Journal of Neural Transmission

https://doi.org/10.1007/s00702-020-02157-3

NEUROLOGY AND PRECLINICAL NEUROLOGICAL STUDIES - REVIEW ARTICLE

Chronic orofacial pain

Sowmya Ananthan1 · Rafael Benoliel1

Received: 8 August 2019 / Accepted: 3 February 2020

© Springer-Verlag GmbH Austria, part of Springer Nature 2020

Abstract
While pain chronicity in general has been defined as pain lasting for more than 3 months, this definition is not useful in
orofacial pain (OFP) and headache (HA). Instead, chronicity in OFP and HA is defined as pain occurring on more than
15 days per month and lasting for more than 4 h daily for at least the last 3 months. This definition excludes the periodic
shortlasting pains that often recur in the face and head, but are not essentially chronic. Although the headache field has
adopted this definition, chronic orofacial pain is still poorly defined. In this article, we discuss current thinking of chronicity
in pain and examine the term ’chronic orofacial pain’ (COFP). We discuss the entities that make up COFP and analyze the
term’s usefulness in clinical practice and epidemiology.

Keywords  Headache · Persistent pain · Facial pain · Migraine · Myalgia · Neuropathic pain

Introduction and/or nuchal ridge, whereas facial pain is defined as pain

occurring mainly or exclusively under the orbitomeatal line,
Chronic orofacial pain (COFP) has been variably defined to anterior to the pinnae and above the neck; (ICHD-3 2018;
describe a group of regional disorders which are persistent ICOP 2020) others include the forehead as part of the face
in nature. In this manuscript, we will analyze the definition (Piper 1995). However, the face and head are both inner-
of COFP as it relates to the more established and widely vated by the trigeminal nerve even if headache is largely via
accepted definitions of chronic headache and chronic pain the first division and orofacial pain via the second and third
in general. We make a clear case for adopting the criteria trigeminal branches. Of itself, this provides a substrate for
used to define ‘chronicity’ in headache to orofacial pain pain referral. Moreover, the face and head have a number
(OFP). Subsequently we examine common (OFP) disorders of important structures adjacent to each other (eyes, ears,
that may become chronic and discuss to what level the term nose, paranasal sinuses, and oral cavity) further complicat-
COFP is of clinical importance and significance. ing pain referral patterns. The trigeminal nerve provides
primary sensory innervation to all of these as well as the
brain, brainstem, and the cavernous sinus further facilitating
Chronic facial pain or headache? pain referral mechanisms at the peripheral and central levels
(Tal et al. 2015).
Discussing facial pain is impossible without a parallel intro- Indeed, there is clinical overlap between chronic orofa-
duction to the overlap and intimate relationship with head- cial pain (COFP) and headache with 60% of patients seek-
ache. Facial pain has generally been separated from head- ing treatment for COFP reporting headaches (Dando et al.
ache based on anatomic definitions. Headache is defined 2006). Moreover, there is a significant relationship in the
as pain occurring only or mainly above the orbitomeatal general population between the frequencies of chronic
facial, head, and cervical pain (Storm and Wanman 2006).
The mechanism can be explained by the neuronal interplay
* Rafael Benoliel between the trigeminal subnucleus caudalis and the upper
benolira@sdm.rutgers.edu cervical spinal cord (trigeminocervical complex), and neu-
1
roplastic changes that occur in response to injury and pain
Department of Diagnostic Sciences, Center
for Temporomandibular Disorders and Orofacial Pain,
(Shinoda et al. 2019; Tal et al. 2015). In clinical practice,
Rutgers School of Dental Medicine, 110 Bergen Street, therefore, headache and facial pain are most often intimately
Newark, NJ 07101, USA

