• 文章类型: Case Reports
    严重的胃肠道出血,由于肝假性动脉瘤(HAPA)对十二指肠的长期压迫,是极其罕见的情况。事实上,当假性动脉瘤直径较小时,诊断可能特别具有挑战性。因此及时有效的诊断和治疗具有重要意义,在这种情况下,内窥镜检查,结合血管内治疗,可以提供一种有效的方法,特别是因为它消除了手术的需要,同时产生有利的结果。
    一名75岁的老人因呕血和黑便出现在医院急诊科。尽管保守治疗,如“酸抑制,流体再供应,止血和输血“,未发现显著改善.急诊胃镜检查随后发现十二指肠球部溃疡(图1),血栓性头部暴露,表面活跃出血。此外,腹部计算机断层扫描(图2)未显示明显的HAPA表现.内镜止血不成功后,我们进行了血管造影(图3),在肝总动脉近端分支血管远端发现了直径为5.56mm的假瘤样扩张.弹簧圈栓塞后(图4),病人的病情好转,出院了,6个月后的随访显示没有复发或并发症的迹象。
    由小的肝假性动脉瘤引起的十二指肠溃疡出血非常罕见,内窥镜干预对此类病例有效。
    UNASSIGNED: Significant gastrointestinal hemorrhages, resulting from long-term compression of the duodenum by a hepatic pseudoaneurysm (HAPA), is an extremely rare condition. In fact, when the pseudoaneurysm is small in diameter, diagnosis can be particularly challenging. Timely and effective diagnosis and treatment is therefore of great significance, and in this case, endoscopy, combined with intravascular therapy, can provide an effective approach, especially since it removes the need for surgery while yielding favorable outcomes.
    UNASSIGNED: A 75-year-old old man presented to the hospital\'s emergency department with hematemesis and black stool. Despite conservative treatments such as \"acid suppression, fluid resupply, hemostasis and blood transfusion\", no significant improvement was noted. Emergency gastroscopy subsequently revealed an ulcer in the duodenal bulb (Figure 1), with an exposed thrombotic head and active bleeding on the surface. In addition, abdominal computed tomography (Figure 2) showed no obvious HAPA manifestations. After unsuccessful endoscopic hemostasis, angiography was performed (Figure 3) and a pseudotumor-like dilatation measuring 5.56 mm in diameter was found at the distal end of the proximal branch vessel of the common hepatic artery. Following spring coil embolization (Figure 4), the patient\'s condition improved and he was discharged from the hospital, with a follow-up after six months showing no signs of recurrence or complications.
    UNASSIGNED: Duodenal ulcer hemorrhages due to small hepatic pseudoaneurysms are very rare, with endoscopic intervention being effective for such cases.
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  • 文章类型: Case Reports
    背景技术超镜夹(OTSC)系统是一类内窥镜夹,其旨在与常规的透镜夹相比提供改进的强度和组织捕获。这些夹子在管理许多胃肠道疾病方面通常是安全有效的,总体不良事件发生率较低。尽管OTSC多年来一直用于治疗消化道出血和肠穿孔,它通常被降级为二线治疗,直到最近才成为胃肠道出血的一线止血治疗.这里的案例报告,我们介绍了导致医源性胃十二指肠动脉(GDA)结扎的OTSC独特不良事件.一名71岁的男子表现为6个月的上腹部腹痛和2周的呕血,最终被诊断为十二指肠溃疡出血.他接受了多次内镜干预,试图控制十二指肠溃疡出血,包括将OTSC放置在可见的容器上。OTSC放置后不久,他因复发性便血而低血压,并咨询了介入放射学对出血进行血管内处理.血管造影显示OTSC已从十二指肠腔部署到GDA的中部,有效地连接GDA,直接血管损伤导致出血.这种出血最终通过线圈栓塞控制。然而,OTSC对GDA中段的医源性结扎使血管内介入术控制出血显著复杂化.结论随着OTSC设备在内窥镜检查套件中的使用越来越普遍,重要的是要分享在临床环境中可能遇到的潜在陷阱,这些陷阱不仅影响内窥镜医师和患者,但其他专业也是如此。
