Rapunzel syndrome:A rare presentation with multiple small intestinal intussusceptions

Bezoars are usually confined to the stomach which is seen in individuals with psychiatric illness like trichotillomania,trichophagia and gastric dysmotility.Long standing bezoars may extend into the small intestine leading to a condition known as Rapunzel syndrome.Diagnosis can be established by endoscopy,ultrasonography and computed tomography scan.Treatment includes improvement of general condition and removal of bezoar by laparoscopic approach or laparotomy.Psychiatric consultation is necessary to treat and prevent relapse.We report a case of Rapunzel syndrome in a 16-yearold girl with trichotillomania.She presented with history of epigastric mass for three months and recent onset of pain abdomen,vomiting and early satiety.Skiagram of abdomen was showing distended stomach and endoscopy revealed trichobezoar.At laparotomy,stomach was distended with trichobezoar and there were multiple small intestinal intussusceptions.Gastrotomy and manual reduction of intussusceptions with the removal of trichobezoar with its tail was done.Patient recovered completely after the procedure.

Multiple bowel intussusceptions from metastatic localized malignant pleural mesothelioma:A case report

Localized malignant pleural mesothelioma (LMPM) is a rare occurrence, and gastrointestinal intra-luminal metastases have not previously been reported. Herein, we report a patient with LMPM who presented with a local recurrence 10 mo after initial en bloc surgical resection. Abdominal computed tomography was performed for intractable, vague abdominal pain with episodic vomiting, which showed a "target sign" over the left lower quadrant. Laparotomy revealed several intra-luminal metastatic tumors in the small intestine and colon and a segmental resection of metastatic lesions was performed. Unfortunately, the patient died of sepsis despite successful surgical intervention. Though local recurrence is more frequent in LMPM, the possibility of distant metastasis should not be ignored in patients with non-specifi c abdominal pain.

Adult sigmoid intussusception resembling rectal prolapse:A case report

BACKGROUND Rectal prolapse arises from benign etiology.When symptoms of internal intussusception mirror those of rectal prolapse,a misdiagnosis is possible,especially under limited clinical presentation.It is crucial to recognize and differentiate rectal prolapse from internal intussusception because the two diagnoses have different prognoses.Here,we describe a case of adult sigmoid intussusception presenting as rectal prolapse.CASE SUMMARY A 64-year-old woman with no known medical history visited a gastrointestinal outpatient department due to hard bloody stool defecation for 1 wk followed by constipation for 3 d.Colonoscopy revealed a huge polypoid ulcerated tumor at the sigmoid colon with lumen stenosis.The patient was admitted due to postprocedural dull abdominal pain.Due to failed colonoscopy reduction and stent insertion,the patient underwent sigmoid colon resection with primary end-to-end anastomosis,with the transverse colostomy pathological report showing adenocarcinoma,pT3N0M0.She recovered well from the operation and was discharged with regular outpatient clinic follow-up.CONCLUSION Presentation and manifestation of sigmoid intussusception may resemble that of rectal prolapse,necessitating careful observation due to distinct prognostic implications.

Appendiceal intussusception complicated by adenocarcinoma of the cecum:A case report

BACKGROUND Appendiceal intussusception is a pathological condition in which the appendix is inverted into the cecum,which may cause symptoms that resemble those of other gastrointestinal disorders and may induce intestinal obstruction.The rarity of this case presentation is the co-occurrence of appendiceal intussusception and cecal adenocarcinoma,a combination that to our knowledge has not previously been reported in the medical literature.This case provides new insights into the complexities of diagnosing and managing overlapping pathologies.CASE SUMMARY A 25-year-old woman presented with persistent periumbilical pain and bloody stools.An initial biopsy showed cecal cancer;however,subsequent colonoscopy and computed tomography findings raised the suspicion of appendiceal intussus-ception,which was later confirmed postoperatively.This unique case was charac-terized by a combination of intussusception and adenocarcinoma of the cecum.The intervention included a laparoscopic right hemicolectomy,which led to the histopathological diagnosis of mucinous adenocarcinoma with appendiceal intussusception.The patient recovered well postoperatively and was advised to initiate adjuvant chemotherapy.This case highlights not only the importance of considering appendiceal intussusception in the differential diagnosis,but also the possibility of appendicitis and the atypical presentation of neoplastic lesions.CONCLUSIONS Physicians should consider the possibility of appendiceal intussusception in cases of atypical appendicitis,particularly when associated with neoplastic presentation.

