Infective Endocarditis – An Observational Study

Abstract Introduction: Despite all the progresses made in the management of infectious and cardiovascular diseases, the incidence of infective endocarditis remains high. Aim: the assessment of etiological, clinical, therapeutic aspects in patients with endocarditis. Material and method: A retrospective observational study in 40 patients with infective endocarditis was conducted, over a period of 5 years. Parameters related to demographic, risk factors, clinical aspects, nature and location of valve damage, bacteriological and therapeutic parameters were assessed. Echocardiography was used to confirm the location of the endocarditis; the aetiology of the disease was identified through the isolation of bacteria from blood cultures by using an automatic BacT/ALERT® system. Results: Patients’ age ranged between 31 - 84 years old. The disease was present predominantly in male patients (67.5%). 70% of the patients were positively diagnosed with endocarditis and 30% with possible endocarditis; the most frequent localization was the native valves in 75% of the cases, compared to the localization in the prosthetic valves, 25%; the aortic valve was involved in 60% of the cases, mitral valve in 40% of the patients. Aetiology of endocarditis was confirmed in 55% of cases as follows: Enterococcus fecalis, speciae, Staphylococcus epidermidis, Escherichia coli, Streptococcus gallolyticus, coagulase-positive methicillin resistant Staphylococcus aureus, coagulasepositive methicillin sensitive Staphylococcus aureus, Staphylococcus lungdunensis, coagulase-negative staphylococci, Streptococcus gordonii, viridans, agalactiae. Antibiotic treatment was administered according to the antibiogram in 55% of the cases. Conclusions: Staphylococcus speciae was the most frequent etiologic agent both in case of native and prosthetic valve endocarditis, with aortic valve predominance.

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Infective endocarditis caused by HACEK group bacteria—a registry-based comparative study

European Journal of Clinical Microbiology & Infectious Diseases ◽

10.1007/s10096-021-04240-3 ◽

2021 ◽

Author(s):

Anna Bläckberg ◽

Christian Morenius ◽

Lars Olaison ◽

Andreas Berge ◽

Magnus Rasmussen

Keyword(s):

Staphylococcus Aureus ◽

Infective Endocarditis ◽

Mortality Rate ◽

Haemophilus Influenzae ◽

Comparative Study ◽

Clinical Characteristics ◽

Prosthetic Valve ◽

Prosthetic Valve Endocarditis ◽

Common Cause ◽

Prosthetic Valves

AbstractInfective endocarditis (IE) caused by bacteria within Haemophilus (excluding Haemophilus influenzae), Aggregatibacter, Cardiobacterium, Eikenella and Kingella (HACEK) is rare. This study aimed to describe clinical features of IE caused by HACEK genera in comparison with IE due to other pathogens. Cases of IE due to HACEK were identified through the Swedish Registry of Infective Endocarditis (SRIE). Clinical characteristics of IE cases caused by HACEK were compared with cases of IE due to other pathogens reported to the same registry. Ninety-six patients with IE caused by HACEK were identified, and this corresponds to 1.8% of all IE cases. Eighty-three cases were definite endocarditis, and the mortality rate was 2%. The median age was 63 years, which was lower compared to patients with IE caused by other pathogens (66, 70 and 73 years respectively, p ≤ 0.01). Patients with IE caused by Haemophilus were younger compared to patients with IE due to Aggregatibacter (47 vs 67 years, p ≤ 0.001). Patients with IE due to HACEK exhibited longer duration from onset of symptoms to hospitalization and had more prosthetic valve endocarditis compared to patients with IE due to Staphylococcus aureus (10 vs 2 days, p ≤ 0.001, and 35 vs 14%, p ≤ 0.001). This is, to date, the largest study on IE due to HACEK. Aggregatibacter was the most common cause of IE within the group. The condition has a subacute onset and often strikes in patients with prosthetic valves, and the mortality rate is relatively low.

