5 6
Revista da Sociedade Br asileir a de Medicina Tr opical 3 8 ( 1 ) :5 6 -5 7 , jan-fev, 2 0 0 5
RELATO DE CASO/CASE REPORT
Intestinal spirochetosis and colon diverticulosis
Espiroquetose intestinal e diverticulose do cólon
Marcus Aurelho de Lima
1, 2, André Luís Barbosa
1,2, Vitorino Modesto dos Santos
2,
and Felipe Prata Misiara
2ABSTRACT
A c a se o f i n te sti n a l sp i ro c h e to si s i n a 6 2 - ye a r- o ld wh i te m a le i s re p o rte d . Th e c o n d i ti o n wa s c h a ra c te ri ze d b y c h ro n i c f la tu le n c e a n d e p i so d e s o f i n te sti n a l h e m o rrh a ge , i n a d d i ti o n to th e e vi d e n c e o f h yp o to n i c d i ve rti c u la r d i se a se , wi th a la rge n u m b e r o f sle n d e r o rga n i sm s i n th e c o lo n e p i th e li u m a n d c ryp ta e . Sp i ro c h e te s we re d e m o n stra te d b y Wh a rti n -Sta rry sta i n . Th e se ro lo gi c te sts f o r syp h i li s a n d HIV we re p o si ti ve . Sp i ro c h e to si s wa s tre a te d wi th p e n i c i lli n G, a n d th e p a ti e n t re m a i n s f re e o f i n te sti n a l c o m p la i n ts 2 0 m o n th s la te r.
Ke y-words: Co lo n . Di ve rti c u lo si s. HIV. In te sti n a l b le e d i n g. Sp i ro c h e to si s.
RESUMO
Um c a so d e e sp i ro q u e to se i n te sti n a l é re la ta d o e m u m h o m e m b ra n c o d e 6 2 a n o s. A c o n d i ç ã o f o i c a ra c te ri za d a p o r f la tu lê n c i a c rô n i c a e e p i só d i o s d e h e m o rra gi a i n te sti n a l, a lé m d a e vi d ê n c i a d e d o e n ç a d i ve rti c u la r h i p o tô n i c a d o s c ó lo n s, c o m n u m e ro so s o rga n i sm o s f i la m e n to so s n o e p i té li o e n a s c ri p ta s d o c ó lo n . Os e sp i ro q u e ta s f o ra m d e m o n stra d o s p e la c o lo ra ç ã o d e Wh a rti n - Sta rry. Os te ste s so ro ló gi c o s p a ra sí f i li s e HIV f o ra m p o si ti vo s. A e sp i ro q u e to se f o i tra ta d a c o m p e n i c i li n a G e o p a c i e n te p e rm a n e c e se m q u e i x a s i n te sti n a i s a p ó s 2 0 m e se s.
Pal avr as-chave s: Có lo n . Di ve rti c u lo se . Esp i ro q u e to se . HIV. Sa n gra m e n to i n te sti n a l.
1 . Canc e r Co mb at Asso c iatio n o f Ce ntr al B r azil, Ub e r ab a, MG; 2 . Unive r sity Ho spital o f Tr iângulo Mine ir o Me dic al Sc ho o l, Ub e r ab a, MG, B r azil.
Addr e ss to: Pr o f. Mar c us Aur e lho de Lima. Disc iplina de Endo c r ino lo gia/CM/FMTM. Ge túlio Guar itá 1 3 0 , Ab adia 3 8 0 2 5 - 4 4 0 Ub e r ab a- MG, B r azil. Te l: 5 5 3 4 3 3 1 8 5 2 5 8 , Fax: 5 5 3 4 3 3 1 2 6 6 4 0 .
e - mail: lima@ me dne t. c o m. b r. Re c e b ido par a pub lic aç ão e m 6 /8 /2 0 0 4 Ac e ito e m 1 7 /9 /2 0 0 4
We report a c ase of intestinal spiroc hetosis assoc iated with
c olon divertic ular disease and HIV infec tion. Although c olorec tal
spiroc hetosis was desc ribed in humans by Harland and Lee2 in 1 9 6 7 , the r e still r e mains so me c o ntr o ve r sy ab o ut c linic al
signific anc e and its asso c iatio n with diarrhea and intestinal
bleeding3 5 6. Here, the first cases were reported by De Brito et al1.
