Journal of Pediatric Surgery Case Reports最新文献

{"title":"Tumoral calcinosis in an 8-year-old girl: A case report","authors":"M. Savadier,&nbsp;N. Maistry,&nbsp;D.S. Harrison","doi":"10.1016/j.epsc.2024.102955","DOIUrl":"10.1016/j.epsc.2024.102955","url":null,"abstract":"<div><h3>Introduction</h3><div>Tumoral Calcinosis is a rare, benign metabolic disorder, characterized by the deposition of calcium phosphate crystals in extra-articular soft tissues. Less than 400 cases have been described globally, with only around 10 % being described in the pediatric population. Large bilateral masses are exceedingly rare.</div></div><div><h3>Case report</h3><div>We present a case of an 8-year-old female with bilateral 15 × 10cm posterior chest wall masses, who presented following a one-year history of enlargement. The patient had no medical or family history. A diagnosis of primary hyperphosphataemic tumoral calcinosis was suspected based on biochemistry and imaging. The patient had a raised phosphate and normal parathyroid hormone level, while ultrasound and Computed Tomography scans showed multilocular, calcified lesions with solid and cystic components. Core needle biopsy confirmed a diagnosis of tumoral calcinosis. Staged surgery was undertaken to resect the masses, which had extensive regional invasion. Clear margins were attained, with a rim of normal tissue on histology. The patient developed recurrent bilateral 15 × 20cm masses one year later, having defaulted post-operative treatment with Acetazolamide and Aluminium Hydroxide. Further surgery and medical management with the above agents was required. Six months post-surgery for the recurrence, no further recurrence was noted, but following poor adherence to her medical therapy another year later, there has been recurrence on the right, measuring 5 × 5cm. Treatment with Acetazolamide and Aluminium Hydroxide continue, with close observation for further growth.</div></div><div><h3>Conclusion</h3><div>A combination of targeted medical therapy with Acetazolamide and Aluminium Hydroxide, and early surgical resection, may limit the risk of recurrence in hyperphosphatemic tumoral calcinosis in the pediatric population.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"114 ","pages":"Article 102955"},"PeriodicalIF":0.2,"publicationDate":"2024-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143148293","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Concurrent esophageal atresia with tracheoesophageal fistula and hirschsprung disease in a term neonate: A case report","authors":"Yidnekachew Getachew ,&nbsp;Jejaw Endale ,&nbsp;Mihret S. Tesfaye ,&nbsp;Hiwote M. Tsega ,&nbsp;Azeb Mulugeta","doi":"10.1016/j.epsc.2024.102949","DOIUrl":"10.1016/j.epsc.2024.102949","url":null,"abstract":"<div><h3>Introduction</h3><div>Concurrent esophageal atresia with tracheoesophageal fistula and Hirschsprung disease is a rare combination of congenital anomalies, with only four cases reported to date in full-term neonates.</div></div><div><h3>Case presentation</h3><div>A 4-day-old full-term neonate from rural Ethiopia was brought to our pediatric surgical unit with symptoms of respiratory distress and excessive frothy secretions. Chest exam revealed intercostal and subcostal retractions along with crepitation in the right posterior chest. The abdomen was distended, and a positive blast sign was noted during the rectal exam. Echocardiography showed a small patent ductus arteriosus. We attempted to pass an orogastric tube, but it was not possible. Chest and abdominal X-rays showed air in the stomach and the orogastric tube in the upper esophagus, suspicious for an esophageal atresia with a tracheoesophageal fistula. The patient had persistent thrombocytopenia, requiring multiple platelet transfusions, and was treated for aspiration pneumonia before he was deemed stable to undergo an operation. He received daily maintenance fluids with electrolytes for nutritional support and underwent daily rectal irrigations. On day of life 17 he was taken to the operating room. We found and ligated a distal tracheoesophageal fistula and did an end-to-end esophageal anastomosis via right thoracotomy. Because of the ongoing need for daily rectal irrigations, we also did a full thickness transanal rectal biopsy and confirmed the diagnosis of Hirschsprung disease. Postoperatively, the patient had no complications and has since started breastfeeding. He continues to receive rectal stimulation and irrigation and is on the wait list for his pull-through operation.</div></div><div><h3>Conclusion</h3><div>A high index of suspicion for Hirschsprung disease is crucial in patients with esophageal atresia and tracheoesophageal fistula, delayed meconium passage and lack of spontaneous bowel function.