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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 -
American Journal of Otolaryngology 2022Actinomycosis is a granulomatous infection that rarely involves the larynx or pharynx. Three cases of actinomycosis of the larynx or pharynx from our institution were... (Review)
Review
INTRODUCTION
Actinomycosis is a granulomatous infection that rarely involves the larynx or pharynx. Three cases of actinomycosis of the larynx or pharynx from our institution were reviewed and a systematic literature review was performed to better define surgical management, antibiotic therapy, risk factors, and incidence of recurrence or complications.
MATERIALS AND METHODS
PubMed/Medline, Cochrane, Embase, and Google Scholar were searched on November 30, 2021 using the terms "laryngeal actinomycosis", "pharyngeal actinomycosis", "actinomycosis AND larynx", and "actinomycosis AND pharynx." Articles which did not describe appropriate sites or were non-English were excluded. Results were collected for demographic information, site(s) of infection, comorbidities, lesion characteristics and treatments.
RESULTS
Along with three cases reported from our institution, 40 unique cases were reviewed from 37 studies for a total of 43 patients (Table 1). 34 (81.0 %) of the patients were male with the highest incidence of infection in the seventh decade (54.8 %). The most common site for the infection was the larynx (69.0 %) followed by the pharynx (16.7 %). Risk factors included a history of radiation therapy, immunosuppression, inhalational irritant, and diabetes (Table 3). The duration of antibiotic therapy varied greatly, from one month to one year and total follow up ranged from 1 month to 2.5 years (Table 1).
CONCLUSIONS
A comprehensive review of the literature on pharyngolaryngeal actinomycosis shows that this infection has increased prevalence within the head and neck cancer patient population. Similar to cervicofacial actinomycosis, these atypical sites have shown favorable responses to extended antibiotic therapy and generally do not require aggressive surgical management.
Topics: Humans; Male; Female; Pharynx; Irritants; Actinomycosis; Larynx; Anti-Bacterial Agents
PubMed: 36029619
DOI: 10.1016/j.amjoto.2022.103609 -
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 -
ANZ Journal of Surgery Jul 2020Abdominal actinomycosis (AA) is a rare infection. The aim of this study was to summarize the evidence available on AA.
BACKGROUND
Abdominal actinomycosis (AA) is a rare infection. The aim of this study was to summarize the evidence available on AA.
METHODS
A systematic review was conducted. Data sources included Trip Database, BIREME, SciELO, Cochrane Library, WoS, MEDLINE, EMBASE, SCOPUS, IBECS and LILACS. Eligibility criteria included: studies related to surgically treated AA, in adult population, without language and sex restriction, published between 1966 and 2019. The following variables were analysed: publication year, age, sex, geographical origin, location of lesions, clinical manifestations, risk factors, species isolated and treatments used.
RESULTS
A total of 1505 studies were initially identified. After scrutinizing titles and abstracts, and checking duplications, 221 articles including 406 subjects with AA were included. All were case reports or series. Mean age of subjects was 49.2 years and 56.2% were female. The highest proportion of articles was published between 2015 and 2019 (18.7%). Publications were predominantly from the USA (12.2%). Structures usually involved were abdominal wall, colon and appendix. The most common presentation was abdominal mass (39.2%). In 42.1% of patients, an associated factor was found, highlighting intrauterine devices (14.3%). The microbiology studies highlighted Actinomyces israelli. Morbidity, recurrence and verified mortality were 18.2%, 1.0% and 2.2%, respectively. Penicillin was the most used antibiotic.
CONCLUSION
Evidence about AA is scarce and dispersed within a reduced range of articles and cases.
Topics: Abdominal Wall; Actinomyces; Actinomycosis; Adult; Female; Humans; Intrauterine Devices; Male; Middle Aged; Risk Factors
PubMed: 32729660
DOI: 10.1111/ans.16141 -
Infectious Diseases (London, England) Jul 2023Whipple's disease is an uncommon chronic systemic disease caused by . The most characteristic findings of late Whipple's disease include diarrhoea, abdominal pain,... (Review)
Review
Whipple's disease is an uncommon chronic systemic disease caused by . The most characteristic findings of late Whipple's disease include diarrhoea, abdominal pain, weight loss, and arthralgias, however, other clinical findings can occur, including lymphadenopathy, fever, neurologic manifestations, myocarditis and endocarditis. The aim of the present study was to systematically review all cases of Whipple's disease-associated infective endocarditis (IE) in the literature. A systematic review of PubMed, Scopus, and Cochrane Library (all published studies up to 28 May 2022) for studies providing data on epidemiology, clinical characteristics as well as data on treatment and outcomes of Whipple's disease-associated IE was performed. A total of 72 studies, containing data for 127 patients, were included. A prosthetic valve was present in 8% of patients. The aortic valve was the most commonly involved intracardiac site followed by the mitral valve. Heart failure, embolic phenomena, and fever were the most common clinical presentations, however, fever occurred in less than 30% of patients. Sepsis was rarely noted. The diagnosis was most commonly performed through pathology through positive PCR or histology in cardiac valves in 88.2% of patients. Trimethoprim with sulfamethoxazole were the most commonly used antimicrobials followed by cephalosporins and tetracyclines. Surgery was performed in 84.3% of patients. Mortality was 9.4%. A multivariate logistic regression analysis model identified presentation with sepsis or development of a paravalvular abscess to be independently associated with increased mortality, while treatment with the combination of trimethoprim with sulfamethoxazole was independently associated with reduced mortality.
