A large cervical lymphatic malformation in a neonate successfully treated with Kampo medicine

Abstract

Introduction: Recently the application of Kampo, or Japanese traditional medicine, to lymphatic malformation (LM) has been reported, but there are no reports of its effects against neonatal LM. We herein report a case of a neonatal huge cervical LM successfully treated with eppikajutsuto and ogikenchuto without tracheostomy or sclerotherapy. Patient concerns: A baby boy with a huge cyst in his right neck was delivered by Caesarean section without a prenatal diagnosis. The cyst extended and slightly compressed the trachea on the 7th day after birth. Diagnosis: The patient was diagnosed with LM by ultrasonography. Interventions: Although we planned sclerotherapy with OK-432 followed by tracheostomy, his parents refused tracheostomy. Therefore, we started the administration of eppikajutsuto (0.5 g/kg/d) on the 26th day of life instead of sclerotherapy. As the cyst was slightly enlarged at the beginning of eppikajutsuto administration, we increased the dose of eppikajutsuto to 0.75 g/kg/d and added ogikenchuto (0.4 g/kg/d), expecting greater regression of the lesion than with the initial regimen. Outcomes: After a few days, the cyst started to diminish in size. Six months later, magnetic resonance imaging showed remarkable regression of the LM without tracheal compression. Lessons: The combination of eppikajutsuto and ogikenchuto was thus found to be effective in the treatment of a neonatal LM. This is the first report to demonstrate that the dose-escalation of eppikajutsuto may therefore be effective without any severe adverse effects, even in neonates.

Effect of Japanese Kampo medicine, eppikajutsuto, in patients with lymphatic malformation

“…To the best of our knowledge, this is the first study to report the effect of eppikajutsuto (EKJT) on reducing lesion volume in mediastinal LM cases in Japan [3] . We also reported a case with a marked reduction in cervical LM after increasing the dosage of EKJT [4] . Other cases of LM reduction with EKJT have also been reported [5,6] .…”

“…[3] We also reported a case with a marked reduction in cervical LM after increasing the dosage of EKJT. [4] Other cases of LM reduction with EKJT have also been reported. [5,6] However, no evidence has been established to evaluate its efficacy, dose dependency, and safety.…”