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"114 ","pages":"Article 102949"},"PeriodicalIF":0.2,"publicationDate":"2024-12-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143147679","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Pleuropulmonary blastoma in A 4-year-Old male: A case report","authors":"Fereshteh Moshfegh ,&nbsp;Zahra Hosseinzade ,&nbsp;Mohammad Javanbakht ,&nbsp;Shahla Ansari ,&nbsp;Ali Manafi Anari ,&nbsp;Khatere Tavajohi","doi":"10.1016/j.epsc.2024.102951","DOIUrl":"10.1016/j.epsc.2024.102951","url":null,"abstract":"<div><h3>Introduction</h3><div>Pleuropulmonary blastoma (PPB) in children can present with symptoms such as chest or abdominal pain, cough, fever, and respiratory distress, often leading to misdiagnosis as infections, pneumothorax, or pneumonia.</div></div><div><h3>Case presentation</h3><div>A 4-year-old male presented with generalized abdominal pain, hematuria, and respiratory symptoms. chest pain, cough, fever, and chills. Despite outpatient treatment with antibiotics, his condition failed to improve, and he was admitted to the hospital. Chest X-ray showed that the right hemithorax was whited out. Chest ultrasound a mass-like area in the right lower lobe. A chest computed tomography (CT) showed a large pleural effusion and a completely collapsed right lung. Laboratory tests showed elevated inflammatory markers and leukocytosis. Contrast-enhanced computed tomography (CECT) showed a large, well-defined heterogeneous mass measuring 98 × 86 mm in the right hemithorax. He was taken to the operating room for a resection. Through a right thoracotomy we found and resected a tumor measuring 3 × 14 × 16 cm arising from the lower lobe, and we resected a small portion of a rib that appeared involved by the tumor. Postoperative studies showed metastatic disease in several ribs. Pleural fluid cytology showed malignant cells, and the histopathology of the specimen confirmed the diagnosis of a type III PPB. Genetic testing was positive for a DICER1 mutation. Chemotherapy with ifosfamide, vincristine, actinomycin-D and doxorubicin was initiated five days after diagnosis. He currently continues his treatment.</div></div><div><h3>Conclusion</h3><div>Chest tumors should be suspected in patients that present respiratory symptoms that do not respond to standard measures and have abnormal findings on plain chest films. PPB should be suspected if a lung mass is found. Patients with confirmed PPB must undergo genetic testing to rule out DICER1 mutations, which are associated with a variety of other malignancies.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"114 ","pages":"Article 102951"},"PeriodicalIF":0.2,"publicationDate":"2024-12-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143148296","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Late presentation of a Type IV sacrococcygeal teratoma in a 16-year-old female: A case report","authors":"Ahmed Eldeib ,&nbsp;Carlos A. Reck ,&nbsp;Koby Herman ,&nbsp;Emily Yang ,&nbsp;Stefani Manis ,&nbsp;Francisca Velcek","doi":"10.1016/j.epsc.2024.102947","DOIUrl":"10.1016/j.epsc.2024.102947","url":null,"abstract":"<div><h3>Introduction</h3><div>Sacrococcygeal teratomas (SCTs) are rare germ cell tumors, predominantly diagnosed in neonates. These tumors are categorized as mature, immature, or malignant, with mature teratomas posing a lower risk for malignant transformation. While symptoms commonly arise from the mass effect, late presentation in adolescents or adults is rare.</div></div><div><h3>Case presentation</h3><div>A 16-year-old female with no significant past medical or surgical history presented with perianal pain, constipation, and fever. On physical exam, the rectum was collapsed by a mass. The laboratory work-up showed leukocytosis and an elevated C-reactive protein. Lactate dehydrogenase (LDH), alpha-fetoprotein (AFP), and beta-human chorionic gonadotropin (β-HCG) levels were within normal limits. A CT scan of the abdomen and pelvis revealed a 12 x 16 × 16 cm complex mass in the presacral region. Further evaluation with a pelvic MRI confirmed the presence of a multiloculated mass with mixed solid and cystic components, rim enhancement, and possible coccygeal involvement suggestive of a sacrococcygeal teratoma. Given the size and location of the mass, we decided for a combined abdomino-perineo-sacral surgical approach. The patient received Ceftriaxone and Metronidazole before the operation. We started with a midline laparotomy and detached the tumor from the intra-abdominal structures, and we continued with a posterior sagittal approach. The tumor appearance was consistent with a mature teratoma and had purulent fluid within the cystic component. We removed the mass completely and did a coccygectomy. The postoperative course was complicated by a retroperitoneal collection and a posterior wound dehiscence that required several washouts, debridement, vacuum-assisted closure therapy, and a long course of antibiotics. She was discharged home on postoperative day 32. She is being followed with yearly MRIs.