You are on page 1of 13

Dysphagia

https://doi.org/10.1007/s00455-020-10137-8

ORIGINAL ARTICLE

Whiplash‑Associated Dysphagia and Dysphonia: A Scoping Review


D. B. Stone1,2,3   · E. C. Ward4 · S. R. Knijnik1,5,6 · H. Bogaardt1 · J. M. Elliott1,3,7

Received: 30 December 2019 / Accepted: 15 May 2020


© Springer Science+Business Media, LLC, part of Springer Nature 2020

Abstract
Swallowing and voice complaints after a whiplash injury have been observed and reported in several studies; however,
variability in study design complicates current understanding of whether dysphagia and dysphonia should be recognised as
potential adverse outcomes. A scoping review was conducted across six databases from 1950 to March 2019. A total of 18
studies were included for review. Data regarding study purpose, design, outcome measures, participant characteristics and
outcomes reported were extracted. Level of evidence (LOE) was assessed by the American Speech-Language Language
Association (ASHA)’s LOE system. All studies were exploratory, with 68% rated as poor (< 3) on quality ratings. Nearly half
(n = 6) were single case reports. Only three studies investigated some type of swallow-related outcome specifically within
the study aim/s. Incidence of swallow-related problems ranged from 2 to 29%, with unspecified complaints of “swallowing
difficulty”, “dysphagia” and fatigue and pain whilst chewing reported. Neither swallowing biomechanics nor the underlying
pathophysiology of swallow or voice complaints was investigated in any study. Four case studies presented post-whiplash
voice complaints; two of which described loss of pitch range. Others described hoarseness, loss of control and weak phona-
tion. Most studies only mentioned swallow- or voice-related deficits when reporting a wider set of post-injury symptoma-
tology and six did not describe the outcome measure used to identify the swallow and voice-related problems reported.
The existing literature is limited and of low quality, contributing to an unclear picture of the true incidence and underlying
mechanisms of whiplash-related dysphagia and dysphonia.

Keywords  Dysphagia · Deglutition · Dysphonia · Voice · Swallowing · Whiplash · Whiplash-associated disorders

Introduction

The sudden and rapid acceleration and deceleration forces


during a motor vehicle collision (MVC) may give rise to
Electronic supplementary material  The online version of this hyperextension and flexion of the cervical spine [1, 2] and a
article (https​://doi.org/10.1007/s0045​5-020-10137​-8) contains so-called “whiplash injury”. In the absence of spinal fracture
supplementary material, which is available to authorized users.

5
* D. B. Stone The Federal University of Health Sciences of Porto Alegre,
daniellebstone@gmail.com Porto Alegre, Brazil
6
1 The Pontifical Catholic University of Rio Grande do Sul,
Faculty of Medicine and Health, School of Health Sciences,
Porto Alegre, Brazil
The University of Sydney, Sydney, NSW, Australia
7
2 Department of Physical Therapy and Human Movement
Speech Pathology Department, Royal North Shore Hospital,
Sciences, Feinberg School of Medicine, Northwestern
Northern Sydney Local Health District, St Leonards,
University, Chicago, IL, USA
NSW 2065, Australia
3
Neuromuscular Imaging Research Laboratory, Kolling
Institute at the Northern Sydney Local Health District,
Sydney, Australia
4
School of Health and Rehabilitation Sciences, The University
of Queensland and Centre for Functioning and Health
Research (CFAHR) Metro South Hospital and Health
Services, Brisbane, QLD, Australia

13
Vol.:(0123456789)
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

and neurological deficit in most cases, the cardinal clini- The evidence base is currently insufficient towards accu-
cal feature of whiplash is neck pain and reduced mobility rately informing the nature or extent of dysphagia or voice
[3–5]. However, the presenting signs and symptoms known changes post-whiplash injury. In addition, patients with
as whiplash-associated disorders (WAD) include but are not WAD are not often assessed or managed by speech-language
limited to, widespread bodily pain, traumatic distress [6–8] pathologists (SLPs). However, if dysphagia and dysphonia
and dizziness and headache [3]. Importantly, up to 50% are found to be potential components of the clinical course,
of those exposed to whiplash following MVC experience then early identification and referral of at-risk patients to
persistent symptomatology [9]. The economic and public SLPs for further assessment and management may ultimately
health burden of WAD is profound [10, 11]. With a high and need to be incorporated into practice guidelines. The aim of
increasing prevalence of WAD worldwide [11], low-levels of this scoping review was to explore what is currently known
life satisfaction [8, 9] and poor treatment outcomes [12], the about swallow and voice complaints after whiplash and what
importance of contributing towards a better understanding of further research is needed to better understand these issues.
the patient-centred poor-recovery profile is greater than ever.
For over 25-years the Quebec Task Force (QTF) clas-
sification system of WAD [13] has guided assessment and Method
prognosis by dividing a range of symptoms into 5 grades;
grades 0–III reflecting no physical signs (0), neck stiffness Design and Preliminary Search
(I) and reduced neck range of movement and point tender-
ness (II) and grades III and IV encompassing more traumatic A rigorous scoping review design [28, 29] was selected due
cervical spine injury including fracture and absent reflexes. to (a) the substantial heterogeneity in the available body of
However, there remains a lack of general agreement about literature, (b) no single clinical question being addressed
the clinical usefulness of this classification [14]. Current and (c) an absence of any previous critique [29, 30]. In line
evidence highlights the heterogeneity of the clinical course with indications for scoping review over systematic review
[7, 15], demonstrating a myriad of physical and psycho- by Munn et al. [30], this study aimed to determine the scope
logical impairments in those that transition from acute to of literature covering swallow and voice problems related
chronic WAD-related disability [8]. Although not formally to whiplash and provide an overview of current themes and
recognised within the QTF classification system, emerging gaps. The scoping review was conducted in accordance with
evidence suggests that voice and swallow complaints may the Preferred Reporting Items for Systematic Reviews and
be among the potential manifestations of under-recognised Meta-Analyses extension for Scoping Reviews (PRISMA-
features from whiplash [16]. ScR) guidelines [28]. However, prior to conducting the
Previous studies have described voice and swallow prob- final literature search, an additional step involving a pre-
lems in acute and chronic whiplash [16–18]; however, dis- liminary literature search was conducted to develop the final
parity exists in the outcomes used to measure and report search terms and inclusion/exclusion criteria. Examination
these problems. As such, little is known about the incidence of known literature revealed issues relating to swallowing
and pathophysiological mechanisms. Alterations in oro- and voice deficits were often only secondary reports within
pharyngeal volume have been reported in individuals pre- highly varied study methodologies. Hence in this prelimi-
senting with poor recovery compared with those nominating nary step, known key papers were identified and their con-
recovery and healthy control participants [19, 20]. Albeit tent and reference lists searched, to determine all possible
preliminary, a conceivable link between self-reported change keywords related to whiplash and the types of studies with
in post-traumatic swallow with oropharyngeal volume has potential to encompass information on any related swal-
been offered [16]. The potential impact of a post-traumatic low or voice-related problem. For example, the preliminary
muscular tensioning has also been raised as an underlying search revealed a large body of literature investigating jaw
aetiology in patients with an idiopathic functional dys- range of motion (ROM) and pain, temporomandibular joint
phagia [21]. Considering the likely impact of pharyngeal (TMJ) biomechanics and the impact of collisional forces on
and laryngeal tensioning on swallow function [22–24], a jaw function. As jaw dysfunction may have an associated
whiplash-induced muscle tension dysphagia and/or dys- impact on bolus mastication, this was included in the final
phonia is plausible. Dysphagia in patients with chronic pain search terms.
(e.g. fibromyalgia) has been previously suggested [25] and
swallowing and voice symptoms have been described in Eligibility Criteria
laryngeal hypersensitivity syndromes [26, 27]. With this in
mind; swallow and voice deficits following whiplash may From the information gained from the preliminary search
contribute to the extensive symptom profile in those with strategy, the study’s final inclusion and exclusion criteria
poor recovery. were determined a priori and are detailed in Table 1.

