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Early necrotizing enterocolitis in a 2-day-old term neonate: A case report 2天大新生儿早期坏死性小肠结肠炎1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-27 DOI: 10.1016/j.epsc.2025.103139
Samuel Kefiyalew Kelbessa , Sinbona Ararsa Keneni , Abdisa Ejeta Gedefa , Demelash Darota Dojamo

Introduction

Necrotizing enterocolitis (NEC) remains one of the most devastating and poorly understood intestinal diseases in neonates.

Case description

A 30-h-old male neonate was born at a gestational age (GA) of 39 weeks and 3 days, weighing 2800 g, with APGAR scores of 7 and 8 at one and 5 min, respectively. After delivery, the baby began breastfeeding and passed meconium within 16 hours, but developed bilious vomiting, abdominal distension, and dark-red rectal bleeding at 24 hours of age. Palpation of the abdomen elicited a crying response from the infant, and Per rectal exam showed blood on the examining catheter. The white blood cell count was 10.25 × 10^9/L, with a neutrophil percentage of 78.6 %. Abdominal imaging was not conducted due to logistical reasons. Given the clinical presentation, a diagnosis of intestinal malrotation with acute midgut volvulus and potential ischemia was made. The infant underwent laparotomy, revealing necrosis of the transverse and descending colon, which was excised, and the ascending colon was anastomosed to the sigmoid colon. A biopsy of the necrotic colon was consistent with necrotizing enterocolitis, revealing coagulative necrosis and pneumatosis cystoides intestinalis. Postoperatively, the infant had a nasogastric tube for five days, progressed to full feeding over a week, and was discharged on day 14, thriving at eight months post-operation.

Conclusion

Necrotizing enterocolitis must be included in the differential diagnosis of term neonates who develop bilious emesis, abdominal distension, and bloody stool.
坏死性小肠结肠炎(NEC)仍然是新生儿中最具破坏性和知之甚少的肠道疾病之一。病例描述1例30 h大男婴,出生时胎龄39周3天,体重2800 g, 1分钟和5分钟APGAR评分分别为7分和8分。分娩后,婴儿开始母乳喂养并在16小时内排出胎便,但24小时时出现胆汁性呕吐、腹胀和深红色直肠出血。腹部触诊引起婴儿的哭闹反应,直肠检查显示检查导管上有血。白细胞计数10.25 × 10^9/L,中性粒细胞占78.6%。由于后勤原因,未进行腹部影像学检查。结合临床表现,诊断为肠旋转不良伴急性中肠扭转及潜在缺血。婴儿接受剖腹手术,发现横结肠和降结肠坏死,切除,升结肠与乙状结肠吻合。坏死结肠的活检符合坏死性小肠结肠炎,显示凝固性坏死和肠囊性肺肿。术后5天进行鼻胃管插管,1周后完全进食,14天出院,术后8个月身体健康。结论坏死性小肠结肠炎应列入足月新生儿胆汁性呕吐、腹胀、便血的鉴别诊断。
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引用次数: 0
Acute appendicitis caused by Enterobius vermicularis in an 8-year-old child: a case report 8岁儿童由蛭状肠虫引起的急性阑尾炎1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-17 DOI: 10.1016/j.epsc.2025.103130
Autumn Bertch , Kimber Boyko , Irina Stout

Background

Enterobius vermicularis is a common pediatric parasite, and appendicitis is a frequent surgical emergency in children; however, it is uncommon for E. vermicularis infection to be the underlying cause.

Case presentation

An 8-year-old girl presented with fever, vomiting, and right lower quadrant abdominal pain. Laboratory evaluation demonstrated leukocytosis (WBC 19.3 × 109/L) with neutrophilia (14.4 × 109/L) and eosinophilia (1.0 × 109/L). Contrast-enhanced CT revealed a dilated, fluid-filled appendix with mucosal thickening and a distal appendicolith, consistent with acute appendicitis. She underwent uncomplicated laparoscopic appendectomy and was discharged the following day, tolerating a regular diet. Histopathology confirmed acute appendicitis with E. vermicularis organisms present. She was prescribed pyrantel pamoate oral suspension 50 mg/mL, 4.8 mL by mouth as a single dose, with a repeat dose scheduled two weeks later, and all household contacts were advised to undergo treatment. Notably, she had a history of recurrent conjunctivitis and vulvovaginitis, prompting referral to ophthalmology to evaluate for rare extraintestinal involvement.