This c ondition has been sc arc ely reported, and is c harac terized by the histologic al appearanc e of a fa lse b rush b o rde r on the
c o lo nic muc o sa, whic h r e pr e se nts a laye r o f spir o c he te s2,
espec ially Bra c hyspi ra a a lb o rgi and B r ac hyspir a pilo sic o li ( Serpulina pilosicoli)5. Spirochetes presumably result from
fecal-oral c ontamination, and the c olonization rate in human c olon
depends upon sanitation c onditions, diet, sexual prac tic es, and
im m une func tio n6. Altho ugh the a sso c ia tio n o f inte stina l
spirochetosis with human immunodeficiency virus infection could favor invasive disease, dissemination to other organs is extremely
rare6.
Our pur po se is to e mphasize unsuspe c te d spir o c he to sis
as a po ssible diagno stic pitfall in patients presenting intestinal
b le e ding asso c iate d o r no t with dive r tic ulo sis.
CASE REPORT
A 6 2 -year-old white male was seen on July 2 0 0 2 , with a
c hr o nic c o mplaint o f flatule nc e and e piso de s o f inte stinal
hemorrhage that oc c urred only during evac uation. The patient
used alc ohol and tobac c o oc c asionally, but denied drug abuse
and there was no history of homosexuality. He was non-febrile
and in a good general state. The heart was normal; pulse rate,
8 0 bpm; blood pressure, 1 4 0 /8 0 mmHg. The lungs, liver, arterial
pulses, and lymph no des were no rmal. The spleen was no t
palpable. A tender lower left abdominal quadrant was found
during palpation. Stool examinations revealed no c onventional
pathogens. A full blood c ount and bioc hemistry determinations
were normal. Proc tologic examination disc losed hemorrhoids
a t 3 a n d 7 h o ur s , with o ut o th e r c h a n ge s o n a n us c o py.
Colonosc opy showed pan-c olon hypotonic divertic ular disease,
and biopsy samples were obtained from the c olon. Histologic al
examinatio n o f the c o lo n b io psies sho wed muc o sa sur fac e
5 7
Ba r bo s a AL e t al
staine d with silve r-me the namine b y Whar tin-Star r y me tho d
( Figur e 2 ) . Ad d i ti o n a l l y, th e r e we r e e d e m a , m o d e r a te i n fl a m m a to r y i n fi l tr a te o f l ym p h o c yte s a n d n u m e r o u s
e o sino phils in the c o r ium. Afte r the histo patho lo gic al data,
th e s e r o lo gic te s ts fo r s yp h ilis a n d HI V we r e p o s itive .
Spir o c he to sis was tr e ate d with pe nic illin G, and the patie nt is fo und fr e e o f inte stinal c o mplaints 2 0 mo nths late r.
Moreover, the study of biopsy allowed us to identify the infectious
process and the spirochetes, which are not detected in the routine stool examinations8. It is noteworthy that spirochetes are difficult
to grow on culture media, which is an important limitation for
the ir c har ac te r izatio n in c linic al pr ac tic e . I n additio n to
immunohistoc hemistry1, the best way to identify spiroc hetes includes ultrastructural study1 4 and polymerase chain reaction1 0,
sophisticated tools are not easily available. In this context, the
positive test for syphilis could constitute another index of suspicion
for general practitioners. Based on clinical suspicion, the diagnosis of intestinal spirochetosis could be confirmed without additional
c osts, bec ause spiroc hetes are easily identified through
silver-methenamine stain. However, Orenstein and Dieteric h ( 2 0 0 1 )
found only four cases of spirochetosis in colon biopsies from 8 2 symptomatic patients with acquired immunodeficiency syndrome8
and conclude that treatable conditions may be overlooked by less
experienced surgical pathologists.
Al th o u g h we c a n n o t d i s c a r d th e h yp o th e s i s th a t
dive r tic ulo s is m a y fa vo r the c o lo niza tio n o f s pir o c he te s , independently fro m the patient’s immune func tio n, in this c ase
the e vide nc e o f inte stinal spir o c he to sis allo we d the suspic io n
o f c o nc o mitant HIV infe c tio n.
We suggest the inc lusion of spiroc hetosis in the differential
diagnosis of intestinal bleeding oc c urring in patients with or without c hanges at c olonosc opy, and biopsy spec imens c ould be
obtained in order to favor the c orrec t diagnosis in suc h c ases.
REFERENCES
1 . De B r ito T, Sa ndo va l MP, Silva AG, Sa a d RC, Co la ia c o vo W. I nte s tina l s p i r o c h e t o s i s : f i r s t c a s e s r e p o r t e d i n B r a zi l a n d t h e u s e o f immuno histo c he mistr y as an aid in histo patho lo gic al diagno sis. Re vista do Instituto de Me dic ina Tr o pic al de São Paulo 3 8 : 4 5 - 5 3 , 1 9 9 6 . 2 . Har land WA, Le e FD. Inte stinal spir o c hae to sis. B r itish Me dic al J o ur nal
3 : 7 1 8 - 7 1 9 , 1 9 6 7 .
