Cervicofacial actinomycoses following sagittal split ramus osteotomy: A case report

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19. St. Haxholdt 0, Krintel JJ, Johannson G: Pre-operative alcohol infusion. Anaesthesia 39:240, 1984 20. Fox AW, Guzmen NJ, Friedman PA: The clinical pharmacology of alcohol, in Barnes HN, Grossman MD, Delbanco (eds): Alcoholism: A Guide for the Primary Care Physician. New York, NY, Springer-Verlag, 1987, pp 29-43 2 1. Hoffman PL, Tatakoff B: Ethanol’s action on brain biochemistry, in Tarter RE, Van Thiel DH: Alcohol and the Brain: Chronic Effects. New York, NY, Plenum, 1985, pp 19-68 22. Corah NL: Assessment of a dental anxiety scale. J Dent Res 48: 496, 1969 23. Kleinknecht RA, McGlynn FD, Thomdike RM, et al: Factor analysis of the Dental Fear Survey with cross validation. J Am Dent Assoc 108:59, 1984 24. Crowne DP, Marlowe D: A new scale of social desirability independent of psychopathology. J Consult Psycho1 24:349, 1960 25. Pallasch TJ: Principles of pharmacotherapy: V. Toxicology and adverse drug reactions. Anesth Prog 36:4 1, 1989 26. Roy-Byrne P. Nutt DJ: Benzodiazepines: Biological mechanisms, in Roy-Byrne P. Cowley DS (eds): Benzodiazepines In Clinical

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Practice: Risks and Benefits. Washington, DC, American Psychiatric Press, 199 1 Hobson JA, Steriade M: Neuronal basis of behavioral state control, in Bloom FE (ed): Handbook of Physiology Section 1: The Nervous System, vol 4. Intrinsic Regulatory Systems of the Brain. Bethesda, MD, American Physiological Society, 1986, pp 710-7’67 Ticky MK, Burch TP, Davis WC: The interaction of ethanol and the benzodiazepine-GABA receptor ionophore complex. Pharmacol Biochem Behav 18:15, 1983 Chan AWK, Lamgun M, Leong FW, et al: Does chronic ethanol confer full cross-tolerance to chlordiazepoxide? Pharmacol Biochem Behav 30:385, 1988 Royce JE: Alcohol Problems and Alcoholism: A Comprehensive Survey. New York, NY, The Free Press, 1989, p 21 Petersen JK, Milgrom P: Pain Relief In the Orofacial Regions. Copenhagen, Munksgaard, 1989 American Dental Association: Treating the active or recovering chemically dependent dental patient. Chicago. IL. American Dental Association, 1989

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1992

Cervicofacial Actinomycoses Following Sagittal Split Ram& Osteotomy: A Case Report GEORGE

WAYNE OZAKI, DDS,* A. OMAR ABUBAKER, DMD, PHD,t C. SOTEREANOS, DMD, MS,* AND GARY T. PATTERSON,

The sagittal split ramus osteotomy (SSRO) has been used for the treatment of various mandibular deformities, including mandibular retrognathia, prognathia, apertognathia, and asymmetry. ‘*2Because of anatomic variations of the mandible, limited access to the surgical site, and technical difficulty of the procedure, there is a potential for many complications. The most common complications associated with the SSRO are bleeding and relapse.3 Other complications include fracture of the mandible, unfavorable split, infection, airway obstruction, necrosis, neurosensory dysfunction, delayed Received from the Department of Oral and Maxillofaciat Surgery, School of Medicine, University of Pittsburgh, Pittsburgh, PA. * Resident. t Formerly, Research Fellow; currently, Assistant Professor, Department of Oral and Maxillofacial Surgery, Medical College of Virginia, Richmond. $ Associate Professor. 5 Assistant Professor of Surgery. Address correspondence and reprint requests to Dr Abubaker: Department of Oral and Maxillofacial Surgery, Medical College of Virginia, Box 566, Richmond, VA 23298. 0 1992 American Association of Oral and Maxillofacial Surgeons 0278-2391/92/5008-0021$3.00/0

