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Necrotizing enterocolitis and optimal surgical timing: Case series 坏死性小肠结肠炎和最佳手术时机:病例系列
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-16 DOI: 10.1016/j.epsc.2024.102906
Mariel Magdits , Gabriella Grisotti , Lan Vu

Introduction

The pathophysiology and management of necrotizing enterocolitis (NEC) with identification of the subset of NEC totalis and the timing of surgical intervention, remains an area of active research and clinical uncertainty. A third of deaths from NEC are attributed to NEC totalis. This case series presents three distinct cases of total or near-total NEC.

Case series

Case 1: A female preterm infant born at 27 weeks of gestation developed sepsis at six days of life, diagnosed with NEC evidenced by portal venous gas and diffuse pneumatosis intestinalis. Surgical exploration 8 weeks later revealed extensive involvement of the entire small bowel, culminating in a fatal outcome. Case 2: A male preterm infant delivered at 24 weeks and 4 days gestation presented with respiratory distress syndrome shortly after birth and developed abdominal distension and ongoing metabolic acidosis and thrombocytopenia. On day 12 post-NEC diagnosis, surgical exploration revealed extensive bowel necrosis involving most of the small bowel and right colon. The patient did not survive the operation due to intraoperative hemorrhage secondary to the degree of acute inflammation. Case 3: A preterm female infant delivered at 25 weeks and 5 days gestation exhibited respiratory failure and a tense abdomen within the first week of life. Upon transfer to a higher level of care, imaging revealed massive pneumoperitoneum and exploration demonstrated extensive bowel necrosis from the mid jejunum to the distal transverse colon. Prompt surgical intervention within 24 hours post-diagnosis resulted in bowel resection and jejunostomy creation, leading to improved clinical condition and ongoing survival.

Conclusion

The presented case series suggests that timing in surgical intervention for NEC may play a role in outcomes.
导言坏死性小肠结肠炎(NEC)的病理生理学和管理,以及全坏死性小肠结肠炎子集的鉴定和手术干预的时机,仍然是一个活跃的研究领域和临床不确定因素。三分之一的 NEC 死亡病例归因于全坏死性小肠结肠炎。本病例系列介绍了三例不同的全NEC或近全NEC病例。病例系列病例 1:一名妊娠 27 周出生的早产女婴在出生六天时出现败血症,诊断为 NEC,表现为门静脉积气和弥漫性肠道积气。8 周后进行手术探查,发现整个小肠广泛受累,最终导致死亡。病例 2:一名妊娠 24 周零 4 天的早产男婴,出生后不久即出现呼吸窘迫综合征,并出现腹胀、持续代谢性酸中毒和血小板减少。NEC 诊断后第 12 天,手术探查发现大面积肠坏死,涉及大部分小肠和右侧结肠。由于急性炎症的程度导致术中大出血,患者未能在手术中存活下来。病例 3:一名妊娠 25 周零 5 天的早产女婴在出生后一周内出现呼吸衰竭和腹部紧张。转到更高级别的医院后,影像学检查发现大量腹腔积气,探查显示从空肠中段到横结肠远端有大面积肠坏死。确诊后 24 小时内及时进行手术治疗,切除了肠道并建立了空肠造口,从而改善了临床状况并延长了存活时间。
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引用次数: 0
Congenital Treves’ field transmesenteric hernia in 7-month-old infant: A case report 7 个月大婴儿的先天性特雷韦斯野外跨肠管疝:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-15 DOI: 10.1016/j.epsc.2024.102905
Abdelrahman S. Elnour , Omer Abuagla , Faisal Nugud , Osman Taha

Introduction

Congenital Treves’ field hernia is the herniation of a visceral segment through a defect in the terminal ileal mesentery, poses a high risk of strangulation and bowel gangrene. Its preoperative diagnosis is challenging due to its rarity, nonspecific presentation, and often inconclusive imaging.

