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Journal of Medical Ultrasonics

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2011 - Vol.38

Vol.38 No.04

Case Report(症例報告)

(0481 - 0487)


A case of bronchopulmonary sequestration with a relatively good prognosis after fetal thoracoamniotic shunting

小松 玲奈1, 中田 雅彦2, 住江 正大3, 早田 桂1, 関野 和1, 岡田 朋美1, 石田 理1, 野間 純1, 吉田 信隆1, 秋山 卓士4

Reina KOMATSU1, Masahiko NAKATA2, Masahiro SUMIE3, Kei HAYATA1, Madoka SEKINO1, Tomomi OKADA1, Makoto ISHIDA1, Jun NOMA1, Nobutaka YOSHIDA1, Takashi AKIYAMA4

1広島市立広島市民病院産科婦人科, 2社会保険徳山中央病院産婦人科, 3山口大学医学部附属病院産科婦人科, 4広島市立広島市民病院小児外科

1Department of Obstetrics and Gynecology, Hiroshima City Hospital, 2Department of Obstetrics and Gynecology, Tokuyama Central Hospital, 3Department of Obstetrics and Gynecology, Yamaguchi University Hospital, 4Department of Pediatric Surgery, Hiroshima City Hospital

キーワード : bronchopulmonary sequestration, thoracoamniotic shunting, hydrothorax, fetal therapy, ultrasonography, prenatal

胎児胸水を伴う肺分画症の予後は不良である.胎児胸水除去及びシャント術を行うことで,胎児水腫や肺低形成が予防でき,予後の改善につながると言われている.胎児胸腔‐羊水腔シャント術が有効であった胎児肺分画症の1例を経験したので報告する.症例は,34歳,経産婦.前医で胎児胸部腫瘤と心臓右方偏位を認め,妊娠28週6日に当科を紹介初診した.胎児超音波検査・MRI検査で左胸郭に43×46×41mmの腫瘤と多量の胸水を認めた.腫瘤は肺葉外に存在し,腹部大動脈から腫瘤へ向かう栄養血管を同定し肺葉外肺分画症と診断した.その他胎児構造異常や胎児水腫を認めず,ウィルス感染は否定的であった.同日胎児胸水を32 ml除去したが,翌日には胸水の再貯留を認めたため,妊娠29週1日に胎児胸腔‐羊水腔シャント術を施行(超音波ガイド下に八光社製ダブルバスケットカテーテルを留置)した.術後両肺は拡張し,胸水の再貯留・分画症肺の増大・羊水過多や切迫早産徴候なく経過した.妊娠38週5日,分娩誘発にて3,110 gの女児をAS9/9で経腟分娩した.児はNICUに入院,人工呼吸管理は不要であったが,日齢1より胸水が貯留し胸腔持続ドレナージを開始,150‐200 ml/日の排液を認めた.日齢8に分画肺摘出術を施行し,経過良好で日齢17に退院した.本症例は無治療であれば胎児水腫などを合併し予後不良であったと予想され,胎児胸腔‐羊水腔シャント術が有用であった.

The prognosis of bronchopulmonary sequestration (BPS) complicated by hydrothorax, fetal hydrops, or pulmonary hypoplasia is generally considered poor. Prenatal thoracocentesis and thoracoamniotic shunting of massive hydrothorax are indicated to decrease perinatal morbidity. This report presents a case of BPS with a relatively good prognosis after thoracoamniotic shunting. A 34-year-old multigravida was referred to the hospital at 28 weeks and 6 days gestation for hydrothorax. Ultrasonography and MRI demonstrated a huge mass in the left thorax complicated by mediastinal shift to the right and large left pleural effusion. A combination including color Doppler ultrasound visualized a vessel arising from the descending aorta to the mass. These findings indicated extralobar BPS. Thoracocentesis was carried out during her first referral, when 32 mL of serous yellowish pleural effusion was removed. However, the pleural effusion reaccumulated on the following day and thoracoamniotic shunting was performed. A doublebasket catheter (16 G, Hakko Medical Products, Japan) was placed percutaneously under ultrasound guidance. The pleural effusion disappeared after shunting, and the bilateral lungs expanded. A female baby weighing 3110 g was successfully delivered with an Apgar score of 9(1’)/9(5’). The patient was transferred to the Neonatal Intensive Care Unit. The pleural effusion reaccumulated postnatally, and the patient required continuous drainage. Tumor resection was performed on the 7th postnatal day. A histopathologic examination revealed BPS. The patient was discharged on the 17th day and was doing well at 3 months of age. Thoracoamniotic shunting is considered to be an effective therapeutic option in cases of BPS complicated by hydrothorax.