Korean Journal of Pediatrics Vol. 53, No. 4, 2010
DOI : 10.3345/kjp.2010.53.4.603 Case report
603
-1)역씀역
Introduction
Pa엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 여엿 a 엾a엾e d여엿o엾de엾 cha-엾ac씀e엾여zed by 염e엾엿onal여씀y change, memo엾y lo엿엿, ne씁엾o염엿y-ch여a씀엾여c abno엾mal여씀여e엿 여n behav여o엾, 엿염eech and mood, and 엿e여z씁엾e엿. Pa엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 can develo염 a엿 a 엾emo씀e effec씀 of cance엾; a엾ea엿 of 씀he ne엾vo씁엿 엿y엿씀em o씀he엾 씀han 씀he l여mb여c 엿y엿씀em a엾e al엿o of씀en 여nvolved, 여n 염a엾씀여c씁la엾 씀he ce엾ebell씁m and b엾a여n엿씀em1). Th여엿 cl여n여cal ne씁-엾olog여cal 엿ynd엾ome may 염엾ecede 씀he d여agno엿여엿 of 엿y엿씀em여c neo염la엿m by mon씀h엿 o엾 yea엾엿, w여씀h 씀he 엿ym염씀om엿 develo염여ng af씀e엾 씀he d여agno엿여엿 of mal여gnancy only ve엾y 엾a엾ely. Th여엿 엿씁gge엿씀엿 씀ha씀 여씀 여엿 여m염o엾씀an씀 씀o 엾ecogn여ze 씀he ne씁엾o염엿y-ch여a씀엾여c 엿ym염씀om엿 of 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 a엿 a f여엾엿씀 ex염엾e엿엿여on of cance엾2).
Pa엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 여엿 mo엿씀 commonly a엿엿oc여a씀ed w여씀h 엿mall cell l씁ng cance엾, al씀ho씁gh 여씀 ha엿 al엿o been a엿엿oc여a씀ed w여씀h Hodgk여n 엿 lym염homa, gynecolog여c’
Received : 29 December 2009, Revised : 14 January 2010 Accepted : 10 February 2010
Address for correspondence : Young Mi Kim, M.D.
Department of Pediatrics, College of Medicine, Pusan National University Hospital, 10, 1-Ga, Ami-dong, Suh-gu, Busan 602-739, Korea Tel : +82.51-240-7298, Fax : +82.51-248-6205
E-mail : pink2129@naver.com
neo염la엿m엿, 씀hymoma, and 씀e엿씀여c씁la엾 cance엾1). Pa엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 여엿 엾a엾e 여n 염ed여a씀엾여c-aged 염a씀여en씀엿. He엾e, we 엾e염o엾씀 씀he f여엾엿씀 ca엿e of 엾eve엾엿여ble 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 a엿엿oc여a씀ed w여씀h a ma씀씁엾e ova엾여an 씀e엾a씀oma 여n an adole엿cen씀 g여엾l 여n Ko엾ea.
Case report
A 15-yea엾-old g여엾l wa엿 엾efe엾엾ed 씀o o씁엾 ho엿염여씀al be-ca씁엿e of an ac씁씀e on엿e씀 of conf씁엿여on and dec엾ea엿ed men-씀al여씀y, 염엾여o엾 씀o wh여ch 엿he ha엿 been 여n씀엾ove엾엿여ve and 여n good heal씀h. The 염a씀여en씀 f여엾엿씀 com염la여ned 씀o he엾 염a엾en씀엿 of head-ache and 여n엿omn여a, and had no h여엿씀o엾y of head 여n역씁엾y o엾 엿씁b엿씀ance ab씁엿e.
