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Ann Thorac Surg 2004;77:2213-2215
© 2004 The Society of Thoracic Surgeons
a Intensive Care Unit, Royal Children's Hospital, Melbourne, Australia
b Cardiac Surgery Unit, Royal Children's Hospital, Melbourne, Australia
c Department of Cardiology, Royal Children's Hospital, Melbourne, Australia
Accepted for publication June 6, 2003.
* Address reprint requests to Dr Tibballs, Intensive Care Unit, Royal Children's Hospital, Parkville, Melbourne, Australia 3052
e-mail: james.tibballs{at}rch.org.au
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Abstract |
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Introduction |
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Alternative therapy with somatostatin, or a long-acting analogue, octreotide, is controversial being reported as "successful" [1–8] or as "unsuccessful" [9, 10].
We report a case of rapid short-term suppression of chylothorax occurring in a newborn postcardiac surgery and in whom lymphangiectasia was demonstrated on lung biopsy.
A 2.1 kg 36-weeks gestation infant had atrial isomerism, double outlet right ventricle, complete atrio-ventricular septal defect, obstructed total anomalous pulmonary venous drainage (TAPVD), severe pulmonary stenosis, hypoplasia of the left ventricle, left superior vena cava, and right aortic arch. Mechanical ventilation and prostaglandin infusion were required within minutes of birth. Chest radiograph showed a coarse bilateral reticular pattern considered secondary to TAPVD (Figure 1). Hydrops was not present.
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Nasogastric feeding was commenced on day 4 with a formula feed (S26, Wyeth Nutrition, Baulkham Hills, NSW, Australia). Only intermittent feeding was possible over days 4–6 due to vomiting associated with bowel malrotation demonstrated by a barium study. On day 6, 225 mL of straw-colored opaque fluid drained from the chest and continued at 110–130 mL per day. The fluid was sterile with a white cell count of 500 x 106/L of which 98% were lymphocytes and had a triglyceride level of 1.8 mmol/L in contrast to a serum level of 0.6 mmol/L. No fluid was detected in the left chest throughout. Feeding was changed on day 6 to Monogen (Scientific Hospital Supplies, Somerton Park, Australia) whose lipid fraction is 93% medium chain triglycerides and 7% long chain triglycerides. Intravenous 50% dextrose was administered from day 2 and amino acids and lipids were administered from day 7. Nasogastric feeding was ceased on day 7.
On day 10 an intravenous infusion of Octreotide (Sandostatin, Novartis Pharmaceuticals Australia Pty Ltd) was commenced at 3 µg · kg–1 · h–1 and increased to 5 µg · kg–1 · h–1 on day 11. This coincided with a reduction of drainage to zero within 16 h. On day 13 the chest drain was removed and the infusion of octreotide ceased because no drainage had occurred for 48 h. Echocardiographic studies on day 10 and a cardiac catheter study on day 12 excluded thrombosis of the central veins and recurrent obstruction of the pulmonary veins. The central venous pressure throughout days 2–11 was 3–9 mm Hg. On day 13 fluid reaccumulated in the right pleural cavity. The pulmonary reticular pattern failed to resolve and an alternative diagnosis to TAPVD was sought. Biopsy of the right lung revealed lymphangiectasia. Octreotide was not recommenced. The chest drain left in situ after biopsy drained 115–230 mL per day until day 16 when the infant succumbed after treatment, considered to be futile, was withdrawn. A postmortem examination was not performed. Chest drainage throughout is shown in Figure 2.
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Comment |
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The efficacy of octreotide therapy for chylothorax is controversial. In this case, chyle drainage ceased completely within 16 h of commencement and did not reoccur until after cessation of octreotide 48 h later. This is consistent with the experimentally observed rapid onset of action of the hemodynamic effects which are evident within minutes [12]. In nine other cases involving infants or children, which resemble ours, chyle loss abated within hours of administration of somatostatin or octreotide in one case [8], stopped after 2–5 days, and allowed removal of chest drains in all cases within 4–6 days [5, 7, 8]. In "successes" of octreotide therapy the dose was variable from 0.5–4 µg · kg–1 · h–1 [5, 7]. Venous hypertension was known to be present in six of these cases [5] and to be absent in two [7].
We question the efficacy of octreotide claimed in some other pediatric case reports when chyle drainage persisted for as long after the initiation of treatment as with traditional treatments. In three "successful" cases of octreotide or somatostatin therapy [4, 6], chyle loss persisted for 10, 15, and 24 days after its commencement. Because these times are similar to the average times (approximately 14 days, range 3–66, n = 40) required for spontaneous resolution during treatment with TPN or oral medium-chain triglycerides [17, 18], it is questionable that the cessation of chyle loss was truly the result of somatostatin therapy. Arguably these three cases may be reclassified as "unsuccessful" octreotide treatment irrespective of whether the dose was high at 3.5–7 µg · kg–1 · h–1 in one case [4] or low at 10–40 µg/kg/d subcutaneously in the remaining two cases [6]. It is not known whether venous hypertension was present. Indeed octreotide therapy in one adult case in which lymph ceased 15 days after the start of eight days of therapy was described by the authors as "noneffective" [9]. Another adult case was notable for the "inefficacy" of subcutaneously administered analogue of somatostatin [10].
Congenital pulmonary lymphangiectasia is a rare condition occurring either as an isolated condition, as part of a generalized condition, or in association with heart disease particularly anomalous pulmonary venous drainage and common pulmonary vein atresia [19]. It may be unilateral. The etiology of chylothorax in our case may have been surgery, lymphangiectasia, or both, although its restriction to the right chest suggests that surgery was, at least, partially responsible. There are no previous reports of the use of somatostatin or analogues in the treatment of lymphangiectasia.
No serious complications or adverse effects have been reported with short-term use of somatostatin or analogues although disturbance of gastrointestinal function and perturbations of glucose metabolism may occur. Prolonged use may cause cholelithiasis [20].
Although it appears appropriate to employ somatostatin or an analogue early after recognition of chylothorax after surgery, its efficacy is yet to be proven. Differences in outcome may be related to different dosing. The dose remains empirical but we recommend approximately 3–5 µg · kg–1 · h–1 intravenously for up to 3–5 days. Causes of venous obstruction should be sought and relieved.
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This article has been cited by other articles:
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  S.-y. Chan, W. Lau, W. H.S. Wong, L.-c. Cheng, A. K.T. Chau, and Y.-f. Cheung Chylothorax in Children After Congenital Heart Surgery Ann. Thorac. Surg., November 1, 2006; 82(5): 1650 - 1656. [Abstract] [Full Text] [PDF]
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