Int Endodontic J - 2021 - Sanner - Patients With Persistent Idiopathic Dentoalveolar Pain in Dental Practice
Int Endodontic J - 2021 - Sanner - Patients With Persistent Idiopathic Dentoalveolar Pain in Dental Practice
DOI: 10.1111/iej.13664
ORIGINAL ARTICLE
1
Dental Office, Frankfurt, Germany Abstract
2
Carolinum, Clinic of Conservative Aim: To assess whether persistent idiopathic dentoalveolar pain (PIDAP), a diagno-
and Preventive Dentistry, Frankfurt am
Main, Germany
sis of exclusion, exhibits common features that can facilitate its diagnosis. PIDAP is
3
Dental Office, Drachten, The defined by the International Classification of Orofacial Pain (ICOP 6.3.) as ‘Persistent
Netherlands unilateral intraoral dentoalveolar pain, rarely occurring in multiple sites, with vari-
4
Clinic of Conservative and Preventive able features but recurring daily for more than 2 h per day for more than 3 months,
Dentistry, Center of Dental Medicine,
in the absence of any preceding causative event’.
University of Zurich, Zurich,
Switzerland Methodology: In this observational study, participants fulfilling the new ICOP di-
agnostic criteria of PIDAP were included, covering 16 years of consecutive data. A
Correspondence
Frank Sanner, Dental Office, Niedenau
systematic, retrospective assessment of patients utilizing the new PIDAP criteria on
50, Frankfurt 60325, Germany. complaints of chronic tooth pain in a referral-based endodontic practice was under-
Email: [email protected] taken. Non-idiopathic cases were excluded on the basis of clinical and radiologic
findings. A modified neuropathic pain questionnaire was used to describe the pain-
ful sensations. Furthermore, allodynia in the periodontal space and sensory changes
in the oral mucosa were assessed using a periodontal probe.
Results: Amongst the 160 patients assessed, 78 (63 women) fulfilled the strict
PIDAP criteria. Pain history of PIDAP included no nocturnal awakening (85%) and
a ‘pulling/dragging’ pain quality (59%). In 69% of the patients with PIDAP, pain was
associated with a root filled tooth at the same site. In 14% of the cases, no endodontic
treatment was performed in the affected quadrant. Mechanical allodynia in the gin-
gival sulcus was observed in 91% of patients with painful teeth or implants.
Conclusions: In this observational study, PIDAP mainly affected females and was
associated with undisturbed sleep and periodontal allodynia.
KEYWORDS
endodontic diagnostics, International Classification of Orofacial Pain, persistent idiopathic
dentoalveolar pain
I N T RO DU CT ION The main reason for this pain is usually endodontic in-
fection (Zehnder & Belibasakis, 2015). However, whilst
Dental pain is a relatively common condition with a sig- toothaches from endodontically involved, i.e. infected
nificant socioeconomic impact (Constante et al., 2012). teeth can be excruciatingly intense, they do not normally
This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided
the original work is properly cited.
© 2021 The Authors. International Endodontic Journal published by John Wiley & Sons Ltd on behalf of British Endodontic Society.
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232 PIDAP DENTAL SIGNS AND SYMPTOMS
last long before the patient seeks dental care (Rechenberg this observational study was on pain history, dental signs
et al., 2016). This form of ‘typical’ tooth pain usually re- and symptoms.
solves after endodontic intervention (Law et al., 2015). The data collected in this study made it possible to
On the other hand, patients with persistent toothache apply the new ICOP criteria for PIDAP and rule out
require dentists to differentiate between odontogenic, other possible causes for chronic or persistent pain to
non-odontogenic and mixed pain. In contrast to ‘typical’ the maximum extent by characterizing this group of
tooth pain, pain with a neuropathic or idiopathic charac- patients.