Case presentation

A previously healthy 7-month-old female infant presented with an 18-h history of intense diffuse abdominal pain, nausea, and bilious vomiting. On examination, she appeared ill and dehydrated, with tachycardia and tachypnea. Her abdomen was grossly distended, tense, and tender, with no visible scars or hernias. Blood tests, including a complete blood count, urea, and electrolytes, were normal except for an elevated white blood cell count. Abdominal X-rays and ultrasound revealed features consistent with distal small bowel obstruction, with no evidence of intussusception. Following resuscitation, an emergency laparotomy was performed for suspected acute abdomen due to distal small bowel obstruction. Intraoperatively, the jejunum and proximal ileum were dilated, and a portion of the ilium was found herniated through a 6 × 4 cm mesenteric defect. After reduction, approximately 25 cm of necrotic small bowel was resected, followed by an end-to-end anastomosis. The postoperative recovery was smooth, and she was discharged on day seven with no signs of short bowel syndrome on follow-up.

Conclusion

Congenital Treves’ field transmesenteric hernia is a rare cause of intestinal obstruction in children that should be considered in cases of mechanical small bowel obstruction.
导言先天性特雷韦氏野疝是指内脏部分通过回肠系膜末端的缺损疝出,极易造成绞窄和肠坏疽。由于其罕见性、非特异性表现和通常不确定的影像学表现,其术前诊断极具挑战性。病例介绍 一名 7 个月大的健康女婴因强烈的弥漫性腹痛、恶心和胆汁性呕吐就诊 18 小时。经检查,她看起来不适且脱水,伴有心动过速和呼吸急促。她的腹部明显胀大、紧张和压痛,没有明显的疤痕或疝气。除了白细胞计数升高外,包括全血细胞计数、尿素和电解质在内的血液化验结果均正常。腹部 X 光片和超声波检查显示与远端小肠梗阻一致,但没有肠套叠的迹象。经过抢救,因怀疑是远端小肠梗阻导致的急腹症,医生为其实施了急诊开腹手术。术中,空肠和回肠近端扩张,发现部分髂骨通过一个 6 × 4 厘米的肠系膜缺损疝出。在缩小手术范围后,切除了约 25 厘米的坏死小肠,随后进行了端对端吻合术。结论先天性特雷韦斯氏野跨肠系膜疝是导致儿童肠梗阻的罕见原因,在机械性小肠梗阻的病例中应加以考虑。
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引用次数: 0
Chronically retained esophageal foreign body, a case report 慢性滞留食管异物,病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-12 DOI: 10.1016/j.epsc.2024.102904
Nardos Mulu Admasu, Fisseha Temesgen Gebru, Tihitena Negussie Mamo, Eden Belay Tilahun, Etsub Abebaw

Introduction

Foreign body ingestion is one of the most common pediatric emergencies among infants and young children. Between 80 % and 90 % of ingested foreign bodies pass spontaneously, 10 %–20 % require endoscopic removal, and about 1 % necessitates an open approach for removal.

Case presentation

A 13-year-old female developed progressive dysphagia over the course of 11 years. The parents reported a history of a foreign body ingestion (a plastic candlepin holder) 11 years before. They were informed that the foreign body would pass spontaneously but they did not recall ever having recovered it. Over the years she was repeatedly taken to various healthcare facilities where chest X-rays were performed and reported as normal, and she was sent home without interventions. When she presented to our clinic, she could swallow chewed solid food only with liquids. A neck examination revealed a bulge on the right anterolateral side. The remainder of the physical examination was unremarkable. An esophagogram revealed an upper esophageal stricture with a diverticulum. Upper GI endoscopy demonstrated a proximal esophageal stricture, while a neck CT scan showed narrowing of the proximal esophagus and a foreign body with a central hole. The impacted foreign body was successfully removed by endoscopy. She was able to swallow solid food for about a year, after which she developed dysphagia again. Endoscopy revealed narrowing at the proximal esophagus. An attempt at esophageal dilation was unsuccessful. She subsequently underwent a cervical exploration, which showed a short proximal esophageal stricture and a diverticulum proximal to the stricture. A diverticulectomy and resection with end-to-end anastomosis were done. Six months after the operation she continues to eat solid foods with some difficulty, but is gaining weight, and remains under close surveillance.

