雙側隔離肺合併肺過度充氣

(1)

2008 19 527-530

300 678

ၡāāࢋ

27 X

ᙯᔣෟĈ࿣ᗓ۱ ( Pulmonary sequestration ) ளҜજਔ ( Aberrant artery )

࿪ཝᕝᆸ ( Computed tomography )

݈֏

"࿣ᗓ۱" ߏ۱ొд൴ֈ࿅඀̚யϠ۞˘჌͌

֍۞Аّ͇ள૱ĂЯࠎι֭՟ѣᓑᘭזϒ૱۞͚

ঈგĂ҃ͷι۞ֻҕֽ໚ߏវೈᒖ۞ր௚જਔĂ ᐖਔа߹఼૱౅࿅۱ᐖਔٕ۰ր௚ᐖਔĂٙͽߏ

˘๴൑Αਕّ۞۱௡ᖐ

¹

Ą࿣ᗓ۱ள૱གྷ૱൴Ϡ дಏ઎˭۱ཧૄغ۞ҜཉĂ൴Ϡдᗕ઎۞࿣ᗓ۱ ՀࠎංѣĂፂԧࣇٙۢĂΪѣ͌ᇴ࣎९ѣԆፋ۞

͛ᚥಡӘ

²

Ąд఺ቔ͛ౢ྆ԧࣇಡӘ˘ҜЯཛొ

়ঽಶᗁĂ҃ຍγ൴ன൑ײӛ྽ᓜԖাې۞ᗕ઎

࿣ᗓ۱Ą

ঽּಡӘ

˘Ҝ 2 7 ໐۞շّĂ൑٩ඵ௫ၚĂϤٺཛొ

ূ൭זާ෧ಶᗁĂ՟ѣЇңײӛ྽াېĄঽଈజ ෧ᕝࠎާّᔰԍۆЪ׀ᓘႹĂ֭ͷјΑ۞߉Җ˞

ღާ۞γࡊ͘ఙĄ༊ॡ਒ొ X Ѝຍγ۞൴னдν

˭۱ཧă͕᝙ޢ͞ѣள૱۞ҕგᇆည ( ဦ˘ )Ą நጯᑭߤॡ൴னᗕ઎˭۱ཧ۞ײӛࢰځពഴ͌Ą дڦडពᇆ጗۞ཛొ࿪ཝᕝᆸବೡ̚൴னᚑࢦᔰ ԍۆЪ׀ࡍ͋Ă֭ͷТॡ൴னᗕ઎˭۱ཧѣଂ਒

ొࢫ͹જਔ̶͚΍۞ளҜજਔͽ̈́ొ̶˭۱ཧ࿅

ޘ·ঈᙷҬ۱ঈཚត̼ ( ဦ˟ )Ąдࢦޙ۞ˬჯ ( 3 dimensional reconstruction ) ࿪ཝᕝᆸବೡ̚ቁ ᄮ˞ࣣрଂፖჽ˯ࢬĂ਒ొࢫ͹જਔѣ׌୧ளҜ જਔ̶͚זν˭۱ćѣ˘୧ளҜજਔ̶͚זΠ˭

۱ ( ဦˬ )ĄঽଈдТ˘͇јΑତצ˞ღާ۞ᔰ ԍ̷ੵఙ֭ͷ՟ѣЇң׀൴াயϠĄ

ঽଈ۞۱Αਕពϯϡ˧۱߿ณ FVC Ĉ 3.91 L(84% ࿰ഇࣃ) ăௐ˘ࡋϡ˧Фঈณ FEV1 Ĉ 3.08 L (78% ࿰ഇࣃ)ă FEV1/FVC ͧதĈ 78.85% ă۱ ዶट᎕ RV / ۱ᓁटณ TLC Ĉ 31.34% (127% ࿰ഇ ࣃ )Ą۱Αਕඕڍࠎѝഇܡ๫ݭೱঈᅪᘣĂ௑Ъ

