Journal of Pediatric Surgery Case Reports最新文献

{"title":"Vascular lesion of the appendix causing torsion: A case report","authors":"Derek Marlor,&nbsp;Bayan Shalash,&nbsp;Vinay Prasad,&nbsp;Kathleen Nicol,&nbsp;Benjamin Kaumeyer,&nbsp;Dana Schwartz","doi":"10.1016/j.epsc.2024.102858","DOIUrl":"10.1016/j.epsc.2024.102858","url":null,"abstract":"<div><h3>Introduction</h3><p>Laparoscopic appendectomy is a common procedure for pediatric surgeons, primarily employed for acute appendicitis. However, this case report presents a unique scenario of a 12-year-old male with symptoms suggestive of appendicitis, diagnosed with an appendiceal mass causing torsion, due to a vascular malformation or reactive vascular proliferation.</p></div><div><h3>Case presentation</h3><p>A 12-year-old male presented with three days of abdominal pain, diarrhea, and emesis. The clinical exam was concerning for appendicitis. Ultrasonography revealed a mildly dilated appendix terminating in a hypervascular, lobulated mass, suggestive of a large solid appendiceal neoplasm. Subsequent CT scan further characterized this lesion that encased the mildly dilated appendix, compressing it and causing reactive appendiceal wall thickening. All laboratory studies were within normal limits. Diagnostic laparoscopy revealed a torsed, hemorrhagic mass originating from the distal aspect of the appendix. He underwent laparoscopic appendectomy, and the entire specimen was removed intact. Pathology reported a non-malignant vascular malformation or reactive vascular proliferation. The patient had an uncomplicated postoperative course and was managed like a standard postoperative appendectomy. One year following this procedure, he was readmitted with a large abdominal mass found to be Burkitt's lymphoma and was started on treatment. Pathology from the original appendiceal resection was reviewed and confirmed to be unrelated.</p></div><div><h3>Conclusion</h3><p>Vascular malformation/proliferation of the appendix is rare and may present similarly to appendicitis. Any mass may serve as a lead point for appendiceal torsion. Simple appendectomy is likely sufficient for such vascular lesions, and many pediatric appendiceal neoplasms.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"110 ","pages":"Article 102858"},"PeriodicalIF":0.2,"publicationDate":"2024-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000861/pdfft?md5=6e69500f1fb47696654b8b604ebf2e9b&pid=1-s2.0-S2213576624000861-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142150515","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":"Spindle cell lipoma, a rare and benign adipocytic tumor: A case report","authors":"Nitin Sajankila ,&nbsp;Amanda Graveson ,&nbsp;Veronica Ulici ,&nbsp;John DiFiore","doi":"10.1016/j.epsc.2024.102863","DOIUrl":"10.1016/j.epsc.2024.102863","url":null,"abstract":"<div><h3>Introduction</h3><p>Spindle cell lipomas are a rare, benign soft tissue mass that are easily treated with surgical excision. Although typically occurring in males and much later in life, in this case report, we present a case of a spindle cell lipoma occurring in a female teenager.</p></div><div><h3>Case presentation</h3><p>The patient is a healthy 16-year-old Female who presented with a soft, mobile, and poorly defined posterior flank mass on physical exam. As it was not well-visualized by ultrasound, a magnetic resonance image (MRI) was obtained, revealing a well-defined, T1 and T2 bright, ovoid lesion in the subcutaneous tissue. On MRI, the mass measured 7.4 x 4 × 4.5 cm and was consistent with a benign fatty mass. The mass was thought to be a lipoma and it was recommended that it be excised to confirm the diagnosis. However, the patient elected to defer due to concerns about undergoing surgery. Two years later she re-presented with a much larger mass in the same region and a surgical excision was then performed. On excision, the mass appeared smooth, glistening, and fatty, measuring 13.5 x 9.8 × 4.5 cm. Histology revealed encapsulated fibroadipose tissue containing both adipocytes and CD34-staining spindle cells, consistent with a diagnosis of spindle cell lipoma. The patient continues to do well 6 months later with no evidence of recurrence.</p></div><div><h3>Conclusion</h3><p>Spindle cell lipoma cannot be differentiated from lipomas pre-operatively.