Prenatally diagnosed large mediastinal lymphangioma: A case report

Tanaka, Hideaki; Masumoto, Kouji; Aoyama, Tomohiro; Sanmoto, Youhei; Ono, Kentaro; Sakamoto, Naoya; Kawakami, Hajime

doi:10.1002/ccr3.1760

Cited by 4 publications

(7 citation statements)

References 10 publications

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“…Up to date, no extensive research has been carried out on this subject. Several echocardiographic examinations in a fetus with lymphangioma allowed for the observation of the lesion and analysis of whether the lymphatic lesion affects cardiac function, e.g., due to compression or flow and cardiac output disturbances [8,23]. In addition, fetal heart echocardiography in the third trimester of pregnancy in a fetus with an extracardiac defect allows us to indicate the time, place, and manner of delivery.…”

Section: Discussionmentioning

confidence: 99%

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The Value of Fetal Heart Evaluation in Fetuses with Rare Congenital Lymphangiomas: A Cohort Study from a Single Tertiary Center across Two Decades (Years 1999–2020)

Kordjalik

Szmyd

Karuga

et al. 2022

JCM

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Lymphangiomas are uncommon, benign (from a histopathology viewpoint) malformations of the lymphatic system with thin-walled vessels; however, these tumors may be dangerous for fetal or neonatal life. They are observed in 1:6000 newborns at birth and in 1:750 spontaneous abortions. We aimed to investigate the role of fetal echocardiography in the prognosis of lymphangioma. Selected data of 19,836 pregnant women studied between 1999 and 2020 were retrospectively analyzed. In total, 32 cases of lymphangioma meeting the following criteria were further analyzed: (1) ultrasound availability from the 1st trimester of pregnancy and (2) nuchal translucency ≤ 2.5 mm. Echocardiographic findings, karyotype, size, and location of the possible lesion were juxtaposed with the clinical follow-up. The statistical analysis was performed using Statistica 13.1 software (StatSoft, Tulsa, OK, USA). Lymphangioma in the analyzed material coexisted with abnormalities in fetal echo in 78% (n = 25) of cases, especially: heart defect in 50% (n = 16) and with normal heart structure with functional changes in 28% (n = 9). Karyotype was available in 50% of the analyzed cases (n = 16). Normal cytogenetic results were observed in 62.5% (n = 10) of cases. In the remaining cases, the following were observed: Turner Syndrome: 25% (n = 4) and Down Syndrome 12.5% (n = 2). The rate of alive newborns was significantly higher among fetuses with isolated lymphangioma in comparison to those with lymphangioma associated with abnormal ECHO examination: 38.46% (n = 5) vs. 15.38% (n = 2; p = 0.037). Abnormal ECHO exam was a poor prognostic sign for fetuses with lymphangioma; therefore, we think it is important to refer these cases for detailed echocardiography in tertiary centers. Moreover, it should be highlighted that in all lymphangioma cases there was an indication to perform the karyotype assessment, as there was a high risk of aneuploidy.

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Section: Discussionmentioning

confidence: 99%

“…They are observed in 1:6000 newborns at birth and in 1:750 spontaneous abortions. In the majority of cases, they involve the neck or axilla, but can also involve other parts of the body, such as: scrotum, retroperitoneum, gluteal region, mediastinum, groin, pelvis, mesentery, omentum and spleen [3][4][5][6][7][8].…”

Section: Introductionmentioning

confidence: 99%

The Value of Fetal Heart Evaluation in Fetuses with Rare Congenital Lymphangiomas: A Cohort Study from a Single Tertiary Center across Two Decades (Years 1999–2020)

Kordjalik

Szmyd

Karuga

et al. 2022

JCM

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“…The initial prenatal images raised the suspicion of pericardiac effusion, similar to the previously reported case. 4 Although mediastinal cLM is rare, it should be considered as a candidate in the differential diagnosis of fetal pericardiac effusion. Prenatal thoracocentesis caused shape changes of the target, which was an asymmetrical unilocular cystic lesion surrounding the heart.…”

Section: Discussionmentioning

confidence: 99%

“…7 Tanaka et al reported a case of neonatal respiratory distress due to mediastinal cLM requiring percutaneous decompression immediately after birth. 4 According to Jung et al, a mediastinal tumor can lead to lung hypoplasia and heart failure due to compression of the heart and the mediastinum. 7 After careful evaluation by MRI and sonography, prenatal percutaneous decompression could be considered for the prevention of fetal hydrops and lung hypoplasia, and emergent respiratory management just after birth could be avoided.…”