</div></div><div><h3>Conclusion</h3><div>Superinfection of mature teratomas can lead to postoperative infectious complications. Pre-operative and postoperative antibiotics are recommended when resecting mature teratomas.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"114 ","pages":"Article 102947"},"PeriodicalIF":0.2,"publicationDate":"2024-12-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143148245","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Repair of “bladder neck” cloaca using a trans-vesicle approach: A case report","authors":"Nathalie Carey ,&nbsp;Luis H. Braga ,&nbsp;Bruno Leslie ,&nbsp;J Mark Walton ,&nbsp;Michael H. Livingston","doi":"10.1016/j.epsc.2024.102946","DOIUrl":"10.1016/j.epsc.2024.102946","url":null,"abstract":"<div><h3>Introduction</h3><div>Cloaca with a common channel greater than 3 cm typically requires urogenital separation rather than total urogenital mobilization. The purpose of this report was to describe our experience managing a female infant with an usual form of cloaca where all structures inserted onto the bladder neck.</div></div><div><h3>Case presentation</h3><div>A female infant with a single perineal opening underwent loop sigmoid colostomy shortly after birth. She voided spontaneously and had no hydrocolpos. She underwent exam under anesthesia, cystoscopy, and cloacogram under a single anesthetic at 5 months of age. This revealed five structures that inserted directly into the bladder: a central rectal fistula, two hemivaginas and hemiuteri bilaterally, and two ureters without hydronephrosis. The outflow tract was a long common channel measuring 5.2 cm. This patient was reviewed in a multidisciplinary setting and underwent repair at 10 months of age. We performed a midline laparotomy and opened the anterior wall of the bladder to visualize the structures that inserted posteriorly. Foley catheter and ureteric stents were placed. The rectal fistula and hemivaginas were mobilized off the bladder internally. Vaginal replacement was performed using a transverse portion of rectum. Additional colonic length was achieved by converting the loop colostomy to an end-loop. The abdomen was closed and posterior sagittal anorectoplasty was performed in a prone position.</div></div><div><h3>Conclusion</h3><div>This infant underwent repair of a “bladder neck” cloaca using a transvesicular approach. She is now 35 months of age and thriving. She developed neurogenic bladder requiring clean intermittent catheterization and is working on fecal continence.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"114 ","pages":"Article 102946"},"PeriodicalIF":0.2,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143147674","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Early separation of parasitic conjoined twins at a tertiary care hospital: A case report","authors":"Ahmad Yani ,&nbsp;Wulan Ayudyasari ,&nbsp;Kshetra Rinaldhy ,&nbsp;Muhamad Luthfi Prasetyo ,&nbsp;Nafissa Amanda Safinati Yani","doi":"10.1016/j.epsc.2024.102945","DOIUrl":"10.1016/j.epsc.2024.102945","url":null,"abstract":"<div><h3>Introduction</h3><div>Conjoined twins is defined as twins that are physically fused in utero and at birth. Parasitic conjoined twins is a particularly rare form of conjoined twins, occurring in fewer than 1 in 1 million live births. The management of parasitic conjoined twins remains a challenge, lacking a universal consensus regarding the optimal timing for separation surgery.</div></div><div><h3>Case presentation</h3><div>A 31-week pregnant woman presented with a fetal emergency. Ultrasound at 27 weeks revealed conjoined abdominopagus twins sharing a single liver. At 31 weeks, only one fetal heartbeat was detected, requiring an urgent cesarean section. Two asymmetric female twins were delivered: Neonate A, who survived with an APGAR score of 7/9, and Neonate B, a stillbirth. Neonate A had ventriculomegaly, while Neonate B exhibited multiple congenital anomalies, including bilateral cleft lip, hypodactyly, hydrocephalus, and hydrops fetalis. Neonate A was stabilized and underwent successful early separation surgery. Postoperatively, Neonate A received intensive care, including respiratory support, broad-spectrum antibiotics, and treatment for a patent ductus arteriosus. At one-month follow-up, Neonate A was stable and recovering well.</div></div><div><h3>Conclusion</h3><div>Early separation of parasitic conjoined twins is recommended in cases with one stillborn or deceased twin, as it improves survival chances for the autosite. Delayed separation is advised for cases with two viable twins, allowing for organ maturation and reducing intraoperative complications.