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Table 1  Inclusion/exclusion criteria
Inclusion Exclusion

P Adults > 18 years with whiplash injury or exposure (aka: cervical Adults whose primary condition is not whiplash injury or exposure
flexion extension, cervical hyperextension-hyperflexion, cervical Individuals requiring the following as a result of whiplash injury:
hyperextension, cervical hyperflexion, cervical acceleration-decel-  Spinal surgery
eration, cervical deceleration, cervical strain, whiplash-associated  Tracheostomy
disorder/s, whiplash syndrome or mandibular whiplash)  Intubation
(Note: If voice or swallow complaints were reported to be of concern
prior to tracheostomy or intubation intervention, the study was
included)
Traumatic brain injury or other significant (non-direct nerve injury)
neurological impairment following whiplash
Spinal or jaw fracture
History of traumatic brain injury, stroke or other neurological
disease/disorder; disability, head and neck cancer, head and neck
surgery or gastro-oesophageal disease
Children < 18 years and Infants
I n/a n/a
C n/a n/a
O Presenting with one or more of the following symptoms: None of the following: dysphagia, swallow deficit, difficulty chewing/
 Dysphagia masticating, odynophagia, jaw or orofacial pain, trismus
 Swallowing/deglutition disorder/deficit None of the following: dysphonia, voice change, voice disturbance,
 Muscle tension dysphagia vocal hoarseness
 Difficulty chewing/mastication Jaw range of movement and/or pain unrelated to swallow-related
 Odynophagia function
 Trismus/reduced jaw range of movement/jaw fatigue explicitly
associated with any chewing or swallow-related function/s
Jaw pain if explicitly associated with any chewing or swallow-
related function/s
 Dysphonia
 Voice change
 Voice disorder/disturbance
 Vocal hoarseness, aphonia, vocal strain
Languages other than: English, Dutch, German, French, Portuguese,
Spanish
D Complete scientific articles Review articles
Single case reports Book chapters
Commentaries
Letters to the editor

PICO participant, intervention, comparison, outcome, design

A search strategy was designed in Medline and is Scoping Review


detailed in Online Appendix  1. Searches were subse-
quently modified for each database. The following main An electronic literature search of all articles published
subject headings were used: whiplash injuries, mandibular between 1950 and March 2019 was conducted independently
whiplash, deglutition, deglutition disorders, voice, laryn- by two authors (DS, SRK) on 29 March 2019. Databases
geal diseases and voice disorders. Additional search terms included PUBMED, MEDLINE, SCOPUS, Web of Science,
included: swallowing, swallow, pharynx, oropharynx, lar- EBSCO, and EMBASE. Reference lists of all selected arti-
ynx, dysphagia and dysphonia. To capture all potential cles were manually searched, with forward and backward
swallow and voice deficits in the absence of a previously citation chasing, for any relevant publications. Deduplication
established whiplash-associated dysphagia or dysphonia, was conducted using the Bramer method [31].
the search strategy was inclusive of any possible deficit Following deduplication, one reviewer (DS) screened
relating to swallow and/or voice, including structures such titles for inclusion. Articles where titles were unrelated to
as cranial nerves and swallow musculature. any inclusion criteria were excluded. Two reviewers (DS,

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

SRK) independently screened the eligibility of remaining Results


articles based on abstract screen. In the case where screening
was unable to determine if a title and/or abstract met crite- Initial search yielded a total of 1758 articles for review as
ria, the article was selected for full-text review. Independent detailed in the PRISMA flow diagram (Fig. 1). Independent
full-text review was conducted by the same two reviewers review was conducted for 57 full-texts. A total of 18 arti-
and assessed based on inclusion/exclusion criteria. In cases cles met inclusion/exclusion criteria and were included for
where the reviewers’ opinions differed for either abstract or review and summarised in Table 2.
full-text review, a third reviewer (HB) was used to achieve
consensus.
Quality Rating

