A case of recurrent intussusception in young adult female: Peutz Jegher syndrome

Authors

  • Brajesh B. Gupta Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Ganesh D. Jadhav Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Pravin Bhingare Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Sanjay Dakhore Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Amit Kodape Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Shweta Gupta Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Manali Jain Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India
  • Karishma Deshmukh Department of General Surgery, Government Medical College, Nagpur, Maharashtra, India

DOI:

https://doi.org/10.18203/2349-2902.isj20230512

Keywords:

Recurrent intussusception, Peutz Jegher syndrome, Colonic polyp, Lead point

Abstract

Peutz Jegher syndrome (PJS) is an autosomal dominant inherited polyposis syndrome, in which multiple characteristic polyps occur in the gastro-intestinal tract associated with mucocutaneous pigmentation especially of vermillion border of the lips. Small bowel obstruction is presenting complaint in half of the cases-intussusception due to polyp being lead point is the high risk for obstruction. A 21 year old female patient with presented to emergency department with complaining of intermittent episodes of abdominal pain and multiple episodes of billious vomiting since 7 days. Patient had history of exploratory laparotomy 10 years back for similar complaints. On examination, melanocytic pigmentation present over vermillion border and palmar aspect. Enhanced computed tomography reveals telescoping of the distal jejunum to proximal ileal loops s/o jejuno-ileal intussusception with ileo-ileal intussusception were noted. On surgical decision, patient again underwent exploratory laparotomy which revealed intussusception with non-viable small bowel segment were found, Small bowel resection was performed and jejuno-ileal anastomosis was done. Patient follow up was maintained to go with colonoscopic evaluation. In conclusion, recurrent intussusception is a rare cause of acute abdomen in adults. A high index of suspicion and appropriate investigations are helpful in prompt diagnosis. Thorough evaluation intra operative and then regular periodic follow up is required in such cases for early detection of disease before complication occurs.

References

Duan SX, Wang GH, Zhong J, Ou WH, Fu MX, Wang FS, et al. Peutz-Jeghers syndrome with intermittent upper intestinal obstruction: A case report and review of the literature. Medicine (Baltimore). 2017;96(17):e6538.

Duan SX, Wang GH, Zhong J, Ou WH, Fu MX, Wang FS, et al. Peutz-Jeghers syndrome with intermittent upper intestinal obstruction: A case report and review of the literature. Medicine (Baltimore). 2017;96(17):e6538.

Tweedie JH, McCann BG. Peutz-Jeghers syndrome and metastasising colonic adenocarcinoma. Gut. 1984;25(10):1118-23.

Chowdhry S, Umrigar DD, Yadav N. Peutz-jeghers syndrome in a child presenting with acute abdomen: a case report, Asian J. Dermatol. 2015;20-4.

Foley TR, McGarrity TJ, Abt AB. Peutz-Jeghers syndrome: a clinicopathologic survey of the "Harrisburg family" with a 49-year follow-up. Gastroenterology. 1988;95(6):1535-40.

Giardiello FM, Trimbath JD. Peutz-Jeghers syndrome and management recommendations. Clin Gastroenterol Hepatol. 2006;4(4):408-15.

Butt N, Salih M, Khan M, Ahmed R, Haider Z, Shah SA. An incidentally discovered asymptomatic para-aortic paraganglioma with Peutz-Jeghers syndrome. Saudi J Gastroenterol. 2012;18:388.

Latchford A, Cohen S, Auth M, Scaillon M, Viala J, Daniels R, et al. Management of Peutz-Jeghers Syndrome in Children and Adolescents: A Position Paper From the ESPGHAN Polyposis Working Group. J Pediatr Gastroenterol Nutr. 2019;68(3):442-52.

Loureiro J, Menegazzo GL, Vergamini L, Pestana RC, Formiga FB, Sousa MCC, et al. Diagnostic difficulty in Peutz–Jeghers syndrome. Colorectal Dis. 2015;35:67-71.

Giardiello FM. Hereditary colorectal cancer and polyp syndromes. Early Diagnosis Treat. New York: Elsevier; 2011: 21-30.

Townsend C, Beauchamp RD, Evers BM, Mattox KL. Sabiston textbook of surgery: The biological basis of modern surgical practice. 20th ed. New York: Elsevier; 2016: 1372.

Downloads

Published

2023-02-24

Issue

Section

Case Reports