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Cervicofacial actinomycosis
Cervicofacial Actinomycosis Stuart W. Leafstedt, MD, Sioux City, Iowa Robert M. Gleeson, MD, Sioux City, Iowa
Actinomycosis is an infectious disease that can present as a pseudotumor mass or cold abscess in the cervicofacial region. This uncommon disease involves the face, neck, and ororespiratory tract and may involve the pulmonary and gastrointestinal tract as well. The variable clinical picture and infrequency of the disease makes diagnosis difficult. Increased alertness of clinicians and microbiologists to the presence of anaerobic organisms as the cause of infection should result in earlier and more frequent diagnosis of actinomycosis. Clinical and Bacteriologic Features Cervicofacial actinomycosis presents as an indurated swelling or pseudotumor mass located near or on the mandible. Actinomycotic infections were first described nearly 100 years ago from the suppurative exudate from lesions of cattle having the disease known commonly to veterinarians as “lumpy jaw.” The organism was given its name Actinomycosis bovis (literally, “ray-fungus of the cow”) by Harz, a botanist. The filamentous organisms had been observed in the granules from the exudate. A similar disease in humans was recognized at about the same time. In 1885, Israel reported in detail thirty-eight patients with cervicofacial actinomycosis. The organism identified in human infections was later named A israelii in his honor. For decades the organism from cattle and the organism from humans were named strictly on the basis of its origin. Studies in the past twenty years confirm that the two species differ. A bovis has not been identified in human infections. A israelii is the most common in humans, but other species can be identified as well: Arachnia propionica, A naeslundii, A viscosus, and A odotolyticus. From the Department of Surgery. St. Luke’s Medical Center. Sioux City, Iowa. Reprint requests should be addressed to Stuart W. Leafstedt, MD, Surgical Consultants, PC, 2919 Hamilton Boulevard, Sioux Cii. Iowa 51104. Presented at the Combined Meeting of the James Ewing Society and the Society of Head and Neck Surgeons, New Orleans, Louisiana, March 25-29.1975.
Actinomyces lies in the intermeTaxonomically, diate subdivision between true fungus and true bacteria. It is a gram-positive, branching, filamentous microorganism related more to true bacteria than to fungus. It is anaerobic to microaerophilic in nature, does not produce spores, and is not acidfast. The mycelia are delicate and easily fragmented in transfer from abscess to slides for Gram stain elevation. Careful microbiologic evaluation is needed to differentiate the organism from Nocardia asteroides and anaerobic diphtheroid bacteria. Actinomyces is worldwide in distribution and can be found as saprophyte within the oral cavity and mucous membranes. It has been cultured from carious teeth, abscessed teeth, tartar, tonsils, and the pulmonary and gastrointestinal tracts. The organisms gain entrance to a microaerophilic or anaerobic atmosphere by penetrating trauma of the soft tissue of the ororespiratory tract. A history of dental procedure often antedates infection but is not always present. Poor oral hygiene, saprophytic endogenous growth of Actinomyces, and minor endoral trauma are the major factors in development of this infection. The infection spreads by burrowing, without regard to fascial planes and infrequently to lymph nodes separated from the primary infection site. Contiguous lymph nodes may become wholly or partially involved with the infection. The abscesses may often involve both bone and soft tissue, producing firm, irregular masses. The overlying skin may become fixed to the mass, and secondary venous congestion results in a reddened or violaceous color. The mass may rupture spontaneously, producing a draining sinus or fistula. Reports from the first five decades of this century indicate this was common. Most recent reports suggest a differential diagnosis of pseudotumor or cold abscess when the inflammation is seen at an earlier stage. Pain is not a prominent feature but may occur when the infection is adjacent to or within a mobile structure of the ororespiratory
The AmericanJournald Surgery
Cervicofacial Actinomycosis
