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Updates on the sporulation process in

Talukdar, P. K., Olguín-Araneda, V., Alnoman, M., Paredes-Sabja, D., & Sarker, M. R. (2015). Updates on the sporulation process in Clostridium species. Research in , 166(4), 225-235. doi:10.1016/j.resmic.2014.12.001

10.1016/j.resmic.2014.12.001 Elsevier

Accepted Manuscript http://cdss.library.oregonstate.edu/sa-termsofuse *Manuscript

1 Review article for publication in special issue: Genetics of toxigenic

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3 Updates on the sporulation process in Clostridium species

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5 Prabhat K. Talukdar1, 2, Valeria Olguín-Araneda3, Maryam Alnoman1, 2, Daniel Paredes-Sabja1, 3,

6 Mahfuzur R. Sarker1, 2.

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8 1Department of Biomedical Sciences, College of Veterinary Medicine and 2Department of

9 Microbiology, College of Science, Oregon State University, Corvallis, OR. U.S.A; 3Laboratorio

10 de Mecanismos de Patogénesis Bacteriana, Departamento de Ciencias Biológicas, Facultad de

11 Ciencias Biológicas, Universidad Andrés Bello, Santiago, Chile.

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14 Running Title: Clostridium formation.

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17 Key Words: Clostridium, , sporulation, Spo0A, sigma factors

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20 Corresponding author: Dr. Mahfuzur Sarker, Department of Biomedical Sciences, College of

21 Veterinary Medicine, Oregon State University, 216 Dryden Hall, Corvallis, OR 97331. Tel: 541-

22 737-6918; Fax: 541-737-2730; e-mail: [email protected]

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1 24

25 Abstract

26 Sporulation is an important strategy for certain bacterial species within the phylum to

27 survive longer periods of time in adverse conditions. All spore-forming have two phases

28 in their life; the vegetative form, where they can maintain all metabolic activities and replicate to

29 increase numbers, and the spore form, where no metabolic activities exist. Although many

30 essential components of sporulation are conserved among the spore-forming bacteria, there are

31 differences in the regulation and the pathways among different genera, even at the species level.

32 While we have gained much information from the most studied spore-forming bacterial ,

33 , we still lack an in-depth understanding of spore-formation in the genus Clostridium.

34 Clostridium and Bacillus share the master regulator of sporulation, Spo0A, and its downstream

35 pathways, but there are differences in the activation of the Spo0A pathway. While Bacillus

36 species use a multicomponent phosphorylation pathway for phosphorylation of Spo0A, termed

37 phosphorelay, such a phosphorelay system is absent in Clostridium. On the other hand, a number

38 of genes regulated by the different sporulation-specific transcription factors are conserved

39 between different Clostridium and Bacillus species. In this review, we discuss the recent findings

40 on Clostridium sporulation and compare the sporulation mechanism in Clostridium and Bacillus.

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2 42 1. Introduction

43 Sporulation is an intriguing bacterial property of a certain low G+C group of Gram-

44 positive bacteria, which have existed from ancient time (2.5 billion years ago) [1]. It is a

45 complex developmental process, which leads to the generation of metabolically dormant spores

46 from vegetative cells [2]. While the exact reason is not known for the decision of bacterial cell to

47 form spores, it has been hypothesized that nutrient depletion or the presence of toxic compounds

48 triggers the sporulation process [3]. Spore formation is a helpful strategy for the spore-forming

49 bacteria leading to survival in unfavorable conditions in the environment or inside the hosts for

50 prolonged periods of time, and to other hosts or environments. Spores can

51 withstand physical and chemical stresses, such as high temperatures, pressures, solvents,

52 oxidizing agents, lytic , irradiation, acceleration, and antimicrobials [4, 5], which could

53 rapidly destroy the vegetative form of the bacterium. In several instances, spores serve as an

54 infective particle in human and animal diseases [6]. Each of these fascinating characteristics led

55 microbiologists to engage their profound interest on dissecting spore structure and the

56 mechanism of spore formation.

57 Extensive studies have been conducted on the sporulation process of Bacillus species,

58 especially for many years, and thus it is regarded as the for

59 sporulation. Due to the availability of techniques for genetic manipulation, molecular

60 microbiologists showed the most interest in this species to illustrate the sporulation mechanism.

61 However, after gaining significant knowledge from B. subtilis, researchers have switched their

62 focus to other spore-forming bacteria, especially Clostridium species. Although, it has been

63 suggested that sporulation in Bacillus and Clostridium employ similar mechanisms based on

3 64 their morphological similarities, studies have shown that there are some differences in the early

65 stages of sporulation process in these two species [2, 7-9].

66 Clostridium species are Gram-positive, anaerobic, spore-forming including

67 strains of importance to human and animal health (C. tetani, C. perfringens, C. botulinum and C.

68 difficile), cellulose degradation (C. phytofermentans and C. thermocellum), solvent production

69 (C. acrtobutylicum and C. beijerinckii) and strains involved in bioremediation (C. cadavaris).

70 This heterogeneous group of Clostridium is divided into 19 clusters [10]. These strains are

71 widely distributed throughout the world in all sorts of environments, but most likely found in

72 and animal intestines in the form of vegetative cells or dormant spores.

73 It has been hypothesized that and Clostridia were in the same class until about 2.5

74 billion years ago when the rise of atmospheric occurred, also known as the ‘great

75 oxidation’ event [1]. Bacilli diverged from the Clostridia as a separate class during that period.

