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Treatment of Cervicofacial Type of Actinomycosis in a Patient with Allergy to Penicillin – Case Report

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clinical case

szymon Frank

1, a, e

, Paweł nieckula

1, B, D

, Monika Jodko

2, e, F

Treatment of Cervicofacial Type of Actinomycosis

in a Patient with Allergy to Penicillin – Case Report

Leczenie pacjentki uczulonej na penicylinę

chorej na szyjno-twarzową postać promienicy – opis przypadku

¹ Department of Oral surgery, Medical University of Warsaw, Poland

² student scientific circle of the Department of Oral surgery, Medical University of Warsaw, Poland

A – koncepcja i projekt badania; B – gromadzenie i/lub zestawianie danych; C – opracowanie statystyczne; D – interpretacja danych; E – przygotowanie tekstu; F – zebranie piśmiennictwa

Abstract

actinomycosis is an infectious disease caused by the gram positive anaerobic bacteria from the actinomyces spe-cies. infection most often develops following tooth extraction or as a result of an injury in the face or neck region. The aim of the study is to present the case of a female patient allergic to penicillin who was clinically treated for actinomycosis. a 50-year-old female patient presented at the Department of Oral surgery of the Medical University of Warsaw for extraction of the roots of the 36 tooth with gangrenous pulp. Medical history revealed an allergy to lignocaine, penicillin and metronidazole. The patient had cervicofacial actinomycosis. The suggested algorithm of treatment in the patient’s case produced the desired effect and resulted in recovery. surgical treatment of the lesion and the administration of doxycycline led to effective healing of the patient (Dent. Med. Probl. 2012, 49,

4, 595–599).

Key words: Actinomycosis, cervicofacial, Actinomyces.

Streszczenie

Promienica jest chorobą zakaźną wywołaną przez Gram-dodatnie bakterie beztlenowe z rodzaju Actinomyces. Zakażenie rozwija się najczęściej po usunięciu zęba lub w następstwie urazu twarzy lub szyi. celem pracy jest przed-stawienie przypadku klinicznego pacjentki z alergią na penicylinę leczonej z powodu promienicy. 50-letnia kobieta zgłosiła się do Zakładu chirurgii stomatologicznej WUM w celu ekstrakcji korzeni zęba 36 z miazgą w stanie zgorzelinowego rozpadu. W wywiadzie ustalono alergię na lignokainę, penicylinę oraz metronidazol. stwierdzono u niej szyjno-twarzową postać promienicy. Zaproponowane postępowanie w tym przypadku przyniosło pożądany rezultat i pacjentka wyzdrowiała. leczenie chirurgiczne zmiany oraz podanie doksycykliny pozwoliło ją całkowicie wyleczyć (Dent. Med. Probl. 2012, 49, 4, 595–599).

Słowa kluczowe: promienica, szyjno-twarzowa, leczenie chirurgiczne i antybiotykowe.

Dent. Med. Probl. 2012, 49, 4, 595–599

issn 1644-387X © copyright by Wroclaw Medical University and Polish Dental society

actinomycosis is a disease caused by Gram- -positive anaerobic or microaerophilic bacteria of the genus actinomyces. as a clinical entity, it was described more than 100 years ago [1, 2]. actino-mycosis is a rare human infectious disease, much more common in animals (pigs, horses, cattle and dogs). The most commonly isolated species of ac-tinomycetes are: Actinomyces israeli, A. meyeri,

A. bovis, A. naeslundii, A. viscous and A. odontoli-tycus [2, 3]. These microorganisms are widespread

in nature. as saprophytes, they dwell in the hu-man body, in the mouth especially in the interden-tal spaces, gingival pockets, airways and gastroin-testinal tract [1, 2].

actinomycosis occurs three times more often in men than in women. This disease usually

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af-fects the age group of 20–50-year-olds. The most common variant form of actinomycosis is the cer-vicofacial form, which makes up approximately 50–70% of cases [4].

