Journal of Pediatric Surgery Case Reports最新文献_第4页

Magnetic functional undiversion in a patient with enteroatmospheric fistula and hostile abdomen: a case report 磁功能不转移治疗肠大气瘘伴敌对腹部1例报告

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2026-01-02 DOI: 10.1016/j.epsc.2026.103171

Cynthia Juliana Hernandez Yate , Nidia Catalina León Rodríguez , Germán Aquilino Mogollón Cruz , Diana Alejandra Holguin Sanabria , Pablo Eliecer Luengas Perez , Isabel Gutierrez Soto

Introduction

Enteroatmospheric fistula in the setting of a hostile abdomen represents a major surgical challenge. Conventional anastomotic techniques are often unfeasible due to friable tissue, extensive adhesions, and high operative risk. Magnetic compression anastomosis has emerged as a minimally invasive alternative for restoring intestinal continuity in complex surgical scenarios.

Case presentation

We report the case of a 14-year-old male with total colonic aganglionosis diagnosed in early childhood and initially managed with a diverting colostomy. Definitive reconstruction was delayed due to loss of follow-up after relocation to a remote region with limited access to specialized pediatric surgical care. At 14 years of age, the patient underwent intestinal reconstruction, which was complicated by an anastomotic leak, multiple laparotomies, and the development of a single enteroatmospheric fistula in the setting of a persistent hostile abdomen.

Given the high surgical risk, the patient was not considered a candidate for conventional reoperation. A custom-made magnetic compression device was therefore designed to adapt to the patient's altered intestinal anatomy and the absence of commercially available magnetic systems for pediatric functional undiversion in the region. The device consisted of two annular neodymium magnets coated with methyl methacrylate, one housed within a convex 3D-printed thermoplastic polyurethane cover, and was placed endoluminally under endoscopic guidance. Also radiographic flow up was made.

The magnets achieved proper alignment and were spontaneously expelled on postoperative day 7. By postoperative day 14, intestinal transit was restored and enteral feeding was resumed without evidence of leak or infection, achieving functional closure of the fistula. Due to severe loss of abdominal wall domain, abdominal wall closure was managed by secondary intention.

Conclusion

Magnetic compression anastomosis seems to be a safe and effective alternative for the management of single enteroatmospheric fistulas.

腹壁不良的肠大气瘘是一个主要的外科挑战。由于组织易碎，粘连广泛，手术风险高，传统的吻合技术往往不可行。在复杂的手术情况下，磁压缩吻合术已成为恢复肠道连续性的一种微创替代方法。我们报告一例14岁男性全结肠神经节病的诊断在儿童早期和最初处理与转移结肠造口。由于搬迁到偏远地区，无法获得专门的儿科外科护理，最终的重建被推迟。14岁时，患者进行了肠道重建，术后出现吻合口漏，多次剖腹手术，并在持续敌对腹部的情况下出现了单一的肠-大气瘘。考虑到手术风险高，患者不考虑常规再手术。因此，我们设计了一种定制的磁性压缩装置，以适应患者肠道解剖结构的改变和该地区缺乏商用磁性系统的儿童功能不转移。该装置由两个涂有甲基丙烯酸甲酯的环形钕磁铁组成，其中一个安装在一个凸3d打印热塑性聚氨酯盖内，并在内窥镜引导下放置。同时也进行了射线成像。术后第7天，磁体达到了正确的排列并自发排出。术后第14天，肠道运输恢复，肠内喂养恢复，无渗漏或感染迹象，实现功能性瘘口闭合。由于腹壁区域的严重丧失，腹壁关闭是由次要意图管理。结论磁压吻合术是一种安全、有效的治疗单肠大气瘘的方法。

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引用次数: 0

Acute appendicitis presenting as acute scrotum in a toddler: A case report 小儿急性阑尾炎表现为急性阴囊：1例报告

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2026-01-02 DOI: 10.1016/j.epsc.2026.103177

Sean Gannon , Ibraheem Abdelraheem

Introduction

The acute scrotum is a common paediatric emergency presentation most often caused by testicular torsion, epididymo-orchitis, or incarcerated inguinal hernia. We present the rare occurrence of it occurring secondary to perforated appendicitis, further complicated by a post-operative pyocoele requiring surgical intervention.

