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Mycopathologia

, Volume 183, Issue 6, pp 879–891 | Cite as

The Curious Case of “Case Report” of Infections Caused by Human and Animal Fungal Pathogens: An Educational Tool, an Online Archive, or a Format in Need of Retooling

  • Jean-Philippe Bouchara
  • Vishnu ChaturvediEmail author
Editorial

Abstract

Case reports describe the unusual occurrence and complications of diseases, diagnostic challenges, and notable therapeutic successes. Some journals have discontinued the case reports, while new case report journals have appeared in recent years. During the eightieth anniversary of Mycopathologia, it is fitting to examine the relevance of the case report since the journal continues to traverse the boundaries of basic and clinical sciences. A random sample of recent case reports and other articles were selected from Mycopathologia. Springer Nature individual article download statistics, and Google Scholar and Scopus citations numbers were compared to assess the reader access and bibliometric impact of case reports. Our analysis indicated that the case report format continues to be a vital element of publication in a cross-disciplinary journal such as Mycopathologia. Medical and veterinary case reports covering fungal pathogens are widely read as evident from their download numbers. The download numbers have a positive correlation with the completeness of the report, the topics and geographic origin of reports have a neutral influence, and the recency leads to lower downloads. There is no discernible trend between the download numbers and the citations of case reports as measured by Google Scholar and Scopus. A specially designed checklist for Mycopathologia case reports and new format MycopathologiaIMAGES are being introduced to improve the quality and relevance of case reports further.

Introduction

Case reports highlight the unusual disease occurrence and complications, diagnostic challenges, and notable therapeutic successes observed by the astute professionals serving at the frontlines of medical and veterinary practices [1, 2, 3, 4, 5, 6, 7, 8, 9, 10]. Case reports have been a regular feature in the most prestigious medical journals including the New England Journal of Medicine and The Lancet. The ongoing massive changes in the scientific publishing industry have also impacted the publication of case reports [11, 12, 13, 14, 15, 16]. Some journals have discontinued the case reports as a feature of their regular contents while new journals, devoted exclusively to case reports, are also being introduced [17, 18, 19, 20, 21, 22, 23, 24]. Amidst these unprecedented changes, there is renewed scrutiny of the scholarly impact of the case reports and the case for their continued publication in journals that carry a mixed format of articles [19, 22]. The case report format is often associated with the variable quality of content, and there is a prevailing view that case reports as a group generate low science citation index measured by the impact factor. However, the overall impact of case reports relative to other publication formats is mixed when an expanded list of benefits and metrics are used for their assessment [25, 26, 27, 28, 29, 30, 31]. In a sign of definite progress, a consensus toward standardized format for the case report has emerged among specific disciplines with the use of checklist to ensure high-quality reporting by the prospective authors [32, 33, 34, 35, 36, 37].

Mycopathologia had a long history of publishing case reports beginning with the first few inaugural volumes [38, 39, 40]. Among these medical and veterinary case reports, there were essential insights into new pathogenic species, new tests, and treatments as well as the undocumented clinical manifestations of well-known pathogenic fungi and fungal diseases. During this eightieth anniversary of publication, we saw it fitting to re-evaluate the relevance of the case report as a regular publication format in Mycopathologia since the journal continues to traverse the boundaries of basic and clinical sciences.

Case Report Analyses

In the following analysis, we focused on the case reports primarily from Mycopathologia and their reception in the scientific community. Recent case reports and other articles were selected at random from 2016 to 2017 issues of Mycopathologia. Springer Nature individual article download statistics were used to gauge the professional reception of each report after publication (Tables 1, 2, 3, 4, 5, 6, 7; Fig. 1). Google Scholar and Scopus citations numbers of respective reports were compared to the download numbers to assess the bibliometric impact. We also used examples from the New England Journal of Medicine to strengthen our conclusions about the bibliographic measurements and impact of the case reports (Table 8).

