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Background:
Systematic Review

Lomentospora prolificans Disseminated Infections: A Systematic Review of Reported Cases

by
Afroditi Konsoula
1,
Aris P. Agouridis
2,3,
Lamprini Markaki
4,
Constantinos Tsioutis
2 and
Nikolaos Spernovasilis
5,6,*
1
Department of Pediatrics, General Hospital of Sitia, 72300 Sitia, Greece
2
School of Medicine, European University Cyprus, 2404 Nicosia, Cyprus
3
Department of Internal Medicine, German Oncology Center, 4108 Limassol, Cyprus
4
“Iliaktida” Pediatric & Adolescents Medical Center, 4001 Limassol, Cyprus
5
Department of Infectious Diseases, German Oncology Center, 4108 Limassol, Cyprus
6
School of Medicine, University of Crete, 71303 Heraklion, Greece
*
Author to whom correspondence should be addressed.
Pathogens 2023, 12(1), 67; https://doi.org/10.3390/pathogens12010067
Submission received: 29 November 2022 / Revised: 26 December 2022 / Accepted: 28 December 2022 / Published: 31 December 2022
(This article belongs to the Special Issue Opportunistic Fungal Infections)
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Abstract

:
Background: Lomentospora prolificans, a rare, highly virulent filamentous fungus with high rates of intrinsic resistance to antifungals, has been associated with different types of infections in immunocompromised as well as immunocompetent individuals. Objective: To systematically address all relevant evidence regarding L. prolificans disseminated infections in the literature. Methods: We searched Medline via PubMed and Scopus databases through July 2022. We performed a qualitative synthesis of published articles reporting disseminated infections from L. prolificans in humans. Results: A total of 87 studies describing 142 cases were included in our systematic review. The pathogen was most frequently reported in disseminated infections in Spain (n = 47), Australia (n = 33), the USA (n = 21), and Germany (n = 10). Among 142 reported cases, 48.5% were males. Underlying conditions identified for the majority of patients included malignancy (72.5%), hemopoietic stem cell transplantation (23.2%), solid organ transplantation (16%), and AIDS (2%). Lungs, central nervous system, skin, eyes, heart and bones/joints were the most commonly affected organs. Neutropenia was recorded in 52% of patients. The mortality rate was as high as 87.3%. Conclusions: To the best of our knowledge, this is the first systematic review conducted on disseminated infections due to this rare microorganism. Physicians should be aware that L. prolificans can cause a diversity of infections with high mortality and primarily affects immunocompromised and neutropenic patients.

1. Introduction

Lomentospora prolificans, formerly known as Scedosporium prolificans or Scedosporium inflatum, is a rare emerging opportunistic pathogen that primarily affects immunocompromised individuals but can also cause infections in healthy populations [1]. It is found in the environment, including soil, decaying organic matter, and contaminated water [2,3]. The first report as a pathogen in humans was in 1984, when Malloch and Salkin isolated this fungus from an immunocompetent patient with osteomyelitis [4].
L. prolificans can grow on standard mycological media such as Sabouraud’s dextrose agar (SDA) or potato dextrose agar (PDA) [5]. Characteristic macroscopic features include olive-gray to black colony morphology and susceptibility to cycloheximide [6]. Microscopic features that may indicate the presence of L. prolificans include visualization of flask-shaped conidiophores which are inflated or swollen at the base, from which single, or clusters of, conidia emerge [6].
L. prolificans infection causes a wide range of clinical manifestations from localized to disseminated infections, depending on the immune status of the infected individual [7]. Disseminated infection usually affects immunocompromised hosts and is accompanied with a high mortality rate, as highlighted in previous reviews [1,8].
L. prolificans is increasingly recognized as a cause of invasive fungal infection in geographic areas such as Australia [9], the United States [10,11], and some parts of Europe [12,13,14]. High rates of intrinsic resistance to several antifungals reduce the possibility of successful recovery [15]. The lack of or difficult access to rapid species-specific diagnostic methods further complicates the treatment of this infection [16].
Herein, we systematically address the literature on all relevant cases of disseminated infections caused by L. prolificans in humans.

2. Materials and Methods

2.1. Study Design

The purpose of this systematic review is to evaluate and better understand the clinical profile and pathogenicity of disseminated infections caused by L. prolificans.
We performed a qualitative synthesis of published articles reporting disseminated infection from L. prolificans in humans.

2.2. Search Strategy

An extensive bibliographic search of Medline via PubMed and Scopus databases was conducted from inception until 31 July 2022. Only articles published in English were included. Initial searches were performed using the following search terms: “(Lomentospora prolificans) OR (Scedosporium prolificans) OR (Scedosporium inflatum)”. Additional studies were identified from the references provided by retrieved studies.

2.3. Inclusion and Exclusion Criteria

The inclusion criteria for our systematic review included articles that contained at least one case of disseminated infection with L. prolificans. Disseminated infection was defined as (1) clinical syndrome consistent with infection and (2) recovery of the isolate from blood samples or microbiological and/or pathological evidence of infection at ≥ 2 noncontiguous sites. Only papers based on humans and written in English were considered eligible.
Studies were excluded if they did not fulfil inclusion criteria; if they reported only localized infection by L. prolificans; or if the infections were not in humans.

2.4. Data Extraction

Studies were independently and thoroughly examined by two investigators (A.K., A.P.A.) and studies’ characteristics (author, year, study design, country, patient age/sex, underlying disease/conditions, clinical manifestations, sample, treatment, outcome) were extracted. Any discrepancy between the reviewers was resolved by consensus. For the review of our analysis, which was designed according to the guidelines of 2020 [17], data extraction was performed with adherence to Preferred Reporting Items for Systematic reviews and Meta-Analysis (PRISMA model). Due to the study design, no institutional Review Board approval was obtained.