76 After that separation, Bacilli remain as an aerobic spore-former whereas Clostridia persisted as

77 an anaerobic-spore former. Different environmental requirements for the growth of these two

78 classes of bacteria may explain why there are differences in the molecular mechanism of

79 sporulation, especially at the initial stages in sporulation. In contrast, signature sporulation genes

80 are still conserved between these two classes long time after their separation indicating that both

81 had originated from the same origin [11, 12].

82 Despite having importance in the field of human and animal health and physiology,

83 cellulose degradation, solvent production and bioremediation, the molecular events in

84 Clostridium sporulation are not well understood, primarily as a result of limited genetic

85 manipulation. Recent developments in molecular techniques such as, high-throughput genome

86 sequencing, genome-wide transcriptional profiling, and directed or random mutagenesis

4 87 techniques such as group II introns for insertional mutagenesis and transposons for random

88 mutagenesis, have enabled researcher to find out the hints of molecular mechanism leads to the

89 sporulation in Clostridium.

90 In this review, we discuss the recent advancements in the sporulation study on four major

91 Clostridium species including C. acetobutylicum, C. botulinum, C. difficile, and C. perfringens.

92 We discuss how the components of the sporulation process differ between Clostridium than

93 Bacillus species. Also, we compare the sporulation process among Clostridium species.

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95 2. Stages of sporulation and spore structure

96 The morphological stages for spore formation are similar in all spore-forming bacteria. In

97 every sporulation event, there are two forms present in the cell; the mother cell and the forespore.

98 The total sporulation process can be divided into seven stages (stage I-VII) [13]. The first stage,

99 called stage 0 is actually the growth of vegetative cells before the beginning of sporulation. In

100 stage I and II, the cell DNA releases as an axial filament and the asymmetric cell division results

101 in forming of two compartments, one with smaller prespore compartment and the other is larger

102 mother cell compartment. Initially, one-third of the DNA material is deposited in the prespore,

103 although the rest of the DNA is rapidly pumped into the prespore compartment via the action of

104 the DNA translocase, SpoIIIE. During stage III, the prespore is engulfed by the mother cell and

105 called forespore, which has inner and outer membranes surrounding and floating as a protoplast.

106 In step IV, (PG) layer synthesizes the primordial germ cell wall and the cortex in

107 the space between inner and outer membranes surrounding the forespore. The outcome of stage

108 V is the formation of the complex structure of known as the spore coat, outside on the

109 surface of the forespore. Despite these changes in the morphological structure of spores, there is

5 110 one more stage to make the newly formed spore more durable. In stage VI, spore’s resistance to

111 UV radiation and heat is established. After going through all these changes, mature spores are

112 liberated from the mother cell into the environment during the stage VII of sporulation.

113 The basic structure of spores and morphological stages are conserved among all spore-

114 forming bacteria. The spore structures contribute to the dormant to sustain in a

115 variety of environmental stresses like high temperature, pressure, extreme pH, and radiation until

116 the spore finds itself in more favorable condition for vegetative growth. Usually, the bacterial

117 genome is deposited inside the central compartment of the spore surrounded by the lipid bilayer

118 covered with a layer of PG, which is known as the germ cell wall. This germ cell wall also serves

119 as the cell wall of vegetative forms after the completion of spores germination. The Germ cell

120 wall is wrapped in a thick layer of another layer of modified PG, termed the cortex. It is essential

121 for the acquisition and the maintenance of the heat resistance [14, 15]. Finally, this cortex layer

122 is encapsed by a multiprotein coat protecting it from the action of the PG-lytic enzymes. In

123 several species such as B. anthracis, B. cereus and C. difficile, this multiprotein coat is further

124 enclosed by another structure known as the exposporium [16-18]. If present, the or

125 otherwise the coat serves as the interacting structure of the spore to the environment. The spore’s

126 inner membrane contains the essential components for spore germination, including various

127 germinant receptors, which interacts with small molecules that trigger germination and a return

128 to the vegetative form [14, 19-21].

129

130 3. Initiation of sporulation

131 The full initiation pathway has been identified in the Bacillus species; however, the clear

132 picture in Clostridium has yet to be elucidated. In Bacillus, a multi-component signal

6 133 transduction system termed ‘phosphorelay’ is present [7]. Proper functioning of this system

134 leads to the activation of a master regulator, Spo0A [22] (Fig. 1). At present, no such

135 phosphorylation system has been found in Clostridium species. Another important component

136 for the initiation of sporulation are kinases, termed orphans, because they lack the cognate

137 response regulator [23]. These orphan kinases are involved in receiving different stimuli, both

138 from the extracellular or intracellular and initiate the process of sporulation. In Bacillus, at least

139 five orphan kinases (KinA - KinE) are present, but the KinA and KinB are the most efficient

140 ones. Each of the kinases is able to respond to different stimuli [24].

141 In Bacillus, all orphan kinases autophosphorylate upon receiving the respective signal(s)

142 and transfer the phosphoryl group from their phosphotransferase to an aspartate residue

143 in Spo0F, a single domain response regulator [24]. The phosphoryl group is then passed to a

144 histidine residue in a phosphotransferase domain within Spo0B. Finally, Spo0B phosphorylates

145 the key sporulation regulator, Spo0A, that eventually starts the second phase of the sporulation

146 process (Fig. 1). There is no evidence for the presence of Spo0F in any Clostridium species [7].