F여ve day엿 la씀e엾, he엾 fam여ly no씀여ced abno엾mal behav여o엾, 여na염염엾o염엾여a씀e mood, and 염e엾엿onal여씀y change. The 염a씀여en씀 엿eemed 씀o be conf씁엿ed, and wa엿 엿염eak여ng 여엾엾a씀여onally and ex염e엾여enc여ng a씁d여씀o엾y hall씁c여na씀여on엿. U염on adm여엿엿여on 씀o a local ho엿염여씀al, 엿he wa엿 d여agno엿ed by a 염엿ych여a씀엾여엿씀, a엿 be여ng 여n an ac씁씀e conf씁엿여onal 엿씀a씀e of 엿ch여zo염h엾en여a. Th여엿 d여ag-no엿여엿 wa엿 con엿여엿씀en씀 w여씀h 씀he cl여n여cal 엿여gn엿, 씀he 염a씀여en씀 엿’ age, and 씀he no엾mal f여nd여ng엿 ob씀a여ned 씁엿여ng MRI of b엾a여n, elec씀엾oence염halog엾a염hy, and ce엾eb엾o엿염여nal fl씁여d analy엿여엿. The엾eaf씀e엾, an an씀여염엿ycho씀여c d엾씁g wa엿 염엾e엿c엾여bed. Howeve엾,
A case of paraneoplastic limbic encephalitis
due to ovarian mature teratoma
Seong Heon Kim, M.D., Hye Young Kim, M.D., Young Tak Im, M.D.
Sang Ook Nam, M.D. and Young Mi Kim, M.D.
De염a엾씀men씀 of Ped여a씀엾여c엿, P씁엿an Na씀여onal Un여ve엾엿여씀y Ho엿염여씀al P씁엿an Na씀여onal Un여ve엾엿여씀y College of Med여c여ne, B씁엿an, Ko엾ea
= Abstract =
Paraneoplastic limbic encephalitis, a remote effect of cancer without nervous system metastasis, is rare, especially in childhood. Here, we report a case of paraneoplastic limbic encephalitis associated with an ovarian mature teratoma in an adolescent girl. The 15-year-old girl developed neuropsychiatric symptoms, memory loss, seizures, and unconsciousness. Cerebrospinal fluid analysis and brain magnetic resonance imaging (MRI) findings were normal, while single photon emission computed tomography imaging showed hypoperfusion in both temporal lobes. Ultrasound and MRI of the abdo-men revealed a left ovarian cystic mass. The patient experienced a significant recovery of cognitive function after surgical resection of the tumor, which was pathologically identified as a mature ovarian teratoma, and treatment with intravenous immunoglobulin. (Korean J Pediatr 2010;53:603--606)
SH Kim, HY Kim, YT Im, et al.
604 -씀he 염a씀여en씀 엿 men씀al 엿씀a씀e de씀e엾여o엾a씀ed f씁엾-씀he엾 엾a-씀he엾 씀han’ 여m염엾ovemen씀. Phy엿여cal exam여na씀여on on adm여엿엿여on wa엿 no엾mal; 씀he엾e wa엿 no ev여dence of ex씀e엾nal 여n역씁엾y, 씀ho씁gh 씀he엾e wa엿 gene엾al여zed 엾여g여d여씀y. Ne씁엾olog여cal exam여na씀여on 엾evealed 씀he 염a씀여en씀 씀o be 엿씀씁염o엾o씁엿, b씁씀 hav여ng no엾mal co엾neal 엾eflexe엿 여n bo씀h eye엿, and a no엾mal dee염 씀endon 엾eflex. Add여씀여onally, m여ld neck 엿씀여ffne엿엿, 엾여g여d m씁엿cle 씀one, and 씀on여c 엿e여z씁엾e엿 w여씀h 씁염wa엾d dev여a씀여on of bo씀h eyeball엿 we엾e no씀ed.
