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Fulminant Hepatic Failure after Chemosaturation with Percutaneous Hepatic Perfusion and Nivolumab in a Patient with Metastatic Uveal Melanoma. 转移性葡萄膜黑色素瘤患者经皮肝灌注和纳武单抗化疗后暴发性肝衰竭
IF 0.9 Pub Date : 2021-03-29 eCollection Date: 2021-01-01 DOI: 10.1155/2021/8870334
Lindsey Teal, Jeffrey Yorio

Immune checkpoint inhibitors, such as nivolumab, a programmed death receptor-1 (PD-1) inhibitor, have dramatically improved the treatment of advanced melanomas. Chemosaturation with percutaneous hepatic perfusion (PHP) delivers chemotherapy in high doses directly to the liver and is a potentially effective treatment modality in metastatic uveal melanoma with liver metastases. Its safety and effectiveness have not been studied in patients also receiving immunotherapy. A 46-year-old male with a history of uveal melanoma of the right eye was found to have liver metastases. He was treated with PHP using high-dose melphalan for 6 months with a partial response followed by progression. Two months after his last PHP treatment, the patient was started on nivolumab. After two doses of nivolumab, the patient developed severe hepatitis that progressed to fulminant hepatic failure and death despite treatment with high-dose corticosteroids and mycophenolate mofetil. Nivolumab and other immune checkpoint inhibitors have been effective in treating advanced melanoma and extending life. However, there are serious immune adverse events that can occur. While hepatitis after taking nivolumab has been documented, fulminant hepatic failure is rare. We believe that prior PHP treatment contributed to the severity of the hepatitis and, ultimately, fulminant hepatic failure. To our knowledge, this is the only case of fulminant hepatic failure secondary to a checkpoint inhibitor with preceding PHP. Specific precautions should be made in patients who have been exposed to PHP in the past, and further studies should be done to assess the safety of using checkpoint inhibitors after PHP.

免疫检查点抑制剂,如nivolumab,一种程序性死亡受体-1 (PD-1)抑制剂,极大地改善了晚期黑色素瘤的治疗。经皮肝灌注化疗饱和(PHP)将高剂量化疗直接输送到肝脏,是转移性葡萄膜黑色素瘤伴肝转移的潜在有效治疗方式。在同时接受免疫治疗的患者中,其安全性和有效性尚未得到研究。46岁男性,右眼有葡萄膜黑色素瘤病史,发现肝转移。患者使用高剂量美法仑治疗PHP 6个月,部分缓解,随后病情进展。在他最后一次PHP治疗两个月后,患者开始使用纳武单抗。两剂纳武单抗治疗后,患者发展为严重肝炎,进展为暴发性肝衰竭和死亡,尽管接受了大剂量皮质类固醇和霉酚酸酯治疗。Nivolumab和其他免疫检查点抑制剂在治疗晚期黑色素瘤和延长生命方面有效。然而,可能会发生严重的免疫不良事件。虽然服用纳武单抗后肝炎已被证实,但暴发性肝衰竭是罕见的。我们认为先前的PHP治疗导致了肝炎的严重程度,最终导致了暴发性肝衰竭。据我们所知,这是唯一的病例暴发性肝衰竭继发于检查点抑制剂与先前的PHP。对于既往暴露于PHP的患者应采取特别的预防措施,并应进一步研究PHP后使用检查点抑制剂的安全性。
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引用次数: 4
A Dose-Response Relationship to Radiotherapy for Cutaneous Lesions of Langerhans Cell Histiocytosis. 朗格汉斯细胞组织细胞增多症皮肤病变放射治疗的剂量-反应关系。
IF 0.9 Pub Date : 2021-03-24 eCollection Date: 2021-01-01 DOI: 10.1155/2021/6680635
Mark K Farrugia, Carl Morrison, Francisco Hernandez-Ilizaliturri, Saif Aljabab