    BACKGROUND The Over-the-Scope-Clip (OTSC) System is a class of endoscopic clips intended to provide improved strength and tissue capture compared to conventional through-the-scope clips. These clips are generally safe and effective in managing many gastrointestinal conditions, with a low overall adverse event rate. Although the OTSC has been used to treat gastrointestinal bleeding and bowel perforations for many years, it often is relegated to second-line therapy and has only recently become a first-line hemostatic therapy for gastrointestinal bleeding. CASE REPORT Here, we present a unique adverse event of the OTSC causing iatrogenic ligation of the gastroduodenal artery (GDA). A 71-year-old man presented with 6 months of epigastric abdominal pain and 2 weeks of hematemesis, and was ultimately diagnosed with a bleeding duodenal ulcer. He underwent multiple endoscopic interventions to attempt to control the duodenal ulcer bleeding, including placement of the OTSC on a visible vessel. Soon after OTSC placement, he became hypotensive with recurrent hematochezia, and Interventional Radiology was consulted for endovascular management of the bleeding. Angiography showed the OTSC had been deployed across the midportion of the GDA from the duodenal lumen, effectively ligating the GDA, causing bleeding due to direct vascular injury. This bleeding was ultimately controlled with coil embolization. However, this iatrogenic ligation of the midportion of the GDA by the OTSC significantly complicated endovascular intervention to control the bleeding. CONCLUSIONS As the OTSC device becomes more commonly used in the endoscopy suite, it is important to share potential pitfalls that may be encountered in the clinical setting that impact not only endoscopists and patients, but other specialties as well.
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  • 文章类型: Case Reports
    获得性免疫缺陷综合征(AIDS)患者消化道出血的原因复杂。在这里,我们介绍了一例由巨细胞病毒引起的AIDS患者十二指肠溃疡引起的危及生命的胃肠道出血的罕见病例。
    一名31岁的男性艾滋病患者于2023年7月18日入院,主诉腹痛38天,间歇性便血12天。在他住院期间,胃肠内镜将消化道出血归因于巨大的十二指肠溃疡。此外,巨细胞病毒(CMV)感染通过宏基因组下一代测序(mNG)证实为溃疡的原因,苏木精-伊红(HE)染色,和活检组织的免疫组织化学(IHC)染色。患者的消化道出血通过介入栓塞停止。经过4周的抗CMV治疗,巨大的十二指肠溃疡被治愈了.
    对于患有消化道出血的艾滋病患者,应考虑CMV引起的胃肠道溃疡。综合方法(mNG,活检组织的HE染色和IHC染色)有利于确诊。除了抗CMV治疗,介入栓塞是止血的一种选择。
    UNASSIGNED: The reasons for gastrointestinal bleeding among patients with acquired immune deficiency syndrome (AIDS) were complex. Here we present an unusual case of life-threatening gastrointestinal bleeding caused by a cytomegalovirus-induced duodenal ulcer in an AIDS patient.
    UNASSIGNED: A 31-year-old male with AIDS was admitted on July 18, 2023, complaining of abdominal pain for 38 days and intermittent hematochezia for 12 days. During his hospitalization, gastrointestinal endoscopy attributed gastrointestinal bleeding to a giant duodenal ulcer. Furthermore, cytomegalovirus(CMV) infection was confirmed as the reason for the ulcer through metagenomic next-generation sequencing (mNGs), hematoxylin-eosin(HE) staining, and immunohistochemistry (IHC) staining for the biopsy tissue. The patient\'s gastrointestinal bleeding was stopped by interventional embolization. Following a 4-week course of anti-CMV treatment, the giant duodenal ulcer was cured.
    UNASSIGNED: For AIDS patients with gastrointestinal bleeding, the CMV-induced gastrointestinal ulcer should be considered. Comprehensive mothods (mNGs, HE staining and IHC staining for biopsy tissue) were benefit for confirmed diagnosis. Beside anti-CMV treatment, the interventional embolization is a choice for hemostasis.