Conclusion

Diagnosing non-radiopaque esophageal foreign bodies is challenging in centers with limited resources. Chronically impacted foreign bodies and their complications are difficult to treat due to the lack of a standardized approach.
导言异物误食是婴幼儿最常见的儿科急症之一。80%-90%的异物可自行排出,10%-20%的异物需要内窥镜取出,约 1%的异物需要开刀取出。其父母称 11 年前曾误食异物(塑料烛台)。他们被告知异物会自然排出,但他们不记得曾经找到过异物。多年来,她曾多次被送往不同的医疗机构进行胸部 X 射线检查,结果均显示正常,于是她被送回了家,没有采取任何干预措施。当她来到我们诊所时,她只能用流质食物吞咽咀嚼过的固体食物。颈部检查发现右侧前外侧有一凸起。其余体格检查结果均无异常。食管造影显示食管上段狭窄,并伴有憩室。上消化道内窥镜检查显示食管近端狭窄,颈部 CT 扫描显示食管近端狭窄,异物中央有孔。内窥镜检查成功取出了异物。她能吞咽固体食物大约一年,之后再次出现吞咽困难。内镜检查发现食道近端狭窄。食管扩张尝试没有成功。随后,她接受了颈部探查,结果显示食管近端狭窄较短,狭窄近端有一个憩室。医生为她做了憩室切除术和端对端吻合术。结论在资源有限的中心,诊断不透射线的食管异物具有挑战性。由于缺乏标准化方法,慢性异物及其并发症很难治疗。
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引用次数: 0
Congenital rib absence with liver herniation: A case report 先天性肋骨缺失伴肝疝:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-10 DOI: 10.1016/j.epsc.2024.102903
Yirgalem Teklebirhan Gebreziher , Feven Mekonen Tadesse , Hadush Tesfay Negash , Berihu Tadish Gebre

Introduction

Congenital rib absence with liver herniation is an extremely rare condition. Only a few cases have been reported, and most of them have been associated with Poland syndrome.

Case presentation

A full-term female newborn, born at 38 weeks of gestation with a birth weight of 3100 g, delivered vaginally following a normal pregnancy was brought to our clinic at 11 days of life for a right lower thoracic bulge. There was no relevant family history. The physical exam was completely normal except for a 4 × 5 cm round protrusion in the right lower anterolateral chest. The ribs in the area were clearly absent. The skin, the subcutaneous tissue and the muscles appeared normal. He had normal vital signs and breath sounds. The abdominal exam was normal. A plain chest X-ray showed normal lung fields, absence of the right lower ribs, a right diaphragmatic eventration, and a soft tissue herniation. A three-dimensional computed tomography (3D-CT) reconstruction confirmed the diagnosis of congenital hypoplastic 6th and 7th right ribs, absent 8th and 9th right ribs, and a foramen in the right 10th rib. It also showed that the visible bulge was the liver protruding through the weakened chest wall. The only other anatomical anomaly was 6th to 9th thoracic butterfly vertebrae. The patient is being managed conservatively and is doing well.