Langerhans cell histiocytosis (LCH) is a rare disease, afflicting approximately 4.6 and 1-2 per 1 million children and adults, respectively. While LCH can involve numerous organ systems such as the lung or bone, it is uncommon for the disease to be limited to the skin. Radiotherapy has an established role for osseous lesions. However, the efficacy and dose for nonosseous manifestations of the disease are not well described. In the current case report, we detail a 49-year-old adult male with skin-limited LCH requiring palliative radiotherapy (RT) to numerous sites for pain control. The patient was initially diagnosed and treated with single agent cytarabine for approximately 6 months. Despite treatment, he had little symptomatic response of his cutaneous lesions. We delivered a single dose of 8 Gray (Gy) to 3 separate skin lesions, including the bilateral groin, right popliteal region, and right axillary lesion, which resulted in pain reduction and partial response at four-month follow-up. Subsequently, we decided to treat the left axillary untreated lesion to a higher dose of 24 Gy in 12 fractions. At four-month follow-up, the left axilla RT resulted in complete clinical response and improved pain control compared to the right axilla. Following RT treatments, the patient was found to have a BRAF mutation, and vemurafenib was initiated. Further follow-up with positron emissions tomography demonstrated complete metabolic response in numerous disease areas, including both axillae. Based on this case report's findings, a higher radiotherapy dose may be more effective for treating cutaneous LCH.

朗格汉斯细胞组织细胞增多症(LCH)是一种罕见的疾病,儿童和成人的发病率分别为4.6 / 100万和1-2 / 100万。虽然LCH可以涉及许多器官系统,如肺或骨,但这种疾病局限于皮肤是罕见的。放射治疗在骨性病变中的作用已经确立。然而,该疾病的非骨表现的疗效和剂量没有很好的描述。在目前的病例报告中,我们详细介绍了一位49岁的成年男性皮肤局限性LCH,需要姑息性放疗(RT)到多个部位来控制疼痛。患者最初接受单药阿糖胞苷治疗约6个月。尽管治疗,他的皮肤病变几乎没有症状反应。我们对3个不同的皮肤病变,包括双侧腹股沟、右腘窝区域和右腋窝病变,给予单剂量8 Gy,在4个月的随访中,疼痛减轻,部分缓解。随后,我们决定将未经治疗的左腋窝病变以24 Gy的高剂量分12次治疗。在四个月的随访中,与右腋窝相比,左腋窝RT产生了完全的临床反应和改善的疼痛控制。在接受RT治疗后,发现患者有BRAF突变,并开始使用vemurafenib。进一步随访的正电子发射断层扫描显示,在许多疾病区域,包括腋窝,有完全的代谢反应。根据本病例报告的结果,较高的放疗剂量可能对治疗皮肤LCH更有效。
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引用次数: 3
Development of Two Types of Skin Cancer in a Patient with Systemic Sclerosis: a Case Report and Overview of the Literature. 两种类型的皮肤癌在系统性硬化症患者的发展:一个病例报告和文献综述。
IF 0.9 Pub Date : 2021-02-26 eCollection Date: 2021-01-01 DOI: 10.1155/2021/6628671
Firdevs Ulutaş, Erdem Çomut, Veli Çobankara

Systemic sclerosis (SSc) is an uncommon rheumatic disease in which the underlying main histopathologic feature is a thickening of the skin due to excessive accumulation of collagen in the extracellular tissue. Fibrogenesis, chronic inflammation, and ulceration may eventually promote skin neoplasms. Although nonmelanoma skin cancer (NMSC) is the most frequent type, there have been restricted case reports and case series with skin cancers in SSc patients in the literature. Herein, we describe a 78-year-old woman diagnosed with diffuse cutaneous systemic sclerosis thirteen years ago and associated nonspecific interstitial pneumonia that was successfully treated with high cumulative doses of cyclophosphamide. She developed basal cell carcinoma and squamous cell carcinoma of the skin in the follow-up. She is still on rituximab treatment with stable interstitial lung disease as indicated by pulmonary function tests and high-resolution chest computed tomography. To our knowledge and a literature search, this is the first reported patient with SSc with two types of skin cancer. In this review, we also aimed to emphasize the relationship between SSc and skin cancer, and possible risk factors for SSc-related skin cancer.

