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Autoimmune interstitial lung disease and hepatitis C, an interesting association: Two case reports 自身免疫性间质性肺病与丙型肝炎:有趣的关联两个病例报告
Pub Date : 2024-05-22 DOI: 10.1016/j.hmedic.2024.100066
Alondra Esthefanía Llamas Domínguez , Julio Augusto Palma Zapata , Jose Luis Parra-Herrera , Juan Manuel Diaz , Silvia Denise Ponce-Campos

Introduction

The hepatitis C virus (HCV) is a hepatotropic and lymphotropic virus. Its infection generates a chronic stimulus to the immune system, giving rise to a wide range of systemic diseases with rheumatological, hematological, neurological, renal, and pulmonary manifestations. Interstitial lung disease (ILD) is one of the most common and harmful direct effects of chronic HCV infection in the lung. However, polyarthralgia and arthritis are the most common rheumatic conditions associated with this infection. There are also less common ones, such as myopathies, including anti-synthetase syndrome (ASS). Although immune syndromes are common in patients with chronic HCV infection, their pathogenesis for some illnesses is often unclear. This case report discusses the association between ILD, hepatitis C, and rheumatologic diseases. We cover clinical manifestations, theories of pathogenesis, and the importance of selecting appropriate treatment for these patients.

Cases presentations

Two clinical cases are presented, both diagnosed with ILD associated autoimmune diseases in patients with HCV infection. The first case is a 76-year-old man with progressive dyspnea, dry cough, muscle weakness, and joint pain in both hands. Physical examination revealed dry velcro-type crackles and the presence of clubbing. Laboratory studies showed a hepatitis C viral load detectable by PCR, positive rheumatoid factor (RF), and anti-citrullinated peptide (anti-CCP). Chest CT scan showed a usual interstitial pneumonia pattern (UIP). Therefore, this patient was diagnosed with ILD and concomitant rheumatoid arthritis (RA) due to HCV infection. The second case is a 44-year-old woman complaining of dry cough and dyspnea, chest tightness, odynophagia, itching, and nasal congestion. On physical examination, bilateral subscapularis fine crackles, mechanic's hands. Blood laboratory analyses revealed a positive hepatitis C antibody test with hepatitis C virus viral load undetectable by PCR, RF: (+), anti-Ro-52 (+); anti-PL-12 (+), anti-Ro/SSA 52 (+). Chest CT scan showed a pattern of organizing pneumonia (OP). As a result, we diagnosed the patient with ILD, ASS, and past infection with the HCV.

Conclusions

HCV and autoimmune diseases affect multiple systems, with lung involvement being a poor prognosis in both cases. Our case reports demonstrate positive outcomes in treating challenging diseases through close patient monitoring and individualized treatment decisions based on their needs, despite the absence of treatment guidelines for these patients.

导言丙型肝炎病毒(HCV)是一种趋肝性和趋淋巴性病毒。感染丙型肝炎病毒会对免疫系统产生慢性刺激,引起一系列全身性疾病,包括风湿病、血液病、神经病、肾病和肺病。间质性肺病(ILD)是慢性 HCV 感染对肺部造成的最常见、最有害的直接影响之一。然而,多关节痛和关节炎是与这种感染相关的最常见的风湿病。还有一些不太常见的疾病,如肌病,包括抗合成酶综合征(ASS)。虽然免疫综合征在慢性 HCV 感染患者中很常见,但某些疾病的发病机制往往并不清楚。本病例报告讨论了 ILD、丙型肝炎和风湿病之间的关联。我们将介绍这些患者的临床表现、发病机制理论以及选择适当治疗的重要性。病例介绍本报告介绍了两个临床病例,这两个病例均被诊断为 HCV 感染患者的 ILD 相关自身免疫性疾病。第一个病例是一名 76 岁的男性,患有进行性呼吸困难、干咳、肌无力和双手关节痛。体格检查发现他有干燥的尼龙搭扣式噼啪声,并伴有跛行。实验室检查显示,PCR 检测出丙型肝炎病毒载量,类风湿因子(RF)和抗瓜氨酸肽(anti-CCP)阳性。胸部 CT 扫描显示为常见间质性肺炎模式(UIP)。因此,该患者被诊断为因感染 HCV 而导致的 ILD 和并发类风湿性关节炎(RA)。第二例患者是一名 44 岁女性,主诉干咳和呼吸困难、胸闷、吞咽困难、鼻痒和鼻塞。体格检查时发现双侧肩胛下细噼啪声、机械手。血液实验室分析显示,丙型肝炎抗体检测呈阳性,PCR检测不到丙型肝炎病毒载量,RF:(+),抗Ro-52(+);抗PL-12(+),抗Ro/SSA 52(+)。胸部 CT 扫描显示为组织性肺炎(OP)。结论HCV和自身免疫性疾病会影响多个系统,肺部受累是两种病例的不良预后。我们的病例报告表明,尽管缺乏针对这些患者的治疗指南,但通过对患者进行密切监测并根据其需求做出个性化治疗决定,在治疗具有挑战性的疾病方面取得了积极成果。
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引用次数: 0
Drug-induced thrombotic microangiopathy associated with eltrombopag 与艾曲波帕相关的药物诱发血栓性微血管病
Pub Date : 2024-05-22 DOI: 10.1016/j.hmedic.2024.100069
Mohammad Tinawi