Conclusion

Congenital intercostal liver herniation with absent ribs is an extremely rare condition and can be managed conservatively in asymptomatic cases. We recommend 3D-CT reconstruction as an effective way to confirm the diagnosis and rule out associated malformations.
导言先天性肋骨缺失伴肝疝是一种极为罕见的疾病。病例介绍 一名足月女婴,妊娠 38 周,出生体重 3100 克,正常妊娠后经阴道分娩,出生 11 天时因右胸下段隆起被送至我院就诊。没有相关的家族病史。体格检查完全正常,只是右胸前下侧有一个 4 × 5 厘米的圆形突起。该区域的肋骨明显缺失。皮肤、皮下组织和肌肉均正常。生命体征和呼吸音正常。腹部检查正常。胸部 X 光片显示肺野正常,右下侧肋骨缺失,右侧膈肌分离,软组织疝。三维计算机断层扫描(3D-CT)重建确诊为先天性右第 6 和第 7 肋骨发育不良,右第 8 和第 9 肋骨缺失,右第 10 肋骨有一个孔。它还显示,可见的隆起是肝脏穿过薄弱的胸壁突出。唯一的其他解剖异常是第 6 至第 9 胸椎。结论先天性肋间肝疝伴肋骨缺失是一种极为罕见的疾病,对于无症状的病例可以采取保守治疗。我们建议将三维 CT 重建作为确诊和排除相关畸形的有效方法。
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引用次数: 0
Esophageal placement of a biliary stent to manage a iatrogenic esophageal perforation: A case report 食管置入胆道支架治疗先天性食管穿孔:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-09 DOI: 10.1016/j.epsc.2024.102898
Saurin Dipak Dani, Dravina Shetty, Abhaya R. Gupta, Paras R. Kothari

Introduction

Post-operative anastomotic stricture can occur after a gastric tube esophagoplasty in children with esophageal atresia, resulting in difficulty swallowing. Endoscopic dilatation is commonly used to treat these strictures. The dilatations carry a risk of esophageal perforation.

Case presentation

A two-year-old male was admitted to the intensive care unit for an esophageal perforation that resulted from an esophageal dilatation. He had a history of long-gap type-C esophageal atresia/tracheo-esophageal, which had been managed with a cervical esophagostomy and a gastrostomy at birth, followed by an esophageal replacement by reverse gastric tube at the age of 17 months. At the age of 20 months, he developed dysphagia. An esophageal stricture was diagnosed by endoscopy. He underwent several balloon dilatations, followed by dilatations with solid dilators. The last dilatation was complicated by the esophageal perforation that prompted his hospital admission. At the time of the admission, we placed a chest tube, started antibiotics, and kept him NPO. We resumed enteral feedings 12 days after the perforation but immediately noticed that the formula was draining out of the chest tube. A surgical repair of the persistent fistula was deemed unsafe. We decided to place a biliary stent to cover the perforation endoscopically. Nasogastric feeds were reinitiated. The stent was kept in place for six weeks, while the patient was fed by a nasogastric tube. After 6 weeks, a contrast study confirmed that the perforation had sealed. Oral feedings were started at that time and the chest tube was removed. He has had no recurrence of the perforation or the stricture since then.

Conclusion

Biliary stents could be an option for the management of iatrogenic esophageal perforations in children.
导言:食道闭锁儿童在接受胃管食道成形术后可能会出现术后吻合口狭窄,导致吞咽困难。内窥镜扩张术通常用于治疗这些狭窄。病例介绍 一名两岁男童因食管扩张导致食管穿孔而被送入重症监护室。他曾患有长间隙C型食管闭锁/气管食管,出生时曾接受过颈部食管造口术和胃造口术,17个月大时接受了反向胃管食管置换术。20 个月大时,他出现了吞咽困难。通过内窥镜检查,他被诊断为食道狭窄。他接受了数次球囊扩张术,之后又用固体扩张器进行了扩张。最后一次扩张因食管穿孔而并发,导致他入院治疗。入院时,我们为他插上了胸管,开始使用抗生素,并保持 NPO。穿孔 12 天后,我们恢复了肠内喂养,但立即发现配方奶从胸管中流出。手术修补持续存在的瘘管被认为是不安全的。我们决定在内镜下放置一个胆道支架来覆盖穿孔。重新开始了鼻饲。支架放置了六周,患者使用鼻胃管进食。6 周后,造影剂检查证实穿孔已经封堵。于是开始口服喂食,并拔掉了胸管。结论胆道支架是治疗儿童先天性食管穿孔的一种选择。
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引用次数: 0
Chest wall reconstruction with biological mesh, rib stabilization, and latissimus dorsi rotation: A case report 用生物网片、肋骨固定和背阔肌旋转重建胸壁:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-05 DOI: 10.1016/j.epsc.2024.102899
Lucía Fein , Bruno Cuturi , Ruver Berazategui , Sylvia Duarte

Introduction

Children with chest wall tumors may require extensive chest wall resections. The reconstruction of large chest wall defects can be surgically challenging. We present a new chest wall reconstruction method.