147 However, a homolog of Spo0B was found in C. tetani, but its function has yet to be known [25].

148 There are more components involved in Bacillus sporulation and those can regulate the

149 phosphorelay system either positively or negatively. An alternative sigma factor, SigH, is a

150 transcription factor and activates Spo0A, Spo0F, KinA and KinE [26]. While Spo0A is activated

151 directly by SigH, Spo0A increases the transcription of spo0H gene encoding SigH by repressing

152 AbrB, a global repressor. In contrast to Bacillus, Clostridium spo0H is constitutively expressed

153 at a low level, without any sign of increase at the onset of spore formation [9].

154 In total, thirty-nine histdine kinases have been found in B. subtilis, of which nine are

155 orphans [7, 24]. Among five sporulation specific histidine kinases, only three have been found to

7 156 contain transmembrane domains [27], which means those three might response to the

157 environmental signal. In an earlier review, Paredes et al [2] described the presence of putative

158 orphan histidine kinases in three Clostridium species; C. acetobutylicum, C. perfringens, and C.

159 tetani based on the completed genome sequences of Clostridium species at that time. These

160 authors used a BLAST- based approach to identify 35 kinases in C. acetobutylicum and six of

161 them are orphans (Table 1). Another study identified two different pathways for sporulation

162 initiation in C. acetobutylicum [28], and by insertional inactivation of kinase genes (single and

163 double mutants), they showed that CAC0903, CAC3319, and CAC0323 directly activate Spo0A.

164 Our group then extended the search for putative orphan histidine kinases in other

165 Clostridium species. We followed the strategy of Paredes et al [2], to find out the orphan

166 histidine kinases from the Clostridium genomes in NCBI database. In C. perfringens Type-A

167 food poisoning strain SM101, six putative orphan histidine kinases (ORFs CPR1953, CPR1493,

168 CPR1316, CPR0195, CPR1055, and CPR1728) were identified based on the BLASTP analyses

169 with kinases from B. subtilis. Knock-out mutations in two of these (CPR1055, and CPR1728)

170 showed sporulation and germination defects suggesting their putative role in activating the

171 Spo0A by phosphorylation (P. Udompijitkul and M. R. Sarker, unpublished).

172 The phosphorelay system might explain why Bacillus is more environmentally versatile

173 than Clostridium. In the process of evolution, Bacillus adopted themselves to the environmental

174 changes by adding different functional genes for reacting to multiple signals. The absence of a

175 phosphorelay system leaves remaining questions about how Clostridia initiate sporulation. One

176 hypothesis is that, Clostridium Spo0A is activated by direct phosphorylation from the orphan

177 histidine kinases. Another possibility is that there might be a different unknown phosphorelay

178 system present in Clostridium.

8 179

180 4. The master regulator, Spo0A

181 The most widely studied and functionally characterized component of the sporulation

182 machinery in spore-forming bacteria is the key regulator, Spo0A. It is a transcription factor that

183 controls the transition of the bacterium into the spore form [2, 26]. The structure and function of

184 Spo0A has been mostly studied in Bacillus species. In that system, Spo0A is activated by

185 Spo0B [29-31]. When the number of phosphorylated Spo0A reaches a threshold [32], the protein

186 binds directly to specific DNA sequences (TGNCGAA) upstream of several early sporulation

187 genes and thus activates the sporulation process [33-35]. This particular DNA sequence is known

188 as ‘0A box’ or the binding site for Spo0A. The structure of Spo0A is critical for its subsequent

189 changes from de-phosphorylation to phosphorylation state and vice-versa. Spo0A has two

190 functional domains, the N-terminal phosphorylation and dimerization domain (receiver), and the

191 C-terminal DNA binding (effector) domain. These two domains are separated by a hinge region

192 [36]. Phosphorylation leads to structural rearrangement that facilitates Spo0A dimerization [37,

193 38], resulting in the disruption of transcription-inhibitory contacts between the receiver and

194 effector domains. This disruption leads to the binding of DNA binding domains to the Spo0A

195 protein. The crystal structure of the DNA binding domain confirms specific and non-specific

196 contacts between Spo0A protein and the consensus sequence [38, 39]. Upon activation, Bacillus

197 Spo0A directly activates 121 genes, including genes required for polar septum formation [34].

198 Spo0A is conserved between both Bacillus and Clostridium [40, 41]. The inactivation of

199 spo0A in several Clostridium species resulted in the blocking of sporulation and synthesis of

200 sporulation-specific sigma factors [9, 42-44]. Transcriptomic and proteomic analyses identified

201 C. dificile Spo0A as a global regulator that regulates metabolic and virulence factors outside the

9 202 sporulation process [45]. Similarly as in B. subtilis [46], Spo0A has a role in formation in

203 C. difficile [47, 48] and C. perfringens [49].

204 One additional function of Spo0A is to regulate different Clostridial [42, 50].

205 Recent works have suggested that it may be responsible for the regulation of A and toxin B

206 production by C. difficile [35, 43, 51, 52]. Study demonstrated that Spo0A positively regulates

207 toxin A and toxin B expression. The spo0A mutant in an erythromycin mutant strain of C.

208 difficile, C. difficile strain 630 delta erm resulted in the productions of toxins [43]. However,

209 another study showed a contradictory result; inactivation of spo0A in 630 delta erm strain had no

210 affect on the toxin production [35]. Deakin et al [51] reported that a C. difficile R20291 spo0A

211 mutant caused more severe disease in a murine model than the wild type strain. Recently, one

212 study has shown the various affects of Spo0A on the toxin production in different C. difficile

213 strains [52]. Spo0A has been implicated in virulence in mice models [43, 51]. In C.

214 acetobutylicum, Spo0A as well as sporulation affect the solvent production [53].

