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Giant mediastinal lymphangioma in a 4-year-old girl: a case report 4岁女童巨大纵隔淋巴管瘤1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-17 DOI: 10.1016/j.epsc.2025.103164
Mary R. Foster , Hyunyoung G. Kim , John J. Doski

Background

Mediastinal lymphangiomas are rare benign congenital malformations of the lymphatic system in children, often presenting with non-specific respiratory symptoms and posing diagnostic challenges.

Case presentation

A previously healthy 4-year-old female presented with progressive chest pain and intermittent dyspnea. Imaging revealed a large multiloculated cystic lesion (9.6 × 7.3 × 10.8 cm) occupying much of the right hemithorax, causing leftward mediastinal shift and compression of adjacent structures. Thoracentesis and right pigtail thoracostomy yielded partial drainage but incomplete resolution. Workup for Echinococcus, Histoplasma, Coccidioides, and tuberculosis was negative. Cyst fluid and blood cultures showed no growth, while cytology of the cyst fluid demonstrated inflammatory cells without evidence of malignancy. She was treated empirically with intravenous antibiotics for presumed obstructive pneumonia. On hospital day 8, she underwent right thoracotomy with complete excision of the multiloculated cystic mass, which was adherent to but separate from the pericardium and lung. There was no lymphatic leak, and the intraoperative chest tube was removed on postoperative day 2. Histopathology confirmed a benign cystic lymphangioma. The patient was discharged on hospital day 11 and remained asymptomatic and recurrence-free at 1-year follow-up.

Conclusion

Mediastinal lymphangioma should be considered in the differential diagnosis of pediatric patients presenting with progressive respiratory symptoms or cystic mediastinal lesions on imaging.
背景纵隔淋巴管瘤是儿童淋巴系统罕见的先天性良性畸形,通常表现为非特异性呼吸系统症状,给诊断带来挑战。病例表现:既往健康的4岁女性以进行性胸痛和间歇性呼吸困难表现。影像学显示大的多室囊性病变(9.6 × 7.3 × 10.8 cm),占据大部分右半胸,导致纵隔向左移位,压迫邻近结构。胸腔穿刺和右辫子开胸术产生部分引流,但不完全解决。棘球绦虫、组织浆体、球虫和肺结核检查均为阴性。囊肿液和血培养未见生长,而囊肿液的细胞学检查显示炎症细胞,无恶性肿瘤证据。她因疑似阻塞性肺炎接受了经验性静脉注射抗生素治疗。住院第8天,她接受了右开胸手术,完全切除了附着但与心包和肺分离的多室囊性肿块。无淋巴漏,术中胸管于术后第2天拔除。组织病理学证实为良性囊性淋巴管瘤。患者于住院第11天出院,随访1年无症状复发。结论小儿纵膈淋巴管瘤在影像学上表现为进行性呼吸道症状或囊性纵膈病变时应考虑鉴别诊断。
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引用次数: 0
Kidney auto-transplantation in a 7-year-old with complex renal artery anatomy and multi-drug-resistant hypertension: a case report 肾脏自体移植治疗7岁复杂肾动脉解剖和多重耐药高血压1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-16 DOI: 10.1016/j.epsc.2025.103162
Jayne Rice , Lily Huang , Peter Abt , Alexander Fairman

Introduction

There are numerous surgical techniques used for revascularization in the setting of renal artery stenosis in children, although formal guidelines do not exist and “standard of care” is dictated by center experience.

Case presentation

We present a case of a 7-year-old male with multi-drug-resistant hypertension in the setting of complex renal artery anatomy. His genetic testing was unrevealing for congenital aortopathies, and his clinical history was not suggestive of vasculitis or other inflammatory conditions. Computerized tomography angiogram (CTA) demonstrated two main renal arteries on the right, both with high grade, proximal stenosis, and on the left his main renal artery was occluded, with robust collateral filling. Nuclear function testing of the left kidney demonstrated <10 % function. The patient initially underwent an aorto-renal bypass using hypogastric artery; however, the bypass became occluded on post operative day four. To salvage the kidney, the patient underwent an auto-transplantation into the pelvis using a syndactylization technique to salvage both right renal arteries. He recovered well and is slowly weaning his anti-hypertensives at his last visit, 9 months post-operative.

