Case Report
Abdominal actinomycosis: a rare complication after cholecystectomy X. Vanoeteren1, K. Devreese2, P. De Munter1 1
Department of General Internal Medicine, University Hospital Leuven, Belgium, 2Department of Clinical Biology and Laboratory, University Hospital Leuven, Belgium We present a single case of abdominal actinomycosis occurring in a 74-year-old female with a history of cholecystectomy 42 months before presentation. In a review of the literature, we present risk factors, clinical characteristics, diagnosis, and treatment of this infection. Abdominal actinomycosis is a rare, chronic, granulomatous infection characterized by the release of ‘sulphur granules’. Actinomyces species should always be part of the differential diagnosis of patients presenting with a history of surgical or invasive procedures, presenting with an abdominal mass. Computed tomography (CT)-guided aspiration with or without core biopsy of this mass is a useful investigation. Diagnosis is often difficult: In less than 10% of cases, the diagnosis is made pre-operatively. Definitive diagnosis is often based on histochemical, macroscopic, and microscopic examination of tissue specimens. The disease should be treated with high doses of intravenous penicillin for 2–6 weeks followed by oral therapy for at least 6–12 months.
Keywords: Actinomycosis abdominalis, Actinomycosis neuii
Introduction Abdominal actinomycosis is a rare, chronic spreading granulomatous and fibrosing infection characterized by the formation of multiple abscesses, draining sinuses, and the release of characteristic ‘sulphur granules’. Inoculation occurs with mucosal disruption, usually following perforation of an abdominal viscus.1,2 Actinomyces spp. also requires the presence of other bacteria, which destroy the overvascularized regions and convert an aerobic environment to an anaerobic one. This allows Actinomyces spp. to migrate, infect, and proliferate easily in already injured tissue.3 Actinomycosis occurs most frequently in the cervicofacial (50–65%), abdominal (20%), and thoracic (15%) regions. Abdominopelvic actinomycosis is increasing in frequency.4 The genus Actinomyces consists of a heterogeneous group of Gram-positive, non-spore-forming, catalase-negative pleomorphic rods that form a significant component of the intestinal commensal flora. The bacteria are generally of low pathogenicity, but may invade damaged mucosa, leading to bacteremia and systemic infection in both healthy individuals and immunocompromised patients.5 Diagnosis is often difficult because the nonspecific symptoms on initial presentation. Imaging is mostly Correspondence to: P. De Munter, UZ Leuven, Belgium. Email: [email protected]
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performed using computed tomography (CT) or magnetic resonance imaging (MRI) and is often suggestive of neoplasm. In less than 10% of cases, the diagnosis is made preoperatively.6 Treatment consists of long-term penicillin and drainage of the abscesses.2 In this case report, we present a single case of abdominal actinomycosis occurring in a 74-year-old female with a history of cholecystectomy 42 months before presentation. In a review of the literature, we present risk factors, clinical characteristics, diagnosis, and treatment of this rare infection.
Case Report A 74-year-old woman presented in the Emergency Department of the University Hospital of Leuven in January 2012 with right thoracic pain and malaise with severe fatigue. The right thoracic pain had been present for a few weeks, was initially respiratoryrelated, but had become continuous since a few days. She also had an intermittently sharp stabbing pain in the right lower thorax quadrant for a few months now. She had an unintentional weight loss of 2 kg over the past 2 weeks. Repeat history taking revealed that the patient had been feeling unwell since discharge from another hospital after a cholecystectomy in 2008. She now presented in the emergency department because of increasingly radiating pain from the right thoracic base towards the ventral and dorsal thoracic half. The pain increased when lying down and with deep inspiration. She was nauseous
