A neonatal case of interruption of the aortic arch with interrupted left subclavian artery complicated with chylothorax

症例は日齢4の男児で,呼吸障害が出現し,胸部X線像にて右側に胸水貯留像を認めた.胸腔穿刺にて白濁オレンジ色の胸水を採取し,組成より乳糜胸と診断した.心音はギャロップリズムを示した.左上肢と両下肢の脈が弱いため左橈骨動脈造影を行い大動脈弓離断症と診断した.心臓カテーテル検査にて,心室中隔欠損および動脈管開存は伴わず,stretched foramen ovaleを認めた.また左鎖骨下動脈起始部は大動脈弓より離断しており,下行大動脈とも離れていた.本例は新生児期にすでに下行大動脈への豊寓な側副血行路を形成していたが,心不全および難治性乳糜胸による呼吸不全のため日齢26に死亡した.大動脈弓離断症にお...

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Published inShinzo Vol. 26; no. 4; pp. 388 - 393
Main Authors Mori, Kenji, Yoshida, Tetsuya, Hayashi, Koji, Nakatsu, Tadanori
Format Journal Article
LanguageJapanese
Published Japan Heart Foundation 15.04.1994
Subjects
PDDT
乳糜胸
大動脈弓離断症
左鎖骨下動脈起始部離断
Online AccessGet full text
ISSN0586-4488
2186-3016
DOI10.11281/shinzo1969.26.4_388

