Rehabilitation of facial dermatologic lesion of dental origin in a pediatric patient

Mohammad Kamran Khan; Mahendra Kumar Jindal

doi:10.4103/jorr.jorr_73_21

CASE REPORT

Year : 2023 | Volume : 15 | Issue : 1 | Page : 48-53

Rehabilitation of facial dermatologic lesion of dental origin in a pediatric patient

Mohammad Kamran Khan, Mahendra Kumar Jindal
Department of Pediatric and Preventive Dentistry, Dr. Ziauddin Ahmad Dental College and Hospital, Faculty of Medicine, Aligarh Muslim University, Aligarh, Uttar Pradesh, India

Date of Submission	05-Nov-2021
Date of Decision	13-Sep-2022
Date of Acceptance	20-Sep-2022
Date of Web Publication	29-Dec-2022

Correspondence Address:
Mohammad Kamran Khan
Hamdard Nagar-A, Civil Line, Aligarh, Uttar Pradesh
India

Source of Support: None, Conflict of Interest: None

Check

DOI: 10.4103/jorr.jorr_73_21

Abstract

Odontogenic cutaneous sinus tract (OCST) of the facial region is rarely found and mostly misdiagnosed because of its variable cutaneous manifestations with the absence of obvious dental symptoms. Usually, patients consider such lesions of dermatologic origin instead of dental origin, and hence, seek its treatment from medical specialists which results in recurrence due to inappropriate treatments without considering the dental etiology, dental history, intra-oral examination, and dental radiographs. The current article presents a case report about chronic OCST at mental region (chin) of the face in a 10-year-old male patient. The source of infection was completely eliminated by endodontic therapy using calcium hydroxide Ca(OH)₂ and by conservative surgical debridement of periapical necrotic granulation tissue without excising the extra-oral sinus-tract orifice. The appropriate dental treatment resulted in the resolution of symptoms and rehabilitation of facial esthetics without any recurrence in follow-ups. Thus, the patient's quality of life was improved after successful dental treatment as evaluated by Child's Perception Questionnaire 8–10.

Keywords: Aesthetic treatment, Child-Perceptions-Questionnaire 8–10, diagnostic dilemma, diagnostic enigma, odontogenic cutaneous sinus tract, paediatric patient, quality of life

How to cite this article:
Khan MK, Jindal MK. Rehabilitation of facial dermatologic lesion of dental origin in a pediatric patient. J Oral Res Rev 2023;15:48-53

How to cite this URL:
Khan MK, Jindal MK. Rehabilitation of facial dermatologic lesion of dental origin in a pediatric patient. J Oral Res Rev [serial online] 2023 [cited 2023 May 31];15:48-53. Available from: https://www.jorr.org/text.asp?2023/15/1/48/365924

Introduction

It is well known that in human beings “Health is a state of complete physical, mental, and social well-being and not just the absence of disease” (WHO 1948). It has been observed that facial appearance influences mental health, social-interaction, and personality development and it also impacts the health-related quality of life (HRQOL) in children and adolescents.^[1] Skin problems in childhood also exert a strong impact on their self-esteem and confidence.^[2]

Odontogenic cutaneous sinus tract (OCST) is an uncommon lesion, manifested mostly as erythematous, crusting or smooth and nontender nodule, or ulcer-like lesion with purulent exudate discharge. These lesions may occur on the skin of body or angle of mandible, chin or cheek regions of the face which can be caused by the sequelae of pulpal necrosis or chronic apical periodontitis due to deeply/grossly carious tooth, traumatized tooth, infected retained root stumps or may be due to residual chronic infection of the jaws.^{[3],[4],[5],[6]}

Cutaneous sinus tracts of odontogenic origin are rarely found.^[7] Hence, its actual identification has been a challenge for dermatologists or general surgeon for establishing the correct diagnosis.^[6],[8] Literature demonstrates that majority of times these lesions are initially misdiagnosed and treated inappropriately because of its rare occurrence and absence of dental symptoms.^[6],[8] The lesions having cutaneous manifestation in the cervico-facial region similar to OCST constitute differential diagnosis such as furuncle, carbuncle, epidermal cysts, infected sebaceous gland, branchial cleft fistula, pyogenic granuloma, salivary gland fistula, deep mycotic infection, actinomycosis, thyroglossal tract fistula, basal cell and squamous cell carcinoma, osteomyelitis, and foreign-body reaction.^[5],[6],[8] These further pose difficulties and diagnostic dilemma in establishing its early true clinical diagnosis and effective treatment.^[5],[6],[8]