Drug-induced thrombotic microangiopathy due to an immune reaction is idiosyncratic and not dose-dependent. The classic example is quinine-dependent antibodies resulting in thrombotic microangiopathy due to activation of endothelial cells. Eltrombopag is a thrombopoietin-receptor agonist indicated in resistant chronic immune thrombocytopenia (ITP). This is the first report of full-blown biopsy-proven thrombotic microangiopathy with acute kidney injury (AKI) and nephrotic syndrome (NS) in a patient with chronic ITP who was initiated on eltrombopag therapy.

免疫反应导致的药物诱发血栓性微血管病是特异性的,与剂量无关。典型的例子是奎宁依赖性抗体激活内皮细胞导致血栓性微血管病。Eltrombopag 是一种血小板生成素受体激动剂,适用于抗药性慢性免疫性血小板减少症(ITP)。这是首例经活检证实患有血栓性微血管病并伴有急性肾损伤(AKI)和肾病综合征(NS)的慢性ITP患者接受艾曲波帕治疗后发生血栓性微血管病的报道。
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引用次数: 0
Mitomycin-induced pulmonary toxicity: A case report and review of literature 丝裂霉素诱发的肺毒性:病例报告和文献综述
Pub Date : 2024-05-21 DOI: 10.1016/j.hmedic.2024.100067
Sam Sugimoto , Hanna K. Sanoff , Lucas S. Wind , Jason R. Mock

Mitomycin is an alkylating chemotherapeutic agent that is used for the treatment of anal cancers. Pulmonary toxicities associated with mitomycin are broad and have been well described and range from acute bronchospasm, diffuse alveolar damage, interstitial pneumonitis, pleural disease to pulmonary veno-occlusive disease1, 2. The following is a case of a 63-year-old woman who suffered mitomycin-related acute lung injury 14 days after receiving the first dose. She had escalating oxygen needs, which eventually required mechanical ventilation. Treatment included high-dose steroids and broad-spectrum antibiotics; however, her respiratory function never recovered, and she died from cardiopulmonary arrest from hypoxia. This case highlights that patients undergoing therapy with mitomycin who develop respiratory symptoms should have this lung toxicity considered and the need for further research to recognize and treat this rare complication.

丝裂霉素是一种烷基化化疗药物,用于治疗肛门癌。与丝裂霉素相关的肺部毒性非常广泛,从急性支气管痉挛、弥漫性肺泡损伤、间质性肺炎、胸膜疾病到肺静脉闭塞性疾病,均有详细描述1, 2。以下是一名 63 岁女性的病例,她在接受第一剂丝裂霉素治疗 14 天后出现与丝裂霉素相关的急性肺损伤。她对氧气的需求不断增加,最终需要机械通气。治疗包括大剂量类固醇和广谱抗生素;然而,她的呼吸功能再也没有恢复,最终因缺氧导致心肺骤停而死亡。本病例强调,接受丝裂霉素治疗的患者如果出现呼吸系统症状,应考虑肺部毒性,并强调需要进一步研究来识别和治疗这种罕见的并发症。
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引用次数: 0
Mycobacterium xenopi infection presenting as cavitary lung nodule in immunocompetent individual- A case report 免疫功能正常者肺部出现空洞性结节的 xenopi 分枝杆菌感染--病例报告
Pub Date : 2024-05-06 DOI: 10.1016/j.hmedic.2024.100065
Sudeep Acharya, Sakura Thapa, Mahreen Anwar, Shamsuddin Anwar, Michel Chalhoub

Mycobacterium xenopi, a slow-growing non-tuberculous mycobacterium uncommonly recognized pathogenic organism especially in the United States. We present a unique and interesting case of pulmonary cavitary lesions and infection in a young patient with no comorbidities.