actinomycetes may become pathogenic as a result of interruption of the barrier of the skin or mucosa. The infection usually develops after tooth extraction or due to injury (chemical, thermal or mechanical) of the facial or neck region. actino-mycosis is spread most commonly by continuity. The immunity and virulence of oral microorgan-isms which are in synergism with actinomycetes affect the course of the specific inflammatory pro-cess. it should be remembered that actinomycosis infection is nearly always combined. Untreated or inadequately treated disease results in significant tissue destruction.

among the culture media predisposed to ac-tinomycosis development, periapical granulation tissue of the teeth with gangrenous pulp, inflamed gingival pockets, chronic purulent tonsillitis, in-flammation of the bone, jaw fractures and infected wounds of the oral mucosa may be mentioned.

characteristic symptoms of actinomycosis are swelling with associated redness and the presence of solid tumors with purulent lesions. in the lat-er stage of the disease those lesions create fistulae excreting druses – light yellow grains with a di-ameter of 0.1–0.5 mm (Fig. 1) [ 5]. The name of the species comes specifically from the presence of characteristic rays in these grains. Then begins granulation tissue formation, which eventually be-comes fibrosis, resulting in the formation of scar tissue.

Case Report

a 50-year-old woman in good general condi-tion reported to the Department of Oral surgery of the Medical University of Warsaw to extract the roots of the 36 tooth (Fig. 2). The woman was gen-erally healthy. allergy to lignocaine, metronida-zole, and penicillin was recorded in an interview. after the use of these drugs, the patient reported maculopapular rash and bronchospasms causing labored breathing.

in the medical history, the patient reported pain occurring around the 36 tooth. The pain had lasted 2 days and increased under pressure on the 36 tooth. in clinical examination in the vestibule of the mouth, palpation tenderness around the 36 tooth was found. Further study found pain on percussion of the 36 tooth in the horizontal and vertical plane, absence of mucosal redness and negative fluctuate symptoms in the vestibule of the mouth around the 36 tooth.

submandibu-lar lymph nodes on the left side were ensubmandibu-larged, though smooth, painless and movable in relation to ground and skin. On the right side they were impalpable. The patient was referred for pantomo-graphic imaging. On the basis of radiological and clinical research, a large carious cavity in the 36 tooth, a trace of filler material in the distal canal of this tooth and periapical change of an osteolysis character around the media root were concluded. in tooth 37 mesial-occlusal secondary caries were found. Tooth 38 had a crown destroyed by caries and pulp in gangrenous disintegration.

Due to the patient’s allergy to lignocaine, an intradermal allergy test to marcaine was per-formed. The reading after 30 minutes and 24 hours was negative, which determined the choice of mar-caine as an anesthetic substance. Under local an-esthesia, extraction of the roots of 36 tooth was

Fig. 1. extraoral fistula excreting purulent content with

the contents of characteristic “actinomycete grains”

Ryc. 1. Przetoka zewnątrzustna z wydzielającą się

tre-ścią ropną, z zawartotre-ścią charakterystycznych „ziaren promieniczych”

Fig. 2. 36 tooth, qualified for extraction Ryc. 2. Ząb 36 – zakwalifikowany do ekstrakcji

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carried out. after that, the tissue granulation was removed. Tooth 38 was planned to be removed in the next stage of treatment.

after a period of ten days after extraction, the patient reported again with swelling of the cheek on the left side, which was painful to the touch (Fig. 3). according to the patient, swelling start-ed to slowly grow three days after the extraction of the roots of the 36 tooth. clinical examination revealed redness and hard, warm swelling of the skin of the left cheek. The patient denied increased body temperature from the date of extraction. The exaggerated cheek was punctured extraorally and a scanty purulent content was obtained. The re-sulting fluid was sent for histopathological exam-ination in the direction of actinomycosis (Fig. 4). subsequently under local anesthesia of 0.5% Mar-caine, the skin coat on the top of the swelling was incised. The cavity of the cheek abscess was

opened and drained. a drain and extraoral ban-dage was established. The next day, hard swelling of the cheek continued without evidence of clini-cal improvement. The roots of the 38 tooth were removed under local anesthesia. Full sanation of the mouth was finished. On the same visit, the ab-scess cavity was flushed with saline. scanty blood- -purulent content was removed. a drain and ban-dage was established.

