Letter 
Prion in Saliva of Bovine Spongiform Encephalopathy–Infected Cattle 
To the Editor: A definitive diagnosis of bovine spongiform encephalopathy 
(BSE) in cattle usually relies on Western blot and immunohistochemical testing 
of samples from the obex region of the brainstem. These conventional diagnostic 
tests can detect the presence of the abnormal (disease-associated) form of the 
prion protein (PrPSc) in brain samples several months before the onset of 
clinical signs; however, there is no appropriate, universal tool for early 
preclinical and antemortem diagnosis of BSE. Furthermore, confirmation of the 
disease is currently only possible by postmortem examination of brain tissues. 
In this study, we used the serial protein misfolding cyclic amplification 
(sPMCA) technique to determine the presence of PrPSc in saliva samples collected 
from BSE-infected cows before and after the onset of disease (1). In a previous 
study (2), we analyzed the tissue distribution of PrPSc in cattle up to 66 
months after they were orally inoculated with a relatively low dose (5 g) of 
homogenized brainstem from animals with naturally occurring BSE in England. In 
2011, after publication of that study and 83.3 months after the cows were 
inoculated, clinical signs of BSE developed in 1 cow (no. 5444); necropsy was 
performed 84.7 months after inoculation. In addition, we used saliva samples 
from 2 BSE-affected cows (nos. 5413 and 5437) (2) to determine the presence of 
PrPSc. We collected saliva samples from animals at 4 monthly intervals, 
beginning in 2009, 56 months after inoculation. Samples were stored at −80°C 
until analysis. Using the sodium phosphotungstic acid precipitation method, we 
concentrated (100-fold) individual 1-mL saliva samples from each time point. We 
then diluted the concentrated samples 1:10 with the normal isoform of prion 
protein substrate containing 0.5% potassium dextran sulfate. Using the sPMCA 
technique as described (1), we amplified the samples in 3–8 tubes, and we used 
Western blot to analyze the proteinase K–treated sPMCA products (2). Using 
Western blot and immunohistochemical tests, we detected the accumulation of 
PrPSc in brains collected at necropsy from the 3 cows examined. In addition, 
using the sPMCA technique, we detected PrPSc signal in 1) saliva samples that 
were concentrated from samples collected from the same 3 cows at necropsy and in 
2) concentrated saliva samples that were collected from 2 of the cows (nos. 5413 
and 5444) at the early clinical stages of disease. 
Figure 
Figure. . Western blot detection, using the serial protein misfolding 
cyclic amplification technique, of the abnormal (disease-associated) form of the 
prion protein (PrPSc) in concentrated saliva samples from 3 cows experimentally 
infected... 
After saliva samples underwent 3 rounds of amplification, we detected PrPSc 
in a saliva sample that was collected from cow number 5437 two months before the 
clinical onset of clinical symptoms (Figure). For 2 of the cows (nos. 5413 and 
5437), the positive ratio of salivary PrPSc at round 4 of amplification 
increased as the disease progressed (Figure). Because PrPSc signal could be 
detected in BSE-infected brain homogenates diluted up to 10−10 after 2 rounds of 
amplification (1), we estimated PrPSc levels in the nonconcentrated original 
saliva samples to be lower than those in BSE-infected brain homogenate diluted 
to 10−12. No PrPSc signal was detected in samples collected from the 3 cows 3–5 
months before the onset of clinical symptoms or from age-matched noninfected 
controls, even after 4 rounds of amplification. We demonstrated the presence of 
PrPSc in saliva of BSE-affected cows during the clinical stage of the disease, 
and in 1 case, at the preclinical or asymptomatic stage. Our findings suggest 
that PrPSc is likely to be detected in the saliva of BSE-affected cattle during 
the clinical stage of disease, after accumulation of PrPSc in the brain. PrPSc 
was found in the salivary glands of BSE-affected cattle at the terminal stage of 
infection (1). Therefore, once the infectious agent reaches the central nervous 
system, it may spread centrifugally from the brain to the salivary glands 
through the autonomic nervous system. Infectivity of saliva and the presence of 
PrPSc in saliva have been reported in other ruminants affected with 
transmissible spongiform encephalopathy. Infectivity of saliva was demonstrated 
in deer with chronic wasting disease (3) and in scrapie-affected sheep (4); the 
immunolabeled PrPSc accumulated in the salivary glands of scrapie-affected sheep 
(5). A low level of PrPSc was detected in concentrated buccal swab samples of 
preclinical scrapie-infected sheep by using sPMCA (6,7). These results suggest 
that small amounts of PrPSc may accumulate in the salivary glands and are then 
secreted into saliva. The presence of infectious prions in saliva may explain 
the facile horizontal transmission of scrapie in sheep (4–6) and chronic wasting 
disease in deer (4,8). There has been no epidemiologic evidence, however, that 
saliva, milk, blood, and cerebrospinal fluid from BSE-infected cattle are 
infectious (9). Nonetheless, the potential risk for BSE transmission by body 
fluids or excretions from BSE-infected cattle is cannot be ruled out by the 
current data. 
