Oral Cutaneous Fistula

Article Author:
Chourouk Chouk
Article Editor:
Noureddine Litaiem
Updated:
6/4/2019 4:04:20 PM
PubMed Link:
Oral Cutaneous Fistula

Introduction

Oral cutaneous fistulas (OCF) are infrequent conditions in which there is a pathologic communication between the oral cavity and the skin. In medical and dental literature, the terms fistulas and sinus tracts are often used to describe the same condition. Chronic dental infections, trauma, dental implant complications, salivary gland lesions, and neoplasms are the most common causes of oral cutaneous fistulas. Affected patients usually seek help from dermatologists or surgeons rather than from dentists. As the clinical cutaneous and oral manifestations are generally scarce and often nonspecific, the diagnosis of OCF requires a high degree of suspicion. Prognosis is excellent when treatment initiates promptly, particularly if there was a dental infection. Otherwise, OCF associated with malignancies can lead to complications and may be life-threatening.

Etiology

Odontogenic cutaneous fistulas are responsible for most cases of oral cutaneous fistulas reported in the literature. They arise as sequelae to bacterial invasion of the dental pulp resulting in apical periodontitis caused by a carious lesion, trauma or other causes. This condition occurs when the pulp becomes necrotic, and the infection spreads into the periradicular area. Subsequently, infection leads to bone resorption and dissects along the path of least resistance from the root apex to finally erupts through the skin.[1]

Cutaneous fistulas and sinuses in the maxillofacial region secondary to osteomyelitis rarely appear in clinical practice.[2] It is more likely to develop in patients with uncontrolled diabetes, in those with osteoradionecrosis who have undergone jaw irradiation and in those with metabolic bone diseases such as osteitis deformans “Paget disease” or osteopetrosis.

An additional cause of OCF is medication-related osteonecrosis of the jaw.[3] Biphosphonates and other anti-resorptive medications, as well as IV antiangiogenic therapies, have been reported to induce osteonecrosis of the jaw.[3] These medications have roles in the treatment of diseases such as lytic bone metastases, malignant hypercalcemia, multiple myeloma, and osteoporosis.

Traumatic fistulas may be the result of injury or surgical repair. In a study by Dawson et al., which explored factors associated with the formation of orocutaneous fistula following head and neck reconstructive surgery, they found that patients who had previous chemoradiotherapy had a significantly higher probability of developing a fistula than those who had not (p = 0.008).[4]

Complications of dental implants usually result from a combination of infection and host of inflammatory responses or a lack thereof. The literature reports a case report of oral cutaneous fistula associated with an osseointegrated dentoalveolar implant which occurred after three months.[5]

There are also reports of fistulas mimicking a brachial cyst caused by an ectopic salivary gland. In one case a 24-year-old man presented with intermittent clear drainage on both sides of the middle neck. The lesion underwent surgical excision, and the pathological examination revealed heterotopic salivary gland tissue.[6]

Periapical actinomycosis is among the rarest forms of actinomycosis in the maxillofacial region. Although rare, it is one of the most common infections that result in an orocutaneous fistula.[7] More rarely OCF is secondary to a neoplasm.  Squamous cell carcinoma is the most common oral cavity neoplasm, and if the fistula occurs, it has a poor prognosis because lymphatic drainage has most likely taken place by the time of diagnosis.

Epidemiology

In the largest study of odontogenic cutaneous fistulas reported by Guevara-Gutiérrez et al., which examined 75 cases during an eleven-year timeframe, the mean age was 45 years. The most affected age group was 51 years and over (28%). Female to male ratio was 1.14 to 1.[1] Many factors were suspected of inducing dental infections and facilitating the formation of sinus tracts such as poor oral hygiene, xerostomia, and unsatisfactory surgical procedures. In the study of Lee et al., in two out of 33 patients, fistulas were related to actinomycosis. In one patient, the fistula was presumed to have been caused by osteoradionecrosis after radiation therapy for mandible cancer.[8]

Histopathology

The possibility of a neoplastic cause may require a histopathological examination of oral cutaneous fistula. A biopsy is also useful for the diagnosis of actinomycosis which has characteristic pseudohyphae appearing as clublike projections that stretch out from a central basophilic staining core [7].

History and Physical

In contrast with acute infections that cause extreme pain, chronic dental infections are often asymptomatic. In the series of Guevara-Gutierrez et al., the authors found the most frequent locations of odontogenic cutaneous fistula were the mandibular angle (36%), the chin (28%), and the cheeks (24%). The predominant morphological lesions were a nodule (52%), followed by an abscess (19%) and fistula (12%). Of the 75 patients, in 74 (99%) the location of the OCF was adjacent to the causative tooth.[1] In a study by Lee and colleagues, 27 patients (81.8%) with oral cutaneous fistulas had been misdiagnosed in previous clinics, resulting in one or more recurrences.

In chronic osteomyelitis with drainage, pain may not be a symptom. In some cases of actinomycosis, yellow granules are observable at clinical examination. Signs of salivary gland infections include swelling, pain, and trismus if the parotid gland is involved.

Evaluation

In case of odontogenic cutaneous fistula, a tooth with a necrotic pulp often has a normal appearance or presents with only slight alterations in its color. Radiographic analysis can be used to show bone loss in the apex of the infected tooth.[9] This type of study may be complemented with the placement of a gutta-percha point.[1] Unless the infection is widespread and severe, imaging studies such as a CT scanning or MRI usually is not necessary.

