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Successful treatment of tumor lysis syndrome associated with hepatic artery infusion chemotherapy in a patient with hepatocellular carcinoma: a case report. 肝动脉输注化疗成功治疗肝癌患者肿瘤溶解综合征1例。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-21 DOI: 10.1186/s13256-024-04928-y
Miao Li, Ying-Ting Zhou, Bi-Wei Yang

Background: Tumor lysis syndrome is a life-threatening complication in the treatment of cancer. However, it rarely occurs in solid tumors, especially in hepatocellular carcinoma.

Case presentation: We present a 52-year-old male Asian patient with advanced hepatocellular carcinoma treated with hepatic artery infusion chemotherapy that resulted in tumor lysis syndrome. The patient developed symptoms of oliguria, seizure, hyperkalemia, hyperuricemia, hypocalcemia, hyperphosphatemia, and increased creatinine. He recovered from it after adequate hydration, correction of metabolic abnormalities, and renal replacement therapy.

Conclusions: This case highlights the importance of maintaining a high index of suspicion of TLS even in solid tumors such as hepatocellular carcinoma, especially with a large tumor burden. It also underscores the need for early intervention in suspected TLS for a successful outcome.

背景:肿瘤溶解综合征是肿瘤治疗中一种危及生命的并发症。然而,它很少发生在实体瘤中,特别是在肝细胞癌中。病例介绍:我们报告一位52岁的亚洲男性晚期肝癌患者,接受肝动脉灌注化疗导致肿瘤溶解综合征。患者出现少尿、癫痫、高钾血症、高尿酸血症、低钙血症、高磷血症和肌酐升高等症状。经充分补水、纠正代谢异常和肾脏替代治疗后,他康复了。结论:本病例强调了即使在肝细胞癌等实体肿瘤中,特别是在肿瘤负荷较大的情况下,也应保持高度的TLS怀疑指数。它还强调需要对疑似TLS进行早期干预,以取得成功的结果。
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引用次数: 0
Polyarteritis nodosa presenting with severe upper gastrointestinal bleeding: a case report. 结节性多动脉炎伴严重上消化道出血1例。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-21 DOI: 10.1186/s13256-024-04892-7
Xin-Yue Xiao, Yun Niu, Ping-Ping Liu, Yu-Ming Zheng, Ming-Gang Zhang, Guo-Chun Wang, Xiao-Di Wang, Fang Liu

Background: Polyarteritis nodosa is a relatively uncommon type of systemic necrotizing vasculitis that primarily affects medium-sized arteries. While gastrointestinal involvement is known in polyarteritis nodosa, heavy gastrointestinal bleeding due to gastric ulceration is relatively uncommon. We present the case of an 81-year-old male of Chinese ethnicity who experienced severe gastrointestinal bleeding as a result of polyarteritis nodosa and an innovative treatment approach for a better patient outcomes.

Case presentation: Upon admission to the medical intensive care unit, the patient underwent a comprehensive diagnostic assessment, including examinations for cardiovascular and dermatological abnormalities, laboratory tests, autoantibody and tumor marker assessments, and imaging studies (such as endoscopies, whole-body computed tomography, and positron emission tomography-computed tomography scans), and a skin biopsy. The patient had tachycardia, hypotension, and extensive skin abnormalities on the lower extremities along with anemia, low platelets, and abnormal renal function. Upper gastrointestinal endoscopy revealed gastric and duodenal ulcers. Additional examinations, including electronic colonoscopy, capsule endoscopy, and whole-body computed tomography, were negative. A positron emission tomography-computed tomography scan showed increased uptake in the arterial walls and skin, which supported the diagnosis of polyarteritis nodosa, later confirmed by a biopsy of the skin on the lower extremities. Methylprednisolone, octreotide, and omeprazole were administered, leading to improvement in gastrointestinal symptoms, ulcer healing, and skin recovery. The patient continued with prednisone for 1 month.

Conclusion: This case serves to inform gastroenterologists about the need to consider polyarteritis nodosa in severe upper gastrointestinal bleeding and underscores the importance of prompt, medication-based treatment for successful patient outcome.

背景:结节性多动脉炎是一种相对少见的系统性坏死性血管炎,主要影响中等动脉。虽然结节性多动脉炎累及胃肠道是已知的,但由胃溃疡引起的大量胃肠道出血相对罕见。我们报告一例81岁的中国男性,由于结节性多动脉炎而经历了严重的胃肠道出血,并提出了一种创新的治疗方法,以获得更好的患者结果。病例介绍:在进入重症监护室后,患者接受了全面的诊断评估,包括心血管和皮肤异常检查、实验室检查、自身抗体和肿瘤标志物评估、影像学检查(如内窥镜检查、全身计算机断层扫描、正电子发射断层扫描-计算机断层扫描)和皮肤活检。患者出现心动过速、低血压、下肢广泛皮肤异常,并伴有贫血、低血小板和肾功能异常。上消化道内窥镜检查显示胃和十二指肠溃疡。其他检查包括电子结肠镜检查、胶囊内窥镜检查和全身计算机断层扫描均为阴性。正电子发射断层扫描-计算机断层扫描显示动脉壁和皮肤摄取增加,支持结节性多动脉炎的诊断,后来通过下肢皮肤活检证实。给予甲基强的松龙、奥曲肽和奥美拉唑,可改善胃肠道症状、溃疡愈合和皮肤恢复。患者持续使用强的松1个月。结论:该病例告知胃肠病学家在严重上消化道出血时需要考虑结节性多动脉炎,并强调了及时、基于药物的治疗对患者成功预后的重要性。
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引用次数: 0
Spinal schwannoma presenting with intraspinal hematoma: a case report and review of the literature. 以椎管内血肿为表现的脊髓神经鞘瘤:1例报告及文献复习。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-21 DOI: 10.1186/s13256-024-04968-4
Zhe Shen, Haoyang Chen, Haosheng Wang, Yaoqi Wang, Tao Li

Background: Spinal schwannomas presenting with an intraspinal hematoma or subarachnoid hemorrhage are extremely rare, and patients often have severe spinal cord compression symptoms. However, the mechanism underlying the bleeding remains unclear.