Hiroyuki Okada, Yuichi Murayama , Noriko Shimozaki, Miyako Yoshioka, 
Kentaro Masujin, Morikazu Imamura, Yoshifumi Iwamaru, Yuichi Matsuura, Kohtaro 
Miyazawa, Shigeo Fukuda, Takashi Yokoyama, and Shirou Mohri 
Author affiliations: Author affiliations: National Agriculture and Food 
Research Organization, Tsukuba, Japan (H. Okada, Y. Murayama, N. Shimozaki, M. 
Yoshioka, K. Masujin, M. Imamura, Y. Iwamaru, Y. Matsuura, K. Miyazawa, T. 
Yokoyama, S. Mohri); Hokkaido Research Organization, Shintoku, Japan (S. Fukuda) 
Acknowledgment 
This work was supported by a grant-in-aid from the BSE and Other Prion 
Disease Project of the Ministry of Agriculture, Forestry and Fisheries, Japan. 
References 
1.Murayama Y, Yoshioka M, Masujin K, Okada H, Iwamaru Y, Imamura M, 
Sulfated dextrans enhance in vitro amplification of bovine spongiform 
encephalopathy PrPSc and enable ultrasensitive detection of bovine PrPSc. PLoS 
ONE. 2010;5:e13152. DOIPubMed 2.Okada H, Iwamaru Y, Imamura M, Masujin K, 
Matsuura Y, Murayama Y, Detection of disease-associated prion protein in the 
posterior portion of the small intestine involving the continuous Peyer’s patch 
in cattle orally infected with bovine spongiform encephalopathy agent. 
Transbound Emerg Dis. 2011;58:333–43. DOIPubMed 3.Haley NJ, Seelig DM, Zabel MD, 
Telling GC, Hoover EA. Detection of CWD prions in urine and saliva of deer by 
transgenic mouse bioassay. PLoS ONE. 2009;4:e4848. DOIPubMed 4.Tamgüney G, Richt 
JA, Hamir AN, Greenlee JJ, Miller MW, Wolfe LL, Salivary prions in sheep and 
deer. Prion. 2012;6:52–61. DOIPubMed 5.Vascellari M, Nonno R, Mutinelli F, 
Bigolaro M, Di Bari MA, Melchiotti E, PrPSc in salivary glands of 
scrapie-affected sheep. J Virol. 2007;81:4872–6. DOIPubMed 6.Maddison BC, Rees 
HC, Baker CA, Taema M, Bellworthy SJ, Thorne L, Prions are secreted into the 
oral cavity in sheep with preclinical scrapie. J Infect Dis. 2010;201:1672–6. 
DOIPubMed 7.Gough KC, Baker CA, Rees HC, Terry LA, Spiropoulos J, Thorne L, The 
oral secretion of infectious scrapie prions occurs in preclinical sheep with a 
range of PRNP genotypes. J Virol. 2012;86:566–71. DOIPubMed 8.Mathiason CK, 
Powers JG, Dahmes SJ, Osborn DA, Miller KV, Warren RJ, Infectious prions in the 
saliva and blood of deer with chronic wasting disease. Science. 2006;314:133–6. 
