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Milky Tea-Colored Pleural Effusion: Empyema Complicated by Pneumothorax Due to Mixed Infection With Mycobacterium tuberculosis and Aspergillus fumigatus. 乳白色茶色胸腔积液:由结核分枝杆菌和烟曲霉混合感染引起的脓胸合并气胸。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-10 DOI: 10.12659/AJCR.951599
Yaya Gong, Hongyan Sun

BACKGROUND Empyema is the accumulation of infected fluid within the pleural cavity, sometimes accompanied by pneumothorax. Bacterial empyema is the most common. Tuberculous and fungal empyema are less common and can occur in immunocompromised patients. Empyema caused by mixed infection with both tuberculosis and fungal pathogens is even less common. CASE REPORT This report describes a 76-year-old male lung cancer patient admitted to the hospital with fever and cough. He was receiving tislelizumab immunotherapy before admission. Chest CT at admission revealed pneumonia. Following empirical antimicrobial therapy, the pneumonia showed no improvement. He refused bronchoscopy; therefore, a sputum sample was delivered for tNGS testing. Sputum tNGS testing indicated mixed infection with Acinetobacter baumannii, Stenotrophomonas maltophilia, Klebsiella pneumoniae, Streptococcus pneumoniae, Aspergillus fumigatus, Aspergillus flavus, and COVID-19. Following adjustment of the antimicrobial regimen based on pathogenetic findings, he developed empyema complicated by pneumothorax. A chest tube was inserted, resulting in improvement of empyema and pneumothorax symptoms. Bacterial, fungal, and Mycobacterium tuberculosis cultures of the pleural effusion were all negative. Further tNGS analysis of the pleural effusion revealed a mixed infection with Mycobacterium tuberculosis and Aspergillus fumigatus. The patient refused further treatment and died 5 days after discharge. CONCLUSIONS Diagnosis of tuberculous empyema and fungal empyema is challenging and the prognosis is poor. In patients with malignant tumors, particularly those receiving immunotherapy, the possibility of Mycobacterium tuberculosis infection and fungal infections should be fully considered when infections occur, and early diagnosis and treatment are essential.

背景:脓胸是感染的液体在胸膜腔内的积聚,有时伴有气胸。细菌性脓胸最常见。结核性和真菌性脓胸不太常见,可发生在免疫功能低下的患者。由结核和真菌病原体混合感染引起的脓胸更不常见。病例报告本报告描述了一位76岁男性肺癌患者因发烧和咳嗽入院。入院前他正在接受替利单抗免疫治疗。入院胸部CT显示肺炎。经经验性抗菌药物治疗后,肺炎无好转。他拒绝支气管镜检查;因此,送痰样本进行tNGS检测。痰液tNGS检测提示混合感染鲍曼不动杆菌、嗜麦芽窄养单胞菌、肺炎克雷伯菌、肺炎链球菌、烟曲霉、黄曲霉和COVID-19。在根据病理结果调整抗菌方案后,他出现了脓胸并发气胸。插入胸管,改善了脓胸和气胸症状。胸腔积液细菌、真菌和结核分枝杆菌培养均为阴性。进一步的胸膜积液tNGS分析显示结核分枝杆菌和烟曲霉混合感染。患者拒绝进一步治疗,出院后5天死亡。结论结核性脓肿和真菌性脓肿诊断困难,预后较差。恶性肿瘤患者,特别是接受免疫治疗的患者,发生感染时应充分考虑结核分枝杆菌感染和真菌感染的可能性,早期诊断和治疗至关重要。
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引用次数: 0
Actinomycotic Cholecystitis and Pancreatitis: Report of an Unusual Case. 放线菌性胆囊炎和胰腺炎:罕见病例报告。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-10 DOI: 10.12659/AJCR.951112
Carlos Eduardo Brantis-de-Carvalho, Sara Mohrbacher, Juliana Valéria Souza Framil, Victor Augusto Hamamoto Sato, Erico Souza de Oliveira, Pedro Renato Chocair