系统性硬化症(SSc)是一种罕见的风湿病,其潜在的主要组织病理学特征是由于细胞外组织中胶原蛋白的过度积累而导致皮肤增厚。纤维形成、慢性炎症和溃疡最终可促进皮肤肿瘤的发生。虽然非黑色素瘤皮肤癌(NMSC)是最常见的类型,但文献中关于SSc患者皮肤癌的病例报告和病例系列有限。在此,我们描述了一位78岁的女性,13年前被诊断为弥漫性皮肤系统性硬化症,并伴有非特异性间质性肺炎,并通过高累积剂量环磷酰胺成功治疗。她在随访中发展为皮肤基底细胞癌和鳞状细胞癌。她仍在接受利妥昔单抗治疗,肺功能检查和高分辨率胸部计算机断层扫描显示她患有稳定的间质性肺病。据我们所知和文献检索,这是第一例伴有两种类型皮肤癌的SSc患者。在这篇综述中,我们还旨在强调SSc与皮肤癌的关系,以及SSc相关皮肤癌的可能危险因素。
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引用次数: 3
Early Gastrointestinal Progression to Immunotherapy in Lung Cancer: A Report of Two Cases. 肺癌早期胃肠道进展到免疫治疗两例报告。
IF 0.9 Pub Date : 2021-02-26 eCollection Date: 2021-01-01 DOI: 10.1155/2021/6692538
Federica Martorana, Katia Lanzafame, Giuliana Pavone, Lucia Motta, Gianmarco Motta, Nicola Inzerilli, Rosaria Carciotto, Giada Maria Vecchio, Antonino Maria Zanghì, Héctor Josè Soto Parra, Gaetano Magro, Paolo Vigneri

Intestinal and pancreatic metastases are rare and often challenging to recognize and manage. Lung cancer patients with enteric involvement usually display poor outcomes. Hyperprogression to immunotherapy represents a concern, even though there is currently no agreement on its exact definition. Gastrointestinal hyperprogression to immune checkpoint inhibitors has not been described so far. In these cases, distinguishing disease-related symptoms from immune-related adverse events may represent a diagnostic conundrum. Here, we report two cases of non-small-cell lung cancer experiencing a rapid pancreatic and colic progression to immunotherapy, respectively. While further investigations to identify biomarkers associated with hyperprogression are warranted, clinicians should be aware of the potential unusual clinical presentations of this phenomenon.

肠道和胰腺转移是罕见的,往往难以识别和管理。累及肠道的肺癌患者通常表现出较差的预后。免疫治疗的超进展引起了人们的关注,尽管目前对其确切定义还没有达成一致。胃肠过度进展到免疫检查点抑制剂目前还没有描述。在这些病例中,区分疾病相关症状和免疫相关不良事件可能是一个诊断难题。在这里,我们报告两例非小细胞肺癌经历快速胰腺和绞痛进展到免疫治疗分别。虽然进一步的研究确定与超进展相关的生物标志物是必要的,但临床医生应该意识到这种现象的潜在不寻常的临床表现。
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引用次数: 2
Response to Treatment with an Anti-Interleukin-6 Receptor Antibody (Tocilizumab) in a Patient with Hemophagocytic Syndrome Secondary to Immune Checkpoint Inhibitors. 抗白细胞介素-6受体抗体(Tocilizumab)治疗继发免疫检查点抑制剂的噬血细胞综合征患者的反应
IF 0.9 Pub Date : 2021-02-12 eCollection Date: 2021-01-01 DOI: 10.1155/2021/6631859
Alejandro Olivares-Hernández, Luis Figuero-Pérez, María A Amores Martín, Lorena Bellido Hernández, Laura Mezquita, María Del Rosario Vidal Tocino, Félix López Cadenas, Felipe Gómez-Caminero López, Roberto A Escala-Cornejo, Juan Jesús Cruz Hernández

Background. Immunotherapy represents one of the fundamental treatments in the management of some types of cancer, especially malignant melanoma. Toxicity derived from increased immune system activity can manifest in multiple organs and systems. We present a case of hematological toxicity, manifested as hemophagocytic syndrome (HPS), which was successfully treated with an anti-interleukin-6 antibody (tocilizumab). Case Report. This case presents a 75-year-old woman diagnosed with metastatic choroidal melanoma, refractory to several lines of treatment. After the failure of the previous lines, ipilimumab was started. After the third dose, she developed grade 2 thrombocytopenia and anemia accompanied by elevated levels of ferritin, triglycerides, and decreased fibrinogen. Hemophagocytosis was observed in the bone marrow biopsy, and a PET-CT showed splenomegaly with increased metabolism. Treatment was based on high doses of corticosteroids and tocilizumab. Four days after the start of treatment, progressive clinical and analytical improvement was observed, achieving total remission of the condition. Discussion. HPS induced by immunotherapy is due to an immunorelated cytokine storm syndrome (CSS). The administration of the anti-interleukin-6 receptor antibody drug acted on this cytokine cascade, leading to stabilization and subsequent remission. For this reason, the use of tocilizumab should be part of the immunotherapy-induced HPS treatment algorithm.