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without abdominal pain or vomiting. She had had a cold since 2 weeks without signs of respiratory distress. Her past history consisted of a breast enlargement in 1973, arterial hypertension treated with antihypertensive medication, and a laparoscopic cholecystectomy in July 2008 with post-operative bile leak, and treated with antibiotics for 10 days (amoxicillin and clavulanic acid). She had lived in Congo from 1963 until 1973. Clinical examination showed a normal abdominal investigation (no tenderness, no pain on pressure), reduced vesicular breathing of the entire right lung with crepitations during in- and expiration, and a known systolic murmur, grade 3/6, best heard in the apical region in left lateral position. Breast examination showed two indurated collections on the left side, attributed to an involuted breast prosthesis. There were no signs of dehydration, no oedema of the ankles, and a normal CVD. Her blood pressure was 130/65 mmHg, heart rate was 88 beats/min, regular, respiratory rate was 18/min, and blood oxygen saturation level was 99% without oxygen. The highest measured temperature during admission was 38.3uC. Laboratory results showed a white blood cell count of 18.46109/l with 90.8% neutrophils (normal value: 4.06109–10.06109/l). C-reactive protein was 195.4 mg/l (normal value: ,5.0 mg/l). Liver set was abnormal with alkaline phosphatase 662 U/l (normal value: ,113 U/l), aspartate transaminase 66 U/l (normal value: ,38 U/l), alanine transaminase 88 U/l (normal value: ,41 U/l), gamma-glutamyl transpeptidase 114 U/l (normal value: ,53 U/l), total bilirubine 1.17 mg/dl) (normal value: ,1.00 mg/dl), and conjugated bilirubine 0.70 mg/dl (normal value: ,0.50 mg/dl). She had some electrolyte abnormalities with a potassium level 3.4 mmol/l (normal value: 3.50–5.10 mmol/l), sodium 128.6 mmol/l (normal value: 135.0–145.0 mmol/l), and chloride 89.4 mmol/l (normal value: 98.0–107 mmol/l). Lactate levels were 27.74 mmol/l (normal value:
Abdominal actinomycosis: a rare complication after cholecystectomy
0.5–1.6 mmol/l) and D-dimers were 2186 mg/l (normal value: ,500 mg/l). Chest X-ray showed a bilateral pleural effusion (right more than left). Ultrasound of the abdomen showed a non-vascularized cystic lesion with air entrapment in its wall (865.565 cm) in segment 5 of the liver, highly suspicious for a liver abscess. A CT abdomen was therefore performed, which showed a large liver abscess with a multi-compartmental pleural effusion on the right (Fig. 1A). The abscess was drained through CT-guided puncture. A drain was left in place (Fig. 1B). The pleural effusion was not evacuated at that time. The puncture fluid was sent for pathological examination and culture. Serologies for Echinococcus and Entamoeba histolytica were negative. Blood cultures were also negative. The patient was admitted to the hospital and was started on intravenous amoxicillin clavulanic acid. During admission, she had one episode of febrility: the temperature range varied between 37.5 and 38.3uC. Microscopic examination and culture of the puncture fluid showed the presence of Gram-positive rods, identified as Actinomyces neuii by matrixassisted laser desorption ionization time-of-flight mass spectrometry. Based on the identification and antimicrobial susceptibility testing, antibiotic treatment could be adjusted to penicillin G (663 mU IV). A CT thorax showed a pleural effusion, encapsulated in the oblique fissure of the right lung. Puncture of the fluid showed empyema (glucose ,2 mg/dl, total protein level of 35 g/l, lactate dehydrogenase 10 989 U/l, amylase 25 U/l, white blood cell count 11.16109/l). Culture (under antibiotic treatment) remained negative. A thoracic surgeon was consulted and 13 days after admission a thoracotomy was performed with
Figure 1 (A) CT abdomen before punction: a large liver abscess is visible in segment 5 (arrow). (B) CT abdomen after punction: the abscess was drained through CT-guided puncture. A drain was left in place (arrow).
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pleural decortication (resection of the visceral and parietal pleura) and drainage of the empyema. The patient was discharged from the hospital 1 month after admission. At that time, she had received 4 weeks IV antibiotics. Treatment with amoxicillin 1 g, three times a day, was continued, with a planned treatment of 6 months. During follow-up consultations, 3 and 6 months after hospitalization, she had no more complaints. Her chest radiographs showed regression of the pleural effusion and an ultrasound of the abdomen showed no collections and a normal liver span. Laboratory results showed a normalization of liver function and C-reactive protein. After 6 months of therapy with amoxicillin 1 g, three times a day, therapy was discontinued.