BACKGROUND Actinomycosis is an uncommon bacterial infection caused by the commensal organism Actinomyces spp., which can become pathogenic upon tissue injury or disruption of the mucosal barrier. While primarily recognized for its cervicofacial and abdominopelvic presentations, actinomycosis can also involve the gallbladder and potentially the pancreas, posing diagnostic and therapeutic challenges due to its ability to mimic other conditions. CASE REPORT We present the case of a 26-year-old woman with a history of recurrent acute pancreatitis and cholecystitis, followed by the development of thrombotic microangiopathy. An atypical hemolytic-uremic syndrome panel revealed gene mutations previously associated with the onset of this condition. Computed tomography, endoscopic ultrasound, and abdominal ultrasound revealed focal enlargement of secondary ducts in the head of the pancreas. While no gallbladder wall thickening was observed, imaging demonstrated a substantial amount of heterogeneous, predominantly hypoechoic and amorphous content within the gallbladder, suggestive of biliary sludge. The patient subsequently underwent cholecystectomy, and histopathological examination of the gallbladder specimen confirmed the presence of Actinomyces spp., observed as sulfur granules and Gram-stained bacilli, as well as varying degrees of mucosal hyperplasia, transmural fibrosis, xanthomatous histiocytes, and Rokitansky-Aschoff sinuses. CONCLUSIONS Following the procedure and appropriate antimicrobial therapy, the patient experienced resolution of the infection, improvement in overall health status, and no new episodes of pancreatitis during the following year. We propose that underlying CFHR3 and CFI gene mutations and initial systemic inflammation precipitated the thrombotic microangiopathy-atypical hemolytic uremic syndrome, and facilitated Actinomyces colonization of the compromised gallbladder, ultimately leading to chronic gallbladder disease and recurrence.

背景:放线菌病是一种罕见的细菌感染,由共生生物放线菌引起,在组织损伤或粘膜屏障破坏时可致病性。虽然放线菌病主要以颈面和腹腔为表现,但放线菌病也可累及胆囊和潜在的胰腺,由于其模仿其他疾病的能力,给诊断和治疗带来挑战。病例报告我们提出的情况下,26岁的妇女与复发性急性胰腺炎和胆囊炎的历史,随后发展为血栓性微血管病。一个非典型溶血性尿毒症综合征面板显示基因突变先前与该病的发病有关。计算机断层扫描、内窥镜超声和腹部超声显示胰腺头部继发导管局灶性扩大。虽然没有观察到胆囊壁增厚,但影像学显示胆囊内有大量不均匀的,主要是低回声和无定形的内容物,提示胆道污泥。患者随后行胆囊切除术,胆囊标本的组织病理学检查证实存在放线菌,观察到硫颗粒和革兰氏染色杆菌,以及不同程度的粘膜增生,跨壁纤维化,黄瘤组织细胞和Rokitansky-Aschoff窦。结论:在手术和适当的抗菌药物治疗后,患者感染得到了缓解,整体健康状况得到改善,在接下来的一年中没有新的胰腺炎发作。我们认为,潜在的CFHR3和CFI基因突变和最初的全身炎症促成了血栓性微血管病变-非典型溶血性尿毒症综合征,并促进了放线菌在受损胆囊的定植,最终导致慢性胆囊疾病和复发。
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引用次数: 0
Successful Treatment of Multifocal Demyelinating Sensory-Motor Neuropathy (Lewis-Sumner Syndrome) With Rituximab: A Case Report. 利妥昔单抗成功治疗多灶性脱髓鞘感觉-运动神经病变(Lewis-Sumner综合征)1例。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-09 DOI: 10.12659/AJCR.950084
Mengcui Gui, Jing Lin, Yue Li, Bo Chen, Bitao Bu, Zhijun Li

BACKGROUND Multifocal acquired demyelinating sensory-motor neuropathy (MADSAM) is recognized as a variant of chronic inflammatory demyelinating polyneuropathy. The primary characteristics of MADSAM include multifocal sensory loss and muscle weakness, which are frequently asymmetrical and predominantly affect the upper limbs. Involvement of the lower limbs is less commonly observed in MADSAM. CASE REPORT A 27-year-old female patient presented with recurrent numbness and weakness in her left lower limb was admitted to our hospital. Her medical history included episodes of left peripheral facial paralysis and lower-limb numbness and weakness, which had previously improved after short-term oral steroid therapy. In addition to motor and sensory peripheral nerve impairment in the left lower limb, the neurological examination revealed atrophy of the tongue muscle and a leftward deviation of the tongue. Cerebrospinal fluid examination and magnetic resonance imaging indicated no abnormalities. Electromyography suggested demyelination of motor and sensory nerves in the left lower limb. Sural nerve biopsy demonstrated demyelination changes and axonal degeneration. A diagnosis of multifocal sensory and motor neuropathy was considered, and the patient was administered corticosteroids and tacrolimus. As the condition progressed, electromyography showed gradual involvement of both lower limbs, leading to the consideration of MADSAM. Despite treatment with corticosteroids and tacrolimus, the patient experienced relapse. Rituximab was initiated, resulting in symptoms improvement and reduced recurrence without adverse events. CONCLUSIONS Corticosteroids, plasma exchange, and immunoglobulins have been demonstrated to be effective treatments for CIDP. In our MADSAM case, rituximab proved effective when the patient did not respond to corticosteroids and tacrolimus. We propose that rituximab may serve as an alternative option for patients with MADSAM.

