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R e v i s t a d a S o c i e d a d e B r a s i l e i r a d e M e d i c i n a T r o p i c a l 2 9 ( 5 ) : 5 0 3 - 5 0 6 , s e t - o u t , 1 9 9 6 .

REIA TO D E CASO

PA RO TID EN LA RG EM EN T D UE TO A D EN O V IRUS

IN FEC T IO N IN PA TIEN T W IT H HUM A N

IM M U N O D EFIC IEN C Y V IRUS IN FEC T IO N

Maria Irma Seixas Duarte, Valdir Sabbaga Amato, Juliane Gomes de Paula,

David Everson Uip, M arcos Boulos and Vicente Amato Neto

The aut h o rs repo rt a ca s e o f ade no v irus - i n d u c e d e n la rg e m e n t o f t he p a ro t id g l a n d inv o lv ing a p a t ie n t in fe c t e d w ith h u m a n im m u n o d e ficie n cy v irus (H IV ). P hy s ical e x a m in a t i o n re v ea led g o o d g e n e ra l co ndit io n, no f e v e r a n d b ilat eral e n la rg e m e n t o f t he p a ro t id regio n, w hich w as o f in c re a s e d co ns is t ency a n d slightly t e n d e r to p alp at io n. H is t o lo g ica l e x a m i n a t i o n o f t he p a ro t i d g l a n d d e m o n s t ra t e d a s light p e ri d u c t a l l y m p h o m o n o n u c l e a r i n fla m m a t o ry infilt ra t e w it h t h e p re s e n c e o f f o c a l p o i n t s o f necro s is . Tests to d e t e rm in e t he p re s e n c e o f f u n g i a n d alco h o l- acid res ist ent b acilli w ere negat iv e. Im m uno his t o chem is t ry f o r cy t o m egalo v irus , heip es s im plex , H I V p 2 4 a nt i gen a n d a de no v iru s s ho w ed po sitiv ity o nly f o r a de no v irus in t he epit helial n u c le i o f n u m e ro u s g l a n d d uct s . Tins is the t hird ca s e o f this ty pe re p o rt e d in t he lit erat ure, ind ic a t i n g t he im p o rt a nce o f i n c lu d in g ade no v irus in t he differen t ial diagno s is o f this co ndit io n.

K ey - w o rds : H u m a n im m u n o d e ficie n cy v irus. A deno v irus . P aro t id g la n d .

T h e i n v o l v e m e n t o f s a liv a r y g la n d s , e sp ec ially the p aro tid g land s, in p atients w ith A ID S has b e e n rec o g n iz ed as o n e o f the m ultip le c linic o p ath o lo g ic m anifestatio ns o f in fec tio n w ith HIV.

Sin c e o f the first d esc rip tio n o f A ID S-related inv o lv em ent o f the p aro tid and su bm and ib u lar g land s d esc rib ed b y Ry an in 198513, a larg e n u m b er o f p atho lo g ies inv o lv ing the p aro tid g land s hav e b e e n rep o rted . Usually, intrap aro tid and p erip aro tid ly m p h n o d es are inv o lv ed , w ith the c o m m o n o c c u rren c e o f hy p erp lastic ly m p had eno p athy and b enig n intraep ithelial le sio n s15.

A d e n o v ir u s e s f r e q u e n tl y a tta c k H IV - in fec ted p atients, c au sing ev ents rang ing fro m asy m p to m atic ex c retio n in u rine and fe c e s to clinic al m anifestatio ns o f v ariab le sev erity 5

W e rep o rt a c ase o f p aro tid enlarg em ent in an H IV - in f e c te d in d iv id u a l a s s o c i a te d w ith ad eno v iru s in the p aro tid d ucts.

D epartam ento de D oenças Infecciosas e Parasitárias e Laboratório de Investigação M édica em M oléstias Infecciosas do H ospital das Clinicas, da Faculdade de M edicina da U niversidade de São Pauio.