异种分枝杆菌是一种生长缓慢的非结核分枝杆菌,在美国尤其罕见。我们介绍了一例独特而有趣的病例,该病例是一名无合并症的年轻患者的肺腔病变和感染。
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引用次数: 0
An unconventional implant treatment – Implant placement in contact with ankylosed root fragments and management of its post operative complications: A case report 非常规种植治疗--接触强直牙根碎片的种植体植入及其术后并发症的处理:病例报告
Pub Date : 2024-04-25 DOI: 10.1016/j.hmedic.2024.100064
Tejaswi kodem , Anusha boddeda , S. spurthi , P. jerusha , Hyndavi balla

Implant treatment presumes that implants are placed in bone, without any contact with root. At ankylosed teeth, complete root removal is often invasive; subsequently, the sites require additional augmentation procedures to complete the treatment. Execution of a proper treatment is an important step for implant procedures. Implant failure occurs when these parameters are not selected properly. Implant failure can be avoided but may occur in few cases. The aim of this report is an unconventional implant placement with ankylosed root fragments, avoiding extractive invasive surgery and bone damage furthermore handling its post operative complications. In this case report an implant was placed in contact with ankylosed root following which patient reported back with exposed implant shoulder which was surgically manged by Coronally advanced flap using nanohydroxyapatite and fish collagen membrane leading to healthy periimplant mucosa. This case report opens up new avenues to implant placement at sites with compromised osteotomy sites to avoid an invasive extraction surgery and management of its post operative complications

种植治疗的前提是将种植体植入牙槽骨中,与牙根没有任何接触。对于强直的牙齿,完全去除牙根往往是侵入性的;随后,这些部位需要额外的增量手术来完成治疗。进行适当的治疗是种植手术的重要步骤。如果这些参数选择不当,种植失败就会发生。种植失败是可以避免的,但在少数情况下可能会发生。本报告的目的是对强直的牙根碎片进行非常规的种植体植入,避免创伤性拔牙手术和骨损伤,并进一步处理术后并发症。在本病例报告中,种植体与强直的牙根接触后,患者反馈种植体肩部外露,通过使用纳米羟基磷灰石和鱼胶原膜进行冠状前移皮瓣手术处理后,种植体周围粘膜变得健康。该病例报告为在截骨部位受损的部位植入种植体开辟了新的途径,从而避免了侵入性拔牙手术及其术后并发症的处理。
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引用次数: 0
Spiny papules on the soles: More than viral warts? 脚底的刺状丘疹:不仅仅是病毒疣?
Pub Date : 2024-04-20 DOI: 10.1016/j.hmedic.2024.100063
Alvaro Prados-Carmona , Francisco Javier de la Torre-Gomar , Ricardo Ruiz-Villaverde
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引用次数: 0
Familial mediterranean fever associated with amyloid storm: A case report with literature review 伴有淀粉样蛋白风暴的家族性地中海热:病例报告与文献综述
Pub Date : 2024-04-16 DOI: 10.1016/j.hmedic.2024.100061
Radwa M. Abdelsattar , Youstina Mohsen , Kholoud Mohamed El Sherif , Emad Samaan

One of the adverse complications of familial Mediterranean fever (FMF) is amyloid nephropathy, which usually progresses silently within 10–20 years into end-stage renal disease (ESRD). However, this case presented with rapid progression to ESRD, and the cause is suspected to be an amyloid storm. There are only a few cases that were reported to have developed this condition.

A 28-year-old male patient with a proven past medical history of FMF for five years was referred to the nephrology service of Mansoura University Hospitals (MUH) with acute kidney injury (AKI), heavy proteinuria, and high serum inflammatory markers. The condition deteriorated with rapidly increasing serum creatinine during admission and necessitated starting hemodialysis. Renal histopathology reported heavy renal amyloid deposition. The patient became hemodialysis dependent in the follow-up visits three months after discharge. This case raises the importance of suspecting an amyloid storm in FMF patients.

家族性地中海热(FMF)的不良并发症之一是淀粉样变性肾病,通常在10-20年内悄无声息地发展为终末期肾病(ESRD)。然而,该病例却迅速发展为 ESRD,其原因被怀疑是淀粉样蛋白风暴。一名 28 岁的男性患者因急性肾损伤 (AKI)、大量蛋白尿和高血清炎症指标被转诊至曼苏拉大学医院(MUH)肾内科,既往病史已证实其患有 FMF 长达五年。入院时病情恶化,血清肌酐迅速升高,不得不开始进行血液透析。肾组织病理学报告显示,患者肾脏淀粉样蛋白沉积严重。出院三个月后的随访中,患者开始依赖血液透析。该病例表明,怀疑 FMF 患者出现淀粉样蛋白风暴非常重要。
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引用次数: 0
Fatal CMV pneumonitis in critically ill COVID-19 patient–A case series of 4 patient COVID-19 重症患者中致命的 CMV 肺炎--4 例患者的系列病例
Pub Date : 2024-04-15 DOI: 10.1016/j.hmedic.2024.100062
Jamil Muqtadir Bhatti , Syed Ali Raza , Fatima Zaina , Syed Ali Abbas , Irshad Batool , Muhammad Owais Shahid , Nazish Hussain

