Debuf Marie-Julie, Claeys Tania, Stalens Jean-Philippe, Cornette Luc
Department of Pediatrics, Université Catholique de Louvain, Brussels, Belgium.
Department of Pediatrics, AZ Sint-Jan Brugge-Oostende, Bruges, Belgium.
J Med Case Rep. 2017 Jun 16;11(1):160. doi: 10.1186/s13256-017-1318-z.
Hematochezia is a frequent symptom in early infancy. However, it occurs very rarely within the immediate neonatal period, and its occurrence before any oral intake is particularly rare. Because of the "congenital" presentation of hematochezia in our patient, we initially considered our case to be a non-classical, potentially severe type of food protein-induced allergic proctocolitis. This diagnosis needs to be confirmed by an abnormal oral challenge test once the hematochezia has disappeared. If such a challenge cannot demonstrate an allergic origin, then the etiology of the hematochezia could be a neonatal transient eosinophilic colitis. Only two similar cases have been described so far.
We report the case of a black baby boy of African origin born at 36 weeks 5 days of gestational age who presented with massive hematochezia immediately after birth. A rectosigmoidoscopy revealed a severe inflammation associated with diffuse eosinophilic infiltration on biopsy. His clinical outcome was favorable after introduction of an amino acid formula diet. We initially considered our case to be a non-classical, potentially severe type of food protein-induced allergic proctocolitis but reintroduction of standard formula milk at the age of 3 months was successful. So, our patient is the first newborn in Europe who fits the diagnosis of "neonatal transient eosinophilic colitis."
We discuss the possible etiology of "congenital" eosinophilic inflammation of the distal colon and conclude that hematochezia in well-looking neonates, in the absence of negative challenge tests later on, is more likely to be a neonatal transient eosinophilic colitis than an allergic proctocolitis. This new entity could be more frequent than previously thought, changing our medical care strategies for this kind of neonatal symptom.
便血是婴儿早期常见的症状。然而,在新生儿期内便血非常罕见,在任何经口摄入之前出现便血则尤为罕见。由于我们的患者便血呈现“先天性”表现,我们最初认为该病例是非典型的、可能严重的食物蛋白诱导的过敏性直肠结肠炎。一旦便血消失,这一诊断需要通过异常的口服激发试验来证实。如果这样的激发试验不能证明过敏起源,那么便血的病因可能是新生儿短暂性嗜酸性粒细胞性结肠炎。迄今为止,仅描述了两例类似病例。
我们报告一例非洲裔黑人男婴,孕36周5天出生,出生后立即出现大量便血。直肠乙状结肠镜检查显示严重炎症,活检发现弥漫性嗜酸性粒细胞浸润。引入氨基酸配方奶后,他的临床结局良好。我们最初认为该病例是非典型的、可能严重的食物蛋白诱导的过敏性直肠结肠炎,但3个月大时重新引入标准配方奶成功。因此,我们的患者是欧洲首例符合“新生儿短暂性嗜酸性粒细胞性结肠炎”诊断的新生儿。
我们讨论了远端结肠“先天性”嗜酸性粒细胞炎症的可能病因,并得出结论:外表良好的新生儿出现便血,若后期激发试验无阴性结果,更可能是新生儿短暂性嗜酸性粒细胞性结肠炎而非过敏性直肠结肠炎。这种新的疾病可能比之前认为的更为常见,这将改变我们针对这类新生儿症状的医疗护理策略。