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Pooled analysis of 1270 infective endocarditis cases in Turkey

The Journal of Infection in Developing Countries ◽

10.3855/jidc.10056 ◽

2019 ◽

Vol 13 (02) ◽

pp. 93-100 ◽

Cited By ~ 1

Author(s):

Arman Vahabi ◽

Funda Gül ◽

Sabina Garakhanova ◽

Hilal Sipahi ◽

Oğuz Reşat Sipahi

Keyword(s):

Infective Endocarditis ◽

Prosthetic Valve Endocarditis ◽

Pooled Analysis ◽

Etiologic Agent ◽

National Database ◽

Coagulase Negative Staphylococci ◽

History Of ◽

National Databases ◽

Culture Negative ◽

Overall Mortality

Introduction: Despite developments in medicine, infective endocarditis (IE) is still associated with significant morbidity and mortality. In this study it was aimed to systematically review the infective endocarditis literature published or presented from Turkey. Methods: To find the published series, one national database (Ulakbim), and three international databases (Scopus, Pubmed and Sci-e) were searched between 31 October-3 November 2014. also, abstracts of congresses by three national congresses were searched for studies regarding infective endocarditis. Results: Data for 1270 patients (38.3% female, mean age 46.2, 28% prosthetic valve endocarditis) with a diagnosis of infective endocarditis were obtained from 21 reports (18 published articles and three congress abstracts). Of the 18 articles, four were in peer-reviewed medical journals indexed in national databases and 14 were in international databases. There was an underlying heart disease in 51.9% and history of dental procedure was 6.7%. Fever, heart murmur and fatigue were present in 94%, 71.4% and 69% respectively. most commonly involved site was mitral valve (43.3%), followed by aortic (33.8%) and tricuspid valve (6.4%). Staphylococcus aureus, coagulase-negative staphylococci and enterococci comprised the 22.8%, 9.7% and 7.5% of the cases while 31.1% were culture-negative. Overall mortality was 23.4%. When we compared series related to years 2008 and before and 2009 and after, the mortality rates were (24.1%-224/931) vs (20.1%-32/159), respectively (p = 0,31). Conclusion: Infective endocarditis is still associated with significant mortality. S. aureus seems to be the most common etiologic agent. There was a slight decrease in the recent years in mortality.

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P3662Relationships between bacteria and echocardiographic manifestations in infective endocarditis - data from the Swedish endocarditis registry

European Heart Journal ◽

10.1093/eurheartj/ehz745.0517 ◽

2019 ◽

Vol 40 (Supplement_1) ◽

Author(s):

A Damlin ◽

K Westling ◽

E Maret ◽

K Caidahl ◽

M J Eriksson

Keyword(s):

Infective Endocarditis ◽

Mitral Valve ◽

Aortic Valve ◽

Tricuspid Valve ◽

Positive Culture ◽

Group B Streptococci ◽

Coagulase Negative Staphylococci ◽

Pulmonic Valve ◽

Prosthetic Valves ◽

Positive Culture Result

Abstract Introduction Echocardiography (ECHO) is a method of choice for direct visualization of infective endocarditis (IE) - induced cardiac manifestations. Purpose To investigate the correlations between certain bacteria causing the IE and specific IE manifestations diagnosed by ECHO in the population from the Swedish Endocarditis Registry. Methods A cohort study based on selected data from the Swedish Endocarditis Registry including patients aged 18 and above, admitted between 2008 and 2017 with positive culture result from blood or surgical material, in total 570 patients (mean age 58±18 years, 33% female, 67% male patients). For numeric variables, Student's t-test was used. Correlations between the variables were estimated using odds ratio (OR) and 95% confidence intervals (CI). P<0.05 was considered significant. Results Of 570 patients 165 (29%) had a history of drug abuse; 21 (4%) had a bicuspid aortic valve and 110 (19%) had prosthetic valves. Significant correlations were seen between patients with aortic valve IE and coagulase negative staphylococci (OR 2.88, p=0.01, CI 1.26–6.88) and enterococcus spp (OR 2.31, p<0.01, CI 1.30–4.16), specifically enterococcus faecalis (2.45, p<0.01, CI 1.34–4.54), mitral valve IE and streptococcus spp (OR 1.92, p<0.01, CI 1.31–2.83), specifically group B streptococci (OR 8.16, p<0.01, CI 1.71–77.03), tricuspid valve IE (OR 6.55, p<0.01, CI 3.93–11.23) and pulmonic valve IE (OR 7.53, p=0.03, CI 1.0–335.25), and staphylococcus spp, specifically staphylococcus aureus (tricuspid valve: OR 6.55, p<0.01, CI 3.93–11.23 and pulmonic valve: OR 9.26, p=0.01, CI 1.22–412.22), abscess and coagulase negative staphylococci (OR 4.13, p<0.01, CI 1.13–12.25), pacemaker or ICD-associated IE and HACEK (OR 7.60, p=0.01, CI 1.66–27.06). Conclusions Significant correlations were found between certain bacteria and specific IE manifestations detected by ECHO, i.e. on aortic valve, mitral valve, tricuspid valve or pulmonic valve, abscess and pacemaker or ICD-associated IE.