Conclusion

Parasitic infection of the appendix should be suspected in pediatric patients with acute appendicitis and eosinophilia to ensure complete treatment with anti-parasitic therapy.
背景蛭状肠虫是一种常见的儿科寄生虫,阑尾炎是儿童常见的外科急症;然而,它是罕见的蠕虫感染是根本原因。病例表现:一名8岁女孩,表现为发烧、呕吐和右下腹部疼痛。实验室检查显示白细胞增多(WBC 19.3 × 109/L),伴中性粒细胞增多(14.4 × 109/L)和嗜酸性粒细胞增多(1.0 × 109/L)。增强CT显示阑尾扩张,充满液体,粘膜增厚,远端阑尾结石,符合急性阑尾炎。她接受了简单的腹腔镜阑尾切除术,并于第二天出院,饮食正常。组织病理学证实急性阑尾炎存在蛭状芽胞杆菌。给她开了帕马酸吡喃酯口服混悬液50 mg/mL, 4.8 mL单剂量口服,计划两周后重复给药,并建议所有家庭接触者接受治疗。值得注意的是,她有复发性结膜炎和外阴阴道炎的病史,促使她转诊到眼科评估罕见的肠外受累。结论小儿急性阑尾炎伴嗜酸性粒细胞增多患者应怀疑阑尾寄生虫感染,以保证抗寄生虫治疗的完全性。
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引用次数: 0
Robotic spleen-sparing distal pancreatectomy in a pediatric patient with a solid pseudopapillary tumor: A case report 机器人保脾远端胰腺切除术治疗小儿实性假乳头状肿瘤1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-11-01 DOI: 10.1016/j.epsc.2025.103135
Autumn Bennitt , Nitin Chilukuri , Michael Leinwand

Introduction

Solid pseudopapillary tumor (SPT) of the pancreas is a rare neoplasm with low metastatic potential that occurs primarily in the pediatric population and is managed by resection via an open or laparoscopic approach.

Case presentation

An 11-year-old female was found to have an incidental distal pancreatic mass on computed tomography (CT) as part of her evaluation following a motor vehicle accident. She complained of vague intermittent abdominal pain but was otherwise asymptomatic. Her abdominal exam was soft, nontender, and without a palpable mass. Magnetic resonance cholangiopancreatography (MRCP) redemonstrated the mass without local invasion. SPECT (Single Photon Emission Computed Tomography)-CT showed lack of radiotracer uptake and thus was not consistent with splenic tissue. She underwent robotic-assisted spleen-sparing distal pancreatectomy using three 8 mm robotic ports across the mid-abdomen and a left lower quadrant 12 mm accessory port. Dissection was performed using the robotic vessel sealer and monopolar electrocautery. The pancreas was transected with a 12 mm endostapler (blue load). Operative time was 5.25 hours, and estimated blood loss was 10 ml. The patient had an uneventful postoperative course and was discharged home on postoperative day 5. Pathology revealed a solid, 3.6 cm × 2.5 cm, well-encapsulated pseudopapillary tumor with a 0.8 mm margin and no lymphovascular or perineural invasion. Our patient has had no evidence of recurrence on imaging over 3 years.

