Friday, 9 October 2009

An Unusual Presentation of a Lingual Ulcerative Lesion - eMedicine Case

It's weekend, minna-san~~~
What do you usually do at weekend?

It's little bit unusual case found at eMedicine Case Presentation about Oral Healt and Dentistry, but I managed to found one of it.
So, here we go...

An Unusual Presentation of a Lingual Ulcerative Lesion
Talib A. Najjar, DMD, MDS, PhD; Prabhjot Singh, B Med Sc, DDS

A 78-year-old man presents to the oral surgery clinic complaining of discomfort along the left lateral border of his tongue. He states that he first noticed a lesion on his tongue approximately 4 weeks ago, and since that time it has progressively become larger and more bothersome. The patient denies having any fevers, chills, drainage at the site of the lesion, or generalized swelling in the head and neck. A blade implant was placed in the left mandible 15 years ago to restore the lower left 1st and 2nd premolar teeth (a blade implant is an older system to replace missing teeth; this has been replaced by the use of single or multiple titanium implants to replace missing teeth). He has no known chronic medical conditions. He has no allergies and does not take any medications. He reports a 30 pack-year history of smoking, as well as occasional alcohol use.

On physical examination, the patient appears well-nourished and in no apparent discomfort. His vital signs include an oral temperature of 99.5°F (37.5°C), a pulse rate of 78 bpm, a blood pressure of 134/88 mm Hg, a respiratory rate of 14 breaths/min, and an oxygen saturation of 98% while breathing room air. The cardiovascular, respiratory, and abdominal examinations are normal. His head and neck examination is negative for any gross swelling or lymphadenopathy. The intraoral examination reveals an indurated nodular mass 1 cm × 1 cm in diameter on the left lateral margin of the tongue with 2 separate areas of ulceration both superior and inferior to the mass (see Figure 1). The mass is not tender to palpation and is without any bleeding or drainage. There is no sublingual elevation, induration, or asymmetry, and his maximal oral opening is measured at 45 mm. Well-healed incisional wounds from his prior restorative work of the left mandible are noted.

The initial workup consists of a panoramic radiograph, a culture swab, and an incisional wedge biopsy of the lesion. The panoramic radiograph shows the blade implant in place in the left mandible (see Figure 2). There is minimal bone destruction around the blade implant and no other bony pathology can be seen. The biopsy specimen is sent for histopathologic diagnosis (see Figure 3).
What is the most likely diagnosis?

Hint: Definitive diagnosis requires close examination of the microscopic biopsy specimen.
a. Thyroglossal cyst
b. Apthous ulcer
c. Lingual carcinoma
d. Actinomycosis

The correct answer for the quiz is d. Actinomycosis

he diagnosis of actinomycosis was made based on the patient's physical examination as well as the histologic and microbiologic evaluation. The wedge biopsy demonstrated marked acute inflammation, microabscess formation, and visible organisms morphologically consistent with actinomycosis (see Figure 3). This case is an atypical presentation of actinomycosis of the head and neck. A more common presentation consists of chronic submandibular swelling, usually brawny induration with fistula formation and purulent drainage. The presentation of actinomycosis on the tongue itself is also unusual. Firm masses with associated ulceration on the lateral border of the tongue in the presence of a significant tobacco and alcohol history is usually associated with malignancy, typically squamous cell carcinoma. Although the presence of a chronic, firm swelling is consistent with actinomycosis infection, malignancy must also be considered. Therefore, in addition to the microbiologic analysis, an incisional biopsy of the lesion must be obtained.

There was no evidence of other cervicofacial, abdominal, pelvic, or pulmonary actinomycosis in this patient. The frequency of cervicofacial, abdominal/pelvic, and pulmonary actinomycosis is 55%, 25%, and 15%, respectively; subcutaneous actinomycosis as well as actinomycosis at other sites accounts for the remaining 5% of cases. Although the pathogenesis is unclear, the 2 primary predisposing factors for the development of an actinomycosis infection are: (1) the presence of an introductory pathway into the tissue, and (2) a suitable environment for the bacteria to thrive. Trauma seems to play an important role in most cases by initiating the portal of entry for the organism. In this case, it was postulated that trauma associated with the blade implant may have been involved. Some investigators have proposed that other microorganisms, such as Staphylococcus aureus, act in a synergistic fashion to create an anaerobic environment for the Actinomyces to multiply. In the cervicofacial area, the infection is frequently of odontogenic origin and can be the result of oromaxillofacial trauma, dental intervention, or poor oral hygiene. Actinomyces israelii reproduces easily in the presence of necrotic tissue and can lead to the clinical emergence of the disease. The presence of dental or periodontal disease and devitalized tissue after trauma or surgery provides an adequate environment for Actinomyces species to flourish. Actinomyces requires the presence of many other types of bacteria to proliferate; the specific ecosystem thus formed has a low oxidoreduction potential that is favorable to anaerobic growth. This ecosystem is formed with polymicrobic "associate" flora working in a synergistic fashion. It destroys local tissue, which is a highly vascularized and aerobic region, and replaces it with a poorly vascularized granulomatous tissue, thereby permitting the development of an anaerobic milieu that is essential to Actinomyces development.[1,2,3,4]