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"114 ","pages":"Article 102945"},"PeriodicalIF":0.2,"publicationDate":"2024-12-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143148248","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Trans-umbilical management of OHVIRA syndrome by a thoracic trocar technique: A case report","authors":"Husam Ibrahimoglu,&nbsp;Ibrahim Uygun","doi":"10.1016/j.epsc.2024.102933","DOIUrl":"10.1016/j.epsc.2024.102933","url":null,"abstract":"<div><h3>Introduction</h3><div>Cervical atresia in cases of obstructed hemivagina and ipsilateral renal agenesis (OHVIRA) syndrome is rare. To prevent complications that may occur due to the association of cervical atresia with OHVIRA syndrome, a left hemihysterectomy and salpingectomy is occasionally needed.</div></div><div><h3>Case presentation</h3><div>A 14-year-old female was referred to us due to abdominal pain. She had never experienced menstrual pain until three periods preceding her presentation. Abdominal ultrasound (US) showed that the left horn of the uterus was hypoplastic and compatible with hematosalpinx. Computerized tomography (CT) of the abdomen showed a 3-cm left endometrial cavity, and an 8-cm septated lesion in the left adnexa. The left ovary could not be visualized. Magnetic resonance (MR) revealed uterus didelphys and a 6–8 cm septated lesion in the left adnexa. The left kidney was not present. The left cervix was absent. Based on these findings, OHVIRA syndrome was diagnosed. She was taken to the operating room for a hysteroscopy, which showed a single cervix, normal uterine mucosa, and a single tubal ostium. No septum was seen in the vagina. She then underwent a laparoscopy, which revealed uterus didelphys, widespread endometriosis, a large left hematosalpinx, left tubal torsion, no left ovary, and a normal right ovary. We proceeded with a left hemihysterectomy and salpingectomy using a transumbilical single-incision approach. The foci of endometriosis were suctioned and the specimen retrieved through the umbilical incision. She recovered well from the operation and was discharged home 5 days later.</div></div><div><h3>Conclusion</h3><div>OHVIRA syndrome should be suspected in patients with renal agenesis and an ipsilateral adnexal mass. Patients with OHVIRA and cervical atresia may need a left hemihysterectomy and salpingectomy, which can be done through a transumbilical single-incision approach.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"112 ","pages":"Article 102933"},"PeriodicalIF":0.2,"publicationDate":"2024-11-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142759215","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Common bile duct cysts in neonates, not always a choledochal cyst: A case report","authors":"Josephine Issenman ,&nbsp;Kathryn Martin","doi":"10.1016/j.epsc.2024.102928","DOIUrl":"10.1016/j.epsc.2024.102928","url":null,"abstract":"<div><h3>Introduction</h3><div>Obstructive jaundice in neonates results from disruption of bile drainage from the liver, which may be related to several conditions. Differentiating between these conditions and intervening rapidly is critical, as the success of treatment for biliary atresia decreases as the infant ages.</div></div><div><h3>Case presentation</h3><div>A 10-week-old term female born at 39 weeks of gestation was brought to the gastroenterology service to assess persistent jaundice. The jaundice was first noted by the patient's mother and pediatrician at 2 weeks of age and was thought to be physiologic. The stool during this period was reported to be normal in color. Laboratory investigations revealed obstructive jaundice (total bilirubin of 9.6 mg/dL; direct bilirubin of 5.6 mg/dL). Imaging demonstrated a cystic dilation of the common bile duct, concurrent dilation of the right hepatic duct, and no visible gallbladder. Given the ultrasound findings and parental report of normally colored stools, the diagnosis of an obstructed choledochal cyst was favored. However, due to the absence of a gallbladder, we obtained a magnetic cholangiography. The findings were similar, and with a presumed diagnosis of a type IV choledochal cyst the patient was taken to the operating room at the age of 91 days. A cystic dilatation of the common bile duct was found, with no distal duct. The structure identified as the right hepatic duct on imaging was found to be an atretic gallbladder. An intraoperative cholangiogram confirmed the diagnosis of cystic biliary atresia, and a Kasai portoenterostomy with Roux-en-Y reconstruction was performed. Postoperatively the hyperbilirubinemia resolved, but her liver function has gradually deteriorated, and she has been referred for a liver transplant.</div></div><div><h3>Conclusion</h3><div>Biliary atresia can present with symptoms and imaging findings that mimic choledochal cysts. Given that the treatment and prognosis of these conditions vary, persistent neonatal jaundice must be worked up expeditiously, and a high index of suspicion for biliary atresia must be maintained.