Article Type : Case Report
Authors : Romdhani M, Hannachi S, El Mnif K, Msakni I, Abid R and Battikh R
Keywords : Actinomyces; Renal; Infection; Histopathology; Antibiotherapy
Actinomycosis is an uncommon, bacterial
infection resulting from anaerobic Gram-positive filamentous bacteria,
belonging to the Actinomyces genus. These bacteria inhabit various parts of the
body such as the respiratory system, digestive system, and female reproductive
tract. The clinical and radiological signs and symptoms of this condition are
diverse, resembling several other diseases, including cancer, which can make
its diagnosis challenging and frequently delayed. It's important to note that
renal involvement in actinomycosis is exceedingly rare. We present a case of a
young man who was admitted to the hospital with renal actinomycosis. The
patient presented with abdominal pain, night sweats and alteration of the
clinical status. He underwent a radical nephrectomy given that the radiological
presentation suggested a tumoral cause. The pathology showed the actinomycosis
specific « sulfure granules». Our patient received antibiotic treatment with
penicillin G initially, followed by a combination of amoxicillin and
doxycycline, which resulted in a favourable outcome.
Actinomycosis is a rare, chronic, and slowly
progressive bacterial infection. It is caused by anaerobic and microaerophilic
bacteria of the genus Actinomyces, mainly Actinomyces israelii [1]. These
bacteria are usual saprophytic germs of the natural cavities of the human
being. They normally colonize the oral cavity, the digestive, the respiratory
and the female genital tract. Renal involvement is exceptional [2]. Clinical
and radiological manifestations are numerous, mimicking several pathologies
including neoplasia, making the diagnosis difficult and often delayed.
We report the case of a 22-year-old man, without
important medical history admitted in our department for symptoms evolving for
six months including abdominal pain, night sweats and alteration of the
clinical status. In ambulatory, he received symptomatic treatment combined with
unspecified antibiotic therapy for a short time without any clinical
improvement. An abdominal CT-Scan was carried out showing a tissue mass in the
upper pole of the right kidney, infiltrating the anterior, posterior, and
superior pararenal space, invading the adrenal and the underlying hepatic
parenchyma. It also showed adenomegaly of the right renal pedicle (Figure 1).
Laboratory showed, leukocytosis neutrophils (WBC by 13.2 × 109/L,
neutrophils by 10.5 × 109/L). Serum C-reactive protein was by 40
mg/L (N <8 mg/l) and creatinemia was normal. The urine culture was sterile
and the HIV serology was negative. In the face of imaging findings, a possible
tumour was suspected. We did not perform a biopsy for fear of tumour cell
dissemination. A laparoscopic radical nephrectomy with adrenalectomy was
therefore done. The specimen was sent to pathology. Macroscopy study had shown
enlarged right nephrectomy specimen removing the adrenal gland measuring
160/110/80mm. On section, the upper renal pole is dissociated by a voluminous
lesion of pseudotumor appearance, 70 mm in length, solid, greyish-white,
necrotic, of heterogeneous consistency, friable and necrotic. This lesion
appears to be fistulized in the peri-renal fat, giving rise to a fibroblastic
reaction that encompasses the adrenal gland (Figure 2). Microscopically, it is
an inflammatory pseudotumor formation consisting of sheets of inflammatory
cells with a predominance of neutrophils creating foci of suppuration. In the
centre of these foci, actinomycotic grains can be seen, forming a fine
filamentous felting (Figure 3). There are associated fistulous tracts in the
peri-renal and adrenal fat. There is no histological sign of malignancy. The
microbiological culture was negative. We started with intravenous antibiotic
therapy with penicillin G (20 MIU/day) for two weeks combined with oral doxycycline
(200 mg/day). We subsequently made the oral relay by amoxicillin (2gx3/day)
combined with doxycycline (200 mg/day) for a total duration of 12 months.
Follow-up consultations reported a complete disappearance of the clinical
signs.
Actinomycosis is an uncommon infection that is not
well recognized and is seldom considered initially. Its diagnosis is
challenging due to its slow progression and diverse range of clinical
manifestations. This complexity arises from the lack of specific clinical and
radiological indicators. It is a bacterial infection caused by Actinomyces
species which are anaerobic, Gram-positive filamentous bacteria [3]. This
microorganism normally inhabits the mucosa of the oropharynx, tracheobronchial
tree, intestines, and female reproductive system as a saprophyte. Despite its
common presence, it rarely causes infections in people with healthy immune
system. It has not been reported to be transmitted from one human to another
[3]. The crucial step in the development of actinomycosis is the penetration of
Actinomyces spp. into various organs and structures due to a mucosal rupture or
breach [3]. This disease is characterized by its tendency to form foci of
suppuration within the tissues, which evolve spontaneously towards
fistulization with an intense inflammatory reaction surrounded by fibrosis. It
extends to adjacent tissues, without respect for anatomical barriers [4]. The
infection primarily occurs in the cervicofacial area in about 60% of cases,
where it typically affects the salivary glands and lacrimal ducts. In
approximately 20% to 25% of cases, it is located in the thoracic region and can
cause lung or chest wall involvement. About 25% of the cases occur in the
abdominal area, with the most common locations being the caecal appendix and
the pelvic region in women. Intrauterine devices are thought to be a
significant contributing factor to the occurrence of the infection in the
pelvic region [5,6]. However, renal actinomycosis is very uncommon, with less
than 25 documented cases in adult patients since 1990 [7]. Renal involvement in
actinomycosis occurs through two mechanisms: the first is a hematogenous
dissemination from other infection sites, while the second mechanism is through
contamination by contiguity, which is facilitated by the bacteria's production
of proteolytic enzymes [8]. In our case, the source of contamination was not
defined. This infection mimics various diseases mainly malignant masses,
tuberculosis and fungal infections. In addition, the evolution is often
insidious, responsible of a diagnostic delay, source of evolved forms [8]. The
manifestations of renal actinomycosis can include a renal abscess, pyonephrosis
with renal calcinosis, or necrotizing papillitis. The majority of reported
cases have occurred in individuals who have a normal immune system [9].
