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Nodular lymphoid hyperplasia simulating ileocolic intussusception: A case report 模拟回结肠肠套叠的结节状淋巴增生:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-27 DOI: 10.1016/j.epsc.2024.102831
James G. Glasser

Introduction

Nodular lymphoid hyperplasia is usually encountered by gastroenterologists during a workup for chronic abdominal pain accompanied by food intolerance, hematochezia, or diarrhea. When an intestinal endoscopy is performed, the usual finding is multiple small submucosal nodules that are identified as lymphoid tissue by biopsy.

Case presentation

A previously healthy 8-year-old boy presented with a several-day history of intermittent abdominal pain and vomiting. He had no history of diarrhea or bloody stools. He was afebrile, and all of the other vital signs were normal. His abdomen was distended and tender, particularly in the right upper quadrant. Blood work was normal, except for an elevated C-reactive protein. His imaging work-up consisted of an abdominal ultrasound that ruled out acute appendicitis and a computerized tomography (CT) scan that showed images suggestive of ileo-colic intussusception, with a soft tissue mass in the right upper quadrant. Due to his age, reduction of the intussusception by insufflation was not attempted. An exploratory laparotomy was done through an incision in the right lower quadrant. We found no intussusception; rather, the wall of the cecum and the base of the appendix were markedly thickened, causing a partial obstruction of the cecal lumen and the ileo-cecal valve. We proceeded with an ileo-cecectomy and ileocolic anastomosis. He was discharged home on the 4th postoperative day. The pathologic and molecular analysis confirmed the diagnosis of follicular lymphoid hyperplasia.

Conclusion

Nodular lymphoid hyperplasia of the ileo-cecal region can mimic an ileo-colic intussusception. It should be included in the differential diagnosis, particularly in patients outside the age range of idiopathic intussusception.

导言:结节性淋巴组织增生症通常是消化内科医生在检查慢性腹痛并伴有食物不耐受、便血或腹泻时遇到的。在进行肠道内窥镜检查时,通常会发现多个黏膜下小结节,活检后可确定为淋巴组织。病例介绍 一个以前身体健康的 8 岁男孩因间歇性腹痛和呕吐就诊数天。他没有腹泻或血便病史。他无发热,其他生命体征均正常。他的腹部胀痛,尤其是右上腹。血液检查正常,只是C反应蛋白升高。他的影像学检查包括腹部超声波检查和计算机断层扫描(CT),前者排除了急性阑尾炎的可能,后者显示的图像提示回肠结肠肠套叠,右上腹有软组织肿块。由于患者年龄较大,没有尝试通过充气来减少肠套叠。我们从右下腹切口进行了探查性开腹手术。我们没有发现肠套叠;相反,盲肠壁和阑尾底部明显增厚,导致盲肠腔和回盲肠瓣部分阻塞。我们为他进行了回盲肠切除术和回结肠吻合术。他在术后第 4 天出院回家。病理和分子分析证实了滤泡状淋巴增生的诊断。应将其纳入鉴别诊断,尤其是特发性肠套叠年龄段以外的患者。
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引用次数: 0
Fetus in fetu in a 1-year-old female: A case report 一名 1 岁女性的胎儿:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-27 DOI: 10.1016/j.epsc.2024.102830
Ayushi Agarwal, Meetu Agrawal, Sana Ahuja

Background

Fetus in fetu (FIF) is an exceedingly rare congenital anomaly characterized by the presence of a partially developed fetus within the body of its twin. It is a rare entity with fewer than 200 cases documented worldwide. Typically presenting as an abdominal mass in early childhood, this condition remains a diagnostic challenge due to its infrequency and varied clinical manifestations.

Case presentation

We present the case of a 1-year-old female child referred to the pediatric surgery department due to an abdominal mass detected by her parents. Physical examination revealed a firm, ill-defined mass in the right upper abdomen. Ultrasonography delineated a heterogeneous, calcified mass in the right upper quadrant. Laboratory investigations (complete blood count, viral markers), including tumor markers (AFP, beta-hCG), were within normal limits.

A midline laparotomy was performed, and intraoperative findings revealed a well-encapsulated mass adherent to the retroperitoneum, adjacent to the liver. Careful dissection was carried out to preserve vital structures, and the mass was successfully removed without rupture. Histopathological examination confirmed the diagnosis of fetus in fetu, displaying a spectrum of tissues reminiscent of embryonic development, including skin, adnexal structures, brain tissue, mature cartilage, adipose tissue, bone marrow elements, and a partially developed vertebral column. After the surgical resection, the patient experienced an uneventful postoperative recovery. At one year of follow up the patient has shown no signs of recurrence.