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 S. Ananthan, R. Benoliel

related. The pain referral patterns are complex, but very is clear that chronic orofacial pain is more prevalent than
commonly seen as the symptoms can overlap, hence this previously thought.
relationship can be bidirectional (Graff-Radford and Abbott A clear definition of COFP is essential so as to allow a
2016; Abouelhuda et al. 2017). common base upon which to study disease burden, outcome,
Based on this, we will be drawing on some headache and comorbidities associated with orofacial pain in a stand-
research to examine the phenomenon of chronic orofacial ardized manner. These comorbidities may include other pain
pain (COFP). Orofacial pain is generally classified based on syndromes and psychosocial disturbances.
the symptomatology and suspected pathophysiology and a
new classification has been recently pubished (ICOP 2020), Chronic pain
see Table 1. Hence the clinical groupings such as muscu-
loskeletal pain (within the temporomandibular disorders), Based on temporal patterns, OFP disorders may, rather sim-
neuropathic pain (post-traumatic and post-herpetic), and plistically, also be further classified as ‘acute’ or ‘chronic’.
odontogenic pain due to a variety of dentoalveolar patholo- Although the term seems self-explanatory, what indeed is
gies (Sharav and Benoliel 2015). OFP may be primary (or ‘chronicity’ in pain and specifically in OFP and headache?
idiopathic) (Treede et al. 2019) or secondary when the pain In general, chronic pain is widely defined as present for more
is due to an identifiable pathological process (Benoliel et al. than 3 months (Olesen et al. 2004, 2007). The definition of
2019). chronicity for pain has endured the test of time and the IASP
The prevalence of pain in the orofacial region has task force on chronic pain recently defined it as “persistent
been reported to be between 16.1 and 33.2% (Horst et al. or recurrent pain lasting longer than 3 months” (Treede
2015), (de Melo Junior et al. 2019). A realistic prevalence et al. 2015). Although simple, the definition according to
for OFP seems to be around 25%, of which about 10% is pain duration is clear, unambiguous, and easily applicable.
COFP (McMillan et al. 2006; Macfarlane et al. 2002a; Ng The definition does not prevent the additional collection of
et al. 2002), and induces significantly reduced quality of relevant and important information and comorbid conditions
life, sleep disturbances, and disability (Shueb et al. 2015; such as psychosocial factors, depression, pain-related dis-
Benoliel et al. 2017; Haviv et al. 2017; Ridgeway et al. tress, and functional impairment. In studies, chronic pain,
2013; Almoznino et al. 2017). Painful temporomandibular irrespective of diagnosis, demonstrates high prevalence of
disorders are quite prevalent with 4.6% of the population serious psychiatric and medical comorbidities (Ratcliffe
reporting these (6.3% in women, 2.8% in men) (Isong et al. et al. 2008; Altindag et al. 2008; Nicholson and Verma 2004;
2008). This is in agreement with the 2009 National Health Wilson et al. 2002; Korszun 2002).
Interview Survey that found that 5% of adults reported pain In the field of headache and OFP, a simple definition
in the face or jaw over a 3-month period. The incidence ratio based on disease duration is incompatible with the temporal
of persistent facial pain was reported at 38.7 per 100,000 characteristics. When strictly adhering to such a definition,
person years, is more common in women and increased with primary orofacial pain and headache disorders are chronic
age (Koopman et al. 2009). Syndromes identified included by definition, since many occur for the life of an individual
trigeminal neuralgia and cluster headache, which were the patient. The particular temporal characteristics of OFP and
most common forms. Paroxysmal hemicrania and glos- HA require a different approach to define chronicity.
sopharyngeal neuralgia were among the rare syndromes. It The headache community defined chronicity for head
pain as headache that occurred (untreated) on at least 50%
Table 1  International Classification of Orofacial Pain (ICOP) of the days for at least 3 months and lasted at least 4 h per
day (ICHD-3 2018). The criterion of a 4-h duration per day
Chapters describing diagnostic criteria for: was used to exclude the paroxysmal headache disorders (e.g.
1. Orofacial pain attributed to disorders of dentoalveolar and anatomi- episodic cluster headache) but has since been shortened to 2
cally related structures
h for uniformity across disorders. These headache disorders
2. Myofascial orofacial p­ aina
are more of an unremitting paroxysmal disorder than a bona
3. Temporomandibular joint (TMJ) p­ aina
fide chronic pain condition. In this manner, chronic headache
4. Orofacial pain attributed to lesion or disease of the cranial n­ ervesa
and OFP disorders can be separated from episodic disorders
5. Orofacial pains resembling presentations of primary h­ eadachesa
based on their time pattern.
6. Idiopathic orofacial p­ aina
Chronic daily headache (CDH) is the term coined in the
Chapter with recommendations on further assessment
headache literature to bring together these disorders based
7. Psychosocial assessment of patients with orofacial pain
on their temporal pattern. Similarly, COFP is an umbrella
From International Classification of Orofacial Pain (ICOP) 2020 term, primarily useful for epidemiology and the study of
a
 Groups that may include subtypes with a chronic temporal pattern as the effect of chronicity on quality of life issues. CDH as a
defined in the text diagnosis is sometimes seen clinically and in the headache