Background

The SARS-CoV-2 epidemic has wreaked havoc on the world's leading healthcare systems, especially in intensive care. While cytomegalovirus (CMV) co-infections are widespread in severely ill patients because of an underlying immune suppression brought on by various causes, their influence on patients riddled with COVID-19 is unknown. Nevertheless, there is a correlation between severe COVID-19 and significant immune suppression, which has an impact on the reactivation of the coronavirus (CMV) and subsequently affects the clinical outcome.

Case presentation

The authors provide a case series of four patients with severe COVID-19-related respiratory failure referred to a high dependency unit (HDU). During their stay in the HDU, all the patients acquired CMV reactivation.

Conclusion

COVID pneumonia and immunosuppressants are used to suppress cytokine storms, but they can also play a role in CMV reactivation and its implications for fatal outcomes.

背景SARS-CoV-2疫情给世界领先的医疗系统,尤其是重症监护系统造成了严重破坏。由于各种原因导致的潜在免疫抑制,巨细胞病毒(CMV)合并感染在重症患者中非常普遍,但其对感染 COVID-19 的患者的影响尚不清楚。然而,重症 COVID-19 与严重的免疫抑制之间存在相关性,免疫抑制会影响冠状病毒(CMV)的再活化,进而影响临床结果。结论COVID 肺炎和免疫抑制剂可用于抑制细胞因子风暴,但它们也可在 CMV 再激活及其对致命后果的影响中发挥作用。
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引用次数: 0
Novel intronic variant in CYBB causing X-linked chronic granulomatous disease: Case report 导致X连锁慢性肉芽肿病的CYBB新型内含子变异:病例报告
Pub Date : 2024-04-13 DOI: 10.1016/j.hmedic.2024.100060
Bridget E. Wilson , Amrita Basu , Keith Sacco , Roshini S. Abraham

Chronic granulomatous disease (CGD) is caused by defective phagocyte NADPH oxidase function, leading to recurrent catalase-positive bacterial and fungal infections, granulomas, and hyperinflammatory manifestations. In some cases of CGD, the identified genetic variant may be novel or discordant with the patient presentation. In these cases, assessment of NADPH oxidase specific protein expression may be beneficial. Here, we present a 16-month-old male presenting with Nocardia and Cutibacterium infection and DHR-based flow cytometry with absent neutrophil oxidative burst. Genetic testing revealed a novel intronic variant in CYBB, so further testing was pursued. The patient had decreased gp91phox and p22phox in neutrophils and monocytes, confirming the diagnosis of X-linked CGD. When available, additional studies, including protein expression and/or functional evaluation can improve genotype-phenotype correlations.

慢性肉芽肿病(CGD)是由吞噬细胞 NADPH 氧化酶功能缺陷引起的,会导致反复出现过氧化氢酶阳性的细菌和真菌感染、肉芽肿和高炎症表现。在某些 CGD 病例中,确定的基因变异可能是新的,或与患者的表现不一致。在这些病例中,评估 NADPH 氧化酶特异性蛋白的表达可能是有益的。在此,我们介绍了一名 16 个月大的男性患者,他患有诺卡氏菌和 Cutibacterium 感染,基于 DHR 的流式细胞术显示中性粒细胞氧化爆发缺失。基因检测发现 CYBB 存在一个新的内含子变异,因此进行了进一步检测。患者中性粒细胞和单核细胞中的 gp91phox 和 p22phox 减少,确诊为 X 连锁 CGD。在有条件的情况下,包括蛋白质表达和/或功能评估在内的其他研究可以改善基因型与表型之间的相关性。
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引用次数: 0
Unusual presentation of recurrent testicular cancer after 13 years of initial presentation 睾丸癌初次发病 13 年后复发的罕见病例
Pub Date : 2024-04-10 DOI: 10.1016/j.hmedic.2024.100058
Ahmed Abdelhakeem, Raza Zarrar, Junaid Anwar

Introduction

Testicular cancers account for < 1 % of solid tumors in males. The majority (95 %) of testicular cancers are germ cell tumors (GCTS), and they are histologically subclassified into seminomas and non-seminomas. 70 % of seminoma patients present with localized stage I disease between the ages of 24 and 34. The estimated 5-year survival rate and 15-years cancer-specific survival of 95 % and 99 %, respectively. The overall crude relapse rate is 15.2–19.3 %, with most relapses occurring in the first two years. We present a case of relapsed metastatic mixed GCTS after 13 years of surgical resection of stage I seminoma.