Conclusion

Robotic spleen-preserving distal pancreatectomy appears to be an effective treatment for children with distal pancreatic tumors.
胰腺实性假乳头状肿瘤(SPT)是一种罕见的肿瘤,具有低转移潜力,主要发生在儿童人群中,通过开放或腹腔镜手术切除治疗。病例介绍:一名11岁的女性在一次机动车事故后,在计算机断层扫描(CT)上发现偶然的胰腺远端肿块。她主诉有隐隐约约的间歇性腹痛,但其他方面无症状。腹部检查柔软,无触痛,未见明显肿块。磁共振胰胆管造影(MRCP)显示肿块无局部浸润。SPECT(单光子发射计算机断层扫描)-CT显示缺乏放射性示踪剂摄取,因此与脾组织不一致。她接受了机器人辅助的保脾远端胰腺切除术,使用三个8毫米的机器人端口穿过中腹部和一个12毫米的左下腹辅助端口。采用机器人血管封口器和单极电灼进行解剖。胰腺用12mm内吻合器(蓝色负载)横切。手术时间5.25小时,估计失血量10ml。患者术后过程平稳,术后第5天出院。病理显示实性,3.6 cm × 2.5 cm,包膜良好的假乳头状肿瘤,边界0.8 mm,无淋巴血管或神经周围浸润。我们的病人在3年多的影像上没有复发的迹象。结论机器人保脾远端胰腺切除术是治疗儿童胰腺远端肿瘤的有效方法。
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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 : 2025-12-01 Epub 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
Hepatic rupture in a neonate with hemophilia B: a case report 新生儿血友病B肝破裂1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-24 DOI: 10.1016/j.epsc.2025.103137
Shuai Liu, Ruo-Yi Wang, Chun-Tian Wang

Introduction

Hepatic rupture in neonates is extremely rare and often fatal. Its association with hemophilia B has scarcely been reported.

Case presentation

A male neonate was born via cesarean section at 39 weeks of gestation with a birth weight of 3.6 kg and Apgar scores of 10 and 10. The prenatal course was without complications and there was no family history of bleeding disorders. At 59 hours of life the patient developed with abrupt abdominal distension, which progressed to hemodynamic shock by 61 hours of life. Bloodwork revealed a hemoglobin level of 4.5 g/dL and severe metabolic acidosis (pH 6.57, lactate 27 mmol/L). A bedside ultrasound combined with abdominal paracentesis confirmed hemoperitoneum. Emergency laparotomy was performed at 63 hours of life. We found a 3-cm fissure on the right hepatic lobe, which was surgically repaired. The patient had a postoperative uneventful recovery and was discharged home in stable condition 2 weeks later. Coagulation function assays confirmed factor 9 activity of 2 %, confirming the diagnosis of hemophilia B. At 6-month of follow-up he has had no recurrent bleeding episodes and remains clinically stable on regular factor replacement therapy.

