Perinatal/Neonatal Case Presentation - Nature

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CASE PRESENTATIONS. The mother was referred to our hospital because of fetal intrauterine growth retardation at 24 weeks and 5 days of gestation. The fetus ...
Perinatal/Neonatal Case . . . Presentation . . . . . . . .

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Treatment of Chylothorax in a Premature Infant Using Somatostatin Maki Goto, MD Kazuya Kawamata Masanao Kitano Ken Watanabe Yosihide Chiba, MD, PhD

We describe a case of spontaneous chylothorax in a premature infant successfully treated by octreotide after other forms of conservative therapy were not effective.

Journal of Perinatology (2003) 23, 563–564. doi:10.1038/sj.jp.7210975

INTRODUCTION Chylothorax in an infant is a comparatively rare disease. However, when difficulty in treatment arises, the disease may induce breathing difficulty and inadequate nutrition. We encountered a case of chylothorax in a premature infant that improved after continuous intravenous injection of octreotide after other conservative therapies were not effective. CASE PRESENTATIONS The mother was referred to our hospital because of fetal intrauterine growth retardation at 24 weeks and 5 days of gestation. The fetus had no anomaly and no pleural effusion. Ultrasonic tomography demonstrated a large placental hematoma. Emergency Caesarean section was carried out because of fetal distress at 26 weeks and 0 days of gestation. The male infant weighed 467 g ( 2.6 SD) at birth and the Apgar score was 1 at 1 minute, 2 at 5 minute, and 4 at 10 minutes. He was intubated immediately after birth and exogenous pulmonary surfactant was instilled. He was managed using a mechanical ventilator and an umbilical catheter was placed. We started mother’s-own breast milk feeding 2 days

Department of Perinatology and Pediatrics, National Cardiovascular Center, Osaka, Japan. Address correspondence and reprint requests to Maki Goto, Department of Perinatology, National Cardiovascular Centre, 5-7-1, Fujisirodai, Suita, Osaka 565-8565, Japan.

after birth. Chest X-ray signs of bronchopulmonary dysplasia were recognized 14 days after birth. At 16 days of age, chest X-ray demonstrated a pleural effusion in the left thoracic cavity (Figure 1). Nonsurgical management consisted of evacuating the pleural space by tube thoracostomy and reducing chyle flow. Chest fluid culture was negative. Chemical analysis of the chest fluid demonstrated total protein (0.9 g/dl), LDH (71 U/l), and triglycerides (43 mg/dl). Cytologic smear of the fluid showed tissue cells and lymphocytes. The respiratory condition stabilized at 18 days after birth and the baby was extubated 22 days after birth. As the pleural effusion became muddy white after breast milk feeding 23 days after birth, we changed to medium-chain triglycerides (MCT) formula. After changing the MCT-formula, the pleural effusion was no longer milky, but the flow remained abundant (60 ml/24 hours). We did not stop the milk feeding because of concern regarding necrotizing enterocolitis due to his immature physiology. At 28 days after birth, we intubated the infant again due to apnea. The pleural effusion flow remained unchanged. At 36 days after birth, we decided to use octreotide. After obtaining informed consent from his parents, octreotide (0.3 mg/ kg/hour) was started by the continuous intravenous infusion, and the chest drainage stopped on the second day (Figure 2). Treatment was continued for a total of 3 days. No side effects, such as hypotension or hyperglycemia were observed throughout the whole treatment. We removed the chest tube 47 days after birth and he was extubated 2 days later. Mother’s milk feeding was reinstituted 51 days after birth, and the pleural effusion did not recur.

COMMENT Chylothorax in an infant is a comparatively rare disease. However, when there is difficulty treating this condition, breathing difficulty will occur. Patients may present with massive lymph drainage that will induce critical losses of fluid, lymphocytes, protein, coagulation factors, and antibodies, thus increasing morbidity and mortality.1 Conservative therapy currently includes the use of a low-fat diet with MCT, or discontinuing enteral feeding and replacing with parenteral nutrition.2,3 Cessation of oral intake stops food and fluid absorption from the gastrointestinal (GI) tract and also stops the stimulation of GI secretions. Less lymphatic volume is thus offered for absorption and flow into the thoracic duct. Lack

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Somatostatin for Treatment of Chylothorax

Figure 3. Effect of intravenous somatostatin on chyle output.

Figure 1. On chest X-ray, BPD findings were recognized, and there was pleural effusion in his left side thoracic cavity.

on its inhibitory GI effects. In addition to reducing hepatic, splanchnic, and portal blood, octreotide has multiple known inhibitory effects on the GI tract. These include inhibition of pancreatic secretion, decreased gallbladder contractility, slowing of intestinal transit time, inhibition of absorption of glucose and amino acids, and inhibition of GI peptide hormone secretion.5,6 There are scattered case reports that support its use in the treatment of thoracic duct injury.1–6 However, there is no previous report describing the effects of octreotide in treating chylothorax in a premature infant. We report a premature infant in whom introduction of somatostatin immediately diminished chyle production, without observed adverse effects.

References

Figure 2. Using octreotide, chest X-ray confirmed that there was no reaccumulation of fluid.

of oral intake thus offers an added advantage in the attempt to conservatively heal the chylothorax.4 However, in a premature infant, lack of oral intake may increase the risk of necrotizing enterocolitis. Somatostatin has the potential to reduce chyle flow (Figure 3). Its mechanism of action remains to be identified but likely centers

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1. Rimensberger PC, Muller-Scenker B, Kalangos A, Beghetti M. Treatment of a persistent postoperative chylothorax with somatostatin. Ann Thorac Surg 1998:253–4. 2. Kelly RF, Shumway SJ. Conservative management of postoperative chylothorax using somatostatin. Ann Torac Surg 2000; 1944–5. 3. Ulibarri JI, Sanz Y, Fuentes C, Mancha A, Aramendia M, Sanchez S. Reduction of lymphorrhagia from ruptured thoracic duct by somatostatin. Lancet 1990; 258. 4. Demos NJ, Kozel J, Scerbo JE. Somatostatin in the treatment of chylothorax. Chest 2001; 964–6. 5. Markham KM, Glover JL, Welsh RJ, Lucas RJ, Bendic PJ. Octreotide in the treatment of thoracic duct injuries. The American Surgeon. 2000;1165–7. 6. Cheung Y-F, Leung MP, Yip M-M. Octreotide for treatment of postoperative chylothorax. J Pediatr 2001; 157–9.

Journal of Perinatology 2003; 23:563–564