Pediatrics 1994 Stamos 525 8

7/30/2019 Pediatrics 1994 Stamos 525 8

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1994;93;525PediatricsJulie Kim Stamos, Kathleen Corydon, James Donaldson and Stanford T. Shulman

Lymphadenitis as the Dominant Manifestation of Kawasaki Disease

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EX PER IEN CE A N D R EA SO N 525

6 . B ala tsk ii AV , Ru sino vich lu l, R esh etn iko va luK . C ase o f tra um atic he -m o tho rax in a ch ild w ith m ultip le car tila g in ous exos toses. Pediatri ia.197352:82-84

7. D risco ll D J, G lick lich LB , G a len W J. C hes t pa in in children : a pro spec-f iv e s tu dy . Pediatrics. 1976;57 :648-651

8 . F leisch er G R , F ichm an KR , H on ig PJ . H em oth orax in a ch ild . A n un-u sua l cause o f ches t pain . C lin P ed ia tr (P hi la ). 1978;17:300-302

9 . W ang A A , Pa tterson HS , H em s tre et M P , e t al . H em o tho rax in a patien tw ith a sth ma. A nn A lle rg y. 1990;64:55-57

10 . B an adio W A , H ailm ich T . Pos t-traum atic pu lm ona ry con tus ion in ch il-dren . Ann Su ng . 1989;18:1050-1052

11 . B ende r TM , Oh K S, M edin a JL , e t a l. Ped iatric che st t r auma . J ThoracImag i ng . 1987;2 :60-67

12 . L au rin 5 , A ro nson S , Sch uller H , e t a l. S po ntan eo us h em oth orax fromb ro nc ho pu lm on ary se qu estr ation . Unusu al an g io graph ic an d patho-log ic-a nato mic fin din gs . P ed ia tr R ad io l. 1980;10:54-56

13 . Jo hnson D E , M il ler L C , Iverson S , et a l. Th e h eal th of ch ild ren adop tedf rom R om ania . J AMA . 1992;268:3446-3451

14 . K end ig EL Jr. E arly d iag no sis o f tu be rcu losis in ch ildh ood . AJDC . 1956;92:558-561

15. L eung AN , M u ller N L , P in eda PR , e t a l. P rim ary tu berculos is inc hildho od: ra dio gra ph ic m an ifes ta t ion s. Rad i o l ogy . 1992;182:87-91

16 . S ta rke JR . Ch ildh ood tu bercu lo sis du ring th e 19 90 s. P ed ia tr R e v . 1992;13:343-353

17. Solom on L . H ereditary m ul tip le ex os toses . I B one Jo in t Su rg . 1963 ;45 :292-304

18. G a rrison RC , U nni KK , M cLeod RA , et a l. C hondrosarcom a a rising inos teochondroma . Canc e r . 1982;49:1890-1897

19 . Sp onselle r PD , T olo VT . Bone , jo in t a nd m u scle pro b lem s. In : O sk i FA ,DeAng e l i s C D , F eig in R D . e t a l, ed s. P rin cip le s a nd P ra ctice o f P ed ia trics,1 9 9 0 . Ph ila delp h ia : J .B . L ipp inco tt C om pany; 1 99 0

20 . M ad igan R , W orrall T , M cC la in E J . C erv ica l co rd com press ion in he -red i tary m u ltip le ex os toses. R eview of th e litera tu re and rep ort o f aca se . J B on e Jo in t S urg . 1974;56 :401-404

21 . H olvelius L . A neu rysm of pop liteal ar tery c au ses b y a ca rtilag inou sexostos is . O rthop S can d. 1975;46 :836-838

22 . H anrahan PS , Ede lm an J, B rash S . Sp ontaneous hem arthro sis associate dw ith an ex ostosis of the ta lus . J Rheumato l . 1987;14:171