Conclusions

Neonatal hepatic rupture may be the initial manifestation of an underlying bleeding disorder such as hemophilia B. Neonates with spontaneous hepatic rupture should undergo testing to rule out coagulation disorders.
新生儿肝破裂极为罕见,通常是致命的。它与B型血友病的关系几乎没有报道。病例介绍1例男性新生儿在妊娠39周通过剖宫产出生,出生体重3.6 kg, Apgar评分为10和10。产前无并发症,无出血性疾病家族史。患者在出生后59小时出现突发性腹胀,并在出生后61小时发展为血流动力学休克。血检显示血红蛋白水平为4.5 g/dL,严重代谢性酸中毒(pH 6.57,乳酸27 mmol/L)。床边超声联合腹部穿刺术证实腹腔积血。在出生后63小时进行了紧急剖腹手术。我们发现右肝叶有一个3厘米的裂缝,手术修复。患者术后恢复顺利,2周后出院,病情稳定。凝血功能检测证实因子9活性为2%,确诊为b型血友病。随访6个月,患者未出现复发性出血,经常规因子替代治疗后临床稳定。结论新生儿肝破裂可能是潜在出血性疾病如b型血友病的初始表现。新生儿自发性肝破裂应检查排除凝血功能障碍。
{"title":"Hepatic rupture in a neonate with hemophilia B: a case report","authors":"Shuai Liu,&nbsp;Ruo-Yi Wang,&nbsp;Chun-Tian Wang","doi":"10.1016/j.epsc.2025.103137","DOIUrl":"10.1016/j.epsc.2025.103137","url":null,"abstract":"<div><h3>Introduction</h3><div>Hepatic rupture in neonates is extremely rare and often fatal. Its association with hemophilia B has scarcely been reported.</div></div><div><h3>Case presentation</h3><div>A male neonate was born via cesarean section at 39 weeks of gestation with a birth weight of 3.6 kg and Apgar scores of 10 and 10. The prenatal course was without complications and there was no family history of bleeding disorders. At 59 hours of life the patient developed with abrupt abdominal distension, which progressed to hemodynamic shock by 61 hours of life. Bloodwork revealed a hemoglobin level of 4.5 g/dL and severe metabolic acidosis (pH 6.57, lactate 27 mmol/L). A bedside ultrasound combined with abdominal paracentesis confirmed hemoperitoneum. Emergency laparotomy was performed at 63 hours of life. We found a 3-cm fissure on the right hepatic lobe, which was surgically repaired. The patient had a postoperative uneventful recovery and was discharged home in stable condition 2 weeks later. Coagulation function assays confirmed factor 9 activity of 2 %, confirming the diagnosis of hemophilia B. At 6-month of follow-up he has had no recurrent bleeding episodes and remains clinically stable on regular factor replacement therapy.</div></div><div><h3>Conclusions</h3><div>Neonatal hepatic rupture may be the initial manifestation of an underlying bleeding disorder such as hemophilia B. Neonates with spontaneous hepatic rupture should undergo testing to rule out coagulation disorders.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"123 ","pages":"Article 103137"},"PeriodicalIF":0.2,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145526285","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
Precocious puberty in a 20-month-old girl secondary to an ovarian juvenile granulosa cell tumor: a case report 继发于卵巢幼年颗粒细胞瘤的20月龄女童性早熟1例报告
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-15 DOI: 10.1016/j.epsc.2025.103133
Syrine Laribi , Myriam Ben Fredj , Sabrine Ben Youssef , Afef Toumi , Sawsen Chakroun , Lassaad Sahnoun

Background

Juvenile granulosa cell tumors (JGCTs) are extremely rare ovarian neoplasms, accounting for a small fraction of pediatric ovarian tumors. Their hormonal activity often manifests as precocious puberty, which may be the first sign of the disease.

Case presentation

We report the case of a 20-month-old girl presenting with persistent vaginal bleeding for one month. She was born at term by cesarean section after an uncomplicated pregnancy, with an unremarkable medical history. Her mother reported breast development starting six months earlier. On examination, the patient appeared in good general condition, with Tanner stage II breast and pubic hair development and no clitoromegaly. Abdominopelvic ultrasonography revealed a large mixed solid–cystic right ovarian mass measuring 68 × 60 mm with intralesional fluid and uterine enlargement. Pelvic MRI confirmed an 80-mm right ovarian mass with both solid and cystic components and no locoregional invasion or abnormal vascular flow. Laboratory tests showed elevated estradiol (68 pg/mL; normal <62 pg/mL), lactate dehydrogenase (269 U/L; normal <200 U/L), and inhibin B (985 pg/mL; normal <200 pg/mL), with normal human chorionic gonadotropin, progesterone, and alpha-fetoprotein levels. The patient underwent right salpingo-oophorectomy to ensure complete excision and to avoid potential tumor cell spillage or future complications., and histopathological examination confirmed a juvenile granulosa cell tumor. Postoperative follow-up over five months showed complete resolution of symptoms and no recurrence.

