A fistula is an abnormal pathway between 2 anatomic spaces or a pathway that leads from an internal cavity or organ to the surface of the body. A sinus tract is an abnormal channel that originates or ends in one opening. An orofacial fistula is a pathologic communication between the cutaneous surface of the face and the oral cavity.
In the literature, the terms fistulas and sinuses are often used interchangeably. Stedman's Medical Dictionary defines a sinus as a fistula or tract leading to a suppurating cavity. Orofacial fistulas are not common, but intraoral sinus tracts due to dental infections are common. When infection or neoplasia is involved, immediate treatment is necessary. Dental infections, salivary gland lesions, neoplasms, and developmental lesions cause oral cutaneous fistulas, fistulas of the neck, and intraoral fistulas.
Chronic dental periapical infections or dentoalveolar abscesses cause the most common intraoral and extraoral fistulas. These dental periapical infections can lead to chronic osteomyelitis, cellulitis, and facial abscesses. Infection can spread to the skin if it is the path of least resistance. Fascial-plane infections, space infections, and osteomyelitis can cause cutaneous fistulas. Fascial-plane infections often begin as cellulitis and progress to fluctuant abscess formation. Compared with the other conditions, fluctuant abscess formation is more likely to result in cutaneous fistulas.
Rarely, a cutaneous lesion such as a furuncle can be misdiagnosed as a sinus tract to the skin of the face. One case report demonstrates this occurrence from a periapical infection from the right central mandibular incisor, which drained to the patient's chin. Because the tooth could not be restored, it was extracted, which resolved the lesion.
Another case with cutaneous manifestations involved a 44-year-old woman with a draining lesion to the skin just lateral to the nasofacial sulcus. Oral antibiotics did not help resolve the lesion. The patient had poor dentition, and a panoramic radiograph showed 2 periapical radiolucencies of the maxillary right lateral incisor and canine. The teeth were extracted, which resolved the lesion. Sheehan et al recommend a dental examination and radiographs to rule out infection of dental origin to the cutaneous face or neck.
Pathophysiology
Origins and spread, salivary gland fistulas, oral antral and oral nasal fistulas, iatrogenic fistulas (eg, dental implant placement), and miscellaneous orocutaneous fistulas are addressed here.
Origins and spread
Dentoalveolar abscesses originate from direct extension or continuity from an acute irreversible pulpitis that spreads beyond the apex of the tooth. Another cause of dentoalveolar abscesses is an acute exacerbation of a chronic apical periodontitis or periapical granuloma. Periapical granulomas may remain quiescent because the inflammatory cells are walled off by connective tissue. A periapical granuloma may be exacerbated if the patient's resistance to the organism decreases or if the number of microorganisms increases. This condition is also termed a phoenix abscess.
Unusual dental malformations may lead to periapical dental infections. Dens in dente or dens evaginatus, an axial invagination of enamel and dentin into the dental papilla, frequently develops periapical infections, which can lead to sinus tract formation. Even more rarely, taurodontism consisting of elongated crowns or apically placed furcations with pulp chambers with increased occlusal-apical height can cause periapical disease, leading to a sinus tract or parulis.
Most of these dental infections remain intraoral, and they most often spread to the facial or buccal side of the alveolar ridge. If the dental roots are closer to the lingual side, as they often are with the maxillary anterior teeth and the lingual roots of the maxillary molars, a palatal sinus tract may develop. When a sinus tract appears as an intraoral papule or pustule, it is called a parulis/gum boil. When a chronic infection is acutely exacerbated or persistent, the infection can spread to the facial skin, most commonly in the area of the chin. Numerous barriers, including bone, muscle, and facial planes, determine the path of infection spread.
Yasui et al reported a cutaneous fistula of dental origin. A 75-year-old Japanese woman presented with the chief complaint of a left-cheek skin lesion with mild pain. A subcutaneous nodule with erythematous skin was on her left cheek. Dental examination demonstrated a radiolucent area in the left-lower first molar periapical region. The tooth was asymptomatic. Antibiotic therapy and endodontic therapy eliminated the subcutaneous nodule. The authors recommend a complete dental evaluation be performed when a subcutaneous facial nodule is encountered.