13
Chronic orofacial pain

literature (Silberstein et al. 1996), but without knowing the The case for adopting CDH criteria
precise diagnosis, the term is clinically of little use in estab-
lishing management strategies. CDH is relatively common The study of the epidemiology, clinical characteristics, treat-
in the population with a worldwide prevalence of about ment outcomes, and associated comorbidities of ‘chronic’
2.5–5%. The vast majority suffers from chronic tension-type pain syndromes requires precise definitions of its temporal
headache (global prevalence of 2–3%) or chronic migraine features.
(1.3–2.5%) (Jensen 2003; Russell 2005). Secondary CDH is We studied the applicability of CDH criteria to COFP and
most usually due to medication overuse (Castillo et al. 1999; found them to be very useful (Benoliel et al. 2010). Further
Zwart et al. 2003), but also includes headache secondary to studies are needed to examine the 4-h cut off point, but in
COFP entities such as temporomandibular disorders (Graff- general, we found this approach produced a more detailed
Radford 2005). resolution of pain patterns in persistent OFP (Benoliel et al.
Primary CDHs are mostly a temporal transformation of 2010). The criteria are applicable to the study of COFP and
their episodic counterparts (Olesen et al. 2004) and may be should be adopted in epidemiologic and clinical trials, bring-
defined as short or long lasting (> 4 h per attack) Silberstein ing orofacial pain in line with headache.
and Lipton 2000 include chronic migraine, chronic trigemi-
nal autonomic cephalgias, chronic tension-type headache,
and new daily persistent headache. The ‘International Classification of Orofacial
Pain’ (ICOP)
Defining chronic orofacial pain ICOP is a multiprofessional, international initiative to estab-
lish a comprehensive classification of orofacial pain and has
The question is how do we define chronic orofacial pain? recently been published (ICOP 2020). This classification is
The issue has been debated in the literature. Attempts at in line with ICHD-3 and ICD-11, and defines common OFP
defining chronic (orofacial) pain using additional criteria diagnoses such as dentoalveolar pain, masticatory myofas-
from common comorbid conditions, such as depression, cial pain, and facial presentations of primary headaches,
have not been adopted due their inherent complexity. Inclu- which are not directly described or recognized by the ICHD
sion criteria employed in studies on COFP differ; pain (Table 1). We have previously stressed the lack of inclusion
of 1 day over the last month, daily or near daily pain for of OFP in ICHD-3 (Benoliel et al. 2008) and ICOP may
the last 3 months (Aggarwal et al. 2007; McMillan et al. provide a bridge between the headache and OFP professions.
2006; Chung et al. 2004; Macfarlane et al. 2002b; Sharav Importantly, ICOP adopts the temporal definitions of
et al. 1987; Lipton et al. 1993; Locker and Grushka 1987; chronicity in OFP as that for headaches. These definitions
Riley and Gilbert 2001). Moreover, features such as attack can be subsequently field tested further to establish the spe-
frequency and duration are often unclear (Aggarwal et al. cific relevance to the study of COFP (Benoliel et al. 2010).
2007; Chung et al. 2004; McMillan et al. 2006). This lack
of uniformity in defining COFP has been emphasized so that
comparing data across studies is consequently problematic General features of COFP
(Macfarlane et al. 2004; Benoliel et al. 2010, 2009).
In the past, an attempt has been made to conceptually When we applied CDH criteria to OFP, we encountered
unify persistent or COFP syndromes under one pathophysi- interesting findings (Benoliel et al. 2010). Attesting to the
ological concept. This proposes that based on chronicity complexity often involved in the diagnosis of orofacial
and other shared features, the diagnostic entities included pain syndromes (Zebenholzer et al. 2005, 2006; Benoliel
in COFP are clinically and therapeutically more similar et al. 1994, 2008), patients arrival for diagnosis and treat-
than they are different (Woda and Pionchon 1999, 2000). ment about 2–3 years after the onset of pain irrespective of
Unfortunately, the precise definition of ‘chronicity’ for the diagnosis or temporal patterns. These figures are similarly
different OFP disorders was poorly defined and inconsistent. reported in the literature for primary headaches and orofa-
Moreover, this concept, although interesting in its approach, cial pain (Bahra and Goadsby 2004; van Vliet et al. 2003;
has not received wide acceptance (Fricton 1999; Okeson Bowsher 2000; de Siqueira et al. 2004; Main et al. 1992;
1999). Mitchell 1980; Taha et al. 1995; Benoliel et al. 2009, 2016;
Haviv et al. 2014, 2017).
When applying CDH definitions, COFP patients were
usually female, with a longer reported time to diagnosis
and more frequently reported waking due to pain. The most