Conclusion

Auto-transplantation can be a useful method to surgically manage complex renal artery stenosis, and salvage failed prior repairs.
在儿童肾动脉狭窄的情况下,有许多手术技术用于血运重建术,尽管没有正式的指南,“护理标准”由中心经验决定。我们报告一例7岁男性多重耐药高血压在复杂的肾动脉解剖设置。他的基因检测未发现先天性主动脉病变,他的临床病史也未提示血管炎或其他炎症。ct血管造影(CTA)显示右侧两条主要肾动脉近端高度狭窄,左侧主要肾动脉闭塞,侧枝充盈。左肾核功能检查显示功能<; 10%。患者最初采用腹下动脉行主动脉-肾旁路术;然而,旁路在术后第四天闭塞。为了挽救肾脏,患者采用并指化技术进行骨盆自体移植以挽救双右肾动脉。术后9个月,他恢复得很好,并在最后一次就诊时逐渐停用抗高血压药物。结论自体肾动脉移植是复杂肾动脉狭窄手术治疗的有效方法,可挽救先前修复失败的肾动脉。
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引用次数: 0
Primary rhabdomyosarcoma of the breast in a teenager: a case report 青少年原发性乳腺横纹肌肉瘤1例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-08 DOI: 10.1016/j.epsc.2025.103158
Wondwosen Mengist Dereje , Emebet Hunie Baze , Asya Mohammed Yesuf , Atsede Abebe , Yohannes Leweyehu Debasu , Eyoel Negash Taddesse

Introduction

Although rhabdomyosarcoma (RMS) is the most common soft-tissue neoplasm in children, primary breast rhabdomyosarcoma is rare, as breast involvement typically occurs through secondary metastasis rather than as a primary tumor.

Case presentation

A 14-year-old girl presented with a progressively enlarging swelling in her left breast over eight weeks. It began as a small, peanut-sized lump that gradually increased in size, followed six weeks later by a left axillary swelling. She sought medical care eight weeks after the breast swelling began. At presentation, she reported fatigue, weight loss, loss of appetite, and night sweats.
On examination, she was malnourished (BMI 18.1 kg/m2). A firm, non-tender left breast mass measuring 5 × 4 cm was palpated, fixed to underlying tissue but not adherent to the skin. A hard, non-tender left axillary lymph node measuring 2 × 1.5 cm was also noted, while the right breast and axilla were normal.
Ultrasound and fine-needle aspiration cytology (FNAC) revealed an undifferentiated high-grade malignant tumor with axillary metastasis. Given lymphatic spread, the disease was considered advanced, and chemotherapy with Adriamycin and cyclophosphamide was initiated.
Because other malignancies such as high-grade non-Hodgkin lymphoma and dysgerminoma were possible, an incisional biopsy was performed before mastectomy. Histopathology showed small round blue cells, and immunohistochemistry confirmed rhabdomyosarcoma.
After diagnosis, the prognosis and treatment options, including radical mastectomy with lymph node dissection, were discussed with the patient and her family. However, they declined surgery and continued chemotherapy alone.
One month later, she returned for her second chemotherapy cycle but had developed back pain and severe debilitation. She did not return afterward and passed away two months later.