The patient was referred to an allergist to per-form allergy tests for penicillin, cephalosporin, clindamycin, clarithromycin, erythromycin and metronidazole. Due to persistent serous-purulent effusion from the abscess cavity, until histopathol-ogy results were received, treatment by lavage of the abscess cavity with saline and casting was con-tinued daily for 10 days. Results of the allergy tests to antibiotics proved to be positive for all inves-tigated. Histopathological examination revealed actinomycosis – an infection of A. israeli. Due to the continued left cheek swelling and redness of the skin of this area, the exaggerated are had been repeatedly extraorally incised and cleaned.

The patient was referred to the Regional Hos-pital of infectious Diseases in Warsaw in order to continue treatment. During hospitalization, which lasted seven days, the oral antibiotic doxycycline was implemented – Unidox 0.1 g, 1 tablet every 12 hours. antibiotic therapy lasted for 4 weeks. in addition, the patient took Trilac

(Lactobacil-lus acidophi(Lactobacil-lus, Lactobacil(Lactobacil-lus delbrueckii, Bifido-bacterium lactis), claritin, calcium, Kaldyum

(po-tassium chloride), Magnezin (magnesium carbon-icum) and Hydroxyzine. after leaving the hospital the patient signed up for a check-up visit to the Department of Oral surgery, Medical University of Warsaw. The clinical picture has improved sig-nificantly. cheek swelling subsided and soft tissue infiltration decreased. 7 days after the patient left the hospital, the intraoral fistula was closed.

The proposed algorithm in this case has brought the desired effect and led to curing the patient (Figure 5, 6).

Discussion

Rapid diagnosis and beginning effective phar-macotherapy in the case of actinomycosis is a nec-essary condition to remission of the disease and prevention of its complications. actinomycosis can spread into the tissues and penetrate into spac-es such as leaf-mandibular, leaf-palatal, parapha-ryngeal and into the orbit. Unlike other suppura-tive infections, this can spread in ways contrary to the natural anatomy. a serious complication is the spread of actinomycosis through the holes of

Fig. 3. swelling of the cheek on the left side, which

appeared 10 days after extraction of 36 tooth

Ryc. 3. Obrzmienie policzka po stronie lewej, które

pojawiło się 10 dni po ekstrakcji zęba 36

Fig. 4. Taking purulent discharge for examination Ryc. 4. Pobranie wydzieliny ropnej do badania

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the skull with the result that it can cause menin-gitis. While on the descending way it can get into the mediastinum.

correct diagnosis and treatment of actinomy-cosis has many difficulties [1, 6, 7]. The clinical picture of the disease and its course is variable and depends on many factors such as immunity, the location of the primary site of the disease and bac-terial virulence. The course of the disease depends on how fast a diagnosis is made and how effective the therapy is. The complications of

actinomyco-sis are often the result of ineffective treatment. it should be noted that the frequent recurrence of a purulent appearance around the face and neck region, occurring without a specific cause, should always be a possible indicator of actinomycosis, and it is advisable to commence the relevant tests in order to form a proper diagnosis.

The first stage of the clinical disease may be characterized by the presence of bad breath (hali-tosis) and hard tissue infiltration, from which it is difficult to obtain purulent content. a disadvan-tage in the diagnosis of actinomycosis is the need for multiple microbiological or histopathologi-cal tests when the primary test result is negative, since this does not exclude actinomycosis and the test should be repeated. Routine blood tests usu-ally show no abnormalities, and sometimes there may be leukocytosis. imaging by computed to-mography and magnetic resonance imaging can give nonspecific results [8]. all these factors con-tribute to difficulties in making an accurate diag-nosis of the present entity.

cervicofacial actinomycosis most often affects the submandibular space, cheeks, teeth, tongue, parotid, lymph nodes, peri-pharyngeal space and around the thyroid gland [2].