Case

A 2-year-old male initially presented with Influenza A infection and subsequently developed right-sided scrotal swelling, erythema, and tenderness. Clinical examination revealed right iliac fossa tenderness, and ultrasonography demonstrated perforated appendicitis with associated right-sided epididymo-orchitis, without a scrotal collection. The patient underwent laparoscopic appendectomy and was commenced on intravenous ceftriaxone and metronidazole. Despite initial improvement, scrotal swelling and inflammatory markers persisted. Ultrasonography six days postoperatively identified a right-sided pyocoele, prompting escalation of antibiotic therapy; however, symptoms failed to improve. Repeat imaging three days later demonstrated interval enlargement of the collection. Surgical drainage was performed via a transverse right scrotal incision, and temporary drain placement. Although a patent processus vaginalis was not identified intraoperatively, its involvement was considered likely given the clinical course. The patient improved following drainage but re-presented with recurrent pyrexia, requiring recommencement of intravenous antibiotics and a prolonged course of outpatient broad-spectrum oral therapy with metronidazole and cefalexin, requiring over six weeks of antibiotic therapy in total.

Conclusion

Patients with acute appendicitis and a concomitant acute scrotum should be evaluated for a patent processus vaginalis, as the acute scrotum may be caused by purulent fluid from the abdomen accumulating in the tunica vaginalis. Surgical drainage of the purulent fluid may be required.

急性阴囊是一种常见的儿科急症，通常由睾丸扭转、附睾-睾丸炎或嵌顿性腹股沟疝引起。我们报告罕见的继发于穿孔性阑尾炎，术后并发脓腔膨出，需要手术干预。病例1:2岁男性，最初表现为甲型流感感染，随后出现右侧阴囊肿胀、红斑和压痛。临床检查显示右侧髂窝压痛，超声检查显示阑尾炎穿孔伴右侧附睾-睾丸炎，无阴囊积液。患者行腹腔镜阑尾切除术，并开始静脉注射头孢曲松和甲硝唑。尽管最初有所改善，但阴囊肿胀和炎症标志物持续存在。术后6天超声检查发现右侧脓腔，促使抗生素治疗升级；然而，症状并没有好转。三天后重复成像显示间歇增大的集合。通过右侧阴囊横向切口进行手术引流，并临时放置引流管。虽然术中未发现阴道突未闭，但考虑到临床病程，其累及被认为是可能的。引流后患者好转，但再次出现反复发热，需要重新静脉注射抗生素，并延长门诊甲硝唑和头孢氨苄广谱口服治疗疗程，总共需要6周以上的抗生素治疗。结论急性阑尾炎合并急性阴囊的患者，可能是由于腹部的化脓性液体积聚在阴道膜内而引起的急性阴囊，应对其进行诊断。可能需要外科引流脓性液体。

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引用次数: 0

Pedunculated mesenchymal hamartoma of the left lobe of liver in a 3-year-old child: a case report 3岁儿童肝左叶有带蒂间充质错构瘤1例

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2026-01-02 DOI: 10.1016/j.epsc.2026.103170

Sweta Rose , Kamal Nain Rattan , Shivani Malik

Background

Hepatic mesenchymal tumor is a rare benign pediatric hepatic tumor, typically presenting in children less than 2 years of age. Although it has a variable imaging appearance, and favorable prognosis if diagnosed and managed correctly.

Case presentation

A 3-year-old male patient presented with abdominal distension, a large abdominal mass with rapid evolution over four weeks, and poor weight gain. His family and medical history were unremarkable. Imaging studies revealed a large solid-cystic mass arising from the left lobe of the liver. Laboratory parameters were within normal limits. He underwent exploratory laparotomy; careful dissection was undertaken to mobilize the left lobe. The left hepatic artery, portal vein, and left hepatic duct were meticulously isolated and controlled. The tumor was removed intact while preserving the surrounding liver parenchyma. Macroscopic and histological examination described an 18 × 13 × 6 cm multicystic mass consistent with mesenchymal hamartoma of the liver (MHL). The patient remains well at 5-month follow-up, with no evidence of lesion recurrence.

Conclusion

Mesenchymal hamartoma should be included in the differential diagnosis of hepatic masses in children. Complete resection is curative.