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Homograft versus Conventional Prosthesis for Surgical Management of Aortic Valve Infective Endocarditis

Innovations Technology and Techniques in Cardiothoracic and Vascular Surgery ◽

10.1097/imi.0000000000000510 ◽

2018 ◽

Vol 13 (3) ◽

pp. 163-170 ◽

Cited By ~ 5

Author(s):

Bobby Yanagawa ◽

Amine Mazine ◽

Derrick Y. Tam ◽

Peter Jüni ◽

Deepak L. Bhatt ◽

...

Keyword(s):

Infective Endocarditis ◽

Aortic Valve ◽

Surgical Management ◽

Meta Analysis ◽

Prosthetic Valve Endocarditis ◽

Prosthetic Valves ◽

Mechanical Valves ◽

All Cause Mortality ◽

Mortality Incidence ◽

Similar Survival

Objective Surgical management of aortic valve infective endocarditis (IE) with cryopreserved homograft has been associated with lower risk of recurrent IE, but there is equipoise with regard to the optimal prosthesis. This systematic review and meta-analysis were performed to compare outcomes between homograft and conventional prosthesis for aortic valve IE. Methods We searched MEDLINE database to September 2017 for studies comparing homograft versus conventional prosthesis. The main outcomes were all-cause mortality, recurrent IE, and reoperation. Results There were 18 included comparative observational studies with 2232 patients (median follow up = 5 [interquartile range: 2–7] years, 30% prosthetic valve endocarditis); four studies were adjusted for baseline differences. There were no differences in perioperative mortality or stroke despite a greater proportion of staphylococcal endocarditis, abscess, and root replacements but less multivalve involvement in the homograft group. Long-term outcomes of all-cause mortality [incidence rate ratio (IRR) = 1.03, 95% confidence interval (CI) = 0.81–1.31, P = 0.83, for unmatched, and IRR = 0.82, 95% CI = 0.36–1.84, P = 0.63, for matched studies], recurrent endocarditis (IRR = 1.01, 95% CI = 0.53–1.93, P = 0.96, for unmatched, and IRR = 1.04, 95% CI = 0.49–2.19, P = 0.92, for matched studies), and reoperation (IRR = 1.60, 95% CI = 0.80–3.21, P = 0.18, for unmatched, and IRR = 3.17, 95% CI = 0.52–19.44, P = 0.21, for matched studies) were not different comparing homograft versus conventional prosthesis. There was a significantly increased need for reoperation with homograft versus mechanical prosthetic valves, but this comparison was based on limited data. Conclusions Homografts and conventional prostheses offer similar survival and freedom from recurrent endocarditis and reoperation for aortic valve IE. Homografts may be associated with greater risk of reoperation compared with mechanical valves.

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The Role of the Coagulase-negative Staphylococci (CoNS) in Infective Endocarditis; A Narrative Review from 2000 to 2020

Current Pharmaceutical Biotechnology ◽

10.2174/1389201021666200423110359 ◽

2020 ◽

Vol 21 (12) ◽

pp. 1140-1153 ◽

Cited By ~ 1

Author(s):

Mohammad A. Noshak ◽

Mohammad A. Rezaee ◽

Alka Hasani ◽

Mehdi Mirzaii

Keyword(s):