Rare etiology of colonic intussusception involving an adult with emphysematous cystic enteropathy:A case report and review of literature

BACKGROUND Pneumatosis cystoides intestinalis(PCI),characterized by a collection of gas-filled cysts in the intestinal wall,is an uncommon but well-known condition in gastroenterology.Abdominal pain is the most frequent symptom associated with PCI.Intussusception represents a potential cause of recurrent abdominal pain or emergency presentation.However,the occurrence of colonic intussusception secondary to PCI is very unusual in adulthood.CASE SUMMARY A 52-year-old male,known with idiopathic PCI,presented seventeen months after initial diagnosis with a new right upper quadrant pain.A computed tomographyscan demonstrated a colonic intussusception at the hepatic flexure.PCI did not progress compared with initial investigation.The patient underwent an emergency right hemicolectomy.CONCLUSION Resection was recommended in this case because PCI proved to be persisting with no identified curable cause.Surgery allowed to address the underlying pathology,the potential relapse of intussusception,and the likely cause of recurrent abdominal pain,either invagination or PCI itself.

Colo-colonic intussusception as a rare complication of colonoscopy with polypectomy: Two case reports

BACKGROUND Colonoscopy is the most frequently used diagnostic and therapeutic tool for the treatment of colorectal diseases.Although the complication rate is low,it can be potentially serious.Intussusception is a rare and severe complication often associated with polypectomy.Only a handful of post-colonoscopy intussusception cases have been reported,making this study a valuable addition to the medical literature.CASE SUMMARY Case 1:A 61-year-old man underwent colonoscopy with polypectomy for chronic abdominal pain.The patient experienced abdominal pain 11 hours later but was still discharged after pain management.He was readmitted due to recurring pain.Computed tomography(CT)showed colo-colonic intussusception.Initial conservative management and attempts at endoscopic reduction failed;therefore,laparoscopic right hemicolectomy was performed.Histopathological examination revealed tubular adenomas in the polyps and inflammation in the resected specimens.Case 2:A 59-year-old woman underwent colonoscopy with polypectomy for a polyp in the transverse colon.She experienced upper abdominal pain,fever,nausea,and vomiting 9 hours after the procedure.Emergency CT and blood tests revealed a colo-colonic intussusception near the hepatic flexure and an elevated white blood cell count.Initial attempts at endoscopic reduction failed and conservative treatment showed no improvement.She underwent successful laparoscopic reduction and recovered uneventfully.Histopathological examination of the resected polyp revealed hyperplasia.CONCLUSION Post-colonoscopy intussusception in adults is rare,and polypectomy may contribute to its occurrence.Early diagnosis is crucial,with prompt CT examination serving as key.After excluding malignancies,conservative management and reduction of intussusception should be considered before surgical bowel resection.

Colon Adenocarcinoma in a Retrograde Intussusception: A Case Report and Literature Review

Background: Intussusception is defined as a telescoping of a proximal gastrointestinal segment with its mesentery to a distal one, only 5% occur in adults and in colon the probability that it is caused by a malignant disease is up to 65%. Only 1% occurs in a retrograde manner, the rest occur in an anterograde manner. Aim: Describe the clinical presentation of an intussusception in the adult patient as well as its most frequent causes and possible complications that influence decision making for a definitive treatment. Case Presentation: A 66-year-old woman diagnosed with colon adenocarcinoma who underwent elective transverse colectomy and colonic anastomosis with an incidental finding of a transverse colon tumor in a retrograde intussusception was studied. Conclusion: In any adult patient with an intussusception especially in colon a neoplasia should be suspected and the affected segment should be resected without being reduced due to the risk of perforation and tumor dissemination.

