bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

1 Multi-locus characterization and phylogenetic

2 inference of Leishmania spp. in from

3 Northwest

4

5 Han Chen1¶, Jiao Li1¶, Junrong Zhang1, Jinlei He1, Jianhui Zhang1, Zhiwan Zheng1, Dali

6 Chen1*, Jianping Chen1*

7

8 1 Department of Parasitology, West China School of Basic Medical Sciences and

9 Forensic Medicine, Sichuan University, Chengdu, Sichuan, China

10

11 * Corresponding author

12 E-mail: [email protected] (DLC); [email protected] (JPC)

13

14 ¶ These authors contributed equally to this work.

15 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

16 Abstract

17 Background: Leishmaniasis caused by protozoan parasite Leishmania is a neglected

18 disease which is endemic in the northwest of China. were considered to be the

19 potential reservoir hosts for mammalian Leishmaniasis, and Leishmania had been

20 detected in lizards from the epidemic area in the northwest of China. To date, few

21 studies are focused on the natural infection of snakes with Leishmania.

22 Methods: In this study, 15 snakes captured from 10 endemic foci in the northwest of

23 China were detected Leishmania spp. on the base of mitochondrial cytochrome b, heat

24 shock protein 70 gene and ribosomal internal transcribed spacer 1 regions, and

25 identified with phylogenetic and network analyses.

26 Result: In total, Leishmania gene was found in 7 snakes. The phylogenetic inference

27 trees and network analysis suggests that the species identification was confirmed as

28 Leishmania donovani, Leishmania turanica and Leishmania sp.

29 Conclusion: Our work is the first time to investigate the natural Leishmania spp.

30 infection of snakes in the northwest of China. Mammalian Leishmania was discovered

31 in snakes and the reptilian Leishmania was closely related to the clinical strains both

32 prompt the importance of snakes in the disease cycle. To indicate the epidemiological

33 involvement of snakes, a wide sample size in epidemic area and the pathogenic features

34 of reptilian Leishmania promastigotes are recommended in the future research.

35 Keywords: Leishmania; ; cyt b; Hsp70; ITS1; phylogeny; northwest China

36 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

37 Introduction

38 Leishmaniasis is a neglected disease caused by infection with flagellate protozoan

39 parasite Leishmania of the family Trypanosomatidae. There are three main forms of

40 leishmaniasis: visceral or kala-azar (VL), cutaneous (CL) and mucocutaneous (MCL).

41 Over 20 Leishmania species known to be infective to humans are transmitted by the

42 bite of infected sand flies of which at least 98 species were proven or probable vectors

43 worldwide [1]. An estimated 2 million new cases and 50 000 deaths occur in over 98

44 countries annually [2]. In China, which is one of 14 high-burden countries of VL [3],

45 there exists three epidemiological types: anthroponotic VL (AVL), mountain-type

46 zoonotic VL (MT-ZVL), and desert-type ZVL (DT-ZVL) [4]. Acknowledging the

47 information on the epidemiology including the vector and reservoir hosts of the

48 disease is better to understand the disease and its control.

49 In view of the topography in the northwest of China, DT-ZVL is found largely in

50 the oases and deserts including the southern and eastern of Xinjiang Uygur

51 Autonomous Region, the western part of the Inner Autonomous Region, and

52 northern Gansu province [5]. Surveillance of the disease reflects that DT-ZVL is

53 persistence with sporadic outbreaks and still not under control now [5]. Sand flies

54 (Phlebotominae) were recognized as the transmission vector in the traditional sense,

55 while ticks (Ixodoidea) had been reported to be the vector in the transmission of canine

56 VL [6–9]. In addition to humans, there are various kinds of hosts, mainly mammals

57 such as canids, hyraxes and rodents, which could be the source of infection as the

58 reservoirs. Because transmission occurs in a complex biological system involving the bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

59 human host, parasite, vectors and in some cases an animal reservoir host, the control of

60 Leishmaniasis is pretty complex. As for the desert in the northwest of China, there is

61 no consistent agreement regarding dissemination of the actors playing the key roles in

62 leishmaniasis.

63 Moreover, reptiles, mainly lizards, were found harboring Leishmania parasites

64 with controversies in the part of spreading the disease [10]. Blood cells of lizards

65 containing amastigotes were first found by Chatton and Blanc in Tarentola mauritanica

66 from southern Tunisia [11] and then several cases from the same lizard species were

67 reported in the next decades [12]. Wenyon provisionally named the leptomonad

68 flagellates in gecko as Leishmania tarentolae [13]. Killick-Kendrick recognized

69 Sauroleishmania as a separate genus for the leishmanial parasites of reptiles [14], later

70 DNA sequence-based phylogenies had clearly placed it within the Leishmania genus as

71 a secondarily derived development from the mammalian species [15–17]. Most studies

72 were focused on the infection of Leishmania parasites in lizards, but few were in snakes

73 except Belova and Bogdanov cultured promastigotes from the blood of five species of

74 snakes in the Turkmen S.S.R. [14,18].

75 In reality, the detection of reptilian Leishmania is rare in China. Zhang et al. was

76 the first team to detect Leishmania via molecular methods from the lizards captured in

77 the northwest of China [19]. Interestingly, besides reptilian Leishmania, mammalian

78 Leishmania species (L. donovani and L. tropica) were involved despite

79 Sauroleishmania was considered not to infect humans or other mammals [20,21]. This

80 may support the potential reservoir role of lizards for human leishmaniasis. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

81 Similarly, we employed the highly sensitive polymerase chain reaction (PCR) to

82 detect the presence of Leishmania spp. in snakes. The sequences of various genetic

83 markers have been used successfully to infer the phylogenetic relationships within

84 Leishmania including the sequences of DNA polymerase α [15], RNA polymerase II

85 [15], 7SL RNA [22–24], ribosomal internal transcribed spacer [25,26], mitochondrial

86 cytochrome b gene [27,28], heat shock protein 70 gene [29,30] and the N-

87 acetylglucosamine-1-phosphate transferase gene [31]. Therefore, this study was based

88 on three genetic loci, i.e., cytochrome b (cyt b), heat shock protein 70 (Hsp70), and

89 internal transcribed spacer 1 (ITS1) region to genetically characterize Leishmania spp.

