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Orofacial phantom pain: theory and

phenomenology
JJ Marbach
JADA 1996;127(2):221-229
10.14219/jada.archive.1996.0172

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Developing a treatment strategy
for patients who are in chronic
pain can be challenging for any
health care professional, particu-
larly when traditional measures
to relieve pain are ineffective.
Pain treatment can be further
complicated by phenomena
known as phantom pain-most
often associated with limb ampu-
tation. The author describes
three phantom syndromes expe-
rienced in the orofacial region:
phantom tooth pain, phantom
bite syndrome and intraoral
stump pain.
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OROFACIAL PHANTOM PAIN:
THEORYAND PHENOMENOLOGY
JOSEPH J. MARBACH, D.D.S.
0 rofacial pain patients frequently confront dentists with difficult
challenges. Given the present competitive practice climate and the
urgency associated with pain, the clinical dilemma faced by the
dentist is expected to be resolved quickly and decisively. Ironically,
the pain sufferer may not always be aware of the pressure his or
her situation places on the clinician. Nevertheless, such situations
impel clinicians to perform procedures designed to eliminate pain
such as endodontic therapy or tooth extraction or to construct new
and "different" dentures. The urgency is almost palpable; however,
such urgent situations create the potential for failure.
The purpose of this article is to increase clinicians' awareness of
three putative neuropathologic facial pain disorders:
- phantom tooth pain;
- intraoral stump pain;
- phantom bite syndrome.
Researchers have studied phantom phenomena for many years,
and several conclusions seem undeniable. In his review article,'
Melzack described four elements that comprise the theory of the
neuromatrix:
- The bodily sensations we perceive in our brain are started and
maintained typically by information derived directly from our bod-
ies. Since phantom sensations feel so vivid, they, too, are probably
subserved by the same neural processes.
- All sensations we feel from our bodies, such as pain, can be felt
without input from the body. We may conclude from this that sen-
sory experiences are an inherent attribute of the neural "hardware"
of the brain. Although external stimuli may trigger sensations, they
do not create them; sensations are produced by the brain itself.
- The sense of self is generated in the central nervous system, not
from the sensations derived though the peripheral nervous system
or spinal cord. The uniqueness we perceive physically as ourselves
is thoroughly and permanently symbolized in the brain, which is
dominated by non-body images.
- The central nervous system processes that regulate the recogni-
tion of the body are genetically specified, though probably modified
by experience.2
JADA, Vol. 127, February 1996 221
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-ClINICAL PRACTICE
Figure 1. Model depicting tooth pulp injury. Solid arrows have more research support, dotted arrows, less
(Adapted from Devor M, Basbaum Al, Bennett GJ, et al.18 Published with permission from John Wiley &
Sons, Inc.).
The neuromatrix theory pro-
vides a framework for under-
standing some of the most per-
plexing pain disorders faced by
clinicians, those of orofacial
phantom phenomena. For clini-
cal purposes, the disorders are
divided into three categories:
- phantom tooth pain3'4;
- intraoral stump pain, such as
pain experienced in edentate
tissue;
- phantom bite syndrome, a
sensation often associated with
the inability to adapt to changes
in dental occlusion.5'6
Physical trauma is a frequent
precursor to phantom pain. Be-
sides trauma and infection, the
face and mouth are also the
sites of many surgical proce-
dures that, under certain cir-
cumstances, may result in ana-
tomically and physiologically
altered tissue. Injured tissue
may exhibit allodynia, a painful
response to a normally non-
painful stimulus, or hyperalge-
sia, an increased pain response
to a stimulus that is normally
painful, such as sunburn. In ad-
dition, patients may report pain
in tissues adjacent to the site of
222 JADA, Vol. 127, February 1996
injury, a phenomenon known as tooth pain. Usually it follows
secondary hyperalgesia.7 dental or surgical procedures
Injured tissue also may display such as pulp extirpations, api-
spontaneous pain or a pain coectomy or tooth extraction.
lacking a known stimulus. Surgery involving tissue adja-
Recent advances in under- cent to teeth, such as exentera-
standing pain provide evidence tion of the contents of the max-
that the pain associated with illary antrum and trigeminal
peripheral rhizotomy, also
tissue injury can result in oro-
may result Reports on tooth pain facial phantom
from such of obscure 4ongin are pain. Phantom
mechanisms relatively re ceent. The tooth pain is char-
as nociceptor telm acterized by per-
sensitization, term phant Pm tooth sistent toothache.
neuroma for- pain was fir-st used in Neither repeated
mation and
altered cen-
1978, and s ince then ment, endodontic treat-
apicoecto-
tral nervous the conditisan has my nor tooth ex-
system pro- been validalted exten- traction render
cessing. sively as a iclinical en- the affected area
These new free of pain. On
findings have tity the contrary, pro-
radically cedures and other
changed our understanding of surgical interventions such as
the diagnosis, pathogenesis and trigeminal rhizotomy and mi-
treatment of phantom tooth crovascular decompression fre-
pain, phantom stump pain and quently exacerbate pain and
other phantom sensations. may even enlarge its distribu-
tion.
PHANTOM TOOTH PAIN Reports on tooth pain of ob-
The most common form of orofa- scure origin are relatively re-
cial phantom pain is phantom cent.8 The term phantom tooth
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CLINICAL PRACTICEm