Discussion Actinomycosis was first discovered by Israel in 1878.1 It is traditionally divided into three forms: cervicofacial, thoracic, and abdominogenital actinomycosis. The most frequent site of infection is the cervicofacial area, accounting for 40%/50% of cases: 15% of actinomyosis occurs in the thorax; 20% occurs in the abdomen and pelvis.7 Abdominal actinomycosis affects abdominal organs, such as the stomach, colon, or liver. Pelvic and abdominal actinomycosis is increasing in frequency.8 It is found worldwide and occurs at any age, but is rare at ages younger than 10. Males are more frequently infected and the peak incidence is between 15 and 30 years.1 Our patient had a large liver abscess in segment 5 with a multi-compartmental pleural effusion on the right lung base. The liver abscess was immediately drained through CT-guided puncture (Fig. 1). A pleural decortication and drainage of the empyema was performed later during admission via thoracotomy. Remarkably, our patient was, in contrast to the majority of patients who present with actinomycosis infections, a 74-year-old female. Since Actinomyces is considered to be a saprophyte in the oral cavity, gastro-intestinal tract, and female genital tract, some surgical triggers such as mechanical wounds, operation, placement of intrauterine contraceptive device, and teeth extraction may act to destroy the mucosal barrier function, resulting in facilitating the invasion of the pathogen into deeper tissue.9 Other predisposing factors include immunosuppression (HIV, diabetes) and neoplasia. No predisposing factors are, however, noted in 50% of cases.10 Humans are natural reservoirs and there is no documented person-toperson transmission of the disease.11 All tissues and organs can be infected once the mucosal barrier is broken.1
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Reports have described intra-abdominal infections related to spilled gallstones after laparoscopic cholecystectomy.12 Gallstone spillage may occur many months before clinical presentation, in line with the delayed clinical course of actinomycosis and the slow growth of the pathogen. Establishment of human infection may also require the presence of other bacteria, which release a toxin or enzyme inhibiting the host defence. This enhances the relatively low invasive power of actinomycosis. At a more advanced stage, the abdominal mass is accompanied with extensive sinus, fistula, and abscess formation, usually draining to the skin.1 In abdominopelvic actinomycosis, aggressive peri-lesional infiltration with a tendency to cross fascial planes or boundaries and extend to the abdominal wall had been described as an important radiological finding. Haematogenous spread may occur, but lymphatic spread is said not to occur because of the large size of the organism.13 Our patient had a laparoscopic cholecystectomy in July 2008 (with post-operative bile leak and need for antibiotics for 10 days). It is most likely that spilled gallstones or bile during this procedure were the cause of the abdominal infection and formation of the liver abscess. She presented in the emergency department 42 months later with right thoracic pain, malaise and unintentional weight loss. This thoracic pain was caused by a large pleural effusion, which was caused by fistula formation and peri-lesional infiltration of the abdominal focus. Actinomycosis can mimic a malignant tumor when seen on CT or MRI. Imaging often shows bowel wall thickening and regional pelvic or peritoneal mass with extensive infiltration. It must be part of the differential diagnosis of patients with a history of surgical or invasive procedures, presenting with abdominal mass, wall thickness of the intestine, and symptoms such as abdominal pain, fever, and an elevated leukocyte count.8,14 Based on the types of infections, it must be assumed that A. neuii causes endogenous infections.16 Our patient also presented with atypical symptoms of thoracic and abdominal pain, unintentional weight loss, and extreme fatigue, which have a broad differential diagnosis. Radiological techniques are inadequate in the diagnosis of abdominal actinomycosis, except for CT, which shows the site and content of the lesions and their relation to adjacent tissue. When actinomycosis is suspected, CT-guided aspiration, with or without core biopsy of suspicious lesions, is a useful investigation.15 Definitive diagnosis is based on histochemical, macroscopic, and microscopic examination of surgical tissue specimens, which reveal yellow sulphur granules and basophilic filament aggregates, respectively.
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Figure 2 Typical appearance of Actinomyces neuii on Gram stain (61000). Unlike the classical Actinomyces species, Actinomyces neuii shows no branching.