2.5. Assessment of Risk of Bias

A systematic assessment of bias in the included studies was performed using the Joanna Briggs Institute (JBI) critical appraisal checklist for case reports [18]. The items used for the assessment of each study were as follows: patient’s demographic characteristics, patient’s history, patient’s current clinical condition, diagnostic tests or assessment methods and the results, the intervention(s) or treatment procedure(s), post-intervention clinical condition, adverse events (harms) or unanticipated events, takeaway lessons. According to the recommendations of the JBI tool, a judgment of “1” indicates low risk of bias, whereas a “0” on any of the included questions negatively affects the overall quality the case reports. An overall score ≤ 49% equals with high risk of bias, 50% to 69% equals with moderate risk of bias, and ≥ 70% equals with low risk of bias. Risk-of-bias assessment was performed independently by 2 reviewers (A.K., A.P.A.); disagreements were resolved by consensus.

2.6. Statistical Analysis

Associations of survival with surgery and neutropenic/immunosuppressant patients were assessed using the Chi-square test (χ2). Statistical significance was set at 5% significance level (p < 0.05). Data were processed and analyzed using IBM SPSS Statistics for Windows, Version 29.0 (Armonk, NY, USA: IBM Corp, USA).

3. Results

3.1. Study Selection

In Figure 1, the PRISMA flow chart reveals the selection process of included studies. With the above-mentioned search terms, we identified 1373 records on Medline via PubMed and 495 additional records on Scopus. After detecting and removing duplicates, 1494 articles remained, among which we initially excluded 1394 because of study design. Subsequently, we examined in detail the remaining 100 articles. Among them, 13 studies were rejected because selection criteria were not met (Supplementary Table S1 and Figure 1). Finally, 87 studies with a total of 142 cases (patients with disseminated L. prolificans infection) were included in our systematic review.

3.2. Study Characteristics

The included studies were published between 1990 and 2022 (Table 1). A total of 142 individual cases from 87 publications of disseminated infection by L. prolificans fulfilled the inclusion criteria. Studies were more frequently reported in Spain (n = 41), Australia (n = 33), the USA (n = 21), Germany (n = 10), Japan (n = 8), USA/Spain (n = 6), France (n = 6), Mexico (n = 6), The Netherlands (n = 2), Canada (n = 2), South Korea (n = 1), Italy (n = 1), Brazil (n = 1), Belgium (n = 1), Thailand (n = 1), Poland (n = 1), and India (n = 1). Among a total of 127 adults, 5 children (defined as patients <16 years old), and 10 patients whose age was not specified, males represented 48.5% and females 45%, while in 6.3% sex was not mentioned. Underlying conditions, identified for the majority of patients, included malignancy (72.5%), hemopoietic stem cell transplantation (HSCT) (23.2%), solid organ transplantation (16%), and AIDS (2%). No underlying condition was reported in four patients. Neutropenia was recorded in 52% of patients. Lungs, central nervous system (CNS), skin, eyes, heart and bones/joints were the most commonly affected organs. Blood cultures were positive in 107 of 142 (75.3%) patients. The majority of patients systematically received amphotericin B, voriconazole, terbinafine, itraconazole, and fluconazole either as monotherapy or in combination therapy. The overall mortality rate was 87.3% (Table 1).

3.3. Clinical Outcomes

After performing the Chi-Square test, an association between surgery and survival was observed (Pearson Chi-Square = 21.044, p < 0.001). More specifically, patients who underwent surgery had a 11.329 times higher probability of surviving [95% CI, (3.388–37.881)]. Moreover, we found that immunocompetent patients had a 10.3 [95% CI, (1.333–83.333)] higher probability of surviving compared with neutropenic/immunosuppressant patients (Pearson Chi-Square = 7.320, p = 0.05).

3.4. Quality Appraisal

The overall quality was very good, as 72 articles had a low risk of bias, while 9 studies had a high risk of bias and 6 studies had a moderate risk of bias. Quality appraisal results are presented in Supplementary Table S2.