Conclusion

Breast rhabdomyosarcoma should be considered in the differential diagnosis of unexplained or rapidly enlarging breast swelling, particularly in young patients.
虽然横纹肌肉瘤(rhabdomyosarcoma, RMS)是儿童中最常见的软组织肿瘤,但原发性乳腺横纹肌肉瘤是罕见的,因为它通常通过继发性转移而不是原发肿瘤累及乳腺。病例介绍一名14岁的女孩,她的左乳房在8周内逐渐扩大肿胀。它开始是一个花生大小的小肿块,逐渐增大,六周后出现左腋窝肿胀。她在乳房开始肿胀的八周后就医。在就诊时,她报告疲劳、体重减轻、食欲不振和盗汗。经检查,她营养不良(BMI 18.1 kg/m2)。触诊左侧乳房5 × 4 cm的硬而无压痛肿块,固定于下方组织,但不粘附于皮肤。左侧腋窝淋巴结硬而无压痛,大小为2 × 1.5 cm,而右侧乳房和腋窝正常。超声及细针穿刺细胞学检查显示为未分化的高级别恶性肿瘤伴腋窝转移。由于淋巴扩散,疾病被认为是晚期,并开始用阿霉素和环磷酰胺化疗。由于可能存在其他恶性肿瘤,如高级别非霍奇金淋巴瘤和异常生殖细胞瘤,因此在乳房切除术前进行了切口活检。组织病理学显示小而圆的蓝色细胞,免疫组化证实为横纹肌肉瘤。诊断后,我们与患者及其家人讨论了预后和治疗方案,包括根治性乳房切除术和淋巴结清扫。然而,他们拒绝手术并继续单独化疗。一个月后,她返回进行了第二次化疗,但出现了背部疼痛和严重的虚弱。之后她再也没有回来,两个月后就去世了。结论乳腺横纹肌肉瘤在鉴别诊断中应考虑原因不明或迅速增大的乳房肿胀,尤其是年轻患者。
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引用次数: 0
Total colonic duplication with vestibular fistula, including one with colorectal triplication: A case series 全结肠复制合并前庭瘘,包括1例结直肠复制:一个病例系列
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-02 DOI: 10.1016/j.epsc.2025.103157
Michaël de Sousa Amaral , Mathilde Glenisson , Sylvie Beaudoin

Introduction

Complete colonic duplication associated with rectovestibular communication is extremely uncommon, whereas colorectal triplication has been documented only in isolated case reports.

Cases presentation

Case 1 involved a 34-day-old term baby girl, who presented with passage of stool through both a normal anus and an accessory vestibular orifice. Dual-tract contrast studies and hydrocolonic MRI demonstrated complete colonic duplication converging at a single cecum into which the terminal ileum inserted. At a postnatal age of 3 months, a combined Pfannenstiel and perineal approach allowed resection of the duplicated rectum and creation of a termino-lateral sigmoid–sigmoid unification. Postoperative recovery was uneventful, and age-appropriate continence was confirmed during follow-up. Case 2 concerned a female neonate delivered at 41 weeks’ gestation who began passing meconium on day two of life through both the anus and a vestibular orifice. Contrast opacification demonstrated complete colonic duplication, with the anterior duplicated segment coursing toward the vagina. At five months, combined perineal dissection and Pfannenstiel exposure revealed total colonic duplication extending to the terminal ileum, associated with duplication of both ceca and appendices. The duplicated rectum was mobilized, inverted, and excised. Intra-operatively, a rectal triplication embedded within the shared rectal wall was also identified and resected. All three rectosigmoid segments were subsequently anastomosed to the orthotopic colon using stapled unification, and the perineal body was reconstructed. Postoperative recovery was favorable with normal bowel function.

Conclusion

In females with a patent anus who pass stool through an accessory vestibular orifice, underlying colonic duplication should be systematically ruled out.
完全结肠复制与直肠前庭通信是极其罕见的,而结直肠复制仅在个别病例报告中有记录。病例1为一34天足月女婴,其表现为大便通过正常肛门和副前庭口。双束对比研究和结肠积水MRI显示完全结肠复制会聚于回肠末端插入的单个盲肠。在出生后3个月时,采用Pfannenstiel和会阴联合入路切除重复直肠,并建立乙状窦-乙状窦终外侧联合。术后恢复顺利,随访中确认适龄尿失禁。病例2涉及一名妊娠41周分娩的女婴,出生第2天开始通过肛门和前庭孔排出胎便。造影剂混浊显示完全结肠复制,前复制段向阴道方向移动。5个月时,会阴解剖和Pfannenstiel暴露显示全结肠复制延伸到回肠末端,盲肠和阑尾都有复制。重复直肠被移动、倒置并切除。术中,我们也发现并切除了一个嵌在共同直肠壁内的直肠三瘤。所有三个直肠乙状结肠段随后用钉状统一与原位结肠吻合,并重建会阴体。术后恢复良好,肠功能正常。结论肛门未闭经副前庭口排便的女性,应系统排除潜在的结肠复制。
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引用次数: 0
Innovative imaging techniques to guide the prenatal and postnatal management of premature omphalopagus conjoined twins: a case report 创新影像技术指导早产儿脐裂连体婴产前产后处理1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-11-26 DOI: 10.1016/j.epsc.2025.103155
Amanda Phares , Ghadeer Alkusayer , Frances Morin , Jessica Liauw , Horacio Osiovich , Robert Baird

Background

The incidence of conjoined twinning is approximately 1:100,000 with omphalopagus anatomy representing 10 % of all cases. With an overall survival rate of <10 % and a 60 % rate of survival in patients who undergo separation, considerable medical, surgical, and ethical planning is required.