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  • 文章类型: Case Reports
    免疫球蛋白G4(IgG4)相关疾病是一种病因不明的全身性炎症,其特征是血清IgG4和受影响组织中IgG4阳性细胞数量增加。通常涉及的位置之一是胰腺;这种情况被称为1型自身免疫性胰腺炎(AIP)。1型AIP,显示胰腺内胆管狭窄,由于胆道造影结果和临床表现相似,可误诊为恶性肿瘤。在极少数情况下并发球后十二指肠溃疡,区分1型AIP和恶性肿瘤更加困难。一名81岁的男性被转诊到我们医院治疗胰头肿块和阻塞性黄疸。血清学和放射学发现与1型AIP和恶性肿瘤一致。胃十二指肠镜检查显示球后十二指肠溃疡具有内镜特征,可引起恶性十二指肠浸润。尽管活检对恶性细胞呈阴性,随后的病变出血提示恶性肿瘤的进展,导致手术切除。胰十二指肠切除术及病理检查提示1型AIP。同时,提示IgG4相关疾病累及溃疡性病变.据我们所知,这是首例1型AIP并发球后十二指肠溃疡的病例,被误诊为恶性肿瘤,并认为是与1型AIP相关的IgG4相关的胃肠道疾病。
    Immunoglobulin G4 (IgG4)-related diseaseis a systemic inflammatory condition of unknown etiology characterized by increases in serum IgG4 and in the number of IgG4-positive cells in affected tissues. One of the commonly involved locations is the pancreas; this condition is known as type 1 autoimmune pancreatitis (AIP). Type 1 AIP, which shows a biliary stricture in the intrapancreatic bile duct, can be misdiagnosed as a malignancy due to similar cholangiography findings and clinical presentation. In rare cases complicated by post-bulbar duodenal ulcers, differentiating between type 1 AIP and malignancies is even more difficult. An 81-year-old male was referred to our hospital for the treatment of a pancreatic head mass and obstructive jaundice. Serological and radiological findings were consistent with both type 1 AIP and a malignancy. Gastroduodenoscopy revealed a post-bulbar duodenal ulcer with endoscopic features that evoked malignant duodenal invasion. Although biopsies were negative for malignant cells, subsequent bleeding from the lesion suggested the progression of malignancy, which led to surgical resection. Pancreatoduodenectomy and pathological examination indicated that type 1 AIP was present. Simultaneously, the involvement of IgG4-related disease in the ulcerative lesion was suggested. To our knowledge, this is the first reported case of type 1 AIP complicated by post-bulbar duodenal ulcers, which was misdiagnosed as malignancy and considered an IgG4-related gastrointestinal disease associated with type 1 AIP.
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  • 文章类型: Case Reports
    背景:腹膜后血肿在接受肾切除术的患者中相对常见。在这里,我们报告了一例不寻常的病例,包括一个巨大的腹膜后血肿和随后的十二指肠溃疡性出血在根治性肾切除术后。
    方法:一名77岁女性因腰痛入院,她患有严重的右肾积水和尿路感染。
    方法:患者被确诊为高级别尿路上皮癌。
    方法:保守治疗无效后,进行了右根治性肾切除术和输尿管残端切除术。患者接受质子泵抑制剂以防止应激性溃疡形成和出血。手术后的第一天,她的胃肠(GI)内镜检查结果正常.手术后的第二天,腹部计算机断层扫描显示腹膜后血肿。值得注意的是,术后14天,发生大量胃肠道出血,胃肠道内窥镜检查发现球部和十二指肠降部溃疡几乎穿孔。
    结果:患者在手术后第15天死亡。
    结论:肾切除术患者腹膜后血肿后可能发生十二指肠溃疡和出血。及时干预可以预防十二指肠溃疡和并发症,因此可能是一个有希望的拯救生命的代祷。
    BACKGROUND: Retroperitoneal hematomas are relatively common in patients undergoing nephrectomy. Herein, we report an unusual case involving a giant retroperitoneal hematoma and subsequent duodenal ulcerative bleeding following a radical nephrectomy.
    METHODS: A 77-year-old woman was admitted to our hospital for lower back pain, and she had severe right hydronephrosis and a urinary tract infection.
    METHODS: The patient was diagnosed and confirmed as high-grade urothelial carcinoma.
    METHODS: After ineffective conservative treatments, a right radical nephrectomy and ureteral stump resection were performed. The patient received proton pump inhibitors to prevent stress ulcer formation and bleeding. On the first day post-surgery, she had normal gastrointestinal (GI) endoscopy findings. On the second day post-surgery, abdominal computed tomography revealed a retroperitoneal hematoma. Notably, 14 days post-surgery, massive GI bleeding occurred, and GI endoscopy identified an almost perforated ulcer in the bulbar and descending duodenum.
