Legal Medicine 14 (2012) 324–327

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Legal Medicine

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Case Report

Carotid artery rupture and cervicofacial actinomycosis

Anne Kummer a,1, Benoît Lhermitte b, Micaela Ödman c, Silke Grabherr a,Patrice Mangin a, Cristian Palmiere a,⇑,1

a University Center of Legal Medicine, Lausanne-Geneva, Rue du Bugnon 21, 1011 Lausanne, Switzerlandb University Institute of Pathology, Centre Hospitalier Universitaire Vaudois, University Hospital, 1011 Lausanne, Switzerlandc Department of Otorhinolaryngology and Cervico-Facial Surgery, Centre Hospitalier Universitaire Vaudois, University Hospital, 1011 Lausanne, Switzerland

a r t i c l e i n f o a b s t r a c t

Article history:Received 13 April 2012Received in revised form 11 June 2012Accepted 14 June 2012Available online 20 July 2012

Keywords:Cervicofacial actinomycosisHemorrhageCarotid artery rupturePostmortem angiographyPostmortem biochemistry

1344-6223/$ - see front matter � 2012 Elsevier Irelanhttp://dx.doi.org/10.1016/j.legalmed.2012.06.002

⇑ Corresponding author. Tel.: +41 021 314 49 61; fE-mail address: cristian.palmiere@chuv.ch (C. Palm

1 These authors are equally contributed as first auth

Cervicofacial actinomycosis is an uncommon, progressive infection caused by bacilli of the Actinomycesgenus. Actinomyces are common commensal saprophytes in the oral cavity which may have medicalimportance as facultative pathogens. Subsequent to local injuries to the oral mucosa, they may penetratethe deep tissues and be responsible for suppurative or granulomatous infections. We herein report a caseof a 65-year-old man who underwent surgery followed by chemotherapy and radiotherapy for a tonsillarcarcinoma. An ulcerous lesion in the base of the tongue developed and spread to the carotid artery wall.The man died of a massive hemorrhage due to left carotid artery rupture. Postmortem computed tomog-raphy angiography performed prior to autopsy allowed the precise localization of the source of bleedingto be detected. Postmortem biochemical investigations confirmed the presence of inflammation associ-ated with local bacterial infection. Histological investigations revealed the rupture of the left carotidartery surrounded by numerous colonies of Actinomyces. Acute and chronic inflammation with tissuenecrosis as well as post-actinic, fibrotic changes were also found in the tissues surrounding the rupturedartery wall.

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1. Introduction

Actinomycosis is an uncommon, though well-documented,suppurative, granulomatous infection of the upper airway andgastrointestinal tract, caused by Gram-positive bacteria of thegenus Actinomyces. The specific etiologic agents include Actinomy-ces israelii, Actinomyces propionica and Actinomyces naeslundii,which may also be part of normal healthy oral flora [1]. Clinicalinfections are relatively rare [2]. Actinomycetic infections involv-ing the cervico-facial region have been described in patients trea-ted for tonsillar, oropharyngeal and laryngeal carcinomas ascomplications of major head and neck oncolologic surgical proce-dures and radiotherapy [1,3–7]. In these cases, cervical actinomy-cosis is believed to occur as the result of a break in the integrityof the mucosa that allows Actinomyces to infect deeper tissues.Radiotherapy of the head and neck may also cause a breakdownof the protective mucosal barriers, facilitating Actinomyces inva-sion [3]. Lastly, debilitating systemic diseases may facilitate thedevelopment of the infection [8].

Friedman et al. [6] reported the unique case of a post-operativecarotid artery rupture caused by an actinomycetic infection in a

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ax: +41 021 341 70 90.iere).

ors to this work.

case of an oropharyngeal carcinoma treated with surgical resectionand adjuvant radiation therapy. The infection caused right carotidartery wall necrosis and the patient suffered from an arterial hem-orrhage into the oropharynx as well as through a pharyngocutane-ous fistula orifice.