Case presentation

A 34-year-old mother was identified as having conjoined twins at 9 weeks gestation. She underwent an early fetal Magnetic Resonance Imaging (MRI) to assess potential for viability and a late second trimester MRI – these identified omphalopagus anatomy with shared liver and a Tetralogy sequence in twin B. 3D modeling of these images guided delivery and separation planning. The twins were born prematurely at 29 weeks and 6 days. Neonatal Inteive Care Unit (NICU) management was instituted at birth and the initial cardiorespiratory status of the twins was stable with minimal circulatory mixing noted. Combined fetal weight was 2.6 kg. At 3 weeks of life, significant circulatory deterioration occurred in twin B. Urgent separation to facilitate treatment for twin B best optimized both patients’ survival and was in keeping with parental wishes. Detailed cross-sectional imaging was expedited and used to facilitate successful separation. Pre-op anesthesia time was 127 minutes and Operating Room time was 290 minutes; estimated blood loss was 150 mL. Twin B subsequently underwent emergent catheterization with pulmonary artery dilation and right ventricle outflow tract stenting. Both twins recovered successfully with twin A being discharged from hospital at 14 weeks old and twin B discharged at 15 weeks old.

Conclusion

Cross-sectional imaging with 3D modeling helps optimize outcomes for conjoined twins.
背景:连体双胞胎的发生率约为1:10万,脐裂解剖占所有病例的10%。总体存活率为10%,分离患者的存活率为60%,因此需要进行大量的医疗、手术和伦理规划。病例介绍一位34岁的母亲在妊娠9周时被确诊为连体双胞胎。她接受了早期的胎儿磁共振成像(MRI)来评估生存能力的潜力,并在妊娠中期晚期进行了MRI检查,这些检查确定了胎儿脐裂解剖与共享肝脏和双胞胎b的四联症序列,这些图像的3D建模指导了分娩和分离计划。这对双胞胎在29周零6天时早产。新生儿重症监护病房(NICU)管理在出生时建立,双胞胎的初始心肺状态稳定,循环混合最小。胎重合计2.6 kg。在出生3周时,双胞胎B出现了明显的循环恶化。紧急分离以促进双胞胎B的治疗,最好地优化了两名患者的生存,并符合父母的愿望。详细的横断面成像加快并用于促进成功的分离。术前麻醉时间127分钟,手术室时间290分钟;估计失血量为150毫升。双胞胎B随后接受了紧急导管置入术,肺动脉扩张和右心室流出道支架置入术。两个双胞胎都成功康复,双胞胎A在14周大时出院,双胞胎B在15周大时出院。结论三维建模的横断成像有助于优化连体双胞胎的预后。
{"title":"Innovative imaging techniques to guide the prenatal and postnatal management of premature omphalopagus conjoined twins: a case report","authors":"Amanda Phares ,&nbsp;Ghadeer Alkusayer ,&nbsp;Frances Morin ,&nbsp;Jessica Liauw ,&nbsp;Horacio Osiovich ,&nbsp;Robert Baird","doi":"10.1016/j.epsc.2025.103155","DOIUrl":"10.1016/j.epsc.2025.103155","url":null,"abstract":"<div><h3>Background</h3><div>The incidence of conjoined twinning is approximately 1:100,000 with omphalopagus anatomy representing 10 % of all cases. With an overall survival rate of &lt;10 % and a 60 % rate of survival in patients who undergo separation, considerable medical, surgical, and ethical planning is required.</div></div><div><h3>Case presentation</h3><div>A 34-year-old mother was identified as having conjoined twins at 9 weeks gestation. She underwent an early fetal <strong>Magnetic Resonance Imaging (MRI)</strong> to assess potential for viability and a late second trimester MRI – these identified omphalopagus anatomy with shared liver and a Tetralogy sequence in twin B. 3D modeling of these images guided delivery and separation planning. The twins were born prematurely at 29 weeks and 6 days. <strong>Neonatal Inteive Care Unit (NICU)</strong> management was instituted at birth and the initial cardiorespiratory status of the twins was stable with minimal circulatory mixing noted. Combined fetal weight was 2.6 kg. At 3 weeks of life, significant circulatory deterioration occurred in twin B. Urgent separation to facilitate treatment for twin B best optimized both patients’ survival and was in keeping with parental wishes. Detailed cross-sectional imaging was expedited and used to facilitate successful separation. Pre-op anesthesia time was 127 minutes and <strong>Operating Room</strong> time was 290 minutes; estimated blood loss was 150 mL. Twin B subsequently underwent emergent catheterization with pulmonary artery dilation and right ventricle outflow tract stenting. <strong>Both twins recovered successfully with twin A being discharged from hospital at 14 weeks old and twin B discharged at 15 weeks old.</strong></div></div><div><h3>Conclusion</h3><div>Cross-sectional imaging with 3D modeling <strong>helps</strong> optimize outcomes for conjoined twins.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"124 ","pages":"Article 103155"},"PeriodicalIF":0.2,"publicationDate":"2025-11-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145617172","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Small bowel obstruction due to Meckel's diverticulitis in a 5-year-old child: a case report 5岁儿童梅克尔憩室炎致小肠梗阻1例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-11-24 DOI: 10.1016/j.epsc.2025.103156
Marion Poget, Sabine Vasseur Maurer

