Spontaneous remission of fully symptomatic visceral leishmaniasis

BMC Infectious Diseases

Pierre Buffet¹  Nathalie Costedoat-Chalumeau²  Zahir Amoura⁴  Marc Thellier¹  Alain Devidas⁶  Nathalie Chartrel³  Gaëlle Leroux⁴  Mathilde Benhamou⁵  Oussama Mouri³ 

[1]UPMC Université Paris 06, Centre Immunologie et Maladies Infectieuses, Paris, F-75005, France

[2]AP-HP, Hôpital Cochin, Service de Médecine Interne. Centre de référence maladies auto-immunes et systémiques rares, Service de Médecine Interne, Paris, F-75014, France

[3]AP-HP, Hôpital Pitié-Salpêtrière, Service de Parasitologie, Paris, F-75013, France

[4]AP-HP, Hôpital Pitié-Salpêtrière, Service de Médecine Interne, Paris, F-75013, France

[5]AP-HP, Hôpital Cochin, Service de Rééducation et Réadaptation, Paris, F-75014, France

[6]Hopital Sud Francilien, Service d’Hématologie, Corbeil-Essonnes, France

关键词: Bone marrow smear;    Giemsa stained;    Real time PCR;    Hemophagocytic lymphohistiocytosis;    Spontaneous remission;    Visceral leishmaniasis;   

Others  :  1232903
DOI  :  10.1186/s12879-015-1191-6

 received in 2015-04-14, accepted in 2015-10-07,  发布年份 2015

【 摘 要 】

Background

Visceral leishmaniasis (VL), i.e., infection with Leishmania sp. associated with high fever, weight loss, massive splenomegaly and markedly altered laboratory parameters, is generally fatal if untreated. The possibility of transient spontaneous remission of fully symptomatic visceral leishmaniasis (VL) has been mentioned but, to our knowledge) has never been documented.

Case presentation

We report the first documented history of a patient with overt, confirmed VL experiencing a complete remission in the absence of any anti-leishmanial therapy. The diagnosis of VL at the time of the self-resolving episode was strongly suspected based on clinical presentation and presence of antileishmanial antibody, then unequivocally confirmed years later by the presence of an amastigote on a stored smear and the positive quantitative PCR with Leishmania-specific primers from the material scraped from this same slide

Conclusion

This report demonstrates that complete spontaneous remission may occur in patients with overt, fully symptomatic VL. VL should therefore be considered in cases of self-resolving or relapsing episodes of fever of unknown origin. Confirmation should be based on both serological tests and specific PCR on a blood sample.

【 授权许可】

   
2015 Mouri et al.

【 图 表 】

Fig 1.

