Actinomycosis is very often misdiagnosed as typically, the
associated laboratory and radiological findings are nonspecific.
Therefore, clinicians should always have a high index
of suspicion in cases in which there is a chronic, indolent
development of a mass lesion with sinus tracts, that progresses
through the tissue planes, and which relapses following short
courses of antibiotics. Early diagnosis may prevent invasive
investigations and radical surgical procedures as the patient
can then simply be treated with oral penicillin.http://www.sajei.co.za/index.php/SAJEIam2014ay201

Geldenhuys, Annelize

Nyapholi, Mohapi

Visser, Adele

UPSpace at the University of Pretoria

130 2013;28(2)South Afr J Epidemiol InfectCase Report: Patient with protracted abdominal painPatient with protracted abdominal painM Nyapholi, A Geldenhuys, A VisserMohapi Nyapholi, MBChB, DTM&H, Senior RegistrarAnnelize Geldenhuys, MBChB, DA(SA), PGDipTM, RegistrarAdele Visser, MBChB, DTM&H, PGDipTM, MMed, FCP(SA), HeadDivision of Clinical Pathology, Department of Medical Microbiology, University of PretoriaNational Health Laboratory Services, Tshwane Academic DivisionE-mail: adele.vis@gmail.comKeywords: actinomycosis, intrauterine devices Peer reviewed. (Submitted: 2012-04-03. Accepted: 2012-08-03.) © SAJEI South Afr J Epidemiol Infect 2013;28(2):130-133IntroductionActinomycosis is very often misdiagnosed as typically, the associated laboratory and radiological findings are non-specific. Therefore, clinicians should always have a high index of suspicion in cases in which there is a chronic, indolent development of a mass lesion with sinus tracts, that progresses through the tissue planes, and which relapses following short courses of antibiotics.1 Early diagnosis may prevent invasive investigations and radical surgical procedures as the patient can then simply be treated with oral penicillin.2Case reportA 38-year-old woman presented with a three-month history of vague lower abdominal pain with associated nausea, vomiting and excessive weight loss. In addition to these symptoms, she also developed pain and paraesthesia in her right leg, not precipitated by any specific movement. Eight years prior, she had been diagnosed with an ovarian cyst which had been progressively enlarging, based on sonographic evaluation. Having obtained the history, the patient was determined to be para 2, gravida 2, and to have had an intrauterine device inserted as a contraceptive measure on three previous occasions between 1999 and 2011.Laboratory investigations confirmed that the patient had normocytic normochromic anaemia, with a haemoglobin level of 10.2 g/dl (12.5-16 g/dl), a mean corpuscular volume of 86.4 fl (78-100 fl) and a mean corpuscular haemoglobin of 25.7 pg (27-31 pg). Both the platelet and white cell counts were elevated, at 613 x 109/l (140 to 370 x 109/l) and 25.9 x 109/l (4-10.5 x 109/l), respectively. Renal function and electrolytes were within normal ranges, but the alkaline phosphatase was slightly elevated at 145  U/L (42-98  U/L), and the serum albumin was low at 15 g/l (35-52  g/l). The patient was confirmed to be human immunodeficiency virus (HIV)-1 negative by the HIV Combi Assay® (Roche Diagnostics, Mannheim, Germany).A speculum examination of the cervix revealed three circular haematomatous lesions. A pelvic mass of 8 cm x 5 cm x 4 cm was also demonstrated on abdominal ultrasound. A provisional diagnosis of ovarian cancer was made and the decision was taken to perform a staging laparotomy. Ascites was aspirated and submitted for adenosine deaminase levels, and measured at 11.3 U/l (> 30 U/l is suggestive of tuberculosis). In addition, samples were submitted for mycobacterial culture and microscopy, which were negative in both instances. During this procedure, a “frozen pelvis” was discovered with extensive adhesions between the bowel, uterus and right Fallopian tube, as well as a semi-solid adnexal mass, measuring 5 cm x 4 cm x 6.4 cm. The differential diagnosis was established to be ovarian carcinoma, and infection with Mycobacterium tuberculosis. Unfortunately, tissue samples were not submitted for mycobacterial culture which would have been the investigation of choice in this setting. A bilateral adnexectomy and subtotal hysterectomy, with resection of small and large bowel parts, including the appendix, was performed and submitted for histological examination. Macroscopically, nodular lesions with central necrosis could be demonstrated in the