Introduction

Meckel's diverticulum is the most common congenital anomaly of the gastrointestinal tract, yet small bowel obstruction caused by its inflammation remains exceptionally rare, especially in children.

Case presentation

A previously healthy 5-year-old girl presented at a regional hospital with fever and abdominal pain for five days, followed by absence of stool and gas passage for 24 hours. Laboratory tests showed leukocytosis (22,000 cells/mm3) and elevated C-reactive protein (200 mg/L). Abdominal ultrasound revealed signs of small bowel obstruction, free intraperitoneal fluid but no visible appendix. Computed tomography demonstrated small bowel obstruction with possible decreased enhancement in a short segment of small bowel located in the subhepatic region, which prompted a transfer of the patient to our center. The patient, who showed stable vital signs and no evidence of peritonitis, underwent an MRI to further clarify the diagnosis. This exam suggested an internal hernia. Exploratory laparoscopy converted to laparotomy revealed a necrotic Meckel's diverticulum, whose thin pedicle was strangulating the small bowel. A wedge resection of the diverticulum was performed. The postoperative course was uneventful, and the patient was discharged on day four. At two-week follow-up, she was asymptomatic.

Conclusion

Meckel's diverticulitis must be included in the differential diagnosis of children who develop a small bowel obstruction.
梅克尔憩室是最常见的胃肠道先天性异常,但由其炎症引起的小肠梗阻仍然非常罕见,特别是在儿童中。病例介绍:一名健康的5岁女孩在地区医院就诊,发热和腹痛5天,随后24小时无大便和气体通过。实验室检查显示白细胞增多(22 000个细胞/毫米3)和c反应蛋白升高(200毫克/升)。腹部超声显示小肠梗阻,腹腔内积液,但未见阑尾。计算机断层扫描显示位于肝下区域的一小段小肠梗阻,可能增强减弱,这促使患者转移到我们的中心。该患者生命体征稳定,无腹膜炎迹象,接受了核磁共振检查以进一步明确诊断。这次检查显示是腹内疝。腹腔镜探查转为开腹检查,发现坏死的梅克尔憩室,其细蒂勒死小肠。行楔形憩室切除术。术后过程顺利,患者于第四天出院。在两周的随访中,她无症状。结论儿童发生小肠梗阻时,应将梅克尔憩室炎列入鉴别诊断。
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引用次数: 0
Corrigendum to “Raising awareness of the CDH1 mutation for the pediatric surgeon and the use of robotic assisted surgery in pediatric general surgery as demonstrated by a total gastrectomy: a case report” [J. Pediatr. Surg. Case Rep. 123C (2025) 103141] “提高儿科外科医生对CDH1突变的认识,并以全胃切除术为例,在儿科普通外科中使用机器人辅助手术:一个病例报告”的更正[J]。Pediatr。外科病例报告123C (2025) 103141]
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-11-10 DOI: 10.1016/j.epsc.2025.103151
Melanie Elhafid , Kristopher Milbrandt , Richard Bigsby
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引用次数: 0
Intestinal malrotation with midgut volvulus and duodenal stenosis in a neonate: a case report 新生儿肠道旋转不良伴中肠扭转和十二指肠狭窄1例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-11-07 DOI: 10.1016/j.epsc.2025.103142
Samuel Kefiyalew Kelbessa , Mihret S. Tesfaye , Hiwot Y. Anley