Conclusion

Ovarian juvenile granulosa cell tumors must be promptly ruled out in females who present precocious puberty.
幼年颗粒细胞瘤(jgct)是一种极为罕见的卵巢肿瘤,仅占儿童卵巢肿瘤的一小部分。他们的荷尔蒙活动通常表现为性早熟,这可能是这种疾病的第一个征兆。我们报告的情况下,20个月大的女孩表现为持续阴道出血一个月。她是在一次没有并发症的怀孕后通过剖宫产足月出生的,没有什么不寻常的病史。她的母亲报告乳房发育开始于六个月前。经检查,患者总体情况良好,Tanner II期乳房和阴毛发育,无阴蒂肿大。腹部盆腔超声示右侧卵巢68 × 60 mm大混合实性囊性肿块,病灶内积液,子宫增大。盆腔MRI证实右侧卵巢肿块,直径80毫米,有实性和囊性成分,未见局部侵犯或血管流动异常。实验室检查显示雌二醇(68 pg/mL;正常<;62 pg/mL)、乳酸脱氢酶(269 U/L;正常<;200 U/L)和抑制素B (985 pg/mL;正常<;200 pg/mL)升高,绒毛膜促性腺激素、黄体酮和甲胎蛋白水平正常。患者行右侧输卵管卵巢切除术,以确保完全切除,并避免潜在的肿瘤细胞溢出或未来的并发症。组织病理学检查证实为幼年颗粒细胞瘤。术后随访5个月,症状完全缓解,无复发。结论出现性早熟的女性应及时排除卵巢幼年性颗粒细胞瘤。
{"title":"Precocious puberty in a 20-month-old girl secondary to an ovarian juvenile granulosa cell tumor: a case report","authors":"Syrine Laribi ,&nbsp;Myriam Ben Fredj ,&nbsp;Sabrine Ben Youssef ,&nbsp;Afef Toumi ,&nbsp;Sawsen Chakroun ,&nbsp;Lassaad Sahnoun","doi":"10.1016/j.epsc.2025.103133","DOIUrl":"10.1016/j.epsc.2025.103133","url":null,"abstract":"<div><h3>Background</h3><div>Juvenile granulosa cell tumors (JGCTs) are extremely rare ovarian neoplasms, accounting for a small fraction of pediatric ovarian tumors. Their hormonal activity often manifests as precocious puberty, which may be the first sign of the disease.</div></div><div><h3>Case presentation</h3><div>We report the case of a 20-month-old girl presenting with persistent vaginal bleeding for one month. She was born at term by cesarean section after an uncomplicated pregnancy, with an unremarkable medical history. Her mother reported breast development starting six months earlier. On examination, the patient appeared in good general condition, with Tanner stage II breast and pubic hair development and no clitoromegaly. Abdominopelvic ultrasonography revealed a large mixed solid–cystic right ovarian mass measuring 68 × 60 mm with intralesional fluid and uterine enlargement. Pelvic MRI confirmed an 80-mm right ovarian mass with both solid and cystic components and no locoregional invasion or abnormal vascular flow. Laboratory tests showed elevated estradiol (68 pg/mL; normal &lt;62 pg/mL), lactate dehydrogenase (269 U/L; normal &lt;200 U/L), and inhibin B (985 pg/mL; normal &lt;200 pg/mL), with normal human chorionic gonadotropin, progesterone, and alpha-fetoprotein levels. The patient underwent right salpingo-oophorectomy to ensure complete excision and to avoid potential tumor cell spillage or future complications., and histopathological examination confirmed a juvenile granulosa cell tumor. Postoperative follow-up over five months showed complete resolution of symptoms and no recurrence.</div></div><div><h3>Conclusion</h3><div>Ovarian juvenile granulosa cell tumors must be promptly ruled out in females who present precocious puberty.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"123 ","pages":"Article 103133"},"PeriodicalIF":0.2,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145324641","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
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 : 2025-12-01 Epub 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。
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引用次数: 0
Syndromic and non-syndromic Hirschsprung disease with sensorineural hearing loss: A case series 综合征型和非综合征型巨结肠病伴感音神经性听力损失:一个病例系列
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-21 DOI: 10.1016/j.epsc.2025.103125
Fareeda Galley , Ebenezer Akomea-Agyin , Robert Sagoe , Anthony Davor , Regina Darko-Asante

Introduction

Neurocristopathies (NCPs) arise from abnormalities in the formation and migration of neural crest cells (NCCs) during fetal development. These lead to clinical manifestations in various body parts where NCCs contribute to development. They include the failure of NCCs to migrate completely into the distal colon, leading to Hirschsprung disease; failure to contribute to inner ear formation, resulting in sensorineural hearing loss; and failure to contribute to pigmentation of various parts, including the iris. These abnormalities can present in combinations found in both syndromic and non-syndromic children.

