Mboyo A, Monek O, Massicot R, Martin L, Destuynder O, Lemouel A, Aubert D
Department of Pediatric Surgery, Centre Hospitalier de Besançon, Hôpital Saint-Jacques, 2, place Saint-Jacques, 25030 Besançon Cedex, France.
Pediatr Surg Int. 1997 Jul;12(5-6):452-4. doi: 10.1007/BF01076967.
A case of cystic rectal duplication revealed on day 2 of life by a low intestinal occluding syndrome is reported. Radiologic imaging (ultrasonography, cystography, rectography) showed a large, retrorectal liquid formation in the pelvis and abdomen, with pelvic compression of the terminal alimentary canal and lower urinary tract. Magnetic resonance imaging demonstrated a liquid formation with clearly defined edges and no medullary involvement, thus ruling out the possibility of a previous meningeal hernia. Biological markers were within normal limits. On day 4, a 9 x 6-cm cystic rectal duplication was removed, followed by a temporary colostomy. Pathologic examination demonstrated typical rectal architecture with ciliated cells. Radiologic and clinical findings at 2-month follow-up were reassuring. This case report is exceptional for the following reasons: (1) As a rule, rectal duplications are relatively rare (70 cases reported in the literature); (2) The means of disclosing a neonatal rectal duplication is unusual (4 cases reported in the literature); (3) The volume of the malformation was considerable; and (4) Heterotopic ciliated epithelium was present.
报告了1例出生第2天因低位肠梗阻综合征而发现的直肠重复畸形囊肿病例。放射学成像（超声检查、膀胱造影、直肠造影）显示盆腔和腹部有一个巨大的直肠后液性肿物，终末消化道和下尿路受到盆腔压迫。磁共振成像显示液性肿物边界清晰，无髓质受累，从而排除了既往脑膜膨出的可能性。生物学标志物在正常范围内。第4天，切除了一个9×6厘米的直肠重复畸形囊肿，随后进行了临时结肠造口术。病理检查显示为具有纤毛细胞的典型直肠结构。2个月随访时的放射学和临床检查结果令人放心。本病例报告罕见的原因如下：（1）通常情况下，直肠重复畸形相对少见（文献报道70例）；（2）发现新生儿直肠重复畸形的方式不寻常（文献报道4例）；（3）畸形体积较大；（4）存在异位纤毛上皮。