DOIPubMed 9.Brown P, Andréoletti O, Bradley R, Budka H, Deslys JP, Groschup M, 
WHO tables on tissue infectivity distribution in transmissible spongiform 
encephalopathies. Geneva: World Health Organization; 2010 [cited 2011 Nov 2]. http://www.who.int/bloodproducts/tablestissueinfectivity.pdf 
Figure Figure. . Western blot detection, using the serial protein 
misfolding cyclic amplification technique, of the abnormal (disease-associated) 
form of the prion protein (PrPSc) in concentrated saliva samples from 3 cows 
experimentally... 
Suggested citation for this article: Okada H, Murayama Y, Shimozaki N, 
Yoshioka M, Masujin K, Imamura M, et al. Prion in saliva of bovine spongiform 
encephalopathy–infected cattle [letter]. Emerg Infect Dis [Internet]. 2012 Dec 
[date cited]. DOI: http://dx.doi.org/10.3201/eid1812.120528 
DOI: 10.3201/eid1812.120528 
DISSERTATION
CHRONIC WASTING DISEASE: A MODEL FOR PRION TRANSMISSION VIA SALIVA AND 
URINE
Submitted by Nicholas James Haley
Department of Microbiology, Immunology and Pathology In partial fulfillment 
of the requirements For the Degree of Doctor of Philosophy Colorado State 
University Fort Collins, Colorado Summer 2010 
ABSTRACT OF DISSERTATION CHRONIC WASTING DISEASE: A MODEL FOR PRION 
TRANSMISSION VIA SALIVA AND URINE
Chronic wasting disease (CWD) of cervids is a prion disease distinguished 
by its high level of transmissibility, wherein bodily fluids and excretions are 
thought to play an important role. Typical of all prion diseases, CWD is 
characterized by the forced conversion of the normal prion protein (PrPC) into a 
misfolded isoform (PrPCWD), which has been shown to accumulate primarily in 
tissues of the lymphoid and nervous systems, though has also been found in other 
peripheral tissues including elements of the cardiovascular, musculoskeletal, 
and urogenital systems. Despite strong evidence that natural infection is 
acquired from the environment, as well as saliva and blood, a more thorough 
evaluation of excreta, including saliva, urine, and feces, is essential for a 
comprehensive foundation for (1) understanding how environmental 
CWDcontamination occurs, (2) developing in vitro assays for the antemortem 
identification of CWD-infected cervids, and (3) demonstrating the pathogenesis 
of the disease in the natural host.
In this dissertation, two approaches are used to identify infectious CWD 
prions and PrPCWD in the bodily fluids and tissues of CWD-exposed white-tailed 
deer: a novel bioassay system using a transgenic mouse line expressing the 
cervid PrP protein (Tg[CerPrP] mice), and a recently developed prion 
amplification assay known as serial iv protein misfolding cyclic amplification 
(sPMCA). In conjunction with immunohistochemistry and western blotting, these 
two assays were used to definitively identify CWD prions in saliva and urine, in 
addition to elements of the lymphoreticular system, central and peripheral 
nervous systems, and urogenital and oropharyngeal tissues. In initial 
experiments, concentrated urine and saliva samples from terminal CWD+ 
white-tailed deer, suspected of harboring infectious CWD prions, was assessed by 
Tg[CerPrP] bioassay and sPMCA. Authentic prion infectivity was detected in urine 
and saliva using both detection systems in the case of urine, though only mouse 
bioassay successfully demonstrated CWD prions in saliva. The concentration of 
abnormal prion protein in bodily fluids was very low, as indicated by: 
undetectable PrPCWD levels by traditional assays (western blot, ELISA) and 
prolonged incubation periods and incomplete TSE attack rates in inoculated 
Tg[CerPrP] mice. These findings helped to extend the understanding of CWD prion 
shedding and transmission and portend the detection of infectious prions in body 
fluids in other prion infections.