Clinical findings that are commonly seen in an actinomycosis infection include suppuration, draining sinuses, fistulas, and the presence of "sulfur granules" (small, white-yellow granular aggregates of bacterial filaments) in exudates or tissues. In patients with cervicofacial actinomycosis there is a high tendency for poor dental hygiene, caries, oral trauma, dental extraction, or dental abscess. The oral condition in this patient was conducive for the development of actinomycosis. A typical clinical presentation is a hard, "woody" swelling in the mandible and neck regions; an associated abscess may be present in contiguous soft tissue. Ulceration and induration in the tongue were noted in this patient, but there was no evidence of abscess formation, fistula tract, or sulfur granule exudate. Pain and mild pyrexia are usually present in these cases, and sinus tracts may occur in long-standing disease. Classically, the infection presents as a slowly enlarging and slightly tender swelling that may become indurated as a result of fibrosis and scar formation. The fibrosis may play an important role in the pathogenicity of the infection, as the microorganisms are virtually protected from the host defense; therefore, they are more resistant to antibiotics. The pathogenicity ranges from an acute form, with rapid onset and purulent drainage from multiple sinus tracts, to a slowly progressing chronic form characterized by indurated fibrosis with little suppuration. The lesion in this patient falls in the latter category, with induration, fibrosis and ulceration of the lingual mucosa. The infection usually occurs from a few weeks to a few months after the organism penetrates through the oral portal of entry (such as a facial bone fracture, periodontal socket, extraction site, pericoronitis, periapical inflammation, root canal treatment, or periodontal surgery).[1,2,3,4]

Histologically, actinomycotic lesions are characterized by mixed suppurative and granulomatous inflammatory changes. There is proliferation of the connective tissues and the presence of sulfur granules. Under the microscope, sulfur granules may appear cauliflowered at low magnification, whereas at higher magnification the inflammatory reaction can be seen. These granules may be visualized in preparation of the biopsy specimen while the test tubes are being rotated manually. They can also be identified by washing sampled material and crushing it between a slide and coverglass after it has been Gram-stained. With Gram staining, these microcolonies contain Gram-positive, filamentous or branching bacteria. Occasionally, companion bacteria may be observed. It is important to stress that similar granules can be found in other bacterial infections, such as Nocardia infection and botryomycosis. Botryomycosis refers to a Staphylococcus aureus infection that mimics actinomycosis. Nocardia may be differentiated from Actinomyces since it is acid-fast when stained. Gel diffusion methods and fluorescent antibody tests (immunofluorescence with fluorescein isothiocyanate antiserum) are also helpful because they can differentiate A. israelii from other filamentous anaerobes that produce granules in tissue. They can be used retrospectively with formalin- and paraffin-embedded biopsy specimens. Biopsies of the granulomatous lesion or of the fistula are especially useful when no purulent material is present and when the diagnosis remains unclear despite laboratory evaluation. In the absence of absolute bacterial identification from culture, the diagnosis must rely on the clinical presentation and histopathologic findings.[4]

Surgical management includes excision/debridement of abnormal or devitalized tissue, such as infected masses and curettage of osteomyelitic bone lesions. Drainage of any existing abscesses or fistula tracts is also necessary, when present. Surgical treatment alone, however, is not sufficient for treatment. Depending on the type of infection, prolonged antibiotic therapy is the cornerstone of management. Classically, these infections are treated with penicillin-type antibiotics; the duration of therapy is targeted to the patient's clinical condition and response to treatment. Penicillin resistance is uncommonly observed. Certain antibiotics (namely, metronidazole, aminoglycosides, co-trimoxazole, or cephalexin) have no role in treating this infection. Limited case reports have reported success with fluoroquinolones, such as levofloxacin or moxifloxacin. This infection may respond to some second or third generation cephalosporin, macrolide, or tetracycline antibiotics.[1,2,4]

The patient in this case was started on a course of oral penicillin VK at 500 mg every 6 hours for 3 weeks for treatment of the actinomycosis infection. Because it was felt that the blade implant and associated prosthesis could be harboring actinomycotic colonies and could seed more bacteria in the left lateral tongue, it was decided to remove the blade implant along with the prosthesis supported by the implant. The patient was followed for approximately 1 month, at which time total resolution of the lesion was observed (see Figure 4).

You are examining a patient who you suspect may have an actinomycotic infection. As part of the history, the patient mentions that he had dental surgery earlier this year. He states that he often has gum infections, likely as a result of his admittedly poor oral hygiene habits. Which of the following factors in this patient's history could contribute to the development of actinomycosis?
a. Trauma
b. Local Bacterial co-infection
c. Poor oral hygiene
d. All of the above

If the patient described above were to be diagnosed with actinomycosis, which of the following choices would be the most appropriate initial course of treatment?
a. Laser surgery
b. Surgical resection
c. Antibiotic therapy
d. Cryosurgery

So what is your answer?
Ganbatte ne~~~

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