Data Extraction and Synthesis Quality indicator ratings for all parameters, overall quality
rating (total score/7) and stage of research are detailed in
Data including study purpose, design, population (N), time Table 3. All 18 papers were categorised within the explora-
post-whiplash, WAD Grade [32] if specified and outcome tory stage of research as per ASHA LOE system guidelines
measure/s were extracted from each eligible study. If WAD [33]. Six of the 18 papers were single case reports, whilst
grade was not specified, level of injury was reflected in five utilised a case control design. Median quality marker
the inclusion criteria from subject description within the was 2/7, indicating a low level of quality for most studies.
article and was not specifically specified. For the purposes
of clearly synthesising findings, papers were stratified as
Patient Populations
either swallow- or voice- related based on the outcomes or
symptoms described. As no study specifically explored dys-
A total of 15,909 individuals aged between 17 and 75 years
phagia, a wide range of terms that encompassed “swallow-
were described across the 18 included studies (Table 2).
related activities/problems” were accepted, consistent with
Two studies investigated over 7000 participants [34, 35].
the nature of a scoping review. Incidence of dysphagia was
There was significant heterogeneity in the time post-whip-
recorded from studies where an overall percentage was cal-
lash; ranging from immediately following initial injury to
culated. Due to the heterogeneity of outcomes reported, all
9-years post. Most individuals sustained whiplash from an
swallow and voice complaints were listed from each paper
MVC. Only two studies [36, 37] included a small sample
and summarised descriptively.
of subjects who sustained injury from a fall. Seven stud-
ies specified WAD grade as determined by the QTF Task-
Methodological Quality force [32], with the majority of these (n = 5) reporting WAD
grades II–III. For most studies, extent of whiplash injury was
The quality of eligible studies was evaluated using the reflected in the inclusion criteria due to a lack of specifica-
American Speech-Language and Hearing Association tion or grading.
(ASHA)’s Levels of Evidence (LOE) rating tool [33]. The
system was created to determine the extent to which a study
Purpose of the Studies
meets a range of quality indicators and designed specifically
for use in non-randomised studies, including single-case
Six studies [21, 38–42] described a single case presentation.
designs. Considering the types of studies included in this
Of these, 3 [39, 41, 42] described voice-related problems,
review, this system was considered most suitable [33]. Two
two reported pain or sensory complaints related to swal-
independent reviewers (DS and SRK) blindly rated each eli-
lowing [21, 40] and one case described a combination of
gible study across eight indicators, including; study design,
swallow- and voice-related difficulties [38]. One study exam-
blinding, sampling, participant comparability, outcomes,
ined eating behaviours following whiplash as the primary
significance, precision and intention to treat. Studies were
aim [18]. Two specifically investigated chewing behaviour
given one point per indicator if the indicator met the high-
after whiplash [37, 43]. A total of four studies [17, 36, 44,
est descriptor; reflective of a higher quality paper. The final
45] examined the characteristics of jaw-related dysfunction
parameter, intention to treat, is only applicable to controlled
through a range of jaw measurements and symptom-based
studies and therefore in this study, a final quality marker
questionnaires. All these utilised a measure of chewing
maximum score of seven was possible [33]. Each study was
behaviour as part of their battery of assessment, however
then characterised by research stage as either effectiveness,
without explicit investigation of chewing as a primary focus.
efficacy, exploratory or policy [33]. Ratings were compared
The remaining studies examined a larger set of post-injury
between reviewers, with consensus achieved after combined
characteristics to investigate the systemic nature of WAD
review in the case of any discrepancy.

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Fig. 1  Prisma chart Records identified through


database search

Identification
(n=1758)

Records after duplicates


removed (n=974)

Screening Records excluded

Records screened (n=921)

(n=974) - Title (622)


- Abstract (295)
- Language (4)

Full-text articles assessed for Full-text articles excluded


Eligibility

eligibility
(n=39)
(n=53)
- No swallow or voice outcomes; unclear
until full-text review (20)
- Jaw articles unrelated to swallow or
chewing, not obvious on initial screening
(5)
Grey Literature - Review article, incomplete article or book
chapter (5)
(n=4) - Subjects not only whiplash (4)
- > WAD Grade II (3)
Inclusion

- Tracheostomy or other acute/severe injury


(2)
Articles included for
synthesis
(n=18)

[18, 34, 46], the rate of recovery across a wide set of symp- swallow-related difficulties and included: The Jaw Disabil-
toms [47] or the relationship between symptoms and acci- ity Checklist [50], a standardised patient-reported outcome
dent mechanisms [48]. None of the selected studies spe- measure in one study [44], measurement of mandible move-
cifically investigated dysphonia as a specific study aim. No ments [36] or amplitude of masseter muscles via electro-
study investigated pharyngeal swallow biomechanics. myography (EMG) [43] during chewing and a timed chew-
ing endurance test [36, 37, 43, 44] (Table 5). Self-reported
Swallowing‑Related Complaints After Whiplash questionnaires designed for the purpose of the study where
swallow-related behaviour was a component of a wider set
There were 16 studies which reported swallow-related of questions related to post-injury symptoms were used in
complaints after whiplash, though more of half [17, 34, 36, a further seven studies [17, 18, 34, 37, 44, 45]. Six studies
38, 44, 46–49] included this information as part of wider did not utilise any outcome measure [21, 38, 40, 46, 48, 49].
investigations into post-injury sequalae or within broader Specifically, six studies specified an incidence rate
investigations related to jaw dysfunction. Swallowing- for different swallow-related problems occurring within
related issues were described using various terms includ- their cohort, ranging from 2 to 29% [17, 47], see Table 6.
ing: problems eating, dysphagia, swallowing difficulty, However, due to the differences in the descriptors used to
odynophagia, perceived pain on chewing and food avoidance calculate incidence, no average across the studies can be
(Table 4). The most frequently described swallow-related calculated. Three of these studies [46, 48, 49] reported inci-
deficit was problems related to chewing, closely followed dence of swallow-related deficit with neither case report or
by the broad descriptor “swallowing difficulty”. Of these, outcome measurement, where swallow-related complaints
only seven reported a specific tool used to identify these were listed purely in a data table summarising a range of