Case presentation

A 2.5-year-old male who developed respiratory symptoms and chest pain was diagnosed with an Askin's tumor of the left chest wall. The tumor originated in the posterior segments of the seventh and eighth ribs. Computed tomography of the chest revealed a large mass filling the left hemithorax, causing flattening of the hemidiaphragm and mediastinal shift. No distant metastases were identified. The patient underwent six cycles of neoadjuvant chemotherapy with vincristine, adriamycin, cyclophosphamide, ifosfamide, and etoposide. Post-chemotherapy, magnetic resonance imaging revealed a significant reduction in tumor size. The patient was taken to the operating room for the resection of the tumor. The posterior segments of the left sixth, seventh, eighth, and ninth ribs were surgically excised. The chest wall was reconstructed using a biological mesh (Permacol™), sutures for costal arch stabilization using the lattice technique, and a rotated latissimus dorsi muscle flap for defect coverage. An 18 Fr pleural drainage was left in place. The patient tolerated the operation well and was successfully extubated 48 hours later. Pain was effectively managed via paravertebral block. The patient was discharged on the 14th postoperative day. The resected specimen had negative margins. He subsequently underwent several more cycles of chemotherapy. Eighteen months after the operation he has developed no scoliosis and has full range of motion on the left chest wall.

Conclusion

A combination of a biological mesh, rib stabilization and latissimus dorsi rotation seems to be a valid technique to cover large chest wall defects.
导言:患有胸壁肿瘤的儿童可能需要进行大面积胸壁切除。大面积胸壁缺损的重建手术具有挑战性。我们介绍了一种新的胸壁重建方法。病例介绍 一名 2.5 岁的男性出现呼吸道症状和胸痛,被诊断为左胸壁阿斯金肿瘤。肿瘤起源于第七和第八根肋骨的后段。胸部计算机断层扫描显示,左侧半胸腔内充满了一个巨大的肿块,导致半膈变平和纵隔移位。未发现远处转移灶。患者接受了长春新碱、阿霉素、环磷酰胺、伊福酰胺和依托泊苷六个周期的新辅助化疗。化疗后,磁共振成像显示肿瘤明显缩小。患者被送入手术室进行肿瘤切除。手术切除了左侧第六、第七、第八和第九根肋骨的后段。使用生物网(Permacol™)重建胸壁,使用格子技术缝合肋弓稳定,并使用旋转背阔肌肌皮瓣覆盖缺损。术中留置了 18 Fr 胸膜引流管。患者对手术的耐受性良好,48 小时后成功拔管。疼痛通过椎旁阻滞得到了有效控制。患者于术后第14天出院。切除的标本边缘呈阴性。随后,他又接受了几个周期的化疗。结论 生物网片、肋骨稳定和背阔肌旋转的组合似乎是覆盖大面积胸壁缺损的有效技术。
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引用次数: 0
Hydrosalpinx leading to fallopian tube torsion in a 10-year-old female: A case report 一名 10 岁女性的输卵管积水导致输卵管扭转:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-05 DOI: 10.1016/j.epsc.2024.102895
Joanna Sajdlowska , Pasha Shenasan , Nicole Clarke , Nishith Bhattacharyya

Introduction

Isolated fallopian tube torsion (IFTT) is a rare complication in the pediatric population, especially among the pre-adolescent and pre-menarcheal population.