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216 5. Sigma (σ) factors

217 After the initial trigger of spore formation, the cell transitions through different

218 morphological stages to form mature spores, facilitated by the contribution of different σ factors.

219 These are the dissociable RNA polymerase subunits that alter the promoter specificity of the

220 RNA polymerase complex under different environmental and growth phase-dependent

221 conditions [26]. Four sporulation-specific σ factors were first identified in B. subtilis [13, 54-56].

222 These σ factors are compartment specific; σ factor F (σF) and G (σG) are forespore specific and

223 regulated by anti- σ factors and anti-anti- σ factors. On the other hand, σ factor E (σE) and K (σK)

224 are mother cell specific, synthesized as precursor protein, which needs to be cleaved for the

10 225 activation. Generally, σF is the first σ factor appeared in the sporulation process by controlling

226 the early stages of the forespore followed by σE in the mother cells. Later, σG and σK have their

227 actions in the formation of mature spores and vegetative cells, respectively (Fig. 1). The

228 sequence similarities to all four σ factors have been identified in C. acetobutylicum [57] and

229 confirmed by PCR based approach [41]. Similar results have also been observed in other

230 Clostridium species; homologs of all four σ factors have been found in C. perfringens [58, 59].

231 This has been confirmed by transcriptional and protein analyses [60, 61].

232

233 5.1 σF

234 In B. subtilis, the RNA polymerase σF is synthesized prior to the formation of polar

235 septum and held inactive until septation is completed [26]. A similar pattern has been found in

236 few Clostridium species (Fig. 1). In B. subtilis, σF encoding gene, spoIIAC, is transcribed by σH

237 associated RNA polymerase during the initiation of sporulation [62, 63]. σF remains inactive in

238 the pre-divisional cell by binding with anti- σ factor SpoIIB, until it is relieved by the anti-anti- σ

239 factor SpoIIA. The functionality of SpoIIA becomes inactive by the phosphorylation with

240 SpoIIAB, which is a kinase and anti- σ factor. In contrast, SpoIIA becomes active by the

241 dephosphorylation with a phosphatase, SpoIIE. The non-phosphorylated SpoIIA interacts with

242 the SpoIIB- σF complex and displace the σF. Released σF becomes active and directs gene

243 expression via the control of different genes. The role of σF in sporulation of different

244 Clostridium species has been demonstrated by gene-knock-out studies. Clostridium mutants

245 lacking σF completely blocked for sporulation and this defect could be restored to nearly wild-

246 type levels by complementation with wild-type sigF gene, indicating that σF is essential for spore

247 formation in these organisms [61, 64, 65].

11 248

249 5.2 σE

250 In both Bacillus and Clostridium species, σE is synthesized as an inactive precursor

251 protein (pro- σE) (Fig. 1), which is activated by the proteolysis event by the protease activity of

252 SpoIIGA [66, 67]. Pro- σE is transcribed from spoIIG operon, in which sigE (earlier named as

253 spoIIGB), is the second gene in the operon, initiated transcription before asymmetric septation

254 and continues after septum formation, only in the mother cell [68]. This indicates that the σE is

255 produced even after the septum formation. The expression of spoIIGA, the first gene of the

256 spoIIG operon, requires the σF -controlled SpoIIR protein. This indicates that the mother-cell-

257 specific σE is controlled by the forespore-specific σF -directed gene transcription and the presence

258 of an intercellular gene transcription pathway [26, 64]. spoIIGA expression is also controlled by

259 the Spo0A after asymmetric division [26, 69]. Like σF, σE mutant strains in C. perfringens and C.

260 difficile showed sporulation defects in sporulation-inducing conditions [60, 64]. C.

261 acetobutylicum σE mutant strain also blocks the sporulation before the asymmetric division [70].

262

263 5.3 σG

264 In B. subtilis, σG is synthesized in the pre-engulfment prespore, but is not activated until

265 the end of stage III (complete engulfment of prespore by mother cell). Transcription of sigG

266 (earlier named spoIIG) is dependent on σF [26, 71]. The products of spoIIIA and spoIIIJ are

267 needed for the release of σG from inhibition in the forespore compartment. spoIIIA is selectively

268 expressed in the mother cell under σE control, whereas spoIIIJ is expressed in the forespore [26,

269 71]. Mutations in sigG blocks the sporulation in C. perfringens and C. difficile [61, 64]. In C.

270 acetobutylicum, sigG mutant halted the sporulation during the maturation stage [70].

12 271

272 5.4 σK

273 σK is known as the late stage (stage IV) mother cell specific σ factor. It regulates spore

274 coat formation during the late-stage of sporulation [72, 73]. An intervening element of 42-kb

275 size, named skin (sigma K intervening) separates sigK into two parts in some Bacillus species

276 [74]. Both halves are required, as mutations in either halves results in the failure of spore

277 formation at late stage [75]. The absence of the skin element does not affect the growth or

278 sporulation suggesting that this element does not contain any genes essential for survival [74].

279 Inside the mother cell, the skin undergoes site-specific recombination to form sigK. Because it

280 has no visible role in survival, it has been assumed that skin is a rudimentary element left from

281 the ancestors [76]. Interestingly, this skin element has been found in C. difficile, and unlike B.

282 subtilis, it is important for sporulation [77]. Although a 47-kb skin element, named skinCt has

283 been identified in C. tetani [78], other known Clostridium genomes do not contain this element

284 and have an uninterrupted sigK.