Cultures can be positive in about 50% of cases, but take up to 2 weeks to grow, because of anaerobic characteristics and slow growth of the organisms.10 Further complicating culture interpretation is that specimens are commonly mixed with other organisms. Often the diagnosis requires surgical intervention to obtain specimens adequate to demonstrate the characteristic findings of sulphur granules and filament aggregates.6 Sulphur granules can be observed in the purulent material in 50% of cases, but these are not pathognomonic for the disease. The differential diagnosis of sulphur granules includes nocardiosis, streptomycosis, chromomycosis, eumycetoma, and botryomycosis.5 Actinomyces granules regularly show a positive reaction with periodic acid Schiff and Grocott’s dye, but the Kossa reaction is negative.14 Molecular identification by 16S PCR gives another level of certainty and allows definitive speciation.5 There are more than 13 different species of actinomycosis, six of which are associated with human disease (israelli, naeslundii, bovis, odontolyticus, viscosus, and Arachnia propionica).6 In our patient, as mentioned earlier, Actinomyces neuii was found (Fig. 2). Actinomyces neuii had proven to be an exception in the genus of actinomyces on account of its aerobic growth, microscopic morphology (no branching), and the types and localization of infections. Abscesses and infected atheromas are the most frequent types of infections, followed by infected skin structures, endophthalmitis, and bacteremia including endocarditis. Abscesses are mostly localized in the mammary, axillary, and inguinal areas. Intraabdominal and intra-thoracic infections are extremely rare with this species. In direct Gram-stained preparations and smears from cultures, A. neuii appears as a diphtheroidal and even coccoid organism without branching. Because of its atypical appearance, A. neuii could easily be misdiagnosed as a corynebacterium. A. neuii grows within 48 hours on aerobically and
Abdominal actinomycosis: a rare complication after cholecystectomy
anaerobically incubated blood agar plates. In our department of microbiology in Leuven, there have been a few reports of A. neuii in the past. Before use of mass spectrometry, A. neuii was identified as a Corynebacterium species. The emerging use of mass spectrometry has resolved in more frequent identification of previously rarely identified species. The disease should be treated with high doses of intravenous treatment for 2–6 weeks followed by oral therapy for at least 6–12 months. The antibiotic of choice is penicillin. In case of allergy or non-response, alternatives include erythromycin, tetracyclin, clindamycin, cephalosporins, meropenem, and chloramphenicol. Frequent confections with other microbes make combination therapy advisable.5 Surgical resection can be necessary if antibiotic treatment is insufficient.8 Combined medical and surgical treatment achieves a cure in about 90% of cases.7 Our patient first received intravenous amoxicillinclavulanic acid, but this was replaced by penicillin G (663 mU IV) after culture became positive for Actinomyces neuii. Blood cultures remained negative. A pleural decortication and drainage of the empyema was performed through thoracotomy 13 days after admission. The patient was discharged from the hospital 1 month after admission after 4 weeks of intravenous antibiotics. Treatment with amoxicillin 1 g, three times a day, was continued, with a duration of 6 months.
Conclusion In this case, we reported a single case of thoracicabdominal actinomycosis occurring in a 74-year-old female with a history of cholecystectomy 42 months before presentation. Actinomyces should always be part of the differential diagnosis of patients presenting with a history of surgical or invasive procedures, presenting with an abdominal mass. CT-guided aspiration with or without core biopsy of this mass is a useful investigation. Definitive diagnosis is often based on histochemical, macroscopic, and microscopic examination of tissue specimens. Sulphur granules can be observed in the purulent material in 50% of cases, but these are not pathognomonic for the disease. The disease should be treated with high doses of intravenous penicillin for 2–6 weeks, followed by oral therapy for at least 6–12 months.
Declaration of Interest No funding was received for this article. There are no commercial relationships or conflicts of interest regarding this article.
Acknowledgements With special thanks to Professor Jan Verhaegen and his team for providing microbiological findings and figures.
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References 1 Wong J, Kinney T, Miller F, Rivera-Sanfeliz G. Hepatic actinomycotic abscesses: diagnosis and management. AJR Am J Roentgenol. 2006;186:174–6. 2 Kocabay G, Cagatay A, Eraksoy H, Tiryaki B, Alper A, Calangu S. A case of isolated hepatic actinomycosis causing right pulmonary empyema. Chin Med J. 2006;119:1133–5. 3 Hsu JT, Lo HC, Jan YY, Chen M. Actinomycosis mimicking recurrent carcinoma after Whipple’s operation. World J Gastroenterol. 2005;11:1722–4. 4 Elazary R, Bala M, Almogy G, Khalaileh A, Kisselgoff D, RavAcha M, et al. Small bowel obstruction and cecal mass due to actinomycosis. IMAJ. 2006;8:653–4. 5 Lall T, Shebab T, Valenstein P. Isolated hepatic Actinomyces: a case report. J Med Case Rep. 2010;4:45. 6 Vodovnik A, Logishetty K. Actinomyces complicating sigmoid diverticular disease: a case report. Cases J. 2009;2:6456. 7 Pusiol T, Morichetti D, Pedrazzani C, Ricci F. Abdominalpelvic Actinomyces mimicking malignant neoplasm. Infect Dis Obstet Gynecol. 2011;2011:747059. 8 Baierlein S, Wistop A, Looser C, Peters T, Riehle HM, Peterli R. Abdominal Actinomyces: a rare complication after laparascopic gastric bypass. Obes Surg. 2007;17:1123–6.