    RESULTS: The patient died on day 15 after surgery.
    CONCLUSIONS: Duodenal ulceration and bleeding might occur following a retroperitoneal hematoma in patients treated with nephrectomy. Timely intervention may prevent duodenal ulcers and complications, and thus could be a promising life-saving intercession.
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  • 文章类型: Case Reports
    我们报告了根据特征性溃疡发现诊断为与SLCO2A1基因(CEAS)相关的慢性肠病的双胞胎病例。这需要8年的时间来诊断。两个双胞胎都有相似的症状,包括贫血和生长障碍,但由于轻度症状被认为与心理病因一致,最初未对胃肠道进行评估。长子的内窥镜检查结果显示,螺旋溃疡疤痕和假憩室形成,十二指肠中没有幽门螺杆菌感染或嗜酸性粒细胞浸润。由于这对双胞胎同时出现不明原因的溃疡,并且第一个出生的孩子患有螺旋状溃疡,CEAS被怀疑。遗传分析和尿液中高水平的前列腺素E主要尿代谢产物导致CEAS的明确诊断。
    We report the case of twins diagnosed with chronic enteropathy associated with the SLCO2A1 gene (CEAS) based on characteristic ulcer findings, which required 8 years to diagnose. Both twins had similar symptoms, including anemia and growth failure but the gastrointestinal tract was not evaluated initially because of mild symptoms that were considered consistent with psychological etiology. The endoscopic findings of the firstborn child showed spiral ulcer scars and pseudodiverticulum formation without Helicobacter pylori infection or eosinophilic infiltration in the duodenum. Since the twins presented with ulcers of an unknown cause simultaneously and the first-born child had a spiral ulcer, CEAS was suspected. Genetic analysis and high levels of prostaglandin E major urinary metabolites in the urine led to a definitive diagnosis of CEAS.
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  • 文章类型: Case Reports
    假性动脉瘤并不常见,但其破裂和出血可导致严重的并发症并致命。我们在这里介绍一例70多岁的男子,他因持续的胃肠道出血被转移到我们医院。在他入院前一个月,他因左膝骨折接受了手术。内镜检查发现十二指肠溃疡血管搏动,突然破裂并导致大量出血。立即进行内镜止血,出血减少。考虑到假性动脉瘤可能发生的高再出血率,患者接受了介入放射学检查,最终诊断为源自胃十二指肠动脉(GDA)的假性动脉瘤;成功栓塞.检测显示患者有幽门螺杆菌感染。我们假设幽门螺杆菌感染导致十二指肠球部溃疡的发生,患者的左膝骨折和一个月前的手术导致了假性动脉瘤的倾向。
    Pseudoaneurysms are uncommon but their rupture and bleeding can lead to serious complications and be fatal. We present here a case of a man in his late 70s who was transferred to our hospital with persistent gastrointestinal bleeding. One month prior to his admission, he had undergone surgery for a fracture to his left knee. Endoscopic examination found pulsating blood vessels on a duodenal ulcer, which suddenly ruptured and caused significant bleeding. Immediate endoscopic haemostasis was administered and the bleeding decreased. Considering the high rate of rebleeding that may occur with a pseudoaneurysm, the patient underwent interventional radiology that culminated in a diagnosis of a pseudoaneurysm originating from gastroduodenal artery (GDA); successful embolization was achieved. Tests showed that the patient had Helicobacter pylori infection. We hypothesised that the H. pylori infection had led to the occurrence of the duodenal bulb ulcer, and the patient\'s left knee fracture and surgery a month previously had contributed to this predisposition for a pseudoaneurysm.
