S Bittmann, C Wittekindt, V Helmstaedter

Abstract

We present the case of a 4-year-old boy who was born with a large cervicofacial lymphatic malformation. It was partially resected in the first year of life and treated with Picibanil/OK-432 injections at the age of three because of recurrence. Eight months later the boy developed a fulminant streptococcal toxic shock syndrome with cardiac arrest. After resuscitation, high dose intravenous antibiotic therapy and artificial ventilation for 2 weeks, he remained with an extensive hypoxic brain damage. As Picibanil/OK-432 is a preparation of lyophilized, low virulence group A Streptococcus pyogenes cells of human origin, the question of a causative relationship for the tragic outcome arises. We therefore report the clinical course and discuss the action and possible side effects of Picibanil/OK-432.

我們報導一例出生時即有頸部巨大淋巴管畸形的四歲男孩，該病人在出生後的頭一年作了病灶的局部切除，並在其3歲時因為病情反復而應用沙培林/OK-432的注射治療。8個月後該男孩出現暴發性鏈球菌中毒性休克並一度出現心跳驟停。經過急救後，雖給予2周的大劑量抗生素靜脈治療和人工通氣，但該病人仍存廣泛的缺氧性腦損傷。由於沙培林是一種人源細胞來源的A組溶血性鏈球菌凍幹低毒性製劑，其與該病人的不良預後的關係尚需置疑。因此，我們報導此例病人的臨床病程，並討論沙培林/ OK-432可能存在的不良反應。