assessment and management of chronic orofacial pain associated with a disease in children: a...

REVIEW ARTICLE

Assessment and management of chronic orofacial pain associated with adisease in children: a multidisciplinary approachViolaine Sma€ıl-Faugeron ([email protected])1,2, Fr�ed�eric Courson1,2, Charles-Daniel Arreto1,3

1.Service d’Odotontologie, Assistance Publique – Hopitaux de Paris, Hopital Bretonneau, Paris, France2.Unit�e de Recherches Biomat�eriaux Innovants et Interfaces, Facult�e de Chirurgie Dentaire, Universit�e Paris Descartes – Sorbonne Paris Cit�e, Montrouge, France3.Centre de Psychiatrie et Neurosciences, Institut National de la Sant�e et de la Recherche M�edicale, UMR U894, Paris, France

KeywordsChronic orofacial pain associated with a disease,Child, Pain measurement, Multimodal therapy,Multidisciplinary pain clinics

CorrespondenceViolaine Sma€ıl-Faugeron, Unit�e de RecherchesBiomat�eriaux Innovants et Interface EA4462, 1 rueMaurice Arnoux, 92120 Montrouge, France.Tel: + 33 1 58 07 67 82 |Fax: + 33 1 58 07 68 99 |E-mail: [email protected]

Received24 October 2012; revised 26 February 2013;accepted 12 April 2013.

DOI:10.1111/apa.12270

ABSTRACTTo summarize the diversity of assessment and management for chronic orofacial pain

associated with a disease (COFPAD) in children. We performed a review of the literature up

to May 2012. Hetero-evaluation and self-assessment are used according to age of children.

Strict management of the cause is not sufficient for children with COFPAD without a

multidisciplinary approach combining pharmacotherapy, psychology and physiotherapy.

Conclusion: The multidisciplinary approach is the key of management for children with

COFPAD.

INTRODUCTIONAs in adults, in children, pain is naturally occurring.According to the International Association for the Studyof Pain, chronic pain is a complex process defined as anunpleasant sensory and emotional experience associatedwith actual or potential tissue damage, or described in termsof such damage, persisting beyond 3–6 months, with apredominant psychosocial dimension. However, diagnosis,assessment and management of chronic orofacial painassociated with a disease (COFPAD) in children are moredifficult for several reasons.

Indeed, diagnosing the pain is difficult because it maybe unrecognized or considered absent owing to difficultiesin communicating with the child. Even with evidence-based medicine, pain continues to be undervalued inchildren.

Assessing chronic orofacial pain in children may presentproblems because children and adolescents express them-selves differently from adults, which may hinder thetherapeutic relationship that is based on reasonable

communication. Assessing pain by verbal communicationin premature babies or in children up to 2 years is aproblem because of their limited capacity to communicate.Pain is biphasic, with a short ‘noisy’ initial phase followedby a phase of psychomotor slowness. Indeed, signs of painmay vary depending on whether it is acute, prolonged orrecurrent. Chronic pain begins with an acute initial phase.Acute pain gives rise to a host of emotional signs: the factthat the child cries, screams, and cannot be consoled is a

Key notes� The diagnosis, assessment and management of chronic

orofacial pain in children associated with a disease areunique and difficult.

� Thus, a multidisciplinary approach is mandatory tomanage the chronic pain.

� A multimodal therapy should answer both distress ofchildren suffering from chronic orofacial pain associatedwith a disease and concerns of pain care providers.

778 ª2013 Foundation Acta Pædiatrica. Published by John Wiley & Sons Ltd 2013 102, pp. 778–786

Acta Pædiatrica ISSN 0803-5253

determining factor in the diagnosis. Emotional signs maybe associated with direct signs: the child shows where ithurts. It is easy to diagnose acute pain in children. Theconcept of ‘time’ is not the same in the child. The signs ofprolonged pain appear very quickly, changing behaviouraland verbal signs that are more subtle and difficult todetect. The child reduces its activity, becomes calm, sad,apathetic, also known as psychomotor slowness. As well,because the psychomotor slowness phase appears veryquickly and remains so, children may refuse to evaluate orreport their pain, which contributes to misunderstandingthe pain. Consequently, different scales are used to assesschronic orofacial pain, each with its own specificity andsensitivity.