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9 Hayashi M, Asakuma M, Tsunemi S, Inoue Y, Shimizu T, Komeda K, et al. Surgical treatment for abdominal actinomycosis: a report of two cases. World J Gastrointest Surg. 2010;2:405–8. 10 Heidt J, Jansen CL, Leyten EM. An abdominal mass: not a ‘clear cut’ case! Netherlands J Med. 2010;68:319–21. 11 Sahay S, Gonzalez H, Luong T, Rahman S. Pancreatic actinomycosis as a cause of retroperitoneal fibrosis in a patient with chronic pancreatitis. Case report and literature review. JOP. 2010;11:477–9. 12 Reichenbach J, Lopatin U, Mahlaoui N, Beovic B, Siler U, Zbinden R, et al. Actinomyces in chronic granulomatous disease: an emerging and unanticipated pathogen. Clin Infect Dis. 2009;49:1703–10. 13 Nozawa H, Yamada Y, Muto Y, Arita S, Aisaka K. Pelvic actinomycosis presenting with a large abscess and bowel stenosis with marked response to conservative treatment: a case report. J Med Case Rep. 2007;1:141. 14 Ong C, Barness S, Senanayake S. Actinomyces turicensis infection mimicking ovarian tumor. Singapore Med J. 2012;53:9–11. 15 Choi MM, Beak J, Lee J, Park S, Lee WS. Clinical features of abdominopelvic actinomycosis: report of twenty cases and literature review. Yonsei Med J. 2009;50:555–9. 16 von Graevenitz A. Actinomyces neuii: review of an unusual infectious agent. Infection. 2011;39:97–100.
Abdominal actinomycosis: a rare complication after cholecystectomy X. Vanoeteren1, K. Devreese2, P. De Munter1 1
Department of General Internal Medicine, University Hospital Leuven, Belgium, 2Department of Clinical Biology and Laboratory, University Hospital Leuven, Belgium We present a single case of abdominal actinomycosis occurring in a 74-year-old female with a history of cholecystectomy 42 months before presentation. In a review of the literature, we present risk factors, clinical characteristics, diagnosis, and treatment of this infection. Abdominal actinomycosis is a rare, chronic, granulomatous infection characterized by the release of ‘sulphur granules’. Actinomyces species should always be part of the differential diagnosis of patients presenting with a history of surgical or invasive procedures, presenting with an abdominal mass. Computed tomography (CT)-guided aspiration with or without core biopsy of this mass is a useful investigation. Diagnosis is often difficult: In less than 10% of cases, the diagnosis is made pre-operatively. Definitive diagnosis is often based on histochemical, macroscopic, and microscopic examination of tissue specimens. The disease should be treated with high doses of intravenous penicillin for 2–6 weeks followed by oral therapy for at least 6–12 months.
Keywords: Actinomycosis abdominalis, Actinomycosis neuii
Introduction Abdominal actinomycosis is a rare, chronic spreading granulomatous and fibrosing infection characterized by the formation of multiple abscesses, draining sinuses, and the release of characteristic ‘sulphur granules’. Inoculation occurs with mucosal disruption, usually following perforation of an abdominal viscus.1,2 Actinomyces spp. also requires the presence of other bacteria, which destroy the overvascularized regions and convert an aerobic environment to an anaerobic one. This allows Actinomyces spp. to migrate, infect, and proliferate easily in already injured tissue.3 Actinomycosis occurs most frequently in the cervicofacial (50–65%), abdominal (20%), and thoracic (15%) regions. Abdominopelvic actinomycosis is increasing in frequency.4 The genus Actinomyces consists of a heterogeneous group of Gram-positive, non-spore-forming, catalase-negative pleomorphic rods that form a significant component of the intestinal commensal flora. The bacteria are generally of low pathogenicity, but may invade damaged mucosa, leading to bacteremia and systemic infection in both healthy individuals and immunocompromised patients.5 Diagnosis is often difficult because the nonspecific symptoms on initial presentation. Imaging is mostly Correspondence to: P. De Munter, UZ Leuven, Belgium. Email: [email protected]
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performed using computed tomography (CT) or magnetic resonance imaging (MRI) and is often suggestive of neoplasm. In less than 10% of cases, the diagnosis is made preoperatively.6 Treatment consists of long-term penicillin and drainage of the abscesses.2 In this case report, we present a single case of abdominal actinomycosis occurring in a 74-year-old female with a history of cholecystectomy 42 months before presentation. In a review of the literature, we present risk factors, clinical characteristics, diagnosis, and treatment of this rare infection.