E n d e re ç o p a ra c o rre s p o n d ê n cia : Pro f “ D r“ M aria Irma Seixas D uarte. D epartam ento de Patologia da Universidade de São Paulo. A venida Dr. A rnaldo 4 5 5 , sala 2 6, 0 1 246- 000 São Paulo, SI? Brasil.

Receb id o para pub licação em 0 3 / 0 1 / 9 6 .

CA SE REPO RT

A 52-y ear o ld m ale h o m o se x u al w as se e n at th e Paulista Institute o f In fec tio u s and Parasitic D isease o n Janu ary 30, 1995, w ith a cerv ical and b ilateral p reau ricu lar tu m o r fo rm atio n starting 6 m o nths p rio r to ad m issio n. H e d en ied p ain, m o u th d ry ness, fev er o r w h eig ht lo ss. H e rep o rted o n ly a sing le ep iso d e o f h erp e s z o ster 1 y ear p rio r to exam inatio n, w ith sp o n tan eo u s reso lu tio n.

P h y s ic a l e x a m in a tio n r e v e a le d a g o o d g e n e ra l c o n d itio n , n o f e v e r an d b ila te ra l e n larg em en t o f th e p aro tid re g io n , w ith inc reased c o n sisten c y and slig htly ten d er to p alp atio n, as w ell as h ep ato m eg aly . Th ere w ere n o ad d itio nal alteratio ns. Blo o d c o u nts sho w ed 1,762 ly m p ho cy tes/ m m 3.

Th e ELISA fo r H IV w as p o sitiv e, w ith later c o nfirm atio n b y W estern b lo t. O n M arch 1, 1995, C D 4 c o u nts w ere 577/ m m 3 and CD g o f 810 m m 3. O n that o c c asio n , to m o g rap hy o f the cerv ical regio n sho w ed d iffuse and hetero g eneo us en larg em en t o f th e p aro tid g lan d s w ith intrag land u lar no d u lar im ag es, so m e o f them o f cy stic o r n ec ro tic ty p e, ad en o m eg aly o f the caro tid c hain, and hep ato m eg aly w ith three im ag es c o m p atib le w ith ang io m as.

H isto lo g ic exam in atio n o f a p aro tid b io p sy d em o nstrated a p erid uctal ly m p ho m o no nu clear

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R e l a t o d e C a s o . D u a r t e M I S , A m a t o VS, P a u l a J G , U ip D E , B o u l o s M , A m a t o N e t o V. P a r o t i d e n l a r g e m e n t d u e t o a d e n o v i r u s i n f e c t i o n i n p a t i e n t w i t h h u m a n i m m u n o d e f i c i e n c y v i r u s i n f e c t i o n . R e v i s t a d a S o c i e d a d e B r a s i l e i r a d e M e d i c i n a T r o p i c a l 2 9 : 5 0 3 - 5 0 6 , s e t- o u t , 1 9 9 6 .

in flam m ato ry infiltrate w ith the p re sen c e o f fo c al n ec ro sis. A se arc h fo r fu ng i b y G ro co tt staining and fo r alc o ho l-ac id -resistan t b acilli (A A RB) b y Z iehl-N eelsen staining w as neg ativ e. Im m u n o h isto c h em istry fo r c y to m eg alo v iru s, h erp e s sim p lex, A A RB, H IV p 24 antig en and a d e n o v i r u s s h o w e d p o s i ti v i ty o n ly f o r ad eno v iru s in the ep ithelial n u c lei o f the o f nu m ero u s g land d u cts (Fig u re 1). It w as n o t p o ssib le to id entify the sero ty p e. Th e p atient w as su b m itted to 500m g/ d z id o v u d ine and c o u rsed satisfacto rily , w ith w eig h t g ain o f 5kg and c o n sid e rab le red u c tio n o f p aro tid size u p o n p h y sic al ex am in atio n six m o nths after d iag no sis. Th e p atient is c u rrently d o ing w ell o n e y e ar b ac k .

