Prenatal features of congenital peribronchial myofibroblastic tumor

Here, we report a case of a congenital peribronchial myofibroblastic tumor (CPMT). A 34-year-old primigravida was referred to our hospital at 31 gestation weeks because of suspected congenital pulmonary airway malformation (CPAM). Fetal ultrasonography showed a mass measuring 4.6 × 4.0 × 3.9 cm with...

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Main Authors: Hitoshi Isohata, Tsutomu Yoshida, Itaru Sanoyama, Yu Yamazaki, Hiroyuki Goto, Yoshihiro Yoshimura, Kyoko Hattori, Takao Shimaoka, Kazuki Sekiguchi, Yoko Onishi, Kiyoshi Tanaka, Daigo Ochiai
Format: Article
Language:English
Published: Elsevier 2024-04-01
Series:Radiology Case Reports
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S1930043324000025
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author Hitoshi Isohata
Tsutomu Yoshida
Itaru Sanoyama
Yu Yamazaki
Hiroyuki Goto
Yoshihiro Yoshimura
Kyoko Hattori
Takao Shimaoka
Kazuki Sekiguchi
Yoko Onishi
Kiyoshi Tanaka
Daigo Ochiai
author_facet Hitoshi Isohata
Tsutomu Yoshida
Itaru Sanoyama
Yu Yamazaki
Hiroyuki Goto
Yoshihiro Yoshimura
Kyoko Hattori
Takao Shimaoka
Kazuki Sekiguchi
Yoko Onishi
Kiyoshi Tanaka
Daigo Ochiai
author_sort Hitoshi Isohata
collection DOAJ
description Here, we report a case of a congenital peribronchial myofibroblastic tumor (CPMT). A 34-year-old primigravida was referred to our hospital at 31 gestation weeks because of suspected congenital pulmonary airway malformation (CPAM). Fetal ultrasonography showed a mass measuring 4.6 × 4.0 × 3.9 cm with mixed high and low echogenicity in the left lung, which was associated with microvascular blood flow in the tumor. Fetal magnetic resonance imaging (MRI) revealed a low-intensity left lobe lung lesion on a T2-weighted image. These findings suggested that the mass was a CPAM with atypical hypointense findings on MRI T2-weighted images or a rare primary pulmonary tumor, such as a CPMT. Unfortunately, the fetus died in utero at 34 gestation weeks due to cardiovascular failure, which could have resulted from direct encasement of the great vessels or cardiac compression due to rapid tumor growth. The autopsy findings confirmed the diagnosis of CPMT. Primary pulmonary tumors, such as CPMT, are extremely rare lung diseases that develop in utero. These tumors often rapidly grow during pregnancy, resulting in intrauterine fetal death. However, if the patient survives surgical mass resection, the prognosis is good. Given the adverse outcomes observed in our case, careful fetal monitoring is required in case of suspected CPMT during the third trimester of pregnancy. Moreover, in case the well-being of the fetus cannot be assured, immediate delivery should be considered, even in the preterm period, followed by surgery.
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spelling doaj.art-ac9e8ddad8b748f3b98ec2719797c6392024-02-28T05:13:05ZengElsevierRadiology Case Reports1930-04332024-04-0119414631467Prenatal features of congenital peribronchial myofibroblastic tumorHitoshi Isohata0Tsutomu Yoshida1Itaru Sanoyama2Yu Yamazaki3Hiroyuki Goto4Yoshihiro Yoshimura5Kyoko Hattori6Takao Shimaoka7Kazuki Sekiguchi8Yoko Onishi9Kiyoshi Tanaka10Daigo Ochiai11Department of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDivision of Molecular Pathology, Department of Comprehensive Medicine, Research and Development Center for New Medical Frontiers, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Pathology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Pediatric Surgery, Kitasato University School of Medicine, Sagamihara, JapanDepartment of Obstetrics and Gynecology, Kitasato University School of Medicine, Sagamihara, Japan; Corresponding author.Here, we report a case of a congenital peribronchial myofibroblastic tumor (CPMT). A 34-year-old primigravida was referred to our hospital at 31 gestation weeks because of suspected congenital pulmonary airway malformation (CPAM). Fetal ultrasonography showed a mass measuring 4.6 × 4.0 × 3.9 cm with mixed high and low echogenicity in the left lung, which was associated with microvascular blood flow in the tumor. Fetal magnetic resonance imaging (MRI) revealed a low-intensity left lobe lung lesion on a T2-weighted image. These findings suggested that the mass was a CPAM with atypical hypointense findings on MRI T2-weighted images or a rare primary pulmonary tumor, such as a CPMT. Unfortunately, the fetus died in utero at 34 gestation weeks due to cardiovascular failure, which could have resulted from direct encasement of the great vessels or cardiac compression due to rapid tumor growth. The autopsy findings confirmed the diagnosis of CPMT. Primary pulmonary tumors, such as CPMT, are extremely rare lung diseases that develop in utero. These tumors often rapidly grow during pregnancy, resulting in intrauterine fetal death. However, if the patient survives surgical mass resection, the prognosis is good. Given the adverse outcomes observed in our case, careful fetal monitoring is required in case of suspected CPMT during the third trimester of pregnancy. Moreover, in case the well-being of the fetus cannot be assured, immediate delivery should be considered, even in the preterm period, followed by surgery.http://www.sciencedirect.com/science/article/pii/S1930043324000025Congenital peribronchial myofibroblastic tumorPrenatal diagnosisFetal MRIUltrasound
spellingShingle Hitoshi Isohata
Tsutomu Yoshida
Itaru Sanoyama
Yu Yamazaki
Hiroyuki Goto
Yoshihiro Yoshimura
Kyoko Hattori
Takao Shimaoka
Kazuki Sekiguchi
Yoko Onishi
Kiyoshi Tanaka
Daigo Ochiai
Prenatal features of congenital peribronchial myofibroblastic tumor
Radiology Case Reports
Congenital peribronchial myofibroblastic tumor
Prenatal diagnosis
Fetal MRI
Ultrasound
title Prenatal features of congenital peribronchial myofibroblastic tumor
title_full Prenatal features of congenital peribronchial myofibroblastic tumor
title_fullStr Prenatal features of congenital peribronchial myofibroblastic tumor
title_full_unstemmed Prenatal features of congenital peribronchial myofibroblastic tumor
title_short Prenatal features of congenital peribronchial myofibroblastic tumor
title_sort prenatal features of congenital peribronchial myofibroblastic tumor
topic Congenital peribronchial myofibroblastic tumor
Prenatal diagnosis
Fetal MRI
Ultrasound
url http://www.sciencedirect.com/science/article/pii/S1930043324000025
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