Table 1

A sampling of Mycopathologia case reports with high downloads (> 200) and high citations (> 3)

Title

Download

Citationsa

References

First two cases of fungal infections associated with multi-drug resistant yeast, Fereydounia khargensis

1100

3/2

[50]

Subcutaneous phaeohyphomycosis cyst associated with Medicopsis romeroi in an immunocompromised host

998

3/3

[52]

Aspergillosis of the heart and lung and review of published reports on fungal endocarditis

840

6/4

[51]

Fusariosis in a patient with acute myeloid leukemia: a case report and review of the literature

591

10/6

[47]

Fatal cases of bloodstream infection by Fusarium solani and review of published literature

405

5/3

[45]

Paracoccidioides brasiliensis PS2: first autochthonous paracoccidioidomycosis case report in Rio de Janeiro, Brazil, and literature review

334

9/4

[41]

Fungal mycotic aneurysm of the internal carotid artery associated with sphenoid sinusitis in an immunocompromised patient: a case report and review of the literature

325

6/5

[53]

Spondylodiscitis due to Aspergillus terreus in an immunocompetent host: case report and literature review

312

4/2

[43]

Meningitis caused by Candida dubliniensis in a patient with cirrhosis: a case report and review of the literature

297

4/4

[54]

Tinea capitis by Microsporum audouinii: case reports and review of published global literature 2000–2016

284

3/3

[42]

The first case of total dystrophic onychomycosis caused by Aspergillus clavatus resistant to antifungal drugs

255

6/4

[48]

Phaeohyphomycosis due to Exophiala jeanselmei: an emerging pathogen in India-case report and review

209

3/3

[46]

Nebulized liposomal amphotericin B for treatment of pulmonary infection caused by Hormographiella aspergillata: case report and literature review

205

5/2

[44]

Spectral manifestation of melanized fungal infections in kidney transplant recipients: report of six cases

201

4/4

[49]

aGoogle Scholar/Scopus

Table 2

A sampling of Mycopathologia case reports with high downloads (> 200) and low citations (< 2)

Title

Download

Citationsa

References

Imported talaromycosis in Oman in advanced HIV: a diagnostic challenge outside the endemic areas

820

2/1

[55]

The concept of ecthyma gangrenosum illustrated by a Fusarium oxysporum infection in an immunocompetent individual

794

2/2

[56]

Fournier gangrene caused by Candida albicans in an infant after cardiac surgery

765

1/1

[57]

Rhizopus arrhizus and Fusarium solani concomitant infection in an immunocompromised host

347

2/1

[102]

Primary cutaneous mucormycosis caused by Rhizopus oryzae: a case report and review of literature

302

3/2

[59]

A case report of penile infection caused by fluconazole- and terbinafine-resistant Candida albicans

294

1/1

[60]

The simultaneous occurrence of histoplasmosis and cryptococcal fungemia: a case report and review of the literature

283

0/0

[61]

Urinary tract infection (UTI) caused by Fusarium proliferatum in an agranulocytosis patient and a review of published reports

283

2/0

[64]

Fungal keratitis due to Beauveria bassiana in a contact lenses wearer and review of published reports

268

2/2

[70]

Fusarium dimerum species complex (Fusarium penzigii) keratitis after corneal trauma

260

1/1

[69]

First case of Trichoderma longibrachiatum CIED (Cardiac Implantable Electronic Device)-associated endocarditis in a non-immunocompromised host: biofilm removal and diagnostic problems in the light of the current literature

260

3/1

[66]

Cunninghamella bertholletiae infection in a HLA-haploidentical hematopoietic stem cell transplant recipient with graft failure: case report and review of the literature

220

1/1

[65]

Pseudallescheria boydii with Aspergillus fumigatus and Aspergillus terreus in a critically ill hematopoietic stem cell recipient with ARDS

210

2/2

[68]

Respiratory tract infection caused by Fonsecaea monophora after kidney transplantation

205

1/0

[62]

Combined orofacial aspergillosis and mucormycosis: fatal complication of a recurrent paediatric glioma-case report and review of literature

201

1/0

[67]

Severe paracoccidioidomycosis in a 14-year-old boy

200

1/1

[63]

aGoogle Scholar/Scopus

Table 3

A sampling of Mycopathologia case reports with low downloads (< 200) and high citations (> 3)

Title

Download

Citationsa

References

Dermatophytosis due to Microsporum incurvatum: Notification and identification of a neglected pathogenic species

199

7/7

[71]

The first case of invasive mixed-mold infections due to Emericella nidulans var. echinulata and Rasamsonia piperina in a patient with chronic granulomatous disease

195

4/4

[72]

Onychomycosis associated with Exophiala oligosperma in Taiwan

165

5/3

[73]

Paracoccidioidomycosis mimicking sarcoidosis: a review of 8 cases

164

5/3

[74]

aGoogle Scholar/Scopus

Table 4

A sampling of Mycopathologia case reports with low downloads (< 200) and low citations (> 2)

Title

Download

Citationsa

References

Ustilago echinata: infection in a mixed martial artist following an open fracture

169

1/1

[85]

Phaeohyphomycosis caused by Rhytidhysteron rufulum and review of literature

167

3/0

[78]