CA SE R EPO R TSW e retrosp ec tive ly rev iew ed th e m ed ica l char ts of I I KD pa-

tie n ts fo r w hom m edical a tten tio n w as sought sp ecific ally be cau seo f m arked cerv ica l lym phadenopathy (T ab le 1). P atien ts w ereid en tified from review of d iv is ion al r eco rds from Janu ary 1983 toD ecem b er 1992 , en com pa ssing approxim a tely 4 50 pat ien ts w ithKD d iagnosed du rin g that per iod . P atien ts ran ged from 2 to 9y ears (m edian = 5 yea rs), and eigh t w ere b oy s. C h ief com p lain tsa t in itia l exam ina tion w ere fever and n eck m ass or to rtico ll is . T hein itia l d iagn os is in 1 0 o f I I p atie n ts w as b acteria l c erv ica l lym ph-aden itis . C e rv ic al m asses rang ed in siz e from 3 X 2 cm to 5 X 7 cmon exam ination an d w e re usual ly describ ed as e ry them ato us , te n-d er to pa lpa tion , bu t no nflu ctu an t. O n adm iss ion , o ne pa tien t h ado nly fev er and lym phad en opa thy ; th e rem a ind er in it ia lly m ani-fe sted tw o o r m ore o ther m ajo r K D diagno stic cr ite ria . E igh t of 11pa tie n ts even tu ally develo ped at le ast four m a jor K D cr iter ia . Th eo the r th ree p atien ts fu lfilled three m ajo r K D c riteria , a nd al l th reedeveloped corona ry abn orm alitie s.

Computed tom ograph y of the n eck w as perfo rm ed in 5 o f 11pa tie n ts b ecause o f co nce rn ab ou t the m agnitu de o f cerv ic al sw e ll-in g (T able 2). F ind ing s included en la rged c erv ica l an d pa raph a-ryng eal lym ph n od es, in cr eased re troph aryng eal spa ce , an d im -pingem ent by nod es o n the o rop harynx and m ajor c erv ical b lo odve sse ls (F ig s I and 2). O n e pat ien t had a low -densi ty reg io n w ith inth e cerv ica l m ass sug ges tin g necro sis or ab sce ss form ation .

F ive of 1 1 pa tie n ts u nde rw ent ne ed le asp ira t ion o f the lym phnode o r re troph aryng eal space becau se o f a su sp ected suppu ra tiveproce ss. In ea ch c ase , flu id w as unobta inab le or th e scan t am oun tob ta ined w as n egative on rou tine b acteria l cu ltur e. In tw o pa -tie n ts , a sp ira tion procedu res w ere per form ed under g ene ral ane s-the sia . P aren te ral an tib io tic s w e re g iven to n ine p atie n ts fo r pre -s ume d bac teria l lym ph ad enitis, w ith ou t b ene fit. T he d iagno sis o fK D w as de lay ed in m o st case s, b ein g estab lish ed a t a m ed ian of 9days (m ax im um = 15 d ays ) af ter the o nse t of fe ver. In trav eno usgamm aglobu lin an d asp irin w ere adm in iste red to n in e pa tien tsw ho responded w ith rap id reso lu tio n o f feve r and o the r s ign s an dsym p tom s, includ ing d ram at ic sub sid en ce o f cerv ica l sw elling .