背景：肝间充质瘤是一种罕见的儿童良性肝脏肿瘤，多见于2岁以下儿童。虽然它有不同的影像学表现，但如果诊断和处理得当，预后良好。1例3岁男性患者，表现为腹胀，腹部有大肿块，四周内发展迅速，体重增加不明显。他的家庭和病史一般。影像学检查显示肝左叶有一大块实性囊性肿块。实验室参数在正常范围内。他接受了剖腹探查术；进行仔细的解剖以活动左叶。左肝动脉、门静脉、左肝管被严密隔离和控制。肿瘤被完整切除，同时保留了周围的肝实质。肉眼和组织学检查显示一个18 × 13 × 6 cm的多囊肿块，符合肝脏间充质错构瘤（MHL）。患者在5个月的随访中保持良好，无病变复发的迹象。结论间充质错构瘤应列入儿童肝脏肿块的鉴别诊断。完全切除是可治愈的。

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引用次数: 0

Ovarian fibroma in a 14-month-old girl: A case report 14个月女童卵巢纤维瘤1例

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2026-01-02 DOI: 10.1016/j.epsc.2026.103172

Jennifer Osher , Caleb Brown , Mary Morgan Barron , Ismael Kaitama , Patrick Costello , Andrew Trecartin

Introduction

Ovarian fibromas are rare in children, and cellular fibromas with mitotic activity are exceptionally uncommon, with fewer than ten pediatric cases reported. Their preoperative differentiation from malignant tumors is challenging.

Case presentation

We report a 14-month-old girl who was hospitalized for malarial fevers in addition to an abdominal mass. She had a prior two-month history of painless abdominal distention with a palpable, non-tender lower abdominal mass. The patient had mild anemia (hemoglobin of 11.2 g/dl). Tumor markers were unavailable. Ultrasound demonstrated an 8 × 5 cm solid pelvic mass. The mass was well circumscribed with a homogeneous echotexture. Laparotomy revealed no evidence of metastatic disease, and the contralateral ovary was normal. A solid tumor of the right ovary without visible normal ovarian stroma was identified. The highly vascular tumor capsule was adherent to the right fallopian tube, which was carefully separated and preserved. The bisected tumor was notable for pale tan homogeneous tissue within the tumor capsule. Histopathology demonstrated a cellular fibroma with focal necrosis and rare mitoses. The patient was tolerating a regular diet and ambulating well within 24 hours postoperatively. Her one-week follow-up visit confirmed no complications. The family was instructed to follow up annually, with plans for serial abdominal ultrasounds and chest radiographs.

Conclusion

As benign ovarian tumors such as cellular fibroma can occur in females as young as one year of age, in the absence of a definitive preoperative diagnosis of malignancy surgeons should prioritize fertility preservation over radical salpingo-oophorectomy.

卵巢纤维瘤在儿童中很少见，具有有丝分裂活性的细胞纤维瘤尤其罕见，据报道儿科病例不到10例。术前与恶性肿瘤的鉴别具有挑战性。我们报告一个14个月大的女孩，她因疟疾发烧和腹部肿块而住院。她有两个月的无痛性腹胀史，下腹有可触及的无痛性肿块。患者有轻度贫血（血红蛋白11.2 g/dl）。肿瘤标志物不可用。超声示8 × 5厘米实性盆腔肿块。肿块边界清晰，回声均匀。剖腹探查未发现转移性疾病，对侧卵巢正常。右卵巢实体瘤，未见正常卵巢间质。高度血管性肿瘤囊贴附于右侧输卵管，仔细分离保存。切分的肿瘤被膜内可见浅褐色的均匀组织。组织病理学表现为细胞纤维瘤伴局灶性坏死和罕见的有丝分裂。患者饮食正常，术后24小时内活动良好。一周的随访证实无并发症。该家庭被指示每年进行随访，并计划进行连续的腹部超声波检查和胸部x光检查。结论良性卵巢肿瘤如细胞纤维瘤可发生于1岁以下的女性，在术前没有明确的恶性肿瘤诊断的情况下，外科医生应优先考虑保留生育能力而不是根治性输卵管卵巢切除术。

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引用次数: 0

Ileo-umbilical enterocutaneous fistula as the first manifestation of pediatric Crohn’s disease: a case report 儿童克罗恩病的第一表现为回脐肠皮瘘1例

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2025-12-30 DOI: 10.1016/j.epsc.2025.103163

Medhat Elsayed , Sarah Magdy Abdelmohsen , Eiman Yassir Musa Hussain , Ekram Imam , Youmna Medhat , Tahani Helali

Background

Enterocutaneous fistula (ECF) is an uncommon and challenging complication of Crohn's disease in the pediatric population.