背景:多灶性获得性脱髓鞘感觉-运动神经病(MADSAM)被认为是慢性炎症性脱髓鞘性多神经病变的一种变体。MADSAM的主要特征包括多灶性感觉丧失和肌肉无力,通常是不对称的,主要影响上肢。下肢受累在MADSAM中较少见。病例报告一位27岁的女性患者因左下肢复发性麻木和无力住进我院。她的病史包括左周围性面瘫和下肢麻木无力,在短期口服类固醇治疗后有所改善。除了左下肢的运动和感觉周围神经损伤外,神经学检查显示舌肌萎缩和舌向左偏。脑脊液检查及磁共振检查未见异常。肌电图提示左下肢运动神经和感觉神经脱髓鞘。腓肠神经活检显示脱髓鞘改变和轴突变性。诊断为多灶性感觉和运动神经病变,并给予患者皮质类固醇和他克莫司。随着病情进展,肌电图显示两下肢逐渐受累,考虑为MADSAM。尽管用皮质类固醇和他克莫司治疗,患者还是复发了。开始使用利妥昔单抗,导致症状改善,复发减少,无不良事件。结论皮质类固醇、血浆置换和免疫球蛋白已被证明是治疗CIDP的有效方法。在我们的MADSAM病例中,当患者对皮质类固醇和他克莫司没有反应时,利妥昔单抗被证明是有效的。我们建议利妥昔单抗可以作为MADSAM患者的替代选择。
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引用次数: 0
Synchronous Presence of Papillary, Medullary, and Anaplastic Thyroid Tumors in a Single Patient: A Rare Case Report. 甲状腺乳头状、髓样及间变性肿瘤同时出现于一名患者:一罕见病例报告。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-09 DOI: 10.12659/AJCR.950677
Mohammed Al Essa, Reema Aldawish, Abdullah Alkhaldi, Ghaidaa Aljbli, Thamer Althunayan, Abdullah Alkarni, Abdullah Alsalamah

BACKGROUND Papillary thyroid carcinoma (PTC) is the most common form of thyroid cancer, while medullary thyroid carcinoma (MTC) and anaplastic thyroid carcinoma (ATC) are far less common. ATC is the most aggressive form, contributing to the majority of thyroid cancer-related deaths. While the coexistence of different pathological types of thyroid malignancies has been reported, including combinations of ATC, PTC, and poorly differentiated thyroid carcinoma, the detection of ATC within extrathyroidal metastatic lymph nodes remains exceedingly rare and of notable clinical significance. CASE REPORT We present a 68-year-old woman evaluated for a progressively enlarging right-sided neck mass which was apparat in clinical examination, with no other associated symptoms or signs. Nasopharyngoscopy was unremarkable. Imaging studies revealed a large cystic and necrotic mass in the right carotid space compressing the internal jugular vein. A core needle biopsy confirmed metastatic PTC. The patient underwent total thyroidectomy with right radical neck dissection. Histopathological evaluation reported a unique combination: a collision tumor composed of PTC, infiltrative follicular subtype, and MTC. Immunohistochemistry confirmed the diagnoses, and molecular analysis revealed a BRAF mutation. The patient was not a candidate for radioactive iodine therapy due to the aggressive nature of the anaplastic component. Thus, she was referred for targeted cancer therapy. CONCLUSIONS This case highlights the synchronous occurrence of PTC, MTC, and ATC in a single patient. The localization of anaplastic transformation solely to lymph nodes without thyroidal involvement underscores the importance of meticulous histopathological assessment. Comprehensive diagnostic workup and molecular profiling are critical in guiding treatment for such complex presentations.