90 detected in 15 snakes, and conduct tree-based species delimitation to infer the

91 phylogenetic positions by comparison with some representative Leishmania sequences

92 retrieved from GenBank. It was the first time to survey the infected snakes of

93 Leishmania in the northwest of China and try to reveal the primary evidence on the

94 epidemic role of snakes for human leishmaniasis.

95

96 Materials and methods

97 Study area and sampling

98 15 snakes identified as 3 species by morphological characters were captured alive

99 by hand and snake hooks at 10 sites across the endemic foci of VL in the northwest of

100 China (Table 1). These sites are located in arid desert areas with the altitude ranging

101 from 537 to 1222 m above sea level. Franchini [32,33] seems to have observed rare

102 amastigotes from the liver of lizards, and Shortt & Swaminath [34] cultured a strain bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

103 Leishmania on NNN (Novy-MacNeal-Nicolle) medium from the liver of lizards.

104 Consequently, a part of the liver was taken from the abdominal cavity of each snake to

105 detect the presence of Leishmania via PCR. In total, 15 liver specimens were collected

106 in Eppendorf tube and stored at -20°.

107 All surgery was performed under sodium pentobarbital anesthesia, and all efforts

108 were made to minimize suffering. The protocol was approved by the ethics committee

109 of Sichuan University (Protocol Number: K2018056).

110

111 Table 1 List of sampling localities, snake species and sample size in this study

Site Label Locality Species sample

size

P1 Shawan county, Tarbagatay lineolatus 1

Prefecture, Xinjiang

P2 Tekes County, Ili Kazak Gloydius halys 1

Autonomous Prefecture, Xinjiang

P3 Tekes County, Ili Kazak Natrix tessellate 1

Autonomous Prefecture, Xinjiang

P4 Yiwu County, Hami, Xinjiang Psammophis lineolatus 1

P5 Yizhou Area, Hami, Xinjiang Psammophis lineolatus 5

P6 Dunhuang, Jiuquan City, Gansu Psammophis lineolatus 1

Province

P7 Heshuo, Bayingol Mongolian Psammophis lineolatus 2 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

Autonomous Prefecture, Xinjiang

P8 Makit County, Kashgar Prefecture, Psammophis lineolatus 2

Xinjiang

P9 Yopurga County, Kashgar Psammophis lineolatus 1

Prefecture, Xinjiang

P10 Yengisar County, Kashgar Psammophis lineolatus 2

Prefecture, Xinjiang

total 15

112

113 DNA extraction, amplification, cloning and sequencing

114 protocols

115 Total genomic DNA was extracted from liver tissues using the commercial kit,

116 TIANamp® Genomic DNA Kit (TIANGEN Bio, Beijing, China) following the

117 protocols of the manufacturer. PCR primers specific for the Leishmania synthesized by

118 Tsingke Biological Technology Co., Ltd (Chengdu, China) were used to amplify cyt b

119 [35], Hsp70 [36] and ITS1 [37] gene fragments by PrimeSTAR Max DNA polymerase

120 (TaKaRa Bio, Shiga, Japan) according to the manufacturer’s instruction. We picked

121 Leishmania strain MHOM/CN/90/SC10H2 as the positive control while preparing the

122 mixture, and the negative control was treated without template DNA. The PCR products

123 were purified by excision of the band from agarose gel using the Universal DNA

124 Purification Kit (TIANGEN Bio, Beijing, China), and ligated into a pGEM®-T vector

125 (Promega Co., Madison, USA) with added the poly (A) tails. After transformation of bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

126 Escherichia coli DH5α competent cells with the ligation product and X-gal blue-white

127 screening, 8 positive colonies were selected by PCR using the plasmid primers T7 and

128 SP6, and grown in LB/ampicillin medium. DNA was extracted and sequenced at

129 Tsingke Biological Technology Co., Ltd (Chengdu, China). The chromatograms were

130 validated and assembled in BioEdit v7.2.6 [38].

131

132 Sequence alignment and phylogenetic analyses

133 GenBank searches were performed to initially identify the species of the original

134 DNA sequences using BLASTn (https://blast.ncbi.nlm.nih.gov/Blast.cgi). And all the

135 nucleotide sequences generated in this study have been submitted to the GenBank

136 database. The sequences were multiple-aligned with a set of Leishmania strains of each

137 locus examined in this study retrieved from the GenBank using ClustalW of MEGA

138 (Molecular Evolutionary Genetic Analysis v7.0.26 [39]) with its default option and

139 refined manually. The alignments were further trimmed to exclude regions with missing

140 data and then distinct haplotypes were defined by DAMBE v7.0.1 [40,41]. The

141 evolutionary history was inferred by phylogenetic tree construction using Bayesian

142 inference (BI). Gaps were treated as missing data and each haplotype was treated as a

143 taxon in the analyses. The program PartitionFinder v2.1.1 [42] was used to select the

144 most appropriated substitution model of all phylogenetic analyses. Bayesian analyses

145 were carried out using the program MrBayes v3.2 [43] which the trees were started

146 randomly. 2 parallel sets of 4 simultaneous Monte Carlo Markov chains (3 hot and 1

147 cold) were run for 10,000,000 generations until convergence was reached (stopval = bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

148 0.01) and the trees were sampled for every 1000 generations. Temperature heating

149 parameter was set to 0.2 (temp = 0.2) and burn-in was set to 25% (burninfrac = 0.25).