The Value of Ultrasound in the Application of Water Enema in Intussusception

Objective:To introduce high-frequency ultrasound diagnostic technology to cooperate with diagnosis and treatment for patients with intussusception,and to observe its clinical application effect.Methods:The study took patients with intussusception as the object of observation,with a total of 52 cases participating,all of which were clinically admitted from December 2022 to December 2023.After enrollment,the patients underwent high-frequency ultrasound detection in sequence,recording the ultrasound imaging characteristics,and referring to the results of surgical or leaky saline enema reset to confirm the diagnosis(the gold standard)to assess the diagnostic efficacy of high-frequency ultrasound technology.Results:Comparing the confirmed diagnostic results of surgery or leaking saline enema restoration,the detection rate of high-frequency ultrasound technology was 98.07%(P>0.05).Among the 52 patients treated with hydropneumatic enema restoration with the cooperation of high-frequency ultrasound monitoring,39 patients were successfully reset in the first treatment(75.00%),while 9 patients were successfully reset(17.30%)after multiple treatments(≥2 times);the remaining 4 patients were unsuccessful and then converted to surgical treatment,and all of them were successfully treated and cured.Conclusion:For the diagnosis of intussusception,high-frequency color Doppler ultrasound has the advantages of high accuracy and simple operation,which provides a scientific and accurate reference basis for the clinical diagnosis and treatment of patients.

Clinical Research of Transumbilical Singleport Laparoscopic Treatment For Pediatric Intussusception

Objective:To explore the feasibility and clinical efficacy of single port laparoscopic surgery through the umbilical cord for the treatment of pediatric intussusception.Method:Clinical data of 38 cases of pediatric intussusception treated with umbilical single port laparoscopic surgery from December 2017 to June 2019 were collected.The surgical method involves placing Trocar through the umbilical incision to establish pneumoperitoneum,inserting a single hole with a 0°laparoscopic operating channel,exploring intussusception,and performing non-invasive forceps to completely reduce it.After the reduction of intussusception,if intestinal malformation is found,the umbilical incision can be expanded to lift the diseased intestinal tract out of the abdominal cavity for resection.At the same time,clinical data of 24 children who underwent traditional porous laparoscopic intussusception surgery during the same period were collected,and the surgical time,intraoperative blood loss,postoperative hospital stay,and satisfaction score of incision aesthetics were compared between the two groups of children.Result:Both groups of patients successfully completed the surgery,and compared with the porous laparoscopic group,the single hole laparoscopic group had a shorter surgical time[(32.4±8.6)minutes vs.(40.6±9.8)minutes,P<0.05],decreased bleeding volume[(5.5±1.5)mL vs.(8.6±2.2)mL,P<0.05],significantly shortened postoperative hospital stay[(4.6±1.2)d vs.(6.2±1.4)d,P<0.05],and significantly increased satisfaction score with incision aesthetics[(4.2±0.8)points vs.(3.2±0.7)points,P<0.05].Follow up for 6 months to 2 years showed no recurrence of intussusception.Conclusion:Transumbilical single port laparoscopic surgery is a safe and effective method for children with intussusception,which is characterized by small trauma,fast recovery,short operation time,and better aesthetic effect.

Clinical features,diagnosis,and treatment of Peutz-Jeghers syndrome:Experience with 566 Chinese cases

BACKGROUND Peutz-Jeghers syndrome(PJS)is a clinically rare disease with pigmented spots on the lips and mucous membranes and extremities,scattered gastrointestinal polyps,and susceptibility to tumors as clinical manifestations.Effective preventive and curative methods are still lacking.Here we summarize our experience with 566 Chinese patients with PJS from a Chinese medical center with regard to the clinical features,diagnosis,and treatment.AIM To explore the clinical features,diagnosis,and treatment of PJS in a Chinese medical center.METHODS The diagnosis and treatment information of 566 cases of PJS admitted to the Air Force Medical Center from January 1994 to October 2022 was summarized.A clinical database was established covering age,gender,ethnicity,family history,age at first treatment,time and sequence of appearance of mucocutaneous pigmentation,polyp distribution,quantity,and diameter,frequency of hospitalization,fre-quency of surgical operations,etc.The clinical data was retrospectively analyzed using SPSS 26.0 software,with P<0.05 considered statistically significant.RESULTS Of all the patients included,55.3%were male and 44.7%were female.Median time to the appearance of mucocutaneous pigmentation was 2 years,and median time from the appearance of mucocutaneous pigmentation to the occurrence of abdominal symptoms was 10 years.The vast majority(92.2%)of patients underwent small bowel endoscopy and treatment,with 2.3%having serious complications.There was a statistically significant difference in the number of enteroscopies between patients with and without canceration(P=0.004,Z=-2.882);71.2%of patients underwent surgical operation,75.6%of patients underwent surgical operation before the age of 35 years,and there was a statistically significant difference in the frequency of surgical operations between patients with and without cancer(P=0.000,Z=-5.127).At 40 years of age,the cumulative risk of intussusception in PJS was approximately 72.0%,and at 50 years,the cumulative risk of intussusception in PJS was approximately 89.6%.At 50 years of age,the cumulative risk of cancer in PJS was approximately 49.3%,and at 60 years of age,the cumulative risk of cancer in PJS was approximately 71.7%.CONCLUSION The risk of intussusception and cancer of PJS polyps increases with age.PJS patients≥10 years old should undergo annual enteroscopy.Endoscopic treatment has a good safety profile and can reduce the occurrence of polyps intussusception and cancer.Surgery should be conducted to protect the gastrointestinal system by removing polyps.