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"109 ","pages":"Article 102863"},"PeriodicalIF":0.2,"publicationDate":"2024-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000915/pdfft?md5=15185c3fc9525686604c90215f82ccf3&pid=1-s2.0-S2213576624000915-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142096393","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":"Ligamentum arteriosum calcification mimicking esophageal perforation: A case report","authors":"Abdulmalek Alzahrani,&nbsp;Mohammed Alsayed,&nbsp;Khalid Nassibi,&nbsp;Ali A. Alzahrani,&nbsp;Samira Alturkistany","doi":"10.1016/j.epsc.2024.102857","DOIUrl":"10.1016/j.epsc.2024.102857","url":null,"abstract":"<div><h3>Introduction</h3><p>The ligamentum arteriosum is a structure that connects the pulmonary artery to the aorta in the developing fetus. After birth it usually undergoes fibrous degenerative changes and calcification. Ligamentum arteriosum calcification (LAC) can be mistaken for an esophageal perforation, especially in patients with suspected esophageal foreign bodies or suspected esophageal injuries.</p></div><div><h3>Case presentation</h3><p>A 16-month-old boy underwent elective surgery for congenital lipomyelomeningocele. The endotracheal intubation was technically difficult due to an anterior position of the larynx. Postoperatively he developed high fever and an elevated white blood cell count, which were concerning for an esophageal perforation. A computerized tomography (CT) scan with oral contrast with the patient in left latera decubitus due to recent lower back surgery was done and showed a hyperdense line near the aortopulmonary window, resembling an esophageal contrast leak. Knowing that LAC was in the differential, and before proceeding with any major interventions for a presumed esophageal injury we did a barium swallow study, which confirmed that there was no esophageal perforation. The patient recovered well.</p></div><div><h3>Conclusion</h3><p>LAC is an uncommon condition that is difficult to distinguish from a contrast leak secondary to an esophageal perforation. A lack of awareness of condition can lead to unnecessary intervention.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"109 ","pages":"Article 102857"},"PeriodicalIF":0.2,"publicationDate":"2024-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S221357662400085X/pdfft?md5=546dca8de1e25476c80d7c1831cc008c&pid=1-s2.0-S221357662400085X-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142096392","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":"Hemorrhagic ulcerative gastric lymphangioma in an infant: A case report","authors":"Mohammad Tareq Rahimi,&nbsp;Abdul Wahab Amanat,&nbsp;Haseeb Rahman,&nbsp;Soghra Khaliqi,&nbsp;Nawaz Sharif Kashaf,&nbsp;Roohullah Hares","doi":"10.1016/j.epsc.2024.102855","DOIUrl":"10.1016/j.epsc.2024.102855","url":null,"abstract":"<div><h3>Introduction</h3><p>Lymphangiomas are congenital malformations of the lymphatic system, mostly presenting in the head and neck during early childhood. Its occurrence in the stomach is exceedingly rare with no reported cases in infants to date. Due to its asymptomatic nature and the limitations of imaging studies in providing a definitive diagnosis, gastric lymphangioma poses diagnostic and therapeutic challenges in children.</p></div><div><h3>Case presentation</h3><p>A 6-month-old female was brought to the emergency department with a three-day history of poor sucking, fever and feeding intolerance. In addition, she had been suffering from frequent non-bilious vomiting and hematemesis for two months. On physical exam, she appeared pale and drowsy. Her abdomen was soft, though slightly distended in the upper region with a small palpable mass in the epigastric area. An upper GI study revealed a dilated stomach and duodenum suggesting a duodenal obstruction. The only abnormal laboratory study was a hemoglobin level of 6 mg/dl. Laparotomy revealed an obstructive band between the duodenum and jejunum causing mild stenosis, which was released. A mass was found within the stomach. Through a gastrotomy we identified a cystic mass involving the body of the stomach and two-thirds of the antrum, with ulceration of its surface. The mass was completely excised and sent for histopathological examination. The stomach was then closed along the greater curvature as in a sleeve-gastrectomy fashion. The pathology report was consistent with an ulcerated lymphangioma. At 18 months of follow up she remains asymptomatic and has appropriate weight gain.</p></div><div><h3>Conclusion</h3><p>Gastric lymphangioma poses both diagnostic and surgical challenges in infants. Although rare in this population, it should be considered in patients presenting frequent vomiting and hematemesis.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"109 ","pages":"Article 102855"},"PeriodicalIF":0.2,"publicationDate":"2024-08-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000836/pdfft?md5=459e0066e32219ac7033a0b53db28aa7&pid=1-s2.0-S2213576624000836-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142058327","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":"Cloacal exstrophy variant in a female: A case report","authors":"Shelley Warner ,&nbsp;Luciana Lerendegui ,&nbsp;Monica D. Chow ,&nbsp;David Lasko ,&nbsp;Miguel Castellan ,&nbsp;Felipe Pedroso","doi":"10.1016/j.epsc.2024.102856","DOIUrl":"10.1016/j.epsc.2024.102856","url":null,"abstract":"<div><h3>Introduction</h3><p>Cloacal exstrophy is a rare birth defect that affects both the genitourinary and gastrointestinal tract, with an estimated incidence between 1:200,000 and 1:400,000 live births. The exact mechanism that leads to this defect is still unknown. Our purpose is to report a case of omphalocele-exstrophy-imperforate anus-spinal defect (OEIS) complex with a rare variant to add to the literature.