285 The homolog of Bacillus σK has been found in the genomes of C. botulinum [79] and C.

286 perfringens [58] and shown to be essential for early stage of sporulation [60, 80] (Fig. 1). Krik et

287 al [80] constructed two sigK mutants in C. botulinum and demonstrated that σK also acts in the

288 early stage of sporulation and plays as a transcriptional activator of Spo0A. σF transcript is also

289 lower in σK muatnts. Also, σK was shown to have role in different stress tolerance such as cold

290 and osmotic stress in C. botulinum ATCC3502 strain [81].

291

292 5.5 Regulation of sigma factors in Clostridium

13 293 Among the Clostridium species, the most studied organism for σ factor expression during

294 sporulation is C. acetobutylicum [9, 41, 82]. In this species, the pattern of σ factor expression and

295 solventogenesis for SpoIIA, σE, σG, and σK matched that in B. subtilis [9], although it was spread

296 over a much longer time (35 h) than that seen in B. subtilis (8 h). Some σ factors have two

297 separate developmental roles during sporulation. For example, in C. acetobutylicum, σK acts both

298 early, even prior to Spo0A expression, and late stages of sporulation, past of σG activation [83].

299 The expression and regulation of all four σ factors in C. perfringens has been evaluated in

300 two separate studies. By introducing mutations in sigE and sigK genes of C. perfringens SM101,

301 Harry et al [60] discovered some differences in expressions and functions of these σ factors

302 during the sporulation of C. perfringens SM101 versus B. subtilis (Fig. 1). 1) Unlike B. subtilis,

303 where σK is the last σ factor expressed during sporulation, normal production of sigF and sigE

304 transcripts in sporulating SM101 cells is dependent on sigK. 2) sigF transcript production was

305 delayed in a SM101 sigE-mutant, but sigF is the first transcript in B. subtilis. 3) sigG transcripts

306 were detected in SM101-sigE and -sigK mutants while sigG transcription in B. subtilis requires

307 both σE and σF. 4) Transcripts of all four σ factor genes were detected much earlier in C.

308 perfringens SM101 than that reported for B. subtilis. Finally, unlike B. subtilis, where expression

309 of spoIIID (a key mother cell specific transcription factor) requires σE-associated RNA

310 polymerase, the transcription and translation of spoIIID in C. perfringens does not require either

311 σK or σE.

312 But these findings were questioned by another study conducted by Li and McClane [61].

313 To investigate the role of σF and σG in sporulation and CPE production in C. perfringens SM101

314 strain, they constructed isogenic sigF and sigG null mutants and corresponding complemented

315 strains. By the Western blot analyses, they showed that there were little or no production of σG

14 316 σE, and σK in sigF mutant, which were completely restored in complementing strain [61]. These

317 findings suggest that regulation of σ-factors in C. perfringens is similar to B. subtilis.

318 In 2013, three extensive studies [64, 84, 85] were conducted on expression and regulation

319 of σ factors in C. difficile. Saujet et al [85] identified 225 genes under the regulation of σ factors

320 by genome-wide transcriptional analyses and promoter mapping. In a separate study, Fimlaid et

321 al [84] has found almost the same number of genes (224) regulated by the sporulation specific σ

322 factors. Pereira et al showed the intra-regulation of σ factors by disrupting each of the σ genes

323 [64]. Collectively, all these studies revealed that mother-cell-specific σ factors, σE and σK are not

324 regulated by forespore-specific σF and σG, respectively [64, 84, 85]. Also, σG is not dependent on

325 σE [64, 84].

326 When comparing the normalized reference genes, Kirk et al [86] demonstrated the

327 expressions of four σ factors in C. botulinum ATCC3502 strain. Expressions of sigF, sigE, and

328 sigG were simultaneously high at the end of the exponential growth phase. Although very low

329 sigK expression was detected during the exponential phase, this expression was peaked after 18

330 h, means very late during the stationary phase.

331

332 5.6 Role of σ factors in toxin production

333 In addition to regulation of sporulation process, σ factors control the expression and

334 production of some clostridial toxins. First example of sporulation and/or σ factors regulated

335 toxin is CPE, an essential for C. perfringens pathogenesis [87]. Sporulation

336 controls the expression of CPE encoding gene (cpe) at the transcriptional level, as both northern

337 blot and reporter construct studies detected cpe transcription in sporulating, but not in vegetative

338 cultures [88, 89]. The transcription of cpe is dependent on three promoters: promoter 1 (P1) was

15 339 proposed to be σK-dependent, while promoter 2 (P2) and promoter 3 (P3) σE-dependent based on

340 the consensus recognition sequences [89]. The presence of these strong promoters probably

341 explains why C. perfringens type A isolates produce such high levels of CPE during sporulation.

342 Since putative σK -and σE-dependent promoters had been identified upstream of the cpe [89],

343 Harry et al [60] constructed sigK and sigE mutants of C. perfringens SM101 and evaluated the

344 importance of σK and σE in cpe expression. Both mutants failed to produce β-glucuronidase when

345 transformed with a recombinant plasmid carrying the cpe promoter fused to β-

346 glucuronidase gene gusA [60], indicating that cpe expression is dependent on σK and σE. In a

347 follow-up study, Li and McClane [61] demonstrated that cpe transcription and CPE production

348 are blocked in an SM101 sigF-mutant, however, normal levels of cpe transcription and CPE

349 production were observed in sporulating SM101 sigG-mutant cultures. Collectively, findings

350 from these two studies showed that only σE, σF, and σK are needed for CPE synthesis.

