Cystic rectal duplication: a rare cause of neonatal intestinal obstruction

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 × 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.     

消化道重复畸形12例

目的探讨消化道重复畸形的临床和病理特点。方法回顾性分析消化道重复畸形共12例。男8例,女4例。收集临床资料,对患儿进行随访,复习病理切片并结合临床分析。结果本组12例均经手术及病理检查证实诊断,重复畸形解剖部位分布:回盲部4例,食管、结肠各2例,胃、空肠、直肠、肛管各1例。术后均恢复正常。10例重复畸形黏膜性质与主肠管相似,2例临床表现为便血、贫血者在病理上均为异位胃黏膜组织。结论手术切除是治疗消化道重复畸形的最好选择。     

Duplication of the rectum: report of four cases and review of the literature

Rectal duplications are rare anomalies. Recently, we observed four cases of rectal duplication, each presenting with different clinical features including chronic constipation, a prolapsing rectal “polyp,” a “growth” from the vulva, and acute retention of urine. The variety of clinical presentations may lead to delay in diagnosis and multiple operations. Accepted: 28 October 1997     

Perforation of jejunal duplication lined with ectopic gastric mucosa

Alimentary tract duplications are rare congenital anomalies that occasionally contain ectopic gastric mucosa that secretes acid peptic juice and can cause pain, ulceration, bleeding and rarely perforation. We report a case of a 2-year-old girl who developed perforation of a jejunal duplication lined with ectopic gastric mucosa who was managed surgically.     

Cystic rectal duplication: a rare cause of neonatal bladder-outlet obstruction and hydronephrosis

 A case of cystic rectal duplication (RD) is presented. A 7-day-old female was admitted with acute urinary retention, voiding difficulty, and abdominal distention since she was 4 days of age. Ultrasound and abdominal computed tomography (CT) demonstrated a huge, cystic mass in the pelvis and abdomen that resulted in acute urinary retention and bilateral hydronephrosis. CT-guided drainage of the lesion followed by transabdominal surgical excision resulted in a cure. Pathologic examination demonstrated a RD lined by respiratory epithelium. Accepted: 14 December 1999     

Complex colon duplication mimicking an obstructed,non-functioning kidney in a newborn with imperforate anus and spinal dysraphism

Gastrointestinal (GI) duplications contain tissue resembling several portions of the GI tract and are associated with vertebral and genitourinary (GU) abnormalities [1–4]. We report a newborn with low, imperforate anus and lumbosacral dysraphism, who presented with a large cystic mass in the left renal fossa and pelvis. The flank mass (felt initially to be a dysplastic kidney and ureter) proved to be a complex GI duplication with histologic evidence of gastric, small bowel, and colonic mucosa, as well as respiratory epithelium and pancreatic tissue.     

Rectum leiomyoma in a 10-month-old female

An unexpected case of leiomyoma (LM) of the rectum in a 10-month-old female patient. The patient presented with a palpable mass and symptoms of intestinal obstruction, constipation and rectal discomfort. Rectal examination revealed a clearly visible mass. The treatment consisted of a surgical resection with wide margins. Pathology reported a leiomyoma. The patient was submitted to a careful clinical evaluation and a continuous follow up.     

Diverticular rectal duplication with heterotopic gastric mucosa in a child: a rare cause of rectal bleeding

This report describes a rare case of rectal bleeding and proctalgia in a child due to diverticular duplication of rectum associated with heterotopic gastric mucosa.     

Immunohistochemical findings in rectal duplication mimicking rectal prolapse

Alimentary tract duplications represent rare anomalies, with only 5 % occurring in the rectum. The variety in clinical presentation may lead to a delay in diagnosis or to incorrect and multiple surgical procedures. We report the clinical, histological and immunohistochemical characteristics of a rectal duplication occurring in a 3-month-old male with an unusual clinical presentation. Using routine histology and immunohistochemistry, the rectal duplication showed the diffuse presence of gastric mucosa with a characteristic immunophenotype (i.e., diffuse cytokeratin 7 positivity and scattered chromogranin immunoreactivity). As far as we know, this is the first report showing an immunohistochemical differentiation pattern of gastric lining in a rectal duplication. Our results, showing the presence of gastric mucosa, are suggestive of a possible origin from the embryonic foregut.     

Anal-canal duplication in a 6-year-old child

 A 6-year-old girl with an anal deformity had been complaining of pruritus around the anus. A small pit at 6 o'clock was diagnosed as an anal duplication. Fistulography showed a 0.5-cm-depth fistula without communication to the rectum. A 1.5-cm diameter presacral mass was shown on computed tomography. There was no sacral anomaly. At fistulectomy, the fistula contained mucous, yellow-white fluid. The base was enlarged, probably because of inflammation, but was resected without any invasion of the rectum. Histology showed squamous epithelium on the surface of the fistula and columnar epithelium and goblet cells in the base, which confirmed the diagnosis of an anal-canal duplication. Accepted: 20 March 2001     

Duplication of the rectum resembling a juvenile polyp.

A five-year-old boy with a rectal mass mimicking a rectal polyp, which proved to be a cystic duplication of the rectum, is presented. In a child with painless rectal bleeding, a mass palpated during rectal examination is usually diagnosed initially as being a rectal polyp. However, the case presented revealed the possibility of rectal duplication.     

Oral Congenital Cystic Choristomas: A Case Report

Solitary choristomas containing gastrointestinal mucosa occur rarely in the tongue and the oral mucosa and usually present as an asymptomatic mass. This report documents the occurrence of two congenital intraoral cystic choristomas in a 5-month-old male infant. The cysts were located on the dorsum of the base of the tongue and left floor of mouth anterior to the lingual cyst. Both cysts were lined by nonkeratinizing squamous epithelium and gastric, intestinal, and respiratory epithelium. The cysts were excised by an oral approach.     