</p></div><div><h3>Case presentation</h3><p>A 28-year-old female with no known medical history had a monochorionic diamniotic twin pregnancy. At 20 weeks of gestation a prenatal ultrasound demonstrated multiple fetal anomalies on twin B, including omphalocele, absent right kidney, non-visualization of bladder, and single umbilical artery. She had a scheduled Cesarean section at 34 weeks and 5 days of gestation. Twin B had a large omphalocele with an intact sac. Below the omphalocele, there was a cecal plate with a prolapsed ileum and a second small orifice corresponding to the hindgut. Two hemivaginas and hemiclitorises were appreciated inferior to the cecal plate but superior to a bladder plate bridge joining both halves. Each of the hemivaginas was connected to a hemiuterus with its own fallopian tube and ovary. The bladder was on both sides of the cecal plate and connected in the midline adopting a crescent shape with the concave side facing cephalad. Genetic testing confirmed an XX karyotype. On day-of-life 5 the patient underwent omphalocele repair and closure of the abdominal wall, tubularization of the cecal plate, and an end colostomy. Additionally, the hemivaginas and hemiclitorises were relocated to an orthotopic position and the bladder plates were joined together and used to cover part of the abdominal wall defect. She was discharged on day of life 42 with a weight of 2.72 kg. She continues to follow up outpatient. Her second stage procedure is still pending.</p></div><div><h3>Conclusion</h3><p>The position of the genital tract superior to the bladder exstrophy observed in this case has not been described before and is not fully explained by the current theories on the development of OEIS.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"109 ","pages":"Article 102856"},"PeriodicalIF":0.2,"publicationDate":"2024-08-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000848/pdfft?md5=b53e9ec1d70b607c6eb43f08b9aa780c&pid=1-s2.0-S2213576624000848-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142117719","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 pneumomediastinum in previously healthy pediatric patients: A case series","authors":"Isabel Cristina Brito Rojas ,&nbsp;Mayra Angelica Hernandez Peñuela ,&nbsp;Martin Alfonso La rotta ,&nbsp;Vanessa Medina Gaviria","doi":"10.1016/j.epsc.2024.102854","DOIUrl":"10.1016/j.epsc.2024.102854","url":null,"abstract":"<div><h3>Introduction</h3><p>Spontaneous pneumomediastinum is defined as the presence of air in the mediastinum not associated with trauma or iatrogenic causes. This pathology is rare in the pediatric population.</p></div><div><h3>Cases presentation</h3><p>Case 1: A previously healthy 15-year-old male was admitted with a 5-day history of sudden-onset chest pain associated with odynophagia. Upon admission, there were no signs of respiratory distress, but slight subcutaneous emphysema in the neck. A neck X-ray demonstrated extensive prevertebral and paracervical air. Chest x-ray showed increased transparency and delineation of the mediastinal contours as well as increased retrosternal space, all compatible with pneumomediastinum. Chest CT showed extensive emphysema of the supra- and infrahyoid anterior neck bilaterally. Neither the esophagram nor the upper endoscopy reveal an esophageal lesion. The patient remained stable without the need for supplemental oxygen, started a liquid diet 72 hours post admission with adequate tolerance, and was discharged home on the fourth day of hospitalization. Case 2: A previously healthy 10-year-old female was admitted to the emergency department with a clinical picture of odynophagia that prior to admission was associated with signs of respiratory distress and desaturation. Chest X-ray showed subcutaneous emphysema in the left cervical, thoracic, and axillary regions. Chest CT scan with oral contrast showed emphysema of the soft tissues of the left chest wall and neck and no extravasation. Upper endoscopy showed no lesions in the upper digestive tract. She resumed oral feedings 2 days post admission and was discharged home 6 days post admission.