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Orphanet Journal of Rare Diseases Jun 2023CNS actinomycosis is a rare chronic suppurative infection with non-specific clinical features. Diagnosis is difficult due to its similarity to malignancy, nocardiosis... (Review)
Review
BACKGROUND
CNS actinomycosis is a rare chronic suppurative infection with non-specific clinical features. Diagnosis is difficult due to its similarity to malignancy, nocardiosis and other granulomatous diseases. This systematic review aimed to evaluate the epidemiology, clinical characteristics, diagnostic modalities and treatment outcomes in CNS actinomycosis.
METHODS
The major electronic databases (PubMed, Google Scholar, and Scopus) were searched for the literature review by using distinct keywords: "CNS" or "intracranial" or "brain abscess" or "meningitis" OR "spinal" OR "epidural abscess" and "actinomycosis." All cases with CNS actinomycosis reported between January 1988 to March 2022 were included.
RESULTS
A total of 118 cases of CNS disease were included in the final analysis. The mean age of patients was 44 years, and a significant proportion was male (57%). Actinomycosis israelii was the most prevalent species (41.5%), followed by Actinomyces meyeri (22.6%). Disseminated disease was found in 19.5% of cases. Most commonly involved extra-CNS organs are lung (10.2%) and abdomen (5.1%). Brain abscess (55%) followed by leptomeningeal enhancement (22%) were the most common neuroimaging findings. Culture positivity was found in nearly half of the cases (53.4%). The overall case-fatality rate was 11%. Neurological sequelae were present in 22% of the patients. On multivariate analysis, patients who underwent surgery with antimicrobials had better survival (adjusted OR 0.14, 95% CI 0.04-0.28, p value 0.039) compared to those treated with antimicrobials alone.
CONCLUSION
CNS actinomycosis carries significant morbidity and mortality despite its indolent nature. Early aggressive surgery, along with prolonged antimicrobial treatment is vital to improve outcomes.
Topics: Humans; Male; Adult; Abscess; Actinomycosis; Central Nervous System Diseases; Treatment Outcome
PubMed: 37269006
DOI: 10.1186/s13023-023-02744-z -
Orphanet Journal of Rare Diseases Apr 2021Hepatic Actinomycosis (HA) is one of the infections that causes disorders in patients when diagnosed untimely and inappropriately. (Review)
Review
BACKGROUND
Hepatic Actinomycosis (HA) is one of the infections that causes disorders in patients when diagnosed untimely and inappropriately.
METHODS
Case reports on HA in patients published between 2000 and April 2020 were gathered by carrying out a structured search through PubMed/Medline.
RESULTS
Through a survey of the Medline database, 130 studies were identified and then, 64 cases with HA were included in the final analysis. Asia had the largest share of cases with 37.5% (24 reports), followed by Europe and the Americas. Affected patients were predominantly males (64%) and the overall mortality rate was 1% with only one male patient in his 50 s dying. Nearly all patients (92%) were immunocompetent. However, in four patients, the use of immunosuppressive medication led to depression of the immune system. Most of the patients (80%) experienced complications. In terms of the complications, the most frequent ones were previous history of abdominal surgery (32%) and foreign bodies in the abdominopelvic region (20%). Actinomyces israelii was the most common pathogen isolated from patients. Abdominal pain (66%), fever (62%), weight loss (48%), night sweat, malaise, and anorexia (14%) over about 3.1 months were the most frequently reported clinical symptoms. Extension to one or more surrounding organs was evident in 18 patients (28%). Histopathologic examination confirmed infection in 67% of the patients and samples obtained from liver puncture biopsy (32%) were most frequently used in diagnosis. Surgery or puncture drainage + anti-infection was the most common method to treat patients and penicillin, Amoxicillin, Doxycycline, and ampicillin were the most frequently used drugs to control infection.
CONCLUSION
HA should be considered in patients with a subacute or chronic inflammatory process of the liver. With accurate and timely diagnosis of infection, extensive surgery can be prevented.
Topics: Actinomyces; Actinomycosis; Asia; Europe; Humans; Male
PubMed: 33931097
DOI: 10.1186/s13023-021-01821-5 -
Neurological Sciences : Official... Feb 2022Whipple's disease (WD) is a systemic, chronic, relapsing disease caused by Tropheryma whipplei, which can mimic signs and symptoms of various clinical entities. Typical... (Review)
Review
OBJECTIVE
Whipple's disease (WD) is a systemic, chronic, relapsing disease caused by Tropheryma whipplei, which can mimic signs and symptoms of various clinical entities. Typical manifestations are represented by gastrointestinal and systemic symptoms, among which neurological ones are frequent. We present the case of a patient with WD and rapidly progressive cognitive impairment and a review of literature aimed to report epidemiological, clinical, neuroimaging, and laboratory findings of cognitive impairment associated with WD.