13
 S. Ananthan, R. Benoliel

common diagnosis was one of the myalgias as defined by rare. Therefore, in the absence of clear etiologic findings fol-
the Research Diagnostic Criteria for TMDs (RDC-TMD). lowing thorough clinical and radiographic examination, pain
Other characteristics were not significantly different from referral from regional structures, or an atypical presentation
those in episodic-type OFP. Moreover, COFP was not dis- or referral pattern from primary or other headache should
tinctive in pain severity or other demographic features. Most be considered, see below (Gross and Eliashar 2015; Sharav
parameters usually associated with the diagnostic process et al. 2017; Benoliel et al. 2008; Gaul et al. 2008; Benoliel
(laterality, severity, muscle tenderness, and waking) were et al. 2015).
associated with specific diagnosis rather than temporal pat- Chronic mucosal pain has been extensively reported to be
terns when employing appropriate statistical methodology. associated with burning mouth syndrome. However, it is still
This stresses the importance of relating to the term COFP unclear whether pain originating from common oral mucosal
as an umbrella term or diagnosis based on a purely temporal lesions can truly be chronic (Abdalla-Aslan et al. 2016).
definition. It is similar to CDH which includes chronic ten-
sion-type headache, chronic migraine, and other disorders.
For tailored management, further classification into specific Temporo‑mandibular disorders
diagnoses is needed.
The umbrella term- temporo-mandibular disorders (TMD)
have been used to describe painful and non-painful disorders
Review of relevant‑specific COFP disorders affecting the masticatory muscles and/ or the temporo-man-
dibular joint. Pain is associated with the TMJ (arthralgia) or
A detailed, inclusive review of all specific COFP disorders the muscles of mastication (myalgia) and is usually chronic
is beyond the scope of this article. We, therefore, aim to with a significant effect on quality of life (Almoznino et al.
provide an overview with carefully selected citations that 2015). Together, these are the most common type of COFP
will allow readers to expand their knowledge as well as sub- disorders, affecting 4–12% of the population and severe
classify the various specific diagnoses that can fall under symptoms are reported by 10% of subjects (Macfarlane
COFP. et al. 2002a; Nilsson et al. 2005). Moreover only 3–11% seek
treatment, with an estimated 7% of the general population
demanding therapy for TMD (Goulet et al. 1995; Schiffman
Orofacial pain attributed to disorders et al. 2014).
of dentoalveolar and anatomically related The diagnosis of myalgia and arthralgia is clinically based
structures on response to palpation of muscles and TMJs. It is, there-
fore, important to note that masticatory muscle and TMJ
Theoretically, chronic dentoalveolar pain may result from tenderness are present in about 15% and 5%, respectively, of
a disorder involving the teeth or associated tissues (pulpal, the population (Gesch et al. 2004). Moreover muscle tender-
alveolar bone, periodontal, and gingival). Causative factors ness may be ‘asymptomatic’ in a third of such cases. It is
would include untreated caries or trauma to a tooth or asso- only when the patient reports pain that can be replicated by
ciated tissues. palpation that we can establish a diagnosis.
Untreated dental decay has been reported as the most Clinically, it is common to see arthralgia and myalgia
common reason for toothache, which can impact routine in the same patient (Almoznino et al. 2019, 2015; Benoliel
daily activities. Toothache is a common problem and, et al. 2011a), they should be identified and treated as sepa-
depending on geographic location, may be highly prevalent rate entities.
(Kakoei et al. 2013). However, it is unclear what propor-
tion of reported toothache is truly chronic and more data are
needed (Kassebaum et al. 2017). From currently available Myofascial–orofacial pain
data, duration of constant toothache is present for 27.6 days
up to seeking care (Kassebaum et  al. 2017). A similar The pathophysiology of myalgia is uncertain. Research sug-
study estimated 55.2 days of tooth pain prior to present- gests that it is likely to involve complex interactions between
ing for treatment to an emergency dental clinic (Whyman genetic, biological, and psychosocial parameters, hence the
et al. 1996). As the above epidemiology suggests that truly term ‘complex biopsychosocial disorder’ (Benoliel et al.
chronic dental pain (i.e. lasting > 3 months or meeting CDH 2011b; Slade et al. 2013, 2016; Smith et al. 2013). Myalgia
criteria) may be extremely rare and caution should be exer- has been subclassified into three categories, all of which
cised when diagnosing such cases (Benoliel et al. 2019). may result in COFP. Localized myalgia presents with pain
Inevitably, the conclusion is that chronic dentoalveolar in a particular area of a muscle (local myalgia). Myalgia
pain due to related pathology is undocumented, or extremely may also refer within (myofascial pain) or beyond muscle