In this article, we describe the case of a fatal carotid rupturelikely caused by an Actinomycetic infection in a patient who suf-fered from tonsillar carcinoma. The patient was treated with che-motherapy and radiation therapy and developed an ulcerouslesion of the soft tissues surrounding the carcinoma site. Despiteulcer treatment with a fascio-cutaneous flap, the patient died se-ven weeks post-surgery of a massive hemorrhage caused by leftcarotid artery wall rupture. Postmortem angiography and autopsyshowed the rupture of the arterial wall and allowed the exactsource of the bleeding to be detected. Histology showed the pres-ence of numerous colonies of Actinomyces as well as acute andchronic inflammation with necrosis in the tissues surroundingthe ruptured artery.

2. Case report

A 65-year-old man was admitted to the hospital with a left ton-sillar squamous cell carcinoma (T2, N1, M0), which was treatedwith chemotherapy (cisplatin) and radiotherapy between Januaryand March 2011. Three months after terminating these treatments

A. Kummer et al. / Legal Medicine 14 (2012) 324–327 325

(July), a magnetic resonance imaging (MRI) revealed the presenceof a persisting, ulcerative lesion of the base of the tongue and inthe tonsillar region, which was suspected as a persisting malignantdisease. Though biopsies proved negative for carcinomatous cells,the patient was re-admitted to the hospital for the surgical exci-sion of the lesion in August. A transmandibular excision of the baseof the tongue and reconstruction via a micro-anastomosed fascio-cutaneous forearm flap was performed. Tissue samples originatingfrom the excised lesion were histologically analyzed to confirm orexclude the existence of a residual carcinoma. Histopathologicalexamination revealed the presence of an ulcerous lesion with mul-tiple Actinomyces colonies and post-actinic changes. No signs ofmalignancy were detected.

The patient developed severe trismus immediately after thesurgical procedure, unsuccessfully treated with physical therapy.Additionally, a flap dehiscence was visualized via endoscopy. Thedehiscence was subsequently sutured and the patient remainedclinically stable. A new dehiscence of the flap was observed oneweek later (28 days post-surgery), which was again sutured. Fiveweeks post-surgery (35 days) the fascio-cutaneous flap had appar-ently integrated into the surrounding tissues with no evidence ofdehiscence or presence of orocervical fistula. The patient remainedclinically stable over the next two weeks and was able to start apartial, liquid diet.

Suddenly, seven weeks post-surgery and while still in hospital,the patient presented a massive, endobuccal hemorrhage originat-ing from the left tonsillar region. The massive bleeding could notproperly be controlled due to the trismus and the patient rapidlylost consciousness, dying minutes thereafter despite resuscitationattempts.

A medico-legal autopsy was requested by the public prosecutorsince the death occurred in the hospital and a correlation betweenthe surgical procedure, hemorrhage and fatal outcome could not beformally excluded.

A postmortem computed tomography angiography was per-formed prior to autopsy and revealed the presence of a ruptureof the left carotid artery located at the carotid bifurcation(Fig. 1). The precise localization of the source of bleeding wastherefore identified.

External examination was unremarkable. Internal examinationshowed the presence of large amounts of blood in the upper andlower respiratory tracts, a left carotid artery wall rupture with acommunication between the lumen of the artery and the left

Fig. 1. Visualisation of the hemorrhage originating from the left carotid artery after the ain the anterior wall of the left carotid artery. (b) Coronal Maximum Intensity Projection rethe extravasation of contrast agent coming from the left carotid artery next to the bifurcaboth carotid arteries and the extravasation of contrast agent indicating the source of t(whithe arrow) due to the presence of a lesion located proximally.

tonsillar region, a hemorrhagic infiltration of the soft tissues sur-rounding the carotid artery and an ulcerous lesion in the left ton-sillar region that involved the left carotid artery (Figs. 2a and b).

Histological examinations confirmed the presence of necrosis inthe tonsillar region extending to the soft tissues surrounding theleft carotid artery wall. The presence of post-actinic, fibroticchanges in the tonsillar and submucosal tissues, especially involv-ing muscle tissue, was obvious as well as acute and chronic inflam-mation. Numerous colonies of Actinomyces were noted in thetonsillar (Fig. 3) and submucosal regions as well as in the tissuessurrounding the left carotid artery. Lastly, histology showed ahemorrhagic infiltration of the left cervical tissues with massiveaspiration of blood in the lower respiratory tract.