Based on the identification of CWD prions in saliva (“prionsialia”) and 
urine (“prionuria”), I next sought to determine whether deer previously exposed 
orally to urine and feces from CWD+ sources, while conventional test-negative, 
may actually be harboring very low level CWD infection not evident in the 19 
month observation period in initial cervid bioassay studies. A selection of 
tissues, including those of the lymphoreticular and both central and peripheral 
nervous systems were fully examined, initially using Tg[CerPrP] bioassay to 
demonstrate true infectivity, and secondarily with sPMCA. Positive controls 
consisted of issues from CWD+ deer exposed orally to saliva; negative control 
tissue sets were collected from deer exposed orally and intracranially to
v
CWD-negative brain. PrPCWD was detected in the tissues of orally exposed 
deer by both sPMCA and Tg[CerPrP] mouse bioassay; each assay revealed very low 
levels of CWD prions previously undetectable by western blot, ELISA, or IHC. 
Serial PMCA analysis of individual tissues identified that obex alone was 
positive in urine/feces exposed deer. PrPCWD was amplified from both LRS and 
neural tissues of positive control deer but not from the same tissues of 
negative control deer. Detection of subclinical infection in deer orally exposed 
to urine and feces (1) suggests that a prolonged subclinical state can exist 
such that observation periods in excess of two years may be needed to detect CWD 
infection, and (2) illustrates the sensitive and specific application of sPMCA 
in the diagnosis of low-level prion infection.
Despite the confirmation of infectious prions in urine and saliva, along 
with conventional test-negative deer exposed to urine and feces, the manner in 
which infectivity is transferred to these excreta is unknown. To address this, I 
went on to apply sPMCA to tissues associated with production and excretion of 
urine and saliva in an effort to seek proximal sources of prion shedding. I 
blindly analyzed oropharyngeal and urogenital tissues, reproducibly 
demonstrating PrPCWD in each tissue examined in 3 rounds of sPMCA; whereas blood 
samples from the same animals and concurrent negative controls remained 
negative. Tissue distribution was affected by route of inoculation and CNS 
burden. The identification of PrPCWD in bodily fluids and conventional-test 
negative tissues – in the absence of detection by conventional methods – may 
indicate the presence of protease-sensitive infectious prions in excretory 
tissues not revealed by assays employing PK digestion or other means to remove 
PrPC reactivity.
vi
The continued evaluation of bodily fluids and peripheral tissues via sPMCA 
may therefore allow additional insights into prion transmission, trafficking, 
and pathogenesis. 
 Nicholas James Haley Department of Microbiology, Immunology and Pathology 
Colorado State University Fort Collins, CO 80523 Summer 2010 
 snip...
In summary, this study demonstrates for the first time amplifiable PrPCWD 
in various organs and tissues associated with prionsialia and prionuria. The 
ultimate source and mechanism of release into bodily fluids remain unknown, 
though elevated levels in both salivary gland and urinary bladder provides 
strong evidence that these tissues play a crucial role in prion excretion. In 
addition, the source and route of inoculation weighed heavily on the terminal 
peripheral distribution of PrPCWD, as did an individual’s apparent central 
nervous system burden. Finally, while this discovery provides evidence for prion 
invasion of peripheral excretory tissues, the timing of infiltration during CWD 
infection and the protease resistance profile of these prions warrant future 
studies in serial pathogenesis and detection of alternate infectious prion 
species. 
Sunday, July 03, 2011 
Prion Disease Detection, PMCA Kinetics, and IgG in Urine from 
Naturally/Experimentally Infected Scrapie Sheep and Preclinical/Clinical CWD 
Deer 
Thursday, June 09, 2011 
Detection of CWD prions in salivary, urinary, and intestinal tissues of 
deer: potential mechanisms of prion shedding and transmission 
CHRONIC WASTING DISEASE: A MODEL FOR PRION TRANSMISSION VIA SALIVA AND 
URINE 
Sunday, December 06, 2009 
Detection of Sub-Clinical CWD Infection in Conventional Test-Negative Deer 
Long after Oral Exposure to Urine and Feces from CWD+ Deer 
Wednesday, March 18, 2009 
Detection of CWD Prions in Urine and Saliva of Deer by Transgenic Mouse 
Bioassay 
*** Tuesday, September 02, 2008 
 Detection of infectious prions in urine (Soto et al Available online 13 
August 2008.) 
Friday, October 26, 2012 
CHRONIC WASTING DISEASE CWD PENNSYLVANIA GAME FARMS, URINE ATTRACTANT 
PRODUCTS, BAITING, AND MINERAL LICKS
TSS
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