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Table 2  Eligible studies
Study (year) N Age (mean) years Time post- WAD grade Healthy con- Swallow- Voice-related
whiplash* trol group related

Anagnostara44 (2005) 1 58 1–14 d n/s No X x


Bordoni21 (2016) 1 30 6 m II No X
Brademann45 (1998) 1 58 1–3.5 m n/s No x
Carroll17 (2007) 7763 18 years or over  > 1 m n/s No X
Chirchiglia46 (2019) 1 42 6 m n/s No X
Ferrari40 (2005) 7462 37.34 (mean, male); 1 m n/a No X
36.83 (mean, female)
Gargan53 (1994) 50 32 (mean) 5 d–2 y n/s No X
Gronquist18 (2008) 50 20–68  > 6 m II–III Yes X
Haggman42 (2002) 12 28–50 1–9 y II–III Yes X
Haggman43 (2004) 50 17–58 1–9 y II–III Yes (and non- X
whiplash
TMJ**)
Helliwell47 (1984) 1 64 0–18 m n/s No X x
Hildingson55 (1989) 35 19–67 4–14 d n/s No X
Kalezic49 (2010) 21 39 (mean) 3–13 y II–III Yes X
Lampa50 (2017) 80 34.1 (mean)  < 1 m 0–III Yes X
Pennie52 (1991) 144 n/s n/s n/s No X
Severinsson51 (2010) 119 19–75 1 m, 1 y 0–III No X
Sturzenegger54 (1995) 117 19–51 1y n/s No X
Waddell48 (2005) 1 27 56 m WAD No x
Totals 15,909 16 4

n/s not specified, d days, w weeks, m months, y years


*Time post-whiplash
**temporomandibular joint dysfunction—WAD group compared to a group of participants with TMJ dysfunction unrelated to whiplash

symptoms but not elaborated on. Furthermore, three studies studies [37, 43] reported up to one quarter [37] to more than
proposed relationships between swallowing issues and other half [43] of individuals exposed to whiplash discontinued a
symptoms or time post-injury. Carroll et al. [17] reported chewing endurance task due to self-reported fatigue. Self-
that people self-reporting difficulty swallowing after whip- reported pain was significantly higher and onset of fatigue
lash were 3.75 times more likely to also experience jaw pain, was significantly earlier than healthy controls [37]. Further
while a study investigating jaw symptoms in the 1st year to this, Kalezic et al. [43] demonstrated a significant increase
after whiplash observed difficulty chewing increased from in autonomic responses of heart rate and arterial blood pres-
4 to 10% of women over a 12-month period. Finally, Gar- sure during the chewing endurance test.
gan et al. [47] reported observing a 16% incidence of self-
reported “dysphagia” within 7 days of injury in their cohort Voice‑Related and Sensory‑Related Complaints
of 50, noting that this reduced to only 2% after 3-months. After Whiplash
In the most explicit of the three studies investigating
swallow-related difficulties related to whiplash as a primary Of the 18 studies, four reported on voice-related outcomes
aim, Gronquist et al. [18] compared 50 WAD and 50 con- and these are detailed in Tables 5 and 7. Additionally, sen-
trol participants and pre- and post-injury complaints, to test sory symptoms following whiplash were highlighted in sev-
the hypothesis that “eating behavior” would be impaired eral studies, including a unilateral burning sensation on the
following whiplash. A significant difference was found in tongue [40], unilateral clicking sensation in the larynx [42]
self-reported difficulties with jaw opening, biting, chewing and pain on swallowing or jaw opening [18, 44].
and swallowing and increased reports of; avoidance of tough
food, requiring smaller pieces, needing breaks during meals, Underlying Mechanisms
aborting meals and social impact, between those exposed
to whiplash and healthy controls (p < 0.0001) and between Potential underlying mechanisms for the swallowing and
pre- and post-injury complaints (p < 0.0001). The two other voice issues observed were discussed in many studies [18,

13
Table 3  Levels of evidence of eligible studies
Study (year) Design Assessor Sampling Subjects Outcomes Significance Precision Intention to treat Quality Research stage
blinding marker score/7

Anagnostara (2005)44 Case study No No Yes Yes No No N/A 2 Exploratory


Bordoni (2016)21 Case study No No Yes No No No N/A 1 Exploratory
Brademann (1998)45 Case study No No Yes Yes No No N/A 2 Exploratory
Carroll (2007)17 Cross sectional No Yes Yes No Yes Yes N/A 4 Exploratory
Chirchiglia (2019)46 Case study No No Yes No No No N/A 1 Exploratory
Ferrari (2005)40 Cohort No No Yes Yes Yes No N/A 4 Exploratory
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Gargan (1994)53 Cohort Yes No Yes No Yes No N/A 4 Exploratory


Gronquist (2008)18 Case control study No No Yes No Yes No N/A 2 Exploratory
Haggman (2002)42 Case control study No No Yes Yes Yes No N/A 3 Exploratory
Haggman (2004)43 Case control study No No Yes Yes Yes No N/A 3 Exploratory
Helliwell (1984)47 Case study No No No Yes No No N/A 1 Exploratory
Hildingson (1989)55 Case series No No No Yes No No N/A 1 Exploratory
Kalezic (2010)49 Case control study No No Yes Yes Yes Yes N/A 4 Exploratory
Lampa (2017)50 Case control study Yes No Yes Yes Yes No N/A 5 Exploratory
Pennie (1991)52 Case series No No No Yes Yes No N/A 2 Exploratory
Severinsson (2010)51 Cohort No No Yes Yes Yes Yes N/A 5 Exploratory
Sturzenegger 1995)54 Case series No No Yes No Yes Yes N/A 3 Exploratory
Waddell (2005)48 Case study No No Yes Yes No No N/A 2 Exploratory