Case description

A 45-year-old male patient with a remote history of localized left testicular cancer was treated with unilateral orchiectomy in 2008. He presented to the ER with a complaint of severe abdominal pain associated with nausea and vomiting. He reported loss of appetite and intentional weight loss of about 15 lbs. over the past six weeks. Computed tomography (CT) of the abdomen and pelvis with contrast showed 14.9 × 11 × 11.3 cm heterogeneous hypodense retroperitoneal mass circumscribes the abdominal aorta and inferior vena cava (IVC). Small bowel obstruction is not excluded.

The patient underwent upper GI endoscopy revealing severe extrinsic stenosis in the third portion of the duodenum. CT-guided biopsy of the retroperitoneal mass results was consistent with poorly differentiated adenocarcinoma. Immunotoxins demonstrated strong stains with CD117, CD30, and CDX2, consistent with mixed germ cell tumors; 85 % seminoma, 5 % embryonal carcinoma, 5 % yolk sac tumor, and 5 % teratoma. Tumor markers testing showed an alpha-feto protein level of 2905 ng/mL (normal level 10–20 ng/mL), Human chorionic gonadotropin serum level of 1062 mIU/mL (normal level 0–3 mIU/mL), and lactic dehydrogenase level of 1554 IU/L (normal level 313–618 IU/L).

Discussion

Our patient presented with relapsed mixed germ cell tumor after 13 years of successful surgical resection and surveillance for unilateral seminoma. Recurrence was in the form of large retroperitoneal mass, most probably arising from micro-metastases to the paraaortic nodes, causing mechanical compression. Similar cases that presented recurrence after a long duration of surveillance have been reported. However, very few cases of recurrent testicular cancer presenting with intestinal obstruction have been reported where a primary testicular cancer initially presented with GI metastases causing mechanical obstruction.

Conclusion

We describe an unusual case of large metastatic retroperitoneal mass originating from a mixed germ cell tumor in a patient who presented with small bowel obstruction after a very long surveillance period. More extended period of surveillance beyond 2–6 years after surgical resection of localized testicular cancer patients may be warranted.

导言睾丸癌占男性实体瘤的 1%。大多数(95%)睾丸癌是生殖细胞瘤(GCTS),在组织学上可分为精原细胞瘤和非精原细胞瘤。70%的精原细胞瘤患者在 24 至 34 岁之间出现局部 I 期疾病。估计 5 年生存率和 15 年癌症特异性生存率分别为 95% 和 99%。总的粗复发率为 15.2%-19.3%,大多数复发发生在头两年。我们报告了一例 I 期精原细胞瘤手术切除 13 年后复发转移的混合型 GCTS 病例。病例描述:一名 45 岁男性患者,远期病史为局部左侧睾丸癌,2008 年接受了单侧睾丸切除术。他到急诊室就诊时主诉腹部剧痛,伴有恶心和呕吐。他说过去六周食欲不振,体重有意减轻了约 15 磅。腹部和盆腔造影剂计算机断层扫描(CT)显示,14.9 × 11 × 11.3 厘米异质低密度腹膜后肿块环绕腹主动脉和下腔静脉(IVC)。患者接受了上消化道内窥镜检查,发现十二指肠第三部分有严重的外源性狭窄。CT引导下的腹膜后肿块活检结果与分化较差的腺癌一致。免疫毒素显示 CD117、CD30 和 CDX2 染色较强,与混合生殖细胞肿瘤一致;精原细胞瘤占 85%,胚胎癌占 5%,卵黄囊肿瘤占 5%,畸胎瘤占 5%。肿瘤标志物检测显示,α-酮蛋白水平为2905纳克/毫升(正常水平为10-20纳克/毫升),人绒毛膜促性腺激素血清水平为1062 mIU/毫升(正常水平为0-3 mIU/毫升),乳酸脱氢酶水平为1554 IU/L(正常水平为313-618 IU/L)。复发表现为腹膜后巨大肿块,很可能是主动脉旁结节的微转移引起的机械性压迫。类似的病例在长期监测后复发的情况也有报道。我们描述了一例不寻常的腹膜后巨大转移性肿块病例,该肿块源于混合性生殖细胞肿瘤,患者在长期监测后出现小肠梗阻。局部睾丸癌患者在手术切除后 2-6 年内可能需要进行更长时间的监测。
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引用次数: 0
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