13
Chronic orofacial pain

boundaries (myofascial pain with referral) (Schiffman et al. or specific jaw movements (Schiffman et al. 2014). These
2014). Clinical presentation includes pain in the masticatory signs usually indicate capsulitis or synovitis.
muscles which can be replicated on palpation, and resulting When discussing TMJ disorders, the term ‘internal
in dysfunction of the masticatory apparatus. Pain, is usually derangement’ is often used as a diagnosis. However, inter-
unilateral, but can be bilateral and is localized in and around nal derangement is a classification or descriptive term, not
the ear, angle/body of the jaw, and the temple region. Hyper- a diagnosis, and includes disc displacements and degenera-
sensitive areas (aka active trigger points) can cause pain tion of the TMJ (Stegenga 2001; Nitzan et al. 2015). For a
referral to distant sites such as the ear and teeth following precise diagnosis, the structures and the mechanical prob-
palpation. The referral pattern depends on the location of the lem must be clearly defined; for example, disc displacement
hypersensitive area and the intensity of pain. This commonly with reduction or without reduction or intermittent locking
leads to misdiagnosis, leading to unnecessary treatment and (Nitzan et al. 2015). These may be accompanied by limited
increased health care utilization (Hobson et al. 2008). mouth opening and all may occur with and without pain
The quality of pain is generally dull and aching with (arthralgia). When there is damage to the condyle and or
moderate intensity that increases on function. Clinically, eminence, we refer to this as joint degeneration or degen-
the mouth opening may be reduced, with deviation of the erative joint disease. This process is associated with clear
mandible while opening the mouth. The muscles most com- tissue damage and disintegration leading to pathological
monly involved with MMP are the masseter, followed by the changes of the joint components and is also referred to as
temporalis (Valentino et al. 2019), and these are the basis for ‘TMJ osteoarthritis’.
clinical diagnosis (Schiffman et al. 2014).
Average pain duration ranges from 5.5 to 18  h (van
Grootel et al. 2005; Benoliel et al. 2008). Typically, MMP Secondary temporomandibular joint pain
is characterized by a chronic course with reported onset
months to several years previously (Kino et al. 2005; Ram- Temporomandibular joint pain attributed
melsberg et al. 2003). Pain occurring on most days of the to arthritis
month is typical of MFP patients (Benoliel et al. 2008).
The sternocleidomastoid and upper trapezius muscles are Systemic autoimmune arthritides such as rheumatoid and
often associated with MMP (Silveira et al. 2015). Indeed, psoriatic arthritides can affect the TMJ. Changes to the bony
MMP is highly correlated with pain of the cervical muscu- structures are seen early on in the disease process, where
lature, as previous studies have found that increased tender- there is destruction of the bone and cartilage of the joint
ness of the neck and body muscles predicted the incidence compartment. This in turn leads to limitation of function.
of TMD (Almoznino et  al. 2019; da Costa et  al. 2015). Therefore, it is important to diagnose these conditions early,
Indeed, patients with MMP had more widespread painful so that treatment can be rendered in a timely manner and
areas associated with chronic neck pain than patients with further damage to the joint structures can be minimized
neck pain alone (Munoz-Garcia et al. 2017). Symptoms of (Cordeiro et al. 2016). The radiographic signs are similar
TMD are also more prevalent in patients with fibromyalgia to those found in DJD, though the progression is faster and
(Ayouni et al. 2019), which is a widespread chronic pain there is involvement of both the TMJ’s. Clinically, there may
disorder. These data would suggest that MMP is part of a be an anterior open bite due to the rapid destruction of both
more generalized disorder with a variable clinical pheno- the condylar heads, leading to autorotation of the mandible.
type. Management of MMP is conservative involving the use The patients will also complain of pain on other joints in
of anti-inflammatory analgesics, physiotherapy, bite plates, their body. For diagnosis, laboratory studies have to be per-
centrally acting analgesics, intramuscular injections (aka formed, which may show increased inflammatory markers
trigger point injections), and cognitive behavioral therapy and rheumatoid factor.
(Svensson et al. 2015). The management of arthralgia is usually conservative
with the use of anti-inflammatory analgesics, physiotherapy,
bite plates, and arthrocentesis (Nitzan et al. 2015).
Temporomandibular joint pain Finally, the chronicity of arthralgia has been less inves-
tigated than myalgia, possibly due to a lack of clear guide-
Primary temporomandibular joint pain lines. In our study, truly chronic arthralgia was found in a
small number of patients attending for treatment (Benoliel
TMJ arthralgia is characterized by pain originating from the et al. 2010). The publication of ICOP will allow temporal
TMJ that is affected by jaw movement, function, or par- patterns to be studied more thoroughly.
afunction, and can be replicated by palpation, joint loading,

13
 S. Ananthan, R. Benoliel

Degenerative joint disease (DJD) neurovascular compression), ‘idiopathic’ (with no neurovas-