ASHA level of evidence rating system


Blinding—assessors blinded or not blinded; Sampling—random sampling adequately OR inadequately described or convenience sample described or not described; Subjects—groups compa-
rable or not comparable OR subjects described or not described; Outcomes—at least one primary outcome measure is valid or reliable or validity unknown/appears reasonable or invalid/unreli-
able; Significance—P-value reported; Precision—effect size or confidence interval described

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Table 4  Negative swallow-related outcomes following whiplash


Study Problems Dysphagia Swallowing Pain related to swal- Chewing Food avoidance Difficulty Tongue pain
eating difficulty low-related behaviour problems* coughing

Anagnostara44 √
Bordoni21 √ √
Carroll17 √
Ferrari40 √
Gargan53 √
Gronquist18 √ √ √ √ √
Hildingson55 √
Lampa50 √ √
Pennie52 √
Sturzenegger54 √ √
Kalezic49 √
Haggman42 √
Haggman43 √
Helliwell47 √
Severinsson51 √
Chirchiglia48 √
Totals: N 1 4 5 3 8 1 1 1

Swallow-related outcomes as described by each study


*Self-reported pain and/or fatigue during chewing, longer duration during chewing task or premature discontinuation of chewing task

21, 38–41, 43], however not specifically investigated in any inflammatory status. Four studies [36, 37, 43, 44] sug-
of the 18 studies included in this review. Anagnostara et al. gested a relationship between neck injury and chewing
[38] described swallowing difficulty and vocal hoarseness in problems after whiplash, however did not discuss potential
a case of retropharyngeal hematoma post-whiplash. Oedema underlying physiology.
of the pre-vertebral space and bulging of the posterior phar- For voice-related problems after whiplash, an underlying
yngeal wall was found at the level of the tongue base on superior laryngeal nerve (SLN) paresis was considered a
computed tomography (CT) of the neck, which the authors possible cause of loss of pitch and vocal control and weak
reported was suggestive of muscular tearing of the longus phonation in one case study [39]. Bilateral vocal fold palsy
colli muscles [38]. They also reported anterior displacement was identified as the cause of weak phonation in a second
of the arytenoid cartilages, compression of the parapharyn- case [41]; considered to be a result of vagal nerve palsy from
geal space and suggested involvement of pharyngeal muscles shearing and stretching forces at the time of acceleration and
after whiplash, given their location at the anterior border of deceleration motions following impact.
the retropharyngeal space. The physiology underlying both sensory and motor com-
Several studies [18, 21, 36, 37, 45] discussed the func- plaints following whiplash was also discussed. Bordoni et al.
tional relationship between the tongue, atlanto-occipi- [21] discussed widespread sensory hypersensitivity as a
tal joint, cervical joints and hyoid bone after whiplash. potential mechanism of tongue tension and pain, specifically,
Gronquist et al. [18] supposed jaw dysfunction may be hyperexcitability in the central nociceptive pathways via the
the result of impaired coordination between jaw and neck trigeminal reflex [21], where myofascial release was pro-
muscles and joints and suggested eating behaviours may posed to reduce trigger point activity in the cervico-occipital
be impaired due to morphological alterations of the oro- region. Hyperexcitability of the autonomic nervous system
pharyngeal region after whiplash. Bordoni et al. [21] pro- was also used to explain the increased autonomic responses
posed cervical and oropharyngeal tensioning as potential observed during a chewing endurance test [43] and likened
factors impacting the tongue and described a reportedly to the central drivers seen in chronic pain, where the spread
successful case of osteopathic treatment and myofascial and maintenance of pain leads to lowered pain and sensory
release of the tongue and hyoid region. Improvement in thresholds. To explain the observations of pain during chew-
self-reported cervical pain on swallowing following treat- ing, Haggman et al. [37] discussed the coupling of the jaw
ment was considered the result of restoration of physi- and neck sensorimotor system as a mechanism for the spread
ological length in the fascial system and reduction in of pain and musculoskeletal dysfunction,

13
Table 5  Outcome measures in eligible studies
Swallow-related outcomes Voice-related outcomes
Study Self-reported Standard Mandibular Measure- EMG Osteopathic Timed Case report No Voice Nasendoscopy Case report No
questionnaire PROM ments during chewing during assessment chewing outcome range outcome
chewing test measure profile measure

Anagnostara44 √ √ √ √
Bordoni21 √ √ √
Brademann45 √ √ √
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Carroll17 √
Chirchigilla46 √ √
Ferrari40 √
Gargan53 √
Gronquist18 √
Haggmann42 √ √
Haggmann43 √ √
Helliwell47 √ √ √ √
Hildingson55 √
Kalezic49 √ √
Lampa50 √ √ √
Pennie52 √
Severinsson51 √
Sturzenegger46 √
Waddell48 √ √ √
Totals: N 7 1 1 1 2 4 3 6 1 2 4 3

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

Table 6  Incidence of swallow-related difficulties Due to large methodological differences, at present the