Introduction

Intestinal malrotation and congenital duodenal stenosis are uncommon causes of intestinal obstruction in newborns. Their simultaneous occurrence is extremely rare and poses a diagnostic and therapeutic challenge.

Case presentation

A 17-day-old full-term male neonate presented with persistent bilious vomiting, feeding intolerance, and poor weight gain since birth. He exhibited jaundice, edema, and abdominal distension, along with direct hyperbilirubinemia. An abdominal ultrasound revealed a whirlpool sign and abnormal positioning of the superior mesenteric artery and vein, indicating intestinal malrotation complicated by midgut volvulus. An exploratory laparotomy was performed, which revealed incomplete rotation and a mobile cecum, with Ladd's bands obstructing the duodenum and a 360° clockwise volvulus. After detorsing the volvulus and performing a Ladd's procedure, the baby showed initial improvement. However, ongoing bilious output from the nasogastric tube raised concerns about proximal obstruction. An abdominal X-ray done 72 hours later confirmed duodenal stenosis. A Heineke-Mikulicz duodenoplasty was subsequently performed, along with the insertion of a trans-anastomotic feeding tube and a decompression nasogastric tube. The neonate gradually advanced to full feeds, with the nasogastric tube being removed on postoperative day seven and the feeding tube on day ten. The neonate was discharged on the fourteenth postoperative day. Follow-up appointments two weeks, three months and six months post discharge indicated that the baby is thriving well.

Conclusion

Duodenal stenosis should be suspected in neonates who have persistent bilious nasogastric output after a Ladd's procedure.
肠道旋转不良和先天性十二指肠狭窄是新生儿肠梗阻的罕见原因。它们同时发生是极其罕见的,并提出了诊断和治疗的挑战。一例17日龄足月男新生儿自出生以来出现持续性胆汁性呕吐、喂养不耐受和体重增加不佳。他表现出黄疸、水肿和腹胀,并伴有直接的高胆红素血症。腹部超声示漩涡征及肠系膜上动静脉定位异常,提示肠旋转不良合并中肠扭转。行剖腹探查,发现盲肠旋转不全,盲肠移动,Ladd氏带阻塞十二指肠,呈360°顺时针扭转。在扭转扭转并进行拉德手术后,婴儿表现出初步的改善。然而,从鼻胃管流出的胆汁引起了对近端梗阻的关注。72小时后腹部x光检查证实十二指肠狭窄。随后行Heineke-Mikulicz十二指肠成形术,同时插入经吻合口喂养管和减压鼻胃管。新生儿逐渐进入饱食期,术后第7天拔除鼻胃管,第10天拔除喂食管。患儿于术后第14天出院。出院后两周、三个月和六个月的随访表明,婴儿发育良好。结论在Ladd术后出现持续胆汁性鼻胃排出物的新生儿应怀疑十二指肠狭窄。
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引用次数: 0
Large aneurysmal dermatofibroma mimicking melanoma in an adolescent: A case report 青少年大动脉瘤性皮肤纤维瘤模拟黑色素瘤:1例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-11-06 DOI: 10.1016/j.epsc.2025.103145
Emma Lim , Andreana Bütter

Introduction

Hemosiderotic dermatofibroma (HDF) is an infrequent variant of dermatofibroma, made up of small blood vessels, hemosiderin deposits, and extravasated erythrocytes. HDF closely resembles melanoma, with irregular borders and colour changes on clinical examination and atypical cell organization on dermoscopy and can pose a diagnostic dilemma.

