective breathing, dysphagia, dysphonia, and speech disorders. Speech therapy is a relatively new field and in-depth research is needed to create assessment protocols and define treatment methods aimed exclusively at this population.
10. 11.
12.
13.
AUTHORS’ CONTRIBUTION 1. Study conception and design: FOR and LDM. 2. Data collection, analysis and interpretation: FOR, CAF, MSMSM and LDM. 3. Drafting the article or revising it critically: FOR, CAF, MSMSM and LDM. 4. Final approval of the version submitted for publication: LDMT.
14.
15.
16.
SUPPLEMENTARY DATA Supplementary data related to this article can be found online at doi:10.1016/j.jvoice.2019.12.013.
17.
REFERENCES 1. Conselho Federal de Fonoaudiologia. Exercício Profissional do Fonoaudi ologo [Internet] 2002 [citado 2010 Jan 10]. Brasília, DF: CFF; 2002. Available from: http://www.crfa8r.org.br/downloads/ epdo1.pdf. Accessed 27 January 2018. 2. Ag^encia Brasil. IBGE: 6,2% da Popula¸c ~ao t^em Algum Tipo de Defici^encia [Internet] 2015. Available from: http://www.ebc.com.br/noti cias/2015/08/ibge-62-da-populacao-tem-algum-tipo-de-deficiencia. Accessed on: Jan 27, 2018. 3. Brasil. Lei n° 7.853/89, de 24 de outubro de 1989. Disp~oe sobre o apoio as pessoas portadoras de defici^encia, sua integra¸c ~ao social, sobre a Coordenadoria Nacional para Integra¸c ~ao da Pessoa Portadora de Defici^encia - Corde, Institui a Tutela Jurisdicional de Interesses Coletivos ou Difusos Dessas Pessoas, disciplina a atua¸c ~ao do Ministério Publico, Define
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20.
21.
Crimes, e dá Outras Provid^encias. Brasília, DF: Diário Oficial; 1989. 24 out. Yamane K, Mazaki T, Shiozaki Y, et al. Collagen-binding Hepatocyte Growth Factor (HGF) alone or with a Gelatin- furfurylamine hydrogel enhances functional recovery in mice after spinal cord injury. Sci Rep. 2018;17:917. Brougham R, David DS, Adornato V, et al. The SCIRehab project: treatment time spent in SCI rehabilitation. Speech-language pathology treatment time during inpatient spinal cord injury rehabilitation: the SCIRehab project. J Spinal Cord Med. 2011;34:186–195. MacBean N, Ward E, Murdoch B, et al. Optimizing speech production in the ventilator-assisted individual following cervical spinal cord injury: a preliminary investigation. Int J Lang Commun Disord. 2009;44:38293. MacBean N, Ward E, Murdoch B, et al. Phonation after cervical spinal cord injury (CSCI): prospective case examinations of the acute and sub-acute stages of recovery. Int J Speech Lang Pathol. 2013;15:312– 323. Starmer HM, Ayoub N, Byward C, et al. The impact of developing a speech and swallow rehab program: improving patient satisfaction and multidisciplinary care. Laryngoscope. 2017;127:2578–2581. Soares E, Marchiori EC, Rodrigues JPB, et al. Performance in the treatment of patients with lesions in spinal medulla. Fono Atual. 2001;5:44–47. Gordan W, Spivak-David D, Adornato V, et al. SCIRehab: the speech language pathology taxonomy. J Spinal Cord Med. 2009;32:307–318. Gordan W, Gerber D, Spivack-David D, et al. Relationship of speechlanguage pathology inpatient rehabilitation interventions and patients characteristics to outcomes following spinal cord injury: the SCIRehab project. J Spinal Cord Med. 2012;35:565–577. Johansson K, Strombergsson S, Robieux C, et al. Perceptual detection of subtle dysphonic traits in individuals with cervical spinal cord injury using an audience response systems approach. J Voice. 2017;31: 126.e7–126.e17. Silva LK. Technology assessment and cost-effectiveness analysis in health care: the adoption of technologies and the development of clinical guidelines for the Brazilian national system. Ci^enc Saude Coletiva. 2003;8:501–520. Higgins JPT, Grenn S. Cochrane Handbook for Systematic Reviews of Interventions. Version 5.1.0. Australia (AUS): Department of Health and Ageing, Austrália [revisado em 2011]. The Cochrane Collaboration. Available from: www.cochrane-handbook.org. Accessed on: October 23, 2013. Oxford Centre for Evidence-Based Medicine: Levels of Evidence (March 2009) [Internet]. 2009 Mar [cited 2014 dez 20]. Available from: http://www.cebm.net/oxford-centre-evidence-based-medicine-levels-evi dence-march-2009. Accessed on January 27, 2018. Downs SH, Black N. The feasibility of creating a checklist for the assessment of the methodological quality both of randomised and nonrandomised studies of health care interventions. J Epidemiol Community Health. 1998;52:377–384. Brasil. Ministério da Saude. Secretaria de Ci^encia, Tecnologia e Insumo Estratégicos. Departamento de Ci^encias e Tecnologia. Diretrizes metodologicas: elabora¸c ~ao de revis~ao sistemática e metanálise de estudos observacionais comparativos sobre fatores de risco e prognostico / Ministério da Saude, Secretaria de Ci^encia, Tecnologia e Insumos Estratégicos, Departamento de ci^encias e Tecnologia. Brasília: Ministério da Sa ude; 2014:132p. Anderson G. The Swedish modification of the tracheostomy tube to permit speech. Paraplegia. 1993;31:203–206. Watson PJ, Hixon TJ. Effects of abdominal trussing on breathing and speech in men with cervical spinal cord injury. J Speech Lang Hear Res. 2001;44:751–762. NygrenBonnier M, Normi LL, Klefbeck B, et al. Experiences of decreased lung function in people with cervical spinal cord injury. Disabil Rehabil. 2011;33:530–536. Tamplin J, Brazzale DJ, Pretto JJ, et al. Assessment of breathing patterns and respiratory muscle recruitment during singing and speech in quadriplegia. Arch Phys Med Rehabil. 2011;92:250–256.