࿪ཝᕝᆸ̚۱ཧ࿅ޘ·ঈ۞ត̼Ą

(2)

఺჌۱௡ᖐ۞ள૱གྷ૱ົጱ࡭ͅᖬ۞ຏߖͽ

̈́̂ณݜҕĂ֭ͷฟ˥̷ੵள૱۞۱௡ᖐߏᇾ໤

۞ڼᒚ͞ёĄҭߏঽଈଂֽౌ՟ѣЇңײӛ྽া

ېĂЯѩঽଈ١඗ซ˘Վ۞ᑭߤٕ۰ฟ˥ڼᒚĂ

ٙͽΪਕޙᛉঽଈؠഇܝ෧੠ᖸĄ

੅ኢ

࿣ᗓ۱˘ਠజ̶ј۱ཧγݭ ( extralobar lung sequestration ) ͽ̈́۱ཧ̰ݭ ( intralobar lung se- questration ) ˟჌ݭၗĄ

۱ཧγ࿣ᗓ۱ጾѣιҋ̎Ԇፋ۞᝙ᆸ҈ቯĂ

֭ͷజᄮࠎߏ˘࣎ܢΐ۞۱Ąιགྷ૱дᑈ׊ॡഇ ಶజ൴னĂЯࠎк྿Ѻ̶̝̣˩۞۱ཧγ࿣ᗓ۱ ᄃ׎΁А͇۞ள૱ѣᙯĂΒ߁ፖ࿣ቯ۞৿ౝĂᗗ

਒Ă۱ள૱׶Аّ͇͕᝙ঽĄ෹࿅ 9 0 % ۞۱ཧ γ࿣ᗓ۱Ҝдν઎˭۱ཧ̝ޢغ༼Ăͷр൴ٺշ

ّĄֻҕ఼૱ଂ͹જਔ̶͚΍ளҜજਔĂ҃ᐖਔ а߹ߏགྷϤ؈ᐖਔăΗ؈ᐖਔăܝᐖਔٕߏටᐖ ਔ

³

Ą

۱ཧ̰࿣ᗓ۱ߏ׶ϒ૱۱௡ᖐ˘੓జΒд᝙

ᆸ҈ቯ̰Ă׏ݭ۞ܑனߏҜٺν˭۱ཧ۞ତܕፖ

࿣۞ొҜĂ఼૱дјѐॡ̖΍னᓜԖাې҃జ൴ ன

¹

Ąдԧࣇঽˠ۞࿪ཝᕝᆸ֭̚՟ѣ᝙ᆸ҈ቯ

۞̶ࠧĂЯѩᔵ൒՟ѣγࡊঽந̷ੵᙋ၁ҭ̪Ξ

ࠧؠࠎ൑াې۞ᗕ઎۱ཧ̰࿣ᗓ۱Ą̂ࡗ 7 4 %

۞۱ཧ̰࿣ᗓ۱۞જਔֻᑕ੓ٺ਒ొࢫ͹જਔ۞

ள૱̶͚҃ͷԆБ՟ѣ͚ঈგજਔֻගĂ͹ࢋᐖ

528

ဦ˘Ĉν˭۱ཧѣள૱۞ҕგᇆညĄ

ဦ˟Ĉᗕ઎˭۱ཧѣଂ਒͹જਔ̶͚΍۞ள૱જਔͽ̈́

࿣ᗓ۱࿅ޘ·ঈĄ

ဦˬĈࢦޙ۞ 3D ࿪ཝᕝᆸବೡĈ਒ొࢫ͹જਔѣ׌୧ ր௚જਔ̶͚זν˭۱ ( ˬ֎ݭٙ޽) ć˘୧ր

௚જਔ̶͚זΠ˭۱ (቏ᐝٙ޽)Ą

(3)