cular compression), and ‘symptomatic’ when caused by a
The DC-TMD and ICOP classify osteoarthritis and osteoar- known disease or space-occupying lesion (ICHD-3 2018).
throsis as subclasses of degenerative joint disease (Schiffman The pain of trigeminal neuralgia can be either exclusively
et al. 2014; ICOP 2020). Under the DCTMD, osteoarthritis paroxysmal or accompanied by concomitant continuous
(OA) is symptomatic, while osteoarthrosis shares all the pain.
same signs and symptoms of joint damage and dysfunction Diagnosis is made based on the clinical presentation,
but with no pain. The presence of ongoing pain is suggested which includes the presence of trigger zones in over 50% of
to reflect inflammation and is reflected in the terminology patients with trigeminal neuralgia (Maarbjerg et al. 2014).
(Alstergren et al. 2018). Indeed, degenerative joint disease This explains why innocuous stimuli trigger the intense pain
or OA has historically been considered a non-inflammatory of trigeminal neuralgia. A refractory period where after a
disorder. However, accumulating evidence indicates the paroxysm of pain attacks cannot be triggered is another dis-
presence of inflammation involving the synovium (Oehler tinguishing feature. TN may remit but usually for only a few
et al. 2002). Sustained intra-articular inflammation induces weeks and invariable returns. The course is chronic with
degeneration of TMJ structures (Wang et al. 2012). There daily or nearly daily pain occurring, some with a constant
is now clear evidence that synovitis is associated with pain background pain.
and dysfunction and that it promotes cartilage destruction Whether TN worsens over time is unclear. Previous stud-
in osteoarthritic joints (Scanzello et al. 2011; Walsh et al. ies suggest that TN is a progressive disease with a poor prog-
2007). nosis (Zakrzewska and Lopez 2004). About 90% of TN cases
The signs of DJD are seen very commonly on radio- reported increased attack frequency and severity (Bowsher
graphs, but do not always correlate with clinical tenderness 2000; Zakrzewska and Patsalos 2002). Furthermore, initial
(Arayasantiparb et al. 2019). Surface erosion with osteo- response to carbamazepine is around 70%, but by 5–16 years
phyte formation, subcortical cysts, and sclerosis are some the response rate is around 20% with 44% of patients requir-
of the common radiographic signs of DJD (Kaimal et al. ing drug combinations or alternative medication (Taylor
2018). Crepitation is heard in the joint during auscultation. et al. 1981). Long-term follow-up of oxcarbazepine-treated
So, while there are a number of TMJ disorders or CTN cases demonstrated a high failure rate necessitating
derangements, not all are painful. Pain (arthralgia) occurs surgery (Zakrzewska and Patsalos 2002). However, recent
with accompanying inflammation of the capsule and/or data indicate that according to current diagnostic criteria the
synovial membrane lining the joint (Ibi 2019). treatment response and long-term prognosis of CTN are very
good (Di Stefano et al. 2014). Clear treatment protocols have
been established (Bendtsen et al. 2019).
Orofacial pain attributed to lesion or disease
of the cranial nerves
Painful post‑traumatic trigeminal
Neuropathic pain has been recently re-defined as “pain aris- neuropathy
ing as a direct consequence of any lesion or disease affecting
the somatosensory system” (Haanpaa et al. 2011). NP may Trigeminal neuropathy is a chronic neuropathic orofacial
result from neuronal injury, active or past systemic or local pain condition caused by trauma to the peripheral branches
diseases (Elad et al. 2015). The most common neuropathic of the trigeminal nerve (Benoliel et al. 2012; ICHD-3 2018).
pain conditions affecting the orofacial area are trigeminal Persistent pain can occur after simple dental procedures
neuralgia and painful post-traumatic trigeminal neuropathy. affecting smaller peripheral branches such as in 3–7% of
patients undergoing endodontic procedures (Nasri-Heir et al.
2015) and 0.3–0.8% of patients undergoing dental implant
Trigeminal neuralgia placement (Vazquez-Delgado et al. 2018) or due to major
trauma such as fractures, which can affect larger nerve bun-
Trigeminal neuralgia is an episodic, short lasting (< 2 min), dles (Joachim et al. 2019; Benoliel et al. 2005). Altered sen-
severe pain condition affecting one or more branches of the sation may occur commonly after third molar extractions
fifth cranial nerve (ICHD-3 2018). The most common pres- in about 0.35–8.4% of patients, although chronic pain is
entation affects the second and third branch of the trigeminal extremely rare (Sarikov and Juodzbalys 2014).
nerve concomitantly (~ 35%), the same branches are affected The pain is located to the dermatome of the affected nerve,
individually in about equal prevalence (~ 16%). Trigeminal though as the condition becomes chronic, the pain may spread
neuralgia has recently been reclassified into ‘classical’ (with to adjacent dermatomes due to the development of central sen-
sitization and alteration in pain modulation (Nasri-Heir et al.