Foreign Body ◽

Infective Endocarditis ◽

Prosthetic Valve ◽

Human Life ◽

Prosthetic Valve Endocarditis ◽

Surgical Site Infections ◽

Coagulase Negative Staphylococci ◽

Native Valve ◽

Native Valve Endocarditis

Coagulase-negative staphylococci (CoNS) are part of the microbiota of human skin and rarely linked with soft tissue infections. In recent years, CoNS species considered as one of the major nosocomial pathogens and can cause several infections such as catheter-acquired sepsis, skin infection, urinary tract infection, endophthalmitis, central nervous system shunt infection, surgical site infections, and foreign body infection. These microorganisms have a significant impact on human life and health and, as typical opportunists, cause peritonitis in individuals undergoing peritoneal dialysis. Moreover, it is revealed that these potential pathogens are mainly related to the use of indwelling or implanted in a foreign body and cause infective endocarditis (both native valve endocarditis and prosthetic valve endocarditis) in patients. In general, approximately eight percent of all cases of native valve endocarditis is associated with CoNS species, and these organisms cause death in 25% of all native valve endocarditis cases. Moreover, it is revealed that methicillin-resistant CoNS species cause 60 % of all prosthetic valve endocarditis cases. In this review, we describe the role of the CoNS species in infective endocarditis, and we explicated the reported cases of CoNS infective endocarditis in the literature from 2000 to 2020 to determine the role of CoNS in the process of infective endocarditis.

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Infective Endocarditis Due to Corynebacterium Species: Clinical Features and Antibiotic Resistance

Open Forum Infectious Diseases ◽

10.1093/ofid/ofab055 ◽

2021 ◽

Vol 8 (3) ◽

Author(s):

Anna Bläckberg ◽

Linn Falk ◽

Karl Oldberg ◽

Lars Olaison ◽

Magnus Rasmussen

Keyword(s):

Antibiotic Resistance ◽

Infective Endocarditis ◽

Prosthetic Valve Endocarditis ◽

Penicillin G ◽

Severe Condition ◽

Ionization Time ◽

Prosthetic Valves ◽

Corynebacterium Species ◽

Corynebacterium Striatum

Abstract Background Corynebacterium species are often dismissed as contaminants in blood cultures, but they can also cause infective endocarditis (IE), which is a severe condition. Antibiotic resistance of corynebacteria is increasing making treatment challenging. Reports on IE caused by Corynebacterium species are scarce and more knowledge is needed. Methods Cases of IE caused by Corynebacterium species were identified through the Swedish Registry of Infective Endocarditis. Isolates were collected for species redetermination by matrix-assisted laser desorption ionization-time of flight and for antibiotic susceptibility testing using Etests. Results Thirty episodes of IE due to Corynebacterium species were identified between 2008 and 2017. The median age of patients was 71 years (interquartile range, 60–76) and 77% were male. Corynebacterium striatum (n = 11) was the most common IE causing pathogen followed by Corynebacterium jeikeium (n = 5). Surgery was performed in 50% and in-hospital mortality rate was 13%. Patients with IE caused by Corynebacterium species were significantly more likely to have prosthetic valve endocarditis (70%), compared with patients with IE due to Staphylococcus aureus or non-beta-hemolytic streptococci (14% and 26%, respectively) (P < .0001). Vancomycin was active towards all Corynebacterium isolates, whereas resistance towards penicillin G was common. Conclusions Corynebacterium species cause IE, where prosthetic valves are mainly affected and surgery is often performed. Corynebacterium striatum is an important causative agent of IE within the genus. Antibiotic resistance of corynebacteria is relatively common but resistance towards vancomycin could not be detected in vitro.

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18F-FDG PET/CT in Infective Endocarditis: Indications and Approaches for Standardization

Current Cardiology Reports ◽

10.1007/s11886-021-01542-y ◽

2021 ◽

Vol 23 (9) ◽

Author(s):

D. ten Hove ◽

R.H.J.A. Slart ◽

B. Sinha ◽

A.W.J.M. Glaudemans ◽

R.P.J. Budde

Keyword(s):