Case presentation

We report the case of a large (5cm) subcutaneous hemosiderotic/aneurysmal DF (H/ADF) that appeared on the flank of 17-year-old, otherwise healthy, male patient. The mass evolved over two years, with purple discoloration noticed three months prior. Clinical examination revealed a palpable moveable mass overlying the left hip in the subcutaneous tissues measuring approximately 4 x 4 cm. Because of both the substantial size of the mass and diagnostic uncertainty, the decision was made to proceed with surgical resection. The lesion was resected without complication, although there was copious bleeding initially. Pathological findings showed a dermatofibroma, cellular subtype, with aneurysmal change. Sections showed a solid, cellular proliferation composed of plump ovoid to spindle cells arranged in a storiform pattern and short fascicles punctuated by areas of hemorrhage and associated hemosiderin pigment deposition. Occasional multinucleated giant cells were seen. At 1 month follow-up, the patient was in good condition, and completely asymptomatic. There was no recurrence.

Conclusion

Pediatric surgeons should be aware that rare dermatofibroma variants, such as aneurysmal dermatofibroma, can clinically resemble melanoma and should be treated with excisional biopsy. Diagnosis should always be confirmed with histopathology and immunohistochemistry.
含铁血黄素性皮肤纤维瘤(HDF)是一种罕见的皮肤纤维瘤,由小血管、含铁血黄素沉积和外渗的红细胞组成。HDF与黑色素瘤非常相似,临床检查时边界不规则,颜色变化,皮肤镜检查时细胞组织不典型,可能导致诊断困境。我们报告一个大的(5cm)皮下含铁血黄素/动脉瘤性DF (H/ADF),出现在17岁,其他健康,男性患者的侧面。这个肿块在两年多的时间里形成,三个月前就出现了紫色。临床检查显示在左髋关节皮下组织上有一个可触及的可移动肿块,大小约为4 x 4厘米。由于肿块的大小和诊断的不确定性,我们决定进行手术切除。病变切除无并发症,虽然有大量出血最初。病理表现为皮肤纤维瘤,细胞亚型,伴动脉瘤样改变。切片显示实性细胞增生,由丰满的卵形细胞到呈故事状排列的梭形细胞组成,短束状细胞被出血区和相关的含铁血黄素色素沉积所打断。偶见多核巨细胞。随访1个月,患者状态良好,完全无症状。无复发。结论小儿外科医生应注意罕见的皮肤纤维瘤变体,如动脉瘤性皮肤纤维瘤,在临床上与黑色素瘤相似,应采用切除活检治疗。诊断必须通过组织病理学和免疫组织化学来证实。
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引用次数: 0
Appendiceal duplication leading to recurrent ileocolonic intussusception: A case report 阑尾重复导致回肠肠套叠1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-11-06 DOI: 10.1016/j.epsc.2025.103146
Erica C. Arnold , Miriam Conces , Raul E. Sanchez , Sara Mansfield , Jennifer H. Aldrink

Introduction

Appendiceal duplication is a rare congenital anomaly of the appendix, often identified incidentally, and is estimated to be present in 0.004–0.009% of the population. Rarely, an appendiceal duplication may act as a lead point for recurrent ileocolonic intussusception.

Case presentation

This case describes a 15-year-old male with celiac disease who initially presented with ileocolonic intussusception for which he underwent exploratory laparotomy for intussusception reduction at a local institution. Intra-operatively, there was concern for an underlying colonic polyp. He subsequently underwent colonoscopy and CT enterography, which both revealed no evidence of a defined pathologic lead point. He then presented two months later to our institution with recurrent ileocolonic intussusception, which was reduced via air contrast enema. He was monitored overnight and discharged the following day. Three days after this, he again developed abdominal pain and was identified to have recurrent ileocolonic intussusception, which was successfully reduced via air contrast enema. Repeat CT enterography identified no pathologic lead point. He was monitored overnight and discharged with plans for outpatient ileocecectomy. One day later, however, his pain recurred, and he was again diagnosed with ileocolonic intussusception via abdominal ultrasound. This was successfully reduced via air contrast enema, and he was taken to the operating room for planned laparoscopic ileocecectomy to prevent further recurrence. Intra-operatively, there was no evidence of a clear pathologic lead point, but there were significant adhesive colonic peritoneal bands noted, as well as an enlarged but not inflamed appendix. On final pathology, he was found to have appendiceal duplication with otherwise benign ileocecal pathology. He recovered uneventfully in the hospital and was discharged on post-operative day two. He has had no further episodes of recurrence for four months post-operatively.