13
Chronic orofacial pain

2015). Patients who develop painful neuropathies present 2004; Schreiber et al. 2004; Senbil et al. 2008; Rossi et al.
with a wide array of clinical symptoms ranging from nega- 2009; Van Alboom et al. 2009; Viana et al. 2013). ICOP
tive symptoms such as analgesia to positive symptoms such deals with these entities and provides a solid base for future
as hyperalgesia (Vazquez-Delgado et al. 2018; Benoliel et al. research.
2012; Svensson et al. 2011). The pain is generally described
as burning, continuous, moderate in intensity with occa-
sional paroxysms that are more severe in intensity (Renton Neurovascular orofacial pain
et al. 2012; Pigg et al. 2013; Baad-Hansen et al. 2008; Renton
and Yilmaz 2011). The condition is extremely recalcitrant to Studying a large group of patients with migraine-like or
therapy (Haviv et al. 2014). Nevertheless, accepted pharmaco- TAC-like pains in the orofacial region, we were able to use
therapeutic protocols (Attal et al. 2010; Dworkin et al. 2010; ICHD criteria to diagnose the majority into an orofacial
Finnerup et al. 2010) are recommended and topical therapies component of a primary headache while a number remained
(Nasri-Heir et al. 2013; Haribabu et al. 2013) may alleviate ‘undiagnosable’ (Benoliel et al. 2008; Haviv et al. 2019 (in
pain. press)), fitting neither the criteria for migraine or TACs. We
named the disorder in these patients “neurovascular orofacial
Orofacial pains resembling presentations pain” (NVOP).
of primary headaches The features that justify the establishment of NVOP as a
distinct entity have been discussed in detail by our group,
Unexplained pain in the orofacial region may be an atypical including the proposition of diagnostic criteria with positive
presentation of a primary headache. ICHD group primary and negative predictive values (Benoliel et al. 2008). The
headache is divided into four groups; migraines, tension- most prominent is oral and/or facial pain location [Benoliel
type headache, trigeminal autonomic cephalgias, and a het- et al. 2008; Haviv et al. 2019 (in press)]. Pain can be accom-
erogenous group termed ‘other primary headaches’. There panied local autonomic signs, specifically tearing, nasal con-
have been no clear reports on an orofacial tension-type head- gestion, a feeling of cheek swelling or fullness (Benoliel
ache, although the discussion on the similarities and dif- et al. 1997) and migrainous phenomena such as pho/phono-
ferences with masticatory myofascial pain (Svensson et al. phobia and nausea (Czerninsky et al. 1999; Benoliel et al.
2015) point to some interesting research avenues. Similarly, 1997; Obermann et al. 2007; Penarrocha et al. 2004). Pain
no clear orofacial equivalents have been reported for ‘other is bilateral in up to a third of patients with NVOP [Haviv
primary headache’. et al. 2019 (in press)]. Initially, unilateral pain was an inclu-
However, there are several reports of migraine-like or sion criteria for our patient selection studies (Benoliel et al.
trigemino-autonomic (TAC)-like pains in the orofacial 1997; Obermann et al. 2007; Penarrocha et al. 2004) leading
region. The differential diagnosis of such disorders includes to biased results and limiting the size of the NVOP group.
a number of entities. A facial or orofacial equivalent to In later studies, bilateral pain was included [Benoliel et al.
migraine without aura (MWoA), has been reported as ‘lower 2008; Haviv et al. 2019 (in press)]. NVOP can present as
half’(Penarrocha et al. 2004), ‘facial’ (Daudia and Jones paroxysmal, continuous, or combined temporal patterns.
2002) or ‘orofacial’(Gaul et al. 2007) migraine. Regarding Based on these, 45% of NVOP patients would be consid-
this type of migraine, ICHD-3 states that “a subset of oth- ered COFP. Of clinical importance is the common finding
erwise typical patients has facial location of pain, which is of dentoalveolar-accompanying NVOP pain with unique
called ‘facial migraine’ in the literature; there is no evidence characteristics that mimic dental pathology. These signs
that these patients form a separate subgroup of migraine and symptoms include spontaneous or evoked dentoalveo-
patients” (ICHD-3 2018). Amongst TACs, an atypically lar pain (e.g. cold allodynia), explaining patient reports of
located cluster headache termed ‘lower’ CH (Cademartiri superimposed short pain attacks (Benoliel et al. 1997; Ober-
et al. 2002) and ‘orofacial’ CH (Gaul et al. 2008) has been mann et al. 2007; Penarrocha et al. 2004; Gaul et al. 2007).
most commonly noted. Maxillary and mandibular expres- Usually there is no dental pathology to explain the pain and
sions of SUNCT/SUNA have also been recognized (Cohen we hypothesize that this is part of the clinical phenotype.
et al. 2006; Goadsby et al. 2010). In their chronic form, all Additionally it has been a consistent finding that NVOP
the above should be considered in the differential diagnosis. patients report a relatively late age of onset [Benoliel et al.
The importance is clear—primary orofacial pain needs no 2008; Haviv et al. 2019 (in press)]. This fairly new diagnos-
dental intervention. Nevertheless the facial, dentoalveolar, tic entity has been included in ICOP.
and maxillary sinus location of pain in these entities often
cause misdiagnosis and subsequent mistreatment (Czernin-
sky et al. 1999; Eross et al. 2007; Foroughipour et al. 2011;
Kari and DelGaudio 2008; Klapper et al. 2000; Perry et al.

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 S. Ananthan, R. Benoliel

Idiopathic orofacial pain Persistent idiopathic facial pain (PIFP)