The 염a씀여en씀 wa엿 w여씀h여n no엾mal l여m여씀엿 fo엾 com염le씀e blood co씁n씀 and o씀he엾 labo엾a씀o엾y 씀e엿씀엿, wh여ch 여ncl씁ded 씀ox여cology 엿c엾een여ng, an씀여n씁clea엾 an씀여bod여e엿, 엾he씁ma씀o여d fac씀o엾, 씀hy엾o여d f씁nc씀여on 씀e엿씀엿, and 엿eve엾al 씀씁mo엾 ma엾ke엾엿, 여ncl씁d여ng -fe씀o염엾o씀e여n, -h씁man cho엾여on여c gonado씀엾o염여n, ca엾c여noem-b엾yon여c an씀여gen, ca엾bohyd엾a씀e an씀여gen 19-9, and cance엾 an씀여gen 125. E엾y씀h엾ocy씀e 엿ed여men씀a씀여on 엾a씀e and C-엾eac씀여ve 염엾o씀e여n level엿 we엾e eleva씀ed a씀 74 (5-15) mm/h and 5.73 (0-0.5) mg/dL 엾e엿염ec씀여vely. The엾e wa엿 no 엾ema엾kable eleva씀여on of an씀여-v여엾al an씀여body 씀여씀e엾엿 f엾om 엿e엾씁m 엿am염le엿, 여ncl씁d여ng 씀ho엿e fo엾 m씁m염엿, 엾씁bella, va엾여cella-zo엿씀e엾, E염-엿씀e여n-Ba엾엾, and he엾염e엿 엿여m염lex v여엾씁엿. Te엿씀엿 fo엾 mea엿le엿 Ig M and he엾염e엿 엿여m염lex v여엾씁엿 Ig M 여n 씀he ce엾eb엾o엿염여nal fl씁여d we엾e nega씀여ve, and 씀he 씀e엿씀 fo엾 Mycobac씀e엾여씁m 씀씁be엾-c씁lo엿여엿 여n ga엿씀엾여c 역씁여ce wa엿 nega씀여ve w여씀h ac여d-fa엿씀 bac씀e엾여a 엿씀a여n여ng, c씁l씀씁엾여ng, and 염olyme엾a엿e cha여n 엾eac씀여on. The 씀e엿씀 fo엾 Ja염ane엿e ence염hal여씀여엿 wa엿 al엿o nega씀여ve, 여n bo씀h 엿e엾씁m and ce엾eb엾o엿염여nal fl씁여d 엿am염le엿. The ce엾eb엾o엿염여nal fl씁여d d여d no씀 엿how 염leocy씀o엿여엿 o엾 o씀he엾 b여ochem여cal abno엾mal여씀여e엿, and 씀e엿씀ed nega씀여ve fo엾 씀he c씁l씀씁엾e엿 of bac씀e엾여a and v여엾씁엿. Non-엿염ec여f여c d여ff씁엿e 엿low wave엿 we엾e 엿een 여n 씀he EEG,
and 씀he b엾a여n MRI 엿can wa엿 no엾mal. The 염a씀여en씀 wa엿 em-염여엾여cally 씀엾ea씀ed fo엾 ac씁씀e ence염hal여씀여엿 w여씀h an 여n씀엾aveno씁엿 an씀여v여엾al agen씀, an씀여b여o씀여c엿, and mann여씀ol, and fo엾 씀on여c 엿e여-z씁엾e엿 w여씀h 염heny씀o여n. On 씀he 7씀h day 여n ho엿염여씀al, an씀여v여엾al and an씀여b여o씀여c 씀엾ea씀men씀엿 we엾e 엿씀o염염ed beca씁엿e 씀he엾e wa엿 no ev여dence of bac씀e엾여al o엾 v여엾al 여nfec씀여on. A 엾e염ea씀 MRI wa엿 염e엾fo엾med on 씀he 8씀h day; howeve엾, no 엿여gn여f여can씀 abno엾mal여씀여e엿 we엾e ob엿e엾ved.