Study N Swallow-related Incidence (N) %
incidence of swallowing-related issues remains unclear.
descriptor Based on the two large cohort studies, it is possible that
swallowing-related issues may occur for one in every 4 or
Carroll17 5999 Swallowing difficulty 359a 6
5 individuals [17]. However, all studies varied widely in
1128 319b 28.4
sample size, nature and time post-injury and used a variety
Hildingson55 35 Dysphagia 2 5.7
of different and non-specific terms to describe complaints
Gargan53 50 Dysphagia 8c 16
(e.g. “difficulty swallowing”, “problems eating”). Many
1d 2
studies also failed to report exactly how swallowing issues
Pennie52 144 Dysphagia 18 27
were detected, thus limiting any comparison of results
Sturzenegger54 28 Dysphagia 2e 7
across studies.
117 10f 16
In the studies identified there was no specific investi-
Ferrari40 7462 Swallowing difficulty 248g 8.5
gation of swallowing biomechanics conducted. As such,
477h 10.5
there is little information to help clinicians understand how
a
 No jaw pain many people may present with a swallowing deficit or the
b
 New onset jaw pain nature of swallow-related impairments. From the data cur-
c
 Within 7-days rently available there is some early evidence to suggest that
d
 After 3-months issues with jaw function and the impact on chewing may be
e, g
 Male a component of such problems. However, in order to deter-
f, h
 Female mine what other factors may be contributing to eating and
swallowing behaviours as a consequence of whiplash injury,
more rigorous inquiry using validated tools and instrumental
Discussion measures of swallowing are needed. Furthermore, includ-
ing routine assessments of swallowing in future prospec-
A scoping review was conducted to broadly examine the tive investigations would help provide clearer information
nature and incidence of dysphagia and dysphonia follow- regarding symptom presentation and its relationship to the
ing whiplash. A total of 18 studies described a range of clinical course.
swallow and voice-related disturbances; however, for the The review highlighted voice problems post-whiplash
majority these were not the specific focus of the research, across four individual case studies, with a range of param-
often embedded within the reporting of a wider set of eters described and varied mechanisms proposed. Interest-
clinical signs and symptoms. Based on this review of ingly, loss of pitch range was reported in two of four stud-
the existing literature, it can be concluded that the cur- ies [39, 42] and could represent a potential area of future
rent understanding of the incidence, characteristics and/ investigation, particularly given the proposal of a potential
or causes of whiplash-related dysphagia and dysphonia is superior laryngeal nerve involvement [39] and other sugges-
extremely limited. tions of nerve injury [41]. Although spasmodic dysphonia
and muscle tension dysphonia were suspected as an under-
lying mechanism [42], there were no specific perceptual

Table 7  Negative voice-related Study Inci- Outcome measure


outcomes following whiplash dence N
(%)
Self-reported voice complaint Nasendoscopy Imaging

Helliwell47 1 Weak phonation Bilateral vocal fold immobility –


Waddell48 1 Loss of control – –
Break in register in singing
Brademann45 1 Loss of head voice Glottal closure –
Poor vocal fold lengthening,
Sagging vocal fold appearance
Anagnostara44 1 Hoarseness – Arytenoids
displaced
anteriorly

– Outcome measure not used for this study

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

or acoustic voice parameters described and no delineation in reaction to increased chewing load [43] were suggestive
made between these two presentations to confidently make of hypersensitivity and lowered pain and sensory thresholds.
this assumption. With such a small number of participants Given that a cardinal feature of chronic WAD is widespread
and heterogeneity in the measuring and reporting of voice pain and higher pain intensity [56] and that dysphagia and dys-
outcomes in the selected studies of this review, this review phonia are typical manifestations seen in laryngeal hypersensi-
cannot confidently conclude that a post-whiplash dyspho- tivity syndromes [26] the possibility of centrally-driven causal
nia exists. Despite this, the available literature points to or contributing factors in post-whiplash swallow and voice
four individuals who self-reported a range of voice prob- complaints is plausible and worthy of further consideration.
lems and despite this, no study in this review specifically Overall, multiple issues were identified with the quality
set out to examine voice function. Future work is needed, of the existing literature, which was rated as low for most
utilising valid and reliable perceptual, acoustic and instru- studies. Over 30% of the included studies were single case
mental measurement to determine whether voice changes reports, where a range of descriptors suggested voice and/or
after whiplash is perhaps an under-recognised feature of the swallow problems were a direct consequence of injury, how-
chronic recovery profile and whether they share a similar ever without use of reliable and valid measurement tools. As
underlying pathophysiology to swallow complaints. observed in the analysis of the 18 papers, there was great
In the existing literature, swallow-related problems con- variability in symptom presentation and the study designs
cerning chewing, fatigue, pain and food avoidance were failed to detail nature and severity across those injured or the
described, and changes in voice quality and function were temporal expression and duration of symptoms across the
also observed. In most studies, several explanations were clinical course. The degree of WAD was also not specified in
proposed to explain these complaints. However as yet, no most studies and this proves a challenge when trying to make
study has specifically explored underlying cause and the comparisons and draw clinical inference into potential risk
evidence base remains at the level of clinical opinion only. factors. These and other methodological weaknesses need to
Many of the causal mechanisms proposed to date appear be systematically addressed in future studies. This scoping
plausible but further research in this area is needed. For review highlighted the need for robust, primary investiga-
example, the suggested impact of pharyngeal and/or lon- tion into swallow and voice deficits following whiplash to
gus colli muscle involvement in whiplash trauma [38] and elucidate these interesting preliminary findings.
the subsequent impact on swallowing could be grounds
for future work. Although the longus colli is not typically Limitations
considered part of the swallowing musculature, dysphagia
and odynophagia are described as key symptoms of longus Given the limited information on swallow and voice defi-
colli tendonitis [51, 52]. The longus colli also has a role in cits reported in the selected studies, where many descrip-
maintaining the cervical spine lordotic curve [53]. Given tors were embedded within larger data sets, the authors
the incidence of swallowing difficulties in cases of curvature acknowledge the potential of having missed some informa-
change [54], its’ contribution to post-whiplash dysphagia is tion reported in papers with similar, brief and heterogenous
plausible [53] but not yet examined. reporting. Despite this, best efforts were made to include
The link between the jaw, cervical spine and swallow all possible terms across our literature review and robustly
musculature was also suggested in several studies [18, 21, search the known literature. Also, to avoid inclusion of
36, 37, 45], though unsupported by any experimental inves- a suspected large number of studies outside the scope of
tigations. The whiplash event may render some tissues vul- this review, studies of post-whiplash jaw dysfunction were
nerable to injury [55]. For example, the thyroid cartilage excluded if unrelated to swallow or chewing deficits. In
is anterior to the sixth cervical vertebrae, where excessive doing so, the authors acknowledge the chance of missing
stretch of the anterior soft tissue of the neck during the information that may have been embedded in the excluded
injury event may result in nerve injury [39]. Further inves- studies. However, full-text review was conducted on any title
tigation into swallow biomechanics post-whiplash and the or abstract where this was unclear and so this risk was mini-
possible association with biomechanical consequences of mised as much as possible.
cervical spine injury is needed to further our understanding
of post-whiplash swallow or voice problems.
Finally, the potential for a centrally-driven component Conclusion
to explain swallow or voice-related problems provides an
interesting area for future exploration. Sensory disturbances The studies in this review highlighted a range of swallow-
in the tongue [21, 40], larynx [42] and/or jaw [18, 44] have and voice-related deficits following whiplash. However, the
been described, albeit briefly, but have not been systemati- study designs and heterogeneity across the available litera-
cally investigated as yet. Heightened autonomic responses ture provides a challenge in determining the true incidence