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  • 文章类型: Journal Article
    某些病原体的感染与癌症发展有关。我们进行了一项病例队列研究,对约2500例食管,胃癌和十二指肠癌,以及胃和十二指肠溃疡,以及在中国加多利生物库研究中随机选择的约2000名受试者的亚队列>50万成年人。我们使用基于珠的多重血清学测定来测量基线血浆样品中针对19种病原体(总共43种抗原)的抗体。使用使用Prentice伪部分可能性拟合的Cox回归评估病原体和抗原特异性抗体与位点特异性癌症和溃疡风险之间的关联。血清阳性率因不同病原体而异,亚组中丙型肝炎病毒(HCV)的0.7%至EB病毒(EBV)的99.8%。与相应病原体血清阴性的参与者相比,幽门螺杆菌血清阳性与非贲门(校正后风险比[HR]2.73[95%CI:2.09-3.58])和贲门(1.67[1.18-2.38])胃癌和十二指肠溃疡(2.71[1.79-4.08])的高风险相关。HCV与十二指肠癌的高风险相关(6.23[1.52-25.62]),乙型肝炎病毒与十二指肠溃疡的高风险相关(1.46[1.04-2.05])。一些疱疹病毒和人乳头瘤病毒的抗体与胃肠道癌症和溃疡的风险有一些关联,但应谨慎解释。这项针对多种病原体的首次研究表明,多种病原体与胃肠道癌症和溃疡的风险有关。这将为将来研究感染在这些疾病的病因中的作用提供信息。
    Infection by certain pathogens is associated with cancer development. We conducted a case-cohort study of ~2500 incident cases of esophageal, gastric and duodenal cancer, and gastric and duodenal ulcer and a randomly selected subcohort of ~2000 individuals within the China Kadoorie Biobank study of >0.5 million adults. We used a bead-based multiplex serology assay to measure antibodies against 19 pathogens (total 43 antigens) in baseline plasma samples. Associations between pathogens and antigen-specific antibodies with risks of site-specific cancers and ulcers were assessed using Cox regression fitted using the Prentice pseudo-partial likelihood. Seroprevalence varied for different pathogens, from 0.7% for Hepatitis C virus (HCV) to 99.8% for Epstein-Barr virus (EBV) in the subcohort. Compared to participants seronegative for the corresponding pathogen, Helicobacter pylori seropositivity was associated with a higher risk of non-cardia (adjusted hazard ratio [HR] 2.73 [95% CI: 2.09-3.58]) and cardia (1.67 [1.18-2.38]) gastric cancer and duodenal ulcer (2.71 [1.79-4.08]). HCV was associated with a higher risk of duodenal cancer (6.23 [1.52-25.62]) and Hepatitis B virus was associated with higher risk of duodenal ulcer (1.46 [1.04-2.05]). There were some associations of antibodies again some herpesviruses and human papillomaviruses with risks of gastrointestinal cancers and ulcers but these should be interpreted with caution. This first study of multiple pathogens with risk of gastrointestinal cancers and ulcers demonstrated that several pathogens are associated with risks of gastrointestinal cancers and ulcers. This will inform future investigations into the role of infection in the etiology of these diseases.
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  • DOI:
    文章类型: Case Reports
    报道了一例IgG4相关疾病,表现为十二指肠溃疡,以改善IgG4相关疾病的认识。一名70岁的男性出现皮肤瘙痒和腹痛四年,大便变黑两个月。四年前,患者因皮肤瘙痒和腹痛入院。血清IgG4为3.09g/L(参考值0-1.35g/L),丙氨酸氨基转移酶554U/L(参考值9-40U/L),谷草转氨酶288U/L(参考值5-40U/L),总胆红素54.16μmol/L(参考值2-21μmol/L),直接胆红素29.64μmol/L(参考值1.7-8.1μmol/L)均升高。腹部CT扫描和磁共振胰胆管造影显示胰腺肿胀,胆总管狭窄,胆道系统继发性阻塞性扩张.患者被诊断为IgG4相关疾病,并每天使用40mg泼尼松治疗。随着黄疸和腹痛的改善,泼尼松逐渐减少至停药。两个月前,病人出现黑便,血常规检查显示严重贫血,消化道出血被诊断出来.患者来到北京医院急诊科,在其他医院治疗后无好转。胃镜检查显示十二指肠球部溃疡1.5厘米。奥美拉唑治疗后,粪便隐血仍呈阳性。进行PET-CT检查,它显示十二指肠壁的代谢活动没有异常,没有发现肿瘤性病变。考虑IgG4相关疾病,患者入院北京医院风湿免疫科进一步诊治。该患者有右下颌下腺肿块切除史和糖尿病。病人入院后,重新评估了血液检查。血清IgG4升高至5.44g/L(参考值0.03-2.01g/L)。腹部增强CT显示胰腺轻度肿胀,异常强化,肝内、肝外胆管扩张及肠系膜上血管周围软组织。我们对十二指肠球茎的IgG和IgG4进行了病理重新评估和染色。免疫组织化学染色显示IgG4阳性浆细胞明显浸润十二指肠组织,IgG4阳性细胞的数量为每个高倍视野20-30个细胞,IgG4/IgG阳性浆细胞比例超过40%。患者接受了40mg每日剂量的静脉注射甲基强的松龙和环磷酰胺治疗,然后十二指肠溃疡就痊愈了.IgG4相关疾病是一种以慢性炎症和纤维化为特征的免疫药物罕见疾病。这是一种全身性疾病,几乎影响到身体的每个解剖部位,通常累及多个器官,临床表现多样。IgG4相关疾病的消化系统表现多为急性胰腺炎和胆管炎,很少表现为胃肠道溃疡。该病例证实IgG4相关疾病可以表现为十二指肠溃疡,并且是十二指肠溃疡的罕见原因之一。