</p></div><div><h3>Conclusion</h3><p>Spontaneous pneumomediastinum is an infrequent benign clinical entity in children, especially in those with no history of lung disease, infections or substance abuse. Conservative treatment is successful in most cases. Rapid reinitiation of oral feedings does not increase complications and allows an early discharge.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"108 ","pages":"Article 102854"},"PeriodicalIF":0.2,"publicationDate":"2024-08-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000824/pdfft?md5=1be1f5ac8aa72d717500d9ce01bfcf3c&pid=1-s2.0-S2213576624000824-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142040142","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":"Sacrococcygeal teratoma and jejunoileal atresia: A case report","authors":"Jamie E. Anderson ,&nbsp;Shinjiro Hirose ,&nbsp;Amelia S. McLennan ,&nbsp;Zahabiya H. Chithiwala ,&nbsp;Payam Saadai","doi":"10.1016/j.epsc.2024.102853","DOIUrl":"10.1016/j.epsc.2024.102853","url":null,"abstract":"<div><h3>Introduction</h3><p>Sacrococcygeal teratomas (SCTs) and jejunoileal atresias are both rare (1 in 35,000–40,000 births and 1 in 5000 births, respectively). We present a case of a concomitant diagnosis of a cystic sacrococcygeal teratoma and a jejunoileal atresia.</p></div><div><h3>Case presentation</h3><p>A twenty-week screening ultrasound identified a cystic pelvic mass (5.5 x 2.8 × 2.2 cm), dilated bowel, and mild polyhydramnios in an otherwise healthy G1P0 woman. Fetal magnetic resonance image showed a multiseptated presacral cystic mass suspicious for an internal sacrococcygeal teratoma and dilated bowel, thought to represent dilated ascending and transverse colon. Scheduled delivery at 39 weeks of gestation was uncomplicated. Postnatal ultrasound and MRI were consistent with prenatal findings. Given the rarity of both congenital anomalies and no known association between SCTs and jejunoileal atresia, the prenatal bowel dilation was thought to be due to partial obstruction secondary to the SCT. The baby thus underwent SCT resection on day-of-life 2. Ongoing obstruction after resection prompted a contrast enema which demonstrated a microcolon with reflux into the terminal ileum but no reflux into more dilated proximal loops concerning for intestinal atresia. On day-of-life 5, the baby underwent exploratory laparotomy which identified a type-2 ileal atresia, and a primary anastomosis was performed. The patient had an uneventful recovery and was discharged home on day-of-life 22.</p></div><div><h3>Conclusion</h3><p>Prenatal findings of dilated bowel should prompt providers to have a high index of suspicion for jejunoileal atresia, even if other diagnoses exist.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"108 ","pages":"Article 102853"},"PeriodicalIF":0.2,"publicationDate":"2024-08-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000812/pdfft?md5=263fd2308eba45283a6c1a18f013d6bf&pid=1-s2.0-S2213576624000812-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141998151","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":"Cat scratch disease requiring incision and drainage: A case report","authors":"Emile Jeunesse ,&nbsp;Natalie Miller ,&nbsp;Lisa Carroll ,&nbsp;Mary Arbuthnot","doi":"10.1016/j.epsc.2024.102851","DOIUrl":"10.1016/j.epsc.2024.102851","url":null,"abstract":"<div><h3>Introduction</h3><p>Cat Scratch Disease (CSD) is a relatively novel disease that often resolves on its own or with antibiotic therapy and rarely requires surgical intervention. We present a case of CSD with a variable presentation that required surgical incision and drainage for resolution.</p></div><div><h3>Case presentation</h3><p>This is a case of a 13-year-old African American male who presented with a 5cm × 8cm right-sided neck mass, with positive IgM titers for <em>Bartonella henselae</em> and parainfluenza virus following reported cat scratches. He was treated with a partial course of clindamycin before the diagnosis of CSD was made, then completed a five-day course of azithromycin. After three weeks with no resolution of systemic symptoms or reduction in mass size or pain, there was concern for malignancy. The mass was ultimately determined to be an enlarged lymph node and abscess via computed tomography and biopsy. It was surgically incised and drained, and a second five-day course of azithromycin was completed to prevent fistula formation. The surgery effectively treated the antibiotic resistant CSD, and resolution of systemic symptoms followed rapidly.</p></div><div><h3>Conclusion</h3><p>This case report highlights a variable presentation of antibiotic resistant CSD, and that biopsy may be indicated in lymphadenopathy to rule out malignancy. Surgery for CSD is rare, but incision and drainage may be required for abscess formation with CSD, especially with concern for mass effect, and we recommend early surgical referral for these situations regardless of diagnosis.