pain was first used in 1978,9

and since then the condition
has been validated extensively
as a clinical entity.34'10-'4 Other
terms for phantom tooth pain,
such as atypical odontalgia,"
idiopathic odontalgia and atypi-
cal facial pain, also are in use."6
Phantom tooth pain resem-
bles other phantom pain syn-
dromes that arise following am-
putation and injury. Some
suggest that neuropathic pains
that have a predominantly
central "generator" comprise
the so-called deafferentation
pain syndromes."7 This theory
does not exclude the possibility
that a peripheral lesion is re-
quired to sustain the pain re-
sulting from the central genera-
tor (Figure 1).18
Data suggest that dental
pulp amputation not only re-
sults in a lesion at the tooth
apex but, consistent with other
nerve injuries, also influences
the central nervous system
(Figure 2). 9-21 Ironically, pulp
extirpation may not only serve Figure 2. Painful areas often Identified by phantom tooth pain patients: 1.
to abolish acute pain, but also nasolabial fold, 2. mental nerve, 3. maxIllary sinus, 4. infraorbital nerve, 5.
might serve as an initiating fac- palpebral nerve, 6. supraorbital nerve, 7. temporalis muscle, 8. occipital
nerve, 9. extemal pterygoid muscle, 10. infranares. Circle size indicates
tor in chronic pain. complaint frequency.
Figure 3 presents criteria
and Figure 4 clinical character- - The pain is described as a consitant, duxll, deep ache with
istics of phantom tooth pain. occasiornal spontaneous sharp pains. There are no refracto-
ry periods.
Other researchers have corrobo-
rated many but not all features - Peripheral stimuli can momentarily exacerbate the pain
but have no prolonged influence. Percussion over the site of
first described by Marbach9 the injured nerve may result in Tinel's sign.
(gender,'3 psychology22). -These stimuli can be of a type normally not nociceptive.
There appears to be a lowered pain threshold (allodynia).
INTRAORAL STUMP PAIN
- Radiographic and laboratory tests are negative.
Stump pain is a frequent se-
quela of limb amputation. Davis - Sleep is undisturbed by pairn or other phantom sensa-
tions. Many patienats report a brief symptom-free period on
cited this pain as a major cause awakening. This period lasts from seconds to about one
of prosthetic limb rejection hour.
among amputees.23 Recently, Figure 3. Criteria for phantom tooth pain.
the connection between stump
pain and pain associated with stump pain.24 No prior reference mucous membrane covering the
denture use was postulated. to intraoral stump pain per se alveolar process or deep within
Sherman and colleagues sug- has been found. the process itself. Stump pain
gested that denture pain is the Patients report that the site does not disappear with time or
intraoral equivalent of limb of intraoral stump pain is in the with adjustments or replace-