Conclusion

FIF is characterized by diagnostic challenges, often requiring postoperative histological confirmation. Histopathological examination confirmed the presence of FIF. Surgical excision, is crucial for favorable outcomes, especially in preventing potential complications. Comprehensive histopathological evaluation remains imperative for accurate diagnosis.

背景胎儿畸形(Fetus in fetu,FIF)是一种极其罕见的先天性畸形,其特点是在双胞胎体内存在一个部分发育的胎儿。胎儿畸形(Fetus in fetu)是一种极为罕见的先天性畸形,其特征是双胎体内存在部分发育的胎儿。该病例通常在幼儿期出现腹部肿块,由于其发病率低且临床表现多样,因此仍是诊断上的难题。体格检查发现,右上腹有一个质地坚硬、界限不清的肿块。超声波检查显示右上腹有一个异质的钙化肿块。患者接受了中线开腹手术,术中发现一个包裹性良好的肿块紧贴腹膜后,与肝脏相邻。为保留重要结构,手术中进行了仔细的剥离,肿块在没有破裂的情况下被成功切除。组织病理学检查证实了胎儿的诊断,显示出一系列胚胎发育的组织,包括皮肤、附件结构、脑组织、成熟软骨、脂肪组织、骨髓元素和部分发育的椎体。手术切除后,患者术后恢复顺利。结论 FIF 的特点是诊断困难,通常需要术后组织学确认。组织病理学检查证实了 FIF 的存在。手术切除是取得良好疗效的关键,尤其是在预防潜在并发症方面。全面的组织病理学评估仍是准确诊断的必要条件。
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引用次数: 0
Association of concomitant congenital diaphragmatic hernia and Hirschsprung's disease with a mutation in the FOXP1 gene: A case report 先天性膈疝和赫氏病同时与 FOXP1 基因突变有关:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-26 DOI: 10.1016/j.epsc.2024.102829
Vyacheslav Lenkov , Jason O. Robertson

Introduction

Forkhead box protein P1 (FOXP1) is a transcriptional factor that plays a role in gene regulation in a wide array of human tissues. Mutations of this gene result in developmental delay, language deficits, brain, cardiac and urogenital anomalies. Some evidence also connects these mutations with congenital diaphragmatic hernia (CDH) and Hirschsprung's disease (HD). This report describes a case of a newborn with a FOXP1 mutation who was diagnosed with both CDH and HD, suggesting the gene's possible association with both phenotypes.

Case report

The patient was born at 38w2d to a healthy mother. Multiple congenital anomalies were discovered prenatally, including Dandy-Walker malformation, bilateral cleft lip and palate, polydactyly and syndactyly. No chromosomic aneuploidies were identified; however, whole exome sequencing analysis detected a missense mutation of FOXP1. The patient passed meconium on day-of-life zero and progressed appropriately. Presentation of CDH manifested at 7 weeks with respiratory failure following delayed intrathoracic herniation. Chest x-ray confirmed the diagnosis, and a small type A defect was identified at the time of surgery. At 12 weeks, worsening abdominal distention prompted workup of Hirschsprung's disease. Suction rectal biopsy confirmed the diagnosis. Both CDH and HD were addressed surgically upon diagnosis. Post-operative courses were uncomplicated.

Conclusion

This report demonstrates an unusual association of CDH and HD in a patient with a FOXP1 mutation. Both diseases were on the mild spectrum and demonstrated delayed presentations. The role of this FOXP1 mutation as a genetic cause of both diseases warrants further investigation.