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Abstract 症例は日齢4の男児で,呼吸障害が出現し,胸部X線像にて右側に胸水貯留像を認めた.胸腔穿刺にて白濁オレンジ色の胸水を採取し,組成より乳糜胸と診断した.心音はギャロップリズムを示した.左上肢と両下肢の脈が弱いため左橈骨動脈造影を行い大動脈弓離断症と診断した.心臓カテーテル検査にて,心室中隔欠損および動脈管開存は伴わず,stretched foramen ovaleを認めた.また左鎖骨下動脈起始部は大動脈弓より離断しており,下行大動脈とも離れていた.本例は新生児期にすでに下行大動脈への豊寓な側副血行路を形成していたが,心不全および難治性乳糜胸による呼吸不全のため日齢26に死亡した.大動脈弓離断症において,左鎖骨下動脈起始部の離断している病型の報告例はなく,Celoriaなどの従来の分類では表現できない症例と考えられたので報告する.
AbstractList 症例は日齢4の男児で,呼吸障害が出現し,胸部X線像にて右側に胸水貯留像を認めた.胸腔穿刺にて白濁オレンジ色の胸水を採取し,組成より乳糜胸と診断した.心音はギャロップリズムを示した.左上肢と両下肢の脈が弱いため左橈骨動脈造影を行い大動脈弓離断症と診断した.心臓カテーテル検査にて,心室中隔欠損および動脈管開存は伴わず,stretched foramen ovaleを認めた.また左鎖骨下動脈起始部は大動脈弓より離断しており,下行大動脈とも離れていた.本例は新生児期にすでに下行大動脈への豊寓な側副血行路を形成していたが,心不全および難治性乳糜胸による呼吸不全のため日齢26に死亡した.大動脈弓離断症において,左鎖骨下動脈起始部の離断している病型の報告例はなく,Celoriaなどの従来の分類では表現できない症例と考えられたので報告する.
Author Nakatsu, Tadanori
Mori, Kenji
Hayashi, Koji
Yoshida, Tetsuya
Author_FL 中津 忠則
吉田 哲也
森 健治
林 弘治
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  organization: Department of Pediatrics, Komatsushima Red Cross Hospital
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References 13) Warren JW, Ralph TH, Paul GQ, et al :Chylous ascites and lymphoma. AMA J Dis Child 1959 ; 98 : 317-329
4) Celoria CG, Patton RB : Congenital absence of the aortic arch. Am Heart J 1959 ; 58 : 407-414
9)河西達夫:大動脈弓の異常に伴うその周囲器官の変化について.解剖学雑誌1962;37:275-292
6)鷲尾正彦,桜井淑史,入沢敬夫,ほか:大動脈弓欠損症の外科治療,心臓1971;3:632-642
11) Boles ET, Robert JI : Spontaneous chylothrax in the neonatal period. Am J Surg 1960 ; 99 :870-877
12)真田喬子,斎藤裕康,藤塚万里子,ほか:Congenitalpolyvalvulardiseaseが認められた乳児乳慶胸の1剖検例,小児科臨床!978;31:126-130
5) Roberts WC, Morrow AG, Braunwald E: Com-plete interruption of the aortic arch. Circulation 1962 ; 26 : 39-59
1) Everts-Sudrez EA, Carson CP : The triad of congenital absence of aortic arch (isthmus aor-tae) , patent ductus arteriosus and intraventri-cular septal defect?a triology. Ann Surg 1959 ; 150 : 153-159
2) Dische MR, Tsai M, Baltaxe HA : Solitary interruption of the arch of the aorta. Clinico-pathologic review of eight cases. Am J Cardiol 1975 ; 35 : 271-277
15)井出郁,小林瑛児,石川昭:新生児乳慶胸を合併したダウン症候群の2例.小児科臨床1992;45:117-121
3)加納達二,山中修,小林清亮,ほか:成人の孤立性大動脈弓離断症の一手術例。呼と循1991;39 : 367-372
10) Van Mierop LHS, Kusche LM : Cardiovascular anomalies in DiGeorge syndrome and impor-tance of neural crest as a possible pathogenetic factor. Am, J Cardiol 1986 ; 58 : 133-137
7) Hutchins GM : Coarctation of the aorta ex-plained as a branch point of the ductus arter-iosus. Am J Path 1971 ; 63 : 203-214
8) Rudolph AM, Heymann MA, Spitznas U:Hemodynamic considerations in development of narrowing of aorta. Am J Cardiol 1972 ;30 : 514-525
14) Yoss BS, Lipsitz PJ : Chylothrax in two mon-goloid infants. Clinical Genetics 1977 ; 12 : 357-360
References_xml – reference: 10) Van Mierop LHS, Kusche LM : Cardiovascular anomalies in DiGeorge syndrome and impor-tance of neural crest as a possible pathogenetic factor. Am, J Cardiol 1986 ; 58 : 133-137
– reference: 13) Warren JW, Ralph TH, Paul GQ, et al :Chylous ascites and lymphoma. AMA J Dis Child 1959 ; 98 : 317-329
– reference: 4) Celoria CG, Patton RB : Congenital absence of the aortic arch. Am Heart J 1959 ; 58 : 407-414
– reference: 3)加納達二,山中修,小林清亮,ほか:成人の孤立性大動脈弓離断症の一手術例。呼と循1991;39 : 367-372
– reference: 6)鷲尾正彦,桜井淑史,入沢敬夫,ほか:大動脈弓欠損症の外科治療,心臓1971;3:632-642
– reference: 12)真田喬子,斎藤裕康,藤塚万里子,ほか:Congenitalpolyvalvulardiseaseが認められた乳児乳慶胸の1剖検例,小児科臨床!978;31:126-130
– reference: 2) Dische MR, Tsai M, Baltaxe HA : Solitary interruption of the arch of the aorta. Clinico-pathologic review of eight cases. Am J Cardiol 1975 ; 35 : 271-277
– reference: 5) Roberts WC, Morrow AG, Braunwald E: Com-plete interruption of the aortic arch. Circulation 1962 ; 26 : 39-59
– reference: 8) Rudolph AM, Heymann MA, Spitznas U:Hemodynamic considerations in development of narrowing of aorta. Am J Cardiol 1972 ;30 : 514-525
– reference: 11) Boles ET, Robert JI : Spontaneous chylothrax in the neonatal period. Am J Surg 1960 ; 99 :870-877
– reference: 1) Everts-Sudrez EA, Carson CP : The triad of congenital absence of aortic arch (isthmus aor-tae) , patent ductus arteriosus and intraventri-cular septal defect?a triology. Ann Surg 1959 ; 150 : 153-159
– reference: 9)河西達夫:大動脈弓の異常に伴うその周囲器官の変化について.解剖学雑誌1962;37:275-292
– reference: 14) Yoss BS, Lipsitz PJ : Chylothrax in two mon-goloid infants. Clinical Genetics 1977 ; 12 : 357-360
– reference: 15)井出郁,小林瑛児,石川昭:新生児乳慶胸を合併したダウン症候群の2例.小児科臨床1992;45:117-121
– reference: 7) Hutchins GM : Coarctation of the aorta ex-plained as a branch point of the ductus arter-iosus. Am J Path 1971 ; 63 : 203-214
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SubjectTerms PDDT
乳糜胸
大動脈弓離断症
左鎖骨下動脈起始部離断
Title A neonatal case of interruption of the aortic arch with interrupted left subclavian artery complicated with chylothorax
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