METHODS
A systematic review of medical literature published until November 22, 2020, was performed. Full-text, peer-reviewed case reports and series in English language presenting patients with WD and cognitive impairment were included. Data concerning demographic, clinical, neuroimaging, and laboratory characteristics were collected and synthesized qualitatively.
RESULTS
The patient was a 54-year-old male who developed rapidly progressive dementia, fluctuating arousal disturbances, and supranuclear ophthalmoparesis associated with chronic diarrhea and fever spikes. T. whipplei was detected in the cerebrospinal fluid, and appropriate antimicrobial therapy was given with progressive clinical benefit. The systematic review of 114 case reports/series identified 147 patients with WD and cognitive impairment; this latter was rarely isolated. Neurological symptoms associated with cognitive decline were psychiatric disturbances, supranuclear ophthalmoplegia, hypothalamic involvement, and consciousness disorders. Brain imaging and cerebrospinal fluid findings were heterogeneous and nonspecific.
CONCLUSIONS
Cognitive impairment represents one of the most common neurological features associated with WD. The clinical suspicion of this disease in patients with rapidly progressive dementia is crucial to guide diagnostic strategies and proper antimicrobial therapy, which may revert the clinical deterioration.
Topics: Cognitive Dysfunction; Dementia; Diarrhea; Humans; Male; Middle Aged; Tropheryma; Whipple Disease
PubMed: 34981284
DOI: 10.1007/s10072-021-05844-5 -
Alimentary Pharmacology & Therapeutics Apr 2015The classical form of Whipple's disease (WD), clinically characterised by arthropathy, diarrhoea and weight loss, is rare. Recently, other more frequent forms of... (Review)
Review
BACKGROUND
The classical form of Whipple's disease (WD), clinically characterised by arthropathy, diarrhoea and weight loss, is rare. Recently, other more frequent forms of Tropheryma whipplei infection have been recognised. The clinical spectrum includes an acute, self-limiting disease in children, localised forms affecting cardiac valves or the central nervous system without intestinal symptoms, and asymptomatic carriage of T. whipplei which is found in around 4% of Europeans. Genomic analysis has shown that T. whipplei represents a host-dependent or opportunistic bacterium. It has been reported that the clinical course of T. whipplei infection may be influenced by medical immunosuppression.
AIM
To identify associations between immunomodulatory treatment and the clinical course of T. whipplei infection.
METHODS
A PubMed literature search was performed and 19 studies reporting on immunosuppression, particularly therapy with tumour necrosis factor inhibitors (TNFI) prior to the diagnosis in 41 patients with Whipple?s disease, were evaluated.
RESULTS
As arthritis may precede the diagnosis of WD by many years, a relevant percentage (up to 50% in some reports) of patients are treated with immunomodulatory drugs or with TNFI. Many publications report on a complicated Whipple?s disease course or T. whipplei endocarditis following medical immunosuppression, particularly after TNFI. Standard diagnostic tests such as periodic acid-Schiff stain used to diagnose Whipple?s disease often fail in patients who are pre-treated by TNFI.
CONCLUSIONS
In cases of doubt, Whipple?s disease should be excluded before therapy with TNFI. The fact that immunosuppressive therapy contributes to the progression of T. whipplei infection expands our pathogenetic view of this clinical entity.
Topics: Anti-Bacterial Agents; Arthritis; Diagnosis, Differential; Humans; Immunoglobulins; Immunosuppressive Agents; Lymphocyte Count; Tumor Necrosis Factor-alpha; Whipple Disease
PubMed: 25693648
DOI: 10.1111/apt.13140 -
Frontiers in Medicine 2024Pulmonary actinomycosis (PA) is a rare type of infection that can be challenging to diagnose since it often mimics lung cancer.
BACKGROUND
Pulmonary actinomycosis (PA) is a rare type of infection that can be challenging to diagnose since it often mimics lung cancer.
METHODS
Published case reports and case series of PA in patients with suspicion of lung cancer were considered, and data were extracted by a structured search through PubMed/Medline.
RESULTS
After analyzing Medline, 31 studies were reviewed, from which 48 cases were extracted. Europe had the highest prevalence of reported cases with 45.1%, followed by Asia (32.2%), America (19.3%), and Africa (3.2%). The average age of patients was 58.9 years, and 75% of all patients were above 50 years old. Male patients (70%) were predominantly affected by PA. The overall mortality rate was 6.25%. In only eight cases, the causative agent was reported, and was the most common isolated pathogen with three cases. Based on histopathological examination, 75% of the cases were diagnosed, and the lobectomy was performed in 10 cases, the most common surgical intervention. In 50% of the cases, the selective antibiotics were intravenous and oral penicillin, followed by amoxicillin (29.1%), amoxicillin-clavulanic acid, ampicillin, levofloxacin, and doxycycline.