背景:甲状腺乳头状癌(PTC)是最常见的甲状腺癌,而甲状腺髓样癌(MTC)和间变性甲状腺癌(ATC)则较少见。ATC是最具侵袭性的形式,是导致大多数甲状腺癌相关死亡的原因。虽然已有不同病理类型甲状腺恶性肿瘤共存的报道,包括ATC、PTC和低分化甲状腺癌的合并,但ATC在甲状腺外转移淋巴结中的检测仍然非常罕见,具有显著的临床意义。病例报告:我们报告一名68岁的女性,因其在临床检查中发现的逐渐增大的右侧颈部肿块,没有其他相关症状或体征。鼻咽镜检查无明显异常。影像学检查显示右侧颈动脉间隙有一个巨大的囊性坏死肿块压迫颈内静脉。核心穿刺活检证实转移性PTC。患者行甲状腺全切除术并右侧根治性颈部清扫术。组织病理学评估报告了一种独特的组合:由PTC,浸润性滤泡亚型和MTC组成的碰撞肿瘤。免疫组织化学证实了诊断,分子分析显示BRAF突变。由于间变性成分的侵袭性,该患者不适合放射性碘治疗。因此,她被推荐接受靶向癌症治疗。结论:该病例强调了PTC、MTC和ATC在同一患者中同时发生。间变性仅局限于淋巴结而不累及甲状腺强调了细致的组织病理学评估的重要性。全面的诊断检查和分子谱分析是指导治疗这种复杂的表现至关重要。
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引用次数: 0
Acute Graft-Versus-Host Disease After Deceased-Donor Liver Transplantation: A Case Report. 死亡供肝移植后急性移植物抗宿主病1例报告。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-08 DOI: 10.12659/AJCR.951031
Min Jae Kim, Keun Soo Ahn, Tae-Seok Kim, Sunggyun Park, Hye Won Lee

BACKGROUND Acute graft-versus-host disease is a rare but highly fatal complication of liver transplantation, with a reported mortality rate exceeding 70%. Most patients are diagnosed at 3 to 6 weeks postoperatively. Early diagnosis remains challenging because of nonspecific symptoms and overlapping presentations of infections and drug reactions. CASE REPORT We report the case of a 48-year-old woman with end-stage liver disease secondary to primary biliary cholangitis who underwent deceased-donor liver transplantation. On postoperative day 16, she developed abrupt-onset anemia without bleeding, followed by a high-grade fever, erythematous rash, profuse diarrhea, and pancytopenia. Blood cultures and viral studies were negative, despite clinical deterioration. Punch biopsy of the trunk on postoperative day 20 confirmed acute graft-versus-host disease. She was treated with high-dose corticosteroids and enhanced immunosuppression but showed no improvement, meeting the criteria for steroid-refractory graft-versus-host disease. She died of multiorgan failure on postoperative day 30. This is one of the earliest biopsy-confirmed cases of graft-versus-host disease reported after deceased-donor liver transplantation. CONCLUSIONS This case highlights the importance of early clinical suspicion of graft-versus-host disease after liver transplantation, even before the full triad of symptoms manifests. Isolated anemia may be an early warning sign. Therefore, prompt histopathological confirmation via skin biopsy is essential. Current treatment strategies for steroid-refractory graft-versus-host disease are limited, underscoring the need for further therapeutic advances.

背景:急性移植物抗宿主病是一种罕见但高度致命的肝移植并发症,据报道死亡率超过70%。大多数患者在术后3 - 6周被确诊。由于非特异性症状和感染和药物反应的重叠表现,早期诊断仍然具有挑战性。病例报告我们报告了一例48岁的妇女,她患有继发于原发性胆道胆管炎的终末期肝病,她接受了死亡供体肝移植。术后第16天,患者出现突发性贫血,无出血,随后出现高热、红斑疹、大量腹泻和全血细胞减少症。尽管临床情况恶化,但血液培养和病毒研究均为阴性。术后第20天躯干穿刺活检证实急性移植物抗宿主病。她接受了大剂量皮质类固醇和增强的免疫抑制治疗,但没有改善,符合类固醇难治性移植物抗宿主病的标准。术后第30天死于多器官衰竭。这是死亡供体肝移植后最早的活检证实的移植物抗宿主病病例之一。结论:该病例强调了肝移植后早期临床怀疑移植物抗宿主病的重要性,甚至在全部三联症状出现之前。孤立性贫血可能是一个早期预警信号。因此,通过皮肤活检及时进行组织病理学确认是必不可少的。目前对类固醇难治性移植物抗宿主病的治疗策略是有限的,强调需要进一步的治疗进展。
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引用次数: 0
Coronary Artery Bypass Grafting in the Presence of Severe Hyperthyroidism: A Case Report. 冠状动脉旁路移植术治疗严重甲亢1例。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-07 DOI: 10.12659/AJCR.950442
Akie Shimada, Taira Yamamoto, Jiyoung Lee, Shizuyuki Dohi, Daisuke Endo, Yuichiro Sato, Yasutaka Yokoyama, Minoru Tabata