Lym phad en itis as th e D om inan tM an ifesta tion of K aw asak i D isea se

K aw asak i d ise ase (KD ) is an acu te feb rile illn essp rim a rily affe c ting in fan ts and young ch ild ren . Its im -po rtan ce re lates to the fa c t th at 20% to 25% o f un -tre ated p atien ts deve lo p co ron ary ab no rm a lit ies tha tc an le ad to m yoca rd ia l in farc tio n o r even to death .1K D is a lead ing cau se o f acqu ired h ea rt d isea se inch ild ren in m any reg io ns , inc lu d in g the U n itedS tate s .2 B ecause th ere a re n o sp ec ific d iag nos tic te s tsfo r K D , th e d iag nos is is e s tab lish ed b y th e p re sence o ffeve r an d fou r o f five cri teria w ith ou t o th er ex p lan a-tio n fo r th e illn e ss : (1 ) no nexuda tiv e co n ju nc tiv al in -jec tio n ; (2 ) o ra l m u co sal ch an ge s ; (3 ) chang es o f th ep erip he ral ex trem itie s; (4 ) ra sh , p rim ari ly trun ca l; an d(5 ) ce rv ic al lym phadenopa th y . C erv ica l adeno pa thyis th e le as t co ns tan t d iag no stic c rite rion , p re sen t inapp ro x im a te ly 50% o f p atien ts , w h erea s each o f theo the r fou r c rite ria is fo un d in a lm o st 90% of pa tien tsw ith cla ss ic K D .3 W e d esc ribe 1 1 ch ild ren w ith K Db rough t to m ed ica l a t ten tio n p rim a rily o r so le ly be -cau se o f fev er and strik in g ce rv ic al lym ph ad en o-pa thy .

Received for pu blicat ion Ju n 28 , 1 99 3; a ccep ted O ct 1 7, 1 99 3.PEDIATR ICS (ISSN 0031 4005) . C opyright 1994 by th e Am erican Acad -em y of P ed iatrics .

F ig 1 . P atien t 3 , illn ess day 10 . C om pu ted tom ography scan d em -on strate s num erous m atted left ce rv ica l nodes (arrow s) im ping ingon the hy popha ryn x (H ) , d isp lac ing the ste rno cle idom asto idmusc le (5 ), an d com pre ssing th e in ternal ju gu lar v ein (IJ ) an dcaro tid arte ries (C A ) .

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526 EX PER IEN C E A ND REASON

TABLE 1. C lin ica l F eatu res of K awa sak i D isea se (KD ) P at ien ts W ho H ave S ev ere C erv ica l L ym phad enopathyPatien t A ge , yr Se x In itia l C om p la in t O th er KD C riter ia D iagnos is,

(D a te of D eve lopm ent (D .n )) D ay of Illn es sI 3 F Fever

Painfu l , red , 6 x 4 cm neck m a ss R ash (D .2 )C onjunctiva l in jec tion (D .4 )P er iph era l eryth em a and ed em a (D .4)

4

2 9 F FeverTor t ico l l is , la rg e n eck m ass Conju nct ival in jection (D .3)Per iph era l eryth em a and pain (D .3 )R ed lip s (D .3 ) ; R a sh (D .5 )

5

3 4 M FeverW arm , red 6 x 3 cm n eck m ass

R ash (D .1 0)Conju nc tiva l in ject ion (D .10)P eriphe ral ery th em a (D .1 0)R ed lip s (D .10 )

10

4 5 M Fev erT end er, r ed 7 x 4 n eck m a ss

C onju nc tiva l in ject ion (D .2)P er iph era l eryth em a (0 .2)R ash (0 .2 )

2

5 8 F FeverNeck pain an d swell ingD iffic ulty sw allow ing

R ash (D .3 )P er iph era l ed em a (0 .8)C onju nc tiva l in ject ion (D .8)R ed lip s (0 .8 )

9

6 4 M FeverTor t ico l l is7 x 5 cm tend er neck m a ss

P er iph era l ed em a (D .6)Peel ing rash (D .15 )

15

7 5 M Fev erTor t ico f f isLa rge , p ain fu l n eck m ass

R ash (D .2 )Conjun ctiva l in jec tion (D .6 ) 13

8 6 M Fev e rNeck m a ss

O ra l m uco sal c han ges (0 .6 )R ash (0 .6 )

11

9 2 M FeverTorticoffisNeck m a ss

R ash (0 .8 )O ral m ucosa l ch anges (D .9 )Conju nc tiva l in ject ion (0 .9 )

10

1 0 5 M Fev er3 x 2 cm ten der n eck m ass

R ash (0 .5 )C onju nc tiva l in ject ion (0 .4 )Per iph era l edem a (D .5)