JADA, Vol. 127, February 1996 223

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-C1INICA1 PRACTICE
ments of the prostheses. The
health and financial conditions
of patients who complain of this
phantom pain benefit when
treatment is directed toward in-
traoral stump pain and focused
away from mechanically based
etiologies.6
It is primarily the elderly
who undergo multiple tooth ex-
tractions and wear dentures.
Sherman found that age and
disease can reduce the thick-
ness of stump tissues to the ex-
tent that a prosthesis may im-
pinge on the underlying
tissues.2" Others suggest that
pain produced by innocuous me-
chanical stimuli, termed me-
chanical allodynia, results from
activation of large-diameter,
low-threshold mechano-recep-
tive afferent fibers.7 Mechanical
allodynia also has been impli-
cated in neuroma pain.26 These
studies offer an explanation for
how light touch or pressure, as
occurs under a denture, can re-
ception and localization of the pain site.
- Phantom tooth pain occurs in both sexes.
-Phantom tooth pain has been reported i
children.
_ There is nlo e:vid6nce currently that phanitom tooth Xpairn is
characterized by a premorbid personality.
Figure 4. Characteristics of phantom tooth pain.
224 JADA, Vol. 127, February 1996
sult in intense pain. Recogni-
tion of intraoral stump pain as
a potential source of prosthesis-
related pain could result in a
considerable acceleration in the
delivery of appropriate treat-
ment for sufferers.
PHANTOM BITE
SYNDROME
Non-painful phantom limb phe-
nomena are common among re-
cent amputees but usually fade
with time.27 Sensations range
from a minor paresthesia to a
vivid sensation of the entire am-
putated part. The patient's per-
ception of the missing part may
or may not be perceived as an
accurate anatomic representa-
tion. When nonrepresenta-
tional, the phantom limb often
feels larger than the original
and may seem to be rotated on
its axis or fixed in an unnatural
position in relation to the axis
of the body.
Until recently, one type of
adults but not in
orofacial phantom sensation,
phantom bite syndrome, has
been viewed as a psychiatric
disorder.5 6'28 This interpretation
has been revised in light of cur-
rent research.125'27
Brief experiences akin to
phantom bite syndrome are al-
most universal. Most people
who have undergone restorative
dental treatment are familiar
with the delicate perceptual as-
sessments associated with the
final adjustments of even a sin-
gle dental filling.
The onset of phantom bite
syndrome can occur at any
stage of dental treatment. How-
ever, patients typically associ-
ate the origin of phantom bite
syndrome with the construction
of extensive dental prostheses.
Adolescents often experience
phantom bite syndrome when
beginning orthodontic treatment.
Patients with phantom bite
syndrome complain of continu-
ous discomfort and are frequent-
ly distressed by the lack of famil-
iarity of their own bite. Recall
Melzack's third premise concern-
ing how we identify ourselves in
the somatosensory complex of
the brain,1 which dynamically
represents the entire body as a
functional map. Earlier work
also theorized that the sensation
derived through tooth contact
acts as an unique identifier for
each individual's recognition of
the self.5 In other words, when
placing one's teeth together, one
feels one's own bite and not that
of another.
Seeking relief from phantom
bite syndrome often becomes
an expensive and lengthy effort
toward restoration of one's orig-
inal, but now "lost," bite.56 Suc-
cess is rarely, if ever, obtained.
Treatment, therefore, should be
focused on prevention, early de-
tection and patient education.
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CLINICAL PRACTICE