BACKGROUND In coronary artery bypass grafting, hyperthyroidism with autoantibodies, as seen in Basedow disease, is uncommon and rarely leads to severe thyrotoxicosis. However, surgery can trigger this condition, and documented cases have resulted in poor outcomes. We describe the case of an incidental hyperthyroidism finding on preoperative evaluation for coronary artery bypass grafting for triple-vessel coronary artery disease. CASE REPORT A 58-year-old man presented with exertional dyspnea, chest pain, and postprandial vomiting. Coronary angiography revealed severe triple-vessel disease. After admission, he experienced additional episodes of chest pain, which required continuous infusion of coronary vasodilators and strict bed rest. Preoperative screening showed severe hyperthyroidism. Thiamazole 80 mg/day and potassium iodide 50 mg/day were initiated but discontinued after 2 weeks because of leukopenia (white blood cell count, 2800×10⁹/L). Although thyroidectomy was considered, coronary artery bypass grafting was prioritized owing to cardiac risk concerns. Perioperative management included continuous hemodiafiltration dialysis, methylprednisolone (1000 mg/day), and slow plasma exchange, starting with surgery to prevent thyroid storm. Intravenous gamma globulin was administered 3 days before and after surgery. Free triiodothyronine and free thyroxine were normalized on postoperative days 3 and 7, respectively. The postoperative course was uneventful, and the patient remained symptom-free at 1-year follow-up. CONCLUSIONS Coronary artery bypass grafting with uncontrolled hyperthyroidism is prone to complications, and a thyroid storm can be fatal. Although surgery should be avoided in cases of uncontrolled hyperthyroidism, in rare cases in which urgent treatment is not feasible, a critical intervention as described in this report may prevent thyroid crisis.

背景在冠状动脉旁路移植术中,甲状腺机能亢进伴自身抗体,如Basedow病,并不常见,很少导致严重的甲状腺毒症。然而,手术可以引发这种情况,并且有记录的病例结果很差。我们描述了一个偶然的甲状腺功能亢进发现术前评估冠状动脉旁路移植术的三支血管冠状动脉疾病。病例报告一名58岁男性表现为用力呼吸困难、胸痛和餐后呕吐。冠状动脉造影显示严重的三支血管病变。入院后,患者再次出现胸痛发作,需要持续输注冠状动脉扩张剂并严格卧床休息。术前筛查显示严重甲亢。开始使用噻马唑80 mg/天和碘化钾50 mg/天,但在2周后因白细胞减少(白细胞计数,2800×10⁹/L)而停用。虽然考虑了甲状腺切除术,但由于心脏风险的考虑,冠状动脉旁路移植术被优先考虑。围手术期处理包括持续血液透析、甲基强的松龙(1000mg /天)和缓慢血浆交换,从手术开始预防甲状腺风暴。术前和术后3天静脉注射丙种球蛋白。游离三碘甲状腺原氨酸和游离甲状腺素分别在术后第3天和第7天恢复正常。术后过程平稳,1年随访患者无症状。结论冠状动脉旁路移植术合并甲状腺功能亢进易发生并发症,甲状腺风暴可致患者死亡。虽然在不受控制的甲亢病例中应避免手术,但在紧急治疗不可行的极少数病例中,本报告所述的关键干预可能会预防甲状腺危象。
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引用次数: 0
Filiform Polyposis of the Terminal Ileum as the Initial Manifestation of Crohn's Disease: A Case Report. 回肠末端丝状息肉病为克罗恩病的最初表现:1例报告。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-06 DOI: 10.12659/AJCR.951544
Muyin Feng, Qili Xiao, Delong Mo, Yuting Xu, Yan Chen, Chuanjian Lu