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22. Brasil. Ministério da Saude. Gabinete do Ministro. Portaria no 4.279, de 30 de dezembro de 2010. Brasil: Estabelece diretrizes para a organnico de iza¸c ~ ao da Rede de Aten¸c ~ao a Sa ude no ^ambito do Sistema u Sa ude (SUS); 2010. 23. Grillo MHMM, Penteado RZ. The impact of voice on the quality of life of elementary school teachers. Pro-Fono R Atual Cient. 2005;17: 311–320. 24. L opez MN, Araujo F. Papel de la terapia logopédica en pacientes con lesi on medular cervical. Med Reabil. 2000;52:13–15. 25. Nemetz MA, Pontes PAL, Vieira VP, et al. Vestibular fold configuration during phonation in adults with and without dysphonia. Rev Bras Otorrinolaringol. 2005;71:6–12. 26. Deguchi S. Mechanism of and threshold biomechanical conditions for falsetto voice onset. PLoS One. 2011;6:e17503. 27. Britton D, Goldstein B, Jones-Redmond J, et al. Baclofen pump intervention for spasticity affecting pulmonary function. J Spinal Cord Med. 2005;28:343–347. 28. Colman ML, Beraldo PC. Study of maximal inspiratory pressure variations in tetraplegics, treated by means of incentive spirometry, in outpatients. Fisioter Mov. 2010;23:439–449. 29. Beom JY, Seo HY. The need for early tracheostomy in patients with traumatic cervical cord injury. Clin Orthop Surg. 2018;10:191–196. 30. Flanagan CD, Childs BR, Moore TA, et al. Early tracheostomy in patients with traumatic cervical spinal cord injury appears safe and may improve outcomes. Spine. 2018;43:1110–1116. 31. Tanaka J, Yugue I, Shiba K, et al. A study of risk factors for tracheostomy in patients with a cervical spinal cord injury. Spine. 2015;41: 764–771. 32. Nakashima H, Yukawa Y, Imagama S, et al. Characterizing the need for tracheostomy placement and decannulation after cervical spinal cord injury. Eur Spine J. 2013;22:1526–1532.
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33. Quesnel A, Veber B, Proust F, et al. What are the perspectives for ventilated tetraplegics? A French retrospective study of 108 patients with cervical spinal cord injury. Ann Phys Rehabil Med. 2015;58:74–77. 34. McCully BH, Fabricant L, Geraci T, et al. Complete cervical spinal cord injury above C6 predicts the need for tracheostomy. Am J Surg. 2014;207:664–668. 35. Santus P, Gramegna A, Radovanovic D, et al. A systematic review on tracheostomy decannulation: a proposal of a quantitative semiquantitative clinical score. BMC Pulm Med. 2014;14:201. 36. Higashi T, Eguchi H, Wakayama Y, et al. Analysis of the risk factors for tracheostomy and decannulation after traumatic cervical spinal cord injury in an aging population. Spinal Cord. 2019;57:843–849. 37. Lee JC, Gross BW, Rittenhouse KJ, et al. A bitter pill to swallow: dysphagia in cervical spine injury. J Surg Res. 2016;201:388–393. 38. Brown R, DiMarco AF, Hoit JD, et al. Respiratory dysfunction and management in spinal cord injury. Respir Care. 2006;51:853–868. 39. Ramczykowski T, Grunong S, Gurr A, et al. Aspiration pneumonia after spinal cord injury. Placement of PEG tubes as effective prevention. Unfallchirurg. 2012;115:427–432. 40. Budisin B, Bradbury CC, Sharma B, et al. Traumatic brain injury in spinal cord injury: frequency and risk factors. J Head Trauma Rehabil. 2016;31:33–42. 41. Sachdeva R, Gao F, Chan CCH, et al. Cognitive function after spinal cord injury: a systematic review. Neurology. 2018;91:611–621. 42. Kushner DS, Alvarez G. Dual diagnosis: traumatic brain injury with spinal cord injury. Phys Med Rehabil Clin N Am. 2014;25:681–696. 43. Rumbach A, Coombes C, Doeltgen S. A Survey of Australian Dysphagia Practice Patterns. Dysphagia. 2018;33:216–226. 44. Sapienza CM, Wheeler K. Respiratory muscle strength training: functional outcomes versus plasticity. Semin Speech Lang. 2006;27: 236–244.