such as the tongue. Primary infections have been reported in lacrimal gland, orbit, larynx, major salivary glands, hypopharynx, tongue, and paranasal sinuses. Central necrosis of the devascularized abscess leads to formation of conglomerate masses of organisms within a yellowish gray fluid that are clinically apparent as “sulfur granules.” Diagnosis of an actinomycotic infection can be made on this basis alone. To establish a diagnosis of actinomycosis the surgeon must suspect the disease and handle the tissue accordingly. Competent bacteriologists, informed of the suspicion, should perform aerobic, anaerobic, and fungal cultures and appropriate stains. The strictly anaerobic character of the organism requires rapid transport of the abscess material to the laboratory for smears and cultures in an oxygen deprived media. Actinomyces is a slow growing organism and may not be identifiably present for as long as fourteen days in anaerobic culture. Culture plates must be maintained for a minimum of two to three weeks before discarding. Biopsy of the abscess wall often identifies sulfur granules in the chronic inflammatory bed, and methanamine silver stains demonstrate the presence of mycelia. Serologic screening may identify Actinomyces prior to recovery of the organism in culture and may be helpful in the future in assessing the clinical response of the patient to treatment. Secondarily infectious organisms are often present in an abscess and may include any of the pathogenic or saprophytic organisms of the oral cavity [I-4]. tract,
Case Reports
Case I. A five and a half year old boy presented in June 1972 with a 5 by 3 cm mass within his right buccal cheek pad of eighteen months’ duration. Needle aspiration and open biopsy revealed a devascularized firm abscess cavity containing sulfur granules with a typical histologic picture. Intravenous penicillin (20,000,OOO units per day) was given for five days and continued orally at 2,000,OOO units per day for three months with complete resolution of the mass. Normal tissue appeared at examination eighteen months later. Case II. An eighteen year old white female presented in June 1973 with a two month history of a gradually increasing nontender mass in the left upper neck below the body of the mandible. One week prior to admission erythema occurred in overlying dermal tissues without temperature elevation. This 4 by 2 cm mass was opened under local anesthesia and sulfur granules were present.
voluma 130. octobw 1975
Immediate transfer of the material to anaerobic culture media yielded A israelii. Treatment consisted of open drainage and packing, intravenous penicillin at dosage of 40,000,OOO units for seven days, and subsequent oral penicillin in decreasing doses from 20,000,OOO units (penicillin G) per day for one month to 5,000,OOO units (penicillin G) per day for four months. The inflammation resolved with no recurrence eighteen months later. Case III. A sixty-two year old white male had four teeth extracted in October 1972. Six weeks later a mass in the right submandibular region developed that did not respond to broad spectrum antibiotics. Panorex mandible radiographs demonstrated rarification and lytic areas in the midportion of the body of the right mandible consistent with tumor or inflammation. A 4 by 3 cm mass contiguous with the submaxillary gland was present with adjacent, lymphadenopathy. Open biopsy of the mass preparatory to possible excision of tumor was accomplished and revealed chronic inflammatory disease. Needle aspiration of the necrotic mandible revealed gram-positive rods consistent with actinomycosis and A israelii grew in culture. Aqueous penicillin, 30,000,OOO units intravenously per day for seven days followed by 16,000,OOO units per day of penicillin G for three months and 10,000,000 units per day for the last three months resulted in complete resolution of the infection of soft tissue and bone. No disease was present at one year. Case IV. A thirty-five year old woman was treated by her private dentist for an inflammation in her left submandibular region for eight weeks with clindamycin (150 mg, four times per day). This inflammation resolved, but another inflammatory area in the submental region gradually developed in the six weeks prior to August 28, 1974; she had used the same antibiotic for six of nine weeks prior to diagnosis. A nontender central midline reddened submental mass was biopsied and cultured and A israelii was grown. Intravenous penicillin, 40,000,OOO units per day for seven days, followed by oral daily doses of 4,000,OOO to 6,000,OOO units of penicillin G for six months was successful in resolving the infection. Only minimal thickening remains at this time.
Case V. A sixteen year old white male presented with a six month history of a mass in his tongue, which previous biopsy had reported as chronic inflammation with foreign body reaction. A 3 by 4 cm tender firm mass was present in the left posterior tongue base, with tender adjacent lymph nodes. In February 1972, biopsy revealed an abscess cavity containing sulfur granules and A israelii grew in culture. Because of allergy to penicillin, treatment with tetracycline intravenously and orally was instituted. The patient’s early response was encouraging but was followed by rapid recurrence of pain and swelling when treatment was discontinued after two months. Administration of erythromycin was instituted hut discontinued because of gastrointestinal disturbance.