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"108 ","pages":"Article 102851"},"PeriodicalIF":0.2,"publicationDate":"2024-08-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000794/pdfft?md5=7f9ec3d17de9db07cfc5e4d052f198ae&pid=1-s2.0-S2213576624000794-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141998150","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":"Ciliated cyst of the liver: A case report","authors":"Yassine Derouich ,&nbsp;Kamal El Haissoufi ,&nbsp;Abdelouhab Ammor ,&nbsp;Houssain Benhaddou","doi":"10.1016/j.epsc.2024.102852","DOIUrl":"10.1016/j.epsc.2024.102852","url":null,"abstract":"<div><h3>Introduction</h3><p>Ciliated cyst of the liver is rarely seen in the pediatric population and considered as a benign tumor often discovered incidentally.</p></div><div><h3>Case presentation</h3><p>We report the case of a 12-year-old child with a history of contact with dogs, admitted to our department of pediatric surgery because of an abdominal trauma. He had no significant medical history. All blood tests were within normal limits. A computed tomography scan was performed and revealed a 60-mm cystic lesion with dense contents in segment IV of the liver, suspicious for a biliary cyst or a hydatid cyst. Magnetic resonance imaging was in line with the diagnosis of a type I hydatid cyst of the liver according to the Gharbi classification. The patient underwent a laparoscopic resection of the cyst without spilling any content in the peritoneal cavity. The pathological examination of the specimen confirmed the presence of a cystic formation lined by ciliated pseudostratified epithelium without signs of infection or malignancy, confirming the diagnosis of a ciliated cyst of the liver. The postoperative course was uneventful. At 6 months of follow up he remains asymptomatic and has a normal abdominal ultrasound.</p></div><div><h3>Conclusion</h3><p>Ciliated cysts should be included in the differential diagnosis of cystic lesions of the liver. The therapeutic management is a subject of debate.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"108 ","pages":"Article 102852"},"PeriodicalIF":0.2,"publicationDate":"2024-08-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000800/pdfft?md5=38390c81f04eac39b42b746d72258ef8&pid=1-s2.0-S2213576624000800-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141979907","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":"Association between visceral myopathy and Hirschsprung's disease: A case report","authors":"María Jesús Nally R ,&nbsp;Isidora Lavado C ,&nbsp;Rodrigo Maluje J ,&nbsp;Miguel Guelfand C ,&nbsp;Daniel Rojo V","doi":"10.1016/j.epsc.2024.102850","DOIUrl":"10.1016/j.epsc.2024.102850","url":null,"abstract":"<div><h3>Introduction</h3><p>The coexistence of visceral myopathy (VM) and Hirschsprung's disease (HD) is exceptionally rare yet clinically significant. Both conditions necessitate full-thickness biopsy for precise diagnosis. Despite potential similarities in presentation, no documented cases have established an association between these two disorders.</p></div><div><h3>Case presentation</h3><p>We present a 3150-g, full-term male newborn, four days old, born without maternal pathologies or antenatal diagnoses. He was admitted to the emergency department with one day of fecal vomiting, lethargy, and decreased intake. On admission, he exhibited hypothermia, tachycardia, and his oxygen saturation was 94%. Physical examination revealed a septic, dehydrated, poorly perfused newborn with a distended abdomen and absent bowel sounds. Abdominal X-ray indicated pneumoperitoneum. Emergency laparotomy revealed colonic perforation necessitating resection and colonic anastomosis, with the abdomen left open and covered by a dressing, followed by a loop-ileostomy in the subsequent surgery. Surgical biopsies, including a later laparoscopic intestinal mapping, confirmed VM and HD. The patient progressed with complete oral feeding, normal bowel transit through the ileostomy, and no associated complications. Currently, the patient awaits resection of the aganglionic segment and rectal pull-through.</p></div><div><h3>Conclusion</h3><p>This case may suggest either an incidental finding or an undiscovered common etiology linking HD and VM in neonatal intestinal perforation. Comprehensive diagnostic approaches, personalized treatments, and further exploration of their genetic and developmental connections are crucial for improving patient outcomes.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"108 ","pages":"Article 102850"},"PeriodicalIF":0.2,"publicationDate":"2024-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000782/pdfft?md5=7c44077484af480ee21eb6b1d772f9f0&pid=1-s2.0-S2213576624000782-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141979908","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}