In some cases, cone-beam studies have been used to find the exact origin and extent of the infection.[10] CT fistulography has also been used to help diagnose an OCF of dental origin.[11]

Treatment / Management

For dental infections, incision and drainage are often necessary. This treatment includes extraction of the affected tooth, pulpotomy, or pulp removal and drainage.[12] If the tooth is salvageable, root canal therapy also known as endodontic therapy is sufficient to eliminate the infection. Antibiotic coverage is necessary to reduce the number of microbes causing the infection. Usually, dentists recommend the use of amoxicillin with clavulanic acid as it is effective for both gram-positive and gram-negative organisms. In the study of Lee et al., no recurrence was encountered after 1-year follow-up in all 33 patients treated with fistulectomy and extraction or endodontic therapy with antibiotic therapy.[8]

In a study by Andrews et al., the authors reported the beneficial use of a negative-pressure vacuum-assisted closure technique (VAC) to facilitate the closure of OCF.[13]

Differential Diagnosis

Because orocutaneous fistulas are not common, misdiagnosis of those from a dental origin is not unusual. In the study of Lee et al., the majority of patients had been misdiagnosed initially. Oral cutaneous fistulas were thought to be an epidermal cyst (24, 2%), furuncle (21.2%), subcutaneous mycosis (15.2%), squamous cell carcinoma (9.1%), basal cell carcinoma (6.1%) and foreign body granuloma (6.1%).[8]

Prognosis

Treatment of odontogenic cutaneous fistulas results in most cases in an excellent prognosis. WIthout infection control, cellulitis can lead to Ludwig angina and cavernous sinus thrombosis.[14] For OCF secondary to neoplasm, poor prognosis is expected because of rapid lymphatic drainage.

Enhancing Healthcare Team Outcomes

Extraoral and dental examinations are required to make a diagnosis of odontogenic cutaneous fistulas. The early detection of dental problems and preventative dentistry are the best deterrents of OCF formation. Healthcare workers including nurse practitioner, physician assistant, and clinician who see oral cavity wounds and ulcers should refer these patients to a dentist for further workup. Most healthcare clinics are not set up for a thorough oral cavity exam.


References

[1] 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. International journal of dermatology. 2015 Jan;     [PubMed PMID: 24134798]
[2] Roche E,García-Melgares ML,Laguna C,Martín-González B,Fortea JM, [Chronic cutaneous fistula secondary to mandibular osteomyelitis]. Actas dermo-sifiliograficas. 2006 Apr;     [PubMed PMID: 16796969]
[3] Ruggiero SL,Dodson TB,Fantasia J,Goodday R,Aghaloo T,Mehrotra B,O'Ryan F, American Association of Oral and Maxillofacial Surgeons position paper on medication-related osteonecrosis of the jaw--2014 update. Journal of oral and maxillofacial surgery : official journal of the American Association of Oral and Maxillofacial Surgeons. 2014 Oct;     [PubMed PMID: 25234529]
[4] Dawson C,Gadiwalla Y,Martin T,Praveen P,Parmar S, Factors affecting orocutaneous fistula formation following head and neck reconstructive surgery. The British journal of oral     [PubMed PMID: 27502876]
[5] Mahmood R,Puthussery FJ,Flood T,Shekhar K, Dental implant complications - extra-oral cutaneous fistula. British dental journal. 2013 Jul;     [PubMed PMID: 23887526]
[6] Ogawa K,Kondoh K,Kanaya K,Ochi A,Sakamoto T,Yamasoba T, Bilateral cervical fistulas from heterotopic salivary gland tissues. ORL; journal for oto-rhino-laryngology and its related specialties. 2014;     [PubMed PMID: 25591615]
[7] Pasupathy SP,Chakravarthy D,Chanmougananda S,Nair PP, Periapical actinomycosis. BMJ case reports. 2012 Aug 1;     [PubMed PMID: 22854234]
[8] Lee EY,Kang JY,Kim KW,Choi KH,Yoon TY,Lee JY, Clinical Characteristics of Odontogenic Cutaneous Fistulas. Annals of dermatology. 2016 Aug;     [PubMed PMID: 27489421]
[9] Witherow H,Washan P,Blenkinsopp P, Midline odontogenic infections: a continuing diagnostic problem. British journal of plastic surgery. 2003 Mar;     [PubMed PMID: 12791368]
[10] White SC, Cone-beam imaging in dentistry. Health physics. 2008 Nov;     [PubMed PMID: 18849696]
[11] Ranga U,Aiyappan SK,Veeraiyan S, A Case of Intermittently Discharging Skin Lesion: Orodentocutaneous Fistula Demonstrated on CT Fistulography. Journal of clinical and diagnostic research : JCDR. 2014 Aug;     [PubMed PMID: 25302272]
[12] 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. International endodontic journal. 2009 Mar;     [PubMed PMID: 19228218]
[13] Andrews BT,Smith RB,Hoffman HT,Funk GF, Orocutaneous and pharyngocutaneous fistula closure using a vacuum-assisted closure system. The Annals of otology, rhinology, and laryngology. 2008 Apr;     [PubMed PMID: 18478840]
[14] Ferrera PC,Busino LJ,Snyder HS, Uncommon complications of odontogenic infections. The American journal of emergency medicine. 1996 May;     [PubMed PMID: 8639212]