背景。免疫疗法是治疗某些类型癌症的基本疗法之一,尤其是恶性黑色素瘤。免疫系统活性增加引起的毒性可在多个器官和系统中表现出来。我们提出一例血液毒性,表现为噬血细胞综合征(HPS),这是成功的治疗与抗白细胞介素-6抗体(托珠单抗)。病例报告。这个病例是一位75岁的女性,被诊断为转移性脉络膜黑色素瘤,对几种治疗方法都难治。在之前的系列失败后,ipilimumab开始了。第三次给药后,患者出现2级血小板减少症和贫血,并伴有铁蛋白、甘油三酯水平升高和纤维蛋白原降低。骨髓活检显示噬血细胞增多,PET-CT显示脾肿大,代谢增加。治疗基于高剂量皮质类固醇和托珠单抗。治疗开始4天后,观察到临床和分析进展改善,达到病情完全缓解。讨论。免疫治疗诱导的HPS是由免疫相关细胞因子风暴综合征(CSS)引起的。抗白细胞介素-6受体抗体药物作用于细胞因子级联,导致稳定和随后的缓解。因此,tocilizumab的使用应该成为免疫治疗诱导的HPS治疗算法的一部分。
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引用次数: 8
Reconstruction with Total Scapular Reverse Total Shoulder Endoprosthesis after Radical Tumor Excision. 肿瘤根治性切除后全肩胛骨反向全肩关节内假体重建。
IF 0.9 Pub Date : 2021-01-25 eCollection Date: 2021-01-01 DOI: 10.1155/2021/1968621
Michael J Harvey, Howard G Rosenthal

Malignant musculoskeletal tumors about the shoulder girdle region involving the scapula are fairly rare, but when diagnosed, challenging and complex surgical treatment may be warranted with the primary goal of improving patient survival. These tumors are typically extensive and infiltrative at the time of presentation, requiring radical resection to achieve margins and obtain local tumor control. Historically, forequarter amputation or flail extremity were the mainstays of treatment in these cases. Presently, with recent advances in diagnostics, imaging, adjuvant therapies, and surgical treatment, many patients presenting with malignant tumors involving the scapula are candidates for limb salvage surgery. Reconstruction with endoprosthesis seems to have gained acceptance as the preferred surgical treatment for such lesions, as this intervention has resulted in improved postoperative function and cosmesis, with an acceptable complication rate. We present our experience with recent advancement in these surgical efforts in the form of shoulder girdle reconstruction with total scapular reverse total shoulder prosthesis after radical tumor excision.

累及肩胛骨的肩带区恶性肌肉骨骼肿瘤相当罕见,但一旦确诊,可能需要具有挑战性和复杂的手术治疗,其主要目标是提高患者的生存率。这些肿瘤在出现时通常是广泛和浸润的,需要根治性切除以达到边缘和获得局部肿瘤控制。历史上,前肢截肢或连枷肢是治疗这些病例的主要方法。目前,随着诊断、影像学、辅助治疗和外科治疗的最新进展,许多患有累及肩胛骨的恶性肿瘤的患者都是肢体保留手术的候选人。假体重建似乎已被接受为此类病变的首选手术治疗方法,因为这种干预措施改善了术后功能和美观,并发症发生率也在可接受范围内。我们介绍了我们在根治性肿瘤切除后肩胛骨反向全肩关节假体重建肩带手术方面的最新进展。
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引用次数: 2
Syndrome of Inappropriate Antidiuretic Hormone in Esophageal Cancer Patient. 食管癌患者抗利尿激素不适宜综合征。
IF 0.9 Pub Date : 2021-01-19 eCollection Date: 2021-01-01 DOI: 10.1155/2021/8131834
Rahmawati Minhajat, Andi Fachruddin Benyamin, Andi Makbul Aman, Syakib Bakri

Syndrome of inappropriate antidiuretic hormone (SIADH) is a disorder of fluid and sodium balance characterized by hypotonic hyponatremia, low plasma osmolality, and increased urine osmolality caused by excessive release of antidiuretic hormone (ADH). Malignancy is one of the most common causes of SIADH, but SIADH in esophageal carcinoma is very rarely reported. In this case report, a 74-year-old male patient of moderate differentiation of squamous cell esophageal carcinoma had a recurrent electrolyte balance disorder despite repeated corrections. The patient experienced improvement after fluid restriction and drug administration.

