Ismail Allyzain, Mboma Lazaro, Ngotta Victor, Nyamuryekung'e Masawa
The Aga Khan University, East Africa Medical College, P. O. Box 2289, Barack Obama Drive, Dar es Salaam, Tanzania.
Muhimbili University of Health and Allied Sciences, P. O. Box 65001, Dar es salaam, Tanzania.
Int J Surg Case Rep. 2021 Jul;84:106159. doi: 10.1016/j.ijscr.2021.106159. Epub 2021 Jun 30.
In this case report from Muhimbili National Hospital, Dar es salaam, Tanzania, we present the unexpected findings of anorectal malformation, colonic atresia, and intestinal malrotation in a 2-day old neonate. This combination is exceedingly rare, with only case reports published in the literature. We describe the challenges in diagnosis and offer our insights based on this experience and review of the literature.
Our patient was a male born at term, weighing 2600 g, diagnosed clinically with a high anorectal malformation. He was planned for colostomy, and we unexpectedly found a collapsed descending colon. Exploration revealed intestinal malrotation and three segments of type I colonic atresia from the mid transverse colon to the sigmoid colon in addition to the high anorectal malformation.
Creating a colostomy in a high anorectal malformation and failure to identify proximal intestinal atresia would result in potentially devastating consequences. Colonic atresia and anorectal malformation will both present as large bowel obstruction. In the extremely rare situation, when occurring in combination, the obvious clinical diagnosis of anorectal malformation will mask the clinical suspicion of the possibility of colonic atresia. Finding a distal bowel air bubble above the pubococcygeal line on an invertogram is useful in identifying proximal atresia preoperatively.
The current report emphasizes the importance of maintaining an awareness of possible associated colonic atresia in neonates with anorectal malformation. An invertogram and intraoperative finding of a collapsed descending colon should prompt evaluation for a proximal obstructing lesion.
在这份来自坦桑尼亚达累斯萨拉姆穆希姆比利国家医院的病例报告中,我们呈现了一名2日龄新生儿出现肛门直肠畸形、结肠闭锁和肠旋转不良的意外发现。这种组合极其罕见,文献中仅有病例报告发表。我们描述了诊断过程中的挑战,并基于此经验和文献回顾提供我们的见解。
我们的患者为足月出生的男性,体重2600克,临床诊断为高位肛门直肠畸形。计划为其行结肠造口术,而我们意外发现降结肠塌陷。探查发现除高位肛门直肠畸形外,还有肠旋转不良以及从中横结肠至乙状结肠的三段I型结肠闭锁。
在高位肛门直肠畸形患者中进行结肠造口术且未识别近端肠闭锁会导致潜在的灾难性后果。结肠闭锁和肛门直肠畸形均会表现为大肠梗阻。在极为罕见的情况下,当两者合并出现时,明显的肛门直肠畸形临床诊断会掩盖对结肠闭锁可能性的临床怀疑。在倒位造影上耻骨尾骨线以上发现远端肠气泡有助于术前识别近端闭锁。
本报告强调了对肛门直肠畸形新生儿可能合并结肠闭锁保持警惕的重要性。倒位造影以及术中发现降结肠塌陷应促使对近端梗阻性病变进行评估。