Finally, in contrast to managing chronic orofacial pain,the cause of acute pain, considered a symptom, is treated.However, chronic pain presents as a syndrome, andtreating the cause is necessary but not sufficient, asillustrated by facial neuralgia. Indeed, chronic pain ismultidimensional, so management should be multidisci-plinary. Pharmacological as well as nonpharmacologicalinterventions (such as physical, behavioural and cognitivetherapies) are key interventions to managing chronicorofacial pain in children, and improving relations amongthe child, parents and care providers can also help. Thus,chronic orofacial pain continues to be poorly understoodand managed.

We aimed to summarize the experience of COFPAD, thediversity of assessment tools and the management strategiesby performing a systematic search of the literature. First, wedefine and characterize chronic pain with reference tochildren, then we present diagnostic procedures and causesof COFPAD. Next, we describe the assessment of chronicpain in children, and, finally, management strategies andthe need for a multidisciplinary approach.

LITERATURE SEARCH METHODSWe reviewed the literature on chronic orofacial pain inchildren published in English and French (for the detailedsearch strategy, see Data S1). Any type of clinical study(case series, cohort study, randomized trial) and reviewswere eligible. We searched MEDLINE via PubMed forarticles published up to May 2012 (with no date restric-tion). We also screened the reference lists of selectedreports and hand-searched tables of contents of thejournals Pain and Paediatrics up to 2012. We consultedtextbooks and conferences proceedings of the Interna-tional Symposium on Paediatric Pain up to 2012. We usedthe liberal accelerated approach for selection of articles inwhich one author screened all abstracts, and a secondauthor reviewed records excluded by the first reviewer;then two authors independently reviewed all full-textarticles in duplicate.

The initial search identified 545 references. After screen-ing the titles and abstracts (when available), we obtainedthe full reports for 221 studies. Finally, we reviewed 84 full-text reports (Fig. 1). As several reports were sometimes

available to support the same statement, we referenced the50 most relevant studies for our review.

RESULTSDefinition and characteristics of chronic painDefinitionSince 1979, the broad consensus is that pain is primarily asubjective experience, as outlined in the definition of painadopted by the International Association for the Study ofPain: ‘an unpleasant sensory and emotional experienceassociated with actual or potential tissue damage, ordescribed in terms of such damage’. Pain is consideredhere as a complex experience with several dimensions.Chronic pain is no longer simply defined as acute painpersisting beyond 3–6 months but as a complex processwith a predominant psychosocial dimension (1,2). Acutepain serves as an essential alarm system to protect thebody’s integrity, a pain signal. Some children with congen-ital insensitivity to pain show trophic disorders at an earlyage. However, throbbing chronic pain causes neither reflexavoidance nor learning. It becomes a pain disease.

Dimensions of chronic painIt is generally accepted that chronic pain has multipledimensions. The sensory-discriminative dimension encom-

Figure 1 Flow of articles in the study.

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Sma€ıl-Faugeron et al. Chronic orofacial pain in children

passes the neurophysiologic mechanisms that allow fordecoding the quality, duration, intensity and localization ofchronic pain (3). The affective-motivational dimensionexpresses the more or less painful or unpleasant tone ofpain. It also includes states of anxiety and depression (3).The cognitive dimension refers to mental processes that arelikely to modulate pain perception. Behavioural and envi-ronmental dimensions include motor and nonverbal man-ifestations (facial expression, agitation or prostration), aswell as any verbal expressions (complaints, moans) of pain.Finally, psychosocial components are taken into account(4), as causes but also consequences of chronic pain (5).Thus, the affective-motivational and cognitive-behaviouralcomponents override the sensory-discriminative compo-nent. Girls could have a higher risk of chronic pain thanboys because of differences in psychosocial factors: girlsmight be more likely to internalize pain and exhibitsymptoms of anxiety or depression (6). Sex hormones couldalso be a contributing factor to differences in pain sensitivitybetween boys and girls (7). However, the multidimensionalaspect of chronic pain is consistent with multidisciplinarymanagement, with close collaboration of psychologists,neurologists, physiotherapists, general practitioners anddentists.