Case Report A 74-year-old woman presented in the Emergency Department of the University Hospital of Leuven in January 2012 with right thoracic pain and malaise with severe fatigue. The right thoracic pain had been present for a few weeks, was initially respiratoryrelated, but had become continuous since a few days. She also had an intermittently sharp stabbing pain in the right lower thorax quadrant for a few months now. She had an unintentional weight loss of 2 kg over the past 2 weeks. Repeat history taking revealed that the patient had been feeling unwell since discharge from another hospital after a cholecystectomy in 2008. She now presented in the emergency department because of increasingly radiating pain from the right thoracic base towards the ventral and dorsal thoracic half. The pain increased when lying down and with deep inspiration. She was nauseous
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without abdominal pain or vomiting. She had had a cold since 2 weeks without signs of respiratory distress. Her past history consisted of a breast enlargement in 1973, arterial hypertension treated with antihypertensive medication, and a laparoscopic cholecystectomy in July 2008 with post-operative bile leak, and treated with antibiotics for 10 days (amoxicillin and clavulanic acid). She had lived in Congo from 1963 until 1973. Clinical examination showed a normal abdominal investigation (no tenderness, no pain on pressure), reduced vesicular breathing of the entire right lung with crepitations during in- and expiration, and a known systolic murmur, grade 3/6, best heard in the apical region in left lateral position. Breast examination showed two indurated collections on the left side, attributed to an involuted breast prosthesis. There were no signs of dehydration, no oedema of the ankles, and a normal CVD. Her blood pressure was 130/65 mmHg, heart rate was 88 beats/min, regular, respiratory rate was 18/min, and blood oxygen saturation level was 99% without oxygen. The highest measured temperature during admission was 38.3uC. Laboratory results showed a white blood cell count of 18.46109/l with 90.8% neutrophils (normal value: 4.06109–10.06109/l). C-reactive protein was 195.4 mg/l (normal value: ,5.0 mg/l). Liver set was abnormal with alkaline phosphatase 662 U/l (normal value: ,113 U/l), aspartate transaminase 66 U/l (normal value: ,38 U/l), alanine transaminase 88 U/l (normal value: ,41 U/l), gamma-glutamyl transpeptidase 114 U/l (normal value: ,53 U/l), total bilirubine 1.17 mg/dl) (normal value: ,1.00 mg/dl), and conjugated bilirubine 0.70 mg/dl (normal value: ,0.50 mg/dl). She had some electrolyte abnormalities with a potassium level 3.4 mmol/l (normal value: 3.50–5.10 mmol/l), sodium 128.6 mmol/l (normal value: 135.0–145.0 mmol/l), and chloride 89.4 mmol/l (normal value: 98.0–107 mmol/l). Lactate levels were 27.74 mmol/l (normal value:
Abdominal actinomycosis: a rare complication after cholecystectomy
0.5–1.6 mmol/l) and D-dimers were 2186 mg/l (normal value: ,500 mg/l). Chest X-ray showed a bilateral pleural effusion (right more than left). Ultrasound of the abdomen showed a non-vascularized cystic lesion with air entrapment in its wall (865.565 cm) in segment 5 of the liver, highly suspicious for a liver abscess. A CT abdomen was therefore performed, which showed a large liver abscess with a multi-compartmental pleural effusion on the right (Fig. 1A). The abscess was drained through CT-guided puncture. A drain was left in place (Fig. 1B). The pleural effusion was not evacuated at that time. The puncture fluid was sent for pathological examination and culture. Serologies for Echinococcus and Entamoeba histolytica were negative. Blood cultures were also negative. The patient was admitted to the hospital and was started on intravenous amoxicillin clavulanic acid. During admission, she had one episode of febrility: the temperature range varied between 37.5 and 38.3uC. Microscopic examination and culture of the puncture fluid showed the presence of Gram-positive rods, identified as Actinomyces neuii by matrixassisted laser desorption ionization time-of-flight mass spectrometry. Based on the identification and antimicrobial susceptibility testing, antibiotic treatment could be adjusted to penicillin G (663 mU IV). A CT thorax showed a pleural effusion, encapsulated in the oblique fissure of the right lung. Puncture of the fluid showed empyema (glucose ,2 mg/dl, total protein level of 35 g/l, lactate dehydrogenase 10 989 U/l, amylase 25 U/l, white blood cell count 11.16109/l). Culture (under antibiotic treatment) remained negative. A thoracic surgeon was consulted and 13 days after admission a thoracotomy was performed with
Figure 1 (A) CT abdomen before punction: a large liver abscess is visible in segment 5 (arrow). (B) CT abdomen after punction: the abscess was drained through CT-guided puncture. A drain was left in place (arrow).