497
Leafstedt
and Gleeson
In May 1972, the patient entered St. Judes Children’s Research Hospital for the first time with recurrent swelling and unilateral left side headaches. He was treated with intravenous lincomycin, 600 mg every eight hours for one week, and subsequently received oral clindamycin, 450 mg, four times a day, for six months. Two weeks after discontinuing treatment he had recurrence of pain and swelling in his tongue and neck and was admitted a second time to that hospital. Skin testing confirmed his extreme allergy to penicillin. Intravenous lincomycin was reinstituted and oral clindamycin followed. A third admission one month later resulted when swelling and pain in his tongue persisted. The patient again underwent biopsy of the tongue on March 6,197s and a portion of the left middle third of the tongue was removed. Administration of clindamycin, 450 mg, four times a day, was continued for twelve months, until February 1974. Symptoms of pain in the tongue and neck and headaches recurred six months later, and after he received clindamycin, 450 mg, four times a day, for six weeks, the symptoms subsided. The patient has no sign of infection at this time, but because of the high dosage of clindamytin required for more than eighteen months, his prognosis is guarded.
Treatment The therapeutic principles of management of actinomycosis are the combination of prolonged antibiotic therapy with penicillin and appropriate surgical drainage of abscesses or excision of draining sinus tracts. The dosage of penicillin must be great if given intravenously, orally, or in combination, and the drug must be given for a prolonged period of time. In vitro sensitivity testing suggests that A israelii is sensitive to several antibiotics, including tetracycline, erythromycin, cephalothin, lincomycin, and clindamycin [6]. The responsiveness of A israelii in vivo is somewhat different. High dosage of all antibiotics has been reported effective in isolated cases [I]. Recent reports of the control of this disease with clindamycin
are most encouraging
[5].
One of the patients in the present report (case V) did respond to clindamycin but only after its administration for more than eighteen months. Active signs of the disease developed in another patient when clindamycin was administered sporadically for six of nine weeks before diagnosis (case IV); in this particular situation the dosage was probably inadequate. When infections with A israelii respond well, the dosage and duration of therapy may be tailored to the patient’s response. Perhaps the most important factor is continual maintenance of therapy for
490
a period time after
the disease is clinically controlled. Clindamycin should now be considered the first drug of choice in those patients allergic to penicillin and also as an alternate initial drug of choice. As with penicillin, the dosage of clindamycin must be adequately high and prolonged to control the disease. The patient must be carefully managed to allow recognition of the side effects of clindamytin, such as gastrointestinal toxicity. If serious problems occur, tetracycline should be substituted. Summary Cervicofacial actinomycosis is a disease that is currently encountered as a pseudotumor entity or cold abscess. The frequent use of antibiotics in treatment of masses in the cervicofacial region may either delay its appearance or disguise the diagnosis. The surgeon must be highly suspicious when making the diagnosis since precautions in handling the biopsy specimens are necessary. Treatment consists of incision biopsy and drainage or aspiration to establish the diagnosis, followed by prolonged penicillin therapy. In those patients allergic to penicillin, clindamycin should be the initial drug of choice. Either medication must be administered for a prolonged period of time, even after the disease is clinically controlled. Acknowledgment: We wish to thank Walter T. Hughes, MD, Professor of Pediatrics and Microbiology, St. Judes Children’s Research Hospital, Memphis, Tennessee, for management assistance and supplying records on one of our patients, and Bruce Woodward, BA, bacteriologist at St. Luke’s Medical Center, Sioux City, Iowa, for his cooperation and competent work in the identification of actinomycosis in all the patients presented herein. References 1. Balows A, Deliaan RM, Dowell VR. Guze LB: Anaerobic Bacteria: Role in Disease, chapt 19 and 43. Springfield, Illinois, Thomas, 1974. 2. Everts EC: Cervicofacial actlnomycosis. Arch CWarygol92: 466.1970. 3. Hervey JC, Cantreil JR, Fisher AM: Actinomycosis: its recognition and treatment. Ann intern Med 46: 666, 1957. 4. Eastridge CE, Prather RJ, Hughes FA. Young JM, McCaughan JJ: Actinomycosis: a 24 year experience. South Med J 65: 636, 1972. 5. Fass RJ, Schotand JF, Hodges GR. Saslaw S: Clindamycin in the treatment of serious anaerobic infections. Ann intern Med 76: 653.1973. 6. Mohr JA, Rhoades ER, Muchmore HG: Actinomycosis treated with lincomycin. JAMA 212: 2260, 1970.