Diagnostic procedures and causes of chronic orofacialpain associated with a disease in childrenNeurophysiology of orofacial painThe generative mechanism of chronic orofacial pain isseldom unique. Indeed, chronic pain is multifactorial, andthe nociceptive mechanism (pain created by tissue damage)and the neuropathic mechanism (pain due to nerve dam-age) are often entangled, with the added physical mecha-nism, which moves the initial pain to chronicity. Chronicpain varies in feeling and is influenced by psychological andenvironmental factors.

Causes of chronic orofacial pain associated with a diseasein childrenDiseases responsible for COFPAD can be categorized intofour groups (Table 1). The first corresponds to chronicorofacial pain associated with local abnormalities. Amongthem are necrotizing ulcerative stomatitis, which has abacterial origin. As well, sinus pain can occur as a chronicclinical disease process in children, which often underlinescystic (Fig. S1a) or neoplastic processes (Fig. S1b). Tempo-romandibular disorders include both nociceptive and cog-nitive processes in chronic pain states, with greateroccurrence in girls than boys (8). Also, chronic inflammatorydisorders of the salivary glands induce chronic pain. Head-aches although rare may cause chronic orofacial pain; infact, these two conditions share underlying pathophysiolog-ical mechanisms and clinical characteristics (9,10). Lastly,pulpitis caused by caries is frequently related to acuteorofacial pain but in some cases pulpitis may cause chronicorofacial pain for instance because of a cracked tooth.

The second causative group corresponds to chronicorofacial pain associated with systemic abnormalities. The

grouping of systemic disorders that cause chronic paincould be broadly organized into two domains. First, auto-immune disorders include Crohn’s disease, Lyme disease,multiple sclerosis and coeliac disease. In Crohn’s disease(11), an inflammatory disease of the small intestine, oralulcerations are present in 30% of cases and hurt longer,with their increased depth and greater hyperplastic margins,than do vulgar ulcers. The most frequent clinical manifes-tation of Lyme borreliosis, or Lyme disease, in children isneuroborreliosis. Children can present Lyme meningitis,facial nerve palsy or Lyme arthritis, during which hyperalgicradiculitis but also facial nerve palsy or arthritis may inducechronic pain. Chronic pain due to trigeminal neuralgia canbe detected as an early symptom of multiple sclerosis (12).Oral aphthous ulcers and dental enamel defects in childrencan reveal coeliac disease and may be acommpanied bylong-lasting pain (13). The second domain of systemicdisorders is connective tissue disorders and includes

Table 1 Causes of chronic orofacial pain in children

Categories of diseases and examples Main orofacial manifestations

Local pathologies

Necrotizing ulcerative stomatitis Orofacial gangrene

Sinus pain Chronic sinusal pain

Temporomandibular disorders Dental and facial pain

Chronic inflammatory disorders

of the salivary glands

Oral pain before or during meal

Systemic pathologies

Autoimmune disorders

Crohn’s disease Oral ulcerations with hyperplastic

margins

Lyme borreliosis Meningitis, facial nerve palsy, arthritis

Multiple sclerosis Trigeminal neuralgia

Coeliac disease Oral aphthous ulcers, dental enamel

defects

Connective tissue disorders

Rheumatic diseases Temporomandibular joint pain

Systemic lupus erythematosus Temporomandibular joint pain

Sj€ogren’s syndrome Painful swelling of bilateral parotid,

chronic swelling of the salivary

glands, xerostomia

Systemic sclerosis Xerostomia, dental caries, mobile teeth,

periodontal disease, mandibular

erosions

Benign tumours

Neurofibromatosis type I Chronic neuropathic pain due to nerve

compression (a mass deforms face,

maxilla and mandible)

Langerhans cell histiocytosis Gingival mucosa, bleeding, ulcerations,

teeth loss, oral candidiasis,

osteolytic lesions

Intraosseous hemangiomas Pain due to nerve compression

Neoplasms

Burkitt’s lymphoma Pain due to tumour: nociceptive and

neuropathic pain, gingival bleeding

Pain due to treatments: xerostomia,

trismus, mucositis,

osteoradionecrosis

Orofacial rhabdomyosarcoma

Acute lymphoblastic leukaemia


Chronic orofacial pain in children Sma€ıl-Faugeron et al.

