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9HWHULQDU\ 6FLHQFH Mucosal mast cell-derived chondroitin sulphate levels in and worm expulsion from FcR γ-knockout mice following oral challenge with Strongyloides venezuelensis Denis

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9HWHULQDU\ 6FLHQFH

Mucosal mast cell-derived chondroitin sulphate levels in and worm

expulsion from FcR γ-knockout mice following oral challenge with

Strongyloides venezuelensis

Denis Nnabuike Onah 1,

*, Yukifumi Nawa 2

1Department of Veterinary Parasitology & Entomology, University of Nigeria, Nsukka, Nigeria

2

Department of Parasitology, Miyazaki Medical College, Kiyotake, Miyazaki 889-1692, Japan

Mucosal mast cell-derived chondroitin sulphates

(sulphated proteoglycans) were assayed in gut washings

and homogenate of FcR γ-knockout (KO) and wild-type

(WT) C57BL/6 mice challenged with Strongyloides

venezuelensis in order to assess their possible role in

secondary immunity against enteric nematodes Groups

of immune KO and WT mice were challenged by oral

gavage with 300 infective larvae (L 3 ) Establishment of

infection was assessed by daily faecal analysis to

determine the number of eggs per gram of faeces (EPG)

and by adult worm recovery on days 5 and 13 post

challenge Mucosal mast cell (MMC) counts were done on

days 5 and 13 post challenge while MMC-derived

chondroitin sulphates in gut washings (days 1 and 5) and

homogenate (day 8) were assayed by high performance

liquid chromatography (HPLC) Results showed that

patent infection occurred in challenged KO but not WT

mice despite significantly higher mastocytosis in jejunal

sections of KO than WT mice (p < 0.001) Similarly but

against prediction, significantly higher concentration of

MMC-derived chondroitin sulphates was observed in gut

homogenate of KO than WT mice (p < 0.05) In contrast,

significantly higher concentration of chondroitin

sulphates was observed in gut washings of WT than KO

mice (p < 0.05) These results suggest that MMC in KO

mice failed to release sufficient amount of sulphated

proteoglycans into the gut lumen as did the WT mice,

which may have been part of the hostile environment that

prevented the establishment in and eventual expulsion of

adult S venezuelensis from the gut of WT mice following

challenge.

Key words: Strongyloides venezuelensis, mast cells, mucosal

immunity, chondroitin sulphates, gut washings, mice

Introduction

The immune expulsion of adult Strongyloides species

from the small intestine of mice and rats is associated with intraepithelial mast cell hyperplasia [1,2,7,14] Mast cells are thought to mediate this expulsion by creating an environment hostile to the establishment and survival of gastrointestinal nematodes through inflammation-associated changes and release of inflammatory mediators [19] The release of these mediators is induced by mast cell degranulation, which in turn is induced by mast cell activation triggered by cross-linking of the high affinity immunoglobulin Fc receptor (FcR) for IgE (FcεRI) with an antigen-IgE immune complex [3,8,11,13] This type of acute inflammation, also known as type I hypersensitivity is therefore thought to depend on mast cells, its FcεRI and on IgE Perturbation in any one of these three components should result in the loss of type I hypersensitivity responses [18] This, in fact, is the case with FcεRI To be expressed on the surface of cells and for signal transduction into the interior of the cell, FcεRI requires the homodimeric γ subunit of FcRs (FcRγ) [5,10,17] Ablation of the γ subunit

by targeted gene deletion results in the loss of FcγRI expression on mast cells coupled with loss of mast cell functions including degranulation and mediator release in the mutant mouse [23] Using these mice we showed that while the wild type (WT) counterparts were able to expel a

primary Strongyloides venezuelensis infection, FcR γ-knockout (KO) mice failed to do so [16] However, the confounding aspect of our study was that both intestinal mastocytosis and serum mouse mast cell protease-I (mMCP-I), the levels of which have negative correlation with nematode burden in sheep [22] were similar Since FcRγ-KO mice fail to assemble FcγRI on their mast cells and therefore are unable to express mast cell degranulation and mediator release [23], we concluded that mMCP-1 may

be released spontaneously without requiring the mast cell

FcγRI cross-linking with the immune complex formed by the parasite antigen/IgE and that mMCP-1 is not involved in