ter presents a greater clinical challenge. The pathophysiol-
ogy (Vickers & Cousins, 2000), classification (ICOP, 2020),
risk factors (Aggarwal et al., 2010), diagnosis, prognosis MATERIALS AND METHODS
and therapy for persistent dentoalveolar pain have been
discussed extensively (Malacarne et al., 2018). However, Patient evaluations
despite newer classifications being published, many open
questions remain. Prospective data collection in all patients with chronic tooth
The new International Classification of Orofacial Pain pain was performed by the principal investigator (F.S.), a
(ICOP) classification emphasizes the difference between trained endodontist and a physician with a focussed inter-
neuropathic and idiopathic pain that can result in per- est in pain, between January 2003 and June 2019. The use
sistent dentoalveolar pain. The latter was the subject of of this censored data for publication was approved by the
this observational study. In this context, ‘Idiopathic’ is de- local ethics commission (LZK Hessen, 01/2020). The ma-
fined as ‘arising spontaneously or from an obscure or un- jority of patients were referred from other endodontists
known cause’ (Merriam-Webster 2021 https://www.merri within Germany because of ongoing pain after treatment
am-webster.com/dictionary/idiopathic). (Figure 1). Patients aged >18 years who had experienced
Patients experiencing persistent dentoalveolar pain pain persisting for more than three months at the time of
experience significant psychological strain that adversely the examination were eligible for inclusion. All 160 patients
affects their quality of life (Shueb et al., 2015). Persistent reported pain related to a tooth or extraction site.
idiopathic dentoalveolar pain (PIDAP), which has been Good-quality periapical radiographs of the painful re-
redefined most recently (ICOP, 2020), is mainly a diagno- gions were available and examined in all patients. Any
sis of exclusion. For the clinician, however, the typical fea- signs of caries, fractures, bleeding on probing, patho-
tures of PIDAP can help to better distinguish this type of logical pocket depths (>3.5 mm), mucogingival lesions,
pain from odontogenic and other possible pain conditions internal or external resorption or possible lesions of end-
at an early stage. odontic origin resulted in the exclusion of the patient from
Thus, the goal of this observational study was to de- the study group (Table 1). Patients suspected to have pain
scribe patients with PIDAP from the dentist's perspective of a dental inflammatory or traumatic nature or due to
and to identify possible common features within this co- other sources of pain related to teeth were excluded from
hort. From a series of consecutive patients complaining of this analysis, as were patients with suspected pain referred
chronic tooth pain in a referral-based endodontic practice, from adjacent anatomic structures, such as acute sinusitis,
patients who fulfilled the ICOP diagnosis of PIDAP (ICOP, sialadenitis, myofascial pain, arthrogenic pain and signs of
2020) were included over a period of 16 years. The focus of trigeminal neuralgia (Figure 1). Patients with a history of
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SANNER et al. 233
external trauma and iatrogenic injuries from dental treat- Pain questionnaire and interview
ments such as local anaesthetic injections, extractions,
root canal treatment, oral surgery, dental implants, or- All patients completed a pain questionnaire for atypical
thognathic surgery and other invasive procedures in the or persistent tooth pain. This questionnaire (Appendix
painful area within 6 months before the onset of pain were S2) consisted of 46 standard questions, first assessing the
also excluded because of possible or likely post-traumatic main location of the pain and then addressing the onset,
trigeminal neuropathic pain (PTTNP) according to ICOP intensity, quality, medications, sleep patterns and other
4.1.2.3 (ICOP, 2020) when sensory changes in a neuroana- possible contributing factors. A history of surgery or
tomically plausible area were observed. trauma to the head and neck, history of root canal treat-
All pain interviews and clinical examinations were ments and diseases of the nervous system were investi-
conducted by the principal investigator. After their dental gated. In patients who underwent root canal treatment
examinations reported here, the patients diagnosed with in teeth associated with the painful site, the sequence
PIDAP were referred to headache and facial pain clinics of events was noted. This questionnaire was based on
or specialized pain therapists. a standard neuropathic pain questionnaire (Krause &
Backonia, 2003) and recommendations made by Goulet
(2001). Additionally, pain-related symptoms of the oro-
Medical history facial autonomic system and symptoms related to other
orofacial structures were addressed. The patients then
The medical history was obtained using a health question- reviewed their questionnaires with their examiners, who
naire that included questions pertaining to current health obtained clarification for ambiguous answers. The 11-
status (Appendix S1) and the use of prescribed drugs point numeric pain rating scale (NRS-11) was used to
taken on a regular regimen, based on the answers given rate pain intensity (Warren Grant Magnusson Clinical
by the patients. Center, 2003).