rheumatoid diseases and systemic lupus erythematosus,Sj€ogren’s syndrome and systemic sclerosis. Rheumatoiddiseases and systemic lupus erythematosus inducetemporomandibular joint pain leading to chronic pain.Furthermore, Sj€ogren’s syndrome can produce chronicpainful swelling of bilateral parotids, chronic swelling ofparotid glands and xerostomia (14). Chronic pain isaccompanied by orofacial manifestations of systemic scle-rosis (scleroderma) like xerostomia, which in turn leadsdental caries, tooth mobility, periodontal disease or man-dibular erosions localizing in the mandibular angle, condy-lar head, corono€ıd process and digastric region (15).

The third causative category corresponds to chronicorofacial pain associated with benign tumours. Neurofibro-matosis type I (or von Recklinghausen’s disease) is amucocutaneous syndrome, the most common localizationbeing the tongue with macroglossia. Lips, gingival mucosa,palate and oral floor localizations are also found. Clinically,young patients have a mass that evolves gradually to deformthe face, the maxilla and the mandible with teeth movement(Fig. S2). When osseous structures are affected, nervecompression by adjacent neurofibromas invalidates chronicneuropathic pain. Another disease in this category isLangerhans cell histiocytosis. The most common extra-thoracic localizations are gingival mucosa with bleedingand ulcerations, teeth loss, oral candidiasis and osteolyticlesions with debilitating pain. Finally, intraosseous heman-giomas (Fig. S3) belong to this category as benign tumoursof vascular nature that can originate and expand insidebone structures; these tumours are usually congenital, rarelyof posttraumatic origin (16).

The final causative category corresponds to chronicorofacial pain associated with cancerous tumours. Chronicpain is present at cancer diagnosis in 30% of cases andresults from neighbouring organs compressed by tumour ortissue infiltration by tumour cells. Three of the most-oftenencountered cancers are Burkitt’s lymphoma, orofacialrhabdomyosarcoma (Fig. S4) and acute lymphoblastic leu-kaemia. Burkitt’s lymphoma comprises 30% of paediatriclymphomas, orofacial rhabdomyosarcoma accounts forapproximately 3.5% of the cases of cancer among childrenaged 0–14 years and 2% of the cases among adolescentsand young adults aged 15–19 years and acute lymphoblasticleukaemia is the most common cancer diagnosed inchildren and represents 23% of cancer diagnoses amongchildren younger than 15 years (17,18) (accessed on Janu-ary 1, 2013; Available from: www.cancer.gov/cancertopics/pdq/treatment/childALL/HealthProfessional/page1/AllPages).

Two types of chronic pain caused by tissue infiltrationoccur in children: bone pain (nociceptive pain; Fig. S5) andneuropathic pain (19). However, other nociceptive pain canbecome chronic, such as gingival bleeding. Nevertheless,pain in children with cancer is mostly iatrogenic (20,21).Among the pain situations caused by treatment are xero-stomia, trismus, mucositis and osteoradionecrosis. Simi-larly, both mucositis and osteonecrosis, which may beinduced by chemo- or radiotherapy and result in inflamedmucuous membrane or infection of the injured bone,respectively, are accompanied by chronic pain (22). Finally,chronic graft versus host disease in paediatric patients afterhematopoietic stem-cell transplantation produces oral

Figure 2 Assessment of chronic orofacial pain in children.



mouth pain with oral mucosa abnormality and apparentlack of salivary gland involvement (23).

Assessment of chronic orofacial pain associated with adisease in childrenAssessment of chronic orofacial pain requires the appraisalof pain intensity by both scales and/or questionnaires(Fig. 2). The tools vary by the age of the child. Hetero-evaluation methods, based on behavioural manifestations ofchildren, are available for children younger than 2 (1 tool),for children 2–4 years old (2 tools) and for disabledchildren (3 tools) (24). Moreover, self-assessment methodsare available for children 4–6 years old (4 tools) andchildren > 6 years old (25,26).