351 The second sporulation-regulated C. perfringens toxin is TpeL, which belongs to the

352 family of large clostridial toxins. No expression of tpeL-gusA fusion was observed in SM101

353 spo0A-or sigE-mutant, indicating that tpeL expression is dependent on the master regulator of

354 sporulation, Spo0A, and the sporulation-specific σ factor, σE [50].

355 A recent study has identified a repressor protein named VirX, which significantly

356 inhibited the sporulation and CPE production in C. perfringens SM101 strain [90]. The higher

357 levels of cpe transcription and CPE production were observed in SM101 virX-mutant compared

358 to wild-type. Also, the transcription level of sigE, sigF and sigK was strongly induced at 2.5 h of

359 culture of the virX mutant, suggesting that VirX negatively regulates the transcription of cpe and

360 production of CPE through the sporulation-specific σ factors [90].

361

16 362 6. Other factors in Clostridium sporulation

363 Sporulation is regulated by bacterial cell density and . The homologues of

364 AgrB and AgrD of the well-studied aureus Agr quorum sensing system have

365 been found in a few Clostridium species [91-93]. Mutations in both putative agrB and agrD in C.

366 sporogenes and C. botulinum result in the reduction of sporulation as well as toxin production

367 [93]. Agr-dependent quorum sensing is also involved in the regulation of sporulation and

368 granulose formation in C. acetobutylicum [92]. Sporulation and CPE production in C.

369 perfringens also seems to be positively regulated by Agr-like quorum-sensing (QS) system [91].

370

371 7. New sporulation genes in Clostridium

372 Sporulation studies are mainly focused on two important spore-forming genera; Bacillus

373 (aerobic) and Clostridium (anaerobic). Moreover, other organisms resembling both aerobic and

374 anaerobic within the firmicutes have a sporulating nature, although the differences in

375 morphology and life style are widely spread. Despite the universal presence of master regulator,

376 Spo0A, and the four cell-type specific σ factors in all spore-forming bacteria, there are number of

377 other genes conserved in all spore-forming bacteria. Phylogenetic analysis with all spore-forming

378 bacteria reveals the core sporulation genes.

379 Both forward and reverse genetics have been applied to find out the genes directly under

380 the control of sporulation factors. In this aspect, again Bacillus is the model strain to find out the

381 sporulation associated genes. Approaches like identification of sporulation specific proteins such

382 as small acid-soluble proteins (SASPs), and transcriptional analyses revealed new sporulation

383 genes [94, 95]. Using phylogenetic analysis, studies found 111 core genes that are highly

384 conserved among the spore-forming bacteria [12]. Spo0A directly regulates the expression of

17 385 121 genes in Bacillus [34] and significantly enhances the expression of over 500 genes [96].

386 Tragg et al [97] analyzed the presence of all gene products in B. subtilis subsp. subtilis 168 with

387 a total of 626 bacterial genomes including 46 genomes of -formers and found that

388 fifty-eight genes are highly enriched among spore-forming bacteria. Eight of the previously

389 unidentified putative sporulation genes in Bacillus species were inactivated and found to have

390 roles in sporulation in Bacillus [97]. The presence of these newly identified sporulation genes

391 were also found in C. perfringens SM101 strain and mutational analyses demonstrated that ylmC

392 and bkdR mutant showed sporulation defect under spore-forming conditions (P. K. Talukdar and

393 M. R. Sarker, unpublished data). In our study, we extended the search of putative sporulation

394 genes in more Clostridium species. We have selected seven proteins (BkdR, CwlD, DapG, YlxY,

395 YlyA, YlzA and Yqhq) from the pool of 127 proteins identified in B. subtilis [12, 97] because: 1)

396 these are conserved among spore formers (mostly Bacillus and Clostridium strains) and mostly

397 not found in other non-spore-forming bacteria, and 2) role of these proteins yet to be determined.

398 The distribution of these seven putative sporulation proteins in different Clostridium species are

399 shown in Table 2.

400

401 8. Spore components involved in spore resistance

402 Different components of spore structure can protect spores from different physical and

403 chemical stresses. For example, the spore coat and the relatively impermeable spore inner

404 membrane contribute in the spore resistance. Spore core’s low water content, and high levels of

405 dipicolinic acid and associated divalent cations are other important factors involved in spore

406 resistance [98, 99]. Spore’s DNA in the core is protected by sporulation-specific proteins named

407 as α/β-type small acid soluble proteins (SASPs). These proteins bind to the DNA and alter its

18 408 chemical and photochemical reactivity, which are important to protect DNA from heat, many

409 genotoxic chemicals, and UV radiation [5, 100-102]. These SASP proteins are highly conserved

410 in both Bacillus and Clostridium species and appear at the same time in sporulation in both genus

411 [103]. Three SASP genes (ssp1, ssp2, and ssp3) have been identified in different C. perfringens

412 food poisoning (FP) and non-food-borne strains [103, 104]. Gene knock-out studies

413 demonstrated that α/β-type SASPs play a major role in mediating resistance of C. perfringens

414 spores to UV radiation, moist heat and chemicals [103, 105], but not to dry heat [103]. C.