Postmortem biochemical results were consistent with thehypothesis of inflammation and local bacterial infection (increasedC-reactive protein, procalcitonin and lipopolysaccharide bindingprotein concentrations in postmortem serum). Other biochemicalparameters tested in postmortem serum, vitreous and pericardialfluid were normal.

Based on the clinical information as well as postmortem inves-tigation results, the cause of death was determined to be hemor-rhagic shock and massive aspiration of blood into the airwaysdue to the left carotid artery rupture. The ulcerous lesion in the left,deep tonsillar region extending to the carotid wall was consideredthe cause of the artery rupture.

Although three factors were considered as possible explana-tions for the rupture of the left carotid artery (the presence ofthe actinomycetic infection in the deep cervical tissues; the post-actinic changes in the tonsillar and surrounding regions; thechemical action of saliva, which had likely infiltrated into the soft,cervical tissues at the time of the dehiscences of the fascio-cutane-ous flap), the actinomycetic infection was estimated to be the mainfactor leading to the vascular rupture. Death was not consideredthe result of medical malpractice and the case was not pursuedfurther by the public prosecutor.

3. Discussion

Actinomycosis is a rare, chronic infection caused by filamen-tous, Gram-positive bacteria from the Actinomycetaceae family(genus Actinomyces). [9]. It progresses slowly and is often misdiag-nosed due to its ability to mimic other clinical conditions, includ-ing malignancy [10]. Actinomyces are commensal of the human

rterial phase of angiography. (a) Axial image showing the source of bleeding (arrow)construction showing both carotid arteries at the level of the bifurcation (stars) and

tion (arrow). (c) 3D-Volume Rendering reconstruction showing the aortic arch (star),he hemorrhage (yellow arrow). The internal carotid artery is only sparly perfused

Fig. 2a. Image showing the left carotid artery (A) and the communication (B)between the lumen of the artery and the left tonsillar region (C). The probe (yellowarrow) passes through the ulcerous lesion in the left tonsillar region.

Fig. 2b. Image showing the fascio-cutaneous flap (D), the base of the tongue (E) andthe ulcerous lesion (F) in the left tonsillar region.

Fig. 3. Gram staining (original magnification 40�). Deep tonsillar tissues: filamen-tous bacteria embedded in a highly necrotic area.

326 A. Kummer et al. / Legal Medicine 14 (2012) 324–327

oropharynx as well as the gastrointestinal and urogenital tracts[11]. Since these microorganisms are not virulent, mucosal disrup-tions are needed to lead to infection, which in turn appear malig-nant-like due to their capacity to invade surrounding tissues andform masses. Actinomycosis is therefore, mainly, an endogenousinfection [10]. Debilitating states such as diabetes, malignancyand immunosuppression are predisposing factors [8,10].

Actinomycosis is classified into distinct clinical forms accordingto the anatomical sites which are infected: orocervicofacial, tho-racic, abdominopelvic, central nervous system, musculoskeletal,and disseminated. A. israelii is the most common human pathogenand is found in most clinical presentations, though certain specieshave been linked to particular clinical syndromes [11]. Cervicofa-cial actinomycosis is the most common form of the disease andmakes up about 50% of all reported cases [12]. Actinomycotic le-sions are usually described as single or multiple abscesses or indu-rated masses with hard fibrous walls and soft, central loculationscontaining white or yellow pus, usually accompanied by slow-increasing inflammatory swelling at the inoculation site [13,14].The lesions are typically painless and hard, thereby mimicing amalignant tumor [15–17]. Regional lymphadenopathy is typicallyabsent until later stages [18]. Cervicofacial actinomycosis often in-volves the mouth and its immediate lymphatic drainage areas,with submandibular and submental lesions or lower in the anteriortriangle [19,20]. Clinical presentations are usually abscessed orindurated masses, though ulcerous lesions of the oral mucosa havealso been reported [14]. The tonsilla, pharynx and larynx are fre-quently involved [21–23]. Actinomycosis can also affect the hardand soft palate, tongue, lips, hypopharynx and gingival, thoughmore rarely [24,25].