不适当的抗利尿激素综合征(SIADH)是一种以低渗性低钠血症、低血浆渗透压和尿渗透压升高为特征的液体和钠平衡紊乱,由过量释放抗利尿激素(ADH)引起。恶性肿瘤是SIADH最常见的病因之一,但食管癌的SIADH很少报道。在本病例报告中,74岁男性中度分化鳞状细胞食管癌患者反复出现电解质平衡紊乱,尽管多次纠正。患者在限制液体和给药后病情有所改善。
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引用次数: 0
Synchronous Occurrence of Splenic Pleomorphic Mantle Cell Lymphoma and Esophageal Adenocarcinoma with Overexpression of BCL1 Protein. 脾多形性套细胞淋巴瘤和食管腺癌同时发生与BCL1蛋白过表达。
IF 0.9 Pub Date : 2020-12-21 eCollection Date: 2020-01-01 DOI: 10.1155/2020/8888829
Dominik Dabrowski, Roberto F Silva, Michael Constantinescu, Rodney E Shackelford, Nestor Dela Cruz, Eric X Wei

Synchronous occurrences of mantle cell lymphoma (MCL), or intermediate lymphocytic lymphoma, and other malignancies are rare. Such cases present diagnostic and especially therapeutic challenges, making them of particular interest to study. We report a case of synchronic MCL and an esophageal tumor in an elderly male patient. Morphologically, the tumors were classified as splenic pleomorphic MCL and adenocarcinoma of the esophagus occurring concurrently. The pleomorphic MCL mimicked diffuse large B cell lymphoma (DLBCL) but lacked larger centroblast- or immunoblast-like cells. Curiously, both tumors overexpressed cyclin D1 by immunohistochemistry. This is an important feature that distinguishes MCL pathologically from two of its closest entities in the differential diagnosis: chronic lymphocytic leukemia and DLBCL, the latter of which mantle cells cannot transform into. The lymphoproliferation revealed IGH/CCND1 translocation by FISH, but the esophageal adenocarcinoma only showed CCND1 aneuploidy without break-apart signals. Since the gastrointestinal (GI) tract is a common site of extranodal involvement by MCL and lymphomatous polyposis can present as GI polyps, adequate care was taken to differentiate the esophageal adenocarcinoma from advanced stagings of MCL, as well as metastatic adenocarcinoma. Despite numerous immunohistochemical stainings studied, only BCL1 was demonstrated to have partial overlap in both tumors. The patient underwent esophagectomy and splenectomy. A subsequent metastatic primary lung squamous cell carcinoma was diagnosed, after which the patient expired. MCL typically presents at an advanced stage and has been deemed incurable with a prognosis of only several years. It is unclear whether the patient succumbed to complications of his MCL or the metastatic squamous cell carcinoma. Furthermore, he was lost to follow-up for a year and only received treatment after his third cancer was diagnosed. We have reviewed previous reports of synchronic mantle cell lymphoma and other solid tumors or hematological malignancies in the literature.