导言叉头盒蛋白 P1(FOXP1)是一种转录因子,在多种人体组织的基因调控中发挥作用。该基因突变会导致发育迟缓、语言障碍、大脑、心脏和泌尿生殖系统异常。一些证据还表明,这些基因突变与先天性膈疝(CDH)和赫斯普隆氏病(HD)有关。本报告描述了一例 FOXP1 基因突变的新生儿,该新生儿被诊断同时患有 CDH 和 HD,这表明该基因可能与这两种表型都有关联。产前发现多种先天性畸形,包括丹迪-沃克畸形、双侧唇腭裂、多指畸形和并趾畸形。没有发现染色体非整倍体,但全外显子组测序分析发现了FOXP1的错义突变。患者在出生后第零天排出胎粪,病情进展顺利。7周时出现CDH,延迟胸内疝后出现呼吸衰竭。胸部X光检查确诊,手术时发现了一个小的A型缺损。12周时,腹胀症状加重,促使他接受赫氏病检查。直肠抽吸活检证实了这一诊断。CDH 和 HD 均在确诊后进行了手术治疗。结论本报告展示了一名 FOXP1 基因突变患者同时患有 CDH 和 HD 的罕见病例。这两种疾病都属于轻度疾病谱,并且表现为延迟发病。这种 FOXP1 基因突变是导致这两种疾病的遗传原因之一,值得进一步研究。
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引用次数: 0
Life-threatening presentation of giant mature teratoma in a 9-year-old female: A case report 一名 9 岁女性的巨大成熟畸胎瘤危及生命:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-16 DOI: 10.1016/j.epsc.2024.102825
José Ortiz-Fullana , Kenneth Aviles-Fernández , Daniel Corsino-Garayua , Victor Ortiz-Justiniano

Introduction

Mediastinal teratomas are rare in children and adolescents; however, they often present diagnostic and therapeutic challenges, given their potential for malignancy. Rapid intervention, particularly surgical excision, is crucial for large, life-threatening masses following clinical stabilization.

Case presentation

A 9-year-old female presented with progressive left costal pain and dyspnea on exertion, culminating in acute respiratory failure prior to admission. Examination revealed a hypopigmented square patch in the left lower quadrant of the abdomen. Initial chest x-ray (CXR) showed complete opacification of the left lung with mediastinal shift to the right. Subsequent chest CT scan confirmed a 14.2cm × 11.2cm x 14.5cm complex, multiloculated, multiseptated mass suggestive of neoplasia. Local mass effect resulted in the obliteration of the left mainstem bronchi, left upper and lower lobe bronchi, lung atelectasis, and a large left pleural effusion. Surgical excision was planned following consultation with Pediatric Anesthesiology and Intensivist. The patient underwent left posterolateral thoracotomy through the fifth intercostal space, during which a large complex cystic mass (17.5 cm × 16.8 cm x 7.7 cm) arising from the thymus was excised after careful dissection from surrounding lung parenchyma and pericardium. Postoperatively, she recovered well in the Pediatric Intensive Care Unit (PICU) and was discharged on postoperative day #8 after pathology confirmed a benign teratoma.

Conclusion

Mediastinal teratomas in pediatric patients presenting with life-threatening signs and symptoms necessitate early recognition, involvement of a multidisciplinary team, and prompt surgical excision to ensure optimal outcomes.

导言纵隔畸胎瘤在儿童和青少年中非常罕见,但由于其具有恶变的可能性,因此在诊断和治疗方面常常面临挑战。病例介绍 一位 9 岁女性因左肋部进行性疼痛和呼吸困难入院,入院前出现急性呼吸衰竭。检查发现左下腹部有一块色素减退的方形斑块。最初的胸部 X 光片(CXR)显示左肺完全不透明,纵隔向右侧移位。随后的胸部 CT 扫描证实了一个 14.2 厘米 x 11.2 厘米 x 14.5 厘米的复杂、多灶、多切面肿块,提示为肿瘤。局部肿块效应导致左主干支气管、左上叶和左下叶支气管阻塞、肺不张和左胸腔大量积液。儿科麻醉科和重症监护室医生会诊后计划进行手术切除。患者接受了经第五肋间的左侧后外侧胸廓切开术,术中仔细剥离了周围的肺实质和心包,切除了胸腺中巨大的复杂囊性肿块(17.5 厘米 × 16.8 厘米 × 7.7 厘米)。术后,她在儿科重症监护室(PICU)恢复良好,经病理证实为良性畸胎瘤后于术后第 8 天出院。
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引用次数: 0
Septic femoral and caval thrombophlebitis secondary to acute appendicitis: A case report 继发于急性阑尾炎的化脓性股静脉和腔静脉血栓性静脉炎:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-11 DOI: 10.1016/j.epsc.2024.102822
Daniel B. Gehle , Philip W. Morgan , Sara A. Mansfield , Regan F. Williams , Howard I. Pryor II

Introduction

Septic thrombophlebitis (STP) of deep veins is a rare condition that harbors significant morbidity in the pediatric population. Most cases of STP in pediatric patients have been associated with head and neck infections such as in Lemierre syndrome.