Cervicofacial actinomycosis have been described in previouspublications in association with laryngeal [1,2,4,5,7] and pharyn-geal [3,6] carcinomas. In most cases, these have been treated withsurgery and radiation therapy. Numerous authors have also re-ported osteoradionecrotic lesions following radiation therapy forhead and neck surgery, which have subsequently been infectedby Actinomyces [26].

Friedman et al. [6] reported a unique case of a post-operativecarotid artery rupture caused by an actinomycetic infection atthe site of an oropharyngeal carcinoma. The patient had been trea-ted with surgical resection and adjuvant radiation therapy andsubsequently developed right carotid artery wall necrosis, causinga hemorrhage into the oropharynx and through a pharyngocutane-ous fistula orifice near the carotid bifurcation. Surgical explorationrevealed a carotid artery rupture located at a necrotic area just be-low the carotid bifurcation.

In the case herein presented, several parallelisms can be identi-fied with the case described by Friedman et al. [6]. Indeed, we alsoobserved a rupture of the carotid artery wall in a patient who hadsuffered from an oropharyngeal carcinoma and had previouslybeen treated with chemotherapy and radiation therapy. Neverthe-less, there have been no cases reported in medico-legal literaturethus far in which the rupture of the carotid artery wall was linkedto a cervico-facial actinomycetic infection. Additionally, the sourceof bleeding and presence of a communication between the oropha-ryngeal region and the carotid artery lumen was precisely detectedby postmortem angiography and subsequently confirmed by theautopsy. Histology showed the inflammation and necrosis of theoropharynx, pericarotid soft tissues and carotid wall as well asthe post-actinic changes of the deep tonsillar tissues. It also evi-denced the presence of Actinomyces in the submucosal and peri-carotid tissues. Lastly, postmortem biochemical investigationsresults were consistent with the hypothesis of a local bacterialinfection with inflammation.

The clinical presentation of this case suggested an actinomyco-sis, with a history of a non-healing ulcerative lesion mimicking acarcinoma recurrence. The evolution was relatively asymptomaticwith the exception of severe trismus. The infiltration of Actinomy-ces was likely facilitated by the mucosal breaches following surgi-cal procedure and radiation therapy.

Radiation-induced lesions are well described in the literatureand, as in the case described by Friedman et al. [6], radiationtherapy may have been a contributing factor to the carotid artery

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rupture. In fact, radiation can damage the vasa vasorum of the lar-ger arteries, weakening the medial layer and predisposing them torupture [3]. Additionally, radiation therapy can cause vascularthrombosis, resulting in the obliteration of small blood vesselswith successive tissue hypoxia, creating a favorable environmentin which anaerobic organisms, such as Actinomyces species, cangrow [4].

Lastly, the chemical action of the saliva which had likely infil-trated into the soft, cervical tissues at the time of the dehiscenceof the fascio-cutaneous flap, was also considered a factor thatmay have caused or contributed to the weakening of the arterialwall as a consequence of enzymatic digestion [27].

Although three factors were considered as possible explanationsfor the rupture of the left carotid artery, the actinomycetic infectionwas estimated to be the main factor leading to the vascular rupture.

The case herein reported emphasizes the usefulness of perform-ing postmortem radiological investigations, especially postmortemangiography, in order to detect bleeding sources with precision.This is paramount in cases of acute hemorrhage with fatal out-comes, when the affected vessels are of small caliber or are locatedin anatomical areas of the body difficult to access [28,29]. Indeed,the detection and precise identification of the vessels responsiblefor bleeding are of utmost importance in the practice of forensicpathology, especially in situations in which professional liabilityin medical malpractice may be implied.

Cervicofacial actinomycosis in forensic pathology is aninfrequent finding and clinical presentations with carotid arteryruptures are unusual. Nevertheless, this case highlights the use-fulness of a multi-disciplinary approach to improve the qualityof the forensic autopsy and render its results more complete.Not only was the detection of the vessel causing the bleedingessential in this case, but also the contributing conditions andpredisposing disorders. All the data from the medico-legal au-topsy allowed the physiopathology of the entire process to beunderstood and thus for the necessary information to be providedto the judicial authorities.

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