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

and nature of whiplash-associated dysphagia and/or dys- 14. Kivioja J, Jensen I, Lindgren U. Neither the WAD-classifi-
phonia. Further studies using standard and instrumental cation nor the Quebec Task Force follow-up regimen seems
to be important for the outcome after a whiplash injury. A
measures of swallow and voice function, implemented pro- prospective study on 186 consecutive patients. Eur Spine J.
spectively over time in large clinical cohorts are needed to 2008;17(7):930–5.
make headway in this field. Understanding the underlying 15. Elliott JM, et  al. Characterization of acute and chronic
biomechanics and pathophysiology of the swallowing and whiplash-associated disorders. J Orthop Sports Phys Ther.
2009;39(5):312–23.
voice changes is also critical to help inform the nature of any 16. Stone D, et  al. Whiplash-associated dysphagia: considera-
interventions. Until such data are available it is difficult to tions of potential incidence and mechanisms. Dysphagia.
determine whether swallow and voice assessment and any 2019;35(3):403–13.
intervention for these issues, is indicated in the management 17. Carroll LJ, Ferrari R, Cassidy JD. Reduced or painful jaw move-
ment after collision-related injuries: a population-based study. J
of this population. Am Dent Assoc. 2007;138(1):86–93.
18. Gronqvist J, Haggman-Henrikson B, Eriksson PO. Impaired jaw
Acknowledgements  The authors would like to acknowledge the exper- function and eating difficulties in whiplash-associated disorders.
tise of Ms Nicola Wormleaton, Librarian at Royal North Shore Hos- Swed Dent J. 2008;32(4):171–7.
pital and Ms Yulia Ulyannikova, Librarian for the Faculty of Health 19. Elliott JM, et al. Magnetic resonance imaging changes in the size
Sciences, The University of Sydney. Their expertise in assistance with and shape of the oropharynx following acute whiplash injury. J
search strategy design was greatly appreciated. Orthop Sports Phys Ther. 2012;42(11):912–8.
20. Elliott J, et al. MRI study of the cross-sectional area for the cer-
vical extensor musculature in patients with persistent whiplash
associated disorders (WAD). Man Ther. 2008;13(3):258–65.
21. Bordoni B, Marelli F, Morabito B. The tongue after whiplash:
References case report and osteopathic treatment. Int Med Case Rep J.
2016;9:179–82.
1. Gay JR, Abbott KH. Common whiplash injuries of the neck. J Am 22. Kang CH, Hentz JG, Lott DG. Muscle tension dysphagia: sympto-
Med Assoc. 1953;152(18):1698–704. mology and theoretical framework. Otolaryngol Head Neck Surg.
2. Crowe H. A new diagnostic sign in neck injuries. Calif Med. 2016;155(5):837–42.
1964;100:12. 23. Depietro JD, et al. Laryngeal manipulation for dysphagia with
3. Merrick D, Stålnacke B-M. Five years post whiplash injury: symp- muscle tension dysphonia. Dysphagia. 2018;33(4):468–73.
toms and psychological factors in recovered versus non-recovered. 24. McGarey PO Jr, et al. Comorbid dysphagia and dyspnea in muscle
BMC Res Notes. 2010;3(1):190. tension dysphonia: a global laryngeal musculoskeletal problem.
4. Falla D, Bilenkij G, Jull G. Patients with chronic neck pain dem- OTO Open. 2018;2(3):2473974X18795671.
onstrate altered patterns of muscle activation during performance 25. Umay E, et al. AB1063 swallowing difficulty in fibromyalgia: real
of a functional upper limb task. Spine. 2004;29(13):1436–40. or myth? Ann Rheum Dis. 2018;77(s2):1643.
5. O’Leary S, et al. Muscle Dysfunction in cervical spine pain: 26. Vertigan AE, Bone SL, Gibson PG. Laryngeal sensory dys-
implications for assessment and management. J Orthop Sports function in laryngeal hypersensitivity syndrome. Respirology.
Phys Ther. 2009;39(5):324–33. 2013;18(6):948–56.
6. Eather A, et al. How are pain and traumatic stress symptoms 27. Caporali C, et al. Early-onset movement disorder as diagnostic
related in acute whiplash associated disorders? A investiga- marker in genetic syndromes: three cases of FOXG1-related syn-
tion of the role of pain-related fear in a daily diary study. Pain. drome. Eur J Paediatr Neurol. 2018;22(2):336–9.
2019;160(9):1954–66. 28. Tricco AC, et  al. PRISMA extension for scoping reviews
7. Walton DM, Elliott JM. An integrated model of chronic (PRISMA-ScR): checklist and explanation. Ann Intern Med.
whiplash-associated disorder. J Orthop Sports Phys Ther. 2018;169(7):467–73.
2017;47(7):462–71. 29. Arksey H, O’Malley L. Scoping studies: towards a methodological
8. Campbell L, et al. Psychological factors and the development of framework. Int J Soc Res Methodol. 2005;8(1):19–32.
chronic whiplash-associated disorder(s): a systematic review. Clin 30. Munn Z, et al. Systematic review or scoping review? Guidance for
J Pain. 2018;34(8):755. authors when choosing between a systematic or scoping review
9. Carroll LJ, et al. Course and prognostic factors for neck pain approach. BMC Med Res Methodol. 2018;18(1):143.
in whiplash-associated disorders (WAD): results of the bone 31. Bramer WM, et al. De-duplication of database search results
and joint decade 2000–2010 task force on neck pain and for systematic reviews in EndNote. J Med Libr Assoc.
its associated disorders. J Manip Physiol Ther. 2009;32(2, 2016;104(3):240–3.
Supplement):S97–S107. 32. Freeman MD, Croft AC, Rossignol AM. "Whiplash associated
10. Holm LW, et al. The burden and determinants of neck pain in disorders: redefining whiplash and its management" by the Que-
whiplash-associated disorders after traffic collisions: results of bec Task Force: a critical evaluation. Spine. 1998;23(9):1043–9.
the bone and joint decade 2000–2010 task force on neck pain 33. Mullen R. The state of the evidence: ASHA develops levels of
and its associated disorders. J Manip Physiol Ther. 2009;32(2, evidence for communication sciences and disorder. (Ameri-
Supplement):S61–S6969. can Speech-Language-Hearing Association). A S H A Leader.
11. Pink J, et al. Economic and health-related quality of life outcomes 2007;12(3):8.
of whiplash associated disorders. Spine. 2016;41(17):1378–86. 34. Ferrari R, et  al. A re-examination of the whiplash associ-
12. Elliott JM, et al. Spinal cord metabolism and muscle water diffu- ated disorders (WAD) as a systemic illness. Ann Rheum Dis.
sion in whiplash. Spinal Cord. 2012;50(6):474–6. 2005;64(9):1337–422.
13. Spitzer WO. Scientific monograph of the Quebec Task Force on 35. Carroll LJ, Cassidy JD, Côté P. The role of pain coping strategies
Whiplash-Associated Disorders: redefining "whiplash" and its in prognosis after whiplash injury: passive coping predicts slowed
management. Spine. 1995;20:1S–73S. recovery. Pain. 2006;124(1):18–26.