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234 PIDAP DENTAL SIGNS AND SYMPTOMS
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SANNER et al. 235
T A B L E 2 PIDAP patient
Sex (female/male) 63/15
characteristics, pain duration, intensity
Age in years (mean ± SD) 49 ± 14
and location
Pain duration in years (median/IQR) 1.5/(1.4)
Mean NRS-11 pain intensity during the last 2 weeks (median/IQR) 3/(2.5)
Maximum NRS-11 pain intensity during the last 2 weeks (median/IQR) 8/(5.9)
Affected jaw (maxilla/mandible) 45/33
Tooth region (molar/premolar/front) 38/25/15
Dentition at the painful site (tooth/edentulous/implant) 75/2/1
History of root canal treatment at the painful site (yes/no) 54/24
Abbreviations: IQR, inter-quartile range (0.25 Quartile, 0.75 Quartile); NRS, Numerical Rating Scale for
pain intensity; SD, standard deviation.
as extractions, oral surgery, dental implants, orthognathic mean: 7.2). The mean average pain score in the same pe-
surgery and other invasive procedures. In 12 cases, per- riod was 3.8 (median: 3), and the mean NRS-11 scores for
sistent pain preceded the root canal treatment, and in 24 female and male patients were 3.8 and 4.1, respectively.
cases, no root canal treatment or other invasive dental Sixty-six of the 78 (85%) patients stated that the pain
procedures were performed on the tooth. In 12 cases, the did not wake them at night. Furthermore, 64 (82%) pa-
root canal treatment preceded the beginning of persistent tients experienced pain-free intervals on waking in the
pain for less than 6 months, but since sensory changes morning before the pain set in. Pain was present during
were found in other quadrants as well, these cases met the daytime for more than 2 h in all patients.
the criteria for PIDAP, not PTTNP (Appendix S3). Cases Patients generally used more than one term to describe
in which the timing of dental procedures was not known the nature of their pain. The most frequently used adjec-
were not included in the study. tives were as follows: ‘pulling/dragging’, reported by 46 of
78 patients (59%); ‘pressing’ by 41 (52%); ‘burning’ by 33
(42%); ‘tingling’ by seven (9%); ‘electric’ by three (4%); and
Medical history and medication usage ‘stabbing’ by 14 (18%). Forty-three (55%) patients reported
experiencing pulsating or throbbing pain at the affected
Other chronic pain conditions reported by patients with site from time to time. When describing the quality of
PIDAP included chronic back pain (8 individuals), irrita- their pain in more words, patients frequently used non-
ble bowel syndrome (3), migraine (4), fibromyalgia (2) and standard, figurative expressions that differed from more
tension headache (1). A history of depression was reported common ways of describing toothaches, such as ‘like
in nine cases and psychosis in one. The most frequently being stabbed with a knife’, ‘like a wound’, ‘like dead tis-
reported medical conditions were thyroid disorders (21), sue that is nevertheless painful’, ‘as if the tooth were to
including 12 cases of Hashimoto's thyroiditis. Eleven pa- explode’ and ‘as if the bones were moving’.
tients had cardiovascular and respiratory disorders. Other
disorders or diseases were present in very small numbers
of patients. Twenty-one patients reported no other health Clinical findings
problems besides the existing persistent intraoral pain.
Drugs taken on a regular basis included l-thyroxin (18), Pressure to the periapical bone exerted by digital palpa-
contraceptive medication (4), drugs used for chronic pain tion was never painful, nor could pain be elicited by ver-
treatment (8), psychotropic drugs (3) and cardiovascular tical tapping on a tooth with an instrument in any case.
drugs (10). Discomfort was elicited by horizontal tapping in six pa-
tients. Mechanical allodynia of the gingival sulcus was
found in 68 of 75 teeth and in one implant. Thus, allo-
Pain characteristics dynia in the periodontium/peri-implant mucosa was ob-
served in 69 of 76 patients with painful teeth or implants
The pain had been present for an average period of (91%). In two of the cases in which the pain affected the
2.7 years before the examination (median: 1.5; range: edentulous region, this sign could not be tested. The pain-
4 months to 13 years). A group of 17 women had pain ful section invariably consisted of only part of the gingival
persisting for more than or equal to five years. The maxi- sulcus and never included its entire circumference. The
mum pain intensity during the 2 weeks prior to assess- same symptoms could frequently be elicited in the sulcus
ment ranged from 2 to 10 on the NRS-11 scale (median: 8; of the adjacent teeth or in the antagonistic or contralateral
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236 PIDAP DENTAL SIGNS AND SYMPTOMS
quadrants. An allodynic area in the gingival sulcus of the pain was present from the beginning and a root canal
neighbouring teeth was present in 53 cases. More than treatment was performed with the intent to help the pa-
one quadrant was affected in 39 cases: two quadrants in 22 tient. This point should be addressed in future studies. Sex
cases, three quadrants in 11 cases and all four quadrants dependence of chronic pain has been frequently described
in six cases. (Mogil, 2020). A previous study reported a predominance
The gingiva and mucosa of all patients were checked of female patients (List et al., 2007), which was also con-
for sensitivity changes to touch by using the tip of a peri- firmed in this study.