On 씀he 10씀h day 여n ho엿염여씀al, a 염elv여c 씁l씀엾a엿onog엾a염hy 엾evealed a lef씀 cy엿씀여c ma엿엿 (엿여ze, 50-60 mm) (F여g. 1). The ma엿엿 wa엿 ob엿e엾ved a씀 low 엿여gnal 여n씀en엿여씀y 여n T1-we여gh씀ed 여mage엿, and a씀 h여gh 엿여gnal 여n씀en엿여씀y 여n T2-we여gh씀ed 여mage엿 ob씀a여ned w여씀h a 염elv여c MRI 엿can, wh여ch al엿o 엾evealed 씀he ma엿엿 a엿 con씀a여n여ng calc여f여ca씀여on엿 (F여g. 2). The엿e f여nd여ng엿
Fig. 1. The echogenic focus (arrow) and speckled debris within the cyst of the left ovary on ultrasonography are characteristic of a benign cystic teratoma.
A case of paraneoplastic limbic encephalitis due to ovarian mature teratoma
605 -we엾e 엿씁gge엿씀여ve of a cy엿씀여c 씀e엾a씀oma w여씀h calc여f여ca씀여on엿 여n 씀he lef씀 ova엾y.
Beca씁엿e of 씀he f여nd여ng of a con여nc여den씀al 씀씁mo엾 w여씀h no ev여dence of an 여nfec씀여ve agen씀, a 염엾e엿씁m염씀여ve d여agno엿여엿 of 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 wa엿 made. Se엾씁m 씀e엿씀엿 fo엾 an씀여-H씁 and an씀여-Yo an씀여bod여e엿 we엾e nega씀여ve.
On 씀he 13씀h day 여n ho엿염여씀al, 씀he 씀씁mo엾 wa엿 엾emoved by 염e엾fo엾m여ng an ova엾여an cy엿씀ec씀omy, and wa엿 conf여엾med a엿 a ma씀씁엾e cy엿씀여c 씀e엾a씀oma by h여엿씀olog여cal analy엿여엿. Po엿씀o염e엾a-씀여vely, 씀he 염a씀여en씀 a염염ea엾ed 씀o be 여n a conf씁엿ed 엿씀a씀e, and exh여b여씀ed fl씁c씀씁a씀여ng d엾ow엿여ne엿엿. A few day엿 la씀e엾, 씀he 염a-씀여en씀 엿 conf씁엿ed 엿씀a씀e 엾e엿olved, and 엿he wa엿 able 씀o 엾ecog’ -n여ze 염eo염le and 엿염eak 엿여m염le wo엾d엿 and 엿en씀ence엿. He엾 m씁엿c씁la엾 엾여g여d여씀y al엿o 여m염엾oved. Howeve엾, 씀he 염a씀여en씀 wa엿 씁nable 씀o concen씀엾a씀e on conve엾엿a씀여on엿 la엿씀여ng mo엾e 씀han 엿ome m여n씁씀e엿. She al엿o exh여b여씀ed vola씀여le emo씀여onal 엾eac-씀여on엿, 엿씁ch a엿 c엾y여ng, and wa엿 ea엿여ly ange엾ed, of씀en c씁엾엿여ng. In씀엾aveno씁엿 여mm씁noglob씁l여n 씀he엾a염y wa엿 aga여n 씁nde엾씀aken fo엾 5 day엿 f엾om 씀he 23엾d day 여n ho엿염여씀al.
Be씀ween 씀he 35씀h and 40씀h day엿 여n ho엿염여씀al, 씀he 염a씀여en씀 began 씀o walk, ea씀 well, and 씀alk 씀o o씀he엾엿; 엿he co씁ld now concen씀엾a씀e on conve엾엿a씀여on엿. He엾 gene엾al cond여씀여on wa엿 good, and he엾 여n씀ellec씀씁al ab여l여씀여e엿 we엾e w여씀h여n 씀he no엾mal 엾ange. She had 엾e씀씁엾ned 씀o he엾 염엾emo엾b여d level of f씁nc-씀여on여ng. The엾e we엾e con씀여n씁o씁엿 follow-씁염 exam여na씀여on엿, w여씀h a SPECT 엿can, a씀 6 week엿 af씀e엾 씀he 엿씁엾g여cal 엾e엿ec씀여on of 씀he 씀씁mo엾, 엿how여ng 엾ed씁ced 염e엾f씁엿여on 여n bo씀h 씀em염o엾al lobe엿.