Vocal Parameters in Individuals with Traumatic Spinal Cord Injury: A Systematic Review *Felipe de Oliveira Rodrigues, *Camila de Alencar Frois, †Max Sarmet Moreira Smiderle Mello, and ‡ Laura Davison Mangilli, *yzBrasília, Brazil Summary: Objective. To analyze and assess studies on the topic and to describe the vocal parameters of patients with traumatic spinal cord injury (SCI) based on the literature. Method. Studies were identified and selected by searching for articles on the subject published in any journal, using pre-established descriptors: “spinal cord injury,” “speech,” “lang$,” “speech-lang$,” and “voice” (“traumatismos da medula espinal,” “fala,” “fona$,” “fonoaud$,” and “voz” in Brazilian Portuguese). All the phases of the study were conducted independently by the researchers and in the event of disagreement, a final decision was reached by consensus. The articles selected were critically assessed based on their objectives, treatment, and assessment criteria and methods, results and conclusions, as well their level of scientific evidence. Results. A total of 70 scientific articles were identified, eight of which were considered valid based on the inclusion criteria. Research on the contribution of speech therapy to patients with SCI is scarce, particularly regarding voice assessment and treatment. Descriptive and observational studies predominated, with a small sample. Data collection was predominated cross-sectional, which made it possible to identify evaluation and intervention techniques, but the methodologies described preclude generalizations. The results indicated that the parameters of the respiratory function and vocal production in patients with traumatic SCI were reduced lung capacity, presence of voice problems, presence of perceived voice problems, and altered activation of accessory respiratory muscle. Conclusion. The vocal parameters of the patients with traumatic SCI can be described, considering the lack of information available. Disorders related to speech therapy vary in accordance with the type and level of injury. It is important to increase scientific production in this area, particularly randomized trials, in order to establish assessment criteria and treatment techniques and improve levels of evidence. Key Words: Spinal cord injury−Breath−Voice−Speech therapy−Evaluation.
INTRODUCTION Speech therapy encompasses, health promotion, prevention, evaluation and diagnosis, patient guidance, treatment (training and rehabilitation), and improving speech-language aspects of the central and peripheral auditory systems, vestibular function, spoken and written language, voice, fluency, articulation, and the swallowing, cervical, and orofacial myofunctional systems. Speech therapists are therefore equipped to deal with communication disorders of any etiology, diagnosing and treating respiratory, voice, or speech impairments and any aspect related to human communication or the orofacial myofunctional system.1 Research conducted by the Brazilian Institute of Geography and Statistics in partnership with the Ministry of Health found that 6.2% of Brazilians have some form of disability.2 The study also shows that 1.3% of the population have a
Accepted for publication December 18, 2019. Conflicts of interest: The authors declare that there is no conflict of interest. Source of funding: This study was financed in part by the Coordena¸c ~ao de Aperfeic¸ oamento de Pessoal de Nível Superior - Brasil (CAPES) - Finance Code 001. From the *Rehabilitation Sciences Program, University of Brasília, Faculdade de Ceil^andia, Brasília, Brazil; yHospital de Apoio de Brasília, Brazil; and the zSpeech Therapy Course and Graduate Rehabilitation Sciences Program, University of Brasília, Faculdade de Ceil^ andia, Brasília, Brazil. Address correspondence and reprint requests to Laura Davison Mangilli, Faculdade de Ceil^ andia, University of Brasília, Centro Metropolitano, Conjunto A Lote 1, Brasília -DF 72220-900, Brazil. E-mail: [email protected] Journal of Voice, Vol. &&, No. &&, pp. &&−&& 0892-1997 © 2019 The Voice Foundation. Published by Elsevier Inc. All rights reserved. https://doi.org/10.1016/j.jvoice.2019.12.013
physical disability, almost half of whom (46.8%) are severely to very severely impaired, and only 18.4% receive rehabilitation.2 Paragraph IIc, Article 2, of Federal Law 7.853/89 guarantees people with disabilities access to healthcare and stipulates the creation of a network of specialized treatment and rehabilitation centers.3 Spinal cord injury (SCI), often caused by physical trauma, results in a critical clinical condition that has major physical and psychological impacts, with irreversible neurological impairment. The annual global incidence is 10−83 cases per million people, lowering quality of life.4 Speech-language disorders can occur following spinal injury and may affect the voice and swallowing.5 Research corroborates the relationship between spinal injury and a high incidence of dysphagia; however, studies on voice disorders are incipient, with research gaps and low scientific evidence levels that demand more in-depth investigation.6−8 Spinal cord injuries affect the extrinsic muscles of the larynx, altering its movement during speech and compromising vocal resonance. This disorder requires multidisciplinary care in which the role of speech therapists is key.9 Speech therapists can provide important treatment to optimize speech and swallowing outcomes through assessment, strategic compensation techniques, dietary recommendations, patient education, and rehabilitation interventions that target communication (speech, voice, resonance) and swallowing.8 Speech therapy is efficient at improving Functional