    A case of IgG4-related disease presented with a duodenal ulcer to improve the understan-ding of IgG4-related diseases was reported. A 70-year-old male presented with cutaneous pruritus and abdominal pain for four years and blackened stools for two months. Four years ago, the patient went to hospital for cutaneous pruritus and abdominal pain. Serum IgG4 was 3.09 g/L (reference value 0-1.35 g/L), alanine aminotransferase 554 U/L (reference value 9-40 U/L), aspartate aminotransferase 288 U/L (reference value 5-40 U/L), total bilirubin 54.16 μmol/L (reference value 2-21 μmol/L), and direct bilirubin 29.64 μmol/L (reference value 1.7-8.1 μmol/L) were all elevated. The abdominal CT scan and magnetic resonance cholangiopancreatography indicated pancreatic swelling, common bile duct stenosis, and secondary obstructive dilation of the biliary system. The patient was diagnosed with IgG4-related disease and treated with prednisone at 40 mg daily. As jaundice and abdominal pain improved, prednisone was gradually reduced to medication discontinuation. Two months ago, the patient developed melena, whose blood routine test showed severe anemia, and gastrointestinal bleeding was diagnosed. The patient came to the emergency department of Beijing Hospital with no improvement after treatment in other hospitals. Gastroscopy revealed a 1.5 cm firm duodenal bulb ulcer. After treatment with omeprazole, the fecal occult blood was still positive. The PET-CT examination was performed, and it revealed no abnormality in the metabolic activity of the duodenal wall, and no neoplastic lesions were found. IgG4-related disease was considered, and the patient was admitted to the Department of Rheumatology and Immunology of Beijing Hospital for further diagnosis and treatment. The patient had a right submandibular gland mass resection history and diabetes mellitus. After the patient was admitted to the hospital, the blood test was reevaluated. The serum IgG4 was elevated at 5.44 g/L (reference value 0.03-2.01 g/L). Enhanced CT of the abdomen showed that the pancreas was mild swelling and was abnormally strengthened, with intrahepatic and extrahepatic bile duct dilation and soft tissue around the superior mesenteric vessels. We pathologically reevaluated and stained biopsy specimens of duodenal bulbs for IgG and IgG4. Immunohistochemical staining revealed remarkable infiltration of IgG4-positive plasma cells into duodenal tissue, the number of IgG4-positive cells was 20-30 cells per high-powered field, and the ratio of IgG4/IgG-positive plasma cells was more than 40%. The patient was treated with intravenous methylprednisolone at 40 mg daily dosage and cyclophosphamide, and then the duodenal ulcer was healed. IgG4 related disease is an immune-medicated rare disease characterized by chronic inflammation and fibrosis. It is a systemic disease that affects nearly every anatomic site of the body, usually involving multiple organs and diverse clinical manifestations. The digestive system manifestations of IgG4-related disease are mostly acute pancreatitis and cholangitis and rarely manifest as gastrointestinal ulcers. This case confirms that IgG4-related disease can present as a duodenal ulcer and is one of the rare causes of duodenal ulcers.