DMD§

wound healing, temporomandibular joint disorders, trismus, malocclusion, and malunion or nonunion of the osteotomized segments.3-6 Although infection has been reported as a complication of the SSRO, infection by actinomyces has not been previously reported. This article will describe such a case and its management. Report of a Case A 25year-old, healthy, white woman originally came for treatment of her retrognathic mandible. Following presurgical orthodontics, she was admitted and subsequently underwent a bilateral SSRO with mandibular advancement. She encountered no intraoperative or early postoperative complications. However, 6 weeks postoperatively, the patient returned with a 2 X 3-cm swelling in the left submandibular region (Fig 1). She denied any history of fever, chills, malaise, intraoral or extraoral drainage, or ulcerations. The swelling was indurated and slightly tender to palpation. Intraoral examination showed an entirely normal mucosa with complete healing of the incision sites. Her workup included a complete blood count with a differential count, urine analysis, electrolytes, blood urea nitrogen, serum creatinine, a facial radiographic series, and a

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resulted in little resolution of the mass and the area of skin over it started to become erythematous. Fine-needle aspiration of the lesion yielded no fluid. The patient was, therefore, taken to the operating room and, under general anesthesia, the mass was completely excised. A portion of the tissue was cultured for both aerobic and anaerobic organisms, while the rest was sent for histologic evaluation. The cultures were reported to be sterile after 1 and 2 weeks. The pathology report of the hematoxylin-eosin-stained tissue reported lymphoid tissue (Fig 2) and scattered areas of fibrous connective tissue, surrounded by both acute and chronic inflammatory cells. The histologic section stained with Brown and Brenn stain showed organisms consistent with actinomyces (Fig 3). The patient completed a 4-week course of 4 million units of intravenous penicillin G every 4 hours. She was then maintained on 500 mg of oral penicillin V four times daily for 2 more months. The patient had complete resolution of the condition and no further complications thereafter.

FIGURE 1. The submandibular mass as seen at the initial presentation (arrows indicate the borders of the mass).

Discussion

computed tomography (CT) scan of the face, including the submandibular region. Results of all laboratory examinations were within normal limits. However, the CT scan showed a round mass just below the inferior border of the mandible on the left side. The mass was well circumscribed, but was not confluent with either the submandibular or the parotid glands. The patient was admitted and given 4 million units of aqueous penicillin G intravenously every 4 hours. After 8 days, the mass was greatly reduced in size, but complete resolution was not accomplished. Because of significant clinical improvement, she was discharged on the ninth day and was prescribed 500 mg oral penicillin V four times daily for 10 days. The patient’s case was followed on an outpatient basis. Six weeks after discharge, the mass began to increase in size and the patient was readmitted for further evaluation. She was given 4 million units of penicillin G intravenously every 4 hours. The laboratory workup revealed no change from the previous admission. Two weeks of antibiotic therapy

Actinomycosis is a chronic infectious disease manifesting both granulomatous and suppurative features.7-9 Historically, these microorganisms were once believed to be fungi. 8*9In fact, their appearance under the light microscope had given rise to the descriptive term “ray fungus.“7 However, as our knowledge of their biochemical function and the histochemicaf techniques improved, it became apparent that they were actually bacteria.‘,” Actinomyces are microaerophilic, grampositive, non-acid-fast, branched, filamentous, nonspore-forming bacteria. 7,9,’’ The most commonly found microorganism is Actinomyces israelii. However, A naeslundii, A viscosus, A odontolyticus, and A propionica have also been shown to cause human diseases.’ Actinomyces is a commensal, mucosal inhabitant of the oropharyngeal and gastrointestinal region.’ Its presence in the absence of clinically evident pathology reflects this microorganism’s low potential for virulence

FIGURE 2. Low-power photomicrograph of the lesion showing the lymphoid tissue (hematoxylin-eosin stain, original magnification X10).

FIGURE 3. High-power photomicrograph showing the dark-stained microorganisms consistent with actinomycoses (Brown and Brenn stain, original magnification X 160).

