JICDRO is a UGC approved journal (Journal no. 63927)

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CASE REPORT
Year : 2020  |  Volume : 12  |  Issue : 2  |  Page : 166-172

Orocutaneous Fistulae Mimicking as Dermatological Lesion: A Diagnostic Dilemma


Department of Oral and Maxillofacial Surgery, SGT University, Gurugram, Haryana, India

Date of Submission28-Dec-2019
Date of Decision16-May-2020
Date of Acceptance01-Jun-2020
Date of Web Publication14-Dec-2020

Correspondence Address:
Dr. Rajeev Pandey
Senior Lecturer, Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, SGT University, Gurugram, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jicdro.jicdro_63_19

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   Abstract 


Orocutaneous lesions of odontogenic origin are rare, and sometimes, they can mimic as dermatological lesions. They are usually misdiagnosed, and as a result, the treatment is delayed leading to the chronicity of the lesion and enlargement of the fistula and bone loss. These cases require multispecialty consultation for proper diagnosis and treatment. Here, we report a case series of orocutaneous fistulae, due to misdiagnosis, the treatment rendered was improper, leading to enlargement of the lesion along with bony defect and loss of tooth.

Keywords: Chronic, dermatological, fistula, Oro cutaneous


How to cite this article:
Pandey R, Kumar J, Bootwala F, Nag M. Orocutaneous Fistulae Mimicking as Dermatological Lesion: A Diagnostic Dilemma. J Int Clin Dent Res Organ 2020;12:166-72

How to cite this URL:
Pandey R, Kumar J, Bootwala F, Nag M. Orocutaneous Fistulae Mimicking as Dermatological Lesion: A Diagnostic Dilemma. J Int Clin Dent Res Organ [serial online] 2020 [cited 2021 Jan 25];12:166-72. Available from: https://www.jicdro.org/text.asp?2020/12/2/166/303408




   Introduction Top


Orocutaneous fistula (OCF) is an unusual but well-designated condition in the literature. It is an abnormal communication between the oral cavity and the facial skin.[1],[2],[3] These lesions clinically appear as carbuncle, epidermal cysts, furuncle neoplasms of the facial skin, or they may mimic lesions of chronic infections such as tuberculosis, cervicofacial actinomycosis, osteomyelitis of the jaws or sweat gland lesions.[2],[3] OCF is often misdiagnosed and mistreated as it mimics lesions of the dermatological origin.[4] Although it is well described in the literature, both physicians and dentists often misdiagnose it. Many times, the physicians do not consider the chronic odontogenic infection such as a periapical abscess or granuloma due to a decayed tooth as the focus of infection and thus leading to the formation of draining sinus tract (during the initial stage of fistula) that ultimately invades the bone and the overlying soft tissue forming a lesion over the facial skin.[3] The treatment plan usually includes antibiotic therapy, multiple biopsies, incision and drainage, steroid therapy, and cosmetic therapy, and hence, the etiology of the lesion is not addressed.[4] This leads to the chronicity of the lesion, changing a communication into an epithelialized tract or fistula. Here, we report a case series of OCF appearing similar to facial dermatological lesions.