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  • 文章类型: Journal Article
    背景:非食道嗜酸性粒细胞性胃肠道疾病(非EoE-EGID)是一种罕见的疾病,其中嗜酸性粒细胞渗入食道以外的胃肠道部分;但是,近年来,非EoE-EGID患者的数量一直在增加.由于其反复复发的慢性过程,它可能会导致营养不良导致发育迟缓,尤其是儿科患者。没有针对非EoE-EGID的既定治疗方法,需要长期全身性皮质类固醇给药。虽然dupilumab的疗效,抗IL-4/13受体单克隆抗体,对于嗜酸性粒细胞性食管炎,据报道,只有少数报告证明其在非EoEEGID中的疗效.
    方法:一个13岁的男孩发展为十二指肠溃疡的非EoE-EGID,以鸡蛋为扳机.他成功地接受了无鸡蛋饮食治疗,质子泵抑制剂,和白三烯受体拮抗剂.然而,15岁时,他出现了上腹痛恶化和进食困难。血液分析显示嗜酸性粒细胞增多;红细胞沉降率升高;C反应蛋白水平升高,总免疫球蛋白E,胸腺和活化调节趋化因子。上消化道内镜显示十二指肠溃疡伴明显的粘膜嗜酸性粒细胞浸润。胃肠道症状甚至在开始使用全身性类固醇后仍然存在,很难减少类固醇的剂量。由于特应性皮炎加重,开始皮下注射dupilumab。三个月后,胃肠道症状消失,5个月后,十二指肠溃疡消失,粘膜嗜酸性粒细胞计数减少。六个月后,全身性类固醇停药,十二指肠溃疡仍无复发。卵挑战测试结果为阴性;因此,停止无蛋饮食.dupilumab治疗后,血液嗜酸性粒细胞计数和血清IL-5,IL-13和eotaxin-3水平降低。血清IL-5和eotaxin-3水平保持在正常范围内,尽管口服泼尼松龙停药后血液嗜酸性粒细胞计数再次增加。
    结论:dupilumab抑制IL-4R/IL-13R介导的信号传导可能会改善非EoE-EGID患者的腹部症状以及内镜和组织学表现,导致停止全身类固醇给药和对致病食物的耐受性。
    BACKGROUND: Non-esophageal eosinophilic gastrointestinal disorder (non-EoE-EGID) is a rare disease in which eosinophils infiltrate parts of the gastrointestinal tract other than the esophagus; however, the number of patients with non-EoE-EGID has been increasing in recent years. Owing to its chronic course with repeated relapses, it can lead to developmental delays due to malnutrition, especially in pediatric patients. No established treatment exists for non-EoE-EGID, necessitating long-term systemic corticosteroid administration. Although the efficacy of dupilumab, an anti-IL-4/13 receptor monoclonal antibody, for eosinophilic esophagitis, has been reported, only few reports have demonstrated its efficacy in non-EoE EGIDs.
    METHODS: A 13-year-old boy developed non-EoE-EGID with duodenal ulcers, with chicken eggs as the trigger. He was successfully treated with an egg-free diet, proton pump inhibitors, and leukotriene receptor antagonists. However, at age 15, he developed worsening upper abdominal pain and difficulty eating. Blood analysis revealed eosinophilia; elevated erythrocyte sedimentation rate; and elevated levels of C-reactive protein, total immunoglobulin E, and thymic and activation-regulated chemokines. Upper gastrointestinal endoscopy revealed a duodenal ulcer with marked mucosal eosinophilic infiltration. Gastrointestinal symptoms persisted even after starting systemic steroids, making it difficult to reduce the steroid dose. Subcutaneous injection of dupilumab was initiated because of comorbid atopic dermatitis exacerbation. After 3 months, the gastrointestinal symptoms disappeared, and after 5 months, the duodenal ulcer disappeared and the eosinophil count decreased in the mucosa. Six months later, systemic steroids were discontinued, and the duodenal ulcer remained recurrence-free. The egg challenge test result was negative; therefore, the egg-free diet was discontinued. Blood eosinophil count and serum IL-5, IL-13, and eotaxin-3 levels decreased after dupilumab treatment. The serum levels of IL-5 and eotaxin-3 remained within normal ranges, although the blood eosinophil counts increased again after discontinuation of oral prednisolone.
    CONCLUSIONS: Suppression of IL-4R/IL-13R-mediated signaling by dupilumab may improve abdominal symptoms and endoscopic and histologic findings in patients with non-EoE-EGID, leading to the discontinuation of systemic steroid administration and tolerance of causative foods.
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