If the infection originates from a region of the maxillary molar, intraoral spread of the infection occurs buccally. When the infection spreads inferior to the superior attachment of the buccinator muscle, it remains in the oral cavity. If the infection path moves superior to this attachment, cutaneous spread with fistula formation is possible. Infection from the maxillary molar teeth can spread to the palate and into the maxillary sinus, depending on the position of the lingual roots of the teeth.
Maxillary dental infections also may spread to the canine fossa, buccinator space, lateral pterygoid space, and infratemporal space. Spread of infection to the lateral pterygoid and infratemporal spaces is associated with trismus. Infection of maxillary premolars almost always stays confined to the oral cavity and most commonly spreads to the buccal side of the alveolar ridge. Infection from the maxillary anterior teeth is usually contained within the oral cavity. Spread of infection superior to the levator anguli oris muscle or orbicularis oris muscle may result in cutaneous spread.
Infection from the mandibular molars is usually confined to the lingual aspect of the oral cavity by the mylohyoid muscle and to the buccal aspect by the inferior attachment of the buccinator muscle. If the infection penetrates to the lingual area inferior to the mylohyoid muscle, infections of the submandibular, sublingual, and submental spaces may result. If the infection spreads inferior to the buccinator muscle attachment, cutaneous spread may occur with fistulation.
Infection of the mandibular premolars is almost always confined by the buccinator muscle in the oral cavity and most commonly spreads to the buccal side. Infection of the mandibular anterior teeth is usually confined to the oral cavity and spreads facially. If the infection spreads below the mentalis muscle, cutaneous spread may occur. Mandibular space infections may involve the submandibular, submental, pterygomandibular, masseteric, lateral and posterior pharyngeal, parotid, and carotid spaces.
Chronic osteomyelitis more frequently drains through an extraoral sinus opening than through an intraoral opening. Osteomyelitis is more likely to develop in patients with uncontrolled diabetes, in those who have undergone jaw irradiation because of a previous malignancy (osteoradionecrosis), and in those with metabolic bone diseases such as Paget disease (osteitis deformans) or Albers-Schönberg disease (osteopetrosis).
Garré osteomyelitis is a unique chronic osteomyelitis with a prominent periosteal inflammatory reaction that follows periapical disease or tooth extraction. It is uncommon, with average age of 13 years. Elimination of pulpal periapical infection through endodontic therapy without endodontic surgery was shown to be an effective treatment. In this case report total bone healing was observed 1 year later.
Lymphatic spread is also common. Lymphadenopathy with movable tender nodes is a common finding with dental infections. Inflammatory lymph nodes caused by dental infection rarely result in cutaneous fistulas.
Salivary gland fistulas
Salivary gland fistulas are rare except with minor salivary gland mucocele. Saliva from damaged salivary glands or ducts finds the path of least resistance but rarely escapes through the skin or mucosa. The parotid duct or Stensen duct comes close to the cutaneous surface as the duct crosses the outer surface of the masseter muscle. A rare submandibular fistula was reported in association with a ranula of the submandibular gland. A case was reported with a cutaneous opening caused by an ectopic salivary gland. This instance mimicked a branchial cleft or branchial cyst fistula. Gerhard et al state that ectopic salivary gland fistulas should be in the differential diagnosis of branchial fistulas.
One interesting case is of a 4-year-old boy with an anterior cervical fistula, which secreted salivalike fluid while he was eating. Head and neck examination revealed an opening posterior to the hyoid bone. The fistula ascended superficially to the anterior cervical muscles, with a cyst anterior to the hyoid bone, continuing to the left submandibular gland. According to Hayasaka et al, no previous reports have described the Wharton duct running from the submandibular gland to the anterior cervical skin.
Trauma, microorganisms, neoplasms, xerostomia, immunosuppression, and malnutrition usually are the cause of infections that result in fistulas from salivary glands. Iatrogenic causes include surgery and radiation therapy. Patients who are ill or debilitated may be prone to these infections. Actinomycosis, syphilis, tuberculosis, salivary calculi, and malignancy are other etiologic agents that cause salivary gland infections. Staphylococcus aureus, Streptococcus viridans, and Escherichia coli most commonly are found in these infections.