4. Discussion

The current systematic review focuses on disseminated infections caused by L. prolificans in humans. To the best of our knowledge, this is the first systematic review conducted on disseminated infections due to this rare microorganism.
L. prolificans is a rare filamentous fungus found primarily in the environment, including soil, decaying organic matter, and contaminated water [2,3]. Regarding the epidemiology of L. prolificans disseminated infection, cases were initially reported in the dry climates of Spain, Australia and the southwestern United States. Recently, however, there have been publications from other countries, specifically Germany, Japan, France, Mexico, The Netherlands, Canada, South Korea, Italy, Brazil, Belgium, Thailand, Poland, and India (Figure 2). Excluded studies due to different language concern cases reported in the aforementioned countries (Supplementary material).
This pathogen can infect both immunocompetent and immunocompromised patients and thus acts both as a primary and an opportunistic pathogen [100]. Skin, soft tissue, muscle, bone, and joint infections are more common in immunocompetent hosts, and infection usually requires disruption of the anatomic barrier by trauma, surgery, or corticosteroid injections [1,101]. Almost all cases presented in this review involve diseases and conditions indicative of severe immunosuppression. Airway colonization is common in patients with cystic fibrosis and lung transplantation [1,102,103]. Structural changes in the airways, long-term immunosuppression, and previous exposure to antifungal drugs contribute to the higher prevalence of L. prolificans in these patient populations [102,103,104].
Disseminated infection is the most common pattern of L. prolificans infection reported, and is associated with very high mortality rate, as shown in our systematic review. Risk factors for dissemination include solid organ transplantation, HSCT, malignancies (especially hematologic), AIDS, neutropenia, and immunosuppressive therapy [1,8,105,106]. The primary location of the fungus, the degree of immunosuppression, and the speed of disease progression determine the clinical outcome. Primary location of the fungus, such as eyes, joint, bone, and skin plays an important role in clinical outcome, since resection of surgically amenable lesions is significantly associated with improved survival [105,107]. This comes in agreement with our results, since those patients who underwent surgery had higher survival rate. The most frequent clinical manifestations of disseminated disease include fever and CNS, heart and/or respiratory involvement, along with skin lesions, particularly numerous erythematous non-pruritic skin nodules with or without a necrotic center [1,7,44].
Several determinants of pathogenesis have a role in the manifestation of disease [100], associated with germination [108], biofilm formation [109], destruction of lung epithelial cells [109], and infiltration of blood vessels [110], resulting in widespread dissemination to distal organs [110]. Important molecules in the fungal cell wall that enhance fungal virulence include peptidorhamnomannan, glucosylceramide, and melanin [111]. The susceptibility of this fungus to innate immunity, particularly to neutrophils, may explain the high rate of prevalence in neutropenic patients [106]. Therefore, correction of neutropenia is of paramount importance, associated with a favorable outcome [26]. At the same time, a weak innate systemic response of microglial cells in the CNS explains the propensity of this fungus to invade and live in the CNS, a phenomenon known as neurotropism [112]. Detection of L. prolificans in clinical specimens relies principally on direct microscopic examination of fresh specimens or histopathologic analysis, together with culture on appropriate culture media [5]. Histopathologic examination can provide valuable evidence of invasive disease, but culture is necessary because different molds share the same characteristics under the microscope [5]. Direct microscopy and culture are the cornerstone of proven fungal infection [113]. A positive culture from the respiratory system in the absence of radiologic or endobronchial changes may indicate colonization [114]. Disseminated infection can be detected with blood cultures. Positive blood culture is rare in most molds, except those capable of angioinvasion with widespread dissemination, such as Scedosporium/Lomentospora and Fusarium species, and zygomycetes such as Rhizopus and Mucor [110] As shown in this systematic review, blood cultures were positive in 107 of 142 (75.3%) patients. However, their diagnostic utility is limited because most blood cultures become positive late in the course of the disease due to slow growth of the microorganism [1]. Molecular techniques, such as PCR, either panfungal or species-specific, followed by DNA sequencing, can detect invasive fungal infections directly from fresh and formalin-fixed paraffin-embedded (FFPE) material, but only in conjunction with histopathologic examination [115,116,117]. Several case reports have mentioned high serum 1, 3-beta-D-glucan (BDG) levels in patients with L. prolificans infection [51,80], while some other reports, mentioned low serum BDG levels [118]. Hence, although this panfungal biomarker (BDG) may be useful in diagnosis when invasive fungal infection is suspected [5], its clinical utility is controversial. Therefore, results should always be interpreted in conjunction with the other diagnostic methods mentioned above. Matrix-assisted laser desorption/ionization time-of-flight is rapid and reliable method for identifying L. prolificans, but is used by only few laboratories [119,120].
Treatment of L. prolificans infection is challenging because this fungus has intrinsic resistance to most antifungal agents used in clinical practice. The treatment strategy for disseminated disease includes a combination of surgical and antifungal therapy, as well as correction of underlying immune deficiencies [121]. Once invasive L. prolificans is suspected or confirmed, surgical removal of infected tissue should be initiated if feasible [121]. Current clinical practice guidelines recommend that first-line antifungal treatment with voriconazole and terbinafine plus or minus other antifungal agents over a period of at least 4 to 6 months is associated with a favorable outcome [121]. According to Jenks et al., combination therapy with voriconazole plus terbinafine may be associated with improved treatment outcomes compared with other antifungal regimens for the treatment of invasive L. prolificans infections [122]. Clinical evaluation, laboratory studies (inflammatory markers, microbiologic studies), and imaging should be reviewed frequently to assess respond to treatment. Frequency depends on the concomitant conditions, disease severity and initial response to treatment.
Inherent resistance to most available treatments raises the need for new classes of antifungal agents [123]. Olorofim, a key enzyme in the biosynthesis of pyrimidines, has the ability to inhibit dihydroorotate dehydrogenase [124]. It is currently in Phase IIB clinical trials for the treatment of invasive mold infections, including L. prolificans, in patients with limited treatment options [124]. The efficacy of olorofim has been demonstrated in in vitro studies and improved clinical outcomes have been observed in two case reports [124,125,126].
This study has several limitations. It was not possible to perform a meta-analysis because all data are based on case reports and small case series. The above limitations could have affected the quality of our findings and conclusions. However, by using the JBI critical appraisal checklist for each article included in our systematic review, we attempted to minimize the risk of bias and increase quality. The geographic distribution of publications that were included in our review probably reflects research and clinical interest rather than presence of the fungus only in these areas and environments. Finally, despite the high number of titles analyzed in our review, several studies on invasive infections by L. prolificans were excluded, as they did not fulfil inclusion criteria. Although excluded, these studies provide important clinical information on these infections [8,102,107,122].

5. Conclusions

Disseminated disease caused by L. prolificans is a rare infection with significant mortality, and should be suspected especially in immunocompromised and neutropenic patients. Early diagnosis and careful interpretation of culture results are important in the management of these patients. Novel antifungal agents and further exploration of therapeutic options are needed to improve the outcome of this highly fatal infection. Healthcare providers treating patients with disseminated fungal infection should be aware of this life-threatening pathogen.

Supplementary Materials

The following supporting information can be downloaded at: https://www.mdpi.com/article/10.3390/pathogens12010067/s1. Table S1: Reasons for exclusion of studies from the systematic review; Table S2: Reported cases and their risk of bias according to the Joanna Briggs Institute (JBI) Critical Appraisal Checklist for Case Reports.