5

11 3 M Fev erT end er, 4 x 4 cm n eck m ass

R ash (0 .5 )C onju nc tiva l in ject ion (0 .4 )P eriphe ral edem a (0 .4)

6

Two pat ien ts d eve lop ed gian t co ronary aneu ry sm s as seq uelae ofKD ; patien t 8 h ad been treated w ith IV GG on illness d ay 11 , andKD w as d iagnosed in pat ien t 7 in 1 983 , befo re th e use of IV GG .Tw o oth er pat ien ts deve lop ed trans ient mild c or on ar y d il at at io ns ;p atien t 6 , in w hom KD w as d iagnosed on illness d ay 15 , w astreated w ith asp ir in a lone , and pa tien t 9 w as g iven IV GG andasp ir in on illn ess day 10 .

D ISCUSS IONA ltho ugh cerv ic al adeno pa thy (a t le a s t 1 .5 cm di-

am eter ) is a m ajo r cr iter io n fo r KD , it is th e lea st com -m on and is p resen t in on ly abou t 50% of case s. A d -eno pa thy is ty p ica lly un ila tera l bu t m ay b e b ila te ra l .Th e nodes a re u sua lly firm and som ewh at tender .They m ay be e ry th em a tou s bu t are non flu ctu an t.Pa tho log ic lym ph node find in gs in K D are fa ir ly non -spec ific and inc lu de fo ca l a re a s o f nec ros is w ith m i-c ro th rom bi in ad ja cen t sm all v esse ls ,4 h yp erp la sia o fT -zon es , a nd m acroph ag e in fil tra tion of B -zones .5

Ou r patients w ere un u sua l in tha t th ey a ll h ad v erym arked lym phad en opa thy , u su a lly in a sso cia tio nw ith e ry th em a and ten de rne ss , and b ecause fev er an dlym phad en op ath y w ere p re sen t sev era l d ays b efo reo the r m an ife s ta t ion s o f K D w ere no ted . E ig h t o f 11p atien ts even tu al ly d ev elop ed a t least fo u r m a jor KDcriteria. A ltho ugh th e rem ain in g th ree fu lfilled on lyth ree c rite ria , each d ev elo ped typ ica l ech ocard io -g rap h ic ev idence o f co ron ary ch ange s . T h ere fo re , thed iagnos i s of KD is rea sonab ly cer ta in in a ll th e se pa -tien ts . C lo ney et a16 rep o rted an au to psy -d iag no sedcase o f KD sim ila r to ou r pa tien ts . B ecause the rap y ino u r pa tien ts w as d ire c ted tow ard b ac te ria l lym phad-

F ig 2 . Pat ien t 6 , illn ess day 15 . C om pu ted tom og raphy scan d em -on strate s very en la rged righ t cerv ica l an d pa rapha ryn geal nodes(ar row s) som ewhat d isp lac ing the la tera l w a ll o f the hypoph arynxand com p re ssin g the in te rna l jugu lar ve in (IJ) . T he retrop haryn -gea l space (RP ) is w id en ed .

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7 . A pr il M M , Bu r n s JC , Newbu rg er JW , et al K awasak i d ise ase c erv icallymphadenopathy . Arch O to lan yng o l H ea d N eck Sun g . 1989;115:512-514

8 . B rio n L , Courto y M , B ache lar t D , et aL M uco cu tan eou s lym ph nod eSyndrome w it h n ec ro ti c pharyngit is. Eu n J Ped i a t n . 1980;135:111-116

9 . K ork is JA , S tillw a ter L B . An unu su al oto la ryn golog ica l prob lem : m u -cocu taneous lym ph nod e syn drom e (K aw asak is syn drom e ) case r epo rt.I Oto l ary ngo l . 1985;14:257-260

10 . K az i A , G au th ier M , Labe l M H , e t aL UVU11 t iS a nd s up ra gl ot ti ti s:ear ly manifesta t ions of K aw asak i d is ease . P ed ia tr In fec t D is I. 1992;120:564-567