Sjögren syndrome, which has a female-to-male ratio of 10:1, is an immunologic disease that causes xerostomia. Patients with this disease have dry eyes, a dry mouth, and, in the secondary form, immunologic connective-tissue disorders, most commonly rheumatoid arthritis. Patients with Sjögren syndrome may be more prone to parotid gland infection, but this infection rarely results in sinus tract formation to the skin or oral mucosa.
Salivary gland stones or sialoliths can be a site of infection. These are most commonly associated with the Wharton duct, the major duct for the submandibular gland. Any gland can be affected. The sialolith blocks the ductal secretion of saliva, causing a fluid buildup that creates a potential site for infection. A ranula or mucous retention phenomenon of the floor of the mouth results from this blockage; when this is large, it is treated by marsupialization. This technique exteriorizes a cyst or other such enclosed cavity by resecting its anterior wall and suturing the cut edges of the remaining wall to adjacent edges of the skin, thereby creating a pouch.
Drage et al presented 3 cases of a migrating salivary stone or sialolith to adjacent tissues, resulting in cutaneous fistulas from salivary gland origin. Two patients were treated successfully surgically, which resulted in resolution of the fistulas.
A mucocele or mucous retention phenomenon occurs when minor salivary gland ducts are damaged. The walling off of mucin with granulation tissue causes a cystlike structure; on the floor of the mouth, this structure is called a ranula. This lesion is usually painless, and the patient often reports that it swells and breaks with a fluid discharge. More than 50% of patients with a mucocele or mucous retention phenomenon are younger than 21 years, and it occurs equally in males and females. Patients may remember biting their lip.
Clinically, the mucocele appears as a clear or bluish fluctuant vesicle. It is most common on the lower lip and can be found in minor salivary glands of the palate and retromolar pad area. It is extremely rare on the upper lip. Physical trauma to the lower lip is the most common cause of mucoceles. Most upper lip swellings are due to cysts, odontogenic infections, and salivary gland tumors. The differential diagnoses include salivary gland neoplasms, especially mucoepidermoid carcinoma, vascular malformation, hemangiomas, and fibrous nodules or fibroma.
Oral antral and oral nasal fistulas
Tooth extraction, tuberculosis, syphilis, leprosy, malignant neoplasms, phycomycoses, midline granuloma (a form of lymphoma), and developmental clefts may cause oral antral and oral nasal fistulas. The most common cause of oral antral fistulas is tooth extraction. Maxillary first molars account for 50% of oral antral fistulas caused by extractions. Maxillary second and third molar extractions account for the other 50%. Prior to extraction, infection of these teeth may create a communication with the antrum. Approximately 10% of all sinusitis cases have a dental origin.
Lopatin et al concluded that an endoscopic approach to chronic maxillary sinusitis of dental origin is a dependable technique associated with less morbidity and a lower rate of complications.
When patients ingest food and liquids, these may enter the nasal cavity and antrum, causing an unpleasant salty taste and fetid breath. Infection may cause sinusitis, which results in throbbing headaches that are aggravated by head movement. Nocturnal cough and epistaxis may result from drainage of exudate to the oropharynx and nose. Swelling and redness over the sinus and pain beneath the eye, especially with palpation, may occur.
In all cases of idiopathic sinusitis, causes such as infection, polyps, and neoplasms should be excluded. Neoplasms, such as squamous cell carcinoma, may manifest as sinusitis until the neoplasm enlarges enough to show signs of malignancy. By this stage, metastasis may have occurred.
Bisphosphonate-related osteonecrosis of the jaws
An additional cause for oral cutaneous fistulas is bisphosphonate-related osteonecrosis of the jaws (BRONJ). Fortunately, only a small percentage of patients develop BRONJ.Bisphosphonates that are intravenously administered are used to treat patients with osteoporosis, patients with cancer who have hypercalcemia associated with malignant disease, and patients with multiple myeloma or metastatic tumors (breast, lung, prostate) in the bones.
Bisphosphonates are bone resorption inhibitors, inhibiting osteoclast activity and thus decreasing vascular supply of oxygen and host defense cells. Malignancy in bone, dexamethasone therapy, and intravenous bisphosphonate therapy increase the chances of developing BRONJ.Oral/dental causes increasing BRONJ include abscesses, periodontal disease, dental caries, exostoses and tori, and dental extractions.