BACKGROUND Filiform polyposis (FP) is a rare pseudopolyp associated with inflammatory bowel disease (IBD), characterized by elongated mucosal projections. It is found primarily in the colon and rectum, and presentation in the ileum is rare. Despite its benign nature, FP can mask underlying chronic intestinal inflammation. CASE REPORT A 52-year-old woman presented with loose stool but no other gastrointestinal symptoms. She was found to have a branched mucosal mass in the terminal ileum and diffuse ulcerations throughout the colorectum. Initial laboratory tests indicated anemia and elevated inflammatory markers. After 3 months of treatment with mesalazine and adjuvant drugs, the symptom of loose stool had improved, but the colonoscopy and imaging examination results show little change from the initial visit. Colonoscopy, imaging, and histopathology supported a diagnosis of Crohn's disease (CD). The ileal mass was identified as filiform polyposis. After multidisciplinary consultation, she was treated with Ustekinumab, leading to significant symptomatic and endoscopic improvement. The mass was subsequently resected via hybrid ESD and confirmed as an inflammatory polyp. CONCLUSIONS This case reveals that although a patient has no significant symptoms related to CD, FP located at the terminal ileum can initially manifest as CD, highlighting the need for through evaluation to detect underlying IBD. Multidisciplinary collaboration is essential for accurate diagnosis and management. FP can be the "tip of the iceberg" in hidden chronic intestinal inflammation, necessitating careful endoscopic and histologic assessment.

背景:丝状息肉病(FP)是一种罕见的与炎症性肠病(IBD)相关的假性息肉,其特征是延长的粘膜突出。它主要发生在结肠和直肠,在回肠的表现很少见。尽管它是良性的,但FP可以掩盖潜在的慢性肠道炎症。病例报告一名52岁女性,以稀便为主,无其他胃肠道症状。她被发现在回肠末端有一个支状粘膜肿块,并在整个结肠直肠有弥漫性溃疡。最初的实验室检查显示贫血和炎症标志物升高。经美沙拉嗪及辅助药物治疗3个月后,稀便症状有所改善,但结肠镜及影像学检查结果较初诊变化不大。结肠镜检查、影像学检查和组织病理学检查支持克罗恩病(CD)的诊断。回肠肿块为丝状息肉病。在多学科会诊后,她接受了Ustekinumab治疗,导致症状和内镜下的显著改善。随后通过混合ESD切除肿块,确认为炎性息肉。结论:该病例表明,尽管患者没有明显的CD相关症状,但位于回肠末端的FP最初可能表现为CD,因此需要通过评估来发现潜在的IBD。多学科合作对于准确诊断和管理至关重要。FP可能是隐性慢性肠道炎症的“冰山一角”,需要仔细的内镜和组织学评估。
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引用次数: 0
Management of Acute Cholecystitis in a Patient With Eisenmenger Syndrome and Abdominal Heterotaxy: A Case Report. 艾森曼格综合征合并腹部异位患者急性胆囊炎的处理:1例报告。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-06 DOI: 10.12659/AJCR.951532
Jes M Sanders, Matthew Harris, Juan Carlos Caicedo, Steven J Schwulst

BACKGROUND Eisenmenger syndrome presents a unique challenge for the acute care surgeon. Even routine operations such as laparoscopic appendectomy or cholecystectomy become challenging due to the cardiopulmonary physiologic changes and anatomic anomalies associated with Eisenmenger syndrome. The care of these patients can be further complicated by the severity of disease, surgical complexity, and the abnormal anatomy associated with the syndrome. CASE REPORT A 35-year-old patient with Eisenmenger syndrome and abdominal heterotaxy presented with acute cholecystitis. She underwent percutaneous cholecystostomy tube placement during her index hospitalization, which was complicated by atrial fibrillation and a cerebrovascular accident due to air embolism. Three months after presentation, she underwent an uncomplicated open cholecystectomy. She was discharged on post-operative day 5, and her course was notable only for a superficial surgical site infection requiring incision and drainage and antibiotics. CONCLUSIONS Our experience managing acute cholecystitis in a patient with Eisenmenger syndrome, abdominal heterotaxy with interrupted inferior vena cava, and bilateral superior vena cava, highlights critical aspects of care of such patients in the context of routine acute surgical care. Pre-operative planning should include optimization of cardiopulmonary function, an individualized anesthetic plan to maintain systemic vascular resistance, and bailout maneuvers in the event of cardiovascular collapse, such as planning for extracorporeal membrane oxygenation with axillary cannulation in the case of our patient.