150 The results of Bayesian analyses were visualized in FigTree v1.4.3 (available at

151 http://tree.bio.ed.ac.uk/software/figtree/).

152

153 Network reconstructions

154 Due to the limit of bifurcating tree to represent intraspecific gene evolution,

155 haplotype networks may more effectively portray the relationships among haplotypes

156 within species. The phylogenetic networks were constructed by using the Median

157 Joining (MJ) network reconstruction method in NETWORK v5.0.0.3 (available at

158 http://www.fluxus-engineering.com/sharenet.htm) [44,45].

159

160 Results

161 Leishmania infection of the captured snakes in the study area

162 Through preliminary determination of identity using BLASTn, the total

163 prevalence of Leishmania DNA detected via PCR was 46.7% (7/15). 5 (33.3%) snakes

164 were detected as positive for cyt b, 4 (26.7%) snakes for Hsp70 and 2 (13.3%) for ITS1.

165 Considering the sensitivity of different loci, two samples from P1 and P2 were

166 amplified successfully in all three loci, while other samples were detected by only one

167 locus in this study. The snakes from P1, 2, 3, 4, 5, 6, 9 were Leishmania-positive of all

168 the 10 foci as shown in Fig 1.

169 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

170 Figure 1 Sampling sites of snakes in Northwest China, along with the current

171 foci of endemicity of anthroponotic visceral leishmaniasis (AVL) and desert-type

172 zoonotic visceral leishmaniasis (DT-ZVL) in China. The site numbers P1-P10

173 correspond to those in Table 1. Solid five-pointed star (★) represents the endemic focus

174 of AVL and solid triangle (▲) represents the endemic focus of DT-ZVL [4,5]. The

175 species of Leishmania detected from the samples are shown for different colors (yellow:

176 L. donovani complex, blue: L. turanica, pink: Leishmania sp.)

177

178 Sequence characteristics

179 After the haplotypes were defined, 10 cyt b sequences, 7 Hsp70 sequences and 10

180 ITS1 sequences were obtained in this study and deposited in the GenBank database

181 under Accession No. MK330198-MK330207, MK330208-MK330214 and

182 MK300708-MK300717, respectively (Table 2). PCR amplification of each target locus

183 resulted in amplicons of the expected sizes as follows: cyt b (543 bp), Hsp70 (738-767

184 bp), and ITS1 (328-334 bp). The GC contents were 23.4% for cyt b, 64.9% for Hsp70

185 and 41.8% for ITS1, respectively. Thus, both their lengths and their GC contents are

186 within the range of corresponding sequences of other Leishmania species.

187

188 Table 2 List of the lizard samples, origin, isolated Leishmania spp. and GenBank

189 accession numbers.

Snake Voucher Origi Isolated Haplotyp GenBank Sequenc

species number n Leishmani e number accession e length bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

a spp. (bp)

(A)cyt b

Psammophi Guo470 P1 L. C1 MK33019 543

s lineolatus 7 donovani 8

L. sp. C6 MK33019 543

9

Gloydius Guo470 P2 L. C1 MK33020 543

halys 8 donovani 0

Natrix Guo470 P3 L. C2 MK33020 543

tessellata 9 donovani 1

L. turanica C4 MK33020 543

2

L. sp C5 MK33020 543

3

L. sp. C6 MK33020 543

4

Psammophi Guo471 P4 L. C3 MK33020 543

s lineolatus 0 donovani 5

L. turanica C4 MK33020 543

6

Psammophi Guo699 P9 L. sp. C6 MK33020 543

s lineolatus 5 7 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

(B) Hsp70

Psammophi Guo470 P1 L. H1 MK33020 738

s lineolatus 7 donovani 8

L. infantum H2 MK33020 738

9

Gloydius Guo470 P2 L. infantum H2 MK33021 738

halys 8 0

Psammophi Guo402 P6 L. sp. H3 MK33021 767

s lineolatus 3 1

L. sp. H4 MK33021 766

2

L. sp. H5 MK33021 767

3

Psammophi Guo402 P5 L. sp. H5 MK33021 767

s lineolatus 6 4

(C)ITS1

Psammophi Guo470 P1 L. sp. I1 MK30070 334

s lineolatus 7 8

L. sp. I2 MK30070 328

9

L. sp. I3 MK30071 332

0 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

L. sp. I4 MK30071 330

1

L. sp. I5 MK30071 330

2

L. sp. I6 MK30071 330

3

L. sp. I7 MK30071 330

4

Gloydius Guo470 P2 L. sp. I8 MK30071 330

halys 8 5

L. sp. I9 MK30071 332

6

L. sp. I10 MK30071 328

7

190

191 Phylogenetic relationship

192 Prior to the phylogenetic analyses, the most adequate models of nucleotide

193 substitution were selected by PartitionFinder v2.1.1: GTR +G and GTR+I+G for cyt b,

194 HKY+I for Hsp70, K80+G for ITS1. Using Bayesian inference method, the trees

195 showed similar phylogenetic topology for all three loci supported by posterior

196 probability (PP) values.

197 Regarding the BI tree inferred from each locus, the phylogenetic analyses inferred bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

198 from the aligned matrix of cyt b (Fig 2 (A)) separated two distance clades, the Viannia

199 isolates apparently formed an independent clade (PP=1.0) outside the clusters of those

200 other species (PP=1.0), while the subgenera Leishmania and Sauroleishmania were still

201 separated into different branches (PP=1.0 and 0.56, respectively). The isolates C1, C2

202 and C3 were clustered with L. donovani and L. infantum (PP=1.0). The isolate C4 was

203 shared an identical cyt b sequence to a L. turanica isolate and they were grouped with

204 L.arabica, L. major, L. tropica, L. aethiopica and L. killicki in a subclade (PP=0.91)

205 separated with L. donovani complex. The isolate C6 shared identical sequences with

206 Chinese Leishmania sp. isolates from Yang et al. [26] was clustered in a separated clade

207 (PP=0.96) together with C5 and other Leishmania sp.

