Morimoto Mari, Sawada Hirofumi, Yodoya Noriko, Ohashi Hiroyuki, Toriyabe Kuniaki, Hanaki Ryo, Sugiura Katsumi, Toyoda Hidemi, Matsushita Kohei, Koike Yuhki, Otake Kohei, Inoue Mikihiro, Uchida Keiichi, Imai Hiroshi, Mitani Yoshihide, Maruyama Kazuo, Komada Yoshihiro, Ikeda Tomoaki, Hirayama Masahiro
Department of Pediatrics, Mie University Graduate School of Medicine, Tsu, Japan.
Anesthesiology and Critical Care Medicine, Mie University Graduate School of Medicine, Tsu, Japan.
Front Pediatr. 2020 Jul 14;8:352. doi: 10.3389/fped.2020.00352. eCollection 2020.
Severe neonatal gastrointestinal diseases such as necrotizing enterocolitis or spontaneous intestinal perforation are potentially lethal conditions which predominantly occur in preterm infants. Cytomegalovirus (CMV), which is known to cause congenital and acquired infections in the newborns, has also been implicated in such severe gastrointestinal diseases in premature infants. However, the pathogenic role of CMV and effect of antiviral therapy in severe gastrointestinal disease in premature neonates is currently unclear. We present an infant, born at 26-weeks' gestation, presented with progressive dyspepsia and abdominal distention after the closure of the symptomatic patent ductus arteriosus at the day of life (DOL) 4, requiring the emergent surgery for ileal perforation at the DOL8. After the surgery, abdominal symptoms persisted and the second emergent surgery was performed for the recurrent ileal perforation at DOL17. Even then the abdominal symptoms prolonged and pathological examination in the affected intestine at the second surgery showed CMV inclusion body. Immunoreactivity for CMV antigen was detected in the specimen at the first surgery on DOL8. Blood and urinary CMV-DNA were detected at DOL28. CMV-DNA was also detected in the dried umbilical cord which was obtained within a week from birth. A 6-week course of intravenous ganciclovir (12 mg/kg/day) was started at DOL34 and then symptoms resolved along with decreasing blood CMV-DNA. Pathological findings characteristic of CMV were not detected in the resection specimen at the ileostomy closure at DOL94. These observations indicate that anti-CMV therapy may be beneficial for some premature infants with severe CMV-associated gastrointestinal diseases and warrants further studies focusing on pathogenic role, diagnosis, treatment and prevention of this underrecognized etiology of severe gastrointestinal diseases particularly in premature neonates.
严重的新生儿胃肠道疾病，如坏死性小肠结肠炎或自发性肠穿孔，是主要发生在早产儿中的潜在致命病症。已知巨细胞病毒（CMV）可导致新生儿先天性和后天性感染，也与早产儿的此类严重胃肠道疾病有关。然而，CMV在早产儿严重胃肠道疾病中的致病作用以及抗病毒治疗的效果目前尚不清楚。我们报告一例妊娠26周出生的婴儿，出生后第4天有症状的动脉导管未闭关闭后出现进行性消化不良和腹胀，出生后第8天因回肠穿孔需要紧急手术。手术后，腹部症状持续存在，出生后第17天因复发性回肠穿孔进行了第二次紧急手术。即便如此，腹部症状仍持续，第二次手术时对受影响肠道的病理检查显示有CMV包涵体。出生后第8天第一次手术的标本中检测到CMV抗原的免疫反应性。出生后第28天检测到血液和尿液中的CMV-DNA。出生后一周内采集的干燥脐带中也检测到CMV-DNA。出生后第34天开始了为期6周的静脉注射更昔洛韦（12mg/kg/天）治疗，随后症状随着血液中CMV-DNA水平的下降而缓解。出生后第94天回肠造口关闭时的切除标本中未检测到CMV特征性的病理表现。这些观察结果表明，抗CMV治疗可能对一些患有严重CMV相关胃肠道疾病的早产儿有益，并且有必要进一步开展研究，聚焦于这种严重胃肠道疾病（尤其是早产儿）未得到充分认识的病因的致病作用、诊断、治疗和预防。