   Case Reports Top


Case Report 1

A 57-year-old male patient PS referred to the Department of Oral and Maxillofacial Surgery with a complaint of a nonhealing fistula over the left side of his lower jaw along with occasional drainage from the fistula [Figure 1]. According to the history provided by the patient, there was a pea-sized swelling over the left side of his lower jaw initially, gradually the swelling started increasing and formed a punctum followed by drainage of pus and then a gradual decrease in size of the swelling. The swelling never disappeared, and the cycle continued for a few weeks. The patient went to a physician who diagnosed it as a skin pustule and prescribed systemic antibiotics and topical ointments to the patient. Due to the treatment, the patient was free of symptoms, but the wound did not heal completely and still had occasional pus discharge. For about 2 years, the patient kept on shifting from one specialist to another along with the consumption of multiple oral drugs, and the patient finally gave up thinking it was his fate to live with an unhealed wound on his face. The patient, after about a year, was referred to our department for pain in one of the lower left back tooth. During the examination, the cutaneous lesion was discovered, and the history was elicited from the patient. Extraoral examination revealed a cutaneous lesion on the left side of lower jaw measuring about 2.5 cm × 2 cm, causing puckering of the skin. Around the lesion, multiple small pustules were seen. The lesion was tender on palpation and firmly adherent to the underlying bone. There was no discharge observed on palpation. Intraoral examination revealed multiple missing teeth in both arches with fair oral hygiene. The lower left second premolar was a root stump and tender on percussion and the lower left second molar was grossly decayed but mildly tender on percussion. The Orthopantogram was advised, and it revealed grossly decayed 37 with a carious lesion involving pulp along with radiolucency at the apex [Figure 2]. Based on clinical and radiological findings, a diagnosis of orocutaneous lesion of odontogenic origin was made. The patient was told about the condition and planned for extraction of 35 and 37 along with the surgical removal of the fistula under local anesthesia [Figure 3]. Surgical findings: Total involution of skin inside the oral cavity with ingrown hairs, and fixity to the underlying bone. The wound was closed in two layers using 4-0 vicryl sutures for the muscle layer and 3-0 silk sutures for the overlying skin.
Figure 1: Case 1: Orocutaneous fistula clinical picture

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Figure 2: Case1: Orthopantogram findings: showing grossly decayed 37 with periapical radiolucency

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Figure 3: Case 1: Surgical excision of fistula. A bony defect can be appreciated by the periapical lesion

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Follow up

Patient was recalled after 1 week for suture removal [Figure 4], and good healing was observed, 6-month follow-up showed no recurrence.
Figure 4: Case 1: One week follow-up of the patient showing good esthetic and resolution of the orocutaneous fistula

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Case Report 2

A 24-year-old female patient NP referred to the Department of Oral and Maxillofacial Surgery with a chief complaint of swelling over the left side of her lower jaw for 3 years [Figure 5]. The patient reported that around 3 years ago, she had developed pain in the lower back left tooth region and subsequently started developing a swelling over the left side of her face, the swelling used to occasionally increase in size, form a punctum and would start draining pus. The patient consulted many physicians and was given numerous courses of antibiotics, which offered her some relief, but she used to develop the swelling and pain again. The patient visited a dentist around a year back and was diagnosed with a decayed lower, left-back tooth. The tooth was thus removed, and the patient was symptom-free for a few days, but she soon developed the same symptoms again. The patient was psychologically burdened and hence stopped seeking treatment and accepted the swelling as a permanent part of her face. After 1 year, the patient came for a routine dental check up and was referred to our department.
Figure 5: Case 2: Orocutaneous fistula clinical picture

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Extraoral examination revealed a localized, erythematous swelling measuring 4 cm × 3 cm over the lower left side of the face. The center of the swelling was blanched with the presence of puckering of the skin. The lesion was mildly tender on palpation and firmly adherent to the underlying bone. There was no discharge observed on palpation. Intraoral examination revealed missing 36 and pericorinitis in relation to tooth 38. The Orthopantogram was advised, and it revealed a periapical abscess in relation to 38 [Figure 6]. Based on clinical and radiological findings, a diagnosis of OCF secondary to buccal migratory abscess was made. The patient was told about the condition and planned for extraction of 38 along with the surgical removal of the fistula under local anesthesia [Figure 7]. The wound was given an extraoral dressing, and a pack was placed over the extraction socket of 38. The patient was recalled after 1 week, and good healing was observed, 1-month follow-up showed no recurrence [Figure 8].
Figure 6: Case 2: Orthopantomogram findings: periapical abscess in relation to 38

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Figure 7: Case 2: Surgical removal of the fistula tract using gauze

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Figure 8: Case 2: 1 month follow up of the patient showing resolution of the lesion. Esthetic reconstruction was planned later on