The American Journalof Surgery
Actinomycosis is an infectious disease that can present as a pseudotumor mass or cold abscess in the cervicofacial region. This uncommon disease involves the face, neck, and ororespiratory tract and may involve the pulmonary and gastrointestinal tract as well. The variable clinical picture and infrequency of the disease makes diagnosis difficult. Increased alertness of clinicians and microbiologists to the presence of anaerobic organisms as the cause of infection should result in earlier and more frequent diagnosis of actinomycosis. Clinical and Bacteriologic Features Cervicofacial actinomycosis presents as an indurated swelling or pseudotumor mass located near or on the mandible. Actinomycotic infections were first described nearly 100 years ago from the suppurative exudate from lesions of cattle having the disease known commonly to veterinarians as “lumpy jaw.” The organism was given its name Actinomycosis bovis (literally, “ray-fungus of the cow”) by Harz, a botanist. The filamentous organisms had been observed in the granules from the exudate. A similar disease in humans was recognized at about the same time. In 1885, Israel reported in detail thirty-eight patients with cervicofacial actinomycosis. The organism identified in human infections was later named A israelii in his honor. For decades the organism from cattle and the organism from humans were named strictly on the basis of its origin. Studies in the past twenty years confirm that the two species differ. A bovis has not been identified in human infections. A israelii is the most common in humans, but other species can be identified as well: Arachnia propionica, A naeslundii, A viscosus, and A odotolyticus. From the Department of Surgery. St. Luke’s Medical Center. Sioux City, Iowa. Reprint requests should be addressed to Stuart W. Leafstedt, MD, Surgical Consultants, PC, 2919 Hamilton Boulevard, Sioux Cii. Iowa 51104. Presented at the Combined Meeting of the James Ewing Society and the Society of Head and Neck Surgeons, New Orleans, Louisiana, March 25-29.1975.
Actinomyces lies in the intermeTaxonomically, diate subdivision between true fungus and true bacteria. It is a gram-positive, branching, filamentous microorganism related more to true bacteria than to fungus. It is anaerobic to microaerophilic in nature, does not produce spores, and is not acidfast. The mycelia are delicate and easily fragmented in transfer from abscess to slides for Gram stain elevation. Careful microbiologic evaluation is needed to differentiate the organism from Nocardia asteroides and anaerobic diphtheroid bacteria. Actinomyces is worldwide in distribution and can be found as saprophyte within the oral cavity and mucous membranes. It has been cultured from carious teeth, abscessed teeth, tartar, tonsils, and the pulmonary and gastrointestinal tracts. The organisms gain entrance to a microaerophilic or anaerobic atmosphere by penetrating trauma of the soft tissue of the ororespiratory tract. A history of dental procedure often antedates infection but is not always present. Poor oral hygiene, saprophytic endogenous growth of Actinomyces, and minor endoral trauma are the major factors in development of this infection. The infection spreads by burrowing, without regard to fascial planes and infrequently to lymph nodes separated from the primary infection site. Contiguous lymph nodes may become wholly or partially involved with the infection. The abscesses may often involve both bone and soft tissue, producing firm, irregular masses. The overlying skin may become fixed to the mass, and secondary venous congestion results in a reddened or violaceous color. The mass may rupture spontaneously, producing a draining sinus or fistula. Reports from the first five decades of this century indicate this was common. Most recent reports suggest a differential diagnosis of pseudotumor or cold abscess when the inflammation is seen at an earlier stage. Pain is not a prominent feature but may occur when the infection is adjacent to or within a mobile structure of the ororespiratory
The AmericanJournald Surgery
Cervicofacial Actinomycosis