The delay in seeking appropriate intervention from dental surgeon for identifying and eliminating the odontogenic source of infection associated with OCST results in the persistence of periapical pathology of the infected tooth leading to continued discomfort, facial esthetic problems, and multiple unsuccessful medical treatment modalities and may also lead to complications such as sepsis, osteomyelitis, and space-infections in patients.^[5],[6],[8]

Paediatricians, dermatologists, and other medical specialists should need to consider dental aspects of OCST lesions when establishing the diagnosis. Articles related to OSCT would definitely help medical specialists in establishing the definitive diagnosis. There is very scarce literature regarding OCST in pediatric patient. So far, no case report has been published in the literature regarding the conservative surgical treatment of odontogenic cutaneous-sinus-tract in a pediatric patient under local anesthesia along with the assessment of the HRQOL before and after the dental treatment. In existing dental literature, the oral HRQOL (OHRQoL) has been assessed by using Child Perception Questionnaire (CPQ8–10) in few case report articles.^[9],[10] The CPQ8–10 was developed to assess the influence of orofacial problems (disorders/diseases) on the quality of life (QoL) of children aged 8–10 years.^[11] Thus, the CPQ8–10 helps in determining the treatment needs, in selecting the therapies, in monitoring the treatment progress, and also in evaluating the outcomes of provided treatment.^[11]

The current article presents a case report on the successful management of OCST of the face of a 10-year-old male with remarkable esthetic healing outcome without any recurrence in follow-ups visits. The dental treatment of the patient showed the improvement in QoL and overall health status as evaluated by using child perceptions questionnaire CPQ8–10.^[11] This case report has been prepared as per the CARE checklist case report guidelines.

Case Report

A 10-year-old male patient presented with a chief complaint of nodular mole-like lesion over the chin region of the face for 1 year. He also complained of foul odor fluid discharge occasionally from the same lesion. His medical and drug history was found nonsignificant. His detailed dental history revealed an event of traumatic dental injury (TDI) over lower anterior teeth during playing at home for about 1.5 years back, but no dental treatment for TDI was sought from the dentist as patient had no acute dental pain. Patient's parents gave a history of previous medical treatment of the presenting skin lesion from the nearby medical practitioners; but yet, the patient was not relieved and the lesion recurred later.

His family and personal history was not significant. Psycho-social history revealed that patient was not attending school regularly and also was not showing much interest in playing activities with his friends since the facial lesion appeared.

On general physical examination, the patient was found healthy systematically. Extra-oral examination revealed a pink-colored elevated crusty nontender dome-shaped nodular-like lesion with black-colored orifice at its center of approximately 1 cm diameter in midline of mental region (chin) of the face [Figure 1]a and [Figure 1]b. Palpation of the area around the lesion elicited purulent fluid discharge. Regional lymph nodes were not palpable. On intra-oral examination, Ellis class-II fractured right lower central incisor teeth 31, 41 were observed [Figure 1]c. Slightly discolored tooth crown of 31 was seen. No tenderness on percussion was present in the involved teeth. Oral hygiene status was poor with high debris, plaque, and calculus score.

Figure 1: Preoperative extra-oral photograph showing the nodulo-papular lesion over the chin region (a and b); and intra-oral view of the lesion demonstrating the fractured tooth 41 (c); and Preoperative radiograph showing the peri-apical radiolucency in relation to teeth 31 and 41 (d)

Click here to view

Radiographic evaluation (intra-oral periapical X-ray) revealed the irregular periapical radiolucency in relation to the root apices of mandibular central incisors teeth [Figure 1]d. Pulp-sensitivity evaluation (electric pulp test and thermal test) demonstrated negative response from mandibular central incisor teeth [Figure 2]a. Gutta-percha cone was used to trace the origin and path of the sinus tract. The radiograph with GP point confirmed the odontogenic nature of cutaneous sinus tract in relation to the periapical lesion of mandibular central incisor teeth [Figure 2]b and [Figure 2]c.

Figure 2: The pulp-sensitivity test (a); Radiographic tracing of dental origin with gutta-percha cone (b and c)

Click here to view

Based on the detailed history, oral examination, and radiographic evaluation, the clinical diagnosis of chronic apical periodontitis in relation to nonvital mandibular central incisor teeth with extra-oral sinus tract on the mental region of the face was established.

