Congenital colonic stenosis diagnosed in adulthood

Baudet, J; Redondo, Ignacio; Arguiñarena, X.; Castro, Vinicius Leite de; Alonso, M.

doi:10.1055/s-0029-1244141

Cited by 4 publications

(4 citation statements)

References 1 publication

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“…Whereas those with lesser degree of obstruction present later with chronic constipation and abdominal distension. CCS has been diagnosed as late as 32 years of age 4. The clinical picture and investigations may simulate HD as in our patient 5.…”

Section: Discussionmentioning

confidence: 51%

Congenital colonic stenosis with absent caecum and appendix: a rare association

Khanna

Yadav

Nandan³

et al. 2018

BMJ Case Reports

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Congenital colonic stenosis is a rare condition with less than 20 cases reported in the literature since 1966. We report an interesting case of a 7-month-old baby girl who presented with features suggestive of acute intestinal obstruction. On exploration, it was a case of ascending colon stenosis with absence of caecum and appendix. A double barrel ileocolostomy was performed. The histopathology confirmed the diagnosis of colonic stenosis and ruled out the presence of Hirschsprung’s disease in the distal colon. The child underwent second stage surgery (stoma closure) after 9 months. In complex cases of congenital colonic stenosis, an early decompressive surgery followed by a delayed second stage closure is recommended in patients with poor general condition.

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Section: Discussionmentioning

confidence: 51%

Congenital colonic stenosis with absent caecum and appendix: a rare association

Khanna

Yadav

Nandan³

et al. 2018

BMJ Case Reports

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“…Symptoms may be present at birth as constipation, abdominal pain, progressive abdominal distension, or failure to pass meconium within 48 hours but could also become manifest later in childhood as colicky abdominal pain, bilious vomiting, and abdominal distension. Baudet et al [ 18 ] even reported a case of CCS diagnosed in adulthood presenting with abdominal pain, nausea, vomiting, and laxatives abuse.…”

Section: Discussionmentioning

confidence: 99%

Multiple Congenital Colonic Stenosis: A Rare Gastrointestinal Malformation

Zambaiti

Chiaramonte

Salerno

et al. 2016

Case Reports in Pediatrics

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Congenital colonic stenosis is a rare pediatric condition. Since 1968, only 16 cases have been reported in the literature. To the authors' knowledge, multiple congenital colonic stenosis has not been previously reported in the literature. We report the case of a 2-month-old male, presented at our Neonatal Intensive Care Unit with a suspicion of intestinal malrotation. Clinical examination revealed persistent abdominal distension. During the enema examination, the contrast medium appeared to fill the lumen of the colon up to three stenotic segments and could not proceed further. Intraoperatively we confirmed the presence of four types of colonic atresia, located in the ascending, transverse, and descending colon, respectively, plus appendix atresia. First surgical steps consisted in resection of proximal stenotic segment, appendix removal, proximal cecostomy, and distal colostomy on ascending colon in order to preserve colonic length. Histopathological examination confirmed the diagnosis of colonic stenosis. Final surgical step consisted in multiple colocolostomy and enteroplasty. A planned two-stage procedure, consisting of resection with colostomy for decompression as the first step and a later anastomosis, is recommended in order to allow bowel length preservation.

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“…In literature, the oldest case of CCS reported was a 32-year male who presented with abdominal pain, nausea, vomiting, and laxative abuse. [ 18 ] Plain radiographs are usually nonspecific and show features of low-intestinal obstruction as dilated bowel loops with multiple air–fluid levels. CCS has to be differentiated from distal ileal atresia, colonic atresia, malrotation, and neonatal HD in the neonate and from HD in infants and children.…”

Section: Discussionmentioning

confidence: 99%

“…Surgical treatment of CCS is the mainstay of therapy including resection of the stenotic segment and either primary or secondary end-to-end anastomosis. In literature,[ 1 2 3 4 5 6 7 8 9 10 11 12 13 14 18 ] resection of the atretic segment and primary end-to-end anastomosis has been described as the preferred approach in left-sided lesions, while in right-sided lesions, staged approach in the form of initial resection and divided colostomy and secondary end-to-end anastomosis or primary resection and end-to-end anastomosis with proximal intestinal diversion was the preferred approach.. However in our opinion, treatment in patients of CCS should be individualized not only on the basis of site of lesion but also on the basis of medical condition of the patient, discrepancy between proximal and distal lumen diameter as suggested by Cox et al .…”

Section: Discussionmentioning

confidence: 99%

Congenital colonic stenosis: A rare gastrointestinal malformation in children

Gupta

Singh

Sunil

et al. 2021

J Indian Assoc Pediatr Surg

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Aims: Congenital colonic stenosis (CCS) is an extremely rare cause of low-intestinal obstruction in neonates/child. We report our experience with seven cases of CCS presenting with low-intestinal obstruction and diagnosed intraoperatively and also propose an algorithm for its appropriate treatment for the adequate outcome. Materials and Methods: It was a retrospective study of seven patients of CCS including two neonates (5-days and 15-days old), four infants (age range – 2–11 months), and one 24-month-old child admitted from 2014 to 2019. Information regarding the age of presentation, clinical presentation, physical findings, radiological and laboratory findings, details of surgery, and outcome was retrieved and analyzed. Results: The male-to-female ratio was 5:2. Patients were initially diagnosed as cases of Hirschsprung's disease in five and ileal atresia in two. A final diagnosis of CCS was made during surgery and histopathological examination of resected stenotic segment. The segment involved was ascending colon in three, transverse colon in two, and sigmoid colon and junction of descending and sigmoid colon each in one patient. Resection of stenotic colonic segment and primary end-to-end anastomosis was performed in two, divided stoma after resection of the stenotic segment and secondary anastomosis in three, and proximal loop terminal ileostomy followed by resection of the stenotic colonic segment and ileocolic anastomosis after 10–12 weeks in two. Conclusions: CCS is a rare but possible cause of large-bowel obstruction, in neonatal, infant, and children particularly when associated with a history of chronic constipation since birth. It should be kept in mind as a differential diagnosis while managing a case of neonatal and pediatric intestinal obstruction, particularly low-bowel obstruction along with a history of chronic constipation. Treatment should be individualized for each patient based on clinical status and associated anomalies to give the best results with less morbidity.

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Congenital colonic stenosis diagnosed in adulthood

Cited by 4 publications

References 1 publication

Congenital colonic stenosis with absent caecum and appendix: a rare association

Congenital colonic stenosis with absent caecum and appendix: a rare association

Multiple Congenital Colonic Stenosis: A Rare Gastrointestinal Malformation

Congenital colonic stenosis: A rare gastrointestinal malformation in children

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