Case Presentation

A 14-year-old female presented with a one-year history of chronic abdominal pain, weight loss, and a 6-month history of persistent feculent umbilical discharge following a prior "abscess" drainage. A CT scan revealed a fistula tract connecting the umbilicus to an inflammatory mass in the ileocecal region. She was optimized with nutritional support and antibiotics to correct her fever and elevated inflammatory markers. She then underwent a laparoscopic ileocecal resection. The bowel was connected to the umbilicus via a fistula tract, not dense adhesions. The bowel was exteriorized, and an ileo-colic anastomosis was performed with a linear stapler. Her recovery was uneventful. Pathology confirmed Crohn's disease. At one-year follow-up, she was in remission on medication, with full wound healing and weight gain.

Conclusion

Crohn's disease should be suspected in patients who have a history of chronic abdominal pain, weight loss, and chronic purulent umbilical discharge, as the discharge can be an entero-cutaneous fistula.

背景肠皮瘘（ECF）是儿科人群中罕见且具有挑战性的克罗恩病并发症。病例介绍：一名14岁女性，慢性腹痛，体重减轻，既往“脓肿”引流后持续脐带脓性分泌物6个月。CT扫描显示连接脐和回盲区炎性肿块的瘘道。她得到了最佳的营养支持和抗生素，以纠正她的发烧和升高的炎症标志物。然后她接受了腹腔镜回盲切除术。肠道通过瘘道与脐部相连，没有密集的粘连。将肠取出，用线性吻合器进行回肠-结肠吻合。她的康复平安无事。病理证实是克罗恩病在一年的随访中，她的药物治疗缓解，伤口完全愈合，体重增加。结论有慢性腹痛、体重减轻、慢性脐脓性分泌物病史的患者应怀疑克罗恩病，因为分泌物可能为肠-皮瘘。

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引用次数: 0

Congenital esophageal stenosis in an eight-year-old boy: a case report 先天性食管狭窄1例8岁男童

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2025-12-30 DOI: 10.1016/j.epsc.2025.103169

Mohammad Shafiqi , Mujtaba Yama , Behnaz Rahimi , Dunya Moghul

Introduction

Congenital esophageal stenosis is one of the rarest congenital anomalies in children, with an incidence of approximately 1 in 25,000 to 50,000 live births. Esophagography is the primary diagnostic tool used for its identification.

Case report

An eight-year-old boy had dysphagia to solid foods since the age of six months. In the two days prior to his presentation to the hospital he developed persistent vomiting even after drinking water. He was malnourished, weak and had a weight of 18 kg. A contrast swallow study was initially interpreted as a caustic injury, but no history of ingestion was found. Due to the persistence of symptoms after two balloon dilatations done two weeks apart, congenital esophageal stenosis was suspected. The patient was taken to the operative room for an exploratory thoracotomy through the right side. We dissected the esophagus and found a fibrotic stenotic segment measuring 2 cm in length. We resected the segment and proceeded with an end-to-end anastomosis using interrupted PDS 3.0 sutures. A contrast swallow study done on postoperative day 5 confirmed no esophageal leak. We initiated clear liquid diet that day and advanced to soft diet the following day. He was discharged home on the 6th postoperative day. During the first year after the operation, we saw him monthly and confirmed that he remained asymptomatic. By the end of the first year his weight was 25 kg, and he continued to be without symptoms.

Conclusion

Congenital esophageal stenosis must be suspected in children who have a long-standing history of dysphagia.

先天性食管狭窄是儿童中最罕见的先天性异常之一，发病率约为1 / 25,000至50,000活产。食管造影是主要的诊断工具。病例报告：一名8岁男孩自6个月大时便出现固体食物吞咽困难。在他到医院就诊前两天，他甚至在喝水后也出现持续呕吐。他营养不良，身体虚弱，体重只有18公斤。对比吞咽研究最初被解释为腐蚀性损伤，但没有发现摄入史。由于间隔两周进行两次球囊扩张后症状持续，怀疑先天性食管狭窄。患者被送往手术室，通过右侧进行探查性开胸手术。我们解剖食道，发现一个长度为2厘米的纤维化狭窄段。我们切除了该节段，并使用中断的PDS 3.0缝合进行端到端吻合。术后第5天进行的对比吞咽研究证实没有食管渗漏。那天我们开始吃清流食，第二天开始吃软食。术后第6天出院。在手术后的第一年，我们每月检查一次，确认他没有任何症状。到第一年结束时，他的体重达到了25公斤，而且他仍然没有任何症状。结论有长期吞咽困难病史的儿童应怀疑先天性食管狭窄。

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引用次数: 0

Tension viscerothorax ensuing blunt abdominal trauma: a case report 钝性腹部外伤致张力性脏器胸1例

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2025-12-24 DOI: 10.1016/j.epsc.2025.103168

Daniel Lamb , Christopher Huffman , Dan Parrish

Introduction

Tension viscerothorax is an infrequent but potentially deadly complication of traumatic diaphragmatic rupture. Abdominal organ herniation into the thoracic cavity results in increased intrathoracic pressure, thus mimicking a tension pneumothorax with subsequent physiological derangements.