背景:艾森曼格综合征对急症外科医生提出了一个独特的挑战。即使是常规手术,如腹腔镜阑尾切除术或胆囊切除术,由于心肺生理变化和解剖异常相关的艾森曼格综合征变得具有挑战性。这些患者的护理可能会因疾病的严重程度、手术的复杂性和与综合征相关的异常解剖而进一步复杂化。病例报告:一位35岁的艾森曼格综合征合并腹部异位的患者表现为急性胆囊炎。她在第一次住院期间接受了经皮胆囊造瘘管置入,并发心房颤动和因空气栓塞引起的脑血管意外。三个月后,她接受了简单的开放性胆囊切除术。她于术后第5天出院,她的病程值得注意的只是手术部位浅表感染,需要切开、引流和抗生素。结论:我们在处理艾森门格综合征患者急性胆囊炎,腹腔异位伴下腔静脉中断和双侧上腔静脉中断的经验,强调了在常规急性外科护理背景下护理这类患者的关键方面。术前计划应包括优化心肺功能,个体化麻醉计划以维持全身血管阻力,以及在发生心血管衰竭时的救助操作,例如本例患者的腋窝插管体外膜氧合计划。
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引用次数: 0
Giant Cell Arteritis/Polymyalgia Rheumatica and Atypical Pulmonary Carcinoid Tumor: A Paraneoplastic Syndrome? 巨细胞动脉炎/风湿性多肌痛和非典型肺类癌:一种副肿瘤综合征?
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-05 DOI: 10.12659/AJCR.950346
Omar Al Tabaa, Sabrina Hamroun, Khaled Al Tabaa, Roxana Poll, Waad Al Sheikh, Edouard Pertuiset

BACKGROUND Giant cell arteritis (GCA) is the most common large-vessel vasculitis in individuals over age 50 years. Although it typically affects extracranial branches of the carotid artery, central nervous system involvement is rare and can manifest with ischemic stroke. Links between GCA and malignancy have been reported, especially hematologic cancers, but paraneoplastic GCA associated with solid tumors remains exceptional and poorly understood. CASE REPORT We describe a 59-year-old man presenting with temporal headaches, right-hand paresthesia, monocular visual loss, and gait instability. Imaging revealed left thalamic ischemia and bilateral vertebral artery stenosis without atherosclerosis. Examination and Doppler ultrasound supported a diagnosis of GCA with associated polymyalgia rheumatica (PMR), although a temporal artery biopsy was negative. Inflammatory markers were only moderately elevated, consistent with reports of GCA cases with ischemic complications. PET-CT incidentally identified a mediastinal mass, confirmed as small-cell pulmonary neuroendocrine carcinoma. Symptoms improved with corticosteroids, but tapering below 15 mg/day caused relapse, requiring methotrexate. Despite partial oncologic response, brain metastases appeared 8 months later. Follow-up vascular imaging showed regression of arterial stenoses under corticosteroids, arguing against atherosclerosis and supporting the inflammatory nature of the lesions. GCA manifestations remained corticosteroid-dependent throughout follow-up. CONCLUSIONS The simultaneity of GCA and lung neuroendocrine carcinoma, persistence of corticosteroid dependence, and lack of remission despite oncologic response strongly suggest a paraneoplastic mechanism. This case emphasizes the need for heightened suspicion of underlying cancer in patients with atypical, biopsy-negative, or treatment-resistant vasculitis. Recognition of paraneoplastic GCA may improve early cancer detection and influence management decisions at the intersection of rheumatology and oncology.

