We describe two cases of premature infants who developed clinical and radiologic evidence of gastric lactobezoars within the same month in our neonatal intensive care unit while both were receiving medium-chain triglyceride-rich formula as part of the management of chylothoraces.
A lactobezoar is an aggregation of milk or formula-based precipitant in the gastric or intestinal lumen. Although they are rare phenomena, case reports have been described as early as the 1950s.1 Lactobezoars may present in a variety of forms with complications including obstructive symptoms, gastric irritation and perforation.2,3 Previous case series have reported prematurity, high-caloric density formulas (>80 kcal/100 ml), casein predominant formula (>60%) and formulas containing medium-chain triglycerides as associated factors in the formation of lactobezoars.2 They are most often diagnosed by radiographic imaging in the setting of an appropriate clinical scenario. Previously described treatment regimens include stopping enteral feeds and placing patients on parenteral nutrition, along with gastric decompression until resolution.2 Lactobezoars may self-resolve without additional intervention, which has led some to theorize that lactobezoars are underdiagnosed.2 A recent case series identifies only 96 reports of lactobezoars since 1959.2
We describe two cases of premature infants who developed lactobezoars in our neonatal intensive care unit. Both infants were receiving medium-chain triglyceride-rich formula as management of chylothoraces. We hypothesize that medium-chain triglyceride-rich formula may be associated with an increased risk of lactobezoar formation due to altered nutritional content.
Patient 1 is a 31-week premature female infant with a prenatal course that was notable for the development of a fetal arrhythmia 3 days prior to delivery. At delivery the infant was noted to be hydroptic and in significant respiratory distress. She was intubated and had three chest tubes placed in the first hours of life. During her hospitalization, further evaluation was notable for multifocal atrial tachycardia, patent ductus arteriosus (PDA), bilateral chylothoraces, respiratory distress syndrome, bilateral multicystic/dysplastic kidneys and long-segment coarctation of the aorta. Owing to her bilateral chylothoraces, enteral feedings were not given and the patient was given total parenteral nutrition to limit lymphatic drainage. The chest tubes were removed after resolution of drainage. On day of life (DOL) 13, she was started on continuous enteral feeds with medium-chain triglyceride-rich formula (20 kcal/oz. Enfaport, Mead Johnson, Glenview, IL, USA). On DOL 19, the infant developed bloody stools without emesis, fevers, change in hemodynamic status and/or change in abdominal exam. Due to the concern for necrotizing enterocolitis, radiographic imaging was obtained and was notable for extensive mottled lucency over the gastric area. Feedings were stopped and the infant was again treated with total parenteral nutrition. Serial abdominal radiographs were obtained. She did not have any further bloody stools and radiographic imaging showed progressive improvement of the mottled lucency consistent with a lactobezoar. On DOL 30, she was restarted on fortified enteral feeds of medium-chain triglyceride-rich formula (24 kcal/oz.). She was eventually transitioned to enteral breast milk feeds that were well tolerated. The rest of her hospitalization was unremarkable for any further feeding intolerance. She was discharged home on DOL 65 taking full volume breast milk feedings by mouth.
Patient 2 is a 25-week gestation premature female whose prenatal course was uncomplicated prior to preterm labor. She initially received total parenteral nutrition that was slowly weaned as nasogastric feedings of breast milk were advanced. Her course was complicated by chronic lung disease, intraventricular hemorrhage, methicillin-sensitive Staphylococcus aureus bacteremia, retinopathy of prematurity, gastroesophageal reflux and PDA requiring ligation on DOL 31. Postoperatively from the PDA ligation she was tolerating full enteral feedings of fortified breast milk (27 kcal/oz.). On DOL 50, she developed a left-sided chylothorax that was evacuated by thoracentesis. Enteral feedings were stopped and she was started on total parenteral nutrition. On DOL 52, nasogastric feedings were restarted with a medium-chain triglyceride-rich formula (20 kcal/oz. Enfaport, Mead Johnson) to limit lymphatic drainage, and by DOL 55 she was tolerating full volume fortified feedings of medium-chain triglyceride-rich formula (24 kcal/oz.). On DOL 58 she presented with non-bilious emesis, following which feedings were stopped and total parenteral nutrition was restarted. She was noted to have a palpable abdominal mass in her left upper quadrant and mild tenderness. Abdominal radiograph revealed bubbly lucencies in the left upper quadrant, initially concerning for pneumatosis (Figure 1), but subsequent abdominal radiographs were more suggestive of a lactobezoar (Figure 2). She had no further emesis and, on DOL 59, feedings of fortified medium-chain triglyceride-rich formula were resumed (24 kcal/oz.) and were well tolerated. She had no further feeding intolerance during her hospitalization. She was discharged home on DOL 156 taking full volume infant formula feedings by mouth.
Enfaport is an infant formula that is designed for infants with chylothorax or long-chain 3-hydroxy-CoA dehydrogenase (LCHAD) deficiency.4 It is a high medium-chain triglyceride formula and is most often used in the setting of chylothorax in our institution. The lipid content of Enfaport formula is 84% medium-chain triglyceride oil, with the rest made up of a soy oil blend. The protein content is a mixture of calcium caseinate and sodium caseinate. The carbohydrate content consists of corn syrup solids. It is prepared and packaged with a caloric concentration of 30 kcal/oz., composed of 41% carbohydrate, 45% fat and 14% protein (35 g/l), which is the highest protein content of commonly manufactured formula preparations.5
Medium-chain triglyceride-based formulas have been used in the setting of chylothorax to utilize the altered absorption of medium-chain triglycerides directly into the portal venous system, bypassing the lymphatic flow and theoretically increasing the likelihood of healing the source of lymphatic leakage.6, 7, 8 In both of our patients, following elimination of feedings and subsequent use of medium-chain triglyceride-rich formula, the chylothoraces resolved without further re-accumulation.
The combination of prematurity and systemic illness in the setting of enteral formula with high-casein content, that is medium-chain triglyceride-based, likely resulted in the formation of the lactobezoars. Both infants did not suffer any untoward long-term affects from the lactobezoars and went on to tolerate full enteral feeds. Increased awareness of the possibility of the development of lactobezoars in infants receiving medium-chain triglyceride oil-based, high-casein formulas such as Enfaport may lead to timely diagnosis and decrease unnecessary evaluations and/or treatment interventions in the appropriate clinical setting.
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We thank Dr James W Collins for his assistance.
The authors declare no conflict of interest.
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Prahl, M., Smetana, D. & Porta, N. Lactobezoar formation in two premature infants receiving medium-chain triglyceride formula. J Perinatol 34, 634–635 (2014). https://doi.org/10.1038/jp.2014.78
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