ARTICLE IN PRESS 2 Independence Measure outcomes in patients with SCI, whose needs range from treatment for severe disability to temporary impairement.10,11 Although self-reported voice function is limited and the risk of voice problems high, people with SCI are seldom referred to a speech therapist for evaluation and treatment. This suggests limited awareness regarding the nature of the disorder and its possible vocal consequences, as well as the use of assessment methods and instruments in clinical speech therapy that are not always suited to this population.12 The number of technologies developed and incorporated in recent decades is directly related to decreased mortality and greater knowledge and information about medical technologies and care costs.13 The amount of knowledge produced on the different effects and consequences of technologies requires increasingly insightful analysis and synthesis in new forms of research to support decision making. The literature suggests that systematic reviews, meta-analysis, decision analysis, and cost-effectiveness analysis are the most effective methods to achieve this.13 Considering the above, the present study addresses verbal communication and the respective speech-language aspects of respiratory function and sound production in patients with SCI. The aim is to qualitatively assess the literature and to describe the vocal parameters of individuals with traumatic SCI. METHOD The search method was established based on the recommendations of the Cochrane Handbook.14 Studies were identified and selected by searching for articles on the subject, published in the database belonging to the regional web portal of the Virtual Health Library (BVS − http://bvsalud.org/). The following descriptors were used: “spinal cord injury,” “speech,” “lang$,” “speech-lang$,” and “voice” (“traumatismos da medula espinal,” “fala,” “fona$,” “fonoaud$,” and “voz” in Brazilian Portuguese). The Boolean operator “AND” was used to combine the descriptors. Database searches were performed independently by two researchers, with no date restrictions. Each of the articles extracted was also analyzed independently by three researchers. The titles and abstracts were read to assess their eligibility for inclusion in the study. Articles not written in English, Spanish, or Portuguese, not directly related to the topic, those with overlapping keywords, literature reviews, and letters to editors were excluded. Those deemed eligible met the following inclusion criteria: (1) original research articles; (2) describing voice and speech parameters in patients with SCI; (3) speech-language interventions targeting the speech and voice of patients with SCI. The chosen articles were critically analyzed in two stages: Stage 1 − Level of evidence and methodological quality It aimed to assess the level of evidence and methodological quality using the Oxford Centre for Evidence-based
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Medicine15 and Downs and Black checklist.16 The Oxford Centre for Evidence-based Medicine Levels of Evidence15 is based on the study design, classifying the evidence into levels 1a, 1b, 1c, 2a, 2b, 2c, 3a, 3b, 4 and 5, and then grades of recommendation (strength). Cochrane reports that the Downs and Black checklist can be used to assess the bias of individual studies included in systematic reviews.17 The checklist was developed and validated to evaluate the methodological quality of randomized and observational studies. It contains 27 questions covering five domains − reporting (nine questions), external validity (three questions), bias (seven questions), confounding/selection bias (six questions), and power (one question).17 When the criterion that characterizes quality is present, a score of 1 should be given in each of the questions and 0 if it is not. The scale was applied independently by three researchers, only to the clinical/observational studies. A single final score was produced for each article. Stage 2 − Critical analysis The articles were critically analyzed in terms of the vocal parameters exhibited by individuals with SCI, considering the following variables: the objectives; methods (number, sex, age range of participants); treatment and assessment methods and criteria; results (vocal parameters), and conclusions of each study. RESULTS A total of 70 scientific articles were identified, eight of which were considered valid based on the inclusion criteria. Specific studies regarding alternative care for patients with SCI and/or those that did not adopt a speech therapy approach were excluded. Figure 1 illustrates the article selection process. Stage 1 − Level of evidence and methodological quality The level of evidence15 and raw score of the clinical/observational studies selected based on the Downs and Black checklist16 are described in Table 1. The checklist was not used for the study by Gordan et al10 due to your descriptive design. Stage 2 − Critical analysis The results of critical analyses are summarized in Table 2. The articles describe the vocal parameters exhibited by individuals with SCI, associated or not with speech therapy interventions. It is important to note that three of the articles assessed5,10,11 form part of a series of studies aimed at patients with SCI and the range of interventions available to them. DISCUSSION Research on the contribution of speech therapy to patients with SCI is scarce, particularly regarding voice assessment
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Arcles idenfied = 70
Repeated based on overlapping keywords = 8 Arcles analyzed (tles and abstracts) = 62
Arcles selected = 13
Arcles not avaliable in their enrety = 5
3
Vocal Parameters in Individuals with Traumatic Spinal Cord Injury
Arcles excluded = 49 (literature reviews = 1; arcles that did not meet the inclusion criteria = 47)
Complete arcles analyzes = 8
FIGURE 1. Article selection process. and treatment. Studies with a small sample and that do not describe assessment and treatment methods, makes the level of evidence low. It is important to increase scientific production in this area, particularly randomized trials, in order to establish assessment criteria and treatment techniques and improve levels of evidence.17 With respect to the type of study, descriptive and observational research predominated. Data collection was crosssectional, which made it possible to identify evaluation and intervention techniques, but the methodologies described preclude generalizations. The results of this study indicated that the parameters of the respiratory function and vocal production in patients with traumatic SCI were reduced lung capacity (vital capacity − inspiratory capacity, expiratory reserve volume),7,19,21 voice