Infective Endocarditis ◽

Fdg Pet ◽

Prosthetic Valve Endocarditis ◽

Cardiac Device ◽

Native Valve ◽

Prosthetic Valves ◽

Cardiac Devices ◽

Pet Ct ◽

Fdg Pet Ct ◽

Disseminated Disease

Abstract Purpose of Review Additional imaging modalities, such as FDG-PET/CT, have been included into the workup for patients with suspected infective endocarditis, according to major international guidelines published in 2015. The purpose of this review is to give an overview of FDG-PET/CT indications and standardized approaches in the setting of suspected infective endocarditis. Recent Findings There are two main indications for performing FDG-PET/CT in patients with suspected infective endocarditis: (i) detecting intracardiac infections and (ii) detection of (clinically silent) disseminated infectious disease. The diagnostic performance of FDG-PET/CT for intracardiac lesions depends on the presence of native valves, prosthetic valves, or implanted cardiac devices, with a sensitivity that is poor for native valve endocarditis and cardiac device-related lead infections, but much better for prosthetic valve endocarditis and cardiac device-related pocket infections. Specificity is high for all these indications. The detection of disseminated disease may also help establish the diagnosis and/or impact patient management. Summary Based on current evidence, FDG-PET/CT should be considered for detection of disseminated disease in suspected endocarditis. Absence of intracardiac lesions on FDG-PET/CT cannot rule out native valve endocarditis, but positive findings strongly support the diagnosis. For prosthetic valve endocarditis, standard use of FDG-PET/CT is recommended because of its high sensitivity and specificity. For implanted cardiac devices, FDG-PET/CT is also recommended, but should be evaluated with careful attention to clinical context, because its sensitivity is high for pocket infections, but low for lead infections. In patients with prosthetic valves with or without additional aortic prosthesis, combination with CTA should be considered. Optimal timing of FDG-PET/CT is important, both during clinical workup and technically (i.e., post tracer injection). In addition, procedural standardization is key and encompasses patient preparation, scan acquisition, reconstruction, subsequent analysis, and clinical interpretation. The recommendations discussed here will hopefully contribute to improved standardization and enhanced performance of FDG-PET/CT in the clinical management of patients with suspected infective endocarditis.

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Characterization of Staphylococci and Streptococci Isolated from Milk of Bovides with Mastitis in Egypt

Pathogens ◽

10.3390/pathogens9050381 ◽

2020 ◽

Vol 9 (5) ◽

pp. 381

Author(s):

Wedad Ahmed ◽

Heinrich Neubauer ◽

Herbert Tomaso ◽

Fatma Ibrahim El Hofy ◽

Stefan Monecke ◽

...

Keyword(s):

Staphylococcus Aureus ◽

Microarray Analysis ◽

Streptococcus Agalactiae ◽

Nile Delta ◽

Meca Gene ◽

Resistance Rate ◽

Coagulase Negative Staphylococci ◽

Flight Mass Spectrometry ◽

Streptococcus Gallolyticus ◽

Broth Microdilution Method

The aim of this study was to characterize staphylococci and streptococci in milk from Egyptian bovides. In total, 50 milk samples were collected from localities in the Nile Delta region of Egypt. Isolates were cultivated, identified using matrix-assisted laser desorption/ionization time-of-flight mass spectrometry (MALDI-TOF MS), and antibiotic susceptibility testing was performed by the broth microdilution method. PCR amplifications were carried out, targeting resistance-associated genes. Thirty-eight Staphylococcus isolates and six Streptococcus isolates could be cultivated. Staphylococcus aureus isolates revealed a high resistance rate to penicillin, ampicillin, clindamycin, and erythromycin. The mecA gene defining methicillin-resistant Staphylococcus aureus, erm(C) and aac-aphD genes was found in 87.5% of each. Coagulase-negative staphylococci showed a high prevalence of mecA, blaZ and tetK genes. Other resistance-associated genes were found. All Streptococcus dysgalactiae isolates carried blaZ, erm(A), erm(B), erm(C) and lnuA genes, while Streptococcus suis harbored erm(C), aphA-3, tetL and tetM genes, additionally. In Streptococcus gallolyticus, most of these genes were found. The Streptococcus agalactiae isolate harbored blaZ, erm(B), erm(C), lnuA, tetK, tetL and tetM genes. Streptococcus agalactiae isolate was analyzed by DNA microarray analysis. It was determined as sequence type 14, belonging to clonal complex 19 and represented capsule type VI. Pilus and cell wall protein genes, pavA, cadD and emrB/qacA genes were identified by microarray analysis.