Cases presentation

We report on two cases that exhibit different combinations of the clinical manifestations of NCPs. The first is a 2-year-old female delivered at term with delayed passage of meconium but presented at 3 months of life with constipation and abdominal distention. She was managed with rectal washouts, and a rectal biopsy confirmed Hirschsprung disease. She had a trans-endorectal pull-through done at 2 years for a short segment Hirschsprung disease. The mother later reported some developmental delays, which led to the identification of the child's inability to respond to her name or startle to loud noise. An ear assessment revealed bilateral sensorineural hearing loss, believed to be congenital in origin. This was managed with a left-sided cochlear implant, and the child is currently undergoing rehabilitation and has yet to develop her speech. The second is an 18-month-old male believed to be the first reported case of Waardenburg-Shah in Ghana. The child was delivered at term with normal passage of meconium but developed constipation after introduction of family feeds at 6 months and was managed at home by parents with herbal enemas till 16 months of age, when enemas could not relieve constipation. The mother presented the child to the hospital on account of chronic constipation and inability to gain weight. Examination revealed a malnourished child with blue colored iris, abdominal distention, and digital rectal examination of an empty rectum with a gush of feces after removing the finger. Rectal washouts were done, and a rectal biopsy confirmed Hirschsprung disease. A hearing assessment revealed bilateral sensorineural hearing impairment, which was managed with hearing aids. The child is still undergoing nutritional rehabilitation towards definitive surgery.

Conclusion

Patients with Hirschsprung disease should undergo early hearing assessment to rule out Hirschsprung-associated sensorineural hearing loss. The combination of Hirschsprung disease and sensorineural hearing loss may or may not be syndromic.
神经嵴病变(ncp)是由胎儿发育过程中神经嵴细胞(ncc)的形成和迁移异常引起的。这些导致临床表现在不同的身体部位,ncc有助于发展。它们包括ncc不能完全迁移到远端结肠,导致巨结肠疾病;不能促进内耳形成,导致感音神经性听力损失;也不能导致包括虹膜在内的各个部位的色素沉着。这些异常可出现在综合征和非综合征儿童的组合中。我们报告了两例表现出不同临床表现组合的ncp病例。第一例为两岁女婴,足月分娩时胎便排出延迟,但3个月时出现便秘和腹胀。她接受直肠冲洗,直肠活检证实有先天性巨结肠。她在2岁时因短段先天性巨结肠病进行了经直肠内拉通手术。这位母亲后来报告说,孩子有一些发育迟缓,这导致孩子无法对她的名字做出反应,也无法对大声的噪音感到惊吓。耳朵评估显示双侧感音神经性听力损失,相信是先天性的起源。这是通过左侧人工耳蜗来解决的,孩子目前正在接受康复治疗,还没有发展她的语言能力。第二例是一名18个月大的男性,据信是加纳报告的首例Waardenburg-Shah病例。患儿足月分娩,胎便正常通过,但6个月时开始家庭喂养后出现便秘,并由父母在家进行草药灌肠治疗,直到16个月时灌肠无法缓解便秘。这位母亲把孩子送到医院,原因是慢性便秘和无法增加体重。检查发现一个营养不良的孩子,虹膜呈蓝色,腹胀,直肠指检显示直肠空,取下手指后便涌出。直肠冲洗和直肠活检证实了先天性巨结肠疾病。听力评估显示双侧感音神经性听力障碍,使用助听器进行治疗。这名儿童仍在接受营养康复治疗,准备接受最终手术。结论先天性巨结肠病患者应尽早进行听力评估,排除巨结肠相关感音神经性听力损失。巨结肠疾病和感音神经性听力损失的结合可能是也可能不是综合征。
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引用次数: 0
Fetal inguinoscrotal hernia: A case series 胎儿腹股沟-阴囊疝:一个病例系列
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-09 DOI: 10.1016/j.epsc.2025.103122
Aliya Y. Ortaaslan , Françoise Rypens , Dickens Saint-Vil

Introduction

Inguinoscrotal hernias (ISH) are common in neonates but rarely seen in the fetal period, with only 21 reported cases to date. We present two cases of fetal ISH diagnosed antenatally, each initially misidentified.