Case report

A10-year-old prepubertal girl with no past surgical history and a history of autoimmune hepatitis, ADHD, hyperlipidemia and constipation presented with a 4-day history of sharp abdominal pain. Initially the pain was localized to the suprapubic region, but it later became diffuse. She was hemodynamically stable, and all laboratory values were unremarkable. An initial ultrasound showed preserved vascular flow to non-enlarged ovaries. Computerized tomography of the abdomen and pelvis with contrast demonstrated an indeterminate fluid structure in the right lower quadrant. A follow-up ultrasound showed right hydrosalpinx measuring 5.1 x 5.7 × 5.6 cm with free fluid adjacent to the right ovary. Magnetic resonance imaging re-demonstrated a tubular structure in the right lower quadrant adjacent to the right ovary with preserved ovaries. The patient underwent diagnostic laparoscopy, which confirmed isolated right fallopian tube torsion with ischemic changes. The Fallopian tube was detorsed and regained proper vascularization. The fluid within the Fallopian tube was evacuated. The right ovary was intact. She recovered well from the operation and was discharged home shortly thereafter. One week after the operation she was free of symptoms.

Conclusion

Fallopian tube torsion should be included in the differential diagnosis of pre-adolescent and pre-menarcheal females with abdominal pain.
导言孤立性输卵管扭转(IFTT)是儿科罕见的并发症,尤其是在青春期前和初潮前人群中。病例报告一名 10 岁的青春期前女孩,既往无手术史,有自身免疫性肝炎、多动症、高脂血症和便秘病史,4 天前出现腹部剧烈疼痛。起初,疼痛局限于耻骨上区域,但后来变得弥漫性疼痛。她的血流动力学稳定,所有化验指标均无异常。最初的超声波检查显示,未增大的卵巢血管血流通畅。使用造影剂的腹部和盆腔计算机断层扫描显示,右下腹有不明液体结构。随访超声波检查显示,右侧卵巢积水面积为 5.1 x 5.7 x 5.6 厘米,右侧卵巢附近有游离液体。磁共振成像再次显示右下腹邻近右卵巢处有管状结构,卵巢保留。患者接受了诊断性腹腔镜检查,确诊为孤立性右侧输卵管扭转并伴有缺血性改变。输卵管被剥离并恢复了正常的血管。输卵管内的液体被排空。右侧卵巢完好无损。手术后她恢复良好,不久就出院回家了。结论输卵管扭转应纳入青春期前和初潮前女性腹痛的鉴别诊断中。
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引用次数: 0
Retroperitoneal Castleman's disease and ovarian torsion: A case report 腹膜后卡斯特曼病与卵巢扭转:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-10-05 DOI: 10.1016/j.epsc.2024.102897
Mingjun Jin , Tengfei Li , Qianhui Yang , Linsheng Zhao , Liang Dong , Jianghua Zhan

Introduction

Castleman's Disease (CD) is a rare lymphoproliferative disorder with diverse clinical manifestations. It is primarily characterized by painless lymph node enlargement, most commonly in the mediastinum, neck, retroperitoneum, axillary regions, and pelvis.

Case presentation

A 12-year-old female was admitted to our hospital with persistent abdominal pain and vomiting that were not alleviated by initial treatments. Physical examination revealed tenderness in the right lower abdomen. Pelvic ultrasound revealed a mass in the pelvic region. Due to persistent pain, she underwent a laparoscopic exploration during which it was discovered that the right ovary was twisted 360°, and it was adjacent to the pelvic mass. It appeared that the mass was pushing the ovary, and this phenomenon could have been the cause of the torsion. The nature of the mass could not be conclusively determined during the intraoperative assessment, but a biopsy with a fine needle was obtained. Pathological examination was suggestive of a tumor related to the lymphatic system. She underwent a pelvic MRI that confirmed a round, soft tissue mass on the right side of the pelvis measuring approximately 63 × 50 × 56 mm. We did a subsequent laparoscopy and completely resected the mass. The patient recovered well and was discharged without complications. At 2 years of follow up she is in good health. The final pathology diagnosis of the mass was CD.

