Intrauterine Therapy for the Acutely Enlarging Fetal Cystic Hygroma

Kuwabara, Yoshimitsu; Sawa, Rintaro; Otsubo, Yasuo; Yoneyama, Yoshio; Asakura, Hitoshi; Araki, Tsutomu; Takeshita, Toshiyuki

doi:10.1159/000075149

Cited by 22 publications

(14 citation statements)

References 12 publications

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“…Intrauterine sclerotherapy can be performed to reduce the size of the lesion. [3] MRI scan may provide useful information about the anatomy of the neck mass and the adjacent airway for the selection of ex utero intrapartum treatment procedure. [4] …”

Section: Discussionmentioning

confidence: 99%

Giant cystic hygroma of the neck with spontaneous rupture

Kaur

Gupta

Amratash

et al. 2007

J Indian Assoc Pediatr Surg

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Cystic hygroma is a disfiguring benign lesion commonly observed in the neck and face regions of children. The common complications of these malformations are respiratory obstruction, dysphagia, infection, and hemorrhage. Here, we present an uncommon complication of spontaneous rupture of a cystic hygroma. A two-day-old male baby presented with a swelling that was cystic, non-compressible, non-pulsatile and the transillumination was negative. With a clinical diagnosis of a massive cystic hygroma of the neck, CT scan was performed that demonstrated a large multiloculated cystic lesion. On the second day after admission, the swelling burst spontaneously with drainage of about 1000 ml of hemorrhagic fluid. The swelling shrunk considerably after that. The patient underwent surgical excision of the lesion and had an uneventful postoperative recovery. The histopathological examination confirmed the diagnosis of cystic hygroma.

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Section: Discussionmentioning

confidence: 99%

Giant cystic hygroma of the neck with spontaneous rupture

Kaur

Gupta

Amratash

et al. 2007

J Indian Assoc Pediatr Surg

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“…It is well known as a biological response modifi er with an anti-tumor effect. Some researchers, most of them were from Japan, used it in treating neonatal and even fetal cystic hygroma with some success [3][4][5][6][7][8] . The pathway of action of OK-432 within the cystic hygroma was thought to be cellular and cytokine-mediated [5] .…”

Section: Fetal Cystic Hygromamentioning

confidence: 99%

“…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] . Moreover, three past cases with fetal cystic hygroma and hydrops fetalis receiving antenatal OK-432 injection died in utero or within one month after birth ( table 2 ).…”

Section: Fetal Cystic Hygromamentioning

confidence: 99%

“…It is caused by the accumulation of lymph due to the failure of the peripheral lymphatics to fl ow into the jugular sacs [2] . OK-432, or Picibanil ® , a lyophilized incubation mixture of the group A Streptococcus pyogenes of human origin (avirulent Su strain), was initially used in treating malignant pleural effusion in the cancer patients, was recently used in treating pediatric, neonatal, and even fetal cystic hygroma and was reported to be effective [3][4][5][6][7][8] .…”

Section: Introductionmentioning

confidence: 99%

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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 sclerotherapy might be an alternative management option. The use of OK-432, as a sclerosing agent, has shown positive results in several published cases of cystic hygroma [6][7][8][9] .…”

Section: Introductionmentioning

confidence: 99%

Intrauterine Treatment of Large Fetal Neck Lymphangioma with OK-432

Mikovic¹,

Simić²,

Egić³

et al. 2009

Fetal Diagn Ther

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Lymphangiomas are benign vascular malformations of the lymphatic system and most commonly present in the neck area. Large lymphangiomas may compress and/or displace the larynx, trachea and esophagus and cause serious respiratory and feeding problems in neonates. Prenatal therapy could eliminate the risks of the mentioned complications. Prenatal therapy may include the EXIT (ex utero intrapartum treatment) procedure. As this procedure has certain risks for both the neonate and mother, the introduction of a safer method is justified. The use of OK-432, as a sclerosing agent, has shown positive results in several published cases of cystic hygroma, but there is no study about the prenatal use of this agent in the treatment of lymphangioma. The aim of this study was to present our experience with intrauterine intralesional injection of OK-432 in the treatment of neck lymphangiomas. Two cases of large multicystic neck lymphangiomas that were closely situated to the fetal airway were treated by single intralesional injection of OK-432. We noticed a progressive decrease in tumor volume throughout gestation. We did not experience any complications and there were no respiratory or feeding problems in the neonates. The esthetical appearance was satisfactory and both children were normal at the age of 2 years and 6 months, respectively. This report suggests that prenatal intralesional injection of OK-432 might be a safe and effective treatment in selected cases with large fetal neck lymphangiomas.

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Intrauterine Therapy for the Acutely Enlarging Fetal Cystic Hygroma

Cited by 22 publications

References 12 publications

Giant cystic hygroma of the neck with spontaneous rupture

Giant cystic hygroma of the neck with spontaneous rupture

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

Intrauterine Treatment of Large Fetal Neck Lymphangioma with OK-432

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