Case presentation

A 3-year-old male presented with a 10-day history of fever, abdominal pain, and diarrhea and was diagnosed with perforated appendicitis. He underwent laparoscopic hand-assisted appendectomy and was discharged on post-operative day 5. He re-presented to the emergency department two weeks later with several day history of abdominal distension and vomiting and imaging demonstrated a high-grade small bowel obstruction. He underwent exploratory laparotomy and ileocecectomy. His postoperative course was complicated by acute respiratory failure secondary to abdominal compartment syndrome requiring decompressive laparotomy. His clinical status improved and he was able to undergo abdominal closure 4 days later. He then developed persistent fevers and was found to have deep venous thrombosis of bilateral femoral veins extending to the infrarenal inferior vena cava consistent with septic thrombophlebitis. He was successfully treated for his deep venous thrombosis with a course of antibiotics and therapeutic anticoagulation and has made a full recovery.

Conclusion

STP of deep veins is generally managed with antibiotics and therapeutic anticoagulation and rarely requires invasive interventions. This case highlights that in young children, the inferior vena cava is small enough caliber to develop thrombosis and that STP of the pelvic veins and inferior vena cava can occur secondary to intraabdominal infection and its sequelae.

导言深静脉血栓性静脉炎(STP)是一种罕见的疾病,在儿童群体中发病率很高。大多数儿童患者的血栓性静脉炎都与头颈部感染有关,例如莱米埃尔综合征。病例介绍一名 3 岁的男性患者因发烧、腹痛和腹泻就诊 10 天,被诊断为阑尾炎穿孔。他接受了腹腔镜手助阑尾切除术,术后第 5 天出院。两周后,他再次来到急诊科就诊,腹胀和呕吐症状持续了数天,影像学检查显示小肠梗阻程度较高。他接受了剖腹探查术和回肠切除术。术后因腹腔间隙综合征导致急性呼吸衰竭,需要进行减压开腹手术。他的临床状况有所改善,4 天后可以进行腹腔闭合手术。随后,他出现持续发烧,并被发现双侧股静脉深静脉血栓形成,一直延伸到肾下腔静脉,与化脓性血栓性静脉炎一致。通过一个疗程的抗生素治疗和抗凝治疗,他成功治愈了深静脉血栓,目前已完全康复。本病例强调,幼儿的下腔静脉口径较小,足以形成血栓,盆腔静脉和下腔静脉 STP 可继发于腹腔内感染及其后遗症。
{"title":"Septic femoral and caval thrombophlebitis secondary to acute appendicitis: A case report","authors":"Daniel B. Gehle ,&nbsp;Philip W. Morgan ,&nbsp;Sara A. Mansfield ,&nbsp;Regan F. Williams ,&nbsp;Howard I. Pryor II","doi":"10.1016/j.epsc.2024.102822","DOIUrl":"https://doi.org/10.1016/j.epsc.2024.102822","url":null,"abstract":"<div><h3>Introduction</h3><p>Septic thrombophlebitis (STP) of deep veins is a rare condition that harbors significant morbidity in the pediatric population. Most cases of STP in pediatric patients have been associated with head and neck infections such as in Lemierre syndrome.</p></div><div><h3>Case presentation</h3><p>A 3-year-old male presented with a 10-day history of fever, abdominal pain, and diarrhea and was diagnosed with perforated appendicitis. He underwent laparoscopic hand-assisted appendectomy and was discharged on post-operative day 5. He re-presented to the emergency department two weeks later with several day history of abdominal distension and vomiting and imaging demonstrated a high-grade small bowel obstruction. He underwent exploratory laparotomy and ileocecectomy. His postoperative course was complicated by acute respiratory failure secondary to abdominal compartment syndrome requiring decompressive laparotomy. His clinical status improved and he was able to undergo abdominal closure 4 days later. He then developed persistent fevers and was found to have deep venous thrombosis of bilateral femoral veins extending to the infrarenal inferior vena cava consistent with septic thrombophlebitis. He was successfully treated for his deep venous thrombosis with a course of antibiotics and therapeutic anticoagulation and has made a full recovery.</p></div><div><h3>Conclusion</h3><p>STP of deep veins is generally managed with antibiotics and therapeutic anticoagulation and rarely requires invasive interventions. This case highlights that in young children, the inferior vena cava is small enough caliber to develop thrombosis and that STP of the pelvic veins and inferior vena cava can occur secondary to intraabdominal infection and its sequelae.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":null,"pages":null},"PeriodicalIF":0.4,"publicationDate":"2024-05-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000502/pdfft?md5=5dfeedace077884139c8e1171148647e&pid=1-s2.0-S2213576624000502-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140947713","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}
引用次数: 0
Successful separation of conjoined twins, one healthy and one stillborn: A case report 成功分离连体婴儿,一个健康,一个死产:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-09 DOI: 10.1016/j.epsc.2024.102823
Nakubulwa Clare , Ajiko Mary Margaret , Henry Mark Lugobe , Alaroker Florence Olwedo Egwau , Epodoi Joseph