Preventive dentistry plays the most important role for prevention of BRONJ. One study showed that complete prevention of this complication is not currently possible. However, preventive dental care reduces this incidence, and nonsurgical dental procedures can prevent new cases. For those who present with painful exposed bone, effective control to a pain free state without resolution of the exposed bone is 90.1% effective using a regimen of antibiotics along with 0.12% chlorhexidine antiseptic mouth rinses.
One study used platelet-derived growth factors (PDGFs). The authors treated 12 patients with refractory BRONJ and a history of long-term bisphosphonate therapy. Each patient had mucosal ulceration with exposed necrotic bone. The treatment also included bone resection. The surgical intervention they used was a marginal resection limited to the alveolar bone. Ten of the patients recovered with complete mucosal and bone healing.
The treatment objectives for patients with an established diagnosis of BRONJ are to eliminate pain, control infection of the soft and hard tissue, and minimize the progression or occurrence of bone necrosis. Patients should have regular check-ups before, during, and after bisphosphonate therapy. Management includes antibiotics, pain control, and chlorhexidine mouth rinses over long periods of time.
BRONJ treatment can be surgical or nonsurgical. Nonsurgical management includes antibiotics, systemic or topical antifungals, antimicrobial rinses, ceasing bisphosphonate therapy, and stopping dental therapy. Surgical solutions for BRONJ are limited due to the patient’s decreased healing ability. Before treatment with an intravenous bisphosphonate, the patient should have a thorough oral examination, extract nonrestorable teeth, complete all invasive dental procedures, and obtain best possible periodontal health. Patients with full or partial dental prostheses should be examined for areas of mucosal trauma. Patients need to be educated as to the importance of dental hygiene and regular dental evaluations, and instructed to report any pain, swelling, or exposed bone.
Patients who take oral bisphosphonates for less than 3 years and have no clinical risk factors, no alteration or delay in dental surgery is necessary. The risk of developing BRONJ with oral bisphosphonates is very small but increases when therapy exceeds 3 years.For patients who are taking oral bisphosphonates for less than 3 years with or without corticosteroids, the prescribing physician should consider discontinuing therapy for 3 months prior to oral surgery, if systemic conditions permit. The bisphosphonate should not be restarted until healing has occurred.
Miscellaneous orocutaneous fistulas
An oral cutaneous fistula leads to esthetic problems due to the continual leakage of saliva from the oral cavity to the face. Malignancy, inflammation, and trauma are the most common causes.
Traumatic fistulas may be due to injury or surgical repair in areas where mucosal and epidermal surface epithelia line the fistula wall. No inflammation is associated with this type of fistula unless an infection develops (see the image below).
Gunshot wound causing an oral cutaneous fistula. Courtesy of Alexander Pazoki, DDS, LSU School of Dentistry, New Orleans, La.
Dental implants can develop infections, leading to intraoral and possibly extraoral sinus tract drainage. One case report described an implant failure causing a peri-implantitis. This was treated successfully without removal of the implant. In most cases, the implant must be removed when peri-implantitis occurs. This case involved an adjacent tooth with apical periodontitis that may have spread from the tooth or the peri-implantitis spread to the tooth, causing its demise. Treatment included a root canal, debriding the apical bone lesion, and using guided bone regeneration. Normal healing occurred and an esthetic result was achieved.
Failed endodontic therapy or treatment after endodontic therapy can be a source of dental infection. Tanalp et al reported a persistent sinus tract occurring after a post and core was placed on an anterior tooth. Two separate root perforations were causing the persistent infection. Granulation tissue was removed, the perforations were sealed with mineral trioxide aggregate, and bone graft was packed in the resorptive bone areas. Four months after treatment, the patient had no signs or symptoms.
Ricucci et al reported 2 cases in which calculus formation was reported as a cause of a persistent sinus tract after root canal therapy. In one case, a sinus tract developed that did not heal after conventional root canal therapy and apical surgery. Extraction of this tooth revealed calculuslike material on the root surface. The other case showed radiographic signs of healing after apicectomy. Histology of the apical biopsy specimen demonstrated a calculuslike material on the surface of the root apex. The presence of calculus on the root surfaces of these teeth may have contributed to endodontic treatment failure.
Neoplastic fistulas result from the penetration of a neoplasm from the oral cavity to the outlying skin. The most common malignancy in the oral cavity is squamous cell carcinoma. Fistulas caused by squamous cell carcinoma have a poor prognosis because of skin lymphatic drainage (see the images below).