415 perfringens ssp2 was expressed in B. subtilis spores lacking one or both major α/β-type SASP

416 and restored the resistance of α-β- spores to UV and nitrous acid and of α- spores to dry heat,

417 indicate the interchangeability of α/β-type SASP in DNA protection in spores [106]. However,

418 similar levels of SASPs production by FP and NFB strains [104] could not explain the reason

419 why spores of NFB isolates exhibit lower heat resistance than spores of FP isolates [107]. To

420 this end, Li and McClane identified a new SASP protein, named Ssp4, in C. perfringens FP and

421 NFB strains [108] and showed that a single substitution at Ssp4 residue 36 is critical;

422 a glycine at 36 residue of Ssp4 in NFB isolate is responsible for mediating spores’ heat

423 sensitivity, while an asparagine at the same site in Ssp4 of FP isolate for spores’ heat resistance

424 [108].

425

426 9. Concluding remarks and future perspective

427 Sporulation is the unique survival strategy for bacterial cell besides other strategies like

428 quorum sensing or biofilm formation. To date, most of the knowledge on sporulation has been

429 gathered from the genus, Bacillus. In contrast, information about sporulation in Clostridium

430 species is still scarce and insufficient to allow a full understanding of the complete regulatory

19 431 circuit of sporulation. Although the early stage of Clostridium sporulation is vastly different from

432 that in Bacillus because of the absence of phosphorelay system, the major regulator for

433 sporulation, Spo0A and the downstream signaling systems are mostly conserved. Studies done

434 on four industrial and pathogenic Clostridium species revealed the intra generic differences in

435 the regulation of sporulation. For understanding the full mechanism of Clostridium sporulation,

436 we have to answer the following questions:

437 1) Is there any phosphorelay system present in Clostridium species?

438 2) If not, how many kinase genes are present and how do they regulate the activation of

439 Spo0A?

440 3) Are these kinases regulating each other or independently working on Spo0A?

441 4) What are the environmental signals triggering sporulation in Clostridium?

442 5) Do kinases environmental signal-specific?

443 Clostridium genetic manipulation is much harder than in Bacillus species. But with the invention

444 of newer molecular tools like TargeTron/ClosTron for genetic manipulation, a wealth of new

445 information on Clostridium sporulation has been gathered. This new information with the

446 comparison with other sporulation mechanism, one can reveal the intrinsic mechanism of

447 sporulation.

448

449 Acknowledgements

450 This work was supported by a grant from the Agricultural Research Foundation of

451 Oregon State University, and by a Department of Defense Multidisciplinary University Research

452 Initiative (MURI) award through the U.S. Army Research Laboratory and the U. S. Army

453 Research Office under contract number W911NF-09-1-0286 (all to M.R.S); and by grants from

20 454 Fondo Nacional de Ciencia y Tecnología de Chile (FONDECYT Grant 1110569), by a grant

455 from the Research Office of Universidad Andres Bello (DI-275-R/13), and by a grant from

456 Fondo de Fomento al Desarrollo Científico y Tecnológico (FONDEF) CA13I10077 to D.P-S.

457 MA was supported by the Ministry of Higher Education in Saudi Arabia. We thank Dr. Daniel

458 D. Rockey for his editorial help.

459

460 461 462 463 464 465 466 467

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757

758

759

34 760 Figure Legends

761 Figure 1. Proposed sporulation model in Bacillus and Clostridium species. The temporal

762 progression of sporulation is shown for B. subtilis, C. difficile, and C. perfringens. In the

763 phosphorelay system of B. subtilis, after receiving the respective signal(s), all orphan kinases

764 autophosphorylate and transfer the phosphoryl group from their phosphotransferase domain to an

765 aspartate residue in Spo0F, a single domain response regulator. Next, the phosphoryl group is

766 passed to a histidine residue in a phosphotransferase domain, Spo0B. Finally, Spo0B

767 phosphorylate the key sporulation regulator Spo0A. There is no evidence of the presence of

768 Spo0F in any Clostridium species. Phosphorylation of Spo0A leads to the activation of a σ factor

769 cascade that acts in both mother cell and forespore. Differential features of the sporulation

770 pathway in C. difficile include the post-translational activation of σG by σF and the absence of

771 proteolytic activation of σK. A notable difference in the C. perfringens sporulation regulatory

772 circuit is the early requirement of σK to generate sufficient active σE. Solid arrows in the putative

773 regulatory cascade indicate confirmed interactions, whereas dotted arrows indicate that the

774 regulatory relationship between the factors has not been tested.

35 775 Table 1: Putative orphan histidine kinase of different Clostridium species. 776 Species/strainsa No. of No. of ORFs of putative orphan histidine kinases Reference histidine orphan kinasesb histidine kinasesc

Clostridium acrtobutylicum ATCC 824 34 7 CAC0903, CAC0323, CAC2220, CAC0317, CAC3319, [2] CAC2730, CAC0437

Clostridium acrtobutylicum EA 2018 39 9 CEAG2234, CEAG0328, CEAG3322, CEAG2739, CEAG0448, This study CEAG0334, CEAG0915, CEAG2551, CEAG0430

Clostridium acrtobutylicum DSM 1731 36 9 SMBG2253, SMBG0325, SMBG3356, SMBG2765, SMBG0920, This study SMBG0446, SMBG0331, SMBG2573, SMBG0428 Clostridium botulinum A str. Hall 34 4 CLC0398, CLC1171, CLC2637, CLC0394 This study

Clostridium botulinum A str. ATCC 32 3 CBO2762, CBO1120, CBO0336 This study 3502

Clostridium botulinum A str. ATCC 34 4 CLB1159, CLB2704, CLB0379, CLB0383 This study 19397

Clostridium difficile 630 51 6 CD630_24920, CD630_14920, CD630_13520, CD630_19490, This study CD630_15790, CD630_09970

Clostridium difficile CD196 48 7 CD196_1365, CD196_1829, CD196_1501, CD196_1216, This study CD196_2338, CD196_2713, CD196_0520

