Effective amelioration of the Lucio phenomenon with adjuvant tofacitinib therapy in a patient with dual infection of Mycobacterium leprae and Mycobacterium lepromatosis: a case report from India
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Introduction. The Lucio phenomenon (LP) is a characteristic reaction pattern seen in patients with diffuse lepromatous leprosy (DLL). Dual infection with Mycobacterium leprae and Mycobacterium lepromatosis in DLL has been confirmed from other endemic countries but not previously documented from India. Conventionally, LP is treated with a high dose of systemic glucocorticoid (GC) and anti-leprosy treatment (ALT). Here we report a case of leprosy lymphadenitis at initial presentation in a patient with LP and DLL due to dual infection with M. leprae and M. lepromatosis who responded favourably to tofacitinib as adjuvant to ALT and systemic GC therapy.
Case report. A 20- to 30-year-old man presented with swelling over the bilateral inguinal region, pus-filled skin lesions with multiple ulcers, fever and joint pain. Post-hospitalization investigations showed the presence of anaemia, leukocytosis, and elevated acute and chronic inflammatory markers. Skin and lymph node biopsies were suggestive of LP and leprosy lymphadenitis. The presence of M. leprae and M. lepromatosis was confirmed by PCR followed by DNA sequencing of PCR amplicons from tissue. Despite anti-leprosy treatment, oral GC and thalidomide therapy, the patient continued to develop new lesions. One month after the commencement of adjuvant tofacitinib, the patient showed excellent clinical improvement with healing of all existing lesions and cessation of new LP lesions.
Conclusion. Our case confirms the presence of dual infection with M. leprae and lepromatosis in India. Lymph node involvement as an initial presentation of DLL should be considered in endemic areas. Tofacitinib may be a promising new adjuvant therapy for recalcitrant lepra reactions.
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Many thanks for your work and for addressing the reviewers' comments appropriately. However, at times, the language remains poor with grammatical errors throughout the manuscript. While it does not take away from the overall understanding of the information, I would recommend a thorough read through during the "proofing" stage of production.
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This study would be a valuable contribution to the existing literature. The reviewers have highlighted minor concerns with the work presented. Please ensure that you address their comments. Reviewers and I have reviewed your manuscript and as you can see from the largely positive comments below, there are a few suggestions on how your manuscript can be improved. The case is very interesting and would be of great interest of clinicians in the field; however it would be beneficial to describe tofacitinib and its potential role in managing these infections briefly in the Introduction as suggested; and validation of the diagnostic processes. Additionally, there are a few grammatical errors that would benefit from a thorough proof-read. Please try and address these and I look forward to receiving your revised manuscript.
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Comments to Author
The authors have described a case with possible mixed infection of M. leprae and M. lepromatosis based on the PCR results: targeting M. leprae specific repetitive sequence RLEP, while for M. lepromatosis, and nested PCR approach was used targeting single copy gene in M. lepromatosis. The authors have cited a reference Ahuja et al 2018, which was later on retracted and authors have not mentioned this in the manuscript despite being aware of this. Major comments are as follows: (i) The authors have not performed sequencing of the PCR product after heminested PCR to confirm the presence of M. lepromatosis, in addition to M. leprae specific amplcons of RLEP. Mere amplification shouldn't be considered as a confirmation of presence of M. lepromatosis. Authors should justify why the sequencing couldn't be …
Comments to Author
The authors have described a case with possible mixed infection of M. leprae and M. lepromatosis based on the PCR results: targeting M. leprae specific repetitive sequence RLEP, while for M. lepromatosis, and nested PCR approach was used targeting single copy gene in M. lepromatosis. The authors have cited a reference Ahuja et al 2018, which was later on retracted and authors have not mentioned this in the manuscript despite being aware of this. Major comments are as follows: (i) The authors have not performed sequencing of the PCR product after heminested PCR to confirm the presence of M. lepromatosis, in addition to M. leprae specific amplcons of RLEP. Mere amplification shouldn't be considered as a confirmation of presence of M. lepromatosis. Authors should justify why the sequencing couldn't be done, as some of the authors on this work have performed sequencing routinely for drug resistance related studies and are familiar with the technology and analysis part. (ii) The authors have not used additional genomic targets which are known to be specific for M. lepromatosis such as hemN gene (later re-annotated as hemW) or the multi-copy genomic targets described for M. lepromatosis genome in subsequent studies etc in recent publications. This should be explained, as these are very important findings and should be confirmed. (iii) There are typographical errors which need to be carefully corrected, such as line 114: "collaborate" to be replaced by "corroborate"? (iv) Auto-capitalization after full stop in Line 135 (M.Leprae and M. Lepromatosis) should be corrected as "M.leprae and M. lepromatosis" throughout the manuscript. (v) Line 109: sever LP from endemic countries. spelling to be corrected to severe
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
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Comments to Author
Congratulations on this case report, I only have one suggestion, in the introductio. I think you should be to make a better explanation of de Tofacitinib and its mechanism to modulate the immune response in these hosts.
Please rate the quality of the presentation and structure of the manuscript
Very good
To what extent are the conclusions supported by the data?
Strongly 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 …
Comments to Author
Congratulations on this case report, I only have one suggestion, in the introductio. I think you should be to make a better explanation of de Tofacitinib and its mechanism to modulate the immune response in these hosts.
Please rate the quality of the presentation and structure of the manuscript
Very good
To what extent are the conclusions supported by the data?
Strongly 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
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