208

209 Figure 2 The majority-rule consensus tree (midpoint rooted) of three genetic loci

210 inferred from Bayesian inference by using MrBayes v.3.2, and the corresponding

211 median-joining networks were implemented by NETWORK v5.0.0.3: (A) Cyt b, (B)

212 Hsp70, (C)ITS1.

213

214 The Hsp70 analysis (Fig 2 (B)) showed a similar structure to the phylogenetic tree

215 of cyt b: Viannia (PP=1.0), Sauroleishmania (PP=1.0) and Leishmania (PP=0.6). The

216 isolates H1 and H2 searching by BLASTn (Megablast) revealed over 98% identity with

217 L. donovani and L. infantum respectively, while both of them were nested within L.

218 donovani complex. In this subclade, the isolates of other species of Leishmania

219 subgenus (except L. mexicana clustered by the PP 1.0) were not in reciprocally bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

220 monophyletic groups. The isolates H3, H4 and H5 were clustered in a separated clade

221 (PP=1.0) with Leishmania sp. from China.

222 For the ITS1-5.8S region, the phylogenetic tree as Fig 2 (C) shown was constructed

223 by three clades with high support (PP=1.0, 0.97 and 1.0, respectively), corresponding

224 to the three subgenera Viannia, Leishmania, and Sauroleishmania. All the 10 fragments

225 from snakes formed a strongly supported cluster with Leishmania sp. isolated from

226 lizards [19] (PP=1.0), and revealed a close relationship of isolates from Sergentomyia

227 minuta in Europe.

228

229 Median Joining network

230 To get additional insight into the relationships among the strains, we analyzed our

231 data sets using the Median Joining algorithm network approach. The MJ analysis of

232 three regions was congruent with the topology described in the BI trees, and no

233 haplotype was shared among the species groups.

234 The network of cyt b in Fig 2 (A) showed that the isolate C6 and a L. donovani

235 strain from Xinjiang, China (HQ908267) seemed to be the central haplotypes. C1

236 (MK330198 isolated from two snakes captured at P1 and MK330200 from P2), together

237 with C2 (MK330201 from P3) and C3 (MK330205 from P4) were most closely related

238 to the central HQ908267, within 2 mutational steps. C4 (from P3 and P4) shared the

239 same haplotype with one strain of L. turanica from Russia, and only 2 and 3 steps away

240 from the Chinese strains L. turanica (from Xinjiang) and L. gerbilli (from Gansu)

241 respectively. The Leishmania sp. isolate C6 (from P1 and P9) as the central haplotype bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

242 shared the same sequences to Leishmania sp. from China with a wide geographical

243 distribution, including Shandong (HQ908255), Sichuan (HQ908260, HQ908264),

244 Gansu (HQ908263, HQ908271).

245 For the Hsp70 region, the haplotype network of Fig 2 (B) could intuitively reflect

246 the genetically small distances between the obtained sequences H1 (MK330208

247 isolated from P1), H2 (MK330209 and MK330210 from P1 and P2, respectively) and

248 the reference L. donovani and L. infantum strains from India, Sudan and the northwest

249 of China. The haplotype H5 shared by the isolates from P5 (MK330213) and P6

250 (MK330214) was the interior haplotype of Leishmania sp. and may be older than other

251 tip haplotypes H3 (MK330211) or H4 (MK330212) which were both from P6). It

252 reflected a close distance between JX021439 (Xinjiang, China) and the interior

253 haplotype as three mutational steps.

254 As shown in the haplotype network of ITS1-5.8S in Fig 2 (C), the Leishmania sp.

255 group was centered around haplotype KU194967 (Dunhuang, China). All haplotypes

256 found in this group of Sauroleishmania isolated from lizards of the northwest of China

257 were very closely related to each other. One strain from Tuokexun (KU194973) shared

258 the same haplotype with I4 (MK300711 from P1). Similarly, two strains from

259 Dunhuang (KU194969, KU194971) and another one from Lukchun (KU194939)

260 shared the same haplotype with I10 (MK300709 from P2). In addition, three strains

261 isolated from Sergentomyia minuta of Europe shared a close distance with the

262 Sauroleishmania group as less than 3 steps.

263 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

264 Discussion

265 This study was the first time to detect the prevalence of Leishmania infection in

266 snakes by DNA sequencing. Our result showed that the snakes from 7 loci of the 10

267 study areas were detected positive for Leishmania. Yopurga county, Hami and

268 Dunhuang are the DT-ZVL epidemic foci [5], while Shawan and Texes county are lack

269 of data currently.

270 Besides one snake (Natrix tessellate) from Texes were detected three Leishmania

271 species (including L. donovani, L. turanica and Leishmania sp.), the snakes from other

272 positive foci were detected 1-2 species. Leishmania sp. was the most widely distributed,

273 while the distribution of the other two species was lack of the significant geographic

274 specificity in this study owing to the limit of the sample size.