The treatment plan consisted of endodontic therapy with intra-canal calcium hydroxide medicament along with conservative surgical debridement of periapical-infected necrotic granulation tissue in relation to traumatized teeth 31, 41 to eliminate the source of infection. Patient's parents were informed and explained about the diagnosis, prognosis, and treatment plan for the lesion. Written informed consent was taken from parents.

Patient showed positive (+) behavior as per Frankel behavior rating scale. Tell-show-do method was employed as the behavior management technique during dental treatment for the pediatric patient.

First, oral prophylaxis with scaling was done. Afterward, access-cavity opening and necrotic pulp extirpation of 31, 41 were done under rubber-dam isolation with copious irrigation of root canals with 2% cholrhexidine [Figure 3]a. Working length was determined followed by cleaning and shaping of canals was done. Calcium hydroxide intra-canal medicament was placed and access cavity was sealed for 2 weeks [Figure 3]b and [Figure 3]c. The collected purulent exudate was sent for microbial culture and antibiotic sensitivity testing. Complete blood count with hemoglobin, clotting time, and bleeding time tests were advised and were found within the normal limits.

Figure 3: The endodontic treatment procedure performed under rubber-dam isolation with intra-canal dressing of Ca(OH)₂ in teeth 31 and 41 (a-c)

Click here to view

Conservative surgical debridement of infected necrotic granulation tissue in the periapical region of teeth 31, 41 was done under local anesthesia and envelope flap was closed with resorbable sutures [Figure 4]a and [Figure 4]b. Simultaneously, obturation of involved teeth with gutta-percha cones was done. Considering the cosmetic perspective, excision of extra-oral sinus-tract was not done to prevent the scarring of the surgical wound. The patient was prescribed antibiotics as per the result of microbial culture and antibiotic sensitivity testing of purulent discharge and clinical presentation of the patient. The patient was sent to home with necessary postoperative instructions. Oral hygiene maintenance and long-term follow-up visits were advised.

Figure 4: Intra-op photographs demonstrating the conservative surgical debridement of periapical infected granulation tissue and flap closure with resorbable suture material (a and b)

Click here to view

On follow-up visits, patient was found asymptomatic and was satisfied with the treatment outcome. Good and rapid healing process with progressive reduction in extra-oral scar and periapical radiolucency was revealed on clinical and radiographic evaluation in the follow-up visits after 1 week [Figure 5]a and [Figure 5]b, 1 month [Figure 5]c and [Figure 5]d, and 3 months [Figure 5]e and [Figure 5]f.

Figure 5: Clinical follow-ups photographs and radiographs showing the progressive healing of the peri-apical and the exra-oral lesion and also demonstrating the excellent cosmetic results of the treatment after 1 week (a and b); 1 month (c and d); 3 months (e and f)

Click here to view

The patient showed significant improvement in his QoL as assessed by CPQ8–10. Patient's parents revealed that his son has started attending school regularly and was also performing normal routine activities happily and confidently with his friends.

Discussion

OCST is the extra-oral manifestation of dental origin infection which may be caused by pulpal necrosis, pericoronitis (periodontal disease), TDI or may be due to the side effects of other previous dental treatment procedures.^[12],[13] Although majority of OCST are manifested intra-orally, the cutaneous manifestations of OCSTs with diagnostic dilemma have also been reported in literature.^[14],[15] The intra-oral or extra-oral manifestation of odontogenic sinus-tract opening depends upon the several factors such as location of root apex of tooth to muscle attachment, fascial sheath, tissue resistance, bacterial virulence, and host immunity.^[16],[17]

Literature demonstrates that mostly mandibular teeth are associated with OCSTs on the mandibular body and mental region of the face.^[14] However, the occurrence of OCST on nose, nasolabial fold, and inner canthus of the eye has also been reported.^[14],[15] These lesions have variable presentations such as nodule, papule, ulcer-like, scar with draining sinus orifice, etc.^{[12],[14],[16]} It has been observed in literature, that mostly patients are unaware or unable to relate the dental symptoms and history with such cutaneous lesions.^[5],[8]

Since, OCSTs are the sequelae of infected necrotic granulation tissue of periapical region of nonvital tooth. Hence, it is prudent to eliminate the odontogenic source of infection for complete healing of extra-oral lesion.^[8] In the present case also, the two nonvital teeth had chronic peri-apical pathology associated with OCST. Hence, endodontic treatment with periapical debridement of infected tissue was carried out to remove the source of infection.