Lichtheimia infection in a lymphoma patient: case report and a brief review of the available diagnostic tools

157

1/1

[82]

Coccidioidomycosis in patients with selected solid organ cancers: a case series and review of medical literature

154

1/1

[84]

Successful antifungal combination therapy and surgical approach for Aspergillus fumigatus suppurative thyroiditis associated with thyrotoxicosis and review of published reports

146

1/0

[80]

Coinfection with Lichtheimia corymbifera and Aspergillus flavus in an immune-competent patient mimicking as pulmonary-renal syndrome

126

1/1

[81]

aGoogle Scholar/Scopus

Table 5

A sampling of Mycopathologia case reports with low downloads (< 200) and no citations

Title

Download

Citationsa

References

Acute disseminated Talaromyces marneffei in an immunocompetent patient

180

0/0

[77]

Fungal keratitis secondary to Trametes betulina: a case report and review of literature

162

0/0

[86]

A 9-month-old girl from Iran with extensive erythematous plaques due to Trichophyton simii, a zoophilic dermatophyte

160

0/0

[79]

Gastroesophageal reflux disease and pulmonary diseases associated with aspergillosis: Is there a connection?

148

0/0

[83]

Invasive Aspergillus laryngotracheobronchitis in an adult with primary CNS lymphoma

139

0/0

[87]

Molecular identification of cutaneous alternariosis in a renal transplant patient

111

0/0

[75]

Acute paracoccidioidomycosis in a 40-year-old man: a case report and literature review

107

0/0

[76]

Nosocomial rhinocerebral mucormycosis: two cases with a temporal relationship

95

0/0

[103]

aGoogle Scholar/Scopus

Table 6

A sampling of Mycopathologia veterinary case reports

Title

Downloads

Citationsa

References

Coccidioidomycosis in biopsies with presumptive diagnosis of malignancy in dogs: report of three cases and comparative discussion of published reports

1000

1/1

[88]

Disseminated candidiasis in a young, previously healthy, dog and review of literature

800

1/1

[104]

Cryptococcus neoformans var. grubii-induced arthritis with encephalitic dissemination in a dog and review of published literature

259

8/8

[90]

Detection of multiple budding yeast cells and a partial sequence of 43-kDa glycoprotein coding gene of Paracoccidioides brasiliensis from a case of lacaziosis in a female pacific white-sided dolphin (Lagenorhynchus obliquidens)

245

7/6

[94]

Paracoccidioides brasiliensis-associated dermatitis and lymphadenitis in a dog

224

6/5

[91]

Evidence of a Prototheca zopfii genotype 2 disseminated infection in a dog with cutaneous lesions

197

1/1

[92]

Intestinal lesion in a dog due to Cryptococcus gattii type VGII and review of published cases of canine gastrointestinal cryptococcosis

177

3/4

[105]

aGoogle Scholar/Scopus

Table 7

A sampling of Mycopathologia articles covering clinical and experimental mycology, and opinions

Title

Download

Citationsa

References

Candida albicans/Staphylococcus aureus dual-species biofilm as a target for the combination of essential oils and fluconazole or mupirocin

1300

2/1

[101]

Challenges in Fusarium, a trans-kingdom pathogen

1100

12/11

[100]

Pathogenic dermatophytes survive in nail lesions during oral terbinafine treatment for Tinea unguium

734

0/0

[99]

Has Aspergillus PCR come to the age of maturity?

714

1/1

[95]

Genotyping of Fusarium isolates from onychomycoses in Colombia: detection of two new species within the Fusarium solani species complex and in vitro antifungal susceptibility testing

453

12/9

[98]

The AAA ATPase Vps4 plays important roles in Candida albicans hyphal formation and is inhibited by DBeQ

398

3/0

[96]

Quantitative real-time PCR and Platelia galactomannan assay for the diagnosis of invasive pulmonary aspergillosis: Bronchoalveolar lavage fluid performs better than serum in non-neutropaenic patients

296

9/9

[97]

aGoogle Scholar/Scopus

Fig. 1

Cloud visualization (word cloud) of key words appearing in the title of Mycopathologia case reports. The visualization was generated using freely availbale online tool with default setting (WordClouds.com). The characterize size reflects the frequency of occurrence of key words in the title of the case reports summarized in Tables 16

Table 8

A limited sampling of case reports from the New England Journal of Medicine

Title

Diagnosis

Page views

Citationsa

References

Case records of the Massachussets General Hospital. Case 5-2016. A 43-year-old man with altered mental status and a history of alcohol use