275 The effect of many factors (e.g., the condition of specimen, the DNA extraction

276 methodology, the length of amplified gene sequences and the PCR protocols) would

277 cause different positive rates. Amplification of multiple gene sequences could

278 significantly increase the comprehensiveness of the Leishmania infections to be

279 investigated in snakes. Various genetic loci had been developed to detect Leishmania,

280 such as kinetoplast DNA (kDNA) genes, nuclear DNA (nDNA) genes and ribosomal

281 RNA (rRNA) genes [25,46,47]. Cyt b, one of the cytochromes encoded on the

282 kinetoplast DNA maxicircle, is considered one of the most useful markers that had

283 delimitated most tested Leishmania species for phylogenetic work. Hsp70 gene, an

284 antigen gene on the chromosome, is the most optimal marker for the species

285 discrimination and phylogenetic inference. ITS1, one of the highly variable regions of bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

286 rRNA gene, have been successfully used to resolve taxonomical questions and to

287 determine phylogenetic affinities among closely related Leishmania species. Therefore,

288 three genetic loci were carried out in this study and the positive rates were different

289 indeed. Two samples from Shawan and Tekes were detected Leishmania sp. and L.

290 donovani by all the three genetic loci while other samples were detected by only one

291 genetic locus. It was suggested that different Leishmania primers might have different

292 selection preferences for the gene amplification. At the same time, the use of different

293 primers to amplify the corresponding gene fragments did significantly improve the

294 detection rate of infected snake samples.

295 It was curious about the spreading mechanism of Leishmania in reptiles. Wilson

296 & Southgate assumed that transmission was achieved by the host eating an

297 infected sand fly [48,49]. Nevertheless, there were some reptile-feeding species of

298 phlebotomine as we known [50,51]. The same as lizards, it was reasonable that snakes

299 could be infected naturally. Furthermore, there were several reports of snake

300 trypanosome [52–54] which was very close to Leishmania in phylogeny. In view of the

301 small sample size of each location and species, the infection rate does not make much

302 sense. Nevertheless, all three detected snake species in the endemic foci of VL were

303 found Leishmania DNA even the sample size of both Gloydius halys and Natrix

304 tessellate was 1. Therefore, a wide range of sample size would be necessary for the

305 further study to enrich our understanding of the natural infections of Leishmania

306 parasites in snakes.

307 One of the most noteworthy findings in the present study was that two mammalian bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

308 Leishmania taxa, i.e., L. donovani complex and L. turanica appeared to occur in snakes.

309 Previously, Zhang et al. had a similar discovery in lizards [19]. They believe lizards

310 played a reservoir role for human leishmaniasis because of the common ancestry of the

311 obtained haplotypes of L. tropica and clinical samples. At present, apart from the above

312 study, there are few studies of mammalian parasites natural infections in reptiles.

313 Belova [10] carried out intensive research on reptilian and mammalian Leishmania and

314 found that the mammalian parasites (such as L. donovani, L. tropica, and some other

315 promastigotes recovered from sandflies) were infective to lizards. To determine that the

316 reptile is reservoir host of human leishmaniasis, the Leishmania strain collection from

317 VL patients in the epidemic foci and phylogenetic analyses with the reptilian strains are

318 needed.

319 On the other hand, the Leishmania sp. isolated from snakes in this study shared

320 the same haplotypes or located in the close proximity to the strains from VL patients or

321 canines in China as the network shown. Indeed, the same Chinese Leishmania sp.

322 isolates were speculated to be closely related to the lizard-infected strains, L. tarentolae,

323 on the basis of COII [55] and cyt b [28] genes. Analogously, L. adleri was found to

324 cause transient CL in humans by Manson-Bahr & Heisch [56], and could infect rodents

325 which the strains were isolated by Adler [57] and Coughlan et al. [58]. L. tarentolae

326 was chosen as a candidate vaccine against VL in virtue of the character that the parasites

327 could invade in human macrophages and exist as amastigotes in mammals with slower

328 replication [59]. These studies seem to be contradictory with the former which indicated

329 that Sauroleishmania was not pathogenic to human [10]. For the reason that the Chinese bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

330 strains from patients or canines were isolated in the 70s to 90s of last century, the

331 medical records were no longer available, and we could not confirm whether the

332 patients got leishmaniasis due to Sauroleishmania or co-infection with other

333 mammalian Leishmania. Thus, to further explore the pathogenicity of Sauroleishmania,

334 it is recommended to isolate and culture the promastigotes from liver or blood of

335 reptiles and study the virulence to mammals.

336 In conclusion, the present study is the first time to investigate the natural

337 Leishmania spp. infection of snakes in the northwest of China. The species

338 identification was primarily achieved by comparisons of the obtained sequences with

339 the GenBank database and determined the belongs with phylogenetic and network

340 analyses, including Leishmania sp., L. donovani, L. turanica. This is the first time to

341 discover mammalian Leishmania in snakes, after the similar finding in lizards. The

342 Sauroleishmania strains in this study closely related to the clinical isolates, which

343 suggests us to be cautious about the widely accepted view that Sauroleishmania is

344 nonpathogenic to human. These findings invite us to ponder the importance of snakes

345 in the disease cycle. However, more snake samples in epidemic foci and the pathogenic

346 features of Sauroleishmania promastigotes are required for indicating their

347 epidemiological involvement.

348

349 Acknowledgments

350 We are grateful to Xianguang Guo (Chengdu Institute of Biology, Chinese

351 Academy of Sciences) for his assistance in fieldwork. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

352

353 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

354 Reference

355 1. WHO | Global leishmaniasis update, 2006–2015: a turning point in leishmaniasis

356 surveillance. In: WHO [Internet]. [cited 16 Nov 2018]. Available:

357 http://www.who.int/leishmaniasis/resources/who_wer9238/en/

358 2. WHO Expert Committee on the Control of the Leishmaniases, World Health

359 Organization, editors. Control of the leishmaniases: report of a meeting of the

360 WHO Expert Committee on the Control of Leishmaniases, Geneva, 22-26 March

361 2010. Geneva: World Health Organization; 2010.