Author Contributions

Conceptualization, A.P.A., C.T., and N.S.; methodology, A.K. and A.P.A.; formal analysis, A.K. and A.P.A.; investigation, A.K., A.P.A. and L.M.; writing—original draft preparation, A.K., A.P.A., and C.T.; writing—review and editing, A.P.A., L.M., and N.S.; supervision, N.S. All authors have read and agreed to the published version of the manuscript.

Funding

This research received no external funding.

Acknowledgments

The authors would like to acknowledge Ognyan Iskrenov for his assistance with Figure 2.

Conflicts of Interest

The authors declare no conflict of interest.

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Pathogens 12 00067 g001 550
Figure 1. PRISMA flow diagram of article selection process.
Figure 1. PRISMA flow diagram of article selection process.
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Pathogens 12 00067 g002 550
Figure 2. Worldwide distribution of L. prolificans disseminated disease.
Figure 2. Worldwide distribution of L. prolificans disseminated disease.
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Table
Table 1. Study characteristics of Lomentospora prolificans infections reported in the literature.
Table 1. Study characteristics of Lomentospora prolificans infections reported in the literature.
AuthorYearStudy DesignCountryPatient Age/SexUnderlying Disease/ConditionsClinical ManifestationsSampleTreatmentOutcome
Aldoss [19]2019Retrospective cohort studyUSA55/FAML, alloHSCTFungemiaBlood culturePOSNA
Alvarez [14]1995Case seriesSpain27/FAMLPneumonia, fungemiaBlood cultureAMBDied
Alvarez [14]1995Case seriesSpain45/FAML, neutropeniaPneumonia, fungemiaBlood cultureAMBDied
Alvarez [14]1995Case seriesSpain79/FALL, neutropeniaPneumonia, pleural effusion, fungemiaBlood cultureAMBDied
Alvarez [14]1995Case seriesSpain54/FAML, neutropeniaPneumonia, fungemiaBlood, tracheal aspirate cultureAMBDied
Álvarez-Uría [20]2020Case seriesSpain25/MHeart transplantationFungemia, CNS, skin, lung involvement Blood, skin, sputum cultureVRC + TRBDied
Ananda-Rajah [21]2008Case reportAustralia58/MALL, neutropeniaPneumonia, fungemia, embolic skin lesionsBlood cultureVRC + TRBDied
Balandin [22]2016Case reportSpain27/MCF, lung transplantationPneumonia, pleural empyema, pulmonary embolism, mycotic emboliBAL, pleural fluid culture, thrombus sample with fungal elementsVRC + TRB + CAS + intrapleural/neb VRC POS + MTF +ANFDied
Barbaric [23]2001Case seriesAustralia10/FALL, neutropeniaPneumonia, fungemia, skin lesionsSkin biopsy, catheter tip, blood cultureAMB + G-CSFDied
Berenguer [12] 1997Case seriesSpain56/MAcute leukaemia, neutropeniaPneumonia, fungemiaBlood, respiratory culturesAMB + ITCDied
Berenguer [12] 1997Case seriesSpain52/MAcute leukaemia, neutropeniaFungemia, lung, eye involvement Blood cultureFLCDied
Berenguer [12]1997Case seriesSpain48/MAcute leukaemia, neutropeniaPneumonia, skin lesionsSkin, bone cultureAMB + FLC + SurgerySurvived
Boan [24]2020Case seriesAustralia71/FCLLPneumoniaUrine, sputum cultureVRC + TRB + ANF + L-AMBDied
Boan [24]2020Case seriesAustralia63/MAML, neutropeniaPneumoniaBlood cultureVRC + TRB + ANFDied
Boan [24]2020Case seriesAustralia25/FAML, alloHSCT, neutropeniaFungemia, osteomyelitisBlood, sternoclavicular joint tissue, urine cultureVRC + TRB + ANF + MTF + Surgical debridement of sternoclavicular jointDied
Boglione-Kerriena [25]2019Case reportFrance61/NAMM, autoHSCTFungemia, eye infection, meningitis, brain abscess, calculus pyelonephritisBlood, urinary tract stone cultureVRC + TRB + MTF + intravitreal VRC + Surgical removal of the urinary stoneDied (not related to the fungal infection)
Bouza [26]1996Case reportSpain74/FAML, neutropeniaFungemia, pneumonia, skin lesionsBlood, skin biopsy cultureAMB + G-CSF + ITCSurvived
Buil [27]2020Case reportThe NetherlandsNA/F-FungemiaBlood, stool cultureNADied
Chiam [28]2013Case reportAustralia9/FAML, neutropenia, BMTEndophthalmitis, fungemiaBlood cultureAMB + G-CSF + intravitreal VRC VRC + CAS + TRB + MTF + VitrectomySurvived
Cobo [29]2017Retrospective cohort studySpain53/MAML, neutropeniaFungemiaBlood cultureVRC + TRBDied
Cooley [9]2007Case seriesAustraliaNAALL, alloHSCT, neutropeniaFungemia, septic arthritisBlood, synovium cartilage, prostate cultureNADied
Cooley [9]2007Case seriesAustraliaNAAML, alloHSCTPneumonia, fungemiaBlood, BAL, lung, sputum cultureNADied
Cooley [9]2007Case seriesAustraliaNANHL, alloHSCT, neutropeniaFungemiaBlood cultureNoneDied
Cooley [9]2007Case seriesAustraliaNAAML, alloHSCTFungemiaBlood, BAL, lung, skin cultureITC + AMBDied