3 /8 (37% )8/8 (100% )8/8 ( 1 0 0%)1 /8 (12 .5% )8/ 8 ( 1 0 0%)0/ 80/ 88/8 ( 1 0 0%)1 /8 (12 .5% )1 /8 (12 .5% )1 /8 (12 .5% )0/ 80/ 84 /5 (80% )2/ 2 ( 1 0 0%)

52 8 EX PER IEN CE A ND REA SO N

H ered itary Pan creatitis in th eC h ild ren o f W est V irg in ia

H ered ita ry pancrea titis is th e second m ost com m oncau se o f ch ron ic p ancrea titis in ch ild ren .1 In th is en -tity , p ancrea titis is c lu stered in fam ilies and in heritedth rough ou t th e genera tio n s , in vo lv in g m an y fam ilym em bers . T he d isea se is tran sm itted a s an au to som aldominan t t ra i t wh ich is no t sex -lin k ed an d ha s lim itedp en etran ce .1 U n for tun ate ly , th e e tio log y of th is fam il-ia l d isea se is unknown. The m ost com m on sym p tomin h ered itary pancrea titis is recu rren t a ttack s o f ab -dom i n a l pa in du e to rep ea ted ep isod es o f acu te p an -crea titis , w h ich la ter m ay re su lt in ex ocr in e and /orendoc r ine pancrea tic in su ffic ien cy . In case s in w h ichpancrea tic h is to lo gy h ad been repo rted , no rm al pan -crea tic tissu e w as rep la ced by d iffu se fib ro s is andc hr on ic in fla m ma tio n.2

W est V irg in ia seem s to be an end em ic area fo rh ered itary p an crea titis . P erh ap s d ue to in tra fam ilia lmarr i age , th e g ene p oo l for th is d isea se app aren tlyha s b een presen t in this reg ion fo r g en era tio n s . D a tap ub lish ed from th is a rea in 1 973 id en tified th ree f am -ily ped ig ree s w ith th is d ise ase .3 In th is s tud y , w epres ent ou r ex pe rience w ith fo u r ad d ition al fam ilyp ed igree s and th e ch ild ren w ho have h ered itary p an -creatitis .

M ATER IA LS A ND ME T H O D SP atien t P op ula tio n

E ight ch i ld ren from four d ifferen t fam ilies not p rev iou s ly re-por ted with h ered ita ry pancre ati tis w ere fo l low ed up in ou r pe -d iatr ic ga stro in test ina l c lin ic in H unt ing ton , W V . T he ir ch artsw ere rev iew ed retrospective ly for the ir c lin ica l fin d in gs , lab ora-to ry resu lts , and treatm en t.

L ab or ato ry In ve stig atio nP an cre atic fun ction w as eva lu ated by th e s ecretin stim ulation

tes t ( fiv e p atien ts) as described by M adrazo-de la Garza et a l4 or byt he b en ti ro m id e te st5 if en do sco py w as refu sed (tw o patien ts). On epatien t refused bo th tests . F our-h ou r g lu cose to lerance tes ts w erep erfo rm ed in all pat ien ts. D iabe tes ins ip id us w as d iagn osed by th ew ater d epr ivation tes t.

RESULTSThe fou r fam ily p ed igrees rep re sen t a to ta l o f 46

a ffec ted m em b ers , o f w hom sev en had pan crea tic su r -g ery (F ig s 1 and 2 ). F iv e m a le and th ree fem a le ch il-Received f or p u b li ca ti on Ju n 24 , 1993; accepted Se p 15 , 1993 .PED IATR IcS (ISSN 0031 4005). Copyrigh t C 1994 by the Am erican A cad -em y of Pediatr ics.