Cerebral mucormycosis

24,931

3/1

[106]

Case 6-2017. A 57-year-old woman with fatigue, sweats, weight loss, headache, and skin lesions

IgG4-related disease

24,475

2/0

[107]

Case 6-2018: A 35-year-old woman with headache, subjective fever, and anemia

Transient aplastic crisis due to parvovirus B19 infection

24,452

2/0

[108]

Case records of the Massachussets General Hospital. Case 4-2016. A 58-year-old woman with a skin ulcer, fever, and lymphadenopathy

Ulceroglandular tularemia

22,473

1/1

[109]

Case records of the Massachussets General Hospital. Case 6-2016. A 10-year-old boy with abdominal cramping and fevers

Adenocarcinoma of colon

20,200

1/4

[110]

Case 5-2017. A 19-year-old man with hematuria and a retroperitoneal mass

Soft tissue sarcoma

19,730

3/2

[111]

Case 5-2018: A 63-year-old man with confusion after stem cell transplantation

Human herpesvirus 6-related encephalitis

15,621

0/0

[112]

Case 4-2018: A newborn with thrombocytopenia, cataracts, and hepatosplenomegaly

Congenital rubella syndrome

12,861

1/1

[113]

Case 4-2017. A 2-month-old girl with growth retardation and respiratory failure

X-linked periventricular heterotopia caused by an FLNA mutation

11,520

0/0

[114]

aGoogle Scholar/Scopus

High Downloads and Citations

Eight of the 11 case reports in the sample set were downloaded more than 300 times each (Table 1). These case reports were also cited 3 or more times in the last 2 years. These case reports contained a balanced account of case history, mycology and pathology investigations, treatment details, and the review of the relevant literature. They originated from Brazil [41, 42], France [43, 44], India [45, 46], Italy [47], Iran [48], Japan [49], Malaysia [50], Poland [51], UK [52], and USA [53, 54]. There were no other discernible features among this group of case reports to account for the high downloads and citations except for a novelty or rarity of fungal pathogens, for example reports of two cases of multidrug-resistant yeast (Fereydounia khargensis) and of black-grain eumycetoma caused by the rare fungal pathogen Medicopsis romeroi [50, 52]. Next, we examined the case reports that had either high downloads and low citations, or low downloads and high citations (Tables 2, 3). Unlike the pattern in Table 1, many case reports with downloads more than 300 were cited once or twice [55, 56, 57, 58, 59]. None of the reports included any unusual pathogens, nor there were any multidrug-resistant pathogens reported except for a case of fluconazole- and terbinafine-resistant Candida albicans [60]. The geographic origin of these case reports were quite widespread: four each originated from Brazil [58, 61, 62, 63] and China [56, 60, 64, 65], two from Italy [66, 67], and one each from Germany [68], Mexico [59], Oman [55], Poland [57], Portugal [69], and Spain [70]. Unlike high download–low citation reports, there are only a few case reports with low downloads and high citations (Table 3). These reports deal with rare pathogens, mixed infection, or unusual clinical presentation in many cases [71, 72, 73, 74]. Seventeen of twenty-four reports appeared in 2016, while only seven were published in 2017, suggesting that a longer duration from the date of publication might have some influence both on downloads and on citations. In summary, the case reports that described rare or novel pathogens, multidrug resistance, or unique clinical presentations, appeared in 2016 than in 2017, were downloaded more often and received higher citations. However, a high download did not predict higher citation or vice versa.

Low Downloads and Citations

Next, we examined whether there was any discernible pattern for the case reports that had low downloads and low or no citations (Tables 4, 5). These case reports were reported from Belgium [75], Brazil [76], China [77], India [78], Iran [79], Italy [80], Pakistan [81], Spain [76], Switzerland [82], UK [83], and USA [84, 85, 86, 87]. The cases included four Aspergillus infections, three pathogens belonging to Mucorales order, two basidiomycetes, one each Alternaria species, Paracoccidioides species, a rare dematiaceous pathogen, and a case series on Coccidioides species. Notably, ten of the fourteen case reports were published in 2017, and only four appeared earlier in 2016. The limited dataset suggested that the geographic location of the investigation, pathogen type, or disease condition showed no correlation with the lower downloads and citation. However, as could be expected, the recency of publication showed some correlation with the lower downloads and citations.