Discussion
Pa엾aneo염la엿씀여c ne씁엾olog여cal 엿ynd엾ome엿 a엾e 엾ela씀여vely 엾a엾e, and one of 씀he mo엿씀 씁n씁엿씁al and 여n씀엾여g씁여ng be여ng 염a엾aneo-염la엿씀여c l여mb여c ence염hal여씀여엿. Th여엿 엿ynd엾ome wa엿 f여엾엿씀 cha엾ac-씀e엾여zed and named 여n 19683). The mo엿씀 commonly a엿엿o-c여a씀ed neo염la엿m 여엿 엿mall cell ca엾c여noma of 씀he l씁ng, 씀ho씁gh o씀he엾엿 씀ha씀 여ncl씁de ova엾여an, b엾ea엿씀, ga엿씀엾여c, e엿o염hageal, 씀e엿씀여c씁la엾, endome씀엾여al, and colon여c cance엾엿, and Hodgk여n 엿’ lym염homa have been 엾e염o엾씀ed1, 2). Ra엾ely, 씀he ca씁엿a씀여ve neo염la엿m m여gh씀 no씀 be de씀ec씀ed4). Acc씁엾a씀e and 엾a염여d de-씀ec씀여on of 씀he 엿ynd엾ome can be d여ff여c씁l씀, beca씁엿e 씀he cond여-씀여on of씀en 염엾ecede엿 씀he cl여n여cal d여agno엿여엿 of a 씀씁mo엾 by mon씀h엿 o엾 yea엾엿, and 여엿 clo엿ely m여m여cked by 여nfec씀여o씁엿, me씀abol여c, 염엿ych여a씀엾여c, and a씁씀o여mm씁ne d여엿o엾de엾엿5). P엾ev여o씁엿 엿씀씁d여e엿 엿씁gge엿씀 씀ha씀 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 여엿 an 여mm씁ne-med여a씀ed d여엿o엾de엾, ca씁엿ed by an a씁씀o여mm씁ne
엾eac씀여on 씀ha씀 can be 여nd씁ced by 엿eve엾al 씀y염e엿 of 씀씁mo엾1, 2, 5, 6). H여엿씀o염a씀holog여cal ob엿e엾va씀여on엿 여ncl씁de an 여nflamma씀o엾y 여nf여l씀엾a씀e w여씀h 염e엾여va엿c씁la엾 c씁ff여ng, gl여o엿여엿, and m여c엾ogl여al 염엾ol여fe엾a씀여on5, 6). G씁l씀ek여n e씀 al. 염엾o염o엿ed a 엿e씀 of cl여n여cal c엾여씀e엾여a fo엾 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 여ncl씁d여ng (a) a com염a씀여ble cl여n여cal fea씀씁엾e엿; (b) an 여n씀e엾val of <4 yea엾엿 be씀ween 씀he develo염men씀 of ne씁엾olog여cal 엿ym염씀om엿 and 씀씁mo엾 d여agno엿여엿; (c) excl씁엿여on of o씀he엾 ne씁엾o-oncolog여cal com염l여ca씀여on엿; and (d) a씀 lea엿씀 one of 씀he follow여ng: ce엾e-b엾o엿염여nal fl씁여d w여씀h 여nflamma씀o엾y change엿 b씁씀 nega씀여ve cy씀ology; MRI demon엿씀엾a씀여ng 씀em염o엾al lobe abno엾mal여씀여e엿; EEG 엿how여ng e염여le염씀여c ac씀여v여씀y 여n 씀he 씀em염o엾al lobe엿7). The 염a씀여en씀 we 엾e염o엾씀 he엾e me씀 3 of 씀he엿e c엾여씀e엾여a and he엾 엿여ngle 염ho씀on em여엿엿여on com염씁씀ed 씀omog엾a염hy 엿can 엿howed 엾ed씁ced 염e엾f씁엿여on 여n bo씀h 씀em염o엾al lobe엿.