Children < 2 years oldThe pain and discomfort scale of the newborn child(Echelle Douleur et Inconfort du Nouveau-n�e, EDIN) wasdeveloped to assess prolonged pain of newborn children,premature infants and children up to 2 years old. The scalewas validated in a sample of 76 preterm infants in France.Common behavioural indices of pain in this scale includefacial, postural and motor behaviours. The scale also coversthe relationship with the caregiver and the possibility ofcomfort (27).

Children 2–4 years oldThe Gustave Roussy Child Pain scale (Douleur EnfantGustave Roussy, DEGR) was developed to assess pain inchildren with cancer aged 2–6 years. The scale includestwo items of complaints formulated by the child, two itemsof psychomotor slowness and five items of physical signs.This is an invaluable tool to monitor changes in treatmentof a young child with much pain. This scale has beensimplified, and a new scale was created: the hetero-evaluation scale of pain in children (H�et�ero-�evaluation dela Douleur de l’Enfant, HEDEN), which was validated ina sample of 152 children with progressive cancer in France(28).

Children 4–6 and >6 years oldQuantitative and qualitative scales of self-reporting areused for children 4–6 and >6 years old. These scales allowfor a global impression of pain in children by an observer.Global pain ratings can be provided by nurses, parents,researchers and other observers. For example, the visualanalogue scale (VAS) (29), the Faces Pain Scale–Revised(FPS-R) and Numerical Rating Scale (NRS) have all beenused as global rating scales. The VAS is most often used inclinical trials. The young patient indicates the level of pain,from ‘no pain’ to ‘pain as bad as it could possibly be’ on ascale in millimetres, so the observer can quantify the painfrom one assessment to another (30). The FPS-R, with ascore ranging from 0 to 10, and the NRS can be used ifchildren have difficulties understanding the VAS. Bothhave been validated (31–33). The Saint-Antoine PainQuestionnaire qualitative scale (Questionnaire DouleurSaint-Antoine) is the French version of McGill pain

questionnaire. Both were validated (34,35) and used inconsultation or with cases difficult to diagnose. In children4–6 years old, one must assess pain of the child with twodifferent scales simultaneously to ensure that the assess-ment is understood and is reliable. In cases of discordantresults or child refusal, one can use the DEGR or HEDENscales described above.

Disabled childrenThe San Salvadour Child Pain checklist (Douleur EnfantSan Salvadour) is the first validated scale for behaviouralassessment of pain in children with multiple disabilities butwithout verbal communication. It has been validated in 50patients with severe mental deficiency in France (36). Thisscale includes two parts: the basic file and the evaluationgrid itself. The basic file describes behaviour and abilities ofthe patient who don’t have chronic pain. The evaluationgrid is completed when the patient’s behaviour is modifiedfrom the basic behaviour or when there is suspicion of thepresence of chronic pain. This scale consists of 10 itemswhich are divided into three subgroups that are external-izing signs of pain (crying, facial expressions), motor signs(protection, enhancement, abnormal tone) and signs ofpsychic regression (ability to interact, analgesic attitudes).Each item is rating from 0 (usual manifestations) to 4(extreme change). From a total of 6 of 40, chronic pain ispresent.

The revised noncommunicating children’s pain checklistis used to measure pain in children with cognitive impair-ments (37). It was evaluated in 71 children with severecognitive impairments in Canada. This scale consists of 27items divided into six categories: vocal expression, socialbehaviour, facial expression, activity, movements of thebody and legs, and physiological signs. Each item is ratedfrom 0 (no pain) to 3 (severe pain), and the total scoreranges from 0 to 81. A score above 11 is an indicator ofchronic pain.

The revised Face Legs Activity Cry Consolability scale isthe third scale for behavioural assessment of pain inhandicapped children. It was validated in 52 children withcognitive impairments in the USA (38). This scale consistsof five categories rated from 0 to 2: facial expression, legmovement, physical activities, vocal expression and toconsole the child. The total score can range from 0 to 10.

Management of chronic orofacial pain associated with adisease in childrenWith an accurate diagnosis enabled by multiple ratingscales, COFPAD is a clinically well defined. Furthermore,various therapeutic means allow for managing chronicorofacial pain in children (39). However, because of itsmultidimensional aspect, chronic pain calls for transversalskills: treatment restricted to the cause is no longersufficient. Consequently, multimodal therapy, combiningphysiotherapy, psychology and pharmacology, is required(Fig. 3). These therapies can be implemented in someunits involved in managing chronic pain. For example,pain clinics allow for close collaboration of various



specialists such as physicians, nurses, psychologists andbiologists.