Introduction

Conjoined twins are rare congenital malformations that occur in identical twins who share both a placenta and an amniotic sac. If one twin dies, emergency separation is necessary to save the other twin.

Case presentation

A pair of conjoined twin girls, one stillborn and one alive, was referred to our facility 18 hours after delivery from a primary health facility. The twins were diagnosed at birth in a primigravida who had obstructed labor and were delivered by emergency cesarean section. They were joined at the lower chest and upper abdomen and had separate umbilical cords. Imaging studies to determine the extent of organ involvement in the twins were not conducted due to the unavailability of the necessary equipment at our facility. By the second day of life, the surviving twin showed sepsis and was given intravenous fluids and antibiotics. On the sixth day of life, they were taken to the operating room for separation. The twins shared part of the abdominal wall, part of the chest wall and had fused livers, all of which were separated. Following the operation, the surviving twin was treated for sepsis and received nutritional rehabilitation. After a hospital stay of six weeks she was discharged home.

Conclusion

An urgent operation to separate conjoined twins when one of them is stillborn is mandatory, even in a setting with limited diagnostic and surgical capabilities.

导言连体婴儿是一种罕见的先天性畸形,发生在共用胎盘和羊膜囊的同卵双胞胎身上。病例介绍 一对连体女婴,一个死胎,一个活胎,在分娩 18 小时后从一家初级医疗机构转到我们医院。这对双胞胎在一名初产妇分娩时被诊断为难产,并通过紧急剖腹产分娩。她们的下胸部和上腹部相连,脐带分离。由于本院没有必要的设备,因此没有进行影像学检查以确定双胞胎器官受累的程度。出生后第二天,存活的双胞胎出现败血症,医生为其静脉输液并使用抗生素。出生后第六天,他们被送往手术室进行分离。双胞胎共用部分腹壁、部分胸壁,肝脏融合,所有这些都被分离了。手术后,幸存的双胞胎接受了败血症治疗和营养康复治疗。结论:即使在诊断和手术能力有限的情况下,也必须对其中一个死产的连体婴儿实施紧急分离手术。
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引用次数: 0
Gastric volvulus in a child with a diaphragmatic eventration: A case report 一名患有膈肌分离症的儿童出现胃扩张:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-06 DOI: 10.1016/j.epsc.2024.102821
Samuel Amosilo Santoso Kesek , Alexandra Sachlan , Karina Priskila Setiawan

Introduction

Pediatric gastric volvulus (GV) with diaphragmatic eventration is a rare but lethal condition.

Case presentation

A 1-year-old girl complained of progressive vomiting, abdominal pain, and distension. On examination, plain abdominal radiograph demonstrated an air bubble sign in the left upper abdomen with elevation of the hemidiaphragm, while water-soluble contrast follow-through imaging showed partial gastric rotation. During exploratory laparotomy, gastric volvulus (GV) with left diaphragmatic eventration was identified. This abnormality was reduced by performing diaphragmatic plication and gastropexy. Postoperatively, the patient recovered without complications and was discharged on the tenth day of treatment.

Conclusion

Pediatric gastric volvulus (GV) with diaphragmatic eventration is a surgical emergency. This case underscores the importance of considering GV in pediatric patients presenting with vomiting and abdominal symptoms, specifically when radiographic evidence suggests gastric distension and diaphragmatic abnormalities.