Squamous cell carcinoma causing an oral cutaneous fistula. Courtesy of Alexander Pazoki, DDS, LSU School of Dentistry, New Orleans, La.Squamous cell carcinoma of the sinus that penetrates the maxillary ridge.
Actinomycosis, although rare, is one of the most common infections that result in a fistula from the oral cavity to the skin. These infections respond to large doses of penicillin or beta-lactam/beta-lactamase inhibitors administered for a minimum of 6 weeks.
Actinomycosis has been documented as a cause of continual, recurrent, periapical disease associated with endodontically treated teeth. One case presented by Jeansonne demonstrated persistent periapical disease with recurrent sinus tracts. No pain or swelling was present after clinically acceptable initial endodontic treatment, but a periapical lesion developed. After routine endodontic retreatment, the periapical lesion persisted and a sinus tract developed. The sinus tract healed with antibiotic therapy but recurred within a few months. The sinus tract recurred and disappeared with antibiotic therapy over a period of 5 years. After histological diagnosis confirmed actinomycosis, the lesion was treated with antibiotics and periapical surgery. It finally resolved in 5 months.
Fistulas may arise from developmental cysts of the neck region, such as thyroglossal duct, dermoid, sebaceous, preauricular, and branchial arch cysts. Nasopalatine duct cysts occasionally secrete fluid to the anterior palate and the site of the duct.
Intracranial extension and a cutaneous sinus tract are rarely seen with craniofacial dermoid cysts. Scolozzi et al reported a case of a 1-year-old girl who was initially seen with a cutaneous fistula of the frontotemporal region, from an intracranial dermoid cyst. The patient was treated surgically with a right lateral orbitotomy by a bicoronal approach. The cyst was seated within the lateral orbital wall, with intracranial extension through the temporal and sphenoidal bones to the dura of the temporal lobe. Histopathologic analysis confirmed the diagnosis of a dermoid cyst. Craniofacial dermoid cysts may rarely be associated with a cutaneous sinus tract and/or intracranial extension. Failure to identify and treat these lesions may lead to recurrent infection with a potential for meningitis or cerebral abscess. The authors strongly recommend CT scanning and MRI before surgical treatment of any cutaneous fistula in the head and neck region.
The thyroglossal duct cyst is the most common of the developmental cysts of the neck. In embryonic development, the duct follows a path from the tongue to the normal position of the thyroid. These cysts have an equal incidence in females and males, and they usually are observed within the first 2 weeks of life. Proliferation of duct tissue may continue, causing enlargement of the thyroglossal tract. If a sinus opening to the neck occurs, the most frequent location is just below the hyoid bone. The epithelial lining may consist of squamous or pseudostratified columnar epithelium. Infection may occur anywhere along the duct tract, causing purulent exudate. Complete surgical removal of the thyroglossal duct epithelium is the treatment of choice; however, complete removal is difficult and recurrence is frequent.
The lateral branchial arch cyst is the most common developmental cyst of the lateral neck. It occurs when the second branchial arch or second pharyngeal pouch is not eliminated in normal development. The endoderm of this pouch normally becomes the tonsil. A cutaneous sinus, a mucosal sinus, or both may occur. Lateral branchial arch cysts occur equally in males and females, and they may be familial. They can be unilateral or bilateral. They may occur in children; ruptured cysts may occur in adults. Usually, the opening is near the anterior border of the sternocleidomastoid muscle just above the sternoclavicular joint. The differential diagnosis includes a fistula or sinus from an infected or cancer-containing lymph node.
Lymph nodes infected with mycobacteria cause scrofula, a condition in which infection spreads from the node to the skin through a sinus tract. This infection most commonly occurs in the neck. Cat scratch disease is another consideration in the differential diagnosis of sinus tracts from lymph nodes.- Acute dental infections cause extreme pain when they occur in a confined area.
- The pulp is confined in a hard structure, namely, the pulp chamber.
- Most nerve receptors in the tooth are type A delta nerve fibers, which detect pain sensation.
- These fibers interpret the pressure due to edema in infection and inflammation as pain.
- Acute periapical inflammation also causes pain when it is confined to a bony space.
- Pain often decreases or disappears when a sinus tract forms, relieving pressure.