362 3. WHO | Leishmaniasis in high-burden countries: an epidemiological update based

363 on data reported in 2014. In: WHO [Internet]. [cited 16 Nov 2018]. Available:

364 http://www.who.int/leishmaniasis/resources/who_wer9122/en/

365 4. Lun Z-R, Wu M-S, Chen Y-F, Wang J-Y, Zhou X-N, Liao L-F, et al. Visceral

366 Leishmaniasis in China: an Endemic Disease under Control. Clin Microbiol Rev.

367 2015;28: 987–1004. doi:10.1128/CMR.00080-14

368 5. Wang J-Y, Cui G, Chen H-T, Zhou X-N, Gao C-H, Yang Y-T. Current

369 epidemiological profile and features of visceral leishmaniasis in people’s republic

370 of China. Parasit Vectors. 2012;5: 31. doi:10.1186/1756-3305-5-31

371 6. Coutinho MTZ, Bueno LL, Sterzik A, Fujiwara RT, Botelho JR, De Maria M, et

372 al. Participation of Rhipicephalus sanguineus (Acari: Ixodidae) in the

373 epidemiology of canine visceral leishmaniasis. Vet Parasitol. 2005;128: 149–155. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

374 doi:10.1016/j.vetpar.2004.11.011

375 7. Paz GF, Ribeiro MFB, Michalsky ÉM, da Rocha Lima ACVM, França-Silva JC,

376 Barata RA, et al. Evaluation of the vectorial capacity of Rhipicephalus sanguineus

377 (Acari: Ixodidae) in the transmission of canine visceral leishmaniasis. Parasitol

378 Res. 2009;106: 523. doi:10.1007/s00436-009-1697-1

379 8. Dantas-Torres F, Lorusso V, Testini G, de Paiva-Cavalcanti M, Figueredo LA,

380 Stanneck D, et al. Detection of Leishmania infantum in Rhipicephalus sanguineus

381 ticks from Brazil and Italy. Parasitol Res. 2010;106: 857–860.

382 doi:10.1007/s00436-010-1722-4

383 9. Solano-Gallego L, Rossi L, Scroccaro AM, Montarsi F, Caldin M, Furlanello T,

384 et al. Detection of Leishmania infantum DNA mainly in Rhipicephalus sanguineus

385 male ticks removed from dogs living in endemic areas of canine leishmaniosis.

386 Parasit Vectors. 2012;5: 98. doi:10.1186/1756-3305-5-98

387 10. Belova EM. Reptiles and their importance in the epidemiology of leishmaniasis.

388 Bull World Health Organ. 1971;44: 553.

389 11. Chatton E, Blanc G. Existence de corps leishmaniformes dans les hématoblastes

390 d’un gecko barbaresque Tarentola mauritanica L Gunth. CR Soc Biol. 1914;77:

391 430–433.

392 12. Sam R. Telford J. Hemoparasites of the Reptilia : Color Atlas and Text [Internet].

393 CRC Press; 2016. doi:10.1201/9781420080414 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

394 13. Wenyon CM. Observations on the Intestinal Protozoa of Three Egyptian Lizards,

395 with a Note on a Cell-invading Fungus. Parasitology. 1920;12: 350–365.

396 doi:10.1017/S0031182000014347

397 14. Killick-Kendrick R, Lainson R, Rioux J-A, Sar’janova VM. The of

398 Leishmania-like parasites of reptiles. 1986;

399 15. Croan DG, Morrison DA, Ellis JT. Evolution of the genus Leishmania revealed

400 by comparison of DNA and RNA polymerase gene sequences. Mol Biochem

401 Parasitol. 1997;89: 149–159. doi:10.1016/S0166-6851(97)00111-4

402 16. Noyes HA, Arana BA, Chance ML, Maingon R. The Leishmania hertigi

403 (Kinetoplastida; Trypanosomatidae) Complex and the Lizard Leishmania: Their

404 Classification and Evidence for a Neotropical Origin of the Leishmania-

405 Endotrypanum Clade. J Eukaryot Microbiol. 1997;44: 511–517.

406 doi:10.1111/j.1550-7408.1997.tb05732.x

407 17. Orlando TC, Rubio MAT, Sturm NR, Campbell DA, Floeter-Winter LM.

408 Intergenic and external transcribed spacers of ribosomal RNA genes in lizard-

409 infecting Leishmania: molecular structure and phylogenetic relationship to

410 mammal-infecting Leishmania in the subgenus Leishmania (Leishmania). Mem

411 Inst Oswaldo Cruz. 2002;97: 695–701. doi:10.1590/S0074-02762002000500020

412 18. Belova EM, Bogdanov OP. [Incidence of leptomonad infection in snakes of the

413 Turkmen SSR]. Med Parazitol (Mosk). 1969;38: 304–306. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

414 19. Zhang J-R, Guo X-G, Liu J-L, Zhou T-H, Gong X, Chen D-L, et al. Molecular

415 detection, identification and phylogenetic inference of Leishmania spp. in some

416 desert lizards from Northwest China by using internal transcribed spacer 1 (ITS1)

417 sequences. Acta Trop. 2016;162: 83–94. doi:10.1016/j.actatropica.2016.06.023

418 20. Rioux JA, Lanotte G, Serres E, Pratlong F, Bastien P, Perieres J. Taxonomy of

419 Leishmania. Use of isoenzymes. Suggestions for a new classification. Ann

420 Parasitol Hum Comparée. 1990;65: 111–125. doi:10.1051/parasite/1990653111

421 21. Akhoundi M, Kuhls K, Cannet A, Votýpka J, Marty P, Delaunay P, et al. A

422 Historical Overview of the Classification, Evolution, and Dispersion of

423 Leishmania Parasites and Sandflies. PLoS Negl Trop Dis. 2016;10: e0004349.

424 doi:10.1371/journal.pntd.0004349

425 22. Zelazny AM, Fedorko DP, Li L, Neva FA, Fischer SH. EVALUATION OF 7SL

426 RNA GENE SEQUENCES FOR THE IDENTIFICATION OF LEISHMANIA

427 SPP. Am J Trop Med Hyg. 2005;72: 415–420. doi:10.4269/ajtmh.2005.72.415

428 23. Guan W, Cao D-P, sun K, Xu J, Zhang J, Chen D-L, et al. Phylogenic analysis of

429 Chinese Leishmania isolates based on small subunit ribosomal RNA (SSU rRNA)

430 and 7 spliced leader RNA (7SL RNA). Acta Parasitol. 2012;57: 101–113.