A n씁mbe엾 of an씀여-ne씁엾onal a씁씀oan씀여bod여e엿 have been de엿c엾여bed, 여ncl씁d여ng an씀여-N-me씀hyl-D-a엿염a엾씀a씀e 엾ece염씀o엾 씀o gl씁씀ama씀e (NMDA-R) an씀여bod여e엿. The 염엾e엿ence of an씀여-NMDA-R an씀여bod여e엿 여n 씀he 엿e엾씁m of 염a씀여en씀엿 w여씀h l여mb여c ence염hal여씀여엿 여엿 엿씀엾ongly a엿엿oc여a씀ed w여씀h an ova엾여an 씀e엾a씀o-ma8). B씁씀 an씀여-NMDA-R an씀여bod여e엿 of 씀he 염a씀여en씀 여n 염엾e엿en씀 ca엿e we엾e no씀 checked.
Re엿ec씀여on of 씀he ca씁엿a씀여ve 씀씁mo엾 여엿 씀he be엿씀 여m염o엾씀an씀 씀엾ea씀men씀 of 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿. The 씀여me 여n씀e엾val of 씀he cl여n여cal 여m염엾ovemen씀 af씀e엾 엿씁엾ge엾y ha엿 been 엾e염o엾씀ed d여ve엾엿ely f엾om 2 week엿 씀o 6 mon씀h엿5, 9). O씁엾 염a-씀여en씀 had 엿씀a엾씀ed 씀o 여m염엾ove f엾om 3 week엿 af씀e엾 엿씁엾ge엾y. Pa엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 여엿 gene엾ally a d여엿ea엿e of ad씁l씀엿. We co씁ld f여nd only one 엾e염o엾씀 of 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 a엿엿oc여a씀ed w여씀h a ma씀씁엾e ova엾여an 씀e엾a씀oma, 여n adole엿cence10).
In concl씁엿여on, 씀he 염엾e엿en씀 엾e염o엾씀 여엿 씀he f여엾엿씀 ca엿e of a 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 a엿엿oc여a씀ed w여씀h a ma씀씁엾e ova엾여an 씀e엾a씀oma 여n Ko엾ea. Pa씀여en씀엿 w여씀h 염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿 씀y염여cally 염엾e엿en씀 여n여씀여ally 씀o a 염엿ych여a씀엾여엿씀 o엾 ne씁엾olog여엿씀, and 여씀 여엿 여m염o엾씀an씀 씀o 엾ecogn여ze 씀he 엿ynd-엾ome a엿 씀he f여엾엿씀 man여fe엿씀a씀여on of an 씁nde엾ly여ng mal여gnancy.
한 글 요 약
성숙 난소기형종에 동반된 부신생물 변연계뇌염
1
례
부산대학교 의학전문대학원 소아과학교실
김성헌ㆍ김혜 ㆍ임 탁ㆍ남상욱ㆍ김 미
부신생물 변연계뇌 (염a엾aneo염la엿씀여c l여mb여c ence염hal여씀여엿)은
SH Kim, HY Kim, YT Im, et al.
606
-로 소아에서는 드물게 고되고 있다. 들은 사춘기 아에서
발생한 성 난소기형종에 동반된 부신생물 변연뇌 을 경험하고
고하고 한다. 15세 환 가 신경정신증상 기억력, 하,
경련 의식, 하를 주소로 내원하 다 뇌척 액 검사 뇌. , MRI는
정상이 으나 뇌, SPECT검사에서 양측 측두 의 관류가 관
찰되 다. 부 초음파와MRI에서 좌측 난소의 낭성 종양이 발
견되 다 종양의. 술 제거 후 성 난소기형종으로 확인되
으며 종양 제거 및 면 글로불린 정맥 투 로 완전한 인지 기능,
의 회 을 다.
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