ਔа߹ߏགྷϤ۱ᐖਔĄࡗ 1 6 % ۞ঽଈົТॡ΍

னк୧જਔ̶͚

³

Ąಶညԧࣇ۞ঽˠВѣˬ୧ள Ҝજਔ̶ֻ͚ᑕĄࡶঽˠଈѣ۱ཧ̰࿣ᗓ۱҃Т ॡ൴ன׎΁Аّ͇ள૱ߏܧ૱͌֍۞Ą

ᗕ઎࠰൴Ϡ࿣ᗓ۱۞ঽּߏܧ૱ց֍۞Ąϫ

݈д͛ᚥ˯Ϊѣ͌ᇴ࣎९జಡӘ࿅

^{2 , 3}

Ą۱ཧ̰࿣

ᗓ۱౵૱֍۞ள૱ߏಏ˘ٕкᝃཚ۞хдĂ׎̚

Βӣ˞ඕ቙௡ᖐĂ൴ۆ௟ࡪ׶۱ڽ௡ᖐĄॲፂঽ நጯᑭߤពϯѣ 9 1 % ࿣ᗓ۱۞۱ڽ௡ᖐ൴ֈ̙

БĂ҃ೀͼٙѣ۞࿣ᗓ۱࠰ົ൴Ϡ͚ঈგౕᗆ

⁴

Ą

ࡶঽଈ൴Ϡͅᖬ۞۱ొຏߖॡĂ݋࿣ᗓ۱ᄃϒ૱

͚ঈგѣΞਕѣొЊߏ࠹఼۞

⁵

Ăٕ۰ߏགྷϤϒ૱

۞۱௡ᖐ׶࿣ᗓ۱̝ม۞۩ঈ఼྽ซˢĄЯࠎд ۱ཧ̰࿣ᗓ۱۞ள૱۱௡ᖐᄃϒ૱۞۱ཧ௡ᖐ׀

д˘੓Ă֭՟ѣ҈ቯ̶ࠧĂ࿣ᗓ۱఼ঈਕགྷϤ׹

ಛϒ૱۞۱ڽซҖĄҭߏ৿ͻϒ૱۞͚ঈგ۞ా

ତĂ۩ঈ̙ٽଵ΍҃टٽౄј࿣ᗓ۱۱ڽ௡ᖐ΍

ன࿅ޘ·ঈன෪ٕ۰ᙷҬ۱ঈཚ۞ត̼

⁶

Ą҃۱ঈ ཚ۞ؠཌྷυืΒӣᅈბ۞۱ڽ۩มᕖ̂׶۱ڽጨ

۞ຫᗼĄٙͽ఺჌ன෪ᑕΪਕჍࠎ࿣ᗓ۱۞Ԋొ

࿅ޘ·ঈன෪

^5,7

Ą

࿣ᗓ۱дᓜԖ˯౵૱ܑன۞াېΒ߁ၙّݜ မăͅᖬ൴Ϡ۱ۆăม໏ّ൴፵ă҈ቯّ਒൭ă ᓘ།ăݜҕ׶ײӛӧᙱĄ۱ཧ̰࿣ᗓ۱۞ঽଈ̂

ࡗѣ 1 5 % ߏ՟ѣাې۞

³

Ăϒтԧࣇ۞ঽּ˘

ᇹĂߏд໰਒ొ X Ѝͯॡ̖ຍγ൴ன۞Ą

࿣ᗓ۱۞ᝥҾ෧ᕝυืА҂ᇋ׎΁А͇۞۱ ᝃཚͽ̈́۱ཚሳĂּт͚ঈგౕᗆ ( bronchial atresia )Ăᝃّᙷཛྷᒛ۞۱ԛј̙։ ( congenital cystic adenomatoid malformation, CCAM )Ă۱ཧ ۱ঈཚ ( lobar emphysema )Ă Swyer-James ტЪ