431 doi:10.2478/s11686-012-0022-9

432 24. Sun K, Guan W, Zhang J-G, Wang Y-J, Tian Y, Liao L, et al. Prevalence of canine

433 leishmaniasis in Beichuan County, Sichuan, China and phylogenetic evidence for bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

434 an undescribed Leishmania sp. in China based on 7SL RNA. Parasit Vectors.

435 2012;5: 75. doi:10.1186/1756-3305-5-75

436 25. Dávila AMR, Momen H. Internal-transcribed-spacer (ITS) sequences used to

437 explore phylogenetic relationships within Leishmania. Ann Trop Med Parasitol.

438 2000;94: 651–654. doi:10.1080/00034983.2000.11813588

439 26. Yang B-B, Guo X-G, Hu X-S, Zhang J-G, Liao L, Chen D-L, et al. Species

440 discrimination and phylogenetic inference of 17 Chinese Leishmania isolates

441 based on internal transcribed spacer 1 (ITS1) sequences. Parasitol Res. 2010;107:

442 1049–1065. doi:10.1007/s00436-010-1969-9

443 27. Asato Y, Oshiro M, Myint CK, Yamamoto Y, Kato H, Marco JD, et al. Phylogenic

444 analysis of the genus Leishmania by cytochrome b gene sequencing. Exp Parasitol.

445 2009;121: 352–361. doi:10.1016/j.exppara.2008.12.013

446 28. Yang B-B, Chen D-L, Chen J-P, Liao L, Hu X-S, Xu J-N. Analysis of kinetoplast

447 cytochrome b gene of 16 Leishmania isolates from different foci of China:

448 different species of Leishmania in China and their phylogenetic inference. Parasit

449 Vectors. 2013;6: 32. doi:10.1186/1756-3305-6-32

450 29. Fraga J, Montalvo AM, De Doncker S, Dujardin J-C, Van der Auwera G.

451 Phylogeny of Leishmania species based on the heat-shock protein 70 gene. Infect

452 Genet Evol. 2010;10: 238–245. doi:10.1016/j.meegid.2009.11.007

453 30. Yuan D, Qin H, Zhang J, Liao L, Chen Q, Chen D, et al. Phylogenetic analysis of bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

454 HSP70 and cyt b gene sequences for Chinese Leishmania isolates and

455 ultrastructural characteristics of Chinese Leishmania sp. Parasitol Res. 2017;116:

456 693–702. doi:10.1007/s00436-016-5335-4

457 31. Waki K, Dutta S, Ray D, Kolli BK, Akman L, Kawazu S-I, et al. Transmembrane

458 Molecules for Phylogenetic Analyses of Pathogenic Protists: Leishmania-Specific

459 Informative Sites in Hydrophilic Loops of Trans- Endoplasmic Reticulum N-

460 Acetylglucosamine-1-Phosphate Transferase. Eukaryot Cell. 2007;6: 198–210.

461 doi:10.1128/EC.00282-06

462 32. Franchini G. Sur les flagelles intestinaux du type Herpetomonas du Chamaeleon

463 vulgaris et leur culture, et sur les flagelles du type Herpetomonas de Chalcides

464 (Gongylus) ocellatus et Tarentola mauritanica. Bull Soc Path Exot. 1921;14: 641.

465 33. Telford SR. Evolutionary implications of Leishmania amastigotes in circulating

466 blood cells of lizards. Parasitology. 1979;79: 317–324.

467 doi:10.1017/S0031182000053725

468 34. Shortt HE, Swaminath CS. Preliminary Note on three Species of Trypanosomidae.

469 Indian J Med Res. 1928;16. Available:

470 https://www.cabdirect.org/cabdirect/abstract/19281000711

471 35. Luyo-Acero GE, Uezato H, Oshiro M, Takei K, Kariya K, Katakura K, et al.

472 Sequence variation of the Cytochrome b gene of various human infecting

473 members of the genus Leishmania and their phylogeny. Parasitology. 2004;128: bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

474 483–491. doi:10.1017/S0031182004004792

475 36. Garcia L, Kindt A, Bermudez H, Llanos-Cuentas A, Doncker SD, Arevalo J, et al.

476 Culture-Independent Species Typing of Neotropical Leishmania for Clinical

477 Validation of a PCR-Based Assay Targeting Heat Shock Protein 70 Genes. J Clin

478 Microbiol. 2004;42: 2294–2297. doi:10.1128/JCM.42.5.2294-2297.2004

479 37. El Tai NO, Osman OF, El Fari M, Presber W, Schönian G. Genetic heterogeneity

480 of ribosomal internal transcribed spacer in clinical samples of Leishmania

481 donovani spotted on filter paper as revealed by single-strand conformation

482 polymorphisms and sequencing. Trans R Soc Trop Med Hyg. 2000;94: 575–579.

483 doi:10.1016/S0035-9203(00)90093-2

484 38. Hall TA. BioEdit: a user-friendly biological sequence alignment editor and

485 analysis program for Windows 95/98/NT. Nucleic acids symposium series.

486 [London]: Information Retrieval Ltd., c1979-c2000.; 1999. pp. 95–98.