Conclusion

CD is often asymptomatic, but symptoms can arise when the enlarged lymph nodes cause compression on surrounding organs.
导言卡斯特曼病(CD)是一种罕见的淋巴组织增生性疾病,临床表现多种多样。该病的主要特征是无痛性淋巴结肿大,最常见于纵隔、颈部、腹膜后、腋窝和盆腔。病例介绍一名 12 岁女性因持续腹痛和呕吐入院,经初步治疗无效。体格检查显示右下腹有触痛。盆腔超声检查显示盆腔区域有肿块。由于持续疼痛,她接受了腹腔镜探查,在探查过程中发现右侧卵巢扭转了360°,与盆腔肿块相邻。肿块似乎在挤压卵巢,这种现象可能是扭转的原因。术中评估无法确定肿块的性质,但还是用细针进行了活检。病理检查显示是与淋巴系统有关的肿瘤。她接受了盆腔磁共振成像检查,结果证实盆腔右侧有一个圆形软组织肿块,大小约为 63 × 50 × 56 毫米。我们随后进行了腹腔镜手术,完全切除了肿块。患者恢复良好,无并发症后出院。随访两年后,她的健康状况良好。该肿块的最终病理诊断为 CD。ConclusionCD 通常没有症状,但当肿大的淋巴结压迫周围器官时就会出现症状。
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引用次数: 0
A novel approach to pediatric chest wall reconstruction using a 3D-printed biodynamic prosthesis: A case report 使用 3D 打印生物动力假体重建小儿胸壁的新方法:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-09-30 DOI: 10.1016/j.epsc.2024.102896
Keerthi Burgi , Nikhil R. Shah , Tammy Stoll , James D. Geiger

Introduction

Chest wall reconstruction in children continues to be a challenging undertaking, aiming to restore both form and function. An optimal chest wall reconstruction material has not been identified that would provide dynamic chest function and would ideally avoid potential long-term sequelae.

Case presentation

A 15-year-old female with Li Fraumeni syndrome and a history of hepatic rhabdomyosarcoma presented with increasing left back and left scapular pain initially attributed to her known scoliosis. Computed tomography revealed a 12 x 6 × 7 cm mass encasing ribs 3–5 extending into the left chest wall and axilla. Image-guided biopsy confirmed a high-grade osteosarcoma without extra-thoracic metastatic disease. She underwent neoadjuvant chemotherapy after which a three-dimensional (3D) anatomic model of the tumor was printed using the post-therapy imaging reconstruction. The surgical team collaborated with Osteobionix (Osteobionics S.L., Santa Lucia de Tirajana, Spain) to create a patient-specific dynamic chest wall reconstruction implant, based on 3D modeling of the planned resection zone. The final implant was 3D printed with a titanium alloy using electron-beam manufacturing technology, sterilized prior to implantation. She underwent left thoracotomy and rib resection with prosthesis fixation to the T3, T4, and T5 vertebral bodies posteriorly and to the sternum anteriorly. More than2 years after the operation, she is doing well without evidence of recurrent disease. She has an excellent cosmetic result, there has been no progression of her scoliosis, and she has returned to competitive sports.