套细胞淋巴瘤(MCL)或中间淋巴细胞淋巴瘤和其他恶性肿瘤的同时发生是罕见的。这些病例提出了诊断和治疗方面的挑战,使他们特别感兴趣的研究。我们报告一例同时性MCL和食道肿瘤在一个老年男性病人。形态学上,肿瘤分为脾多形性MCL和食管腺癌合并发生。多形性MCL模拟弥漫性大B细胞淋巴瘤(DLBCL),但缺乏较大的成中心细胞或免疫母细胞样细胞。奇怪的是,通过免疫组化,两种肿瘤都过表达cyclin D1。这是一个重要的特征,从病理学上区分MCL与两种最接近的鉴别诊断实体:慢性淋巴细胞白血病和DLBCL,后者的套细胞不能转化。淋巴增生FISH显示IGH/CCND1易位,而食管腺癌仅显示CCND1非整倍体,无分离信号。由于胃肠道是MCL累及结外的常见部位,而淋巴瘤性息肉病可表现为胃肠道息肉,因此需要充分注意区分食管腺癌与晚期MCL以及转移性腺癌。尽管研究了大量的免疫组织化学染色,但只有BCL1在两种肿瘤中被证明有部分重叠。患者行食管切除术和脾切除术。随后被诊断为转移性原发性肺鳞状细胞癌,之后患者死亡。MCL通常出现在晚期,被认为是无法治愈的,预后只有几年。目前尚不清楚患者是否死于MCL并发症或转移性鳞状细胞癌。此外,他失去了一年的随访,直到他的第三次癌症被诊断出来后才接受治疗。我们回顾了文献中关于同时性套细胞淋巴瘤和其他实体肿瘤或血液系统恶性肿瘤的先前报道。
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引用次数: 1
Hepatocellular Carcinoma: First Manifestation as Solitary Humeral Bone Metastasis. 肝细胞癌:首次表现为孤立的肱骨骨转移。
IF 0.6 Q4 ONCOLOGY Pub Date : 2020-12-01 eCollection Date: 2020-01-01 DOI: 10.1155/2020/8254236
Sumera Bukhari, Kristine Ward, Michael Styler

Hepatocellular carcinoma (HCC) most commonly presents with abdominal pain or mass, fever of unknown etiology, weight loss, and decompensation of known liver disease or at an asymptomatic stage through surveillance. Rarely, presenting symptoms can be exclusively related to extrahepatic metastases. Herein, we write a case of a patient with no known liver disease, presenting with a pathological fracture of the proximal humerus bone secondary to a massive solitary metastasis from HCC. This case represents an unusual appendicular skeletal metastasis in a patient with unknown primary HCC, successfully treated with sorafenib. The prognosis of HCC patients with extrahepatic metastasis is poor, and in the presence of bone metastases, the mean survival rate is severely reduced. However, the multikinase inhibitor sorafenib has been the standard of treatment. Recently, there has been developments of other therapeutic class of drugs (i.e., immune check inhibitors), which have shown promising benefits and better side effect profiles. Still, there is a need for further studies, owing to challenges in recognizing cellular and molecular markers.

肝细胞癌(HCC)最常见的症状是腹痛或肿块、病因不明的发热、体重减轻、已知肝病的失代偿或通过监测处于无症状阶段。在极少数情况下,出现的症状可能完全与肝外转移有关。在此,我们记录了一例无已知肝病的患者,其肱骨近端骨病理性骨折是继发于肝癌的巨大单发转移瘤。该病例是原发性不明的 HCC 患者中的一个不常见的阑尾骨骼转移瘤,患者接受索拉非尼治疗后获得成功。有肝外转移的 HCC 患者预后较差,如果出现骨转移,平均生存率会严重下降。然而,多激酶抑制剂索拉非尼一直是治疗的标准药物。最近,其他治疗类药物(即免疫检查抑制剂)也有了新的发展,显示出良好的疗效和更好的副作用。不过,由于在识别细胞和分子标记物方面存在挑战,因此仍需进一步研究。
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引用次数: 0
Delays in Diagnosis of Pulmonary Lymphangitic Carcinomatosis due to Benign Presentation. 良性表现对肺淋巴管癌诊断的延误。
IF 0.9 Pub Date : 2020-11-24 eCollection Date: 2020-01-01 DOI: 10.1155/2020/4150924
Swati Pandey, Shishir Ojha

The diagnosis of lymphangitic carcinomatosis is challenging due to the manifestation of nonspecific symptoms and radiographic abnormalities that bear similarity to those of interstitial lung disease. Herein, we report the case of a 53-year-old woman diagnosed with lymphangitic carcinomatosis from metastatic gastric adenocarcinoma, 3 months after her initial presentation.

淋巴管癌的诊断具有挑战性,因为其表现为非特异性症状和影像学异常,与肺间质性疾病相似。在此,我们报告一例53岁的女性,在她最初的表现3个月后,被诊断为转移性胃腺癌的淋巴管癌病。
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引用次数: 2
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Case Reports in Oncological Medicine
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