problems (voice quality, pitch, loudness, and maximum duration of speech),7 perceived voice problems (quality of the voice)7,21 and greater recruitment of accessory respiratory muscle and higher accessory muscle activation in changing from soft to loud speech.21 According to Brasil,22 healthcare involves providing services and initiatives aimed at health promotion and disease prevention, rehabilitation, and treatment. Thus, the present study aimed to summarize and critically analyze scientific production on voice assessment and training in patients with SCI. Traumatic SCI causes physical, social, and emotional damage. Its effects vary, but evidence demonstrates the occurrence of several pathologies, such as depression followed by a decline in communication and social isolation.7,23 Disorders related to speech therapy vary in accordance with the type and level of injury.5,7,11,20,24 Cervical-level injuries are the most severe and affect the respiratory system, swallowing, speech and voice, and patients with complete SCI exhibit greater impairment and a worse prognosis than those with incomplete injuries. Although less severe, thoracic spinal cord injuries require careful monitoring, since these patients achieve better safe swallowing outcomes during the acute period and are then discharged, meaning important rehabilitation and quality of life aspects may be overlooked.7,20,24 Phonation is the result of aerodynamic energy generated by airflow and converted into acoustic energy via antagonistic forces that act on the myoelasticity of the vocal cords, creating a push-pull effect of synchronized opening and closing. The myoelasticity of the vocal cords and the Bernoulli effect are the main forces involved in the vibration of the vocal cords and sound production.25,26 Reduced air flow will therefore result in less subglottic pressure, altering sound production.12 The vulnerability of patients with SCI during the acute phase is associated with the paralysis of intercostal and other expiratory muscles, which may hinder coughing and expectorating phlegm, thereby increasing the risk of pneumonia and other respiratory complications.27 In complete
TABLE 1. Evidence Level15 and Downs and Black Checklist16 Results Anderson18 Watson Gordan Brougham Nygren- Tamplin Gordan Macbean and et al10 et al5 Bonnier et al21 et al11 et al7 19 20 Hixon et al Evidence levels - OCEBM15 Reporting Methodological External Validity quality - Downs Bias and Black Confounding checklist16 variables/ Selection bias Power Total
4 3 0 0 0
4 7 0 1 0
2A -
2C 6 0 2 0
4 3 0 1 0
4 6 0 2 1
2C 7 1 2 0
4 6 0 2 0
0 3
0 8
-
0 8
0 4
0 9
0 10
0 8
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TABLE 2. Articles Selected, with Their Respective Objectives, Methods and Conclusions Reference Country Journal Anderson18 Sweden Paraplegia
Watson and Hixon19 United States JSLHR
Gordan et al10 United States The Journal of Spinal Cord Medicine
Brougham et al5 United States The Journal of Spinal Cord Medicine
Objective
Methods
Results/Conclusion
Describe the benefits of Participants: 16 patients Respiratory and vocal parameters: not described; the Swedish (11 men) with spinal cord The tailor-made tracheostomy tubes provided modification of the injuries, requiring better adaptation, easy maintenance and low tracheostomy tube to ventilation or not; cost, in addition to improving speech and voice permit speech. Procedures: customized quality. The high-quality tube material makes it silver, silicone or silicone possible to use the same custom-made tubes for rubber tracheostomy tubes longer. for each patient. Study the effects of Participants: 3 men Respiratory and vocal parameters: (1) spirometry: abdominal trussing (24−41 years) with values were reduced based on normality as clinical treatment complete cervical spinal inspiratory capacity was the least reduced, and cord injury (C5−C6); expiratory reserve volume was the most reduced Procedure: a custom-built component of the vital capacity; (2) capacity of device was used to truss oral reading: not described (only the comparison the anterolateral with trussed and untrussed conditions) abdominal wall - the effects The results showed that abdominal trussing of trussing on lung volume increased vital capacity, presumably by and during oral reading optimizing diaphragm function. Longer utterance (average sound pressure duration for oral reading was observed with level, syllables per increased inspiratory capacity. utterance, pause duration) The findings of this study support clinical were studied. impressions that trussing can be used to improve speech in individuals with abdominal paralysis or paresis and diminished breathing function for speech. Describe a treatment Speech language pathology Respiratory and vocal parameters: not described; classification system clinicians developed a The SLP taxonomy consists of 7 intervention (taxonomy) developed taxonomy to provide a categories that address deficits - speech by speech language “system of rules and units” production for patients with artificial airway, pathologists to that would describe key motor speech and voice, swallowing, cognitiveexamine the effects of interventions in the communication, and communication - and the speech language rehabilitation of patients associated exercises and tasks that patients pathology (SLP) with SCI. perform. interventions on SCI Collaborative efforts of SCIRehab SLP rehabilitation technicians to develop the taxonomy provided outcomes. the opportunity to extensively share practice patterns and treatment methods. The clinicians identified multiple treatment inconsistencies, but also learned about differences in practice patterns. The different treatment approaches were consolidated into a practical number of taxonomic and the comprehensive SLP taxonomy for SCI rehabilitation was developed. Describe the nature and Participants: 600 patients with Respiratory and vocal parameters: not described; distribution of speech- traumatic spinal cord injury - 40% of patients received SLP treatment; of language pathology with different neurological these, 57% received intensive therapy; the activities during SCI level and injury integrity; remainder were primarily evaluated or received rehabilitation and Procedures: data was less intensive treatment. discuss predictors of collected from patient - cognitive-communication and swallowing the amount of time medical records. Speech therapy were the most common activities. spent in specific SLP language pathologists The significant variability in cognitive SLP treatment activities. entered details about treatment, which is not well explained by patient sessions times and activity and injury characteristics, sets the stage for descriptions were future research to associate treatments with considered. results.