Section: Discussionmentioning

confidence: 99%

“…There have only been seven cases in total, including our own case, of prenatally diagnosed mediastinum cLM with perinatal treatment (Table 1). 1,4,[6][7][8][9] Muraskas et al first reported prenatal thoracic decompression of mediastinal cLM with hydrops fetalis. 6 Percutaneous ultrasound-guided thoracic decompression was performed two times prenatally.…”

Section: Discussionmentioning

confidence: 99%

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A fetus with large mediastinal cystic lymphatic malformation managed with prenatal serial thoracocentesis and postnatal sclerotherapy

Muromoto

Sugibayashi

Ozawa

et al. 2022

J of Obstet and Gynaecol

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We report a very rare case of large fetal mediastinal cystic lymphatic malformation (cLM), formerly called lymphangioma, that was managed with prenatal serial thoracocentesis and postnatal sclerotherapy. A fetal pericardial effusion-like lesion gradually increased the size until it occupied more than half of the thoracic cavity. Thoracocentesis was performed three times, namely at 31 and 35 weeks of gestation, and also just before the delivery for decompression. The characteristic shape changes of the lesion were observed after thoracocentesis. A boy delivered by planned cesarian section at term required respiratory support after birth. Postnatal magnetic resonance imaging (MRI), which showed a large cystic lesion with septa and hemorrhage without solid components, led to the diagnosis of cLM. The fluid from the cyst continued to be drained. Neonatal sclerotherapy using OK-432 was effective and the infant recovered well. Fetal thoracocentesis may be useful for the diagnosis and management of a large mediastinal cLM.

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Fetal Cystic Lymphatic Malformations

Nzelu,

Panayotidis,

Smith

et al. 2024

J of Ultrasound Medicine

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ObjectiveEvaluate pregnancy and neonatal outcomes with fetal cystic lymphatic malformations (LMs), excluding those arising from the posterior neck, to facilitate patient counseling.MethodA systematic review was performed in accordance with PRISMA guidance. Case series and case reports published between 2000 and 2022 were included.ResultsSixty‐five studies (96 fetuses) met the inclusion criteria. The average gestational age at diagnosis was 25.5 weeks with the commonest location being the anterior neck (28%). All patients were diagnosed with LM using two‐dimensional (2D) ultrasound. Prenatal progression in LM size, presence of intralesional bleeding, or fetal hydrops occurred in 70% (41/59), 9% (5/59), and 3% (2/59), respectively. Chromosomal and structural abnormalities were reported in 4% (2/52) and 2% (2/96), respectively. Overall livebirth rate was 94% (79/84); 12/96 resulted in termination and 5/84 in in utero demise.The average gestational age of delivery was 37.7 weeks. Exactly 19% (15/79) had a vaginal birth, of which shoulder dystocia occurred in one infant. Ex utero intrapartum treatment (EXIT) procedure was performed in 13% (10/79). Postnatal treatment commonly involved surgical excision 38% (30/79), sclerotherapy in 21.5% (17/79), or combination of both in 11.4% (9/79). Of those with reported follow‐up, 4 died within 1 year, 1 developed heart failure at 2 years of life, and the remaining 44 had normal developmental outcomes.ConclusionFetal cystic LMs, excluding those in the posterior neck, are not commonly associated with chromosomal, or additional structural abnormalities. They usually increase in size before delivery with only a minority developing complications. The good developmental outcome was reported in all survivors.

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Prenatally diagnosed large mediastinal lymphangioma: A case report

Cited by 4 publications

References 10 publications

The Value of Fetal Heart Evaluation in Fetuses with Rare Congenital Lymphangiomas: A Cohort Study from a Single Tertiary Center across Two Decades (Years 1999–2020)

The Value of Fetal Heart Evaluation in Fetuses with Rare Congenital Lymphangiomas: A Cohort Study from a Single Tertiary Center across Two Decades (Years 1999–2020)

A fetus with large mediastinal cystic lymphatic malformation managed with prenatal serial thoracocentesis and postnatal sclerotherapy

Fetal Cystic Lymphatic Malformations

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