Cooley [9]2007Case seriesAustraliaNAMDSSinusitisSputum, maxillary sinus, pericardium, myocardium, kidney, skin, lung cultureITC + TRB + SurgeryDied
Cooley [9] 2007Case seriesAustraliaNAAML, neutropeniaChest wall cellulitis, skin nodules, soft tissue infectionChest wall, Hickman catheter cultureVRC + TRB + SurgerySurvived
Damronglerd [30]2014Case reportThailand17/MMDS, AML, neutropeniaSkin lesions, pneumonia, sinusitis, fungemiaSkin biopsy, sinus, tracheal suction, blood cultureVRC + TRBDied
de Battle [31]2000Case reportSpain45/ΜAcute multilinear leukaemia, neutropeniaFungemia, mycotic emboli, pneumonia, pleuritic effusion, skin lesionBlood cultureAMBDied
DeSimone [10]2021Retrospective cohort studyUSA59/MLung transplantationSkin and subcutaneous infection, fungemia Blood, urine, bilateral lower extremity skin (autopsy), lung (autopsy) cultureVRC + MICA + Surgical debridementDied
DeSimone [10]2021Retrospective cohort studyUSA56/FLung transplantationEndophthalmitis, septic arthritisBilateral knee synovial tissue, right ankle joint aspiration, aorta (autopsy) cultureCAS + VRC + AMB + TRB + ALB + Surgical debridementDied
Elsayed [32]1999Case reportCanada28/FALL, neutropeniaFungemiaBlood cultureFLC + AMBDied
Farag [33]1992Case reportAustralia72/FNHL, neutropeniaFungemia, skin lesionsBlood, CSF cultureAMB + FCSDied
Feltkamp [34]1997Case reportThe Netherlands42/MAML, neutropeniaFungemia, pneumonia, brain emboli, skin lesionsBlood, CSF, BAL, sputum, skin biopsy cultureAMB + FCSDied
Gosbell [35]1999Case seriesAustralia68/MAΜL, neutropeniaFungemia, pneumoniaNasal swab, blood cultureAMBDied
Gosbell [35]1999Case seriesAustralia33/FAML, neutropeniaFungemia, pneumonia, meningoencephalitis, endophthalmitis, renal/myocardial/brain abscesses, mycotic aneurysm Blood, CSF cultureL-AMB + ITC + FLC + AMB (intraocular injection)Died
Gosbell [35]1999Case seriesAustralia48/FAΜΜL, neutropeniaFungemia, skin lesionsBlood, skin lesion cultureL-AMB (only one dose given)Died
Gosbell [35]1999Case seriesAustralia46/MNHL, neutropeniaFungemia, pneumoniaBlood cultureAMBDied
Gow-Lee [36]2021Case reportUSA63/MNHL, neutropenia, autoHSCTPneumonia, fungemia, septic arthritisBAL, blood, synovial fluid cultureVRC + MICA + TRB + GM-CSF + L-AMB + Surgical debridement/amputationDied
Grenouillet [37]2009Case seriesFrance68/MNHL, neutropeniaFungemia, pneumoniaSputum, blood cultureAMB + ITCDied
Grenouillet [37]2009Case seriesFrance44/MCML, alloHSCTFungemia, gingival abscessGingival abscess, blood, urine, trachea cultureVRC + TRBDied
Grenouillet [37]2009Case seriesFrance67/MNHL, alloHSCTFungemia, pneumoniaBlood, urine, BAL cultureVRC + CASDied
Guerrero [38]2001Case seriesSpain45/FAML, neutropeniaFungemia, skin lesions, pneumoniaBlood cultureNoneDied
Guerrero [38]2001Case seriesSpain64/MAML, neutropeniaFungemia, pneumonia, cerebral abscessesBlood cultureAMB + ITCDied
Guerrero [38]2001Case seriesSpain27/FAML, neutropeniaFungemia, pneumonia, pleural effusion, meningoencephalitis, skin lesionsBlood cultureL-AMB + ITCDied
Guerrero [38]2001Case seriesSpain72/FAML, neutropeniaFungemia, pneumoniaBlood cultureL-AMB + ITCDied
Guerrero [38]2001Case seriesSpain72/FAML, neutropeniaFungemiaBlood cultureNoneDied
Hanmantgad [39]2017Case reportUSA71/MAML, neutropeniaFungemiaBlood cultureG-CSFDied
Howden [40]2003Case reportAustralia53/FMM, BMT, neutropeniaSinusitis, osteomyelitis, discitis, aneurysmSinus, lumbar spine, hepatic artery wall cultureITC + Surgical decompression of sinusitis ITC +TRB + Laminectomy/surgical debridement VRC + TRB + GM-CSF + Excision of hepatic artery aneurysmSurvived
Jain [41]2017Case reportUSA65/MAML, neutropeniaPneumonia, fungemia, skin lesionsRespiratory, blood, scrotal lesion cultureL-AMB + POS + ISADied
Kimura [42]2010Case reportJapan58/FAML, neutropeniaPneumonia, fungemiaBlood cultureMICA + G-CSFDied
Kubisiak-Rzepczyk [43]2013Case reportPoland21/FALL, alloHSCTFungemiaBlood cultureVRCDied
Maertens [44]2000Case reportBelgium77/MAML, neutropeniaPneumonia, renal abscess, skin lesionsBAL, abscess cultureAMBITC + VitrectomyDied
Marin [45]1991Case reportSpain66/MAML, neutropeniaPneumonia, fungemia, endophthalmitis, skin lesionsSkin lesions, blood, urine, vitreous cultureAMB Died
Westerman [46]1999Case reportAustralia65/FAML, neutropeniaFungemiaBlood, sputum, faecal cultureAMB Died
McKelvie [47]2001Case reportAustralia59/MAML, neutropeniaEndophthalmitis, fungemia, pneumoniaBlood cultureIntravitreal AMB + AMB + VRCDied
Nambiar [48]2017Case reportUSA65/MNHL, neutropeniaFungemiaBlood cultureNoneDied
Nenoff [49]1996Case reportGermany60/MAIDS, Burkitt lymphoma, neutropenia-Kidney, spleen, myocardium tissue autopsy cultureFLC + G-CSFDied