OZAIU ET AL

or invasion.9x12*13 Actinomycosis does not appear to be an opportunistic or communicable disease.’ Cervicofacial actinomycosis may involve the mandible and its overlying tissue, the parotid gland, the tongue, and the maxillary sinus.7~9~‘3-‘6 The mandible tends to be involved more frequently than the maxilla.9~” A breach in the integrity of the mucosa by direct trauma or following a fracture, a tooth extraction, root canal therapy, periodontal or periapical lesion, or some intraoral surgical procedure is thought to be the most likely portal ofentv.9J

1.16.17

On entry, the organisms may stay localized, or they may spread without regard for facial planes to the salivary glands and bone, occasionally causing a tract sinus or fistula of the face or neck?~‘8*‘gSerious complications of cervicofacial actinomycosis include osteomyelitis of the mandible, aspiration into the lungs, and entry into In our case, the portal of entry was the meninges. 7,9~10 thought to be through the surgical incision during the osteotomy, because no other breach in the mucosa was reported between the time of the osteotomy and presentation of the lesion. The presence of lymphoid tissue in the excised specimen indicates that the organisms spread from the portal of entry to a submandibular lymph node, where the lesion presented as a submandibular mass. Cervicofacial actinomycosis may appear clinically as a slowly enlarging, purple to dark red, firm swelling that may be tender and indurated, occasionally with zones of fluctuance.7,9s’2 Often there is abscess formation, with one or more sinus tracts present, causing purulent exudate to drain on the skin surface.’ Patients with this disease generally do not have pain, but they can have nonspecific signs and symptoms such as fever, chills, nausea, vomiting and possibly symptoms related to gastrointestinal irritation.9.‘o The histologic hallmark of an actinomycosis lesion is the granuloma. In the center of the granuloma there is abscess formation, within which are the characteristic colonies of microorganisms.g,20 Histologically, an isolated microbial colony may appear as a central mass of intertwining filaments, with radiating peripheral filaments producing a clublike appearance.9 Actinomyces are rather delicate organisms that are often difficult to culture. For this reason, positive cultures are not always found with actinomycosis infections, especially if the organisms are exposed to air for prolonged periods prior to culturing. Microscopic evaluation of the discharge from the abscesses frequently show organisms that appear crystalline yellow in color and stain positively for periodic acidSchiff.7,9.‘0 These yellow grains, called “sulfur granules,” are pathognomonic for the disease. Since sulfur granules are not always present, the diagnosis of actinomycosis is also dependent on the clinical presenta-

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tion and demonstration of the organisms in culture or tissue biopsy. Brown and Brenn stain, used to differentiate between gram-positive and gram-negative bacteria in tissue sections, 2’ also can be used as an additional diagnostic tool to demonstrate actinomyces in tissue sections. 22 This may be beneficial in situations where there is a strong clinical suspicion of the disease despite negative culture results and negative hematoxylin and eosin-stained histologic sections, which is what occurred in this patient. Fortunately, the Brown and Brenn-stained tissue sections showed histologic evidence of actiaomyces. Actinomyces infections produce a reactive inflammatory response, which causes an area of necrosis and scar tissue around the abscess. This results in a decrease in the vascular supply to the affected region and makes penetration of antibiotics difficult. Consequently, twofold therapy, including antibiotics and surgery, is necessary. After the diagnosis has been made, the lesion should be surgically removed and the surrounding area thoroughly debrided.‘0,23 Following surgical intervention, aggressive antibiotic therapy should be continued. The antibiotic of choice is penicillin G, which should be given 3 to 4 million units intravenously every 4 hours for 2 to 4 weeks. This should be followed by 0.5 to 1 g of penicillin V four times a day for 4 to 6 more weeks.23-27In more serious cases of cervicofacial infections, some authors recommend the initiation of intravenous penicillin G, 5 to 20 million units daily, for 5 to 6 weeks.23-27In patients allergic to penicillin, tetracycline orally 500 mg four times a day for 8 to 16 weeks, or erythromycin orally 500 mg given four times per day for 6 months, is reported to be effective.” In this case, the initial response to antibiotic therapy alone was poor. Not until surgical intervention followed by high doses of intravenous antibiotics, was there complete resolution of the submandibular mass. Since actinomyces may be difficult to isolate, and the infection can mimic a neoplasm or common infectious processes, there is frequently a prolonged course using inadequate antibiotic regimens, with multiple failed attempts at a cure. This case, as well as many other reports in the literature, illustrates the common diagnostic delay with this disease. It also shows that actinomyces, a common and normal inhabitant of the oral cavity, can be seeded through surgical incisions used in various intraoral procedures. It is possible that routine, preoperative intraoral preparation, in conjunction with postoperative antibiotics, is the reason for the rarity and subsequently the relatively few reported cases of facial actinomycosis. However, perhaps the difficulty in making a positive microbiologic or microscopic diagnosis suggests that actinomycosis may be a more common infection than is generally believed.

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