487 39. Kumar S, Stecher G, Tamura K. MEGA7: Molecular Evolutionary Genetics

488 Analysis Version 7.0 for Bigger Datasets. Mol Biol Evol. 2016;33: 1870–1874.

489 doi:10.1093/molbev/msw054

490 40. Xia X. DAMBE7: New and Improved Tools for Data Analysis in Molecular

491 Biology and Evolution. Mol Biol Evol. 2018;35: 1550–1552.

492 doi:10.1093/molbev/msy073

493 41. Xia X. DAMBE6: New Tools for Microbial Genomics, Phylogenetics, and bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

494 Molecular Evolution. J Hered. 2017;108: 431–437. doi:10.1093/jhered/esx033

495 42. Lanfear R, Frandsen PB, Wright AM, Senfeld T, Calcott B. PartitionFinder 2:

496 New Methods for Selecting Partitioned Models of Evolution for Molecular and

497 Morphological Phylogenetic Analyses. Mol Biol Evol. 2017;34: 772–773.

498 doi:10.1093/molbev/msw260

499 43. Ronquist F, Teslenko M, van der Mark P, Ayres DL, Darling A, Höhna S, et al.

500 MrBayes 3.2: Efficient Bayesian Phylogenetic Inference and Model Choice

501 Across a Large Model Space. Syst Biol. 2012;61: 539–542.

502 doi:10.1093/sysbio/sys029

503 44. Bandelt HJ, Forster P, Röhl A. Median-joining networks for inferring intraspecific

504 phylogenies. Mol Biol Evol. 1999;16: 37–48.

505 doi:10.1093/oxfordjournals.molbev.a026036

506 45. Polzin T, Daneshmand SV. On Steiner trees and minimum spanning trees in

507 hypergraphs. Oper Res Lett. 2003;31: 12–20. doi:10.1016/S0167-6377(02)00185-

508 2

509 46. Feridoun Mahboudi MA Mohsen Abolhassani, Mohammad Yaran, Hamid

510 Mobtaker. Identification and Differentiation of Iranian Leishmania Species by

511 PCR Amplification of kDNA. Scand J Infect Dis. 2001;33: 596–598.

512 doi:10.1080/00365540110026746

513 47. Victoir K, Bañuls AL, Arevalo J, Llanos-Cuentas A, Hamers R, Noël S, et al. The bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

514 gp63 gene locus, a target for genetic characterization of Leishmania belonging to

515 subgenus Viannia. Parasitology. 1998;117: 1–13.

516 48. Wilson V, Southgate BA. Lizard leishmania. Biol Kinetoplastida. 1979;2: 241–

517 268.

518 49. Bates PA. Transmission of Leishmania metacyclic promastigotes by

519 phlebotomine sand flies. Int J Parasitol. 2007;37: 1097–1106.

520 doi:10.1016/j.ijpara.2007.04.003

521 50. Stuckenberg BR. New fossil species of Phlebotomus and Haematopota in Baltic

522 Amber (Diptera: Psychodidae, Tabanidae). Ann Natal Mus. 1975;22: 455–464.

523 51. Morayma Solórzano Kraemer M. Systematic, palaeoecology, and

524 palaeobiogeography of the insect fauna from Mexican amber. Palaeontogr Abt A.

525 2007; 1–133. doi:10.1127/pala/282/2007/1

526 52. Wenyon CM. A Trypanosome and Haemogregarine of a Tropical American Snake.

527 Parasitology. 1908;1: 314–317. doi:10.1017/S0031182000003619

528 53. Ayala SC, Atkinson C, Vakalis N. Two New Trypanosomes from North American

529 Snakes. J Parasitol. 1983;69: 391–396. doi:10.2307/3281242

530 54. Viola LB, Attias M, Takata CSA, Campaner M, Souza WD, Camargo EP, et al.

531 Phylogenetic Analyses Based on Small Subunit rRNA and Glycosomal

532 Glyceraldehyde-3-Phosphate Dehydrogenase Genes and Ultrastructural bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.

533 Characterization of Two Snake Trypanosomes: Trypanosoma serpentis n. sp. from

534 Pseudoboa nigra and Trypanosoma cascavelli from Crotalus durissus terrificus. J

535 Eukaryot Microbiol. 2009;56: 594–602. doi:10.1111/j.1550-7408.2009.00444.x

536 55. Cao D-P, Guo X-G, Chen D-L, Chen J-P. Species delimitation and phylogenetic

537 relationships of Chinese Leishmania isolates reexamined using kinetoplast

538 cytochrome oxidase II gene sequences. Parasitol Res. 2011;109: 163–173.

539 doi:10.1007/s00436-010-2239-6

540 56. Manson-Bahr PEC, Heisch RB. Transient Infection of Man with a Leishmania (L.

541 adleri) of Lizards. Ann Trop Med Parasitol. 1961;55: 381–2.

542 57. Adler S. The Behaviour of a Lizard Leishmania in Hamsters and Baby Mice. Rev

543 Inst Med Trop Sao Paulo. 1962;4: 61–4.

544 58. Coughlan S, Mulhair P, Sanders M, Schonian G, Cotton JA, Downing T. The

545 genome of Leishmania adleri from a mammalian host highlights chromosome

546 fission in Sauroleishmania. Sci Rep. 2017;7: 43747. doi:10.1038/srep43747

547 59. Breton M, Tremblay MJ, Ouellette M, Papadopoulou B. Live Nonpathogenic

548 Parasitic Vector as a Candidate Vaccine against Visceral Leishmaniasis. Infect

549 Immun. 2005;73: 6372–6382. doi:10.1128/IAI.73.10.6372-6382.2005

550 bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license. bioRxiv preprint doi: https://doi.org/10.1101/511162; this version posted January 3, 2019. The copyright holder for this preprint (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made available under aCC-BY 4.0 International license.