Burning mouth syndrome The diagnostic criteria for PIFP include the presence of daily
or near daily pain that is initially confined but may sub-
Burning mouth syndrome (BMS) is a rare intraoral pain sequently spread (ICHD-3 2018; ICOP 2020). Pain cannot
condition that is poorly understood and often misdiag- be attributed to any pathological process for a diagnosis of
nosed (Salerno et al. 2016). BMS is common in peri and PIFP. Clearly this is a loose and ambiguous definition and
post- menopausal women and occurs in the absence of any may allow the misclassification of a large number of chronic
organic findings, when it is often termed primary BMS. It facial pain disorders. As such, it is often regarded as a ‘waste
is important to rule out local or systemic causes of burning basket’ diagnosis and one of exclusion. However, it is impor-
such as candida infections, uncontrolled diabetes (Verhulst tant for clinicians to clearly distinguish PIFP from disorders
et al. 2019), avitaminoses, iron deficiency, and thyroid dis- that may mimic it such as trigeminal neuralgia with per-
ease (Talattof et al. 2019). Medications that patients take sistent background pain, painful traumatic trigeminal neu-
commonly may cause oral burning as a common side effect. ropathies, myofascial pain, and others (Sotorra-Figuerola
In the presence of such potentially causative factors, the dis- et al. 2016). Accurate diagnosis is key to succesful therapy
order is termed as secondary BMS. Appropriate diagnostic and prevents potentially serious consequences. The preva-
tests are often required to establish the diagnosis. lent theory is that PIFP is a disproportionate reaction to a
Patients predominantly have minimal burning on waking mild injury, but the exact pathophysiology is still unclear.
but as the day progresses, the burning sensation increases In a deviation from ICHD-3, the definition of PIFP in ICOP
in intensity, reaching peak levels during the evening hours. allows mild sensory changes to be present. The aim is to
The most frequent location is the anterior two-thirds of the allow further research into the pathophysiology of PIFP and
tongue, though in some patients, the burning may be pre- its relationship to PTTN. This change is justified based on
sent in the palate or gingiva, sometimes even the lips may hypoesthesia in studies using quantitative sensory testing
be involved. Eating spicy foods may aggravate the burning (QST) (Siqueira et al. 2013; Baad-Hansen et al. 2013; Fors-
sensation. BMS patients suffer from poor sleep quality and sell et al. 2007).
increased prevalence of anxiety and depression, although Pain in PIFP is usually deep, poorly localized, radi-
cause and effect has not been clearly established (Adamo ating, and mostly unilateral although up to 40% of cases
et al. 2018; Davies et al. 2016). may describe bilateral pain (Maarbjerg et al. 2016). PIFP
The pathophysiology is still poorly understood. It is sug- is commonly described as aching, burning, throbbing, and
gested that three groups may occur all of which link BMS often stabbing (Siqueira et al. 2013; Lang et al. 2005; Baad-
to neuropathic pain (Jaaskelainen 2012; Kolkka-Palomaa Hansen et al. 2013; Forssell et al. 2007; Maarbjerg et al.
et al. 2015). The largest subgroup (50–65%) display intraoral 2016; Pfaffenrath et al. 1993). Severity, usually mild to
peripheral small-diameter fiber neuropathy. The second sub- severe (rated 7 on an 11-point VAS), may be aggravated by
group (20–25%) requires careful neurophysiologic examina- emotional stress (Maarbjerg et al. 2016).
tion to identify them, but is clinically indistinguishable from Most PIFP patients report persistent, long lasting (years)
the other two subgroups. Subclinical lingual, mandibular, or daily pain (Maarbjerg et al. 2016) that tends to spread, in a
trigeminal system pathology is revealed with these tests. The non-dermatomal pattern, with time. Typically, pain charac-
third subgroup (20–40%) displays characteristics of central teristics, location, and associated features change over time.
pain that is hypothesized to involve hypofunction of dopa- Rarely, some PIFP patients report pain free or remission
minergic neurons in the basal ganglia (Jaaskelainen 2012). periods (Maarbjerg et al. 2016). Often, PIFP may coexist
A hypothetical relationship to the function (or dysfunction) with other chronic orofacial pain or headache syndromes
of neuronal pathways relating to the sense of taste has been (Maarbjerg et al. 2016).
investigated and has evidence backing this hypothesis (Eliav
et al. 2007; Formaker and Frank 2000; Grushka et al. 2003;
Just et al. 2010; Kim et al. 2019; Kolkka-Palomaa et al. Conclusions
2015). Management should be evidence based and often
requires multiple, complimentary, and concomitant therapies COFP should be regarded as an umbrella term covering indi-
(e.g. medication and behavioral therapy) (de Souza et al. vidual and specific disorders that have a common chronic
2018; Feller et al. 2017; McMillan et al. 2016). temporal pattern. It is certainly a useful tool to examine dis-
ease burden. In a clinical setting, however, establishing a
diagnosis of COFP would have no therapeutic indication:
each subcategory of COFP requires precise diagnosis and

13
Chronic orofacial pain

tailored management protocols. The adoption of criteria 2010 revision. Eur J Neurol 17(9):1113–e1188. https​://doi.org/1
used for headache chronicity (Benoliel et al. 2019) is rec- 0.1111/j.1468-1331.2010.02999​.x
Ayouni I, Chebbi R, Hela Z, Dhidah M (2019) Comorbidity between
ommended and indeed are followed in the recent publication fibromyalgia and temporomandibular disorders: a systematic
of ICOP (ICOP 2020). review. Oral Surg Oral Med Oral Pathol Oral Radiol 128(1):33–
42. https​://doi.org/10.1016/j.oooo.2019.02.023
Baad-Hansen L, Leijon G, Svensson P, List T (2008) Comparison of
clinical findings and psychosocial factors in patients with atypi-
Compliance with ethical standards  cal odontalgia and temporomandibular disorders. J Orofac Pain
22(1):7–14
Conflict of interest  Dr. Ananthan declares no conflicts of interest. Dr. Baad-Hansen L, Abrahamsen R, Zachariae R, List T, Svensson P
Benoliel is remunerated by Quintessence Publishing for serving as edi- (2013) Somatosensory sensitivity in patients with persistent
tor in chief. idiopathic orofacial pain is associated with pain relief from
hypnosis and relaxation. Clin J Pain 29(6):518–526. https​://doi.
org/10.1097/AJP.0b013​e3182​68e4e​7
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