病例介绍 一名 1 岁女童主诉进行性呕吐、腹痛和腹胀。检查时,腹部平片显示左上腹有气泡征,半膈抬高,水溶性造影剂随访成像显示部分胃旋转。在探查性开腹手术中,发现了伴有左侧膈肌连通的胃扩张(GV)。通过膈肌成形术和胃切除术缩小了这一异常。结论小儿胃扩张(GV)伴膈肌连突是外科急症。本病例强调了在小儿患者出现呕吐和腹部症状时考虑胃扩张的重要性,特别是当放射学证据显示胃扩张和膈肌异常时。
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引用次数: 0
Perforated Meckel's diverticulum as an uncommon cause of pneumoperitoneum in the newborn: A case report 梅克尔憩室穿孔是新生儿腹腔积气的罕见病因:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-06 DOI: 10.1016/j.epsc.2024.102820
William G. Lee , Shannon T. Wong-Michalak , Richard C. Judelson , Eveline H. Shue , Juan Carlos Pelayo , Eugene S. Kim

Introduction

Pneumoperitoneum in neonates is an ominous sign of gastrointestinal (GI) perforation which often requires emergent surgical intervention. Although Meckel's diverticulum (MD) is the most common congenital anomaly of the GI tract, spontaneous MD perforation leading to pneumoperitoneum in the immediate postnatal period is an extremely rare occurrence. This report describes the case of a perforated MD presenting as pneumoperitoneum on day of life (DOL) one.

Case presentation

A 1-day-old 36 week gestational age male presented with acute abdominal distention and guarding upon examination. Two-view abdominal radiographs revealed pneumoperitoneum without pneumatosis intestinalis, portal venous gas, or bowel dilation. Exploratory laparotomy was performed and bilious ascites was encountered – secondary to a perforated MD in the distal ileum. The perforated MD and adjacent small bowel were resected and a double-barrel ileostomy was created. He recovered successfully and without complication in the acute postoperative period. Histopathologic findings were consistent with a perforated MD with ectopic pancreatic tissue.

Conclusion

This rare case highlights the importance of early surgical intervention for neonates with pneumoperitoneum secondary to a perforated MD. Heightened clinical suspicion for a perforated MD should be considered among term/late preterm neonates presenting with pneumoperitoneum and low suspicion for necrotizing enterocolitis.

导言:新生儿腹腔积气是胃肠道(GI)穿孔的一个不祥征兆,通常需要紧急手术干预。虽然梅克尔憩室(MD)是最常见的先天性消化道畸形,但在新生儿期自发性MD穿孔导致腹腔积气的情况却极为罕见。本报告描述了一例在出生后第 1 天(DOL)出现腹腔积气的 MD 穿孔病例。病例介绍:一名出生 1 天、胎龄 36 周的男性患者在接受检查时出现急性腹胀和警惕性增高。双视野腹部X光片显示腹腔积气,但无肠积气、门静脉积气或肠管扩张。患者接受了剖腹探查术,结果发现胆汁性腹水--继发于回肠远端MD穿孔。他切除了穿孔的 MD 和邻近的小肠,并进行了双管回肠造口术。他在术后急性期恢复顺利,没有出现并发症。组织病理学检查结果显示,新生儿腹腔积气与胰腺组织异位的MD穿孔一致。对于出现腹腔积气的足月期/晚期早产新生儿,临床上应高度怀疑 MD 穿孔,而对坏死性小肠结肠炎的怀疑度较低。
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引用次数: 0
Long-term survival of a patient with giant omphalocele and left congenital diaphragmatic hernia: A case report 巨脑疝和左侧先天性膈疝患者的长期存活:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-06 DOI: 10.1016/j.epsc.2024.102819
Soma Sakamura , Shohei Honda , Takafumi Kondo , Insu Kawahara , Kazutoshi Cho , Akinobu Taketomi

Introduction

The combination of a giant omphalocele (GO) and congenital diaphragmatic hernia (CDH) is extremely rare and results in high mortality and morbidity rates in neonates. The most difficult period for survival is the immediate postnatal period, with approximately one-third of newborns dying within the first 48 h after birth.