Topics: Humans; Whipple Disease; Endocarditis, Bacterial; Anti-Bacterial Agents; Trimethoprim; Sulfamethoxazole; Sepsis
PubMed: 37198913
DOI: 10.1080/23744235.2023.2214610 -
Movement Disorders : Official Journal... Nov 2018Whipple's disease, affecting the CNS, can cause a wide variety of symptoms. Movement disorders are very prevalent, and some are pathognomonic of the disease. This... (Review)
Review
Whipple's disease, affecting the CNS, can cause a wide variety of symptoms. Movement disorders are very prevalent, and some are pathognomonic of the disease. This systematic review analyzed all published cases of movement disorders because of CNS Whipple's disease, providing detailed information on clinical and associated features. We have also attempted to address sources of confusion in the literature, particularly related to differing uses of the terminology of movement disorder. This comprehensive overview of Whipple's disease-induced movement disorders aims to aid neurologists in recognizing this very rare disorder and successfully reaching a laboratory-confirmed diagnosis in order to initiate appropriate therapy. © 2018 International Parkinson and Movement Disorder Society.
Topics: Databases, Bibliographic; Humans; Magnetic Resonance Imaging; Male; Middle Aged; Movement Disorders; Ocular Motility Disorders; Whipple Disease
PubMed: 30338868
DOI: 10.1002/mds.27419 -
Practical Neurology Oct 2018A 63-year-old man presented with a 2-month history of progressive right-sided exophthalmos, painful ophthalmoplegia and fevers. As more features developed, he was... (Review)
Review
A 63-year-old man presented with a 2-month history of progressive right-sided exophthalmos, painful ophthalmoplegia and fevers. As more features developed, he was diagnosed with giant cell arteritis, then Tolosa-Hunt syndrome, and transiently responded to corticosteroids. A bland cerebrospinal fluid and highly metabolically active brain (F)-fluoro-D-glucose-positron emission tomography suggested lymphoma. Biopsy of the mass showed sulphur granules with Gram-positive filamentous bacteria with -like colonies. cavernous sinus infections are rare and indolent. They often mimic non-infective causes including other inflammatory and infiltrative conditions, vascular and neoplastic causes, particularly lymphoma. Clinicians should consider infective cavernous sinus syndromes in people with a fluctuating painful ophthalmoplegia that responds poorly to corticosteroids. The term Tolosa-Hunt syndrome is problematic and should be retired or used only with reservation.
Topics: Actinomyces; Actinomycosis; Cavernous Sinus; Humans; Magnetic Resonance Imaging; Male; Middle Aged; Sinusitis
PubMed: 29650638
DOI: 10.1136/practneurol-2017-001844 -
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 -
Pain Mar 2008This systematic literature review aims to assess the prognostic value of psychological factors in the development of late whiplash syndrome (LWS). We included... (Review)
Review
This systematic literature review aims to assess the prognostic value of psychological factors in the development of late whiplash syndrome (LWS). We included prospective cohort studies that provided a baseline measure of at least one psychological variable and used outcome measures relating to LWS (i.e. pain or disability persisting 6 months post injury). A search of electronic databases (Pubmed, Medline, Cinahl, Embase and Psychinfo) up to August 2006 was done using a predetermined search strategy. Methodological quality was assessed independently by two assessors. Data extraction were carried out using a standardised data extraction form. Twenty-five articles representing data from 17 cohorts were included. Fourteen articles were rated as low quality with 11 rated as adequate quality. Meta-analysis was not undertaken due to the heterogeneity of prognostic factors, outcome measures and methods used. Results were tabulated and predefined criterion applied to rate the overall strength of evidence for associations between psychological factors and LWS. Data on 21 possible psychological risk factors were included. The majority of findings were inconclusive. Limited evidence was found to support an association between lower self-efficacy and greater post-traumatic stress with the development of LWS. No association was found between the development of LWS and personality traits, general psychological distress, wellbeing, social support, life control and psychosocial work factors. The lack of conclusive findings and poor methodological quality of the studies reviewed highlights the need for better quality research. Self-efficacy and post-traumatic distress may be associated with the development of LWS but this needs further investigation.
Topics: Community Health Planning; Disability Evaluation; Humans; Prognosis; Prospective Studies; Psychology; Risk Factors; Severity of Illness Index; Statistics as Topic; Whipple Disease
PubMed: 17570588
DOI: 10.1016/j.pain.2007.04.035