Cases presentation

A 25-year-old woman (G6P3A2) was referred for a solid scrotal mass detected at 34 weeks of gestation and suspected to be teratoma. Repeat Ultrasound (US) at 35 weeks, diagnosed an isolated right-sided inguinoscrotal hernia containing peristaltic bowel loops and mesenteric vascularization without any other anomaly. Patient was born in a peripheral hospital before ultimately undergoing inguinal herniorrhaphy at 10 days of life.
In a 27-year-old woman (G1P0), US at 22 weeks revealed bilateral nephromegaly. Biweekly follow-up was performed. At 33 weeks, follow-up US detected a distended rectum containing heterogeneous material. Fetal MRI confirmed bilateral nephromegaly, normal rectal content, and diagnosed a right inguinoscrotal hernia with collapsed bowel loops in the scrotum and an associated left hydrocele. The infant was delivered by cesarean section at 36 weeks at our tertiary pediatric hospital. The infant underwent bilateral nephrectomy and simultaneous bilateral inguinoscrotal hernia repair at 2 months of age.

Conclusion

Fetal inguinoscrotal hernia is rare but should be considered in the differential for a fetal scrotal or perineal mass.
腹股沟阴囊疝(ISH)在新生儿中很常见,但在胎儿期很少见,迄今仅有21例报告。我们提出了两例胎儿ISH诊断产前,每个最初被误诊。病例介绍一名25岁的女性(G6P3A2)在妊娠34周时发现实心阴囊肿块,怀疑是畸胎瘤。35周时复查超声(US),诊断为孤立性右侧腹股沟阴囊疝,包含肠蠕动环和肠系膜血管,无其他异常。患者出生在外围医院,最终在出生后10天进行腹股沟疝修补术。27岁女性(G1P0), 22周超声显示双侧肾肥大。每两周随访一次。在33周的随访中,超声检查发现直肠膨胀含有异质物质。胎儿MRI证实双侧肾肿大,直肠内容物正常,并诊断为右侧腹股沟阴囊疝伴阴囊肠袢塌陷,伴有左侧鞘膜积液。该婴儿于36周时在我院第三儿科医院剖宫产。婴儿于2月龄时行双侧肾切除术及双侧腹股沟阴囊疝修补术。结论胎儿腹股沟-阴囊疝是罕见的,但在鉴别胎儿阴囊或会阴肿块时应予以考虑。
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引用次数: 0
Juvenile granulosa cell tumor in a teenager: a case report 青少年颗粒细胞瘤1例
IF 0.2 Q4 PEDIATRICS Pub Date : 2025-12-01 Epub Date: 2025-10-15 DOI: 10.1016/j.epsc.2025.103131
Ian Cummings-Ruiz , Nicole A. Martínez-Martínez , Kenneth Avilés-Fernández , Yaiel Rodríguez-Avilés , Victor Ortiz-Justiniano

Introduction

Juvenile granulosa cell tumors (JGCTs) are rare ovarian sex cord–stromal neoplasms that primarily affect young females and are often detected early due to hyperestrogenic manifestations.

Case presentation

A 17-year-old female with a history of seizures and hypothyroidism presented with a 5-month history of intermittent abdominal pain, nausea, anorexia, and constant lower-back discomfort associated with progressive abdominal distention. Physical examination revealed marked abdominal distention with striae. Laboratory studies demonstrated elevated lactate dehydrogenase (LDH) and alkaline phosphatase (ALP), as well as beta-human chorionic gonadotropin (β-hCG). An abdominopelvic computed tomography (CT) scan revealed a 35.7 × 26 × 19.1 cm predominantly cystic adnexal mass with eccentric solid components and no evidence of ascites or lymphadenopathy. Tumor markers, including alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA), were negative. Surgical exploration via periumbilical incision revealed a large cystic mass arising from the left adnexa. Controlled decompression with a Veress needle allowed safe mobilization of the tumor, yielding 4.8 L of cystic fluid that was sent for cytological evaluation. The mass was then excised from the abdominal cavity, and a left salpingo-oophorectomy was performed. Pathology confirmed a JGCT with an intact capsule, classified as stage IA according to the Federation of Gynecology and Obstetrics (FIGO) criteria. Postoperatively, the patient recovered well and remained asymptomatic, with no recurrence during a 3-year follow-up period.