*Corresponding author

Tel: +234-42-770106

E-mail: denisonah2003@yahoo.co.uk

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adult S venezuelensis expulsion [16] However, because

mastocytosis has a strong association and often corresponds

with the time of worm expulsion [14], we stated that mast

cells must be involved in the process in a manner yet to be

elucidated We then speculated that failure of FcRγ-KO mice

to expel S venezuelensis might be related to inability of their

mast cells to degranulate and release inflammatory

mediators other than mMCP-1 [16] Sulphated goblet cell

mucins [7] and experimental introduction of glycosaminoglycans

of the type produced by mucosal mast cells [12] have been

shown to prevent the establishment of S venezuelensis

infection and mediate its expulsion from infected mice

Chondroitin sulphates are the major proteoglycans

contained by mucosal mast cells in mouse [25] In this study

therefore, we assayed the amounts of chondroitin sulphates

in gut homogenate and washings of FcRγ-KO mice and their

WT counterparts following oral challenge with S.

venezuelensis This was in order to ascertain (i) if

differences exit in their concentrations in the gut of KO and

WT mice and (ii) whether differences in worm

establishment and expulsion following challenge might be

related to any differences in their concentrations within the

gut niche occupied by the parasite in the two mouse types

Materials and Methods

Animals

FcRγ-knockout C57BL/6 male mice of 8 weeks old were

generous gifts from Professor Toshiyuki Takai and Dr

Masao Ono (Tohoku University, Sendai, Japan)

Age-matched specific pathogen free wild type C57BL/6 mice of

the same sex were purchased from Japan SLC (Shizuoka,

Japan) All animals were kept in our laboratory animal unit

for 2 weeks to acclimatize before use at 10 weeks old

Before and during the period of the experiment they were

supplied with feed and water ad libitum

Parasite

The strain of S venezuelensis originally isolated from a

brown rat in Okinawa Prefecture, Japan [6] and established

as a laboratory strain [20] was used in this study Infective

third stage larvae (L3) used for infecting experimental

animals orally (oral gavage) were obtained from the lungs of

mice given primary infection and sacrificed on day 3 post

infection Briefly, 10 C57BL/6 male mice were each

infected with 5000 L3 of S venezuelensis obtained by the

filter-paper faecal culture method and sacrificed on day 3

post-infection by anaesthetic overdose using diethyl ether

The lungs were removed by dissection, shredded with

fine-tipped thumb forceps in fine-meshed coffee strainers placed

in a beaker of warm PBS and incubated at 37o

C for 3 h

Emerged lung L3 were recovered by centrifugation,

re-suspended and washed three time in fresh warm (37o

C) PBS, counted and adjusted to 1500 lung L/ml

Infection of experimental animals

All animal groups for challenge were primed with 2000 L3

subcutaneously 30 days before challenge For uniform treatment, every mouse in both the challenge and challenge control (primary infection) groups was treated orally with 20-mg/kg mebendazole (Sigma, St Louis, MO) on day 26 post priming to get rid of any residual adult worms from the priming dose On day 30 post priming, each animal in the challenge and control groups was infected with 300 lung-recovered L3 contained in 200µl PBS and introduced directly into the stomach using a stomach needle with blunt oval tip

Experimental protocol

Thirty knockout (KO) and thirty wild-type (WT) mice were used for the experiment Twenty each of the KO and

WT mice were primed as stated above For challenge, primed KO and WT mice with 10 each of their nạve controls were infected as described above The challenge groups were sacrificed 5 mice each on day 1, 5, 8 and 13 post challenge for sample collection while 5 each of their controls were sacrificed on day 5 and 13 post infection Daily faecal egg counts expressed as eggs per gram of faeces (EPG) and adult worm recovery to establish whether patent infection occurred were carried out until day 13, and on day

5 and 13 post infection respectively in both challenge and control animals In the challenge animals only, histology for MMC numbers was carried out on day 5 and 13, while various chondroitin sulphates and mouse mast cell

protease-1 (mMCP-protease-1) in gut washings were assayed on day protease-1 and 5 post challenge Chondroitin sulphates in gut homogenate were assayed on day 8 post challenge

EPG and adult worm recovery

As stated above, faecal samples for daily EPG was collected only from individual animal of the groups sacrificed on day 13 post challenge To ascertain worm establishment, adult worms were recovered from sacrificed challenge and control animals on day 5 and 13 post infection The entire small intestine of each sacrificed animal was isolated and processed for adult worm recovery The methods used for EPG and adult worm recovery, were

as previously described [9,20]

Histology and serology

Jejunal pieces were taken and histological sections prepared, stained and mucosal mast cells were enumerated

as previously described [16] on day 5 and 13 post challenge Also samples of gut washings as described below were collected on days 1 and 5 post challenge and analysed for mMCP-1 concentration by ELISA as described [16]

Proteoglycan assay in gut washings and homogenate

The entire small intestine isolated from each of the

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sacrificed animal on the days specified above was washed

out twice with a total volume of 10 ml sterile distilled water

Each sample was then centrifuged at 350 g for 10 min to

remove debris The supernatant was frozen until used For

gut homogenate, approximately 15 cm of the upper small

intestine was cut off, washed out as above and minced using

a homogeniser (Polytron homogeniser, Kinematika AG,

Littau, Switzerland) The minced samples were suspended

in 5 ml sterile PBS, centrifuged as above and the supernatant

was frozen until used For the assay of the various

chondroitin sulphates, each sample was removed from the

freezer, thawed under room temperature and was then

processed and analysed by high-performance liquid

chromatography according to the methods of Yoshida et al.