巨细胞动脉炎(GCA)是50岁以上人群中最常见的大血管炎。虽然它通常影响颈动脉颅外分支,但累及中枢神经系统是罕见的,可表现为缺血性中风。GCA与恶性肿瘤之间的联系已被报道,特别是血液癌,但与实体瘤相关的副肿瘤GCA仍然是罕见的,并且知之甚少。病例报告我们描述了一个59岁的男性表现为颞头痛,右手感觉异常,单眼视力丧失和步态不稳定。影像学显示左丘脑缺血,双侧椎动脉狭窄,无动脉粥样硬化。尽管颞动脉活检呈阴性,但检查和多普勒超声支持GCA合并多肌痛风湿病(PMR)的诊断。炎症标志物仅中度升高,与有缺血性并发症的GCA病例的报道一致。PET-CT偶然发现纵隔肿块,证实为小细胞肺神经内分泌癌。皮质类固醇治疗可改善症状,但低于15毫克/天的剂量会导致复发,需要甲氨蝶呤。尽管有部分肿瘤反应,8个月后出现脑转移。后续血管成像显示皮质类固醇治疗后动脉狭窄消退,证明动脉粥样硬化不存在,支持病变的炎症性质。在整个随访过程中,GCA的表现仍然依赖于皮质类固醇。结论:GCA与肺神经内分泌癌同时发生,皮质类固醇依赖持续存在,尽管肿瘤反应,但仍未缓解,这强烈提示了一种副肿瘤机制。本病例强调,对于非典型、活检阴性或治疗抵抗性血管炎患者,需要加强对潜在癌症的怀疑。对副肿瘤性GCA的认识可以提高早期癌症的检测,并影响风湿病学和肿瘤学交叉领域的管理决策。
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引用次数: 0
Paraduodenal Hernia With Intestinal Obstruction During Pregnancy. 妊娠期十二指肠旁疝伴肠梗阻。
IF 0.7 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2026-02-05 DOI: 10.12659/AJCR.951298
Yuhang You, Fangxin Wan, Guodong Song

BACKGROUND Paraduodenal hernia (PDH) is a rare internal hernia, accounting for 50% to 55% of internal hernias but only 0.2% to 0.9% of intestinal obstructions. Right-sided PDH is less common, occurring in approximately 25% of cases. CASE REPORT This case describes a 33-year-old pregnant woman at 20 weeks 6 days gestation who was admitted with a 15-day history of abdominal distension accompanied by nausea and vomiting. The patient had postprandial exacerbation of intermittent abdominal pain, bilious vomiting, and significant weight loss (5 kg), with a history of similar symptoms during previous pregnancies. Abdominal contrast-enhanced computed tomography revealed clustered bowel loops in the right upper quadrant and medial displacement of the superior mesenteric vein, which is consistent with right-sided PDH complicated by intestinal malrotation. After conservative treatment failed, the patient opted for pregnancy termination followed by laparoscopic surgery. Intraoperative exploration confirmed complete absence of fusion between the ascending mesocolon and the posterior peritoneum, resulting in a wide hernia defect through which bowel loops had herniated into the space lateral to the ascending duodenum. The procedure included reduction of herniated contents, adhesiolysis, and fixation of the ascending mesocolon. The patient's recovery was uneventful, and she was discharged on postoperative day 5. At 3-month follow-up, no abnormalities were noted. CONCLUSIONS Given its nonspecific clinical presentation, PDH is frequently misdiagnosed or diagnosed late. Abdominal computed tomography facilitates early diagnosis and timely intervention, while laparoscopic repair offers favorable outcomes.

十二指肠旁疝(PDH)是一种罕见的内疝,占内疝的50% ~ 55%,但仅占肠梗阻的0.2% ~ 0.9%。右侧PDH不太常见,约占25%。病例报告:该病例描述了一名33岁妊娠20周6天的孕妇,因15天的腹胀伴恶心和呕吐病史入院。患者餐后间歇性腹痛加重,胆汁性呕吐,体重明显减轻(5 kg),既往妊娠有类似症状。腹部增强ct显示右上象限肠袢聚集,肠系膜上静脉内侧移位,与右侧PDH合并肠道旋转不良相符。保守治疗失败后,患者选择终止妊娠并行腹腔镜手术。术中探查证实升肠系膜与后腹膜之间完全没有融合,导致肠袢疝入升十二指肠外侧间隙。手术包括复位疝内容物、粘连松解和固定上升肠系膜。患者恢复顺利,术后第5天出院。随访3个月,未见异常。结论PDH临床表现非特异性,易误诊或诊断较晚。腹部计算机断层扫描有助于早期诊断和及时干预,而腹腔镜修复提供了良好的结果。
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引用次数: 0
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American Journal of Case Reports
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