Surgical treatments of recurrent small intestine metastatic melanoma manifesting with gastrointestinal hemorrhage and intussusception:A case report

BACKGROUND Melanoma is the most aggressive form of skin cancer,with a tendency to metastasize to any organ.Malignant melanoma is the most frequent cause of skin cancer-related deaths worldwide.Small intestine cancers especially small intestine metastases are relatively rare.Small intestine metastases are seldom described and likely underdiagnosed.Intussusception is most common in pediatric age,and in adults are almost 5%of all cases.CASE SUMMARY A 75-year-old man with a history of acral malignant melanoma was admitted to the Gastroenterology Department of our hospital,complaining of intermittent melena for 1 mo.Magnetic resonance enterography showed partial thickening of the jejunal wall and formation of a soft tissue mass,indicating a neoplastic lesion with jejunojejunal intussusception.The patient underwent partial small bowel resection.Pathological findings and immunohistochemical staining indicated small intestine metastatic melanoma.The patient refused further anti-tumor treatment after the surgery.Ten months after the first surgery,the patient presented with melena again.Computed tomography enterography showed the anastomotic stoma was normal without thickening of the intestinal wall,and routine conservative treatment was given.Three months later,the patient developed melena again.The patient underwent a second surgery,and multiple metastatic melanoma lesions were found.The patient refused adjuvant anti-tumor treatment and was alive at the latest follow-up.CONCLUSION Small intestine metastatic melanoma should be suspected in any patient with a history of malignant melanoma and gastrointestinal symptoms.

Cohort analysis of pediatric intussusception score to diagnose intussusception

BACKGROUND Intussusception is a primary cause of intestinal obstruction in young children.Delayed diagnosis is associated with increased morbidity.Ultrasonography(USG)is the gold standard for diagnosis,but it is operator dependent and often unavailable in limited resource areas.AIM To study the clinical characteristics of intussusception including management and evaluation of the diagnostic accuracy of abdominal radiography(AR)and the promising parameters found in the pediatric intussusception score(PIS).METHODS Children with suspected intussusception in our center from 2006 to 2018 were recruited.Clinical manifestations,investigations,and treatment outcomes were recorded.AR images were interpreted by a pediatric radiologist.Diagnosis of intussusception was composed of compatible USG and response with reduction.The diagnostic value of the proposed PIS was evaluated.RESULTS Ninety-seven children were diagnosed with intussusception(2.06±2.67 years,62.9%male),of whom 74%were<2 years old and 37.1%were referrals.The common manifestations of intussusception were irritability or abdominal pain(86.7%)and vomiting(59.2%).Children aged 6 mo to 2 years,pallor,palpable abdominal mass,and positive AR were the parameters that could discriminate intussusception from other mimics(P<0.05).Referral case was the only significant parameter for failure to reduce intussusception(P<0.05).AR to diagnose intussusception had a sensitivity of 59.2%.The proposed PIS,a combination of clinical irritability or abdominal pain,children aged 6 mo to 2 years,and compatible AR,had a sensitivity of 85.7%.CONCLUSION AR alone provides poor screening for intussusception.The proposed PIS in combination with common manifestations and AR data was shown to increase the diagnostic sensitivity,leading to timely clinical management.