F i g u re 1 - P aro t id g l a n d w ith po s it iv e i m m u n o s t a i n i g f o r a d e n o v iru s in t he n u c le i a n d cy t o plas m o f t he d u ct a l ep it helium . Im uno his t o che m ical, a d en o v irus m o n o c lo n a l c h e m ic o n 1 / 1 0 0 c a b c .X 6 3 0 O M .

D ISCUSSIO N

T h e in v o lv em ent o f the p aro tid g land s in H IV -infected o r A IDS p atients m ay b e p o tentially

d u e to a l a r g e n u m b e r o f c o n d i ti o n s . L y m p h o e p ith e lial le sio n s ( ly m p h o e p ith e lia l sialo p hathy) w itho ut the no n-salivary co m p o nents o f Sjo g ren sy nd ro m e hav e b e e n rep o rted , to g e th e r w ith ly m p h o e p ith e lia l c y sts an d h y p e rp lastic ly m p h ad e n o p ath y b o th w ith in intrap aro tid and p erip aro tid ly m p h n o d e s14, T h e se find ing s hav e b e e n asso c iated w ith the d etectio n o f p 24 antigen by imm uno histo chem istry in p aro tid ly m p ho id le sio n s2. O th er co nd itio ns rep o rted w ere ad en o id c y stic c arc in o m a17, n o n- H o d g kin’s ly m p ho m a9, K ap o si’s sarc o m a12, and ac u te o r c h ro n ic sialo ad enitis2.

It is estim ated that ap p ro xim ately 12% o f p a tie n ts w ith A ID S h av e a c tiv e ad e n o v iru s infectio ns8 and that this v irus is iso lated fro m the u rine o f u p to 2 0 % o f the in fec ted p atients5, altho u gh ad eno v iruses usually d o no t p articip ate sig n ific an tly in o p p o rtu n istic p ath o lo g ic al p ro cesses. H o w ev er, ad eno v iru ses hav e b e e n d esc rib e d in p atients w ith A ID S in d ifferent situ atio ns su c h as g astro intestinal in fec tio ns’ 11, resp irato ry in fec tio ns o ften co m p lic ating c ases o f acu te resp irato ry insu fficiency 16, d issem inated in fec tio ns w ith liv er n ec ro sis9, renal in fec tio n w ith n e c ro tiz in g tu b u lo in te rstitial n e p h ritis7, en c ep h alitis and d issem inated ep end y m itis1, and related to g ranu lo m a annu lare-like skin lesio ns3.

N o e f f e c tiv e i n v iv o c h e m o th e r a p e u tic a g e n ts a g a in s t a d e n o v iru s e s e x is t to d ay . Ribavirin has in v itro activity against ad eno v iru ses b u t sy stem ic treatm ent w ith this ag en t req u ires ad d itio nal stud y 10. D u e to the lac k o f sp e c ific m ed icatio ns o f p ro v en c lin ic al e ffic ac y fo r the treatm ent o f ad eno v iru s in fec tio n an d b ase d o n the p o ten tial c o -p artic ip atio n o f H IV in the p ath o g en esis o f p aro tid enlarg em ent in th ese p a tie n ts , w e a d m in i s te r e d a n ti r e tr o v ir a l therap y w ith z id o v u d ine fo r o u r c ase in sp ite o f n o t hav ing d em o nstrated p 24 an tig en in p aro tid . A Z T treatm ent red u c ed the siz e o f the tu m o r- lik e f o rm a tio n s an d p ro d u c e d an ac c e p tab le c o sm e tic and sy m p to m atic result, as rep o rted in p rev io usly 15. Fu rther inv estig atio n is n eed ed to d efin e the b e st d iag n o stic and therap eu tic ap p ro ac h to the v ario u s ty p es o f p aro tid in v o lv em ent in H IV -infected o r A ID S p atients.

Tw o o th er c ases o f ad eno v iru s-ind u ced enlarg em ent o f the p aro tid g land inv o lv ing H IV -infected p atients hav e b e e n rep o rted in the literature. T h e se tw o p atients, in c o n trast to o u rs, had ad v anc ed H IV in fec tio n , w ith a p rev io u s histo ry o f o p p o rtu nistic in fec tio ns6.