CONCLUSION
The non-specific symptoms resemble lung cancer, leading to confusion between PA and cancer in imaging scans. Radiological techniques are helpful but have limitations that can lead to unnecessary surgeries when confusing PA with lung cancer. Therefore, it is important to raise awareness about the signs and symptoms of PA and lung cancer to prevent undesirable complications and ensure appropriate treatment measures are taken.
PubMed: 38523909
DOI: 10.3389/fmed.2024.1356390 -
BMC Infectious Diseases Jan 2024Actinomyces turicensis is rarely responsible of clinically relevant infections in human. Infection is often misdiagnosed as malignancy, tuberculosis, or nocardiosis,...
BACKGROUND
Actinomyces turicensis is rarely responsible of clinically relevant infections in human. Infection is often misdiagnosed as malignancy, tuberculosis, or nocardiosis, therefore delaying the correct identification and treatment. Here we report a case of a 55-year-old immunocompetent adult with brain abscess caused by A. turicensis. A systematic review of A. turicensis infections was performed.
METHODS
A systematic review of the literature was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. The databases MEDLINE, Embase, Web of Science, CINAHL, Clinicaltrials.gov and Canadian Agency for Drugs and Technology in Health (CADTH) were searched for all relevant literature.
RESULTS
Search identified 47 eligible records, for a total of 67 patients. A. turicensis infection was most frequently reported in the anogenital area (n = 21), causing acute bacterial skin and skin structure infections (ABSSSI) including Fournier's gangrene (n = 12), pulmonary infections (n = 8), gynecological infections (n = 6), cervicofacial district infections (n = 5), intrabdominal or breast infections (n = 8), urinary tract infections (n = 3), vertebral column infections (n = 2) central nervous system infections (n = 2), endocarditis (n = 1). Infections were mostly presenting as abscesses (n = 36), with or without concomitant bacteremia (n = 7). Fever and local signs of inflammation were present in over 60% of the cases. Treatment usually involved surgical drainage followed by antibiotic therapy (n = 51). Antimicrobial treatments most frequently included amoxicillin (+clavulanate), ampicillin/sulbactam, metronidazole or cephalosporins. Eighty-nine percent of the patients underwent a full recovery. Two fatal cases were reported.
CONCLUSIONS
To the best of our knowledge, we hereby present the first case of a brain abscess caused by A. turicensis and P. mirabilis. Brain involvement by A. turicensis is rare and may result from hematogenous spread or by dissemination of a contiguous infection. The infection might be difficult to diagnose and therefore treatment may be delayed. Nevertheless, the pathogen is often readily treatable. Diagnosis of actinomycosis is challenging and requires prompt microbiological identification. Surgical excision and drainage and antibiotic treatment usually allow for full recovery.
Topics: Adult; Humans; Middle Aged; Actinomyces; Actinomycosis; Anti-Bacterial Agents; Brain Abscess; Canada
PubMed: 38245682
DOI: 10.1186/s12879-024-08995-w -
Journal of Gastrointestinal and Liver... Sep 2018Whipple's disease (WD) is known as an infrequent, systemic, chronic infection caused by the actinomycete Tropherima whipplei (T. whipplei). The disease is frequently...
Whipple's disease (WD) is known as an infrequent, systemic, chronic infection caused by the actinomycete Tropherima whipplei (T. whipplei). The disease is frequently characterized by a long prodromal and protean extra-intestinal phase, which often causes misdiagnosis and inappropriate treatments. Herein, we describe the case a 62-year-old man with a histological diagnosis of WD established when oral steroid treatment was started due to rheumatic manifestations, triggering intestinal symptoms. Systematic review of the literature was performed to include studies where WD was eventually diagnosed on duodenal biopsies. Three patients' subgroups were identified according to the clinical presentation.
Topics: Anti-Bacterial Agents; Biopsy; Duodenoscopy; Duodenum; Humans; Intestinal Mucosa; Middle Aged; Tropheryma; Whipple Disease
PubMed: 30240479
DOI: 10.15403/jgld.2014.1121.273.fra -
Cureus Jun 2023Central nervous system (CNS) actinomycosis is a rare, serious, life-threatening, suppurative infection caused by Actinomyces species. Actinomyces are anaerobic...
Central nervous system (CNS) actinomycosis is a rare, serious, life-threatening, suppurative infection caused by Actinomyces species. Actinomyces are anaerobic Gram-positive bacteria, which can be normally isolated from the polymicrobial flora of the gastrointestinal- and genital tracts. They are considered very low virulent bacteria to humans. However, they can lead to several types of local or disseminated infections, if certain pathologic states or immunodeficiency occur. Intracranial abscesses caused by Actinomyces meyeri are rarely reported in adults. In this case report, we describe a 66-year-old woman who presented to the emergency department due to progressive complaints of altered sensorium and low-grade fever, due to an A. meyeri-related brain abscess. The only risk factor was represented by immunodeficiency due to the therapy with Methotrexate and steroids.
PubMed: 37525793
DOI: 10.7759/cureus.41204