[24] and Shinmei et al [21].

Statistical analysis

Data were compiled and subjected to descriptive statistics

while differences between group-means were obtained by

the Students t-test using Microsoft Excel Statistical

Toolpack Differences at p = 0.05 were considered significant.

Results

Eggs per gram of faeces (EPG)

The mean daily EPG of animals sacrificed on day 13 post

challenge are presented in Fig 1 All control WT and KO

animals as well as all challenged KO mice persistently shed

eggs in their faeces until the day of sacrifice In contrast,

there were no eggs in the faeces of all challenged WT mice

One significant observation however, is that the oral route of

infection with lung-recovered L3 does not seem to be an

efficient means of establishing patent infection judging from

the level of EPG of the animals (less than 1000 at peak EPG)

when compared with the EPG of mice infected by the

subcutaneous route in which peak EPG usually runs in tens

of thousands [15,16]

Adult worm recovery

Consistent with the EPG result, control WT and KO as

well as challenged KO mice developed persistent patent S venezuelensis infection The mean number of adult worms

recovered from animals sacrificed on days 5 and 13 are shown in Fig 2 As expected, no adult worms were recovered from any of the challenged WT mice on both days, which agreed with the zero EPG recorded for this group Again, the very few number of adult worms recovered from these mice indicate that oral implantation of third stage larvae is not very efficient in establishing patent infection

Intestinal mast cell numbers and serum mMCP-1 concentration

Mast cells were enumerated in jejunal sections prepared

on days 5 and 13 post oral challenge Results (Table 1a) show that significantly higher numbers of mast cells were

counted in KO than WT mice on day 5 (p < 0.01) and day

13 (p < 0.001) On the other hand, there was no significant difference (p > 0.05) in the amount of mMCP-1 concentration

in gut washings of challenged KO and WT mice on days 1 and 5 post challenge (Table 1b)

Sulphated proteoglycan concentration in gut washings and homogenate

Results of the assay for various sulphated proteoglycans (Chondroitin sulphates A, C, D, E, and total chondroitin sulphate) in gut washings of challenged KO and WT mice sacrificed on days 1 and 5 are presented in Figs 3a and 3b respectively The results show that significantly higher concentrations of the chondroitin sulphates occurred in the

gut washings of WT than KO mice on these days (p < 0.05).

In contrast, significantly higher concentrations of these

Table 1.

(a) Mucosal mast cell number/10 villus crypt unit (Mean ± SD) in FcRγ-KO and WT mice challenged orally with 300

lung-recovered S venezuelensis L3

(b) Mean ± SD of mMCP-1 level (ng/ml) in gut washings of FcRγ-KO and WT mice challenged orally with 300 L3 of S.

venezuelensis

mMCP-1 Concentration (ng/ml)

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sulphated sugars were obtained in gut homogenate

preparations of KO than WT mice on day 8 post-challenge

(p < 0.05, Fig 3c).

Discussion

Primary infection of mice with S ratti and S.

venezuelensis results in the development of a strong

immunity, which completely aborts a patent infection

following secondary challenge of the animals with the parasites [4,20] This strong immunity depends on CD4+

T cells which make Th2 cytokines and induce mast cell-dependent gut inflammatory responses and changes in gut physiology, all of which act in concert to create an environment hostile to worm establishment in and their eventual expulsion from the intestinal niche [15] Our prediction was that an important part of this inflammatory response and change in gut physiology resulting in worm expulsion is the presence of sulphated proteoglycans released by degranulating mast cells Therefore, if FcRγ-KO mice are unable to undergo mast cell degranulation and mediator release [23], the enabling hostile environment for the ablation of secondary infections will be lacking in primed KO animals Consequently, if sulphated proteoglycans play a role in worm expulsion, then the introduction of migrating L3 into a hostile intestinal environment in primed