Nielsen [50]1993Case reportUSA17/MAML, neutropeniaFungemia, pneumonia, skin lesionsBlood, skin, lung tissue cultureAMBDied
Nishimori [51]2014Case reportJapan71/FAML, neutropeniaFungemiaBlood, fecal cultureMICAL-AMBDied
Penteado [52]2018Case reportBrazil17/MX-linked chronic granulomatous disease, AlloHSCTFungemia, pneumoniaBlood, urine cultureVRCDied
Pickles [53]1996Case seriesAustralia41/MAML, neutropeniaPneumoniaKidney, lung, liver autopsy cultureAMBDied
Rabodonirina [54]1994Case reportFrance50/FLung transplantationFungemia, pleural effusion, pneumoniaPleural fluid, central venous catheter, blood cultureAMBDied
Reinoso [55]2013Case reportSpain35/FAML, neutropeniaFungemia, pneumonia, pleural effusion, endophthalmitis, orbital cellulitisVitreous fluid culture/PCR, blood cultureVRC + TRB + VitrectomyDied
Rivier [56]2011Case reportFrance70/MMDS, AML, neutropeniaFungemiaSputum, blood cultureG-CSFPOSCASDied
Rolfe [11]2014Case seriesUSA44/MAML, alloHSCTFungemiaBAL, blood, skin cultureVRC + AMBDied
Salesa [57]1993Case reportSpain56/FAML, autoHSCT, neutropeniaFungemia, skin lesionsBlood cultureAMB + GM-CSFDied
Simarro [58]2001Case reportSpain34/FAML, neutropeniaFungemia, pneumoniaBlood cultureL-AMBDied
Simarro [58]2001Case reportSpain20/FALL, neutropeniaFungemiaBlood cultureAMBDied
Song [59]2010Case reportSouth Korea8/MALL, neutropeniaFungemia, pneumonia, skin lesionsBlood cultureITCDied
Sparrow [60]1992Case reportAustralia3/MNeuroblastoma, autoHSCTSkin lesions, fungemiaSkin biopsy, blood, urine, endotracheal tube, faeces, throat swab cultureAMBDied
Spielberger [61]1995Case reportUSA32/FAML, AlloHSCT, neutropeniaPneumonia, fungemiaSputum, blood cultureAMB + ITCDied
Stefanovic [62]2016Case reportCanada44/MHemophagocytic lymphohistiocytosis, NHL, neutropeniaPneumonia, fungemiaBAL, blood cultureVRC + MICANA
Tapia [63]1994Case seriesSpain45/MMM, autoHSCT, neutropeniaFungemia, meningism, pneumoniaBlood cultureNoneDied
Tapia [63]1994Case seriesSpain49/MAML, neutropeniaPneumonia, hemiplegiaBAL culture, autopsy lung, liver, kidneys, brain (ischemic lesion) fungal invasionAMB + ITC + Surgical resection of lung noduleDied
Teh [64]2019Retrospective cohort studyAustralia68/MCLLFungemiaBlood cultureCASDied
Tey [65]2020Case reportAustralia60/FCLL, neutropeniaFungemia, pneumonia, septic emboli brain, skin, chestBlood cultureVRC + TRB + G-CSFDied
Tong [66]2007Case reportAustralia61/MAML, alloHSCTFungemia, endophthalmitisBlood cultureCAS + VRC + TRB + intravitreal VRCDied (no evidence of fungal infection in autopsy)
Trubiano [67]2014Case reportAustralia67/ΜAMLFungemia, endophthalmitisVitreous fluid, eye, temporal lobe specimen cultureCAS + VRC + TRB + intravitreal VRC + Vitrectomy/enucleation/temporal lobectomySurvived
Valerio [68]2021Case reportSpain25/ΜHeart transplantationFungemia, pneumonia, skin lesionsBlood, catheter tip, tracheal aspirate, skin biopsy cultureL-AMB + VRC + TRBDied
Whyte [69]2005Case reportAustralia8/FALLPneumonia, septic arthritis, osteomyelitis, discitis, epidural fluid collectionLung biopsy, joint aspirate, laminectomy, disc debridement specimens cultureL-AMBVRC + TRB + G-CSF + Laminectomy/disc debridement/surgical joints washoutsSurvived
Wilson [70]2022Case reportAustralia43/FAML, neutropeniaFungemia, pneumonia, skin lesions, septic arthritis, osteomyelitis, intracerebral lesionsBlood, synovial fluid cultureVRC + TRB + MTF + Debridement/synovectomy/arthroscopic washoutDied
Wise [71]1993Case reportAustralia53/MRenal transplantationPneumonia, peritonitisPeritoneal, wound swabs, pleural, ileostomy, jejunal fluid cultureAMBMICDied
Wood [72]1992Case seriesAustralia52/MAMLFungemia, endophthalmitis Vitreous aspirate, urine, blood, skin biopsy culture, autopsy renal abscess cultureAMB + FCSDied
Wood [72]1992Case seriesAustralia46/MALL, neutropeniaFungemiaBlood from Hickman catheter cultureNoneDied
Strickland [73]1998Case seriesUSA51/FBreast cancer, autoHSCT, neutropeniaFungemia, pneumonia, pericardial effusion, pleural effusionBlood culture, autopsy specimens (heart, lung, liver)AMB Died
Carreter de Granda [74]2001Case reportSpain52/FMM, BMT, neutropeniaFungemia, endocarditis, endophthalmitis, brain mycotic aneurysmBlood, valve specimen cultureL-AMB + ITC + Valve replacementDied
Freeman [75]2007Case seriesUSA24/FHyper IgE syndromePneumonia, cerebritisLung tissue autopsy culture, cerebritis/ pyelonephritis with budding hyphae autopsyAMB + POSDied