Nonpainful, nonocclusal oro- will increase with an aging popu- mandibular joints and maxil-
facial phantom sensations also lation eager and able to afford lary sinusitis are sometimes
are sources of complaint.5'22 such services. Most likely, the confused with phantom tooth
Other sensations include non- rates of phantom tooth pain pain.
specific sticking sensations, also will rise. The diagnosis of atypical
such as a sensation similar to a facial pain or atypical facial
wooden toothpick fragment or DIFFERENTIAL neuralgia is used commonly, de-
DIAGNOSES
splinter stuck in the gingiva. spite the deliberate rejection of
Some patients report a percep- The clinician should differenti- the term by the International
tion that certain teeth and oral ate phantom tooth pain from Association for the Study of
soft tissues feel enlarged and the typical neuralgias, the most Pain." Atypical facial pain is di-
misaligned. It may be that common of which is trigeminal agnosed by exclusion of the
those individuals who suffer neuralgia.31 Trigeminal neural- signs and symptoms of the typi-
from both pain and nonpainful gia's paroxysmal pain, confined cal neuralgias. The etiology of
phantom phenomena are more to the distribution of the tri- atypical facial pain is attributed
likely to emphasize their pain geminal nerve, differs from the frequently to psychological fac-
symptom, leaving nonpainful dull constant
sensations under-reported. pain of phan-
tom tooth pain.
EPIDEMIOLOGY OF Unlike phan-
PHANTOIIM TOOTH PAIN
tom tooth pain,
Bonica repeatedly emphasized which can de-
the importance of collecting velop with the
data on the incidence and prev- arrival of the
alence of chronic pain disor- first permanent
ders.28 Only one study could be tooth, trigemi-
found that gathered epidemio- nal neuralgia
logic data on phantom tooth usually first ap-
pain.29 The study yielded a rate pears in the
of eight cases per 256 women (3 fifth decade of Figure 5. Radiolgraph of a 22-year-old woman with
percent) who developed phan- life. phantom tooth pain of four years' duration. The mo-
tom tooth pain following endo- Otherpain-
Other lars were
pain- first extraccted after endodontic therapy of the
and seconc d molars. All teeth were treated en-
dontic treatment (only partial ful, typical neu- dodontically an id with apicoectomy.
data on men were collected). ralgias are as-
Other cases met subthreshold sociated with
levels (three of four criteria) for acute herpes
a diagnosis of phantom tooth zoster, post-
pain. By providing evidence herpetic neu-
of a genetic predisposition to ralgia, genicu-
deafferentation pain,30 the focus late neuralgia
of attention was transferred and, of course,
from the tooth to the individual toothache due
as the unit at risk. It appears to pulpitis. In
that a vulnerable person is at addition, mus-
risk each time endodontic ther- culoskeletal
apy is performed. Genetic pre- disorders such
disposition could help explain as temporo-
.
Figure 6. Radiolgraph of a 42-year-old woman with
~~phantom tooth Ipain of two years' duration. Most
the increasing number of pa- mandibular teeth were trealted with endodontic therapy. At the
tients who undergo extensive, pain and dys- initial visit, seveare pain persisted in all the right
mandibular teelth and the right maxillary molars, the
unsuccessful endodontic treat- function
functiosyn-syn-
right side of thea face and regions of the neck. A
ment (Figures 5, 6). drome,15 arthri- total of 23 sepairate root-canal treatments and three
Per capita endodontic treat- tis of the extractions werre performed for relief of pain.
History indicateDs that the original complaint was
ment in the United States likely temporo- that of myofascAal face pain.