Conclusion

Our titanium-based 3D-printed, patient-specific implant seems to be a valuable option for the reconstruction of the chest wall in children who require an extensive chest wall resection.
导言儿童胸壁重建仍然是一项具有挑战性的工作,其目的是同时恢复外形和功能。病例介绍 一位患有李-弗劳米尼综合征并有肝横纹肌肉瘤病史的 15 岁女性因左背部和左肩胛骨疼痛加剧而就诊,最初的原因是她已知的脊柱侧弯。计算机断层扫描显示,一个 12 x 6 x 7 厘米的肿块包裹着 3-5 根肋骨,并向左胸壁和腋窝延伸。图像引导下的活组织检查证实这是一个高级别骨肉瘤,没有胸腔外转移性疾病。她接受了新辅助化疗,之后利用治疗后的成像重建打印了肿瘤的三维(3D)解剖模型。手术团队与 Osteobionix 公司(Osteobionics S.L.,西班牙圣卢西亚德蒂拉亚纳)合作,根据计划切除区域的三维建模,制作了患者专用的动态胸壁重建植入物。最终植入物采用电子束制造技术用钛合金进行三维打印,并在植入前进行消毒。她接受了左侧开胸手术和肋骨切除术,假体后部固定在T3、T4和T5椎体上,前部固定在胸骨上。术后两年多,她的情况良好,没有复发的迹象。结论我们基于钛的三维打印患者特异性植入物似乎是需要大面积胸壁切除的儿童重建胸壁的重要选择。
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引用次数: 0
Intraductal papilloma presenting as a breast mass in an 8-month-Old male infant: A case report 8 个月大男婴乳房肿块中出现的导管内乳头状瘤:病例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2024-09-28 DOI: 10.1016/j.epsc.2024.102894
Maen Kamal , Nesma Ghanim , Raima Hashmi , Zachary Patterson , Qazi Azher , Antonio J. Williams Sr

Introduction

Intraductal papilloma is an exceptionally rare condition in the pediatric population, particularly among males in infancy and early childhood, typically most prevalent among women aged between 35 and 55 years presenting as sanguineous or serosanguineous nipple discharge and a discrete breast mass. Despite its historical classification as benign, emerging research indicates a potential for malignant transformation, prompting a reevaluation of its clinical significance.

Case presentation

We present the case of a healthy 8-month-old male infant with a right subareolar breast swelling that appeared non tender, well-circumscribed, smooth, mobile, rubbery, without any associated nipple discharge or regional lymphadenopathy, clinically measuring 10 × 10 mm in diameter, and exhibited a bluish green hue with no associated changes in overlying skin. Ultrasound confirmed presence of a right retro areolar breast mass, characterized as a multiseptated complex cyst-like structure with tubular anechoic formations within, measuring 21 x 20 × 6 mm. The findings were suggestive of possible duct ectasia classified as Bi-Rads Category 3 with a benign probability. Due to parental concern for increase in size over 2 months, the mass was resected under general anesthesia through a subareolar incision. Microscopic analysis confirmed dilated cystic ducts and an intraductal papilloma with calcifications. The patient recovered well from the operation, and follow-up at nine months, he has experienced no recurrences.

Conclusion

Although rare, this case underscores the importance of including intraductal papilloma in the differential diagnosis of breast nodules in male infants.
导言导管乳头状瘤在儿科人群中是一种极为罕见的疾病,尤其是在婴幼儿期的男性中,通常在 35 至 55 岁的女性中最为常见,表现为血色或血清样乳头溢液和离散性乳房肿块。本病例中,一名 8 个月大的健康男婴右侧乳晕下乳房肿胀,无触痛,边界清楚,光滑,可移动,呈橡胶样,无任何相关的乳头溢液或区域淋巴结病,临床上直径为 10 × 10 毫米,呈蓝绿色,上覆皮肤无相关变化。超声波检查证实了右侧乳晕后肿块的存在,其特征为多隔复合囊肿样结构,内有管状无回声形成,大小为 21 × 20 × 6 毫米。检查结果提示可能存在导管异位,被归为 Bi-Rads 3 类,良性可能性大。由于父母担心肿块会在两个月内增大,因此在全身麻醉下通过乳晕下切口切除了肿块。显微镜分析证实,囊性导管扩张,导管内乳头状瘤伴有钙化。患者术后恢复良好,9 个月后随访,未见复发。结论虽然罕见,但该病例强调了将导管内乳头状瘤纳入男婴乳腺结节鉴别诊断的重要性。
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引用次数: 0
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Journal of Pediatric Surgery Case Reports
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