Case presentation

We present the case of a newborn girl with a GO and congenital left-sided Bochdalek hernia who survived beyond the neonatal period and underwent radical surgery. The female neonate, weighing 2946 g, was delivered by scheduled Cesarean section at 37 weeks gestation. She had a left-sided Bochdalek CDH containing a small portion of the small bowel and a GO containing a large portion of the liver. There were no other associated malformations, and the pulmonary hypoplasia and pulmonary hypertension in the neonatal period were not fatal. We performed an operation to repair the diaphragmatic hernia, initially approaching it from her abdominal wall defect. We performed multiple operations to close the abdominal wall. Initially, during the neonatal period, we closed it with a skin layer; however, poor weight gain occurred afterward. Considering gastroesophageal reflux as a complication, we performed mesh-assisted abdominal wall closure and fundoplication at 10 months of age, successfully resolving the underlying disease and complications.

Conclusion

Repair of the CDH from the abdominal wall defect provided good visual field and safer primary direct closure. Without using artificial materials in the CDH until closure of the GO avoided infection and resulted in a favorable outcome in this case.

导言巨大脐疝(GO)和先天性膈疝(CDH)并发症极为罕见,导致新生儿死亡率和发病率居高不下。新生儿存活最困难的时期是出生后不久,约有三分之一的新生儿在出生后 48 小时内死亡。本病例是一名患有巨脑疝和先天性左侧 Bochdalek 疝的新生女婴,她在新生儿期后存活下来并接受了根治手术。这名新生女婴体重 2946 克,在妊娠 37 周时通过剖腹产手术顺利分娩。她患有左侧Bochdalek CDH,包含一小部分小肠和一个包含大部分肝脏的GO。没有其他相关畸形,新生儿期肺发育不全和肺动脉高压并不致命。我们对她进行了膈疝修补手术,最初是从她的腹壁缺损处入手。我们进行了多次手术来闭合腹壁。最初,在新生儿期,我们用皮肤层进行了闭合,但之后体重增加缓慢。考虑到胃食管反流是并发症之一,我们在患儿10个月大时为其实施了网状辅助腹壁闭合术和胃底折叠术,成功解决了基础疾病和并发症。该病例在闭合GO前未在CDH处使用人工材料,避免了感染,取得了良好的疗效。
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引用次数: 0
Porcelain gallbladder in a 4-year-old male: A case report 一名 4 岁男性的瓷胆囊:病例报告
IF 0.4 Q4 Medicine Pub Date : 2024-05-04 DOI: 10.1016/j.epsc.2024.102817
George Jarrouj , Lyonell B. Kone , Brian P. Adley , Vincent Franco Biank , Brian Alan Jones

Introduction

Porcelain gallbladder (PGB) is a gallbladder with calcium encrusted on its inner wall. PGB is rare in the pediatric population with only four cases reported in the literature. We present a patient who is the youngest reported case of porcelain gallbladder.

Case presentation

A 4-year-old full-term male with a history of chronic constipation presented with abdominal pain prompting a plain abdominal X-ray which revealed an incidental calcified gallbladder. This was confirmed with an abdominal ultrasound (US) and computed tomography (CT) that identified calcifications in the gallbladder wall. An elective laparoscopic cholecystectomy was performed, given the reported association of PGB with gallbladder cancer. Final pathology confirmed calcification of the gallbladder, along with evidence of cholelithiasis, and cholecystitis, but no dysplasia or malignancy. The patient was discharged on post-operative day 2 without any complications.

Conclusion

A PGB is a rare finding in the pediatric population, with prior studies indicating an association with malignancy and cholecystitis in the adult population. However, there is not enough data to support the association of PGB with future pathologies in the pediatric population and surgical intervention may not be warranted.

导言瓷胆囊(PGB)是指胆囊内壁有钙质包裹。瓷胆囊在儿童中非常罕见,文献中仅报道过四例。病例介绍一名4岁的足月男婴,有慢性便秘病史,因腹痛而接受腹部X光检查,结果显示胆囊钙化。腹部超声波(US)和计算机断层扫描(CT)证实了这一点,并在胆囊壁上发现了钙化。鉴于有报道称 PGB 与胆囊癌有关联,患者接受了择期腹腔镜胆囊切除术。最终病理证实胆囊钙化、胆石症和胆囊炎,但没有发育不良或恶变。患者于术后第 2 天出院,未出现任何并发症。结论:PGB 在儿科人群中较为罕见,之前的研究表明它与成人人群中的恶性肿瘤和胆囊炎有关。然而,目前还没有足够的数据支持 PGB 与儿科人群未来的病变有关,因此可能不需要进行手术干预。
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Journal of Pediatric Surgery Case Reports
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