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Case Report 3

A 26-year-old male patient IS referred to the Department of Oral and Maxillofacial Surgery with a chief complaint of a nonhealing wound over the right side of his lower jaw along with occasional drainage from the wound. The patient was previously treated in our department for fracture of the symphysis region of the mandible 6 months ago. The patient, after 1 month started complaining of pain in the jaw region and development of a pea-sized swelling, which has turned into an unhealing wound over the lower third of his face. The patient consulted a nearby hospital and was advised antibiotics and painkillers, which provided no relief to the patient. The patient reported back to us for further treatment. Extraoral examination revealed a cutaneous lesion on the right side of the lower border of the mandible over the symphysis region, measuring about 2 cm × 2 cm, causing puckering of the skin [Figure 9]. The lesion was tender on palpation and firmly adherent to the underlying bone. There was no discharge observed on palpation. Intraoral examination revealed fair oral hygiene. The orthopantogram was advised, and it revealed a two-hole with gap miniplate over the superior border and four holes with gap miniplate over the inferior border of the mandible in the symphysis region [Figure 10]. Based on clinical and radiological findings, a diagnosis of OCF secondary to hardware failure was made. The patient was told about the condition and planned for surgical removal of the mini plates and the lining of fistula under local anesthesia. A vestibular incision was given extending from 44 to 34, and a full-thickness mucoperiosteal flap was raised. The two miniplates were removed, and the fistula tract was identified and removed [Figure 11]. The intraoral incision was closed with 3-0 silk sutures, and a dressing was placed over the extraoral wound. The patient was recalled after 1 week, and good healing was observed, 1-month follow-up showed no recurrence [Figure 12].
Figure 9: Case 3: Oro cutaneous lesion clinical picture

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Figure 10: Case 3: Orthopantogram findings: Showing mini plates at the anterior mandible region

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Figure 11: Case 3: Surgical picture just before removal of mini plates the reason for Oro cutaneous fistula

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Figure 12: Case 3: One month follow up showed no recurrence of the Oro cutaneous fistula

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Case Report 4

A 44-year-old female patient SR referred to the Department of Oral and Maxillofacial Surgery with a chief complaint of a pea-sized swelling over the right side of her lower jaw for 1 year. The patient reported that around 1 year ago, she had developed pain in the lower front tooth region and started developing a swelling over the chin. The swelling started increasing in size and started draining pus. The patient consulted many physicians and was given numerous courses of antibiotics, which offered her relief. The patient reported to us for removal of a decayed front tooth, as she wanted a denture. While evaluating the patient, we noticed the extraoral swelling, took a detailed history and performed a detailed clinical examination. On examination: Extraoral examination revealed a cutaneous lesion on the right parasymphysis region of the lower jaw measuring about 1.5 cm × 2 cm, causing puckering of the skin [Figure 13]. The lesion was tender upon palpation and firmly adherent to the underlying bone. There was no discharge observed upon palpation. Intraoral examination revealed multiple missing teeth in both arches with fair oral hygiene. The lower right lateral incisor and canine were grossly decayed without any tenderness. The Orthopantogram was advised, and it revealed grossly decayed 42 and 43 with a carious lesion involving pulp along with radiolucency at the apex [Figure 14]. Based on clinical and radiological findings, a diagnosis of orocutaneous lesion of odontogenic origin was made. Patient was told about the condition and planned for extraction of 42 and 43 along with the surgical removal of the fistula under local anesthesia [Figure 15]. Surgical findings: Total involution of skin inside the oral cavity with in-grown hairs, and fixity to the underlying bone. The wound was closed in two layers using 4-0 vicryl sutures for the muscle layer and 3-0 silk sutures for the overlying skin. Follow-up: patient was recalled after 1 week for suture removal, and good healing was observed [Figure 16], 6 months follow-up showed no recurrence.
Figure 13: Case 4: Clinical picture of orocutaneous fistula

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Figure 14: Case 4: Orthopantogram revealed grossly decayed 42 and 43 with carious lesion involving pulp along with radiolucency at the apex

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Figure 15: Case 4: Fistula tract was surgically removed along with the extraction of 42 and 43 tooth

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Figure 16: Case 4: One week follow-up showed good healing and resolution of the orocutaneous fistula

