Severe type 1 upgrading leprosy reaction in a renal transplant recipient: a paradoxical manifestation associated with deficiency of antigen-specific regulatory T-cells?

Autor: Alberto José da Silva Duarte, Francine Brambate Carvalhinho Lemos, Neusa Y. Sakai-Valente, Maria Angela Bianconcini Trindade, Gil Benard, Flávio Jota de Paula, Ana Vieira
Jazyk: angličtina
Rok vydání: 2017
Předmět:
0301 basic medicine
Graft Rejection
Male
medicine.medical_treatment
030106 microbiology
Case Report
Leprostatic Agents
Dapsone
T-Lymphocytes
Regulatory
lcsh:Infectious and parasitic diseases
Clofazimine
03 medical and health sciences
0302 clinical medicine
Immune Reconstitution Inflammatory Syndrome
Leprosy
Immune reconstitution syndrome
medicine
Humans
lcsh:RC109-216
030212 general & internal medicine
Mycobacterium leprae
Skin
Immunosuppression Therapy
Antigens
Bacterial
biology
business.industry
Immunosuppressors
Immunosuppression
Renal transplantation
Middle Aged
medicine.disease
biology.organism_classification
Kidney Transplantation
Transplantation
Infectious Diseases
Treatment Outcome
Granuloma
Immunology
Prednisone
Drug Therapy
Combination
Rifampin
business
Regulatory T cell
Rifampicin
medicine.drug
Type 1 reaction
Zdroj: BMC Infectious Diseases, Vol 17, Iss 1, Pp 1-6 (2017)
BMC Infectious Diseases
ISSN: 1471-2334
DOI: 10.1186/s12879-017-2406-9
Popis: Background Due to its chronic subclinical course and large spectrum of manifestations, leprosy often represents a diagnostic challenge. Even with proper anti-mycobacteria treatment, leprosy follow up remains challenging: almost half of leprosy patients may develop reaction episodes. Leprosy is an infrequent complication of solid organ transplant recipients. This case report illustrates the challenges in diagnosing and managing leprosy and its reactional states in a transplant recipient. Case presentation A 53-year-old man presented 34 months after a successful renal transplantation a borderline-tuberculoid leprosy with signs of mild type 1 upgrading reaction (T1R). Cutaneous manifestations were atypical, and diagnosis was only made when granulomatous neuritis was found in a cutaneous biopsy. He was successfully treated with the WHO recommended multidrug therapy (MDT: rifampicin, dapsone and clofazimine). However he developed a severe T1R immediately after completion of the MDT but no signs of allograft rejection. T1R results from flare-ups of the host T-helper-1 cell-mediated immune response against Mycobacterium leprae antigens in patients with immunologically unstable, borderline forms of leprosy and has been considered an inflammatory syndrome in many aspects similar to the immune reconstitution inflammatory syndromes (IRS). The T1R was successfully treated by increasing the prednisone dose without modifying the other immunosuppressive drugs used for preventing allograft rejection. Immunological study revealed that the patient had a profound depletion of both in situ and circulating regulatory T-cells and lack of expansion of the Tregs upon M. leprae stimulation compared to T1R leprosy patients without iatrogenic immunosuppression. Conclusions Our case report highlights that leprosy, especially in the transplant setting, requires a high degree of clinical suspicion and the contribution of histopathology. It also suggests that the development of upgrading inflammatory syndromes such as T1R can occur despite the sustained immunosuppressors regimen for preventing graft rejection. Our hypothesis is that the well-known deleterious effects of these immunosuppressors on pathogen-induced regulatory T-cells contributed to the immunedysregulation and development T1R. Electronic supplementary material The online version of this article (doi:10.1186/s12879-017-2406-9) contains supplementary material, which is available to authorized users.
Databáze: OpenAIRE
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