Intestinal Invagination in Adults: About a Case at the “Mother-Child” Hospital Le Luxembourg in Mali

Intussusception is a pathology of infants and young children. Its occurrence in adults is very unusual. In the vast majority of cases, it is secondary to a benign or malignant tumor. Small bowel malignancies are relatively rare, 1% - 5% of malignant tumors of the gastrointestinal tract. Acute intussusception, a rare pathology in adults, often presents with non-specific symptoms. Contrary to children, the organic origin remains preponderant in the elderly, where the origin of neoplasia must be suspected in first intention. We report the case of a 49-year-old patient admitted urgently from the Luxembourg “Mère Enfant” hospital center in Bamako, Mali, for an occlusive syndrome. Several abdomino-pelvic ultrasounds were performed in several clinics with mixed conclusions: Hydro-aeric distension and a cessation of matter and gas without a clearly visible mass (04/09/2022);Demonstration of the central hydro-aeric levels wider than high containing fine and regular folds corresponding to connivents whose aspect is in factor of left acute small intestine occlusion, to be completed by the surgical opinion (06/09/2022) and a third ultrasound showed a normal appearance of the liver, gallbladder, kidneys, pancreas, spleen, bladder and prostate;diffuse aero-colic distention without lesion of secondary appearance (21/11/2022). A frontal standing ASP dated 11/25/2022 showed hail-like hydro-aeric levels. Surgical management consisted of an open oncological resection of an obstructive bowel tumor with ileo-ileal invagination. The anatomopathological and immunohistochemical study of the surgical specimen concluded to diffuse small cell non-Hodgkin’s lymphoma, without vascular embolism. The resection limits pass into the healthy zone.

Burkitt Lymphoma Presenting as Ileocolic Intussusception in an Adult

Adult intussusception is rare, highly associated with a malignant lead point, and often requires emergent surgical management. We report the case of a 44-year-old male who presented with generalized abdominal pain and was found to have early ileocolic intussusception secondary to a large ileocecal mass. Biopsies of the mass and an enlarged cardiophrenic lymph node, as well as pleural fluid cytology were all consistent with Burkitt lymphoma (BL). Curiously, the patient’s abdominal exam was reassuring, and the intussusception and malignant bowel obstruction resolved over 36 hours with conservative management alone. With a Burkitt lymphoma international prognostic index (BL-IPI) score of 2, the patient proceeded to treatment with combination chemoimmunotherapy and attained a complete response after four cycles. There was no bowel perforation or recurrent intussusception throughout treatment. Thus, this report marks the first reported case of adult BL-associated intussusception to resolve with non-invasive management and establishes a precedent for conservative management in select patients.

Ileocolic Intussusception Prolapsing from the Rectum in von Recklinghausen's Disease

Intussusception is a paediatric condition that rarely presents in adults. We report an exceptional case of ileocolic intussusception prolapsing from the rectum in an adult with Von Recklinghausen′s disease. A 31-year-old man with von Recklinghausen′s disease presented to emergency department with a history of severe abdominal pain, vomits and rectal bleeding. Abdominal computed tomography showed intestinal obstruction probably due to a sigmoidorectal intussusception. Ileum, appendix, cecum and ascending colon were found to be intussuscepting through transverse, descending and sigmoid colon and prolapsing from the rectum during an emergent laparotomy. A right hemicolectomy was performed. An anatomical pathology examination revealed a neurofibroma of the appendix as lead point for intussusception. Intussusception in adults requires early surgical resection regardless of the nature of the initial case. Neurofibroma of the appendix is very rare;although it is benign, prompt resection is recommended because of a high risk of appendicitis and malignant transformation.