odontal probe to slightly touch the gingiva and mucosa in
the painful quadrant, first on the non-painful contralat-
eral side and then on the affected painful side. In 9 cases, Medical history
patients reported numbness near the site of the pain
after repeated comparisons with the non-painful contra- Thyroid disorders (27%), chronic pain conditions (23%),
lateral side. None of the patients had been aware of this and depression (10%) were reported by the patients.
numbness before the examination. Twenty-eight patients Because this information was questionnaire-based, its
showed sensitivity changes other than numbness in the completeness and accuracy were not assured. It was only
painful quadrant beyond the gingival sulcus; mechani- included to describe the group of patients under investi-
cal allodynia was present in five patients. Seven patients gation in more general terms. Nevertheless, the relatively
reported reduced sensitivity to touch (hypoesthesia) and high frequency of thyroid disorders in this cohort warrants
15 reported discomfort when touched with a periodontal further research. An examination of a large sample of self-
probe compared to the non-painful side. In seven patients, reported chronic pain (McWilliams et al., 2003) found that
more than one sensitivity change in the fixed gingiva was the incidence of apparent thyroid disease amongst indi-
present concurrently. viduals affected by chronic pain was 5.5%.
The painful sensation sometimes increased as a result
of the examination, especially on contact with the gingival
sulcus. A total of 32 patients reported an increase in pain Pain characteristics
levels after completion of the examination.
The maximum pain intensity of the PIDAP pa-
tients under investigation that was reported for the
DI S C US S I O N two weeks prior to clinical assessment was similar
to that of a cohort of endodontic emergency patients
In this observational study, strict PIDAP criteria were ap- with infection-related pain (median of 8 on the NRS-
plied to illuminate the condition from the dentist's per- 11 scale) (Rechenberg et al., 2016). However, and more
spective. This allowed the identification of PIDAP features importantly, the finding that the pain did not wake
that have not been described previously, including the ab- patients at night in these individuals with PIDAP sets
sence of nocturnal pain and the presence of allodynia in their condition apart from infection-related tooth pain.
otherwise healthy periodontal sites. In the study by Rechenberg et al. (2016), 80% of emer-
gency patients experiencing acute pulpitis and 83% of
those with acute apical periodontitis stated that their
PIDAP patients pain woke them at night. An increase in intensity when
lying down or resting is typical of inflammation-related
Until recently, the nomenclature for the conditions under pain. Atypical odontalgia that affects patients mostly at
investigation was not standardized (Nixdorf et al., 2012). and throughout the day, on the other hand, has been
A new ICOP was introduced in 2020 (ICOP, 2020), which reported previously (Rees & Harris, 1979) but has never
was followed here. Studies on atypical odontalgia in endo- been specifically assessed.
dontic patients have reported an occurrence rate of <5% Similar pain descriptors as in this study have been
(Campbell et al., 1990). An association between persistent reported previously in the literature (List et al., 2007). A
toothaches and endodontically treated teeth has also been striking typical pain characteristic in the population stud-
reported (Nixdorf et al., 2010). ied here is the burning pain quality, which affected 42% of
The association between root canal-treated teeth and the patients. Burning pain is also generally described as a
non-odontogenic tooth pain could be explained by either characteristic of neuropathic pain (Collaca et al., 2017).
pathophysiological changes under special circumstances Undisturbed sleep and a pain-free interval after waking
following root canal treatment that led to pain chronifica- are typical characteristics of neuropathic pain (Odrcich
tion or, more likely, indicating that non-odontogenic tooth et al., 2006).
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SANNER et al. 237
A delayed onset of pain following irritation (41%) has Although extraoral QST has been standardized (Matos
also been described in neuropathic pain. In this regard, et al., 2011), and intraoral QST has been described (Baad-
mechanisms similar to those of ‘wind-up’, a progressive, Hansen et al., 2015), more practical means for dental prac-
frequency-dependent increase in excitability, may play a titioners are needed and developed in the form of QualST
role (Coste et al., 2008). (Baad-Hansen et al., 2013).