Considering the various factors (viz. chronicity of the periapical lesion, extra-oral sinus, status of the roots of involved incisors, favorable accessibility to anterior mandibular region, and co-operative behavior of the paediatric patient), the endodontic treatment employing the calcium-hydroxide dressing followed by surgical debridement of infected necrotic granulation tissue from the periapical region of the affected incisors was done to eliminate the source of infection and to facilitate healing.

Extra-oral excision of sinus-tract was not performed to have minimal scar on the chin. The treatment resulted in very satisfactorily periapical and extra-oral healing with excellent cosmetic outcome within 3 months' follow-up.

The advantages of using the calcium hydroxide as intra-canal medicament in endodontics are well known.^{[18],[19],[20]} Ca(OH)₂ causes effective disinfection of the infected root canals of the nonvital teeth due to its efficient antimicrobial actions (bactericidal properties).^{[18],[19],[20]} It has been reported in existing literature that the cleaning, shaping, and irrigation steps of the endodontic therapy can be combined with the use of intra-canal calcium hydroxide dressing to achieve the effective disinfection of the infected root-canals.^{[18],[19],[20]} Prior to endodontic surgery, the placement of intra-canal calcium hydroxide medicament into the nonvital teeth with periapical lesion has been described in the literature to have efficient disinfection of inaccessible areas of the canal and consequently favorable healing outcome of the periapical surgery and the prevention of recurrence of signs and symptoms.^{[18],[19],[20]} Hence, in the presenting clinical case, Ca(OH)₂ was used as intra-canal inter-appointment dressing before commencing the periapical surgery.

Majority of infections in the periapical and endodontic infections are poly-microbial in nature, and the microbial culture mostly demonstrates the growth of anaerobes or facultative anaerobes such as streptococcal species from the infected tissue.^[8],[16] Chronic infections such as tuberculosis (scrofula) and actinomycosis are also included in the differential diagnosis because such lesion also clinically manifest as cutaneous draining sinus.^[21] Microbiological culture of the draining purulent exudate also helps in the identification of the microbial flora and thus, can rule-out the suspicion of other microbial infection like fungal or syphilitic infections.^[8] In the present case, microbial culture and antibiotic sensitivity test of the collected purulent exudate was done to rule out nonodontogenic infections and then to prescribe the correct antibiotic to the patient.

Radiographic evaluation is very critical for establishing the true and definitive diagnosis of OCST in patients having no apparent dental symptoms. Intra-oral radiographs help in identifying the periapical pathology in asymptomatic tooth. Radiograph with inserted gutta-percha cone aids in confirming the involved tooth with the OCST lesion.^[6],[22] In some clinical situations, computed tomography scan and cone-beam computed tomography scan can be used for the radiographic assessment of the source of infection.^[15],[22]

In literature, surgical excision of cutaneous sinus-tract along with the extraction of involved tooth was suggested because earlier lining of sinus-tract was thought to be made of epithelium.^[21] However, Grossman et al. (1988) reported that such sinus-tracts are lined by granulation tissue instead of epithelium.^[22] Therefore, surgical excision of extra-oral sinus-tract is not recommended.^[22] In the present case also, cutaneous sinus-tract was not excised to avoid scarring from extra-oral surgical excision.

Endodontic therapy is the treatment of choice for restorable tooth while extraction is done for nonrestorable tooth. When source of infection is appropriated treated then cutaneous sinus-tract orifice heals spontaneously.^[7] Literature demonstrates that mostly patients with OSCTs receive multiple inappropriate treatment modalities such as dermatologic surgeries for excision of sinus-tract, unnecessary usage of antibiotics, and steroid because misdiagnosis made by medical specialists.^[3],[17] The lesion gets recur if the dental source of infection is not addressed appropriately.^[3],[17]

