BackgroundNeck lymphangioma is the most common lymphatic malformation of the newborn, representing about 5% of benign tumours of infancy and childhood. Due to its action on angiogenesis and cellular growth, the use of sirolimus (m-Tor inhibitor) has been proposed for treatment, as it has produced satisfactory results in some studies in children and in a few cases in neonatology.PurposeTo describe the use of sirolimus as treatment for a left laterocervical lymphangioma in a newborn.Material and methodsWe report the case of a term male newborn (3.3 kg, 51 cm, 0.22 m2) with a left laterocervical mass (13 cm) diagnosed on a prenatal ultrasound. According to the neonatal cases in the literature, an oral solution of sirolimus 1 mg/mL was started at 0.8 mg/m2/day (divided into 12 hourly doses) for 10 days of life. Sirolimus plasma levels were monitored (LC/MS/MS), with a desired target of 4–8 µg/L, as well as adverse effects and the evolution of the mass.Results Day Dose Levels (ng/mL) Triglycerides (mg/dL) Cholesterol-LDL (mg/dL) 10.08 mg bid672860.08 mg bid2389941018.531999128.7382140.1 mg qd190.1 mg qd8103123400.1 mg qd4.1167470.15 mg qd550.15 mg qd7.418096The first 2 months of treatment are described. On day 6, treatment was interrupted owing to high plasma levels. On day 14, sirolimus was reintroduced at a lower dose and once daily schedule (estimated elimination half-life of 33 hours, concordant with the neonatal literature). Posterior levels were correct and the dose was titrated according to them and the weight gain of the child. Regarding side effects, the patient experienced hypertriglyceridaemia and hypercholesterolaemia, with unaltered hepatic and renal functions associated with high sirolimus levels. He also had occasional nausea and vomiting that were appropriately managed. Posterior controls showed normalisation of triglycerides and cholesterol levels. From the beginning of treatment, the cervical mass showed a progressive reduction in size and a marked reduction in consistency.ConclusionSirolimus can be a useful option for the treatment of lymphatic malformations with few short term side effects. More data are needed to characterise the pharmacokinetics of sirolimus in the neonatal population, in order to define optimal dosing.No conflict of interest
Results A total of 177 patients were reviewed, with a mean age of 63.4±16.4 and 32.8% were women. Almost half of the patients 48.6% (n=86) had an ostearticular infection: bacteriemia accounted for 36.2% (n=64). The rest of the infections were related to the central nervous system 3.4% (n=6), endovascular system 3.4% (n=6) and others 8.4% (n=15).Patients excluded: eight due to neutropaenia (n=169), 15 due to thrombocytopaenia (n=162) and 14 due to AKI (n=163) prior to vancomycin therapy.Neutropaenia was developed in seven patients (=1:24), thrombocytopaenia in 12 patients (=1:14) and AKI in 26 patients (=1:6). The prevalence of nephrotoxicity is described as common (1:100-1:10) in the summary product characteristics (SPC). However, neutropaenia and thrombocytopaenia are classified as rare undesirable effects (1:10.000-1:1.000). Conclusion The prevalence of AE related to vancomycin therapy is higher than reported in SPC. In our study neutropaenia was reported in 7:169 patients, thrombocytopaenia in 12:162 and AKI in 26:163.The difference between SPC and our clinical practice is considerable. However, it should be noticed that only patients monitored by PD were reviewed, and therefore the number of patients included is low. It is of high importance to continue reporting any AE related to vancomycin therapy to the appropriate pharmacovigilance institution in order to better understand the toxic profile of the drug. REFERENCES AND/OR ACKNOWLEDGEMENTSNo acknowledgements.No conflict of interest.
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