(Continued)
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TABLE 2. (Continued ) Reference Country Journal
Objective
Nygren-Bonnieri et al20 Sweden Disability and Rehabilitation
Explore and describe decreased lung function in people with cervical spine injuries (CSCI).
Tamplin et al21 Australia Arch Phys Med Rehabil
Develop measurement techniques to quantitatively assess breathing patterns and respiratory muscle recruitment during singing and speech in quadriplegia.
Gordan et al11 United States The Journal of Spinal Cord Medicine
Describe associations of patient characteristics and speech-language pathology (SLP) interventions provided during inpatient SCI rehabilitation with outcomes at discharge and 1 year after injury.
Macbean et al7 Australia International Journal of SpeechLanguage Pathology
Examine changes in phonation and quality of life in the acute and sub-acute stages of recovery after cervical spinal cord injury (CSCI).
Methods
Results/Conclusion
Participants: 33 people with Respiratory and vocal parameters: note described; cervical spinal cord injury Experience related to decreased lung function (between levels C4 and C8); was grouped into three domains: limitations in Procedures: a semibreathing function, ability to cough and voice structured interview guide function. was constructed with the Breathing, the ability to cough and voice function focus on experience of were perceived as limited in most of the patients symptoms related to with CSCI. Most had adapted to the situation and decreased lung function. developed their own strategies for coping, but their limitations were not necessarily experience or referred to as a problem. Participants: six subjects Respiratory and vocal parameters: reduced lung with motor complete C5−7 capacity (vital capacity, 71% vs 102% of quadriplegia + six healthy predicted; P = 0.028); more perceived voice individuals problems (VHI score, 22.5 vs 6.5; P = 0.046), Procedure: a single 45−60greater recruitment of accessory respiratory minute assessment session muscle (loud and soft volumes) (P = 0.028); and (Questionnaire - Voice higher accessory muscle activation in changing Handicap Index (VHI); from soft to loud speech (P = 0.028); respiratory function The results showed that participants with (spirometry and respiratory quadriplegia exhibited the expected respiratory pressure meter); vocal dysfunctions and self-perceived vocal assessment (surface impairment. electromyography (EMGs), plethysmographya, audio data). Participants: 1376 patients Respiratory and vocal parameters: not described; with traumatic spinal cord Patients with SCI who also have traumatic brain injury; injury and therefore have the greatest need for Procedures: initial interventions to address cognitiveevaluation; cognitivecommunication limitations (CCLs) receive the communication treatment most SLP cognitive-communication treatment and swallowing exercises. and show the greatest improvement during Each session was rehabilitation according to the Functional documented by the speech Independence Measure (FIM), but cognitive language pathologists. functioning remained impaired at discharge. This likely explains the consistent finding that more hours of SLP treatment is associated with lower cognitive FIM scores at discharge. Participants: two patients Respiratory and vocal parameters (change was with cervical spinal cord present across measures for both cases): reduced injury + four control lung capacity (vital capacity, 80% and 40% vs 86, individuals; 41% of mean controls); more voice problems Procedure: participants (perceptual ratings of connected speech were were assessed in a quiet considered within normal limits for case one and setting, at three stages in case two speech to be mild − moderately during recovery: 1 week impaired across pitch, loudness, and voice quality after mobilization; variables, with the exception of pitch level 6 months after initial (severely impaired; maximum duration remained evaluation; 12 months after at the lower end of the normal range for case one initial evaluation (11 seconds) and in the case two was severely (spirometry; impaired (5.18 seconds); case one considered that electroglottography; the impairment of his voice rated as mild or mildcomputerized speech to-moderate, and voice function considered to be laboratory + effective and not having any substantial impact on questionnaires). his daily life, and case two considered his voice to be moderately impaired)
(Continued)
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TABLE 2. (Continued ) Reference Country Journal
Objective
Methods
Results/Conclusion In both cases change was observed over time in terms of the extent and pattern of recovery, with most deficiencies remaining relatively stable over the 12-month period. Subtle changes were observed in respiratory function, laryngeal function and phonation over time (case 1) and moderate to severe deficits in overall respiratory function and breathing patterns for speech, laryngeal function and phonation (case 2).