such as the tongue. Primary infections have been reported in lacrimal gland, orbit, larynx, major salivary glands, hypopharynx, tongue, and paranasal sinuses. Central necrosis of the devascularized abscess leads to formation of conglomerate masses of organisms within a yellowish gray fluid that are clinically apparent as “sulfur granules.” Diagnosis of an actinomycotic infection can be made on this basis alone. To establish a diagnosis of actinomycosis the surgeon must suspect the disease and handle the tissue accordingly. Competent bacteriologists, informed of the suspicion, should perform aerobic, anaerobic, and fungal cultures and appropriate stains. The strictly anaerobic character of the organism requires rapid transport of the abscess material to the laboratory for smears and cultures in an oxygen deprived media. Actinomyces is a slow growing organism and may not be identifiably present for as long as fourteen days in anaerobic culture. Culture plates must be maintained for a minimum of two to three weeks before discarding. Biopsy of the abscess wall often identifies sulfur granules in the chronic inflammatory bed, and methanamine silver stains demonstrate the presence of mycelia. Serologic screening may identify Actinomyces prior to recovery of the organism in culture and may be helpful in the future in assessing the clinical response of the patient to treatment. Secondarily infectious organisms are often present in an abscess and may include any of the pathogenic or saprophytic organisms of the oral cavity [I-4]. tract,
Case Reports
Case I. A five and a half year old boy presented in June 1972 with a 5 by 3 cm mass within his right buccal cheek pad of eighteen months’ duration. Needle aspiration and open biopsy revealed a devascularized firm abscess cavity containing sulfur granules with a typical histologic picture. Intravenous penicillin (20,000,OOO units per day) was given for five days and continued orally at 2,000,OOO units per day for three months with complete resolution of the mass. Normal tissue appeared at examination eighteen months later. Case II. An eighteen year old white female presented in June 1973 with a two month history of a gradually increasing nontender mass in the left upper neck below the body of the mandible. One week prior to admission erythema occurred in overlying dermal tissues without temperature elevation. This 4 by 2 cm mass was opened under local anesthesia and sulfur granules were present.
voluma 130. octobw 1975
Immediate transfer of the material to anaerobic culture media yielded A israelii. Treatment consisted of open drainage and packing, intravenous penicillin at dosage of 40,000,OOO units for seven days, and subsequent oral penicillin in decreasing doses from 20,000,OOO units (penicillin G) per day for one month to 5,000,OOO units (penicillin G) per day for four months. The inflammation resolved with no recurrence eighteen months later. Case III. A sixty-two year old white male had four teeth extracted in October 1972. Six weeks later a mass in the right submandibular region developed that did not respond to broad spectrum antibiotics. Panorex mandible radiographs demonstrated rarification and lytic areas in the midportion of the body of the right mandible consistent with tumor or inflammation. A 4 by 3 cm mass contiguous with the submaxillary gland was present with adjacent, lymphadenopathy. Open biopsy of the mass preparatory to possible excision of tumor was accomplished and revealed chronic inflammatory disease. Needle aspiration of the necrotic mandible revealed gram-positive rods consistent with actinomycosis and A israelii grew in culture. Aqueous penicillin, 30,000,OOO units intravenously per day for seven days followed by 16,000,OOO units per day of penicillin G for three months and 10,000,000 units per day for the last three months resulted in complete resolution of the infection of soft tissue and bone. No disease was present at one year. Case IV. A thirty-five year old woman was treated by her private dentist for an inflammation in her left submandibular region for eight weeks with clindamycin (150 mg, four times per day). This inflammation resolved, but another inflammatory area in the submental region gradually developed in the six weeks prior to August 28, 1974; she had used the same antibiotic for six of nine weeks prior to diagnosis. A nontender central midline reddened submental mass was biopsied and cultured and A israelii was grown. Intravenous penicillin, 40,000,OOO units per day for seven days, followed by oral daily doses of 4,000,OOO to 6,000,OOO units of penicillin G for six months was successful in resolving the infection. Only minimal thickening remains at this time.
Case V. A sixteen year old white male presented with a six month history of a mass in his tongue, which previous biopsy had reported as chronic inflammation with foreign body reaction. A 3 by 4 cm tender firm mass was present in the left posterior tongue base, with tender adjacent lymph nodes. In February 1972, biopsy revealed an abscess cavity containing sulfur granules and A israelii grew in culture. Because of allergy to penicillin, treatment with tetracycline intravenously and orally was instituted. The patient’s early response was encouraging but was followed by rapid recurrence of pain and swelling when treatment was discontinued after two months. Administration of erythromycin was instituted hut discontinued because of gastrointestinal disturbance.
