An Unusual Case of Bowel Obstruction: Diagnosis and Treatment

Question: A 61-year-old man was admitted to the Emergency Department with progressive crampy periumbilical pain accompanied with nausea, abdominal distention, and vomiting for 2 days. He experienced recurrent abdominal pain, vomiting, and constipation for more than 40 years with around 2 episodes per year. Symptoms were not related to diet or labor but could be relieved by enema. Air fluid levels could be indicated by computed tomography (CT) or KUB examination during the attack of abdominal pain. Physical examination demonstrated hypoactive bowel sound. Laboratory test showed an increase of C-reactive protein (33.99 mg/L), procalcitonin (0.68 ng/mL), and D-dimer (0.85 mg/L FEU), whereas other parameters were within normal range. Abdominal radiography showed several air fluid levels in the middle-lower abdomen (Figure A), and further enhanced abdominal CT showed a suspicious soft tissue mass in the right-lower quadrant (Figure B and C, yellow arrow). The mesenteric vessels are normal and no obvious obstruction point was discovered (Figure B and C). No special finding was observed under gastroscopy and colonoscopy, except a gastric fundic gland polyp and 2 inflammatory polys in cecum and ascending colon. Abdominal pain and vomiting were relieved with the treatment of fasting, enema, parenteral nutrition, and antibiotics (Cefonicid). Considering repeated intestinal obstruction attacks, no etiology was found with repeated laboratory tests, endoscope, and abdominal imaging examinations, laparoscopic exploration was performed with the consent of the patient. During the exploration, extensive adhesion of omentum and small intestine to the right-side abdominal and pelvic wall was noted. The right-side colon and gall bladder were also involved in the adhesion. The ileum was adhered, between which a mass of 3.5 cm x 2.5 cm x 2.0 cm was seen in the right-lower quadrant (Figure D). The bowel lumen proximal from this point was dilated. Partial ileal and lesion resection were then performed with adhesiolysis. Written informed consent was obtained from the patient for publication of this article and any accompanying images. The resected mass was sent for pathologic examination (Figure E). Based on the laboratory, imaging, and pathologic findings, what is the most likely diagnosis? What is the final diagnosis in this patient? See the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI. The pathologic analysis demonstrated multiple curved tubule structures with thickened surrounding basement membrane in the resected mass. No epithelial component was observed in the small tube cavities but some larger epithelioid cells could be seen. These supported a diagnosis of cryptorchidism with no obvious atypia or signs of malignancy. The patient’s postoperative course was uneventful and he was discharged 10 days after surgery. Bowel obstruction due to intraperitoneal adhesion is common but it is rarely caused by cryptorchidism. Cryptorchidism occurs in around 3% of full-term male neonates and more than half of the patients would experience “spontaneous descent” by the age of 1 year. Most patients could be diagnosed at their early age, based on the typical clinical manifestations and the absence of a testis in the scrotum. However, in our presented case, the patient, who was married and had 1 daughter, was first diagnosed in his sixties due to small bowel obstruction. During the chronic disease course, he was admitted to the hospital more than 10 times and the abdominal imaging indicated the obstructive point kept changing over time. Although the patient had no risk factors for slow motility, such as electrolyte disturbances, specific medications, and abdominal surgery, paralytic ileus was unlikely to be the diagnosis. Still, no obvious obstructive point was found this time but a suspicious soft tissue mass in the CT scan, which might be the “key” for this case. Although the patient denied a history of abdominal surgery or trauma, we considered abdominal adhesion could be a possible cause of his recurrent symptoms and a surgical inspection was needed. The finding of abdominal adhesion was anticipated but cryptorchidism was out of our expectation. Intestinal obstruction related to cryptorchidism in adulthood is so rare that only a few reports could be found in a literature search.1Satoskar S. Kashyap S. Ziehm J. et al.Cryptorchidism as an obscure cause of adhesive small bowel obstruction in an adult, a case report.Int J Surg Case Rep. 2021; 86106319Crossref PubMed Scopus (1) Google Scholar,2Kim C.W. Min G.E. Lee S.H. Small bowel obstruction caused by cryptorchidism in an adult.Ann Surg Treat Res. 2017; 93: 281-283Crossref PubMed Scopus (2) Google Scholar All patients including the current case were in their sixties and experienced similar symptoms of nausea and abdominal pain, which finally required surgical intervention. Specially, the patient in the current case suffered from a chronic disease course for more than 40 years, whereas both of the other cases reported acute episodes. Another case report came from a 26-year-old patient manifesting bowel obstruction, with the coexistence of cryptorchidism and abdominal cocoon syndrome, which is identified as a rare disease with dense abnormal fibrous collagen membrane wrapping abdominal contents.3Song W.J. Liu X.Y. Saad G.A.A. et al.Primary abdominal cocoon with cryptorchidism: a case report.BMC Gastroenterol. 2020; 20: 334Crossref PubMed Scopus (2) Google Scholar Although cryptorchidism might contribute to the obstructive symptoms in this patient, abdominal cocoon syndrome would be considered as the most important “culprit.” The diagnosis of cryptorchidism with bowel obstruction depends on a comprehensive consideration of clinical manifestations, imaging, and pathology, and it is challenging before surgical inspections. However, more careful physical examination, such as palpation of the scrotum on both sides, could largely avoid many detours and should not be omitted even in a middle-aged married male patient.