াĂ͚ঈგᝃཚĂ͚ঈგᕖૺাĂ۱ۆĂ۱ᓘ ႹĂજᐖਔ გ׶၆ϒ૱۱௡ᖐ۞ள૱ր௚જ ਔҕ୵ֻᑕ

⁶

Ą

ᓜԖ෧ᕝ۞ෞҤΒ߁਒ొ X ЍĂ࿪ཝᕝᆸବ ೡĂ८ჃВॎĂ͚ঈგᛷᇆ׶ҕგᛷᇆ

¹

Ą਒ొ

X Ѝགྷ૱ពϯள૱۞۱௡ᖐٕ۰ҕგ۞ౚᇆĂЯ

҃҂ᇋז࠹ᙯ۞෧ᕝĂּтĈ࿣ᗓ۱ਕܑனညߏ ۱ۆ˘ᇹ۞˘๴ౚᇆă˘ਠ۱ཚ๴ăΒӣঈ୵ࢬ

۞ཚ๴ٕ۰ᝃཚĄ౵૱р൴дν઎˭۱ཧ۞ૄغ

ొ

⁵

Ąֻ҃ҕ۞ளҜજਔд X Ѝ໰ͯΪਕઊႬజ

൴னĂυืд࿪ཝᕝᆸ˯̖ਕځព۞జԱ΍ֽĄ

࿪ཝᕝᆸବೡΞͽ୻຾۞࠻ז࿣ᗓ۱۞͚ঈ

გᄃ۱௡ᖐඈкត۞ঽநྋ࣠ඕၹĂซ҃ᑒӄޙ ϲ෧ᕝĄ༊࿪ཝᕝᆸବೡពϯ΍Ҝٺ˭۱ཧ΍ன ᝃཚٕ۱ঈཚĂ֭ͷѣளҜજਔֻҕ۞ኑᗔ۱௡

ᖐள૱ॡĂಶࢋ҂ᇋזߏ࿣ᗓ۱

⁶

Ą

८ჃВॎјညநኢ˯ޝዋٺ࿣ᗓ۱׶Ч჌ត ளݭ۞෧ᕝĂЯࠎ၆ٺҕგ׶۱၁ኳள૱۞Ч࣎

ᆸࢬ۞ᇆညౌѣໂָ۞ӔனĄЯѩιΞͽഴ͌၆ ҕგᛷᇆ۞ᅮࢋ

⁸

Ąҭߏঽˠ۞ײӛજүົд८ ჃВॎҕგౄᇆ˯யϠ઄ᇆĂЯ҃ࢨט˞ι۞ᑕ ϡ

⁹

Ą

ϫ݈ᓲ୊ё࿪ཝᕝᆸ ( Spiral CT ) ႙႙פ΃

˞็௚۞࿪ཝᕝᆸăҕგᛷᇆ׶८ჃВॎĄιѣ

ೀ࣎ᐹ๕ĂΒ߁˞གྷϤᓠͯࢦޙăˬჯϲវၹဦ ᒔ଀ፋវඕၹࢦ௡ćྵԣి۞ବೡΞͽഴֹ͌ϡ ᗉᐖ጗׶ពᇆ጗۞጗ณćΞТॡෞҤ۱၁ኳ׶ঈ

྽۞ඕၹćͽ̈́ᆊ᏿ͧ८ჃВॎܮآ

^{1 0 , 11}

Ą҃ͷ

࿪ཝᕝᆸҕგౄᇆ ( CT angiography ) ߏ઱˘ಏ˘

ᑭߤಶΞͽሀᑢ΍જਔֻҕăᐖਔа߹ᄃ۱௡ᖐ ត̼۞ᇆညĂགྷϤˬჯϲវၹဦΞͽֹᓜԖᗁर Հ˞ྋ࠹ᙯ۞ྋ࣠ၹౄĂЯѩΞઇࠎ࿣ᗓ۱۞෧ ᕝᄃฟ˥݈ෞҤ۞౵ָᏴፄ

¹²

Ą

ঽଈд਒ొ X Ѝ˯Ϊ࠻זν˭۱ѣ˘୧ள૱

ҕგĂҭ౅࿅࿪ཝᕝᆸ̙ҭਕ࠻זᗕ઎۱၁ኳ࿅