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   Discussion Top


An extraoral opening, sinus, or fistula of facial region is a very common finding. Affected patients usually seek help from dermatologists or surgeons rather than from dentists. In children and adolescents, Oro Cutaneous Fistulae are more common as their alveolar processes are not fully developed, with spaces between the teeth and bone that facilitate infections. Therefore, the correct diagnosis is essential for the complete resolution of the lesion. The finding of these case reports shows that improper treatment of such lesions will cause incomplete healing and chronicity of the lesion. Therefore, any such lesion in the cervicofacial region should include odontogenic infection as a cause and should be ruled out.

The infection arises in the tooth-bearing region of the jaw and spreads in the bone and periodontal ligament. If no treatment is done during this period, the infection spread further and involves the cortical plate of the bone and later perforate the cortical plates and spread in the fascial space to localize as an abscess or may develop into cellulitis. From there, the pus or the infected foci may drain through extraoral skin and form a sinus which later epithelize as fistula or mucosa inside the mouth depending on the infection tracks and muscle attachment. The morphological appearance of OCF varies according to the duration of the fistula. This may be attributed to the constant struggle of the body to stop the spread of the infection and initiation of the healing process. It may appear initially as an abscess or sinus opening with pus discharge which, if not treated (etiology is not removed) can lead to epithelisation of the sinus tract and become an ulcerated opening or nodulocystic lesion. In many cases, it may heal, leading to dimpling of the surrounding skin and a very small opening, which can be diagnosed only on careful examination. Many times it may form a firm nodule.

Differential diagnosis of orocutaneous fistula includes dental infection, actinomycosis, osteomyelitis, foreign-body, local skin infection, salivary gland and duct fistula, neoplasm, pyogenic granuloma, lymph node inflammation, sweat gland inflammation, penetrating wounds, congenital fistulae, infected cysts, tuberculosis.[5],[6],[7]

The article has described four case reports, and the findings show that improper treatment of such lesions will cause incomplete healing and chronicity of the lesion. The four case reports described in the article emphasizes the varying etiology of such lesions and the importance of accurate history and clinical examination, both extraoral and intraoral. Patients PS and SR had presented with a chief complaint of long-standing lesions in the lower third of their respective faces. The lesions were asymptomatic as the patients had visited multiple specialists and had undergone multiple analgesic-anti inflammatory-antibiotic regimens. The patients had become symptomless, but the persistent presence of the lesion was still bothersome. The patients had consulted other specialists, but it was futile as the etiology was still not addressed which was pulpal degeneration due to untreated dental caries followed by the development of a periapical lesion which then progresses to the alveolar bone, cortical bone, periosteum and then to the surrounding soft tissue over a period of time. Both the patients had considered this to be a permanent fixture on their respective faces and hence had given up all hopes of getting the lesion to be treated. Patient NP was socially and psychologically burdened for 3 years because of the swelling present over her face, which although was not painful but occasionally discharged pus and had episodes of acute exacerbation. The patient had reported for a routine dental checkup but was discovered with the lesion, which on detailed clinical history and evaluation revealed to be of odontogenic in origin. Patient NP, unlike the PS and SR, had visited a dentist multiple times but was misdiagnosed as a periapical lesion secondary to lower left first molar, which was eventually extracted. The patient had remained symptom-free for some time but became symptomatic again after some time. The main etiology attributed to it was pericoronitis due to impacted third molar, which had developed into a buccal migratory abscess and following the path of least resistance, developed into a fistula. The chronicity of the lesion, despised multiple treatment attempts, and the episodes of its acute exacerbation led to the patient continuing her treatment. Patient IS was treated for symphysis fracture with open reduction internal fixation under local anesthesia. The patient was a migrant laborer and soon relocated to his village after the surgery. The patient, upon the development of the swelling in the chin region, visited a local hospital and was given medications for the same. The patient was not satisfied with the treatment and reported back to our department soon. The patient was previously treated in the department, and the patient was diagnosed with hardware failure and an OCF. Unlike the other three patients where the etiology was odontogenic in origin, this fistula was due to hardware failure secondary to trauma.