Conclusion

The case report signifies that early and correct diagnosis and appropriate dental treatment of OCST by a dental surgeon are crucial for the complete resolution of such lesion without any recurrence. Medical professionals must always take dental opinion and consultation for such suspicious cutaneous lesion of dental origin for detailed dental evaluation. In this case report, OCST in pediatric patient was managed successfully with conservative surgical endodontic treatment to remove completely the source of infection. Thus, the resolution of patient's symptoms, healing of chronic periapical lesion, and rehabilitation of facial esthetics resulted in improved overall health and QoL of the patient. Furthermore, patient regained self-confidence, self-esteem, and social interactions.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Masnari O, Schiestl C, Rössler J, Gütlein SK, Neuhaus K, Weibel L, et al. Stigmatization predicts psychological adjustment and quality of life in children and adolescents with a facial difference. J Pediatr Psychol 2013;38:162-72.
2.	Vivar KL, Kruse L. The impact of pediatric skin disease on self-esteem. Int J Womens Dermatol 2018;4:27-31.
3.	Yadav S, Malik S, Mittal HC, Puri P. Odontogenic cutaneous draining sinus. J Craniofac Surg 2014;25:e86-8.
4.	Ozdemir A, Guven G, Dilsiz A, Sencimen M. Diagnosis and treatment of mandibular extraoral sinus of periodontal origin in a 9-year-old boy: A case report. J Indian Soc Pedod Prev Dent 2008;26:76-8. [PUBMED] [Full text]
5.	Chhabra A, Chhabra N. Dental infection mimicking dermatological lesion: Three case reports of cutaneous fistulae and sinus tracts on face. Indian Dermatol Online J 2018;9:441-4. [PUBMED] [Full text]
6.	Figaro N, Juman S. Odontogenic cutaneous fistula: A cause of persistent cervical discharge. Case Rep Med 2018;2018:1-3.
7.	Tidwell E, Jenkins JD, Ellis CD, Hutson B, Cederberg RA. Cutaneous odontogenic sinus tract to the chin: A case report. Int Endod J 1997;30:352-5.
8.	Chen K, Liang Y, Xiong H. Diagnosis and treatment of odontogenic cutaneous sinus tracts in an 11-year-old boy: A case report. Medicine (Baltimore) 2016;95:e3662.
9.	de Oliveira LR, Bezerra TM, Soares TR, Risso PD, Maia LC. Complicated crown fracture treatment and its impact on quality of life related to oral health: Report of two cases. Rev Cient do CRO-RJ (Rio de Janeiro Dent J) 2019;4:120-24.
10.	Piassi E, Antunes LS, Andrade MR, Antunes LA. Quality of Life Following Early Orthodontic Therapy for Anterior Crossbite: Report of Cases in Twin Boys. Case Rep Dent. 2016;2016:3685693.
11.	Jokovic A, Locker D, Tompson B, Guyatt G. Questionnaire for measuring oral health-related quality of life in eight- to ten-year-old children. Pediatr Dent 2004;26:512-8.
12.	Uppal A, Thaku S, Chauhan D, Singhal P, Doneria D. Extraoral cutaneous sinus tracts of dental origin: A report of two pedodontic cases. Int Dent Med J Adv Res 2017;3:1-4.
13.	Pasternak-Júnior B, Teixeira CS, Silva-Sousa YT, Sousa-Neto MD. Diagnosis and treatment of odontogenic cutaneous sinus tracts of endodontic origin: Three case studies. Int Endod J 2009;42:271-6.
14.	Kumaran MS, Narang T, Dogra S, Bhandari S. Odontogenic cutaneous sinus tracts: A clinician's dilemma. Indian Dermatol Online J 2020;11:440-3. [Full text]
15.	Vermani M, Kalia V, Singh S, Garg S, Aggarwal S, Khurana R, et al. Orocutaneous fistula or traumatic infectious skin lesion: A diagnostic dilemma. Case Rep Dent 2015;2015:353069.
16.	Mishra R, Khan TS. Cutaneous sinus tract in association with traumatic injury to the teeth. Int J Clin Pediatr Dent 2013;6:205-7.
17.	Johnson BR, Remeikis NA, Van Cura JE. Diagnosis and treatment of cutaneous facial sinus tracts of dental origin. J Am Dent Assoc 1999;130:832-6.
18.	Mohammadi Z, Dummer PM. Properties and applications of calcium hydroxide in endodontics and dental traumatology. Int Endod J 2011;44:697-730.
19.	Bhalla VK, Chockattu SJ. Intracanal delivery of calcium hydroxide: A literature review. Saudi Endod J 2021;11:1-6. [Full text]
20.	Noor AE, Amin N, Mustari Shanta T. Combined endodontic surgery associated with intra canal medicament (calcium hydroxide) is necessary for chronic periapical lesions – A case report. Int J Appl Dent Sci 2015;1:133-5.
21.	Chowdri NA, Sheikh S, Gagloo MA, Parray FQ, Sheikh MA, Khan FA. Clinicopathological profile and surgical results of nonhealing sinuses and fistulous tracts of the head and neck region. J Oral Maxillofac Surg 2009;67:2332-6.
22.	Grossman LI, Oliet S, Del Rio CE. Endodontic practice. 11^th edition. Lea & Febiger, Philadelphia, 1988: p. 179-227.