3 . He ine RG, War d PB , Mik o sk a AS, B e nne tt- Wo o d V, Ro b ins- B r o wne RM, Hampso n DJ . B r ac hyspir a aalb o r gi infe c tio n in fo ur Austr alian c hildr e n. Jo ur nal o f Gastr o e nte r o lo gy and He pato lo gy 1 6 : 8 7 2 -8 7 5 , 2 0 0 1 . 4 . Ko r n e r M, Ge b b e r s J O. Cl i n i c a l s i gn i fi c a n c e o f h u m a n i n te s ti n a l
spir o c he to sis - a mo r pho lo gic appr o ac h. Infe c tio n 3 1 : 3 4 1 - 3 4 9 , 2 0 0 3 . 5 . Ma c a i gn e l G, Da vi d F, B o i vi n J - F, Ch e i a b S , Ch a ye tte C, De p l u s R .
Spir o c héto se c o lo -r ec tale, une c ause po ssib le mais inhab ituelle de diar r hée c hr o niq ue ? Pr e sse Me dic ale 3 1 : 4 5 8 -4 6 1 , 2 0 0 2 .
6 . Marthinsen L, Willén R, Carlén B, Lindberg E, Värendh G. Intestinal spiroc hetosis in e igh t pe dia tr ic pa tie n ts fr o m So uth e r n Swe de n . Ac ta Pa th o lo gic a , Mic robiologic a, et Immunologic a Sc andinavic a 1 1 0 :5 7 1 -5 7 9 , 2 0 0 2 . 7 . Nak amur a S, Kur o da T, Sugai T, Ono S, Yo shida T, Ak asak a I, Nak ashima F,
Sa s o u S. Th e fir s t r e po r te d c a s e o f in te s tin a l s pir o c h e to s is in J a pa n . Patho lo gy Inte r natio nal 4 8 : 5 8 - 6 2 , 1 9 9 8 .
8 . Or e n s te in J M, Die te r ic h DT. Th e h is to pa th o lo gy o f 1 0 3 c o n s e c utive c o l o n o s c o p y b i o p s i e s fr o m 8 2 s ym p to m a ti c p a ti e n ts wi th a c q u i r e d immuno de fic ie nc y syndr o me : o r iginal and lo o k - b ac k diagno sis. Ar c hive s o f Patho lo gy and Lab o r ato r y Me dic ine 1 2 5 : 1 0 4 2 - 1 0 4 6 , 2 0 0 1 .
9 . Pe ghini PL, Guc c io n J G, Shar ma A. Impr o ve me nt o f c hr o nic diar r he a afte r tr e atm e nt fo r inte stinal spir o c he to sis. Dige stive Dise ase s and Sc ie nc e s 4 5 : 1 0 0 6 -1 0 1 0 , 2 0 0 0 .
1 0 . Shah RN, Sto so r V, B adve S. Co lo n b io psy in a patie nt with diar r he a – Po ssib le e tio lo gic age nt. Ar c hive s o f Patho lo gy and Lab o r ato r y Me dic ine 1 2 5 : 6 9 9 - 6 7 0 , 2 0 0 1 .
Fi gu r e 1 - De tai l of col on e pi the l i u m wi th n u m e r ou s sl e n de r b aci l l i adhe r e d to the m u cosa su r f ace ( ar r owhe ads) ( HE x 4 0 0 ) .
Fi gu r e 2 - The spi r oche te s appe ar i n col on m u cosa stai n e d b y si l ve r-m e the n ar-m i n e ( ar r ows) ( Whar ti n -Star r y x 4 0 0 ) .
DISCUSSION
This patie nt pr e se nte d with b r ight inte stinal b le e ding,
similarly to several other reports3. Although the exac t sourc e
o f b le e ding c o uld no t b e e stab lishe d thr o ugh e ndo sc o pic examination, the c olon divertic ular disease c ould play a role
in this c ase . The diagno sis o f inte stinal spir o c he to sis was
established by the histologic study of c olon biopsy1 2 3 4 5 6 7 8 9 1 0.
In spite o f the appar e nt r e spo nse to pe nic illin G, with
r e so lutio n o f his sympto ms4 .9, we c o uld no t e ntir e ly asc e r tain
the ab se nc e o f o the r e ve ntual patho ge ns asso c iate d with HIV infe c tio n5. No twithstanding, the patie nt had no vo m iting,
abdo minal pain, diarrhea, o r lo ss o f weight, the mo st frequent