right adnexa only. Despite numerous leiomyomas, similar nodular lesions were not found in the uterus. On microscopic examination of haematoxylin and eosin stain of the right adnexa with ovary, multiple collections of filamentous bacteria, with surrounding inflammatory response and granuloma formation, could be demonstrated (Figure 1 a and b). These collections of bacteria were Gram-positive, positive for periodic acid-Schiff (Figure 1 c and d) 131 2013;28(2)South Afr J Epidemiol InfectCase Report: Patient with protracted abdominal painand Grocott’s dye positive, and negative with Ziehl-Neelson stain. The remainder of the samples that were evaluated microscopically did not show the same pathology, except for the serosal surface of the appendix. A diagnosis of ovarian actinomycosis was made and the patient was treated with parenteral penicillin.DiscussionActinomycosis is a chronic suppurative granulomatous condition that is caused by a group of Gram-positive filamentous anaerobic bacteria. Species that are commonly associated with human infections are Actinomyces israelii, A. naeslundii, A. odontolyticus, A. viscosus, A. meyeri and A. gerencseriae.3 At times referred to as “the most undiagnosed disease”,4 this infection can mimic a wide range of conditions and is often missed, if not actively excluded.1 It should be included in the differential diagnosis of most cases of unconfirmed malignancy as it has been described in this context on numerous occasions.4,5Clinical manifestationsReported prevalence varies significantly in different geographic regions,6,7 although South African data are lacking. Immunosuppression, as a result of various conditions, has largely been cited as a risk factor for this condition.1,8 Generally, six clinical forms have been described, with highly varying prevalence.1 The most common form is orocervicofacial actinomycosis, which is present in up to 50% of cases.9 Typically, this is associated with previous dental manipulation or trauma, as well as poor dental hygiene.10 Thoracic actinomycosis is the second most common form, accounting for approximately 15-20% of cases.9,11 However, the clinical picture can vary greatly, delaying the initial diagnosis.12 This is usually secondary to the aspiration of oropharyngeal secretions, but can also be secondary to direct extension from cervicofacial or abdominal infection. Haematogenous spread has also been described.6Abdominopelvic actinomycosis accounts for an estimated 20% of cases, of which the majority present with Figure 1 a-d: Microscopic appearance of lesions found in the right ovary of the patientFigure 1 a: Haematoxylin and eosin stain showing infiltration by Actinomycetes (black arrow), with surrounding inflammatory cell infiltrate and granuloma formation (white arrow)Figure 1 b: Closer view of the haematoxylin and eosin, showing the filamentous nature of the infiltrateFigure 1 c: Periodic acid-Schiff stain, showing Actinomycetes infiltration (black arrow), with surrounding inflammation (white arrow)Figure 1 d: Closer view of the filamentous infiltrate on the periodic acid-Schiff 132 2013;28(2)South Afr J Epidemiol InfectCase Report: Patient with protracted abdominal painappendicitis.13,14 In most instances, it has been postulated to be secondary to gastrointestinal perforations, neoplasia or surgery.1 Direct extension can occur into the pelvis,1 although most of the primary pelvic cases occur in association with intrauterine contraceptive devices (IUDs).15,16 Despite this classic association, more than 30-million patient years of IUDs have been reported worldwide.16 Currently, there are less than 100 case published reports in the English literature.4 Very rarely, the central nervous system can be involved via either direct extension or haematogenous spread.1 Of these cases, brain abscesses seem to be the most common presentation. Meningitis, subdural empyema and epidural abscesses are less common.17 Rare manifestations include musculoskeletal18,19 and disseminated disease.18DiagnosisTypically, initial findings in affected patients are non-specific and include anaemia, leucocytosis and features of inflammation, like a raised C-reactive protein or erythrocyte sedimentation rate.3,11,16 Elevated alkaline phosphatase, as described in this case, has been noted in cases of hepatic actinomycosis.20Radiological findings suffer the same non-specificity that is returned by general laboratory testing. Typically, these findings are similar to those of inflammation