Fernandez-Guerrero [76]2011Case reportSpain29/FALLEndocarditis, septic arthritis, osteomyelitis, mycotic aneurysm, endophthalmitisBlood, vitreous fluid, embolus, valve vegetations cultureVRC + L-AMB + TRB + Embolectomy/valve replacementDied
Kelly [77]2016Case reportAustralia75/FOvarian carcinomaEndocarditis, cerebral emboli, fungemiaBlood cultureVRCDied
O’ Hearn [78]2010Case reportUSA38/FHeart transplantationEndophthalmitisVitreous specimen, chest wall lesion cultureIntravitreal AMB/VRC + VRC + TRB + VitrectomySurvived
Ochi [79]2015Case reportJapan66/FAML, neutropeniaFungemia, sinusitis, pulmonary/splenic emboli, endocarditisBlood, sputum, CSF cultureFLCVRC + L-AMBVRC + TRB + G-CSFDied
Ohashi [80]2011Case reportJapan58/MMonoclonal gammopathy of undermined significanceFungemia, pneumoniaBlood, sputum cultureITCL-AMBMICA + VRCDied
Sayah [81]2013Case reportUSA70/FLung transplantationPericarditis, mycotic aneurysm, pneumoniaBAL, pericardial cultureVRC + TRB + MICA + PericardiectomyDied
Smita [82]2015Case reportIndia50/MPacemaker implantation, diabetesFungemia, endocarditisBlood, valve tissue specimen cultureL-AMBVRC + POSVRC + TRB + Valve replacementSurvived
Tascini [83]2006Case reportItaly75/MPacemaker implantationEndocarditis, pneumoniaTips of the lead cultureVRC + Pacemaker removalSurvived
Uno [84]2014Case reportJapan35/MRenal transplantationFungemia, endocarditis, meningitis, pneumoniaBlood, sputum, CSF cultureITRA + MICAL-AMB + VRCDied
Wakabayashi [85]2016Case reportJapan64/FChronic osteomyelitisFungemia, endocarditis, endophthalmitis, osteomyelitis Blood cultureFLCDied
Ahmad [86] #2010Case reportUSA50/MRheumatic diseaseFungemia, brain emboliBlood cultureL-AMB + Valve replacementDied
Spanevello [87]2010Case reportAustralia28/FAcute undifferentiated leukemia, neutropeniaPseudoaneurysm, cerebral hemorrhage Blood, sinus material cultureVRC + TRBDied
Beldarrain [88]2000Case reportSpain42/FAML, neutropeniaFungemia, pneumonia, ischemic brain infarct Blood cultureFLCDied
Guadalajara [89]2018Case reportSpain36/FMultiple sclerosis, glucocorticoidsMycotic cerebral aneurysm, ischemic stroke Fungal structures in the arterial wall of ruptured aneurysm, thrombus, larynx, small intestineNoneDied
Tamaki [90]2016Case reportJapan62/MAML, neutropenia, alloHSCTMeningitis, fungemiaBlood, CSF culture and PCRMICAL-AMB + VRCDied
Takata [91]2020Case reportJapan70/FAMLEndophthalmitis, brain aneurysm, fungemiaBlood culture, fungal structures in the arterial wall of the aneurysmCAS + AMBVRC + Aneurysm resectionDied
Marco de Lucas [92]2006Case seriesSpain37/MAML, alloHSCT, neutropeniaOrbit cellulitis, multiple brain lesions, pneumoniaAutopsyAMB + ITC + FLCDied
Marco de Lucas [92]2006Case seriesSpain66/MAML, neutropeniaMultiple brain lesions, pneumoniaAutopsyAMB + ITC + FLCDied
Marco de Lucas [92]2006Case seriesSpain45/MMM, alloHSCT, neutropeniaArterial brain thrombosis, pneumoniaBlood culture, AutopsyAMB + ITC + FLCDied
Marco de Lucas [92]2006Case seriesSpain18/FMDS, AlloHSCT, neutropeniaPansinusitis, orbital cellulitis, multiple brain lesions, pneumoniaAutopsyAMB + ITC + FLCDied
Marco de Lucas [92]2006Case seriesSpain36/MAML, alloHSCT, neutropeniaMultiple brain lesions, pneumoniaAutopsyAMB + ITC + FLCDied
Marco de Lucas [92]2006Case seriesSpain52/FMM, autoHSCT, neutropeniaEndocarditis, subarachnoid hemorrhage, bilateral panuveitis, pneumoniaBlood cultureAMB + ITC + FLCDied
Elizondo-Zertuche [93] 2017Case seriesMexico48/FCML, BMTFungemiaBAL, urine, blood cultureITC + CASDied
Elizondo-Zertuche [93] 2017Case seriesMexico61/MAIDSFungemiaBlood cultureFLCDied
Elizondo-Zertuche [93] 2017Case seriesMexico47/FCMLSepsisBAL, vitreous cultureNoneDied
Elizondo-Zertuche [93] 2017Case seriesMexico57/FRenal transplantationFungemiaBlood cultureAMBDied
Elizondo-Zertuche [93] 2017Case seriesMexico67/MAMLFungemiaBlood, peritoneal fluid cultureFLC + AMBDied
Elizondo-Zertuche [93] 2017Case seriesMexico40/MAMLFungemiaBlood cultureAMBDied
Idigoras [94]2001Case seriesSpain44/FAML, neutropeniaFungemia, pneumonia, conjunctival effusion, cutaneous eruptionBlood cultureAMBDied
Idigoras [94]2001Case seriesSpain55/FBreast cancer, autoHSCT, neutropeniaFungemiaBlood cultureITC + G-CSFSurvived
Idigoras [94]2001Case seriesSpain28/MAIDSFungemia, pneumonia BAL, blood, urine, feces, sputum cultureNoneDied
Idigoras [94]2001Case seriesSpain65/MAML, neutropeniaFungemia, skin lesions, pneumoniaBlood, sputum cultureFLC + ITC + AMB + G-CSFDied
Idigoras [94]2001Case seriesSpain56/FAML, neutropeniaFungemia, pneumoniaBlood cultureFLC + G-CSFDied
Idigoras [94]2001Case seriesSpain28/MAMLFungemia, spondylodiscitis, abdominal abscess, skin lesions, cholecystitisBlood, wound, abscess cultureFLC + ITC + AMB + G-CSF + TRB + VRC + Abscess drainageDied