Probing of the gingival sulcus is usually not painful. In
the present study, a high percentage of patients (91%) had
Clinical findings this sensory abnormality, often not only in the painful tooth
but also in other teeth and quadrants. Neurophysiological
Persistent idiopathic dentoalveolar pain was the most research on this topic has shown that trauma to one den-
frequent diagnosis amongst patients with chronic den- tal nerve may alter the processing of pain in neighbouring
toalveolar pain. In addition to nociceptive and inflam- teeth and other quadrants (Kondo et al., 1992; Sabino et al.,
matory pain, as is the case with odontogenic pain, from 2002). Sabino et al. (2002) reported a correlation between
a pathophysiological point of view, there are two other the invasiveness of dental procedures and neuroplastic
pain types frequently implicated in chronic pain, namely changes in the brainstem. The invasiveness of the pro-
neuropathic pain (Finnerup et al., 2016) and neuroplastic cedures was correlated with the extent of changes on the
pain (Kosek et al., 2016). same and contralateral sides. In the present study, allody-
The ICOP classification describes the type of pain nia in the gingival sulcus, which could be a result of neu-
under investigation as idiopathic. roplastic changes, was also found in 68% of neighbouring
The diagnosis of non-odontogenic toothaches is usu- teeth and in 50% of other quadrants. More insights can be
ally a differential diagnosis based on the absence of any gained into this topic by obtaining better knowledge of the
explanatory tooth-related pathology. Although the sign innervation of the junctional and oral epithelium within
of non-response to local anaesthesia was not examined in the sulcus and the periodontal ligament, the sensory
this study, such non-response at the site of the pain (List changes within the periodontal space, and the changes in
et al., 2006) can be helpful in the diagnostic process. the nerve following separation of the pulp from its nerves,
as they occur during endodontic treatment.
The inclusion of mechanical allodynia within the gin-
Sensory changes gival sulcus into the already described PIDAP diagnostic
features may provide additional important diagnostic
Simple methods available to any general dentist can be ap- utility.
plied to detect gross sensory changes in the gingiva and
mucosa. Somatosensory tests such as the ‘Quantitative
Sensory Test’ (QST) (Maier et al., 2010) and the ‘Qualitative Strengths, limitations and outlook
Sensory Test’ (QualST) (Baad-Hansen et al., 2013) have
been described for sensory testing. The present data col- The strengths of this study include the fact that an at-
lection started earlier than the description of the sensory tempt was made to systematically exclude nociceptive
tests described above. Nevertheless, a different aspect of odontogenic pain, referred pain, facial headaches and
sensory testing has been emphasized. A simpler approach post-traumatic neuropathic pain to the maximum ex-
using a periodontal probe was followed. The area con- tent possible. The inclusion and exclusion criteria were
nected most closely to the dental nerve and pulp is the chosen to avoid creating a mixture of different charac-
periodontium and hence the gingival sulcus. Pulpal and teristics within the group with otherwise unexplained
periodontal innervation develop concurrently with the non-odontogenic pain (headache variants, referred pain,
development and eruption of the dentition. Innervation odontogenic pain) and to identify characteristics of spe-
occurs during tooth eruption (Fristad et al., 1994). The cific groups that may be due to changes in the pain pro-
periodontal space is characterized by unique innervation. cessing system typical of PIDAP.
Sensory changes in this region related to PIDAP have not Nevertheless, this observational study was limited by
yet been studied, nor do the intraoral variants of QST the fact that it was not a cross-sectional study, and the
and QualST cover this aspect. Quantitative sensory test- pain and treatment history depended on the patient's
ing is mentioned as a means for further specifying PIDAP memory. As a result, no conclusions can be drawn re-
into 6.3.1 with and 6.3.2 without somatosensory changes. garding the prevalence of the conditions under investi-
Further discrimination of PIDAP depending on sensory gation. Furthermore, there was a selection bias because
findings must be taken into consideration, since this clas- patients were mostly seen in endodontic clinics (Figure
sification is very dependent on the methods involved. 1), which represents a specific cohort and may not be
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238 PIDAP DENTAL SIGNS AND SYMPTOMS
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