【 参考文献 】

• [1]Pearson R, de Queiroz Sousa A, Jeronimo SB. Leishmania species: visceral (Kala-Azar), cutaneous and mucosal leishmaniasis. In: Mandell, Douglas, and Bennett's Principles and practice of infectious diseases. Volume 2. Mandell GL, Bennett JE, Dolin R, editors. Churchill Linvingstone, Philadelphia; 2000: p.2831-2845.
• [2]den Boer ML, Alvar J, Davidson RN, Ritmeijer K, Balasegaram M. Developments in the treatment of visceral leishmaniasis. Expert Opin Emerg Drugs. 2009; 14(3):395-410.
• [3]Bern C, Maguire JH, Alvar J. Complexities of assessing the disease burden attributable to leishmaniasis. PLoS Negl Trop Dis. 2008; 2(10): Article ID e313
• [4]Mary C, Faraut F, Lascombe L, Dumon H. Quantification of Leishmania infantum DNA by a real-time PCR assay with high sensitivity. J Clin Microbiol. 2004; 42(11):5249-5255.
• [5]Davidson RN, di Martino L, Gradoni L, Giacchino R, Gaeta GB, Pempinello R, Scotti S, Cascio A, Castagnola E, Maisto A et al.. Short-course treatment of visceral leishmaniasis with liposomal amphotericin B (AmBisome). Clin Infect Dis. 1996; 22(6):938-943.
• [6]Brustoloni YM, Lima RB, da Cunha RV, Dorval ME, Oshiro ET, de Oliveira AL, Pirmez C. Sensitivity and specificity of polymerase chain reaction in Giemsa-stained slides for diagnosis of visceral leishmaniasis in children. Mem Inst Oswaldo Cruz. 2007; 102(4):497-500.
• [7]Fisa R, Riera C, Lopez-Chejade P, Molina I, Gallego M, Falco V, Ribera E, Portus M. Leishmania infantum DNA detection in urine from patients with visceral leishmaniasis and after treatment control. Am J Trop Med Hyg. 2008; 78(5):741-744.
• [8]Ozerdem D, Eroglu F, Genc A, Demirkazik M, Koltas IS. Comparison of microscopic examination, rK39, and PCR for visceral leishmaniasis diagnosis in Turkey. Parasitol Res. 2009; 106(1):197-200.
• [9]Fraga TL, Brustoloni YM, Lima RB, Dorval ME, Oshiro ET, Oliveira J, Oliveira AL, Pirmez C. Polymerase chain reaction of peripheral blood as a tool for the diagnosis of visceral leishmaniasis in children. Mem Inst Oswaldo Cruz. 2010; 105(3):310-313.
• [10]Gradoni L, Soteriadou K, Louzir H, Dakkak A, Toz SO, Jaffe C, Dedet JP, Campino L, Canavate C, Dujardin JC. Drug regimens for visceral leishmaniasis in Mediterranean countries. Trop Med Int Health. 2008; 13(10):1272-1276.
• [11]Evans TG, Teixeira MJ, Sousa Ade Q, Pearson RD. Short report: extended follow-up of the natural history of persons infected with Leishmania chagasi. Am J Trop Med Hyg. 1995; 53(4):360-361.
• [12]Badaro R, Jones TC, Carvalho EM, Sampaio D, Reed SG, Barral A, Teixeira R, Johnson WD. New perspectives on a subclinical form of visceral leishmaniasis. J Infect Dis. 1986; 154(6):1003-1011.
• [13]Jeronimo SM, Teixeira MJ, Sousa A, Thielking P, Pearson RD, Evans TG. Natural history of Leishmania (Leishmania) chagasi infection in Northeastern Brazil: long-term follow-up. Clin Infect Dis. 2000; 30(3):608-609.
• [14]Jha TK. Evaluation of diamidine compound (pentamidine isethionate) in the treatment resistant cases of kala-azar occurring in North Bihar, India. Trans R Soc Trop Med Hyg. 1983; 77(2):167-170.
• [15]Sundar S, Murray HW. Gamma interferon in the treatment of Kala-azar and other forms of Leishmaniasis. J Assoc Physicians India. 1995; 43(5):348-350.
• [16]Romero IC, Saravia NG, Walker J. Selective action of fluoroquinolones against intracellular amastigotes of Leishmania (Viannia) panamensis in vitro. J Parasitol. 2005; 91(6):1474-1479.
• [17]Bianciardi P, Fasanella A, Foglia Manzillo V, Trotta T, Pagano A, Sorino S, Gradoni L, Oliva G. The efficacy of enrofloxacin, alone or combined with metronidazole, in the therapy of canine leishmaniasis. Parasitol Res. 2004; 93(6):486-492.
• [18]Cortazar TM, Coombs GH, Walker J. Leishmania panamensis: comparative inhibition of nuclear DNA topoisomerase II enzymes from promastigotes and human macrophages reveals anti-parasite selectivity of fluoroquinolones, flavonoids and pentamidine. Exp Parasitol. 2007; 116(4):475-482.
• [19]da Silva MR, Stewart JM, Costa CH. Sensitivity of bone marrow aspirates in the diagnosis of visceral leishmaniasis. Am J Trop Med Hyg. 2005; 72(6):811-814.
• [20]Gagnaire MH, Galambrun C, Stephan JL. Hemophagocytic syndrome: A misleading complication of visceral leishmaniasis in children--a series of 12 cases. Pediatrics. 2000; 106(4): Article ID E58
• [21]Henter JI, Horne A, Arico M, Egeler RM, Filipovich AH, Imashuku S, Ladisch S, McClain K, Webb D, Winiarski J et al.. HLH-2004: Diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis. Pediatr Blood Cancer. 2007; 48(2):124-131.
• [22]Rajagopala S, Dutta U, Chandra KS, Bhatia P, Varma N, Kochhar R. Visceral leishmaniasis associated hemophagocytic lymphohistiocytosis--case report and systematic review. J Infect. 2008; 56(5):381-388.