In utero Evaluation and the Long-Term Prognosis of Living Infants with Cystic Hygroma

Fujita, Yasuyuki; Shoji, Shuichi; Nakayama, Hideki; Taguchi, Tomoaki; Suita, Sachiyo; Nakano, Hitoo

doi:10.1159/000053948

Cited by 23 publications

(6 citation statements)

References 14 publications

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“…A study of about 1,320 fetuses with a euploid karyotype and increased nuchal translucency in the first trimester reported worse outcome in 61.7% of cases with nuchal size larger than 6.5 mm (13), as observed in our series. It should be noted that cystic hygroma has a worse prognosis when associated with hydrops fetalis, as confirmed here (21). We confirm the good prognosis of cystic hygroma measuring <6 mm, unless associated with hydrops fetalis.…”

Section: Discussionsupporting

confidence: 87%

“…There are few reports on the long‐term prognosis of infants with cystic hygroma. Fujita et al (21) reported developmental retardation in 60% of cases (3/5) during 4–90 months of follow‐up. Malone et al (15), in a prospective multicentred study of 132 cases of fetal cystic hygroma, estimated the prevalence, natural history and long‐term paediatric outcome (median: 25 months, range: 12–50 months).…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Characteristics and outcome of fetal cystic hygroma diagnosed in the first trimester

Graesslin

Derniaux

Alanio

et al. 2007

Acta Obstet Gynecol Scand

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Section: Discussionsupporting

confidence: 87%

Section: Discussionmentioning

confidence: 99%

Characteristics and outcome of fetal cystic hygroma diagnosed in the first trimester

Graesslin

Derniaux

Alanio

et al. 2007

Acta Obstet Gynecol Scand

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“…Generalized edema and hydrops may be the cause of left atrium dysfunction and aorta due to a compression effect leading to fetal death. In the literature, only a few studies have reported the resolution of hydrops and healthy newborns (11); the majority of the studies demonstrate that hydrops is associated with poor fetal outcomes (6,10,12,13). On the other hand, the resolution of nuchal edema with a normal karyotype is a good prognostic marker in the absence of any coexisting malformation.…”

Section: Discussionmentioning

confidence: 99%

Analysis of cystic hygroma diagnosed in the first trimester: Single-center experience

Yakıştıran

Altınboğa

Canpolat

et al. 2020

J Turkish German Gynecol Assoc

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Objective: To evaluate the obstetric outcomes of fetuses with cystic hygroma other than karyotype abnormalities and structural malformations. Material and Methods: We conducted a retrospective study based on the review of medical records of pregnant women in whom ultrasonographic diagnosis of fetal cystic hygroma was established in the first trimester from January 2014 to October 2018. All patients were offered genetic counselling and prenatal invasive diagnostic procedures to obtain fetal karyotype. For ongoing pregnancies fetal echocardiography and detailed second trimester sonographic anomaly screening was performed by a perinatologist/pediatric cardiologist. The demographic characteristics of the women and the results of the karyotype analysis were obtained from the database of our hospital and correlated with the obstetric outcomes. Results: Within a five-year period, there were 106 cases of fetal cystic hygroma. Of those, fetal cardiac malformations were detected in four and micrognathia in one fetus. Eighty-five women underwent fetal invasive procedures and karyotype abnormalities were detected in 52 of the cases. Fetal outcomes of 33 cases with normal karyotype and 21 cases in whom karyotyping analysis were not performed due to patient refusal were enrolled into the study. Obstetric outcomes of 21 women who refused karyotyping consisted of 13 livebirths, seven missed abortions, and one fetal death, whereas those of 33 women with normal karyotype were; 12 livebirths, 12 missed abortions, two hydrops fetalis, and five fetal deaths. Nineteen of 33 fetuses with a normal karyotype and eight of 21 fetuses in whom karyotyping was not performed were terminated. Conclusion: The presence of cystic hygroma carries a high risk for fetal karyotype abnormalities and cardiac malformations. The postnatal outcomes of the fetuses with cystic hygroma appeared to be correlated with the absence of structural malformations and karyotype abnormalities.

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“…An 81.5% fetal demise rate was observed in fetuses with cystic hygroma [9] . The prognosis is found to be poorer if associated with chromosomal abnormalities, hydrops fetalis, or structural abnormalities despite a case with hydrops fetalis and cystic hygroma was reported to regress spontaneously after birth [6,18,19] . There were only two successful cases treated antenatally by OK-432 ever reported in the literature but neither of them was associated with hydrops fetalis [7,8] .…”

Section: Fetal Cystic Hygromamentioning

confidence: 99%

Antenatal Treatment of Chylothorax and Cystic Hygroma with OK-432 in Nonimmune Hydrops fetalis

Chen

Chen²,

Shih³

et al. 2005

Fetal Diagn Ther

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Objectives: To present our experience of using OK-432 in treating fetal cystic hygroma and chylothorax complicated with nonimmune hydrops fetalis. Methods: OK-432 (Picibanil^®) was injected into the fetal pleural cavity or fetal cystic hygroma. Results:Patient 1: A 23-year-old, gravida 2, para 1, was found to have a recurrent fetal chylothorax at GA 29 weeks. Serial amnioreduction and thoracocentesis was performed at GA 31, 32, 33, and 34 weeks. Intrapleural OK-432 injection was performed twice at GA 33 and 34 weeks. Cyanosis and respiratory distress were noted immediately after birth (GA 34 weeks). The baby expired despite of aggressive neonatal resuscitation. Patient 2: A 26-year-old, gravida 2, para 1, was found to have a cystic hygroma of her fetus at GA 17 weeks. Karyotype of the cystic fluid and the amniocytes were 46, XY. Fetal ascites developed at GA 22 weeks. OK-432 injection into the tumour was performed at GA 23 weeks. Stabilization of the cystic hygroma was noted throughout the pregnancy (about 3.5 cm in diameter). Serial fetal paracentesis and/or amnioreduction were performed. Karyotype of the ascites was again 46, XY. Maternal dietary modification with medium chain triglyceride was also prescribed. Chylothorax developed and the baby was born by cesareans at GA 32 weeks. Resolution of pleural effusion, ascites, and regression of cystic hygroma were noted since the 2nd day after birth. The baby had survived beyond 4 months of age at submission. Conclusion: Combination of antenatal OK-432 injection, maternal dietary modification, serial thoracocentesis plus paracentesis, together with amnioreduction and tocolysis, appeared to contribute to the success of antenatal treatment. Fetal pulmonary expansion may determine the immediate neonatal survival.

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In utero Evaluation and the Long-Term Prognosis of Living Infants with Cystic Hygroma

Cited by 23 publications

References 14 publications

Characteristics and outcome of fetal cystic hygroma diagnosed in the first trimester

Characteristics and outcome of fetal cystic hygroma diagnosed in the first trimester

Analysis of cystic hygroma diagnosed in the first trimester: Single-center experience

Antenatal Treatment of Chylothorax and Cystic Hygroma with OK-432 in Nonimmune Hydrops fetalis

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