PharmacotherapyIn recent years, pharmacological treatments of COFPADare determined by collaboration of nursing and medicalstaff. The equimolar mixture of oxygen and nitrous oxide(EMONO) has anxiolytic, euphoric, amnestic and anal-gesic properties. The mixture induces conscious sedationby inhalation, which allows for performing some diag-nostic procedures (e.g. punctures) or other local treat-ments (e.g. osteoradionecrosis) (40). In addition,EMONO can be used as a painkiller (41). When thegenerative mechanism of the pain is predominantlynociceptive, pain of low or medium intensity may betreated with some nonopioid or weak opioid analgesics(42). Two different medications are usually combined toobtain a better analgesic effect. Powerful opioid analgesicsmay be used for severe, intense chronic pain. When thegenerative mechanism of the pain is essentially neuro-genic, management with co-analgesics such as antidepres-sants or anticonvulsants may be required. Opioids,antidepressants and anticonvulsants present hazards forchildren and adolescents, close monitoring should beincluded in the treatment plan.

However, with chronic pain and particularly cancerouspain, different types of generative mechanisms are com-bined, and multidisciplinary therapy is the key to manageCOFPAD. Even though nondrug therapies play a key role

and are inseparable from pharmacological therapies, theyare not used enough (43).

Physical, behavioural and cognitive therapiesFor chronic orofacial pain during temporomandibular jointdisorders in adults, efficacy was demonstrated for transcu-taneous electrical nerve stimulation. One study assessedvibratory analgesia in 17 patients with painful temporo-mandibular disorders (TMD). Results of 20- and 100-Hzvibration were compared with data from a no-vibrationcontrol condition. The results showed that vibratory anal-gesia occurred in TMD chronic pain conditions. VASratings and standardized drawings showing painful regionsboth showed that pain was reduced by 100-Hz, but not by20-Hz, vibration. About 70% of patients reported reducedpain during vibratory stimulation. In children, anotherstudy assessed the effectiveness of a transcutaneous electri-cal nerve stimulation unit. The effects of electronic dentalanaesthesia and local anaesthesia for deep cavity prepara-tions in primary molars were compared in 25 children aged7–9 years by using the Eland Colour Scale. The patients hadsymmetric teeth requiring Class I cavity preparation. One ofthe teeth was treated with electronic anaesthesia and theother with local anaesthesia. The tooth and method wereselected randomly. There was no statistically significantdifference between the groups in the perception of pain.During tooth preparation, 56% preferred transcutaneouselectrical nerve stimulation, 36% preferred local anaesthe-sia (44).

Figure 3 Multidisciplinary approach to management of chronic pain in children.



For the young child, sucking sweet solutions, especiallysucrose and glucose, with or without nutrients have shownanalgesic efficacy. In fact a Cochrane review (45), based onthree studies (220 infants), concluded that sucrose given indosages of 0.5–2 mL of 12–50% solution administeredapproximately 2 min prior to single heel lance was safeand efficacious in providing pain relief. Pain scores weresignificantly reduced in infants who were given sucrose(mean difference �1.64, 95% CI �2.47, �0.81 at 30 sec and�2.05, 95% CI �3.08, �1.02 at 60 sec after heel lance).Although the mechanism of action is not fully understood,the effect cannot be ignored. Skin-to-skin contact (46), andbreastfeeding during procedures (47,48) have shown anal-gesic efficacy too. For the older child, relaxation strategiescan reduce muscular tension and alleviate psychological ormental stress. Hypnotic techniques involve visualizationand mental imagery (49). The association of these tech-niques with EMONO provides a focus on another thing,which gives the impression of having a pleasant dream. Themost common techniques used in hypno-analgesia involvedirect suggestion of anaesthesia or analgesia, displacementof the pain or dissociation. The distraction operates onlyone hypnotic mechanism, ignoring a part of the body.