SCI, all the muscles below the damage are paralyzed, including respiratory muscles, with spasticity also compromising lung function.12,27,28 As such, patients with SCI may experience breathing difficulties and problems such as dysphagia and dysphonia.10 In that case an early tracheostomy should be considered, ensuring patient survival in the acute phase and improving respiratory condition in the chronic phase.29 Tracheostomy within seven days of intubation is recognized as a good treatment option because it may improve respiratory outcomes in patients with traumatic cervical spinal cord injury and it was also associated with fewer ventilator days, lesser time to decannulation, shorter ICU stays, and lower mortality.30−32 It has been reported that 11%−77 % of patients with cervical spinal cord injury require tracheostomy depending on the level of injury31,32 and long-term mechanical ventilation was required for 6.5%.33 McCully34 found that tracheostomy is unlikely in patients with incomplete SCI, regardless of the level of injury. Decannulation of patients with SCI can be a challenge considering that decannulation protocols vary based on the patient’s unique requirements and institutional policies. However, an earlier decannulation may suggest that early tracheostomy patients are more quickly achieving respiratory independence regarding cough strength and secretion management.29,35 In clinical practice cough effectiveness and ability to tolerate tracheostomy tube capping are the most considered parameters.35 Failure to decannulate and death due to respiratory dysfunction are common in patients with a C4 complete SCI because diaphragm dysfunction continues for a long period. Conditions such smoking history, age >45 years, comorbid lung diseases, and active pneumonia can make decannulation process more difficult.32,36 However there is an extremely high possibility of decannulation in C5−7 paralysis patients who can perform a shoulder shrug shortly after injury.32 The dysphagia is common in patients with spinal injuries. Respiratory dysfunction is a major cause of morbidity and mortality in SCI, compromising the muscles of respiration and reducing vital capacity, cough efficacy, and chest wall
compliance. Lung infections are a serious concern in spinal injuries and preventing pneumonia is essential to lowering mortality and shortening hospital stays. In addition, research on respiratory function in this population is important in order to provide more effective interventions.37−39 Reduced respiratory function after cervical spine injuries can indirectly result in voice disorders.12,19−21 Complete injuries cut off motor and sensory function below the level of damage, whereas some residual function remains in incomplete SCI. Cervical injuries between C1 and C3 severely limit respiratory function, while innervation of the diaphragm is partially maintained in SCI below C3; however, the intercostal and abdominal muscles are paralyzed, reducing vital capacity.12 Additionally, although passive expiration is preserved (since it occurs largely via elastic and not muscular force), forced expiration is generally significantly compromised. Although spinal injuries do not generally compromise laryngeal motor function or the mucous membrane of the vocal folds, impaired respiratory function can indirectly affect the voice due to the paralyzed intercostal and abdominal muscles, limiting lung volumes and capacities as well as subglottic pressure control.12 Lower lung capacities and respiratory pressure and significantly greater voice impairment (Voice Handicap Index) in quadriplegics. Electromyographic data indicated significantly greater activation of the sternocleidomastoids and diaphragm during high (compared to low) vocalization in controls and of the trapezius in quadriplegic patients.21 Atypical recruitment of these muscles indicates compensation in the phonation process.19−21 Additionally, abdominal wall paralysis or paresis in individuals with cervical spine injuries, as well as neurological disorders such as muscular dystrophy. The resulting decline in inspiratory capacity decreases vital capacity, compromising the respiratory function required for speechand affecting reading and prosody.19 Experience related to decreased lung function was grouped into three domains by Nygren-Bonnieri et al20 limitations in breathing function, ability to cough, and voice function. The predominant theme of all the participant’s interviews was that although the limitations were significant
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in most cases compared to the situation before injury, they were not necessarily experienced or referred to as a problem. This corroborates the fact that spinal cord injuries compromise respiratory capacity, affecting voice and speech functions. Vocal changes are not caused by dysfunctions in the glottis, but rather its aerodynamics. Factors that influence dysphonia include respiratory function, limited vocal range, reduced loudness, and compensatory changes in phonation among patients with SCI, prompting patients to avoid oral communication.24 Even incomplete cervical spine injuries can cause speech and voice changes, which negatively affect quality of life in these patients. The effect on speech production differs in accordance with the nature of the injury (level and integrity) and the presence of other contributing factors, such as vocal cord paralysis.7 Comorbid traumatic SCI and traumatic brain injury (TBI) can produce a complex clinical spectrum with devastating consequences and can be a major contributor and confounder to impaired cognition40,41 posing important challenges in the rehabilitation of a dual diagnosis SCI patient. Physical, cognitive, and/or emotional impairments are expected as a result from a TBI. Neuropsychological symptoms may result from brain injury and/or may be related to nonorganic factors.42 A high frequency of TBI co-occurring with traumatic SCI of 39.6% was reported40 but previous estimates have been ranging wide from 10% to 74%.42 Potential co-contributors include concomitant brain injury, psychological or somatic comorbidities, decentralized cardiovascular control, and sleep apnea.41,42 Noncompliance, inability to learn, maladaptive reactions to SCI, and poor motivation are neuropsychological symptoms of a missed TBI diagnosis that may be perceived