JADA, Vol. 127, February 1996 225

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_C LINICAL PRACTICE

tors.32 Many associate atypical logic nerve pain states in hu- are generated exclusively in the
facial pain with female gender, mans.37 An electron microscopic peripheral nervous system.
depression, anxiety and hyste- analysis of nerve tissue exposed Devor and several colleagues
ria, although evidence for these to the same type of injury dis- objected to the term deafferen-
correlations is lacking.432 Clini- plays a near-complete loss of tation pain because, in humans,
cians have turned to a psycho- large myelinated fibers distal to rarely is the source of pain
logical explanation for phantom the injury. There also was dam- demonstrably not an afferent
phenomena because the disor- age to small myelinated fibers, input entering the central ner-
ders do not follow the tradition- suggesting that changes in all vous system.18 As evidence they
al patterns asso- calibers of pe- cited the persistence of afferent
ciated with the ner- activity in several peripheral
biomedical Musculosk eletal dis- ripheral
wietadig- vous system nerves even after complete
model of dis- ouders suclh as ter- axons may nerve transection. In the case of
ease.33 poromandiibular pain contribute to dental pulp amputation, the foci
Furthermore, neuropatholog- of abnormal impulse generation
atypical facial and dysfun ction syn- ic pain in cer- could be neuroma formation at
pain patients drome,5s atithu tie of tain types of the root apex. Devor also object-
are recalcitrant the temperemandibu- findings, injury.38 These ed to the phrases wound heal-
in undergoing along ing, used here (Figure 1), and
conventional lar joints amid m il- with evidence complete healing, used else-
treatments, and lary sinusitis are that allodynia where by Marbach.3 He argued,
examination re- sometimes confused associated correctly, that this cannot be
veals high rates with neuro- the case, since axons have been
of psychiatric co- with phanhDm tooth pathologic cut and the normal target that
morbidity, par- pain3 pain condi- they could potentially regener-
ticularly depres- tions involve ate into, the dental pulp, is
sion.33-36 Atypical altered central gone. The terms healing and re-
facial pain may indeed be a case nervous system processing,7 covering were used by Marbach
of misclassification. Examination argue for a combined peripher- to indicate the clinical absence
of the atypical facial pain litera- al-central nervous system etiol- of pain, not the regeneration of
ture suggests that many of ogy for phantom tooth pain. axons. Others suggested that
those patients diagnosed with Devor and others have pro- deafferentation pain is a useful
atypical facial pain meet crite- posed a working model of term. They emphasized the clin-
ria for phantom tooth pain. neuropathologic pain.18 Figure 1 ical similarities between vari-
is adapted from this model. It ous central and deafferentation
PATHOPHYSIOLOGICAL posits changes in both the pe- syndromes. 17,39,40
MECHANISMS OF OROFA-
CIAL PHANTOM PAIN ripheral and central nervous Melzack's theory of "a geneti-
systems. In the first hypothesis, cally built-in neuromatrix for
Phantom tooth pain is persis- "central sensitization" persists the whole body" postulates a
tent, allodynic and frequently, after nerve injury. No further continuous exchange of infor-
but not always, delayed in triggering signal is necessary. mation from the environment
onset. These characteristics, Central sensitization is a term and peripheral nervous system
combined with the fact that the used to signify changes in the to the brain.' Applying the theo-
dental pulp has been entirely central nervous system, only ry to teeth, one could predict
amputated, argue for the role of some of which have been de- that there exists a genetic pro-
a central nervous system mech- scribed. The second hypothesis gram governing the neuro-
anism.3 18 This hypothesis is requires a repeat or renewal of matrix code for the loss of the
strengthened by the fact that the triggering mechanism be- deciduous but not the perma-
phantom tooth pain is rare. cause the period of central sen- nent teeth. A study of kittens
Recent evidence shows that in- sitization following peripheral reports that "natural tooth deaf-
jury to a peripheral nerve in the injury fades. ferentation" associated with ex-
rat results in a pain syndrome In the third hypothesis, foliation of the primary teeth
similar to that of neuropatho- painful neuropathologic signals may differ considerably from