KO and WT mice would most likely result in a patent infection in the former but not in the latter Results of this study largely support our predictions First, significantly higher concentrations of chondroitin sulphates were present

in gut washings of WT, in which the challenge infection was sterile than in KO, in which persistent patent infection was established Secondly, there was no significant difference in the concentrations of mMCP-1 in gut washings taken from both mouse types irrespective of the fact that significantly higher numbers of intraepithelial mast cells were counted in

KO than in WT animals

Taken together, it can be inferred that (a) mMCP-1 may be released by mast cells in a manner not dependent on degranulation i.e., not requiring the antigen-IgE-FcεR cross-linking and triggering, (b) mMCP-1 may not be important in the mucosal immune mechanisms resulting in the expulsion

of adult S venezuelensis from the gut and (c) mucosal mast

cell-derived glycosaminoglycans (chondroitin sulphates) are apparently involved in and therefore play a role in the

prevention of the establishment of adult S venezuelensis in

and their eventual immune expulsion from the gut These

agree with the suggestions of Onah et al [16] regarding

mMCP-1 and its possible role in worm expulsion as well as with their speculation that FcRγ-KO mice are probably

unable to expel primary S venezuelensis infection as a result

of failure of their mast cells to degranulate and release mediators other than mMCP-1 In fact, that significantly higher concentrations of chondroitin sulphates were obtained from the homogenized gut tissue of KO (which contained more mast cells) than WT is an added support that the intraepithelial mast cells in KO contain them but are not releasing them in enough quantities into the gut lumen to effect worm expulsion In addition, we have similar evidence that when immune KO and WT mice are

challenged by subcutaneous introduction of 3000 S venezuelensis L3, fewer larvae are recovered from the lungs

of KO than in WT animals 3 days later yet patent infection

Fig 1 Daily numbers (mean ± SD) of eggs per gram of faeces

(EPG) in immune and control FcRγ-KO and WT mice

challenged orally with 300 lung-recovered L3 of S venezuelensis

and sacrificed on day 13 post challenge

Fig 2 Number of adult worms (mean ± SD) recovered from the

small intestines of immune and control FcRγ-KO and WT mice

challenged orally with 300 lung-recovered L3 of S venezuelensis

and sacrificed on days 5 and 13 post challenge

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occurs only in the latter animals In this infection protocol

serum mMCP-1 was also similar but sulphated

proteoglycans were found to be significantly higher in the

WT than KO animals Moreover, our results in this study

agree with and support those of Maruyama and his

associates [12] who showed that glycosaminoglycans of the

type produced by mucosal mast cells significantly inhibited

the invasion and establishment of adult S venezuelensis

implanted into the duodenum of nạve mice

In conclusion, defective secondary immunity against S.

venezuelensis in FcRγ-KO mice is associated with

significant decrease in the amount of mast cell-derived

chondroitin sulphates released into the gut lumen, suggesting that the sugars are intimately associated with worm expulsion It is our opinion that extensive further studies into the role of sulphated sugars of the type produced

by mucosal mast cells in parasitic gastrointestinal nematode expulsion is essential and worthy of support as they seem attractive candidates for anthelmintic drug investigation and development

Acknowledgments

We thank Mrs Eri Ono for excellent technical assistance

Fig 3 (A) Chondroitin sulphate (ChS) concentration (µg/ml) (mean ± SD) in gut washings of FcRγ-KO and WT mice challenged orally

with 300 lung-recovered L3 of S venezuelensis and sacrificed on day 1 post challenge ChS-A:E = Chondroitin sulphate A-E;

Us-Ch = Unsulphated chondroitin (B) Us-Chondroitin sulphate (Us-ChS) concentration (µg/ml) (mean ± SD) in gut washings of FcRγ-KO and

WT mice challenged orally with 300 lung-recovered L3 of S venezuelensis and sacrificed on day 5 post challenge

ChS-A:E = Chondroitin sulphate A-E; Us-Ch = Unsulphated chondroitin (C) Chondroitin sulphate (ChS) concentrations (µg/ml)

(mean ± SD) in gut homogenates of FcRγ-KO and WT mice challenged orally with 300 lung-recovered L3 of S venezuelensis and

sacrificed on day 8 post challenge ChS-A:E = Chondroitin sulphate A-E; Us-Ch = Unsulphated chondroitin

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and Professor Toshiyuki Takai and Dr Masao Ono (Tohoku

University, Sendai, Japan) for generously providing the

mutant mice used in this study DNO was a JSPS

postdoctoral research fellow and funding for this work was

provided by the Grant-in-Aid for scientific research from the

Ministry of Education, Culture, Sports, Science and

Technology, Japan We also thank the University of Nigeria,

Nsukka for granting DNO the study leave which enabled

him undertake the fellowship in Japan

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