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pleural decortication (resection of the visceral and parietal pleura) and drainage of the empyema. The patient was discharged from the hospital 1 month after admission. At that time, she had received 4 weeks IV antibiotics. Treatment with amoxicillin 1 g, three times a day, was continued, with a planned treatment of 6 months. During follow-up consultations, 3 and 6 months after hospitalization, she had no more complaints. Her chest radiographs showed regression of the pleural effusion and an ultrasound of the abdomen showed no collections and a normal liver span. Laboratory results showed a normalization of liver function and C-reactive protein. After 6 months of therapy with amoxicillin 1 g, three times a day, therapy was discontinued.
Discussion Actinomycosis was first discovered by Israel in 1878.1 It is traditionally divided into three forms: cervicofacial, thoracic, and abdominogenital actinomycosis. The most frequent site of infection is the cervicofacial area, accounting for 40%/50% of cases: 15% of actinomyosis occurs in the thorax; 20% occurs in the abdomen and pelvis.7 Abdominal actinomycosis affects abdominal organs, such as the stomach, colon, or liver. Pelvic and abdominal actinomycosis is increasing in frequency.8 It is found worldwide and occurs at any age, but is rare at ages younger than 10. Males are more frequently infected and the peak incidence is between 15 and 30 years.1 Our patient had a large liver abscess in segment 5 with a multi-compartmental pleural effusion on the right lung base. The liver abscess was immediately drained through CT-guided puncture (Fig. 1). A pleural decortication and drainage of the empyema was performed later during admission via thoracotomy. Remarkably, our patient was, in contrast to the majority of patients who present with actinomycosis infections, a 74-year-old female. Since Actinomyces is considered to be a saprophyte in the oral cavity, gastro-intestinal tract, and female genital tract, some surgical triggers such as mechanical wounds, operation, placement of intrauterine contraceptive device, and teeth extraction may act to destroy the mucosal barrier function, resulting in facilitating the invasion of the pathogen into deeper tissue.9 Other predisposing factors include immunosuppression (HIV, diabetes) and neoplasia. No predisposing factors are, however, noted in 50% of cases.10 Humans are natural reservoirs and there is no documented person-toperson transmission of the disease.11 All tissues and organs can be infected once the mucosal barrier is broken.1
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Reports have described intra-abdominal infections related to spilled gallstones after laparoscopic cholecystectomy.12 Gallstone spillage may occur many months before clinical presentation, in line with the delayed clinical course of actinomycosis and the slow growth of the pathogen. Establishment of human infection may also require the presence of other bacteria, which release a toxin or enzyme inhibiting the host defence. This enhances the relatively low invasive power of actinomycosis. At a more advanced stage, the abdominal mass is accompanied with extensive sinus, fistula, and abscess formation, usually draining to the skin.1 In abdominopelvic actinomycosis, aggressive peri-lesional infiltration with a tendency to cross fascial planes or boundaries and extend to the abdominal wall had been described as an important radiological finding. Haematogenous spread may occur, but lymphatic spread is said not to occur because of the large size of the organism.13 Our patient had a laparoscopic cholecystectomy in July 2008 (with post-operative bile leak and need for antibiotics for 10 days). It is most likely that spilled gallstones or bile during this procedure were the cause of the abdominal infection and formation of the liver abscess. She presented in the emergency department 42 months later with right thoracic pain, malaise and unintentional weight loss. This thoracic pain was caused by a large pleural effusion, which was caused by fistula formation and peri-lesional infiltration of the abdominal focus. Actinomycosis can mimic a malignant tumor when seen on CT or MRI. Imaging often shows bowel wall thickening and regional pelvic or peritoneal mass with extensive infiltration. It must be part of the differential diagnosis of patients with a history of surgical or invasive procedures, presenting with abdominal mass, wall thickness of the intestine, and symptoms such as abdominal pain, fever, and an elevated leukocyte count.8,14 Based on the types of infections, it must be assumed that A. neuii causes endogenous infections.16 Our patient also presented with atypical symptoms of thoracic and abdominal pain, unintentional weight loss, and extreme fatigue, which have a broad differential diagnosis. Radiological techniques are inadequate in the diagnosis of abdominal actinomycosis, except for CT, which shows the site and content of the lesions and their relation to adjacent tissue. When actinomycosis is suspected, CT-guided aspiration, with or without core biopsy of suspicious lesions, is a useful investigation.15 Definitive diagnosis is based on histochemical, macroscopic, and microscopic examination of surgical tissue specimens, which reveal yellow sulphur granules and basophilic filament aggregates, respectively.