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"112 ","pages":"Article 102928"},"PeriodicalIF":0.2,"publicationDate":"2024-11-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142722106","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Spontaneous enterocutaneous fistula in a neonate: A case report","authors":"Yirgalem Teklebirhan Gebreziher ,&nbsp;Yoseph Equar Assefa ,&nbsp;Hadush Tesfay Negash ,&nbsp;Fekrey berhe Gebru","doi":"10.1016/j.epsc.2024.102929","DOIUrl":"10.1016/j.epsc.2024.102929","url":null,"abstract":"<div><h3>Introduction</h3><div>Spontaneous enterocutaneous fistulas (ECF) are extremely rare in children. To our knowledge, only two cases have been reported in the literature.</div></div><div><h3>Case presentation</h3><div>A male born vaginally at 34 weeks of gestation was referred to our hospital at 11 days of life due to a 4-day history of feculent discharge from a right upper quadrant wound. His birth weight was 1.6 kg, and his Apgar scores were 8 and 9 at 1 and 5 minutes, respectively. He had a black blister in the right upper quadrant of the abdomen at birth, which started to ulcerate and increase in size in the following days. He was initially treated with intravenous antibiotics and wound care. On day of life 7, the wound began discharging feculent material. On arrival, his vital signs were within normal limits. A 4 × 4 cm ulcerated wound was noted in the right upper quadrant of the abdomen, with two separate fistula sites, consistent with ECF. The rest of the examination was unremarkable. Abdominal X-ray was normal. The patient was taken to the operating room for an abdominal exploration, which was done through a supraumbilical transverse laparotomy separate from the area of the ECF. We found a 0.5 × 0.5 cm perforation on the hepatic flexure of the colon and a large perforation in the ileum, 40 cm proximal to the ileocecal valve. The colonic perforation was primarily repaired, the perforated ileal segment was resected, and the intestinal continuity restored with an end-to-end anastomosis. The postoperative recovery was uneventful, and he was discharged on the sixth postoperative day. Histopathological examination of the resected bowel revealed normal bowel mucosa, with no evidence of ectopic gastric or pancreatic tissue.</div></div><div><h3>Conclusion</h3><div>Spontaneous ECF is an uncommon occurrence in neonates and children. Early surgical intervention is an effective management approach.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"112 ","pages":"Article 102929"},"PeriodicalIF":0.2,"publicationDate":"2024-11-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142705813","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Inflammatory myofibroblastic tumor of the sigmoid colon: A case report","authors":"Suleiman Ayalew ,&nbsp;Michael A. Negussie ,&nbsp;Messay Gebrekidan ,&nbsp;Dinksira Bekele Deneke ,&nbsp;Mulat Asrade Alemayehu ,&nbsp;Mesfin Tesera Wassie","doi":"10.1016/j.epsc.2024.102927","DOIUrl":"10.1016/j.epsc.2024.102927","url":null,"abstract":"<div><h3>Introduction</h3><div>Inflammatory myofibroblastic tumors (IMTs) are rare tumors primarily affecting children and young adults. IMT of the sigmoid colon is particularly rare, with very few cases reported in the literature.</div></div><div><h3>Case presentation</h3><div>A 2-year-old boy presented with a three-month history of painless, progressive abdominal swelling that began in the right lower abdomen and extended across to the left. He also experienced significant weight loss and reduced appetite, though there were no symptoms of nausea, vomiting, diarrhea, constipation, or rectal bleeding. On examination, a 10 cm firm, non-tender, and non-mobile mass was noted in the right lower quadrant and hypogastric region. No other abnormalities were detected. Laboratory results revealed severe anemia (hemoglobin: 6.1 g/dL) and elevated white blood cell (17,400/μL) and platelet (965,000/μL) counts. An abdominal ultrasound showed a large mass with associated lymphadenopathy, prompting further evaluation with contrast-enhanced CT. The scan revealed an avidly enhancing mesenteric mass near the superior mesenteric artery with minimal ascites and inguinal lymphadenopathy. The patient underwent laparotomy, during which a highly vascular mass originating from the sigmoid colon was resected, followed by colocolic anastomosis. Histopathological examination identified an intermediate-grade inflammatory myofibroblastic tumor with focal mucosal infiltration. Immunohistochemical staining confirmed the diagnosis, showing positivity for vimentin, smooth muscle actin, and focal desmin. Postoperatively, the patient recovered well, tolerated oral intake by day three, and was discharged on day six after a blood transfusion. At follow-up, he remained asymptomatic, with no recurrence on ultrasound and normalized hemoglobin levels.</div></div><div><h3>Conclusion</h3><div>IMT can occur in the gastrointestinal tract in children. Complete surgical resection is curative.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"112 ","pages":"Article 102927"},"PeriodicalIF":0.2,"publicationDate":"2024-11-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142705152","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}