Successful treatment of recurrent visceral leishmaniasis relapse in an immunocompetent adult female with functional hypopituitarism in Bangladesh

Tabiha Binte Hannan,Zazeba Hossain, Utshab Roy, S. M. Mahbubur Rahman, Md Sadiqur Rahman, Sadia Sabah,Md. Abu Rahat,Rajashree Chowdhury,Faria Hossain,Dinesh Mondal, Shampa Saha, Tahniyah Haq,Md Rafiqul Alam,Fazle Rabbi Chowdhury

PLOS NEGLECTED TROPICAL DISEASES(2024)

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摘要
Background Currently available treatment options are mostly effective in achieving long-term cure in visceral leishmaniasis (VL) patients. However, there have been reports of recurrence of this illness in both immunosuppressed and immunocompetent patients.Case presentation We report the first case of recurrent VL relapse in a 19-year-old immunocompetent female with functional hypopituitarism (hypogonadotropic hypogonadism with central hypothyroidism) from Bangladesh, who has been treated three times previously with optimal dosage and duration- liposomal amphotericin B (LAmB) alone and in combination with miltefosine. We treated the patient successfully with a modified treatment regimen of 10 mg/kg body weight LAmB for two consecutive days along with oral miltefosine for seven days as loading dose. For secondary prophylaxis, the patient received 3 mg/kg body weight LAmB along with oral miltefosine for seven days monthly for five doses followed by hormonal replacement. The patient remained relapse free after 12 months of her treatment completion.Conclusion In the absence of protective vaccines against Leishmania species and standard treatment regimen, this modified treatment regimen could help the management of recurrent relapse cases. Visceral leishmaniasis is a globally recognized neglected tropical disease, and Bangladesh is a major contributor of the global burden of this illness. In this article, we present a case on a 19-year-old immunocompetent female with pubertal delay, who has been diagnosed as a case of visceral leishmaniasis for 11 years and presented to us with recurrent relapses despite being treated with standard treatment regimens. The patient also developed functional hypopituitarism due to this chronic illness, which hindered her personal, familial, as well as social life. In scarcity of evidence of a standard treatment regimen for recurrent relapse cases, we treated her with high dose liposomal amphotericin B and miltefosine, along with secondary prophylaxis combining these two drugs with excellent result. Her pubertal delay was also addressed with utmost importance to re-establish her social confidence. This article could encourage physicians to outline an effective regimen to deal with these difficult-to-treat cases.
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