13
D. B. Stone et al.: Whiplash-Associated Dysphagia and Dysphonia

36. Haggman-Henrikson B, Zafar H, Eriksson PO. Disturbed jaw 50. Reiter S, et al. Masticatory muscle disorders diagnostic criteria:
behavior in whiplash-associated disorders during rhythmic jaw the American Academy of Orofacial Pain versus the research
movements. J Dent Res. 2002;81(11):747–51. diagnostic criteria/temporomandibular disorders (RDC/TMD). J
37. Häggman-Henrikson B, et  al. Endurance during chewing in Oral Rehabil. 2012;39(12):941–7.
whiplash-associated disorders and TMD…temporomandibular 51. Steinbach L. Calcium hydroxyapatite deposition disease (HADD).
disorders. J Dent Res. 2004;83(12):946–50. Encylopaedia of diagnostic imaging. Berlin: Springer; 2008. p.
38. Anagnostara A, et al. Traumatic retropharyngeal hematoma and 803–806.
prevertebral edema induced by whiplash injury. Emerg Radiol. 52. Bannai T, Seki T, Shiio Y. A pain in the neck: calcific tendinitis
2005;11(3):145–9. of the longus colli muscle. The Lancet. 2019;393(10185):e40.
39. Brademann G, Reker U. Paralysis of the superior laryngeal 53. Papadopoulou S, et al. Dysphagia associated with cervical spine
nerve after whiplash trauma. Laryngo- Rhino- Otologie. and postural disorders. Dysphagia. 2013;28(4):469–80.
1998;77(1):3–6. 54. Tian W, Yu J. The role of C2–C7 and O-C2 angle in the devel-
40. Chirchiglia D, et al. The neck-tongue syndrome following cervical opment of dysphagia after cervical spine surgery. Dysphagia.
whiplash injury. Interdiscip Neurosurg Adv Tech Case Manag. 2013;28(2):131–8.
2019;15:38–40. 55. Siegmund GP, et al. The anatomy and biomechanics of acute and
41. Helliwell M, et al. Bilateral vocal cord paralysis due to whiplash chronic whiplash injury. Traffic Injury Prev. 2009;10(2):101–12.
injury. Br Med J Clin Res Ed. 1984;288(6434):1876–7. 56. Curatolo M, et al. Central hypersensitivity in chronic pain after
42. Waddell RK. Chiropractic care for a patient with spasmodic dys- whiplash injury. Clin J Pain. 2001;17(4):306–15.
phonia associated with cervical spine trauma. J Chiropr Med.
2005;4(1):19–24. Publisher’s Note Springer Nature remains neutral with regard to
43. Kalezic N, et al. Cardiovascular and muscle activity during chew- jurisdictional claims in published maps and institutional affiliations.
ing in whiplash-associated disorders (WAD). Arch Oral Biol.
2010;55(6):447–53.
44. Lampa E, et al. Effects on jaw function shortly after whiplash
trauma. J Oral Rehabil. 2017;44(12):941–7. D. B. Stone  BAppSc(SpPath), MAppSc, PhD Candidate
45. Severinsson Y, Bunketorp O, Wenneberg B. Jaw symptoms and
signs and the connection to cranial cervical symptoms and post- E. C. Ward  PhD
traumatic stress during the first year after a whiplash trauma. Disa-
bil Rehabil. 2010;32(24):1987–98. S. R. Knijnik  BSpHLSc, MAppSc
46. Pennie B, Agambar L. Patterns of injury and recovery in whiplash.
Injury. 1991;22(1):57–9. H. Bogaardt  PhD
47. Gargan MF, Bannister GC. The rate of recovery following whip-
lash injury. Eur Spine J. 1994;3(3):162–4. J. M. Elliott  PT, PhD, FAPTA
48. Sturzenegger M, Radanov BP, Di Stefano G. The effect of acci-
dent mechanisms and initial findings on the long-term course of
whiplash injury. J Neurol. 1995;242(7):443–9.
49. Hildingsson C, Hietala SO, Toolanen G. Scintigraphic find-
ings in acute whiplash injury of the cervical spine. Injury.
1989;20(5):265–6.

13

You might also like