- In chronic osteomyelitis with drainage, pain may not be a symptom.
- An intraoral sinus tract or parulis may be raised or appear as a red-to-yellow ulcer that bleeds easily and exudes pus.
- If infection from the mandible remains confined to the oral cavity or if the infection spreads to the skin, the site of fistulation may be distant from the intraoral infection site.
- In some cases of actinomycosis, yellow granules (often called sulfur granules) are observed at clinical examination. These granules have a characteristic histologic appearance.
- Signs and symptoms of salivary gland infections include swelling, pain, and trismus if the parotid gland is involved. Major salivary gland fistulas are diagnosed by means of probing or sialography.
- Poor oral hygiene and trauma cause most dental infections.
- Compared with other individuals, patients who are immunocompromised, those who are receiving chemotherapy, and those with blood dyscrasias are more likely to have dental infections.
- Xerostomia leads to additional caries due to increased salivary acidity. This effect enhances the growth of cariogenic bacteria and increases the adherence of plaque to the teeth.
- Gram-positive bacteria and gram-negative microorganisms such asStreptococcus mutans; Staphylococcus epidermidis; S aureus; andPorphyromonas, Actinomycoses, Bacteroides, and Fusobacterium species are found in dental infections and periodontal infections.
- Reportedly, an occult root fracture that resulted from excessive endodontic sealer caused an infection and a chronic fistula lasting more than a year. When the root fracture was discovered and treated, the cutaneous sinus resolved within 1 month.
Medical Care
To properly treat any infection, drainage is necessary to decrease the number of microbes and reduce the amount of substrate on which they grow. Antibiotic coverage is necessary to eliminate or reduce the number of microbes causing the infection.- With most dental infections, penicillin is the drug of choice. Penicillin and amoxicillin with or without clavulanic acid are administered empirically to treat the infection before culture and sensitivity results are available. If the patient has an allergy to penicillin, erythromycin (next antibiotic of choice), azithromycin, clarithromycin, or clindamycin can be administered.
- Amoxicillin is often used because of its rapid absorption in the gastrointestinal tract. Amoxicillin with clavulanic acid (Augmentin) is effective in broad-spectrum infections with both gram-positive and gram-negative organisms.
- Doxycycline is effective in the treatment of periodontal disease. The combination of amoxicillin and metronidazole is also effective in treating severe periodontitis in individuals who are HIV positive.
- Intravenous medications that are useful in the treatment of serious facial and orbital infections include nafcillin; cefazolin; ceftriaxone; vancomycin; levofloxacin; and beta-lactam/beta-lactamase inhibitors, including piperacillin/tazobactam, ticarcillin/clavulanate, and ampicillin/sulbactam.
- The treatment of osteomyelitis and actinomycoses infections may require the intramuscular or intravenous administration of penicillin G, followed by oral antibiotics for 6 weeks to 6 months.
- The removal of sequestered or necrotic bone also is indicated.
- Hyperbaric oxygen may be necessary in patients with severe osteomyelitis and osteoradionecrosis. Hyperbaric oxygen is used to promote vascularization, osteogenesis, and collagen synthesis.
Surgical Care
- Dental infections: Incision and drainage is often necessary. This treatment includes extraction of the affected tooth, pulpotomy, or pulp removal and drainage. If the tooth is salvageable, endodontic therapy usually eliminates the infection. In more serious infections, an incision into the soft tissue with dissection may be necessary. Effective drainage in indurated cellulitis infections can be difficult.
- Mucoceles: Treatment includes removal of the fluid-filled sac and surrounding minor salivary glands. This treatment has an excellent prognosis for cure.
- Oral antral fistulas: Repair these fistulas as soon as possible to prevent the spread of infection and patient discomfort. Waiting until any infection is resolved before repair is best. Decongestants and intensive antibiotic therapy may be needed. Wider incision of the sinus or nasal antrostomy may be necessary to drain the infection more rapidly and promote healing. Removal and curettage of the fistula also aids healing and clearing of infection. If a cleft or fistula from the oral cavity to the sinus is too large for surgical closure, prosthetic devices such as dentures and obturators can be used to prevent nasal speech and aspiration of liquids and food.
- Cutaneous fistulas: Scarring may occur. Plastic or oral and maxillofacial surgery can be performed to address scarring.
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