497
Leafstedt
and Gleeson
In May 1972, the patient entered St. Judes Children’s Research Hospital for the first time with recurrent swelling and unilateral left side headaches. He was treated with intravenous lincomycin, 600 mg every eight hours for one week, and subsequently received oral clindamycin, 450 mg, four times a day, for six months. Two weeks after discontinuing treatment he had recurrence of pain and swelling in his tongue and neck and was admitted a second time to that hospital. Skin testing confirmed his extreme allergy to penicillin. Intravenous lincomycin was reinstituted and oral clindamycin followed. A third admission one month later resulted when swelling and pain in his tongue persisted. The patient again underwent biopsy of the tongue on March 6,197s and a portion of the left middle third of the tongue was removed. Administration of clindamycin, 450 mg, four times a day, was continued for twelve months, until February 1974. Symptoms of pain in the tongue and neck and headaches recurred six months later, and after he received clindamycin, 450 mg, four times a day, for six weeks, the symptoms subsided. The patient has no sign of infection at this time, but because of the high dosage of clindamytin required for more than eighteen months, his prognosis is guarded.
Treatment The therapeutic principles of management of actinomycosis are the combination of prolonged antibiotic therapy with penicillin and appropriate surgical drainage of abscesses or excision of draining sinus tracts. The dosage of penicillin must be great if given intravenously, orally, or in combination, and the drug must be given for a prolonged period of time. In vitro sensitivity testing suggests that A israelii is sensitive to several antibiotics, including tetracycline, erythromycin, cephalothin, lincomycin, and clindamycin [6]. The responsiveness of A israelii in vivo is somewhat different. High dosage of all antibiotics has been reported effective in isolated cases [I]. Recent reports of the control of this disease with clindamycin
are most encouraging
[5].
One of the patients in the present report (case V) did respond to clindamycin but only after its administration for more than eighteen months. Active signs of the disease developed in another patient when clindamycin was administered sporadically for six of nine weeks before diagnosis (case IV); in this particular situation the dosage was probably inadequate. When infections with A israelii respond well, the dosage and duration of therapy may be tailored to the patient’s response. Perhaps the most important factor is continual maintenance of therapy for
490
a period time after
the disease is clinically controlled. Clindamycin should now be considered the first drug of choice in those patients allergic to penicillin and also as an alternate initial drug of choice. As with penicillin, the dosage of clindamycin must be adequately high and prolonged to control the disease. The patient must be carefully managed to allow recognition of the side effects of clindamytin, such as gastrointestinal toxicity. If serious problems occur, tetracycline should be substituted. Summary Cervicofacial actinomycosis is a disease that is currently encountered as a pseudotumor entity or cold abscess. The frequent use of antibiotics in treatment of masses in the cervicofacial region may either delay its appearance or disguise the diagnosis. The surgeon must be highly suspicious when making the diagnosis since precautions in handling the biopsy specimens are necessary. Treatment consists of incision biopsy and drainage or aspiration to establish the diagnosis, followed by prolonged penicillin therapy. In those patients allergic to penicillin, clindamycin should be the initial drug of choice. Either medication must be administered for a prolonged period of time, even after the disease is clinically controlled. Acknowledgment: We wish to thank Walter T. Hughes, MD, Professor of Pediatrics and Microbiology, St. Judes Children’s Research Hospital, Memphis, Tennessee, for management assistance and supplying records on one of our patients, and Bruce Woodward, BA, bacteriologist at St. Luke’s Medical Center, Sioux City, Iowa, for his cooperation and competent work in the identification of actinomycosis in all the patients presented herein. References 1. Balows A, Deliaan RM, Dowell VR. Guze LB: Anaerobic Bacteria: Role in Disease, chapt 19 and 43. Springfield, Illinois, Thomas, 1974. 2. Everts EC: Cervicofacial actlnomycosis. Arch CWarygol92: 466.1970. 3. Hervey JC, Cantreil JR, Fisher AM: Actinomycosis: its recognition and treatment. Ann intern Med 46: 666, 1957. 4. Eastridge CE, Prather RJ, Hughes FA. Young JM, McCaughan JJ: Actinomycosis: a 24 year experience. South Med J 65: 636, 1972. 5. Fass RJ, Schotand JF, Hodges GR. Saslaw S: Clindamycin in the treatment of serious anaerobic infections. Ann intern Med 76: 653.1973. 6. Mohr JA, Rhoades ER, Muchmore HG: Actinomycosis treated with lincomycin. JAMA 212: 2260, 1970.
The American Journalof Surgery