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Figure 2 Typical appearance of Actinomyces neuii on Gram stain (61000). Unlike the classical Actinomyces species, Actinomyces neuii shows no branching.
Cultures can be positive in about 50% of cases, but take up to 2 weeks to grow, because of anaerobic characteristics and slow growth of the organisms.10 Further complicating culture interpretation is that specimens are commonly mixed with other organisms. Often the diagnosis requires surgical intervention to obtain specimens adequate to demonstrate the characteristic findings of sulphur granules and filament aggregates.6 Sulphur granules can be observed in the purulent material in 50% of cases, but these are not pathognomonic for the disease. The differential diagnosis of sulphur granules includes nocardiosis, streptomycosis, chromomycosis, eumycetoma, and botryomycosis.5 Actinomyces granules regularly show a positive reaction with periodic acid Schiff and Grocott’s dye, but the Kossa reaction is negative.14 Molecular identification by 16S PCR gives another level of certainty and allows definitive speciation.5 There are more than 13 different species of actinomycosis, six of which are associated with human disease (israelli, naeslundii, bovis, odontolyticus, viscosus, and Arachnia propionica).6 In our patient, as mentioned earlier, Actinomyces neuii was found (Fig. 2). Actinomyces neuii had proven to be an exception in the genus of actinomyces on account of its aerobic growth, microscopic morphology (no branching), and the types and localization of infections. Abscesses and infected atheromas are the most frequent types of infections, followed by infected skin structures, endophthalmitis, and bacteremia including endocarditis. Abscesses are mostly localized in the mammary, axillary, and inguinal areas. Intraabdominal and intra-thoracic infections are extremely rare with this species. In direct Gram-stained preparations and smears from cultures, A. neuii appears as a diphtheroidal and even coccoid organism without branching. Because of its atypical appearance, A. neuii could easily be misdiagnosed as a corynebacterium. A. neuii grows within 48 hours on aerobically and
Abdominal actinomycosis: a rare complication after cholecystectomy
anaerobically incubated blood agar plates. In our department of microbiology in Leuven, there have been a few reports of A. neuii in the past. Before use of mass spectrometry, A. neuii was identified as a Corynebacterium species. The emerging use of mass spectrometry has resolved in more frequent identification of previously rarely identified species. The disease should be treated with high doses of intravenous treatment for 2–6 weeks followed by oral therapy for at least 6–12 months. The antibiotic of choice is penicillin. In case of allergy or non-response, alternatives include erythromycin, tetracyclin, clindamycin, cephalosporins, meropenem, and chloramphenicol. Frequent confections with other microbes make combination therapy advisable.5 Surgical resection can be necessary if antibiotic treatment is insufficient.8 Combined medical and surgical treatment achieves a cure in about 90% of cases.7 Our patient first received intravenous amoxicillinclavulanic acid, but this was replaced by penicillin G (663 mU IV) after culture became positive for Actinomyces neuii. Blood cultures remained negative. A pleural decortication and drainage of the empyema was performed through thoracotomy 13 days after admission. The patient was discharged from the hospital 1 month after admission after 4 weeks of intravenous antibiotics. Treatment with amoxicillin 1 g, three times a day, was continued, with a duration of 6 months.
Conclusion In this case, we reported a single case of thoracicabdominal actinomycosis occurring in a 74-year-old female with a history of cholecystectomy 42 months before presentation. Actinomyces should always be part of the differential diagnosis of patients presenting with a history of surgical or invasive procedures, presenting with an abdominal mass. CT-guided aspiration with or without core biopsy of this mass is a useful investigation. Definitive diagnosis is often based on histochemical, macroscopic, and microscopic examination of tissue specimens. Sulphur granules can be observed in the purulent material in 50% of cases, but these are not pathognomonic for the disease. The disease should be treated with high doses of intravenous penicillin for 2–6 weeks, followed by oral therapy for at least 6–12 months.
Declaration of Interest No funding was received for this article. There are no commercial relationships or conflicts of interest regarding this article.
Acknowledgements With special thanks to Professor Jan Verhaegen and his team for providing microbiological findings and figures.
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