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1075. A Retrospective Comparison of Native Valve Endocarditis and Prosthetic Valve Endocarditis in a Large Tertiary Care Teaching Hospital From 2007 to 2015

Open Forum Infectious Diseases ◽

10.1093/ofid/ofy210.912 ◽

2018 ◽

Vol 5 (suppl_1) ◽

pp. S322-S322

Author(s):

Hoi Yee Annie Lo ◽

Anahita Mostaghim ◽

Nancy Khardori

Keyword(s):

Risk Factors ◽

High Risk ◽

Aortic Valve ◽

Teaching Hospital ◽

Prosthetic Valve ◽

Prosthetic Valve Endocarditis ◽

Native Valve ◽

Prosthetic Valves ◽

Dental Procedures ◽

History Of

Abstract Background Studies comparing native valve and prosthetic valve endocarditis (NVE and PVE) have mixed findings on the risk factors and outcomes between the two cohorts. This retrospective review of infective endocarditis (IE) at a teaching hospital in the United States aims to compare the clinical and microbiological features between NVE and PVE. Methods Patients were retrospectively identified from 2007 to 2015 using appropriate IE-related ICD-9 codes. Cases that met definite Modified Duke Criteria for IE were further classified as either PVE or NVE, and were reviewed for epidemiology, causative organism(s), affected valves and associations, risk factors, dental procedures in the past 6 months, and 30-day mortality. Results A total of 363 admissions met criteria for definite endocarditis, with 261 NVE cases and 59 PVE cases. Forty-three cases that were either associated with an infection involving both native and prosthetic valves or intracardiac devices were omitted from this study. Most risk factors, such as hemodialysis and intravenous drug use, did not show any significant difference amongst the two groups. IE involving the aortic valve as well as a previous history of IE were more likely to be seen in PVE (both P < 0.0001). Dental procedures done in the preceding 6 months before IE admission were more likely to be associated with PVE than NVE (P = 0.0043). PVE showed a higher likelihood of 30-day mortality compared with NVE (P = 0.067). The causative organisms of PVE were more likely to be caused by common gut pathogens such as Klebsiella and Enterobacter species. Conclusion PVE cases had a significantly higher chance of involving the aortic valve as well as having a history of IE. PVE cases were also significantly more likely to be associated with a dental procedure done in the preceding 6 months than with the NVE cases. This implies that patients with prosthetic valves, who are currently covered under the 2007 AHA guidelines to receive prophylaxis prior to dental procedures, are still at a high risk of developing PVE. It may be prudent to reconsider adding a post-procedure dose of antibiotics, instead of a single preprocedure dose, to extend the protection of this high-risk population with prosthetic valves. Furthermore, PVE cases showed higher rates of 30-day mortality compared with NVE with near significance, which is likely multifactorial. Disclosures All authors: No reported disclosures.

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Infective endocarditis complicating transcatheter aortic valve implantation

Heart ◽

10.1136/heartjnl-2019-315338 ◽

2020 ◽

Vol 106 (7) ◽

pp. 493-498 ◽

Cited By ~ 2

Author(s):

Daniel Harding ◽

Thomas J Cahill ◽

Simon R Redwood ◽

Bernard D Prendergast

Keyword(s):

Infective Endocarditis ◽

Aortic Valve ◽

Transcatheter Aortic Valve Implantation ◽

Surgical Intervention ◽

Imaging Techniques ◽

Prosthetic Valve Endocarditis ◽

Low Risk ◽

Transcatheter Aortic Valve ◽

Aortic Valve Implantation ◽

Valve Implantation

Infective endocarditis complicating transcatheter aortic valve implantation (TAVI-IE) is a relatively rare condition with an incidence of 0.2%–3.1% at 1 year post implant. It is frequently caused by Enterococci, Staphylococcus aureus and coagulase negative staphylococci. While the incidence currently appears to be falling, the absolute number of cases is likely to rise substantially as TAVI expands into low risk populations following the publication of the PARTNER 3 and Evolut Low Risk trials. Important risk factors for the development of TAVI-IE include a younger age at implant and significant residual aortic regurgitation. The echocardiographic diagnosis of TAVI-IE can be challenging, and the role of supplementary imaging techniques including multislice computed tomography (MSCT) and positron emission tomography (18FDG PET) is still emerging. Treatment largely parallels that of conventional prosthetic valve endocarditis (PVE), with prolonged intravenous antibiotic therapy and consideration of surgical intervention forming the cornerstones of management. The precise role and timing of cardiac surgery in TAVI-IE is yet to be defined, with a lack of clear evidence to help identify which patients should be offered surgical intervention. Minimising unnecessary healthcare interventions (both during and after TAVI) and utilising appropriate antibiotic prophylaxis may have a role in preventing TAVI-IE, but robust evidence for specific preventative strategies is lacking. Further research is required to better select patients for advanced hybrid imaging, to guide surgical management and to inform prevention in this challenging patient cohort.

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