The treatment for actinomycosis is surgi-cal drainage of abscesses and long-term antibi-otic therapy. The result of treatment depends on the patient’s general condition, their sensitivity to drugs and the severity and duration of the dis-ease [5]. The drug of choice for actinomycosis re-mains penicillin [2, 8–12]. in severe cases, crystal-line penicillin administered intravenously for 4–6 weeks in combination with surgical drainage and then many months of treatment with oral penicil-lin [2] are recommended.

in the case discussed, because of the patient’s allergy to penicillin, research commenced in order to choose the appropriate antibiotic. Finally, be-cause of the patient’s allergy to penicillins, cepha-losporin, clindamycin, clarithromycin, erythro-mycin and metronidazole, the decision was made to start doxycycline therapy. in the case discussed, the use of doxycycline in a Unidox preparation 100 mg twice daily for four weeks led to successful treatment of the patient. The fistula was closed.

Fig. 5. status after treatment – extraoral view Ryc. 5. stan po leczeniu – widok zewnątrzustny

Fig. 6. status after treatment – intraoral view Ryc. 6. stan po leczeniu – widok wewnątrzustny

References

lancella

[1] a., abbate G., Foscolo a.M., Dosdegani R.: Two unusual presentations of cervicofacial actinomy-cosis and review of the literature. acta Otorhinolaryngol. ital. 2008, 28, 89–93.

Zyzak

[2] K., Kuchar e., Remion J., szenborn l.: actinomycosis of the neck in 16-year-old boy. Przegl. Petriatr. 2011, 41, 94–96.

carinci

[3] F., Jessica Polito J., Pastore a.: Pharyngeal actinomycosis: a case report. Gerodontology 2007, 24, 121–123.

schaal

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Kaszuba

[5] M., Tomaszewska R., Pityńska K., Grzanka P., Bazan-socha s., Musiał J.: actinomycosis mimick-ing advanced cancer. Pol. arch. Med. Wewn. 2008, 118(10), 581–584.

Rzeszutko

[6] M., Jabłecki J.: actinomycosis as a cause of Hepatic abscess – case Report and Review of literature. adv. clin. exp. Med. 2004, 13, 865–867.

Ziora

[7] K., Geisler G., Oświęcimska J., Zajęcki W., legaszewski T., Kluczewska e., Pikiewicz-Koch a., Dyduch a.: acute suppurative Tyroiditis in the course of cervicofacial actinomycosis – the case Report. endokrynol. Pediatr. 2007, 6, 2(19).

Volante

[8] M., contucci a.M., Fantoni M., Ricci R., Galli J.: cervicofacial actinomycosis: still a difficult dif-ferential diagnosis. acta Othorinolaryngol. ital. 25, 2005, 116–119.

Belmont

[9] M.J., Behar P.M., Wax M.K.: atypical presentations of actinomycosis. Head neck 1999, 264–268. Vesely

[10] B.T., Hyza P., Koncena J., Kuklinek i., Kozak J., Ranno R.: Unusual case of resistant actinomycosis following facial trauma. acta chir. Plast. 2005, 47, 119–123.

Kolebacz

[11] B., stryjewska-Makuch G., Grzegorzek T.: cervicofacial actinomycosis – case reports. Otolaryngol. Pol. 2004, 58, 1019–1022.

aguirrebengoa

[12] l., Romana M., lopez l., Martin J., Montejo M., Gonzales De Zarate P.: Oral and cervi-cofacial actinomycosis. Presentation of five cases. enferm. infecc. Microbiol. clin. 2002, 20, 53–56.

Address for correspondence:

szymon Frank

Department of Oral surgery WUM nowogrodzka 59 02-006 Warszawa Poland Tel.: +48 22 502 12 42 e-mail: szymon_frank@tlen.pl Received: 3.02.2012 Revised: 27.08.2012 accepted: 25.10.2012

Praca wpłynęła do Redakcji: 3.02.2012 r. Po recenzji: 27.08.2012 r.

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