during rehabilitation. It is possible to expect other cognitive symptoms like difficulties with concentration, attention, memory, word finding/speech, perception, information processing, and executive functions. Emotional symptoms may include irritability, liability, anxiety, and depression.42 The highest incidence rate of dual diagnosis reported are for those between 15 and 24 years old. Motor vehicle collisions were most likely to yield a comorbid TBI diagnosis, followed by falls. Patients with cervical and thoracic injuries showed a very similar frequency of comorbid TBI.40 A study conducted in the United States on the taxonomy of speech-language pathology in people with SCI demonstrated that speech disorders were more prevalent in the respiratory and laryngeal subsystems of speech production.5,10,11 Respiratory dysfunctions include shallow inspiration and/or shorter, less controlled expiration, whereas laryngeal injuries may manifest themselves in prosodic disorders related to speech rate, duration, stress, and/or accent patterns. Additionally, vocal cord paralysis can be caused by damage to the peripheral nerve of the superior and/or recurrent laryngeal nerve.10
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Based on a previously developed taxonomy for stroke rehabilitation, speech language pathology clinicians developed a taxonomy to provide a “system of rules and units” that would describe key interventions in the rehabilitation of patients with SCI. Time is recorded for each category, and supplementary information focuses on cueing needs and family involvement that help describe and guide intervention selection.10 The patients could receive intensive therapy (more than five sessions during rehabilitation) or punctual interventions (one to five sessions). At the intensive treatment, the patients received an average of 16.1 hours of SLP, and significant differences were observed in the time spent on each activity among neurological injury groups. The cognitive-communication and swallowing therapy were the most common activities. The patients with motor levels of injury at C1−C4 spent more time in swallowing therapy, whereas those with low tetraplegia and paraplegia focused the greatest percentage of time on cognitive-communicative work.5 Associations between patient characteristics and speechlanguage pathology (SLP) interventions provided during inpatient SCI rehabilitation with outcomes at discharge and 1 year after injury. SLP treatment factors explain a small amount of variation in Functional Independence Measure, participation and mood. Variation explained by treatment factors for cognitive outcomes at discharge increased when the patient group was more homogeneous. More time in SLP cognitive communication interventions had a negative relationship, while longer length of stay was positive.11 Based on the results of this review, research is still incipient and as a result, there are no specific assessment and treatment methods that target communication in this population. In this case, the future research and targeted clinical practice should be based on prior knowledge of speech-language pathology (SLP) in general and closing the gap in knowledge regarding SLP directly related to this population. Methods must be established for this specific intervention considering evidence-based practice to ensure better rehabilitation results in patients with SCI. Dysphagia is generally treated during the acute period of injury, with better established evaluation and treatment procedures. Given the severity of SCI and its traumatic impact on those affected, voice and speech become secondary complaints, but remain a vital part of the rehabilitation and inclusion process, contributing to providing patients with a new perspective on life. Speech is the most important form of human communication and social inclusion. As such, even when speech disorders go unnoticed or ignored by patients, proper care should be provided at all levels to ensure their health and quality of life.43 Several studies highlight the need for respiratory training in patients with SCI as a foundation for speech therapy. This is based on the idea that increasing respiratory capacity improves expectoration, thereby reducing cases of bronchoaspiration and enhancing aerodynamics to ensure better quality vocalization and clearer, more fluent speech. It is
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Journal of Voice, Vol. &&, No. &&, 2019
important to investigate which patients might benefit from this therapeutic approach, considering complete high-level injuries and the likelihood that the muscles in question will not be reactivated. In these cases, voice modulation and resonance should be considered as a means of compensating for the compromised airflow.44 Atypical muscle activation was observed during loud speech, indicating spontaneous compensation and raising the question of whether this pathway might be a therapeutic alternative or harmful behavior.21 In this respect, speech assessments using well-defined controlled methods are important in this population in order to provide medical staff with valuable information on patient prognosis and how best to meet their needs. Objective assessment and treatment techniques are needed and should be based on research that specifically targets this pathology.
4.
5.
6.
7.
8.
9.
CONCLUSION The vocal parameters of the patients with traumatic SCI can be described, considering the lack of information available. Disorders related to speech therapy vary in accordance with the type and level of injury. Individuals with SCI exhibit stomatognathic system disorders such as ineffective breathing, dysphagia, dysphonia, and speech disorders. Speech therapy is a relatively new field and in-depth research is needed to create assessment protocols and define treatment methods aimed exclusively at this population.
10. 11.
12.
13.
AUTHORS’ CONTRIBUTION 1. Study conception and design: FOR and LDM. 2. Data collection, analysis and interpretation: FOR, CAF, MSMSM and LDM. 3. Drafting the article or revising it critically: FOR, CAF, MSMSM and LDM. 4. Final approval of the version submitted for publication: LDMT.
14.
15.
16.
SUPPLEMENTARY DATA Supplementary data related to this article can be found online at doi:10.1016/j.jvoice.2019.12.013.
17.
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