Case presentation

A 15-year-old female was medevacked to our hospital after sustaining significant blunt thoracoabdominal trauma secondary to a high-speed Motor vehicle crash (MVC). On arrival the patient was hemodynamically unstable and hypoxic despite being endotracheal intubation (ETT) intubated with ongoing bag valve mask (BVM) ventilation. A left thoracostomy was emergently performed, oxygen saturations improved momentarily thereafter. Exam revealed a prominent seatbelt sign and left thoracoabdominal bruising with abrasions. Computer tomography (CT) of the chest, abdomen and pelvis revealed a left traumatic diaphragmatic rupture, left hemothorax with mediastinal shift, grade 2 splenic laceration, grade 3 left kidney laceration, grade 2 left liver lobe laceration, grade 1–2 pancreatic injury, and an unstable L1-L2 flexion injury concerning for possible spinal cord injury, along with a comminuted left scapular fracture. The patient was taken for an emergent left anterior thoracotomy, revealing omentum, stomach, and spleen within the left thoracic cavity obscuring the diaphragmatic injury. A midline laparotomy was performed, to allow visualization and repair of the diaphragm which revealed transected jejunum. A temporary abdominal closure system was placed. Two days later she returned for washout with anastomosis. The patient ultimately underwent additional orthopedic surgeries, along with a prolonged hospitalization including rehabilitation but was ultimately discharged home.

Conclusion

Tension viscerothorax must be considered in children who sustain severe blunt thoracoabdominal trauma, as it can cause life-threatening physiological derangements.

张力性脏器胸是外伤性膈破裂的一种少见但可能致命的并发症。腹部器官疝入胸腔导致胸内压力增加，从而模仿紧张性气胸并伴有随后的生理紊乱。病例介绍：一名15岁的女性在高速机动车碰撞（MVC）后持续严重钝性胸腹外伤，被紧急送往我院。到达时，患者血流动力学不稳定且缺氧，尽管气管内插管（ETT）插管并持续进行袋阀面罩（BVM）通气。紧急进行了左开胸术，此后氧饱和度短暂改善。检查发现一个明显的安全带痕迹和左胸腹挫伤和擦伤。胸部、腹部和骨盆的计算机断层扫描（CT）显示左侧创伤性膈破裂，左侧血胸伴纵隔移位，2级脾撕裂伤，3级左肾撕裂伤，2级左肝叶撕裂伤，1-2级胰腺损伤，以及可能的脊髓损伤的不稳定L1-L2屈曲损伤，同时伴有左侧肩胛骨粉碎性骨折。患者接受紧急左前开胸手术，发现左胸腔内的大网膜、胃和脾脏，掩盖了膈肌损伤。进行中线剖腹术，使横切空肠的横切膈可见并修复。放置了一个临时腹部闭合系统。两天后返回冲洗吻合。患者最终接受了额外的骨科手术，以及包括康复在内的长期住院治疗，但最终出院回家。结论儿童严重钝性胸腹外伤可引起危及生命的生理紊乱，必须考虑张力性脏器胸。

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引用次数: 0

Letter to editor: Raising awareness for children with CDH1 mutations 致编辑的信：提高对CDH1突变儿童的认识

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2025-12-24 DOI: 10.1016/j.epsc.2025.103167

Jeremy L. Davis

引用次数: 0

Extralobar pulmonary sequestration with a transdiaphragmatic gastric attachment: a case report 经膈胃附著的肺叶外肺隔离一例报告

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2025-12-18 DOI: 10.1016/j.epsc.2025.103166

Rayan A. Rahmani , Johanna M. Borst , David F. Grabski , Amir M. Alhajjat , Mehul V. Raval

Introduction

Extralobar pulmonary sequestration (EPS) has been reported in intradiaphragmatic or intraabdominal locations, but simultaneous involvement of multiple compartments is exceedingly rare. This is the first reported case to our knowledge with simultaneous intrathoracic, transdiaphragmatic, and gastric involvement.