Diagnosis of orocutaneous lesions can be achieved using clinical and radiographical findings. Clinical diagnosis is based on the history of the lesion, which will directly or indirectly relate to toothache or swelling of the region weeks or months before the lesion developed.[3],[5],[6] History will also include multiple treatments with incomplete resolution. On intraoral examination, the presence of a carious tooth, deep pockets, tooth with apical periodontitis or abscess, are all possible etiological factors to the lesion. Radiological examination plays an important role in the diagnosis of OCF.

Modalities include

Computed tomography (CT), fistulography, cone-beam CT, orthopantomogram and gutta percha points may be inserted into the fistula to trace direction and depth of fistula and Intra Oral Periapical (IOPA) radiograph can be taken which will trace out the main etiological factor and the tooth involved.[7],[8]

Treatment option includes both conservative as well as radical surgeries. Conservative modalities include: root canal treatment of the offending tooth with or without periapical surgery depending upon the periapical radiolucency and if the fistula can be allowed to heal by itself. If the offending tooth is not salvable, then extraction of the tooth is recommended, and self-healing of the tract is allowed. Antibiotic coverage is necessary to reduce the number of microbes causing the infection. Amoxicillin with clavulanic acid is most commonly prescribed as it is effective for both gram-positive and gram-negative organisms. Radical surgical treatment includes surgical excision of the tract till the bone along with the extraction of the tooth. This is recommended in cases similar to the above case to correct the cosmetic defect and early resolution of the lesion. Both modalities are based on cutting down the pathway between the skin and the infected area and eliminating the primary source of infection.[6] Prompt treatment results in excellent prognosis, particularly in case of dental infection. If the infection is left untreated, cellulitis can lead to Ludwig angina and cavernous sinus thrombosis.


   Conclusion Top


Physicians easily misdiagnose OCF of odontogenic origin. Successful treatment strategy includes careful history taking, meticulous examination, and radiographic examination. Treatment should be based on the elimination of the cause and restoration of skin and oral cavity integrity.

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 Top

1.
Ranga U, Aiyappan SK, Veeraiyan S. A case of intermittently discharging skin lesion: Orodentocutaneous fistula demonstrated on CT fistulography. J Clin Diagn Res 2014;8:ZD09-11.  Back to cited text no. 1
    
2.
Samir N, Al-Mahrezi A, Al-Sudairy S. Odontogenic cutaneous fistula: Report of two cases. Sultan Qaboos Univ Med J 2011;11:115-8.  Back to cited text no. 2
    
3.
Pandey R, Gupta R, Bhagat N, Islam JI. Oro cutaneous fistula of a periodontal origin. Indian J Dent Adv 2017;9:69-72.  Back to cited text no. 3
    
4.
Guevara-Gutiérrez E, Riera-Leal L, Gómez-Martínez M, Amezcua-Rosas G, Chávez-Vaca CL, Tlacuilo-Parra A. Odontogenic cutaneous fistulas: Clinical and epidemiologic characteristics of 75 cases. Int J Dermatol 2015;54:50-5.  Back to cited text no. 4
    
5.
Figaro N, Juman S. Odontogenic cutaneous fistula: A cause of persistent cervical discharge. Case Rep Med 2018;2018:3710857.  Back to cited text no. 5
    
6.
Tian J, Liang G, Qi W, Jiang H. Odontogenic cutaneous sinus tract associated with a mandibular second molar having a rare distolingual root: A case report. Head Face Med 2015;11:13.  Back to cited text no. 6
    
7.
Baba A, Okuyama Y, Shibui T, Ojiri H. Odontogenic cutaneous fistula mimicking malignancy. Clin Case Rep 2017;5:723-4.  Back to cited text no. 7
    
8.
Lee EY, Kang JY, Kim KW, Choi KH, Yoon TY, Lee JY. Clinical characteristics of odontogenic cutaneous fistulas. Ann Dermatol 2016;28:417-1.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16]



 

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