Cystic duplication of the caecum mimicking intussusception.

Diagnosis of gut duplications are often only made intraoperatively as they are uncommon and do not feature strongly in the differential diagnoses. A case of caecal cystic duplication mimicking intussusception is discussed with the aim of improving diagnostic accuracy in future cases. A 7 month old boy presented with the classical triad of abdominal pain, bleeding per rectum and a palpable mass. Barium enema revealed a mass in the caecum suggestive of an intussusceptum. Repeated attempts at hydrostatic reduction were unsuccessful as the mass was immobile. Laparotomy revealed cystic duplication of the caecum without intussusception. The bleeding per rectum was due to an ulcer in the colonic epithelium overlying the cyst.     

Gastrointestinal duplications

Gastrointestinal duplications are rare congenital lesions that can develop anywhere along the alimentary tract and may present in the newborn period as an abdominal mass. They are differentiated from other intraabdominal cystic lesions by the presence of a normal gastrointestinal mucosal lining. Multiple theories have been proposed to account for these lesions; however, no single theory adequately explains all the known duplications. They are most frequently single, tubular, or cystic and located on the mesenteric side of the native alimentary tract structure. Symptoms often are related to the location of the duplication; oral and esophageal lesions can create respiratory difficulties, whereas lower gastrointestinal lesions may cause nausea, vomiting, bleeding, perforation, or obstruction. Treatment is resection with care taken to protect the common blood supply of the native structures. Occasionally, a partial resection with mucosal excision is required to preserve intestinal mucosa.     

Asymptomatic gastric duplication in a child: report of a new case and review of the literature

Antral duplication cyst with an antral mucosal lining accounts for 2.2% of all gastric duplications, which represent 4–8% of all alimentary tract duplications. They usually become symptomatic before 2 years of age. We report a 6-year-old child with no previous history of symptoms who was admitted to our hospital because of frequent vomiting and acute abdominal pains for the previous 2 days. Abdominal examination revealed a mass in the epigastric area.     

Rectal localization of Burkitt's lymphoma]

BACKGROUND: Rectal tumors are rare in childhood and among them malignant tumors are even less common. Only eight cases of primary rectal lymphomas were reported in children, with various presenting signs and histology. Burkitt's lymphomas are among these cases. CASE REPORT: A five-year-old child presented with hematochezia and unusual constipation. The rectal examination showed a voluminous intra rectal mass. Radiographic and pathologic examinations led to the diagnostic of Burkitt's lymphoma with medullary involvement. Complete remission was obtained after initial chemotherapy but a local relapse occurred and the child died eight months later. CONCLUSION: Hematochezia associated with unusual constipation impose a rectal examination. Early diagnosis of a rectal tumor may allow patients survival.     

Gastrointestinal system duplications in children (19 cases)]

Digestive duplications are a rare condition in children, characterized by an important anatomoclinical polymorphism. Diagnosis suspected on clinics and evoked by radiology, require histological confirmation. PATIENTS AND METHODS: Nineteen children with duplications were managed in our department, from 1989 to 2001: their hospital chart was retrospectively studied. RESULTS: Ten of our patients were less than one year of age, ten were boys and nine were girls. Clinical signs consisted of pain and abdominal mass, with some transit disturbs, and these signs led to ultrasonography and tomodensitometry. In one case alimentary tract duplication was revealed by digestive hemorrhage, leading to scintigraphic study. The digestive duplications were localized on the stomach in one case, the duodenum in two cases, the jejuno-ileum in twelve cases, the colon in two cases, and the rectum in two cases. Five digestive duplications were tubular, with one communicating duplication. All cases benefited from surgical treatment, and resection procedure was chosen according to duplication type and site. Histological study showed eight cases of ectopic mucosa, six ectopic gastric mucosa and two pancreatic ectopic mucosa. CONCLUSION: Diagnosis and surgical precocious treatment of digestive duplications are the only way to warn complications of this benign pathology.     

Type 1 rectal duplication: associated congenital malformations and ultrasound diagnosis

Type I duplication of the rectum is a rare condition that in most reported cases does not occur in association with other congenital malformations. We describe three cases, two of which had duplications of other structures: the first had a functioning supernumerary pelvic kidney, the second a duplication of the bladder, urethra, and vagina; the third had no associated malformations but was unusual, because the duplication cyst lay anterior to the rectum, and unique in that the diagnosis of an intra-abdominal cyst was made by ultrasonic examination. Correspondence to: G. M. A. Hendry     

Ultrasonographic demonstration of gastric duplication in infancy

A case of gastric duplication in an infant is reported. Preoperative ultrasound demonstrated a cystic mass lesion with a thin inner echogenic (mucosa) and a wider outer hypoechoic rim (muscle layer). These findings correlated well with the resected specimen.     

Multifocal gastrointestinal stromal tumor (GIST) of the stomach in an 11-year-old girl

A previously healthy 11-year-old girl presented with an 8-month history of anemia and left upper quadrant abdominal pain. US examination demonstrated a 9-cm cystic mass with a fluid-fluid level in the left upper quadrant with unclear organ of origin. Abdominal MR imaging demonstrated a complex cystic mass, likely arising from the stomach. Additional T2 hyperintense submucosal lesions were identified in the gastric wall. Surgical excision confirmed the diagnosis of multifocal gastric gastrointestinal stromal tumor (GIST). MR imaging was helpful in suggesting a gastric origin of the primary mass and in demonstrating multifocal disease within the stomach.