Current cognitive and behavioural therapies are mainlyintended as learning strategies to cope with situations. Forthe child and the family, trust and a clear explanation oftreatment and lesions are essential to break the viciouscycle of pain and anxiety. Increasing pain control isessential: to automatically stop thinking of the pain, formotivation and for calming. The impact of paediatricchronic pain on parents’ health-related quality of life andfamily functioning can be assessed by the family impactmodule (50). It was validated in 332 children and theirparents (288 mothers, 170 fathers). Children were sufferingfrom chronic pain of different aetiologies. The proposedtotal impact score was positively correlated with thepaediatric quality of life, and negatively correlated withpain catastrophizing, functional disability and child emo-tional and behavioural problems. Compared to fathers,mothers of youth with chronic pain reported worse scores.

Treatment and prevention of orofacial pain due to cancertreatmentHere, we address more specifically the effect of preventiveand curative orofacial chemo-radiotherapy in children. Oneof the most-often encountered cancers is acute lympho-blastic leukaemia. Oral care of children with from thisdisease aims at decreasing pain and discomfort, decreasingthe risk of infection and of bleeding, reducing soft-tissueinflammation and restoring masticatory functions. Using asoft toothbrush in combination with a mouthwash contain-ing chlorhexidine or bicarbonate solutions is stronglyrecommended (51). To deal with mucositis, brushing isperformed by swabbing with gauze soaked in bicarbonateor chlorhexidine solutions combined with an antifungalagent and an anaesthetic (52). Moreover, treating xerosto-mia is imperative: pilocarpine, salivary substitutes and oralgels or mouthwashes are used (53). After cancer treatment,

oral exercises should be implemented or continued toreduce the risk and severity of trismus. Irreversible osteo-radionecrosis may occur, but a curative treatment isunfortunately lacking, and orofacial hygiene can quicklycontribute to this major complication. Any dental thera-peutics should be initiated only with endorsement of theattending onco-hematologist.

CONCLUSIONCOFPAD is an unrecognized syndrome and poorly man-aged, especially in children. A multidisciplinary approach tomanage COFPAD requires appropriate management sched-uling by health care practitioners. During diagnosis, prac-titioners may have difficulties because of unrecognized or‘silent’ pain owing to difficult communication with children.Besides, clinicians may be misled by the psychomotorslowness related to chronic pain and the scarcity ofcomplaints.

Moreover, assessing pain can be difficult when the childrefuses to evaluate the pain. Assessment of chronic pain isburdensome because of the variety of tools based on age.Hetero-evaluation methods based on behavioural manifes-tations are used for young children or those with difficultyunderstanding directions. Self-assessment scales are usedfor children > 6 years old. COFPAD could be misinter-preted, thus leading to uncertain treatment.

Finally, strict treatment of the cause is not sufficientwithout a multidisciplinary approach combining pharma-cotherapy, psychology and physiotherapy. The managementof COFPAD should be improved with systematic andrigorous assessment by medical staff, but training nursingstaff to deal with pain in children and its specificities canimprove the assessment of COFPAD. To complete thisprocess, children and their close relatives must be educatedfor collaboration in the doctor–patient relationship. Thiscan improve the therapeutic environment when nonphar-macological techniques are uncommonly used with chil-dren requiring assistance from a third party. Finally, amultidisciplinary approach with pharmaceutical-free ther-apeutics must be used to manage chronic orofacial pain andmay favour conditions encouraging interdisciplinary prac-tice.

ACKNOWLEDGEMENTSWe thank Professor Sylvie Testelin (Department of Maxil-lofacial Surgery, Amiens Hospital, France) and ProfessorMicheline Robbe (Department of Reconstructive Plasticand Aesthetic Surgery, Amiens Hospital, France) for clin-ical pictures. We thank Laura Smales (BioMedEditing,Toronto, Canada) for editing the manuscript.

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SUPPORTING INFORMATIONAdditional Supporting Information may be found in theonline version of this article:

Figure S1 a/Intra-sinus cyst – b/Intra-sinus tumour.Figure S2 Von Recklinghausen’s disease.Figure S3 Intraosseous hemangiomas.Figure S4 Mandibular sarcoma.Figure S5 Osseous tumour.Data S1 Search equation.



assessment and management of chronic orofacial pain associated with a disease in children: a...

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