Congenital chylothorax in a patient with 21 trisomy syndrome

Yamamoto, Toshiyuki; Koeda, Tatsuya; Tamura, Akiko; Sawada, Hiroshi; Nagata, Ikuo; Nagata, Naoki; Ito, Takashi; Mio, Y.

doi:10.1111/j.1442-200x.1996.tb03733.x

Cited by 12 publications

(9 citation statements)

References 9 publications

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“…However, lymphocytosis can be also discovered in other conditions such as tuberculosis or viral infection. Our two cases proved to non-chylous effusion after milk feeding and total parenteral nutrition containing intralipid with medium-chain TG (6).…”

Section: Discussionmentioning

confidence: 61%

“…Several cases have been reported about congenital or fetal pleural effusion with chromosomal anomaly such as Down syndrome and Turner syndrome (8)(9)(10)(11). Most of these pleural effusion were chylothorax or associated hydrops fetalis (6,9). Hence, karyotyping is indicated in a fetus or newborn with isolated pleural effusion for the evaluation of associated chromosomal anomaly (8,11).…”

Section: Discussionmentioning

confidence: 99%

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Isolated Non-chylous Pleural Effusion in Two Neonates

et al. 2003

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Isolated pleural effusion, so called primary pleural effusion denotes a pleural effusion without documented etiology such as a cardiac, inflammatory, iatrogenic problem or fetal hydrops. Chromosomal anomaly such as Down syndrome may be associated with isolated pleural effusion. The content of the isolated pleural effusion is mostly chylous, and isolated non-chylous pleural effusion in neonate is rare. We experienced 2 cases of isolated non-chylous pleural effusion. They had neither cardiac problem nor other sign of hydrops fetalis. Imaging diagnosis was done by plain chest radiography and subsequent ultrasonogram. One of them was diagnosed to Down syndrome by karyotyping. They were fared well after diagnostic and therapeutic thoracentesis. We describe 2 cases of non-chylous pleural effusion and review a few English-language case reports of this entity.

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Section: Discussionmentioning

confidence: 61%

Section: Discussionmentioning

confidence: 99%

Isolated Non-chylous Pleural Effusion in Two Neonates

et al. 2003

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“…There was no evidence of hydrops in either case and one had a significant cardiac defect. Subsequently, several cases of trisomy 21 have been reported with bilateral chylothoraces and non-immune hydrops detected antenatally in the absence of cardiac defects (Rodeck et al, 1988;Ilagan et al, 1992;Ho et al, 1989;Yamamoto et al, 1996).…”

Section: Discussionmentioning

confidence: 99%

Variable levels of mosaicism for trisomy 21 in a non-immune hydropic infant with chylothorax

et al. 1999

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We report the first case of mosaic trisomy 21 with non‐immune hydrops fetalis and bilateral chylothoraces. Prenatal fetal blood karyotype analysis of 15 fetal cells revealed a 46,XX karyotype. Aggressive prenatal management, including fetal thoracocentesis and pleuro‐amniotic shunt, was performed. A clinical phenotype of Down syndrome was apparent after the gross oedema had subsided. Subsequent chromosome study of neonatal blood lymphocytes showed mosaic trisomy 21 with 23 per cent trisomic cells. Review of the initial fetal blood sample identified trisomy in 5 per cent of 134 cells. Follow‐up study at five months showed no trisomy 21 in 100 cells. This case illustrates the variable levels of mosaicism manifest in the peripheral blood of an infant with obvious Down syndrome phenotype, and the limitation of cytogenetic analysis of peripheral lymphocytes alone in prenatal and postnatal detection of low levels of mosaicism. Copyright © 1999 John Wiley & Sons, Ltd.

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“…Since then, based on a Medline search, a total of 11 cases of congenital pleural eusion associated with trisomy 21 have been reported in the English literature, 9 of which are in the form of congenital chylothorax [1,2,3,4,5,6,7]. In this report we present a further case of congenital pleural eusion associated with trisomy 21 which was also caused by chylothorax as in the majority of the other cases.…”

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confidence: 84%

Chylothorax and respiratory distress in a newborn with trisomy 21

et al. 2001

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Trisomy 21 may present with congenital chylothorax giving rise to symptoms of respiratory distress.Trisomy 21 or Down syndrome is the most common chromosome aberration in the newborn. Its high incidence has led to the better understanding of congenital diseases seen in association with this syndrome which commonly involve the cardiovascular and gastrointestinal systems. However, one of the less,if not the least, known associated condition is the pleural eusion, mostly congenital chylothorax, which is a disease characterised by lymphatic¯uid collection in the thoracic cavity of the newborn due to the leakage from the thoracic duct. We report on a newborn with trisomy 21 referred to our hospital with signs and symptoms of respiratory distress.The patient,a male newborn weighing 3,500 g at birth, was born to a 33-year-old woman gravida 3 para 3 at 39 weeks gestation by normal spontaneous vaginal delivery. He had respiratory ®ndings such as tachypnoea and cyanosis after birth and was therefore followed up for the ®rst 3 days of life after which he was discharged due to the disappearance of these symptoms. He was referred to our hospital on the 4th day of life with the same respiratory ®ndings. On admission, his body temperature was 36.7°C, pulse rate 134/min and respiratory rate 102/min. He was hypotonic and had atypical facies with low-set ears, epicanthic folds, high-arched palate and short ®ngers. Peroral cyanosis and poor breath sounds and rales were noted on the right side of the chest, together with pansystolic murmur in the mesocardiac area. The liver was 4 cm below the right costal margin.Initial laboratory ®ndings were as follows: Hb 23 g/dl, haematocrit 71%, WBC 12800/mm 3 , platelets 55000/ mm 3 , dierential blood count 52% neutrophils, 48% lymphocytes. CRP was negative. AP chest X-ray revealed a right sided pleural eusion. Thorax CT showed signi®cant pleural eusion in the right and minimal pleural eusion in the left side of the chest. ECHO showed a patent foramen ovale. Chromosome analysis revealed a 47 XY+21 karyotype. The cranial and abdominal ultrasound, TORCH screen, and thyroid hormones were all normal. Thoracentesis yielded a yellowish¯uid of pH 7.0 and density of 1010 with 6300/mm 3 leucocytes (100% lymphocytes), 90 mg/dl triglyceride, 35 mg/dl cholesterol and 2.4 g/dl protein. There were no bacteria on the gram stain and the culture was negative.Polycythaemia was treated with partial exchange transfusion. Thrombocytopenia resolved thereafter. He required two thoracenteses to relieve respiratory distress. After starting milk feeds, the yellow transparent thoracentesis¯uid became opaque and chyliform associated with increased triglycerides; therefore breast feeding was stopped and medium-chain triglyceride formula was commenced. After 6 days, the pleural eusion on the chest ®lms resolved gradually and disappeared. Breast feeding was reinitiated and the patient was discharged 14 days after the admission. He is still on breast milk and in a healthy state.The pathogenesis of congenital chylothorax is s...

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Congenital chylothorax in a patient with 21 trisomy syndrome

Cited by 12 publications

References 9 publications

Isolated Non-chylous Pleural Effusion in Two Neonates

Isolated Non-chylous Pleural Effusion in Two Neonates

Variable levels of mosaicism for trisomy 21 in a non-immune hydropic infant with chylothorax

Chylothorax and respiratory distress in a newborn with trisomy 21

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