Veterinary Case Reports

A limited analysis of veterinary case reports is described in Table 6. The reports included six canine cases [88, 89, 90, 91, 92, 93] and one involving a Pacific dolphin [94]. The cases were reported from Brazil for three of them, or from Italy, Japan, Mexico, and the Netherlands for the others. Unlike human cases discussed earlier, all veterinary case reports generated high downloads and notable citations. Additionally, five of the seven case reports were from 2017, which was surprising as the recency showed a negative correlation with the downloads and citations for human case reports. Further examination of a larger dataset of veterinary case reports is needed to discern if their reading and citations follow a different pattern from the medical case reports, in relation to the very low frequency of mycological examination of pets and of course of wild animals, and subsequently, to the limited number of related publications compared to case reports of human infections.

Articles Other Than Case Report

In Table 7, we present the analysis of a few Mycopathologia articles covering clinical and experimental mycology, and the opinions. The articles were from Brazil [95], China [96, 97], Colombia [98], Japan [99], the Netherlands [100], and Poland [101]. Interestingly, an impressive number of downloads of a recent article did not lead to high citations [101], while high downloads of a 2016 article garnered impressive citations. Cautiously, one could discern that the downloads and citations do not always correlate well for many article types in Mycopathologia as goes for case reports.

Case Reports in New England Journal of Medicine

Among the most acclaimed scientific publications, the New England Journal of Medicine includes one case report for almost every week of its yearly publication. We did a random sampling of nine reports for the last 3 years reported in the same month (Table 8). The authors of these case presentations are from the Boston area, and affiliated with the local medical institutions. As expected, these reports were downloaded many thousands of times. Interestingly, recency and topics had no influence on the rates of downloads with a mucormycosis case being most downloaded in this small set. Strikingly, tens of thousands of downloads later, there were still very few citations of these cases. These observations suggest that case reports continue to have an educational role, and it will be a fallacy to subject evaluation of their impact on a single metric such as citation number.

Checklists

As mentioned in the opening section, many journals have instituted a checklist for quality control of case reports being submitted for publication. The essential elements of writing a case report with the help of the checklist are covered in detail elsewhere [32, 33, 34, 35, 36]. After examining variable quality of the case reports being submitted to Mycopathologia, we propose to introduce a checklist to help the prospective authors with the preparation and publication of high-quality case reports (Table 9). We also observed that not all case reports need the length and description of a typical article to convey their important messages. Therefore, we are introducing MycopathologiaIMAGES as a new feature in the journal for the communication of brief clinical observations of interests to the physicians and mycologists (Table 10).
Table 9

A checklist for submission of case report to Mycopathologia

 

Yes

No

Original observation

Ethical statement included

Patient demographic anonymized

Title includes the term for medical and veterinary condition being described and the correct name of the fungal pathogen (italicized)

Title and abstract do not include superlatives such as ‘new’, ‘first’, ‘unique’, etc.

Patient treatment and follow-up included

Confirmatory, probable or possible diagnostic criteria defined

Mycology

  

Any author a specialist in fungal diagnostics?

Culture positive with characteristic microscopy

Culture confirmed with molecular tests

Fungus name conform to current nomenclature

Nucleotide sequence deposited in a known database

Antifungal susceptibility test results included

Antifungal test result interpretation provided with reference to the guidance documents (CLSI, EUCAST)

  

Histopathology

  

Any author a specialist in histopathology?

High-resolution images included

Specialized fungal stains used

Differential histopathology diagnosis discussed

Table 10

MycopathologiaIMAGES checklist

Original, unpublished observation being reported with ethical statement

Report of potential interest to a mixed audience of physicians and laboratory scientists

Brief title (three-four keywords)

One paragraph description not to exceed 500 words

No abstract, literature citation, bibliography or hyperlinks

Maximum of four images with short legends (colored required for free online publication)

Author and primary affiliation at the end of the paragraph (name, degree, primary institution name, city, state, country)

Conclusions

Case reports constitute an essential element of publication in a cross-disciplinary journal such as Mycopathologia. Medical and veterinary case reports covering fungal pathogens are widely read as evident from their download numbers. The completeness of the case report appears to have a positive effect on the downloads; the topics and geographic origin have a neutral influence; and recency shows a negative correlation. There is no correlation between downloads and the citations of case reports as measured by Google Scholar and Scopus. The use of the checklists for the authors and a new format termed MycopathologiaIMAGES are likely to improve the quality and relevance of case reports.

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Copyright information

© Springer Nature B.V. 2018

Authors and Affiliations

  1. 1.Host-Pathogen Interaction Study Group, SFR ICAT 4208, Univ Angers, Univ Brest, Institute of Biology in Health, IRISCHUAngersFrance
  2. 2.New York State Department of HealthWadsworth CenterAlbanyUSA

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