dren , a ged 2 to 1 8 y ear s (m ed ian 8 .5 year s), a re ro -po r ted in th is s tudy . T h e m ost com m on m an ifesta tionw as recu rren t a tta cks o f acu te panc reati tis , fo llow edby ch ro n ic d iarrh ea . L ab ora to ry d a ta sh ow ed no rm a lserum e le ctro ly te , ca lc ium , ch o le stero l , a nd liv er en -zym e leve ls (asp arta te am in o tran sfe ra se , a lan in e am i-notransferase). The sign ifican t c lin ica l sym p tom s andlabo rato ry in ve s tiga tio ns a re summ arized in th eTab l e . An assoc ia tion o f h ered ita ry pancrea titis w ithd iab e tes rn s ip id u s w a s foun d in on e ch ild a t a ge 7(F ig 1 ).

D I SCUSS IONH ered ita ry pan crea titis (H P ) is a ch ron ic d isease

w h ich u ltim a te ly lead s to pancrea tic insuffic iency.The m o st com m on m an ifesta tion is rep ea ted attack so f acu te pan crea titis . A ll o u r ped ia tr ic pa tien ts h av em an ife s ted th e d isea se in th is m an ner, r esu ltin g inm u ltip le adm iss ion s. F iv e o f sev en p atien ts had c lin i-ca l sym p tom s o f m a lab sorp tio n and ha d abno rma lpancrea tic s tim u la tio n te sts. P ancrea tic insuffic iencyw as d iagn osed in tw o patien ts b e fore th e a ge o f 3y ea rs . A ll five pa tien ts re sp on ded to p ancrea tic en -zyme s upp lemen ta t ion .

Pancrea tic p seud ocysts o ccu r com m on ly in H P ,w ith an estim ated in cid en ce in u p to 5 0% of p atien ts .6In o u r serie s , on ly o ne ch ild (fam ily H , F ig 1 ) had th re epancrea tic p seud ocysts w h ich h ad regressed spon ta -n eou sly . T he estim a ted ra te o f regress io n of p seud o-cys ts of a ll etio lo g ie s is 20% .

Exc lud ing o ur e igh t p atien ts , th e p ed ig ree s rep ro -sen t a to ta l o f 3 8 m em bers w ho h ad hered ita ry pan -c rea titis . S ev en pa tien ts (F ig s 1 and 2) h ad a p ar tia l orto ta l p an crea tec tom y , o r o th er pancrea tic d ra ina geprocedu re . D iab ete s mell i tus and recurren t ab dom i-na l p a in w ere d iagnosed in severa l o th er m em bers.T he ou tcom es o f p rev iou s g en era tion s sugg est tha tch ild ren w ith H P w ifi b e p ron e to s ign ifican t futurem orb id ity d ue to pan crea tic ou tle t ob stru ctionan d /o r p seud ocyst form a tion tha t m ay requ i re sur -ge ry . U nfo rtuna te ly , endo c rine /exo crine pancrea t i cinsuffic iency and p an crea tic m alignancy (estim a ted5% ) is th e lo ng-te rm course o f th e d isea se .1

T he m olecu la r d e fect in H P is as y et unknown . D e

TABLE . Clin ical and L abo ratory D ataClin iCal s ymp toms

D iar rhe a, fou l sto o lA bdom in al pa in + recu rre nt p anc rea titisG row th (> 5% tile)Po lyuria

Laborato ry d ataE lev ate d am ylase /lip aseHyp e r c a l c em iaHyper l ip idemiaAbdomin a l ultrasound

EdemaDilated duc tCystsCalcu l i

Pancreat ic fu nc tion tes tA bno rm al g lucose to leran ce test (4 h)Abno rma l secretin testA bnorm al ben tirom ide test

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1994;93;525PediatricsJulie Kim Stamos, Kathleen Corydon, James Donaldson and Stanford T. Shulman

Lymphadenitis as the Dominant Manifestation of Kawasaki Disease

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Online ISSN: 1098-4275.Copyright 1994 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005.American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, 60007.has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by thePEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it

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Pediatrics 1994 Stamos 525 8

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