or malignancy.11 As a rule, lymphatic spread does not occur. Therefore, regional lymphadenopathy is a rare finding.21 No radiological features are considered to be specific for actinomycosis19,20,22 and the diagnosis is rarely established preoperatively based on the radiological findings.4Histopathology can greatly facilitate in establishing a diagnosis of actinomycosis. The demonstration of sulphur granules, consisting of Gram-positive filamentous organisms, is highly supportive of this diagnosis.1 However, up to 46% of cases have no sulphur granules23 and sulphur granules may also be present in a range of other conditions, including nocardiosis,17 streptomycosis, chromomycosis, eumycetoma and botryo-mycosis.24 Typically, actinomycosis stains positive with both periodic acid-Schiff stain and Grocott’s dye, as both are specific for carbohydrates that are found in the cell wall.23,25-33 These two positive stains distinguish actinomycosis from nocardiosis and streptomycosis.34 The differential diagnosis will also include infection with mycobacteria. Therefore, it is suggested that a Ziehl-Neelson stain is also performed. This will be negative in cases of actinomycosis.23 The gold standard for diagnosis remains direct isolation of the organism from a clinical sample.1 However, the culture is fraught with difficulties and is successful in less than 50% of cases.20 Difficulties include administration of the antibiotic therapy prior to sampling, overgrowth by other microbes, as well as analytical difficulties.21 Sulphur granules remain the optimal sample to obtain, but tissue may also be submitted. Once sampled, the specimen should be transported to the laboratory urgently35 to enable rapid inoculation of the culture media and incubation in anaerobic conditions.5 Culture should be followed by identification of the organism, as this condition is caused by a heterogeneous group of bacilli. This can be carried out through biochemical testing (commercially available kits),36 molecular genetic methods,37 fluorescent in situ hybridisation37 or mass spectrometry.38ConclusionThe current epidemiology of actinomycosis in South Africa has not been studied thoroughly. As immunosuppression is cited as a risk factor for acquiring this infection, clinicians should have a high index of suspicion in cases in which there is the chronic, indolent development of a mass lesion with sinus tracts, that progresses through the tissue planes and which Figure 2 b: A closer view of Grocott’s dye positivity, showing filamentsFigure 2 a: Grocott’s dye-positive133 2013;28(2)South Afr J Epidemiol InfectCase Report: Patient with protracted abdominal painrelapses following short courses of antibiotics. Early diagnosis may prevent invasive investigations and radical surgical procedures as these patients can simply be treated with oral penicillin. Conflict of interestThe authors do not report any conflict of interest. DeclarationNo external funding was utilised for the purpose of this study. Dr Visser’s work is supported by the Discovery Foundation.AknowledgementsAcknowledgement is given to Dr JE Dinkel, Senior Specialist, Department of Anatomical Pathology, University of Pretoria/National Health Laboratory Services, Tshwane Academic Division, for assistance with the histopathology diagnosis. References1. Wong V. Actinomycosis. BMJ. 2011;343:d6099.2. Smith A, Hall V, Thakker B, Gemmell C. Antimicrobial susceptibility testing of Actinomyces species with 12 antimicrobial agents. J Antimicrob Chemother. 2005;56(2):407-409.3. Acevedo F, Baudrand R, Letelier L, Gaete P. Actinomycosis: a great pretender. Case reports of unusual presentations and a review of the literature. Int J Infect Dis. 2008;12(4):358-362.4. Laish I, Benjaminov O, Morgenstern S, et al. Abdominal actinomycosis masquerading as colon cancer in a liver transplant recipient. Transp Infect Dis. 2012;14(1):86-90.5. Pusiol T, Morichetti D, Pedrazzani C, Ricci F. Abdominal-pelvic actinomycosis mimicking malignant neoplasm. Infect Dis Obst Gynaecol. 2011;2011:747059.6. Russo T. Agents of actinomycosis. In: Mandell G, Bennett J, Dolin R, editors. Principles and practice of infectious diseases. 