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R e l a t o d e C a s o . D u a r t e M I S , A m a t o V S, P a u l a J G , U ip D E , B o u l o s M , A m a t o N e t o V. P a r o t i d e n l a r g e m e n t d u e t o a d e n o v i r u s i n f e c t i o n i n p a t i e n t w i t h h u m a n i m m u n o d e f i c i e n c y v i r u s i n f e c t i o n . R e v i s t a d a S o c i e d a d e B r a s i l e i r a d e M e d i c i n a T r o p i c a l 2 9 : 5 0 3 - 5 0 6 , s e t - o u t , 1 9 9 6 .

T h e p re sen t rep o rt is im p o rtant b e c au se it su p p o rts the hy p o thesis that ad eno v iru s sho u ld b e c o n sid ered in th e d ifferential d iag no sis as an ag ent cau sin g en larg em en t o f the p aro tid g land am o n g H IV -infected p atients w h o m ay o r m ay n o t h av e p ro g ressed to A ID S.

RESUM O

O s a ut o re s rela t am u m cas o d e a u m e n t o da g l â n d u la p a ró t i d a o ca s io na do p o r adeno v irus , em p a c ie n t e infect ad o p e lo v írus d a im u n o d e fic iê n c ia h u m a n a . A o e x a m e fís ico , este s e ap re s ent av a em b o m es tado geral, a fe b ril e co m a u m e n t o b ilat eral d e parõ t idas, d e co nsistência aum ent ad a e d is cre t a m en t e d o l o ro s a s ã p a l p a ç ã o . O e x a m e h i s t o l ó g i c o d a p aró t ida dem o ns t ro u dis creto infiltrado inflam at ó rio lin fo m o n o n u c le a r p e ri d u c t a l co m p re s e n ç a d e fo co s d e necro s e , as p e s q uis a s p a ra f u n g o s e b acilo s ãlco o l ã c i d o re s i s t e n t e s f o r a m n e g a t iv a s . A t é c n i c a d e im uno - h is t o q uím ica p a ra cito m egalo v írus , b e ip es s im ples, a n t í ge n o p 2 4 d o H I V e a deno v irus , s o m en t e e v i d e n c i o u p o s í t i v i d a d e p a r a o ú lt im o . E s t e é o t e rce iro cas o des crit o n a lit erat ura, d es t a ca n d o a im p o rt â ncia d e i n c lu ir o ad e no v ím s , n o diagnó s t ico d if ere nc ia l, des t e aco m et im ent o .

P a l a v ra s - c h a v e s : V íru s d a i m u n o d e f i c i ê n c i a h u m a n a . A de no v irus . P aró t ida.

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9. Ioachim HL, Ryan JR, Blaugrund SM. Salivary gland lymph nodes. T he site o f lym phadenopathies an d l y m p h o m as a s s o c i a te d w i th h u m an im m unodeficiency virus infection. A rchives of Patholology and Laboratory M edicine

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R e l a t o d e C a s o . D u a r t e M I S , A m a t o VS, P a u l a J G , U ip D E , B o u l o s M , A m a t o N e t o V. P a r o t i d e n l a r g e m e n t d u e t o a d e n o v i r u s i n f e c t i o n i n p a t i e n t w i t h h u m a n i m m u n o d e f i c i e n c y v i r u s i n f e c t i o n . R e v i s t a d a S o c i e d a d e B r a s i l e i r a d e M e d i c i n a T r o p i c a l 2 9 : 5 0 3 - 5 0 6 , s e t - o u t, 1 9 9 6 .

adenovirus serotype in a patient w ith AIDS. The 17. Z eitlen S, Shaha A. Parotid manifestations o f HIV Journal o f Infectious D isesases 16 0 :3 4 9 - 3 5 1 , infection. Journal of Surgical O ncology

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All subjects lived in Mambai municipality an end em ic