Clostridium difficile BI1 48 7 CDBI1_07760, CDBI1_06975, CDBI1_06215, CDBI1_11090, This study CDBI1_09440, CDBI1_12120, CDBI1_14040

Clostridium perfringensstr. 13 27 8 CPE1757, CPE1512, CPE1316, CPE0207, CPE1986, CPE1987, [2] CPE0951, CPE0870

36 Clostridium perfringensATCC13124 30 10 CPF2640, CPF2010, CPF1764, CPF2241, CPF2242, CPF1195, This study CPF0198, CPF1243, CPF1523, CPF0863

Clostridium perfringensSM101 24 9 CPR1023, CPR1953, CPR1954, CPR1493, CPR1316, CPR0195, This study CPR1807, CPR1055, CPR1728 777 778 a Strains were selected for identifying putative orphan histidine kinases based on the available whole genome sequence (WGS) data and respective with the 779 importance of the strain in research purpose. To date, in total 3 WGS for C. acetobutylicum (C. acetobutylicum ATCC 824, C. acetobutylicum EA 2018, and C. 780 acetobutylicum DSM 1731), 13 for C. botulinum (C. botulinum A str. Hall, C. botulinum A str. ATCC 3502, C. botulinum A str. ATCC 19397, C. botulinum F 781 str. Langeland, C. botulinum B1 str. Okra, C. botulinum A3 str. Loch Maree, C. botulinum B str. Eklund 17B, C. botulinum E3 str. Alaska E43, C. botulinum Ba4 782 str. 657, C. botulinum A2 str. Kyoto, C. botulinum F str. 230613, C. botulinum BKT015925, C. botulinum H04402 065), 4 for C. difficile (C. difficile 630, C. 783 difficile CD 196, C. difficile 2007855, and C. difficile BI1), and 3 for C. perfringens (C. perfringens str. 13, C. perfringens ATCC 13124, and C. perfringens 784 SM101) are available. 785 b The total no. of kinases were determined by searching with key word ‘histidine kinase’ for each of the respective strain in NCBI database. 786 c Orphan histidine kinases were identified by identifying the kinases with no adjacent response regulatory protein. 787 788 789

37 790 Table 2: Putative sporulation proteins in different Clostridium species.

Species Orthologs of putative sporulation proteins in Clostridiuma

BkdR CwlD DapG YlxY YlyA YlzA YqhQ Clostridium acrtobutylicum #c + + + + +/- + +/- Clostridium asparagiforme +/- − +/- +/- + +/- − Clostridium bartlettii +/- + + + + +/- +/- # + + + + +/- + +/- Clostridium bolteae + + + +/- + − + Clostridium botulinum # + + + + + + + + + + + +/- + +/- Clostridium cadaveris − − − − − − − Clostridium carboxidivorans + + + + +/- + +/- Clostridium cellulolyticum # − +/- + + +/- + + Clostridium cellulovorans # +/- +/- + + +/- + + Clostridium chauvaei + + + + +/- +/- +/- Clostridium clostridioforme + +/- + + +/- + + Clostridium colicanis +/- + + + +/- + +/- Clostridium difficile # + + + + +/- +/- + Clostridium hathewayi + + + + +/- + + Clostridium hylemonae − + + + +/- + + Clostridium innocuum +/- +/- − +/- +/- +/- − Clostridium kluyveri # + + + + +/- + +/- Clostridium leptum − +/- + + +/- +/- + Clostridium methylpentosum − +/- + +/- +/- + +/- Clostridium nexile − + + + +/- − + # + + + + +/- + +

38 Clostridium papyrosolvens + +/- + + +/- +/- +/- Clostridium paraputrificum − − − − − − − Clostridium perfringens# + + + + − + +/- Clostridium ramosum +/- +/- +/- +/- +/- +/- − Clostridium scatologenes − − − − − − − Clostridium scindens − +/- − + +/- +/- + ATCC + + + + +/- + + 9714 + + + + +/- + +/- Clostridium sticklandii # − − − − − − − Clostridium tetani # + + + + +/- + +/- Clostridium thermocellum # +/- + + + +/- + + Clostridium tyrobutyricum + +/- + +/- +/- + +/- 791 792 a These 7 putative sporulation proteins were identified from the phylogenetic analysis in B. subtilis [12, 97]. From the pool of 127 Proteins, we have selected 793 these 7 proteins because 1) these are conserved among spore formers (mostly Bacillus and Clostridium strains) and mostly not found in other non-sporulating 794 bacteria, and 2) role of these proteins yet to be determined. 795 b Orthologs of sporulation proteins were identified by the BLASTP analyses with B. subtilis strain 168 genome and the presence (+) or absence (−) were listed for 796 different Clostridium species. (+/-) indicates the proteins those have very low identity or have different or unknown functions than it’s respective protein of B. 797 subtilis strain 168. The functions of the proteins in B. subtilis strain 168 are as follows: BkdR, transcriptional regulator; CwlD, N-acetylmuramoyl-L-alanine 798 amidase; DapG, aspartokinase I; YlxY, putative deacetylase; YlyA, hypothetical protein; YlzA, hypothetical protein; and YqhQ, hypothetical protein. 799 c # after species name indicates the WGS are annotated and published for at least one of the strains for these species in NCBI database. The rest of the species do 800 not have any full-annotated genome available for any of their strains. 801 802 803 804 805 806 807 808

39 809

810 811 812

40 Figure 1

Fig. 1.