As the case of our patient, the majority of patients
affected by renal actinomycosis are individuals with a normal immune system and
no pre-existing medical conditions. They exhibited non-specific clinical
symptoms such as abdominal pain, weight loss, fever, weakness, and night sweats,
in descending order of frequency. Hematuria and urinary symptoms were less
commonly observed in these cases [7]. Biology tests are not particularly useful
and lack specificity for diagnosing actinomycosis. However, patients often
exhibit a biological inflammatory response with an increase in C- reactive
protein levels and an accelerated erythrocyte sedimentation rate. Additionally,
an increase in gamma globulin levels has also been reported. In some cases,
hyperleukocytosis with a predominance of neutrophils is observed, although this
is not always the case. This abnormality was also observed in the patient we
examined. The radiological manifestations of actinomycosis are varied and lack
specificity, making it difficult to distinguish from malignant tumours.
Nevertheless, radiological examinations play a crucial role in diagnosing and
assessing the extent of the infection, identifying potential complications, and
monitoring the efficacy of treatment [7]. The CT scan can reveal a bulky tumour
that is heterogeneous in texture and exhibit early enhancement after the
injection of contrast material. The tumour spread to nearby structures [8].
There is no definitive test to detect the presence of Actinomyces bacteria, and
serological tests are not particularly useful. Culturing the bacteria is a
lengthy process that requires anaerobic conditions [8]. The process of
isolating and identifying Actinomyces species can be difficult, especially when
hindered by factors such as prior antibiotic treatment, improper specimen
transportation, insufficient culture conditions, and inadequate incubation
periods. Routine culture techniques and typical incubation times may fail to
detect Actinomyces species, as they are often delicate and slow to grow,
thriving in anaerobic environments. To improve sensitivity, direct
Gram-staining is crucial and has been found to be more effective than culture
methods [10].
In 1999, Hyldgaard-Jensen and al. reported the case of a 45-year-old patient who underwent an ultrasound guided biopsy for a large multinodular mass of the right kidney, finding the “grains” specific of the Actinomycosis, which has enabled a rapid and appropriate antibiotic therapy [11]. In 2004, Dhanani and al. described the case of a 64-year-old man presenting a heterogeneous right flank mass containing air-fluid levels, and an inflammatory reaction involving the right colon and psoas muscle in the CT scan. An empirical antibiotic therapy was prescribed and the repeated CT showed decreased size and inflammation surrounding the mass. The central location of the residual mass imposed a fine needle aspiration showing the « sulfur granules » with no evidence of an urothelial malignancy [12]. However, in many cases, the diagnosis wasn’t straightforward and nephrectomy was performed [7,13,14].
Figure 1: Abdominal CT scan showing
an upper polar renal tissue mass invading the right adrenal gland and the lower
border of the liver (arrow).
Figure 2: Nephrectomy with adrenalectomy specimen (macroscopic aspect): The upper renal pole is dissociated by a voluminous lesion of pseudotumor appearance (star) with fistula (arrow).
Figure
3:
Microscopic appearance of renal actinomycosis with actinomycotic filaments
within a granuloma (PAS coloration, medium power view).
On the treatment of renal actinomycosis, antibiotic
therapy is the rule. Two principles of antibiotic therapy for actinomycosis
have been widely applied for decades: high doses and prolonged duration of
antibiotics. They are explained by the fact that the actinomycosis follicle is
surrounded by a poorly vascularized zone of fibrosis, which impedes
antibiotics. Similarly, antibiotic penetration is reduced in necrotic and scar
tissue [15]. Optimal tissue concentrations are then difficult to achieve. This
is why it is necessary to prolong the duration of antibiotic therapy, in high
doses, in order to treat existing foci and avoid recurrences. Curative
treatment of this infection is based on antibiotic therapy with penicillin G
for a period ranging from one month after disappearance of symptomatic
symptoms, to 12 months. Other antibiotics are also effective: ampicillin,
tetracyclines, clindamycin, chlorampenicol and rifampicin [8].
Renal actinomycosis is a rare condition characterized
by a gradual progression, often resulting in the identification of large masses
with extension and inflammation of nearby tissues. In cases where
imaging does not exhibit typical features of renal cell carcinoma, it is
advisable to consider ultrasound-guided biopsies to avoid unnecessary surgical
interventions. However, for locally advanced or pseudotumoral forms, surgical
intervention may be necessary. If medical treatment alone is pursued, it should
be administered for an extended period, and regular abdominal CT scans are
essential to monitor the regression of the infection.