Factors determining operative reduction in intussusception in children aged less than fourteen years

Aim: To delineate the factors associated with operative reduction of intussusception, in contrast to enema reduction only without surgical intervention, in children aged less than 14 years. Methods: We retrospectively reviewed the records of all children aged <14 years who experienced intussusception between 2002 and 2012 and were treated at a single hospital in Qatar. Clinical outcomes were correlated with age, sex, clinical symptoms, diagnosticand treatmentrelated characteristics, and length of hospital stay. Results: A total of 141 children were diagnosed and treated for intussusception over 11 years. The male to female ratio of intussusception was 1.4:1. The length of hospital stay was longer in patients who underwent operative reduction than those that did not. Conclusion: Delay in presentation did not decrease the success of radiologic reduction and did not increase the risk of operative intervention and bowel resection.

Stapled transanal rectal resection for obstructed defecation syndrome associated with rectocele and rectal intussusception

AIM:To evaluate the safety and efficacy of stapled transanal rectal resection(STARR),and to analyze the outcome of the patients 12-mo after the operation.METHODS:From May 2007 to October 2008,50 female patients with rectocele and/or rectal intussusception underwent STARR.The preoperative status,perioperative and postoperative complications at baseline,3,6 and 12-mo were assessed.Data were collected prospectively from standardized questionnaires for the assessment of constipation[constipation scoring system,Longo’s obstructed defecation syndrome(ODS)score system,symptom severity score],patient satisfaction (visual analogue scale),and quality of life(Patient Assessment of Constipation-Quality of Life Questionnaire).RESULTS:At a 12-mo follow-up,significant improvement in the constipation scoring system,ODS score system,symptom severity score,visual analog scale and quality of life(P<0.0001)was observed.The symptoms of constipation improved in 90%of patients at 12 mo after surgery.The self-reported definitive outcome was excellent in 15(30%)patients,fairly good in 8(16%),good in 22(44%),and poor in 5(10%).CONCLUSION:STARR can be performed safely without major morbidity.Moreover,the procedure seems to be effective for patients with obstructed defecation associated with symptomatic rectocele and rectal intussusception.

The evolution and refinement of vasoepididymostomy techniques

Obstructive azoospermia secondary to epididymal obstruction can be corrected by microsurgical reconstruction with vasoepididymostomy （VE）. Although alternative management such as epididymal or testicular sperm aspiration in conjunction with intracytoplasmic sperm injection is feasible, various studies have established the superior cost-effectiveness of VE as a treatment of choice. Microsurgical VE is considered one of the most technically challenging microsurgeries. Its success rate is highly dependent on the skills and experience of the surgeons. Various techniques have been described in the literature for VE. We have pioneered a technique known as longitudinal intussusception VE （LIVE） in which the epididymal tubule is opened longitudinally to obtain a larger opening to allow its tubular content to pass through the anastomosis. Our preliminary data demonstrated a patency rate of over 90%. This technique has been widely referenced in the recent literature including robotic-assisted microsurgery. The history of the development of different VE approaches, the oreoDerative evaluation along with the techniaues of various VE will be described in this article.

Anterograde jejunojejunal intussusception resulted in acute efferent loop syndrome after subtotal gastrectomy

Postoperative intussusception is an unusual clinical entity in adults,and is rarely encountered as a complication following gastric surgery.The most common type after gastric surgery is retrograde jejunogastric intussusception,and jejunojejunal intussusception has been rarely reported.We report a case of anterograde jejunojejunal intussusception after radical subtotal gastrectomy with Billroth Ⅱ anastomosis in a 38-year-old Korean woman with early gastric cancer,and include a review of the literature on this unusual complication.

Inverted Meckel’s diverticulum manifested as adult intussusception: Age does not matter

Adult intussusception due to Meckel’s diverticulum (MD) is an uncommon cause of intestinal obstruction. However, the surgeon should still be suspicious of this condition since the non specific symptoms and the rarity of it make a preoperative diagnosis uncertain. Considering the secondary nature of adult intussusception and the necessity of early surgical intervention to avoid morbidity and mortality, we report two cases of intussusception due to MD in adults. A diverticulectomy using a TA stapler was performed in the first patient. In the second patient extensive fibrosis of the adjacent mesentery and thickening of jejunal mucosa were observed, so a segmental resection of the small bowel or affected ileal part and a hand-sewn anastomosis was performed. The postoperative period along with the long term follow-up was uneventful for both patients. The decision between diverticulectomy vs bowel resection can be based on the intussuscepted bowel condition. Early surgical intervention may ensure a favorable outcome.