Jenks [95]2018Retrospective cohort studyUSANA/NANHLFungemiaBlood cultureMICA + L-AMBDied
Jenks [95]2018Retrospective cohort studyUSANA/NAChronic granulomatous diseaseFungemiaBlood cultureVRC + TRBSurvived
Vagefi [96]2005Case reportUSA56/FLung transplantationPneumonia, endophthalmitisBronchial bruising, vitreous cultureVRC + TRB + intravitreal AMB/VRCDied
Johnson [97]2014Retrospective cohort studyUSA54/MMutlivisceral transplantationNAAutopsy heart, pericardium,
pleura, kidneys, brain		AMB + CAS + VRCDied
Johnson [97]2014Retrospective cohort studyUSA51/FMutlivisceral transplantationNAAutopsy pericardium, eyes, dermis, heart, kidneys, pancreasAMB + CAS + VRCDied
Nasif [98]2021Case reportUSA48/MRenal transplantationThigh, brain, shin abscesses, femoral artery mycotic aneurysmThigh, brain, shin abscesses culturePOS + AMB + SurgeryTRB + VRC + SurgeryDied
Tintelnot [13]2009Retrospective cohort studyGermany54/FRenal transplantationFungemia, pneumonia, skin lesions, sepsisBlood cultureL-AMB + FCS + MICDied
Tintelnot [13]2009Retrospective cohort studyGermany53/FAMLFungemia, pneumonia, skin lesions, endophthalmitis, sepsisBlood cultureAMB + FCSDied
Tintelnot [13]2009Retrospective cohort studyGermany61/FLong term corticosteroidsFungemia, pneumoniaBlood, tracheal secretions cultureNoneDied
Tintelnot [13]2009Retrospective cohort studyGermany44/FCML, BMTFungemia, pneumonia, sepsisBAL, urine, catheter, blood cultureCASDied
Tintelnot [13]2009Retrospective cohort studyGermanyNA/MBMTFungemia, endophthalmitis, sepsisBlood culturePOSDied
Tintelnot [13]2009Retrospective cohort studyGermany40/MAMLFungemia, sepsisBlood cultureAMBDied
Tintelnot [13]2009Retrospective cohort studyGermany64/MAMLFungemia, brain involvementBlood cultureNoneDied
Tintelnot [13]2009Retrospective cohort studyGermany60/MChronic idiopathic myelofibrosis, BMTFungemia, sepsisBlood, BAL cultureVRC + CASDied
Tintelnot [13]2009Retrospective cohort studyGermany47/FCOPD, lung transplantationEndophthalmitis, sepsisBAL, vitreous fluid culturePOS + CASL-AMBVRCDied
Husain [99] *2005Case seriesUSA/Spain55/ΜSmall bowel transplantationPeritoneum involvementNAAMBDied
Husain [99] *2005Case seriesUSA/Spain40/MKidney/pancreas transplantationCNS, pulmonary involvementNAVRCSurvived
Husain [99] *2005Case seriesUSA/Spain51/FSmall bowel transplantationAneurysmNAAMB + VRC + CASDied
Husain [99] *2005Case seriesUSA/Spain17/MLiver transplantationPulmonary involvementNAVRCDied
Husain [99] *2005Case seriesUSA/Spain44/FHeart transplantationPulmonary, sinus, skin involvementNAAMBDied
Husain [99] *2005Case seriesUSA/Spain68/MKidney/liver transplantationSkin involvementNAVRCSurvived
AML: acute myeloid leukemia, ALL: acute lymphoblastic leukemia, AMML: acute myelomonocytic leukemia, NHL: non-Hodgkin lymphoma, CML: chronic myeloid leukemia, MM: multiple myeloma, MDS: myelodysplastic syndrome, ΒΜΤ: bone marrow transplantation, AlloHSCT: allogenic hemopoietic stem cell transplantation, AutoHSCT: autologous hemopoietic stem cell transplantaton, COPD: chronic obstructive pulmonary disease, AMB: amphotericin B, L-AMB: liposomal amphotericin B, VRC: voriconazole, TRB: terbinafine, POS: posaconazole, CAS: caspofungin, MTF: miltefosine, ANF: anidulafungin, ITC: itraconazole, ALB: albaconazole, FLC: fluconazole, FCS: flucytosine, ISA: isavuconazole, MIC: miconazole, MICA: micafungin, NA: not applicable. * This study includes six solid organ recipients with L. prolificans infection affecting many systems, but it is not clearly stated if dissemination is present. # Information extracted from other articles [77,85].
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Konsoula, A.; Agouridis, A.P.; Markaki, L.; Tsioutis, C.; Spernovasilis, N. Lomentospora prolificans Disseminated Infections: A Systematic Review of Reported Cases. Pathogens 2023, 12, 67. https://doi.org/10.3390/pathogens12010067

AMA Style

Konsoula A, Agouridis AP, Markaki L, Tsioutis C, Spernovasilis N. Lomentospora prolificans Disseminated Infections: A Systematic Review of Reported Cases. Pathogens. 2023; 12(1):67. https://doi.org/10.3390/pathogens12010067

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Konsoula, Afroditi, Aris P. Agouridis, Lamprini Markaki, Constantinos Tsioutis, and Nikolaos Spernovasilis. 2023. "Lomentospora prolificans Disseminated Infections: A Systematic Review of Reported Cases" Pathogens 12, no. 1: 67. https://doi.org/10.3390/pathogens12010067

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