ޘ·ঈᄃк୧ளҜજਔֻҕĂགྷϤ࿪ཝˬჯϲវ ࢦ௡Հਕ୻຾࠻זજਔ۞ֽ໚ᄃ֕ШĂ၆ٺ͘ఙ

݈۞ෞҤܧ૱ѣᑒӄĄд͛ᚥ˯֭՟ѣᙯٺ࿣ᗓ ۱ଈ۰۞۱Αਕ࠹ᙯಡጱĂώঽଈдநጯᑭߤॡ ᗕ઎˭۱ཧײӛࢰ˭ࢫ҃۱Αਕྏរពϯ۱ዶट

᎕ᆧΐ۞ѝഇܡ๫ݭೱঈᅪᘣĂౌ׶࿪ཝᕝᆸ̚

ܑன۱၁ኳ࿅ޘ·ঈ௑ЪĄ

д۱ཧ̰࿣ᗓ۱̚ຏߖߏ౵૱֍۞࡭ঽࣧ

ЯĂٙͽ̂ొЊ૞छౌޙᛉ˘ό෧ᕝజቁؠĂࠤ Ҍߏ൑াې۞࿣ᗓ۱ౌᑕྍ̷ੵĂͽ࿰֨ຏߖă ݜҕͽ̈́ϏֽΞਕயϠ۞ᒛাត̼ĄЯࠎ˘ό࿣

ᗓ۱൴ϠຏߖĂಶົᅮࢋ߉Җྵ̂۞͘ఙĂညߏ ۱ཧ̷ੵćࡶ՟ѣயϠຏߖॡ݋Ξͽ߉Җྵ̈۞

͘ఙĂּт۱༼̷ੵٕߏແې̷ੵ

^1,2

Ąтڍঽଈ

൴Ϡ˞ᚑࢦ۞ײӛඃ࢝াېಶυื੺˯߉Җღާ

͘ఙĂͽ྿ז౵ָ۞࿰ޢඕڍ

¹³

Ąдঽଈ߉Җγ ࡊ͘ఙ̷ੵ࿣ᗓ۱݈Ăщଵҕგᛷᇆٕߏ࿪ཝᕝ ᆸҕგౄᇆቁᄮ̪൒ߏυื۞ĂЯࠎтѩΞͽ൴ னሕд۞਒ొٕ۰ཛొ۞ளҜજਔĂࢫҲฟ˥ॡ

۞ӧᙱޘ

^3,6,12

Ą

529

(4)

ѣొЊጯ۰ᄮࠎдາϠ׊ॡഇ൴ன۞࿣ᗓ۱

ົҋҖੜ̼෇ᒺĂЯѩޙᛉֹϡજਔং๫ ( tran- scatheter arterial embolization, TAE ) ۞͞ёԯֻҕ જਔܡ๫Ăֹ࿣ᗓ۱ҋ൒෇ᒺĂЯѩјࠎڼᒚາ Ϡ׊࿣ᗓ۱ྵщБ҃׀൴াྵ͌۞ആ΃͞ڱ

^14,15

Ą ҭдјˠĂ఍ந࿣ᗓ۱౵р۞ڼᒚᏴፄ̪ߏ૟ঽ իԆፋ۞̷ੵ

¹³

Ą

༊਒ొ X Ѝͯ̚ѣள૱ॡĂ੼ޘ۞ᘃႷߏд

࿣ᗓ۱۞෧ᕝ࿅඀̚౵ࢦࢋ۞ࢋ৵Ąซ˘Վ۞࿪

ཝᕝᆸᑭߤΞͽᙋځள૱۞જਔֻᑕ׶࿣ᗓ۱۞

௡ᖐඕၹĂ҃གྷϤ࿪ཝˬჯࢦ௡Հਕ୻຾࠻זજ ਔ۞ֽ໚ᄃ֕ШĄጐѝેҖ̷ੵ͘ఙߏࠎ˞ࢫҲ ݜҕᄃຏߖඈሕд۞Ϡ׻ރ਎Ă֭ͷΞͽ̈́ѝа

ೇϒ૱۞۱ΑਕĄ

ણ҂͛ᚥ

1.Lin CH, Lin CT, Chen CY, Peng HC, Chen HC, Wang PY.