Conclusion

JGCT should be considered as a differential diagnosis of large ovarian tumors in adolescents, even in the absence of hormonal manifestations.
青少年颗粒细胞瘤(jgct)是一种罕见的卵巢性索间质肿瘤,主要发生在年轻女性身上,通常因雌激素分泌过多而早期发现。17岁女性,有癫痫发作和甲状腺功能减退病史,5个月来间歇性腹痛、恶心、厌食,持续腰背部不适伴进行性腹胀。体格检查显示明显腹胀伴条纹。实验室研究表明乳酸脱氢酶(LDH)和碱性磷酸酶(ALP)以及β-人绒毛膜促性腺激素(β-hCG)升高。腹部计算机断层扫描(CT)显示一个35.7 × 26 × 19.1厘米的主要囊性附件肿块,有偏心实体成分,无腹水或淋巴结病的证据。肿瘤标志物,包括甲胎蛋白(AFP)和癌胚抗原(CEA)均为阴性。经脐周切口探查发现左附件有一个巨大的囊性肿块。用Veress针控制减压,使肿瘤安全活动,产生4.8 L囊性液,用于细胞学评估。然后从腹腔切除肿块,并进行左侧输卵管卵巢切除术。病理证实为JGCT,包膜完整,根据妇产科学联合会(FIGO)标准分类为IA期。术后患者恢复良好,无症状,随访3年无复发。结论即使没有激素表现,jgct也可作为青少年卵巢大肿瘤的鉴别诊断手段。
{"title":"Juvenile granulosa cell tumor in a teenager: a case report","authors":"Ian Cummings-Ruiz ,&nbsp;Nicole A. Martínez-Martínez ,&nbsp;Kenneth Avilés-Fernández ,&nbsp;Yaiel Rodríguez-Avilés ,&nbsp;Victor Ortiz-Justiniano","doi":"10.1016/j.epsc.2025.103131","DOIUrl":"10.1016/j.epsc.2025.103131","url":null,"abstract":"<div><h3>Introduction</h3><div>Juvenile granulosa cell tumors (JGCTs) are rare ovarian sex cord–stromal neoplasms that primarily affect young females and are often detected early due to hyperestrogenic manifestations.</div></div><div><h3>Case presentation</h3><div>A 17-year-old female with a history of seizures and hypothyroidism presented with a 5-month history of intermittent abdominal pain, nausea, anorexia, and constant lower-back discomfort associated with progressive abdominal distention. Physical examination revealed marked abdominal distention with striae. Laboratory studies demonstrated elevated lactate dehydrogenase (LDH) and alkaline phosphatase (ALP), as well as beta-human chorionic gonadotropin (β-hCG). An abdominopelvic computed tomography (CT) scan revealed a 35.7 × 26 × 19.1 cm predominantly cystic adnexal mass with eccentric solid components and no evidence of ascites or lymphadenopathy. Tumor markers, including alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA), were negative. Surgical exploration via periumbilical incision revealed a large cystic mass arising from the left adnexa. Controlled decompression with a Veress needle allowed safe mobilization of the tumor, yielding 4.8 L of cystic fluid that was sent for cytological evaluation. The mass was then excised from the abdominal cavity, and a left salpingo-oophorectomy was performed. Pathology confirmed a JGCT with an intact capsule, classified as stage IA according to the Federation of Gynecology and Obstetrics (FIGO) criteria. Postoperatively, the patient recovered well and remained asymptomatic, with no recurrence during a 3-year follow-up period.</div></div><div><h3>Conclusion</h3><div>JGCT should be considered as a differential diagnosis of large ovarian tumors in adolescents, even in the absence of hormonal manifestations.</div></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"123 ","pages":"Article 103131"},"PeriodicalIF":0.2,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145323968","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
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Journal of Pediatric Surgery Case Reports
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