Pelvic and abdominal-wall actinomycotic infection by uterus gateway without genital lesions

Introduction
Actinomycosis is a rare chronic infectious disease caused by gram-positive bacteria of the Actinomycetaceae family. It is usually found in the gastrointestinal flora and female genital tract (1, 2, 3). The inflammatory lesions are mostly unique, occurring mainly in the cervical and facial regions (50-70%), as well as in the thoracic (15-20%) and abdominal-pelvic areas (10-20%). The characteristic lesion occurs as an infiltrative mass, displaying micro-abscesses or abscesses which sometimes may lead to skin fistulae. As these microorganisms are not virulent, they require a break in the integrity of the mucous membranes by surgery (oral surgery, bowel or pelvic surgery), by foreign bodies (e.g., chicken or fish bones, IUDs) or triggered by other infections during which actinomycetes is introduced into the deep tissues by mucosal perforation (typhoid fever, amoebiasis, perforated appendicitis, perforated diverticulitis) (1, 2).
The diagnosis is suggested by imaging techniques and confirmed by pathological examination of the specimens obtained from deep needle aspirate or biopsy specimens (specific sulphur granules are pathognomonic). The diagnosis can also be established bacteriologically – although less frequent – by placing the specimens obtained from draining sinuses or puncture under anaerobiotic conditions. The differential diagnosis is made with other chronic inflammatory conditions (tuberculosis, Chron’s disease) but mostly with malignant tumours. The treatment requires medical and surgical care.

Case report
A 36-year-old female was admitted from gynaecology department for the presence of an abdominal-pelvic mass, with no clear relation (clinical or imagistic) with genital organs. For the last 3 weeks the patient has been complaining of a hypo-gastrium painful tumour with fever and urinary frequency. The patient’s physiological and pathological history is nonspecific except an IUD for the last 3 years.
Clinical findings are normal, except for a palpable, solid and poor delimited hypo gastric mass of 5 to 6 cm, originating in the abdominal wall structures. The vaginal examination reveals the genitals within normal limits, but following digital rectal examination a second tumour belonging to the rectal wall on the right side is discovered. The tumour was 3-4 cm in size, 6 cm above the dentate line, localized extra-mucosal and mobile.
Laboratory tests were within normal limits except for a mild anaemia (11.2 g/dl) and a leucocytosis (11 800 /mm3).
CT scan reveals above the pubis, at the level of the rectum abdominal muscles, a non-homogeneous mass of 3 cm in size, with central necrosis extended through the anterior aspect of the urinary bladder wall (Fig. 1, 2).
During surgery, via sub-umbilical median incision, a tumoural mass of 5-6 cm in size was discovered in the muscular-aponeurotic layer above the pubic bone. The tumour was of solid consistency, poorly delimited, and developed through the rectum abdominal muscle, its aponeurosis, pro-peritoneal tissue and peritoneum. Approximately 4 cm of the anterior-superior aspect of the urinary bladder wall was involved also. The genitals were unaffected. On the right lateral rectal wall a second tumour of 2 to 2 cm in size with the same characteristics was discovered.
The parietal mass, including a segment of the urinary bladder wall, underwent tumorectomy as well as the lateral rectal tumour. The defect of the urinary bladder wall was closely followed by suture-cistoraphy. The specimen of the parietal tumour is poorly delimited, yellow-grey in colour, with small areas of central necrosis. The extemporaneous pathological examination of the parietal specimen showed a sarcoma-like pattern.
The outcome of the pathologic examination of paraffin-embedded tissue samples from both tumoural specimens indicates inflammatory lesions characteristic of actinomycosis. The Intra-Uterine Device (IUD) was removed and placed under anaerobiotic cultures conditions, yet without identifying bacterial colonies from the Actynomyces group. For the next 14 days the patient was put on antimicrobial therapy with Ampicilin. (30 mg/kg/day). The postoperative outcome was uneventful.

Discussions
Actinomycosis is a rare chronic bacterial infection (1,300,000 cases in the USA and 1,100,000 in the Netherlands and Germany) caused in human subjects mainly by Actinomyces israeli. Other isolated species of the genera (e.g. Actinomyces meyeri, odontolyticus and naeslundii) may cause similar clinical syndromes (1, 2, 3, and 4). They require a break in the integrity of the digestive or genital mucous membranes in order to invade and cause human illness. The widespread use of all types of IUDs has led to an increased number of actinomycotic infections via genital organs which were reported in the medical literature (3, 5, 6, and 7).
The particularity of this patient resides in the presence of two lesions (urinary bladder and abdominal wall and also rectal wall), located in the vicinity of the uterus bearing an IUD, but without any damage of genital organs (ovaries, uterus or fallopian tubes). Although starting from the gateway of the actinomycotic lesions, the literature lists parauterine lesions as well, without affecting the fallopian tubes or the ovaries. (3).
Diagnosis of Actinomycosis of the abdomen and pelvis is difficult because the lesion frequently shows unique or multiple tumour-like aspect. They are poorly delimited, typically contiguously spread, frequently ignoring tissue planes and invading surrounding tissues or organs. (1, 2, 3). These characteristics make the differentiation (clinical and imagistic) between them and neoplasic tumours very difficult (7,8,9). The confusion with a malignancy was made with respect to this patient, due to the intraoperatory macroscopic appearance of the lesion and the extemporaneous pathological examination aspect of the specimen.
In clinical forms of actinomycosis with multiple abscesses and sinus tracts clinical findings, imagistic, bacteriologic and microscopic tests (with discharge of sulphur granules that are pathognomonic) could give a more rapid diagnosis than in tumour-like forms.
Although the infection responds to a wide range of antimicrobial therapy, which can eventually lead to full recovery, most patients end up on the operating table, as was the case of our patient. Even in the cases involving advanced lesions (3) (especially with the pseudotumoural forms) on account of the difficulties encountered in establishing the diagnosis.

Conclusions
The presence of an Intra-Uterine Contraceptive Device (IUD) should be taken as a sign of a possible actinomycotic infection, often localized in the internal genital organs and pelvic structures.
In some patients, the lesions can affect structures and organs close to the IUD-bearing uterus, without damaging the internal genital organs.
The identification of the sulphur granules in the specimen sampled via puncture from a supposedly actinomycotic provides significant clues to the diagnosis and points out to the possibility of a treatment based on antibiotics, thus avoiding surgery.
When diagnosing an abdominal or pelvic mass in patients with a history that might have triggered lesions of the digestive mucosa (recent or remote bowel surgery, infectious diseases or caused by external foreign bodies), the possibility of an actinomycotic lesion must always be taken into consideration.

References
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3. Russo T. Agents of Actinomycosis. In: Principles and Practice of Infectious Diseases, 5th edition, Mandell, Douglas and Bennett’s editors. New York: Ed. Churchill Livingston; 2000. p. 2645-2652.
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8. Elazary R, Bala M, Almogy G, Khalaileh A, Kisselgoff D, Rav-Acha M, et al. Small bowel obstruction and cecal mass due to actinomycosis. Isr Med Assoc J. 2006;8(9):653-4.
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