Case presentation

A 21-month-old male was prenatally diagnosed with a left lung lesion via ultrasound. A computed tomography (CT) scan with contrast performed at 3 months of age at an outside center was consistent with a left lower lobe hybrid lesion. The family did not wish to undergo surgery at that time. A repeat CT scan with contrast was performed at 15 months of age for better visualization of the anatomy. The patient underwent video-assisted thoracoscopic surgery, which revealed an extrapulmonary mass with feeding vessels from the aorta and a fibrous transdiaphragmatic connection to the stomach. Intraoperative upper endoscopy confirmed no esophageal or gastric luminal involvement. The lesion was resected, and the diaphragm was primarily closed thoracoscopically. Pathology demonstrated extrapulmonary sequestration with type 2 congenital pulmonary airway malformation (CPAM)-like features. The patient recovered well and was discharged on postoperative day two.

Conclusion

Congenital lung lesions may have transdiaphragmatic extensions to the digestive tract not detectable by preoperative imaging studies.

肺叶外肺隔离（EPS）在膈内或腹内位置有报道，但同时累及多个肺室是非常罕见的。据我们所知，这是第一例胸内、横膈膜和胃同时受累的病例。一个21个月大的男婴在产前通过超声诊断为左肺病变。在3个月大时，在中心外侧进行的计算机断层扫描（CT）与造影剂扫描符合左下叶混合性病变。家人当时不希望接受手术。在15个月大时进行了重复CT扫描和对比，以便更好地观察解剖结构。患者接受电视胸腔镜手术，发现肺外肿块，有主动脉供血血管和经膈纤维连接胃。术中上腔镜检查证实没有食管或胃腔受累。病变被切除，横膈膜主要在胸腔镜下闭合。病理表现为肺外隔离伴2型先天性肺气道畸形（CPAM）样特征。患者恢复良好，术后第2天出院。结论先天性肺病变可经膈肌延伸至消化道，术前影像学检查无法发现。

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引用次数: 0

Heteropagus twins: A case series 异翼龙双胞胎：一个案例系列

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports

Pub Date : 2025-12-17 DOI: 10.1016/j.epsc.2025.103165

Martial Olivier Moulot , Jean Marie Konan , Agnime Reine - Christine Akproh , Julie Konan , Manuela Adjoba Ehua , Kouamé Soroboua Agbara

Introduction

Heteropagus (parasitic) twinning is an exceptionally rare congenital anomaly characterized by an incompletely formed parasitic twin attached to a more developed autosite. We present the first case series from our region, illustrating unusual anatomical variants and complex visceral associations seldom reported in the literature.

Case presentation

Over the last two decades, 4 newborns with heteropagus twinning were surgically managed. The first patient was a full-term newborn with a well-developed third lower limb implanted in the right iliac region and a rudimentary upper limb attached above the pubis. Both additional limbs were excised successfully with an uneventful recovery. The second patient presented with duplicated genitalia, double bladder, colonic duplication, patent ductus arteriosus and a ventricular septal defect). Complete surgical correction led to a favorable long-term outcome. The third patient had a gluteal parasitic limb associated with a sacrococcygeal teratoma, renal dysplasia, and genital duplication. He died on day of life 11 from cardiorespiratory failure before any resection was attempted. The fourth patient had an epigastric parasitic twin with a large omphalocele containing rudimentary intestine. Separation was completed at one month of age, but the patient died on the first postoperative day.

Conclusion

Heteropagus (parasitic) twinning can present with multiple phenotypic variants. Visceral and particularly cardiac malformations play a key prognostic role.

异位（寄生）双胞胎是一种非常罕见的先天性异常，其特征是一个不完全形成的寄生双胞胎附着在一个更发达的自体上。我们提出了我们地区的第一个病例系列，说明了文献中很少报道的不寻常的解剖变异和复杂的内脏关联。在过去的二十年中，有4例新生儿异位双胞胎接受了手术治疗。第一位患者为足月新生儿，发育良好的第三条下肢植入右髂区，上肢发育不全，附于耻骨上方。两个额外的肢体被成功切除，并顺利恢复。第二例患者表现为双生殖器、双膀胱、结肠复制、动脉导管未闭和室间隔缺损。完全的手术矫正导致了良好的长期结果。第三例患者有臀寄生肢，并伴有骶尾骨畸胎瘤、肾发育不良和生殖器官重复。他在出生第11天死于心肺衰竭，还没来得及切除手术。第4例患者为上腹部寄生双胞胎，伴大脐膨出，含未发育的肠。1个月大时完成分离，但患者在术后第一天死亡。结论异卵（寄生）双胞胎可存在多种表型变异。内脏畸形，尤其是心脏畸形在预后中起着关键作用。

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引用次数: 0