7th ed. Philadelphia: Elsevier Churchill Livingstone, 2010; p. 3209-3219.7. Smego R, Foglia G. Actinomycosis. Clin Infect Dis. 1998;26(6):1255-1261.8. Dominguez D, Antony S. Actinomyces and nocardia infections in immunocompromised and non-immunocompromised patients. J Nat Med Ass. 1999;91(1):35-39.9. Weese W, Smith I. A study of 57 cases of actinomycosis over a 36-year period. A diagnostic “failure” with good prognosis after treatment. Arch Intern Med. 1975;135(12):1562-1568.10. Schaal K, Lee H. Actinomycete infections in humans: a review. Gene. 1992;115(1-2):201-211.11. Mabeza G, Macfarlane J. Pulmonary actinomycosis. Eur Respir J. 2003;21(3):545-551.12. Goussard P, Gie R, Kling S, Beyers N. Thoracic actinomycosis mimicking primary tuberculosis. Pediatr Infect Dis J. 1999;18(5):473-475. 13. Agarwal K, Sharma U, Acharya V. Microbial and cytopathological study of intrauterine contraceptive device users. Indian J Med Sci. 2004;58(9):394-399.14. Cayley J, Fotherby K, Guillebaud J, et al. Recommendations for clinical practice: actinomyces like organisms and intrauterine contraceptives. Br J Fam Plann. 1998;23(4):137-138.15. Choi M, Baek J, Lee J, et al. Clinical features of abdominopelvic actinomycosis: report of twenty cases and literature review. Yonsei Med J. 2009;50(4):555-559.16. Fiorino A. Intrauterine contraceptive device-associated actinomycotic abscess and Actinomyces detection on cervical smear. Obstet Gynecol. 1996;87(1):142-149.17. Smego R. Actinomycosis of the central nervous system. Rev Infect Dis. 1987;9(5):855-865.18. Felz M, Smith M. Disseminated actinomycosis: multisystem mimicry in primary care. South Med J. 2003;96(3):294-299.19. Ha K, Lee H, Kim H, et al. Abdominal actinomycosis: CT findings in 10 patients. AJR Am J Roentgenol. 1993;161(4):791-794.20. Kim T, Han J, Hoh W, et al. Thoracic actinomycosis: CT features with histopathologic correlation. AJR Am J Roentgenol. 2006;186(1):225-231.21. Bennhoff D. Actinomycosis: diagnosis and therapeutic considerations and a review of 32 cases. Laryngoscope. 1984;94(9):1198-1217.22. Pickhardt P, Bhalla S. Unusual nonneoplastic peritoneal and subperitoneal conditions: CT findings. Radiographics. 2005;25(3):719-730.23. Brown J. Human actinomycosis. A study of 181 subjects. Hum Pathol. 1973;4(3):319-330.24. Ormsby A, Bauer T, Hall G. Actinomycosis of the cholecystic duct: case report and review. Pathology. 1998;30(1):65-67.25. Grocott R. A stain for fungi in tissue sections and smears using Gomori’s methenamine-silver nitrate technique. Am J Clin Pathol. 1955;25(8):975-979.26. Lequerré T, Nouvellon M, Kraznowska K, Bruno MC. Septic arthritis due to Actinomycosis naeslundii: report of a case. Joint Bone Spine. 2002;69(5):499-501.27. Harsch IA, Benninger J, Niedobitek G, et al. Abdominal actinomycosis: complication of endoscopic stenting in chronic pancreatitis? Endoscopy. 2001;33(12):1065-1069.28. Hansen T, Wagner W, Kirkpatrick CJ, Kunkel M. Infected osteoradionecrosis of the mandible: follow-up suggests deterioration in outcome for patients with Actinomyces-positive bone biopsies. Inter J Oral Maxillo Fac Surg. 2006;35(11):1001-1004.29. Kim TS, Han J, Choi JC, et al. Actinomycosis: CT features with histopathologic correlation. Amer J Roentgenol. 2006;186(1):225-231.30. Acquaro P, Tagliabue F, Confalonieri G, et al. Abdominal wall actinomycosis simulating a malignant neoplasm: case report and review of the literature. Worl J Gastrointes Surg. 2010;2(7):247-250.31. Kawamura N, Shimada A, Morita T, et al. Veteri. Rec. Intraperitoneal actinomycosis in cat. 2005;157(19):593-594.32. Andreani A, Cavazza A, Marchioni A, et al. Bronchopulmonary actinomycosis associated with hiatal hernia. Mayo Clin Proc. 2009;84(2):123-128.33. Hefny AF, Sebastian M, Torab FC, Joshi S, Abu-Zidan FM. Actinomycosis of the gallbladder: case report and review of the literature. Asia J Surg. 2005;28(3):230-232. 34. Kamprath S, Merker A, Kuhne-Heid R, Schneider A. Abdominale aktinomykose bei IUD. Zentralbl Gynakol Journal. 1997;119(1):21-24.35. Brook I. Actinomycosis: diagnosis and management. South Med J. 2008;101(10):1019-1023.36. Miller P, Wiggs L, Miller J. Evaluation of API An-IDENT and RapID ANA II systems for identification of Actinomyces species from clinical specimens. J Clin Microbiol. 1995;33(2):329-330.37. Hansen J, Fjeldsoe-Nielsen H, Sulim S, et al. Actinomyces species: a Danish survey on human infections and microbiological characteristics. Open Microbiol J. 2009;3:113-120.38. Fass R, Scholand J, Hodges G, Saslaw S. Clindamycin in the treatment of serious anaerobic infections. Ann Intern Med. 1973;78(6):853-859.

Patient with protracted abdominal pain

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Patient with protracted abdominal pain

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