Haemophagocytic lymphohistiocytosis due to Burkholderia pseudomallei in a primigravida

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Abstract

Introduction. Melioidosis is caused by Burkholderia pseudomallei, a Gram-negative, saprophytic bacillus, commonly found in soil or contaminated water. As infection with this bacterium produces a wide variety of clinical manifestations the organism is aptly called the ‘great mimicker’. Even though it is non-fastidious and an easily cultivable organism, it can be misidentified in automated identification systems.

Case report. A 24-year-old primigravida presented with complaints of fever and myalgia of 45 days’ duration. She was diagnosed to have haemophagocytic lymphohistiocytosis (HLH) based on clinical and laboratory parameters. Blood and bone marrow culture sent to the microbiology laboratory grew non-fermenting Gram-negative bacilli which were misidentified as Burkholderia cepacia by matrix-assisted laser desorption ionization-time of flight mass spectrometry (MALDI-TOF MS) technology. It was subsequently identified as B. pseudomallei by 16S rRNA gene sequencing. The patient was commenced on intensive phase therapy with intravenous ceftazidime for 2 weeks, followed by maintenance therapy with oral trimethoprim and sulfamethoxazole for 3 months. In view of HLH, she was treated with intravenous dexamethasone for 2 weeks which was later switched to oral dexamethasone for a period of 6 weeks. She responded well to the treatment, but had to undergo medical termination of her pregnancy as there was severe intrauterine growth restriction of the fetus.

Conclusion. Prognosis of melioidosis is excellent if early diagnosis and appropriate antibiotic treatment is provided. In this era of automation, it is important to determine if the suspected pathogen is listed in the database of the automated identification system.

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  1. I am pleased to tell you that your article has now been accepted for publication in Access Microbiology. The work presented is clear, well written and contributes to the literature. Thank you for addressing all reviewers comments satisfactorily and in a timely manner.

  2. Thank you for submitting your manuscript for publication in Access Microbiology. It has been examined by expert reviewers who have concluded that the work is of potential interest to the readership of Access Microbiology. However, based on the comments received, it is clear that a major revision of this manuscript will be required before a decision can be made on its publication. I will be pleased to consider a revised manuscript along with a document including a point by point response to each of the reviewers comments. Your revised manuscript may be returned to one or more of the original reviewers, along with your itemised response to the reviewers’ comments. I look forward to receiving the revised manuscript.

  3. Comments to Author

    The case report Hemophagocytic lymphohistiocytosis due to Burkholderia pseudomallei in a primigravida was reviewed. Though an interesting observation, several points need to be addressed 1. How many points of the (Henter et al Criteria 2004) were fulfilled for arriving at a diagnosis, Could it be possible Hemophagocytic lymphohistiocytosis 2. What were the results of bench side tests, Gram stain, triple disc screening 3. Was the duration of intensive phase (16days) sufficient, keeping in mind the clinical presentation and concurrent steroid therapy 4. Any follow up laboratory findings of CBC, prior to starting Co-trimoxazole, which also caries a risk of cytopenia 5. incomplete Literature review and failure to cite prior reports of Hemophagocytic lymphohistiocytosis due to Burkholderia pseudomallei

    Please rate the quality of the presentation and structure of the manuscript

    Satisfactory

    To what extent are the conclusions supported by the data?

    Partially support

    Do you have any concerns of possible image manipulation, plagiarism or any other unethical practices?

    No

    Is there a potential financial or other conflict of interest between yourself and the author(s)?

    No

    If this manuscript involves human and/or animal work, have the subjects been treated in an ethical manner and the authors complied with the appropriate guidelines?

    Yes

  4. Comments to Author

    1. Description of the case(s) Needs significant improvement. 2. Presentation of results Not adequate 3. How the style and organization of the paper communicates and represents key findings Not adequate 4. Literature analysis or discussion Lines 144 onwards, let the authors restrict to the relevance to the results obtained in the given patient. Like serology needs to be discussed in detail, which hasn't been conducted in the given case etc. 5. Any other relevant comments 1. This organism is found near the coastal areas, a line may be added about the residence of the patient. Though at present there are reports of B. pseudomalliei infection from the central area of India also. 2. Lines 28-29: The authors stated that it was misidentified as B. cepacia. Looking at the typical colony morphology, a reliable guess can be made about the differential diagnosis of dry colonies producing organisms and B. cepacia isn't amongst those two/three organisms. 3. Lines 130: Which antibiotic was specifically to this patient and for how long? Please describe it discreetly. 4. It needs linguistic improvement and the minor grammatical errors to be corrected.

    Please rate the quality of the presentation and structure of the manuscript

    Poor

    To what extent are the conclusions supported by the data?

    Partially support

    Do you have any concerns of possible image manipulation, plagiarism or any other unethical practices?

    No

    Is there a potential financial or other conflict of interest between yourself and the author(s)?

    No

    If this manuscript involves human and/or animal work, have the subjects been treated in an ethical manner and the authors complied with the appropriate guidelines?

    No: Not applicable