Conclusion

Appendiceal duplication is a rare congenital anomaly that should be considered as a potential etiology for otherwise unexplained recurrent ileocolonic intussusception.
阑尾重复是一种罕见的先天性阑尾畸形,通常是偶然发现的,估计在0.004-0.009%的人群中存在。极少情况下,阑尾重复可作为回肠肠套叠复发的先导点。病例介绍:本病例描述了一名15岁的乳糜泻男性患者,他最初表现为回肠肠套叠,并在当地机构接受了剖腹探查术以减少肠套叠。术中,有潜在结肠息肉的担忧。随后进行结肠镜检查和CT肠造影检查,均未发现明确的病理导点。两个月后,他以复发性回结肠肠套叠就诊,经空气造影灌肠治疗。他接受了彻夜监护,并于第二天出院。3天后,患者再次出现腹痛,确认为复发性回肠肠套叠,经空气造影灌肠成功消除。重复CT肠造影未发现病理导联点。他接受了一整晚的监护,出院时计划进行门诊回肠切除术。然而,一天后,他的疼痛复发,他再次通过腹部超声诊断为回肠结肠肠套叠。通过空气造影剂灌肠成功地减少了这种情况,并将他带到手术室进行计划的腹腔镜回盲切除术,以防止进一步复发。术中,没有明确的病理线索,但有明显粘连的结肠腹膜带,以及阑尾肿大但未发炎。在最后的病理检查中,他被发现有阑尾重复和其他良性回盲病理。他在医院恢复得很顺利,并于术后第二天出院。术后4个月无复发。结论阑尾重复是一种罕见的先天性异常,可作为复发性回肠肠套叠的潜在病因。
{"title":"Appendiceal duplication leading to recurrent ileocolonic intussusception: A case report","authors":"Erica C. Arnold ,&nbsp;Miriam Conces ,&nbsp;Raul E. Sanchez ,&nbsp;Sara Mansfield ,&nbsp;Jennifer H. Aldrink","doi":"10.1016/j.epsc.2025.103146","DOIUrl":"10.1016/j.epsc.2025.103146","url":null,"abstract":"<div><h3>Introduction</h3><div>Appendiceal duplication is a rare congenital anomaly of the appendix, often identified incidentally, and is estimated to be present in 0.004–0.009% of the population. Rarely, an appendiceal duplication may act as a lead point for recurrent ileocolonic intussusception.</div></div><div><h3>Case presentation</h3><div>This case describes a 15-year-old male with celiac disease who initially presented with ileocolonic intussusception for which he underwent exploratory laparotomy for intussusception reduction at a local institution. Intra-operatively, there was concern for an underlying colonic polyp. He subsequently underwent colonoscopy and CT enterography, which both revealed no evidence of a defined pathologic lead point. He then presented two months later to our institution with recurrent ileocolonic intussusception, which was reduced via air contrast enema. He was monitored overnight and discharged the following day. Three days after this, he again developed abdominal pain and was identified to have recurrent ileocolonic intussusception, which was successfully reduced via air contrast enema. Repeat CT enterography identified no pathologic lead point. He was monitored overnight and discharged with plans for outpatient ileocecectomy. One day later, however, his pain recurred, and he was again diagnosed with ileocolonic intussusception via abdominal ultrasound. This was successfully reduced via air contrast enema, and he was taken to the operating room for planned laparoscopic ileocecectomy to prevent further recurrence. Intra-operatively, there was no evidence of a clear pathologic lead point, but there were significant adhesive colonic peritoneal bands noted, as well as an enlarged but not inflamed appendix. On final pathology, he was found to have appendiceal duplication with otherwise benign ileocecal pathology. He recovered uneventfully in the hospital and was discharged on post-operative day two. He has had no further episodes of recurrence for four months post-operatively.</div></div><div><h3>Conclusion</h3><div>Appendiceal duplication is a rare congenital anomaly that should be considered as a potential etiology for otherwise unexplained recurrent ileocolonic intussusception.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"123 ","pages":"Article 103146"},"PeriodicalIF":0.2,"publicationDate":"2025-11-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145526286","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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Journal of Pediatric Surgery Case Reports
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