Pulmonary sequestration. Chin Med J (Taipei) 1994; 53: 168-72.

2.Juettner F, Pinter H, Lammer G, Popper H, Friehs G. Bilateral intralobar pulmonary sequestration: therapeutic implications.

Ann Thorac Surg 1987; 43: 660-2.

3.Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung se- questration: report of seven cases and review of 540 published cases. Thorax 1979; 34: 96-101.

4.Riedlinger WF, Vargas SO, Jennings RW, et al. Bronchial atre- sia is common to extralobar sequestration, intralobar sequestra-

tion, congenital cystic adenomatoid malformation, and lobar emphysema. Pediatr Dev Pathol 2006; 9: 361-73.

5.Zylak CJ, Eyler WR, Spizarny DL, Stone CH. Developmental lung anomalies in the adult: radiologic-pathologic correlation.

Radiographics 2002; 22: S25-43.

6.Ikezoe J, Murayama S, Godwin JD, Done SL, Verschakelen JA.

Bronchopulmonary sequestration: CT assessment. Radiology 1990; 176: 375-9.

7.Karantanas AH, Gossios K, Kontoyiannis D. Intralobar lung se- questration and surrounding hyperaeration. Eur Radiol 2001; 11:

348. 8.Felker RE, Tonkin ILD. Imaging of pulmonary sequestration.

Am J Roentgenol 1990; 154: 241-9.

9.Doyle AJ. Demontration of blood supply to pulmonary seques- tration by MR angiography. Am J Roentgenol 1992; 158: 989- 90.

10.Frush DP, Donnelly LF. Pulmonary sequestration spectrum: A new spin with helical CT. Am J Roentgenol 1997; 169: 679-82.

11.Jose F, Roberto A, L DM, Pedro P. Diagnosis of pulmonary se- questration by spiral CT angiography. Thorax 1998; 53: 1089- 92.

12.Kang M, Khandelwal N, Ojili V, Rao KLN, Rana SS.

Multidetector CT angiography in pulmonary sequestration. J Comput Assist Tomogr 2006; 30: 926-32.

13.Ayed AK, Owayed A. Pulmonary resection in infants for con- genital pulmonary malformation. Chest 2003; 124: 98-101.

14.Lee KH, Sung KB, Yoon HK, et al. Transcatheter arterial em- bolization of pulmonary sequestration in neonates: long-term follow-up results. J Vasc Interv Radiol 2003; 14: 363-67.

15.Tokel Ka, Boyvat F, Varan B. Coil embolization of pulmonary sequestration in two infants: a safe alternative to surgery. Am J Roentgenol 2000; 175: 993-5.

530

Bilateral Pulmonary Sequestration with Hyperaeration Ĉ Case Report

Yen-Kun Ko, Chien-Lung Hsiao, Chih-Hung Lin, and Yu-Jen Lee

¹

Pulmonary sequestration is a relative rare congenital abnormality. Mostly, this congenital lesion occurs main- ly in the basal segments of the lower lobes and is more frequently unilateral. Bilateral pulmonary sequestration is a rare abnormality. We report a case of a 27-year-old male visited our hospital because of abdominal pain. He was found asymptomatic bilateral pulmonary sequestration within the lower lobes accidentally, which was con- firmed by computed tomography. There are few reports in the literature about bilateral pulmonary sequestration with hyperaeration. The treatment of choice is complete excision of the lesion to prevent the tendency of infec- tion, hemorrhage, and the long-term neoplastic changes. ( J Intern Med Taiwan 2008; 19: 527-530 )

Division of Chest Medicine, Cathy General Hospital Shiji Branch;

1

Division of Chest Medicine, Cathay General Hospital, Hsinchu Branch