226 JADA, Vol. 127, February 1996

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CLINICAL PRACTICE-

the "sudden insult of deafferen- of an arm amputee, so, too, can ences between the phantom
tation imposed by the simple pain be produced in the face by tooth pain sample and with the
endodontic procedure in the the similar neural patterns. control groups were higher
adult cats."'41 This observation scores on a measure of demoral-
provides independent evidence TREATWMENT ization. Because demoralization
for a difference between decidu- Treatment based on two meth- can be interpreted as a conse-
ous and permanent tooth loss. ods has been extensively re- quence as well as an antecedent
The neuromatrix theory also viewed.3 Oral medications influ- of chronic pain," this study does
could account for the lack of re- ence afferent impulses that not show that phantom tooth
ported phantom tooth pain culminate in central synaptic pain cases are characterized by
among children. excitability. The second, nerve a specific premorbid personality.
The neuromatrix theory block injections, is directed at
suggests an alternative to the the changes in the chemistry of SOCIAL AND MEDICO-
LEGAL FACTORS
psychodynamically oriented transported substances in the
view of phantom bite syn- peripheral nervous system. The social difficulties that arise
drome's etiology. Prior work Another focus involves pro- out of chronic pain conditions
was based on chronic patients, fessional education that targets may result in frustration and
whose statuses were often bi- prevention and early diagnosis. resentment on the part of both
ased by emotional reports of This approach limits unneces- the patient and the clinician.
failure related to sary deaf- Phantom phenomena account
extensive dental Just as Ube!brain can ferentation for a considerable amount of
interven- produce dDo,mmands and promotes litigation.
tions.5,6,42 The early treat- Besides psychological mor-
sparse literature for move ent to ment. Neuro- bidity, chronic pain patients
was confined to limbs tha tlhave been surgical treat- often experience strained social
psychoanalytic amputate 'd, so, too, ments should interactions that result from
theory. In con- be avoided, as unsolved problems related to
can pamn be hproduced they are asso-
c
trast, the neuro- their clinical management. In a
matrix theory in the fac by the ciated with series of studies, facial pain pa-
posits that the similar neguiral pat- high morbidi- tients identified dentists and
knowledge of ty.43 Topical physicians as the chief source of
every cusp and terns. drug applica- their perceived estrangement
groove compris- tion has shown and feelings of being psychologi-
ing the individu- promise in cally flawed.45 Fifty-two percent
al's dental occlusion resides in early trials with some neuro- of respondents reported having
the brain as a coherent unit or pathic pain syndromes. been told by a clinician that their
occlusal neurosignature. Thus, facial pain could be imaginary.
for some individuals, alteration PSYCHOLOGICAL CON- Feelings of social stigma are
SIDERATIONS
of the dental occlusion results not the result of the patient's
in a neural input that the neu- The symptoms of phantom tooth personality problems. Feelings
romatrix cannot recognize as pain often are considered to be of estrangement result from
one's own bite. Regardless, at of psychological origin by those consulting clinicians' pejorative
onset the result is often the unfamiliar with its clinical labeling of the facial pain symp-
same: the person engages in a physical characteristics. One toms as a psychological disor-
lifelong search for the "correct" study compared 115 phantom der. When this happens, patients
bite. Some of these patients de- tooth pain cases with a group of are placed in a situation ripe for
velop myofascial face pain sec- 151 myofascial facial pain cases pejorative labeling by clinicians
ondary to the phantom bite syn- and 137 non-pain controls on a or even by close relatives.
drome. Just as the brain can variety of personality character- Indeed, facial pain patients re-
produce commands for move- istics.4 Only one personality fac- ported estrangement in and dis-
ment to limbs that have been tor, locus of control, statistically satisfaction with intimate rela-
amputated or painful fatigue in differentiates the three groups. tionships, particularly with
a tightly clenched phantom fist The chief psychological differ- spouses.46

JADA, Vol. 127, February 1996 227

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-CLINICAL PRACIICE
Together, these two studies
indicate that many facial pain
patients, in a search for a
meaningful explanation of their
chronic pain, encounter nega-
tive reactions from clinicians,
family and friends. Clinicians,
in turn, have been shown to re-
sent chronically ill patients for
whom they play a caretaking
role.45 They tend to depersonal-
ize those who do not improve
over a long period. The sense of
stigma that arises from such la-
beling not only causes alienation,
but may also compel sufferers to
undergo treatments that may be
inappropriate in an effort to le-
gitimize their complaints.
Patients unfamiliar with the
potential side effects of dental
treatment may assume their
symptoms are the result of sub-
standard care. Alternatively,
well-intentioned clinicians
sometimes undertake cases for
which the are unprepared.
Education can help alleviate
these problems. Strategies to
deal with medico-legal disputes
are discussed elsewhere in de-
tail.47,48
CONCLUSION
Progress in the management of
orofacial phantom phenomena,
both painful and nonpainful, is
undergoing rapid acceleration.
These formerly enigmatic clinical
states are yielding their secrets
to neuro- and behavioral scien-
tists. The construct of the neuro-
matrix provides a hypothesis-
generating foundation for further
investigation.
Phantom pains represent
complex phenomena that do not
behave in a linear fashion and
cannot be explained by the re-
ductionism of the mind-body
split. Wlhen assessing phantom
pain, the sum of the parts is in-
sufficient in understanding the
228 JADA, Vol. 127, February 1996
whole; in these cases, mind-
body interactions are as impor-
tant as the parts. Strong evi-
dence favors a neurological
basis for phantom pain, which
should be pursued with further
research. Research into the con-
sequences of chronic pain of an
unknown origin also is vital. .
Dr. Marbach is the Robert and Susan
Carmel Professor in Algesiology, University of
Medicine and Dentistry of New Jersey,
Department of Oral Pathology, Biology and
Diagnostic Sciences and Department of
Psychiatry. He is also a clinical professor,
School of Public Health, Columbia University,
New York City. Address reprint requests to
Dr. Marbach, 110 Bergen St., University
Heights, Newark, N.J. 07103-2714.
This research was supported by National
Institute of Dental Research Grant No.
DE05989.
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