Case presentation: We present the case of a 53-year-old Chinese female diagnosed with a T12 schwannoma accompanied by an intratumoral hematoma. The patient suddenly experienced unbearable pain in the lower limbs. An emergency operation was necessary, and during surgery, we resected a tumor and evacuated a hematoma. We found a spinal nerve root fracture, intratumoral congestion, tumor capsule rupture, and bleeding. Pathological analysis indicated a schwannoma.

Conclusion: Injury to the nerve roots and vessels during motion, nerve root torsion, twisting, venous obstruction, tumor congestion, swelling, and capsule rupture are important processes in spinal schwannoma hemorrhage. Early diagnosis and proactive surgery are key points for treatment.

背景:脊髓神经鞘瘤表现为椎管内血肿或蛛网膜下腔出血极为罕见,患者通常有严重的脊髓压迫症状。然而,出血的机制尚不清楚。病例介绍:我们报告一例53岁的中国女性被诊断为T12神经鞘瘤并伴有瘤内血肿。病人突然感到下肢难以忍受的疼痛。紧急手术是必要的,在手术中,我们切除了一个肿瘤并清除了血肿。我们发现脊髓神经根骨折,肿瘤内充血,肿瘤囊破裂,出血。病理分析为神经鞘瘤。结论:运动损伤神经根和血管,神经根扭转、扭曲、静脉阻塞、肿瘤充血、肿胀、囊破裂是脊髓神经鞘瘤出血的重要过程。早期诊断和积极手术是治疗的关键。
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引用次数: 0
Postorgasmic illness syndrome benefiting from omalizumab and antidepressant: a case report. 性高潮后疾病综合征受益于奥玛单抗和抗抑郁药:一个病例报告。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-21 DOI: 10.1186/s13256-024-04986-2
Kasım Okan, Ragıp Fatih Kural, Meryem Demir, Furkan Cot, Mustafa Dinckal, Sevgi Altay, Nihal Mete Gökmen

Background: Postorgasmic illness syndrome is characterized by flu, rhinitis, conjunctivitis, loss of appetite, muscle weakness, and fatigue after ejaculation, lasting 2-7 days. The multidisciplinary treatment approach, incorporating omalizumab and antidepressants, has rarely been documented in literature.

Case presentation: In this article, we present the case of a 33-year-old single Turkish male with postorgasmic illness syndrome, characterized by typical clinical symptoms and a positive autologous semen test. Notably, his serum estrogen and progesterone levels were elevated. The patient's Beck anxiety score was 42 points, the depression scale score was 37, and suicidal thoughts. Fresh autologous semen taken at the hospital was diluted with 0.9% saline, and prick and intradermal skin tests were performed.

Conclusion: The patient's symptoms improved significantly with the combination of omalizumab and escitalopram. This case not only provides a new perspective on the management of postorgasmic illness syndrome but also highlights the potential roles of allergic, psychiatric, and endocrinological mechanisms in the etiology and treatment of this complex condition.

背景:高潮后疾病综合征以流感、鼻炎、结膜炎、食欲不振、肌肉无力、射精后疲劳为特征,持续2-7天。多学科治疗方法,结合奥玛珠单抗和抗抑郁药,很少有文献记载。病例介绍:在这篇文章中,我们提出了一个33岁的土耳其男性性高潮后疾病综合征的病例,其特点是典型的临床症状和阳性的自体精液测试。值得注意的是,他的血清雌激素和黄体酮水平升高。患者的贝克焦虑得分为42分,抑郁量表得分为37分,还有自杀念头。取医院新鲜自体精液,用0.9%生理盐水稀释,进行点刺和皮内皮肤试验。结论:奥马单抗与艾司西酞普兰合用可显著改善患者的症状。本病例不仅为性高潮后疾病综合征的治疗提供了新的视角,而且强调了过敏、精神病学和内分泌学机制在这一复杂疾病的病因和治疗中的潜在作用。
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引用次数: 0
Exponential decline, ceiling effect, downregulation, and T-cell response in immunoglobulin G antibody levels after messenger RNA vaccine boosters: a case report. 信使RNA疫苗增强剂后免疫球蛋白G抗体水平的指数下降、天花板效应、下调和t细胞反应:一例报告
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-20 DOI: 10.1186/s13256-024-04889-2
Harukazu Hirano, Hiroshi Asada

Background: Vaccine protection against severe acute respiratory syndrome coronavirus 2 infection reduces gradually over time, requiring administration of updated boosters. However, long-term immune response following up to the sixth dose of the messenger RNA vaccine has not been well studied.

Case presentation: We longitudinally determined anti-spike protein immunoglobulin G antibody levels in a 69-year-old Japanese man 76 times (first to sixth dose) to investigate their dynamics. Regarding the messenger RNA BNT162b2 vaccine, first to fourth doses were identical monovalent vaccines, and fifth and sixth doses were identical bivalent vaccines. T-cell responses after fourth and fifth doses were studied using T-SPOT. Immunoglobulin G levels peaked at 1-2 weeks after second to sixth dose, declining exponentially after each dose. The decline was approximated using the formula f (t) = Ae-t/τ + C. Time constant τ increased with each booster vaccination, indicating a decreasing rate of antibody titer decay with increasing number of doses. Baseline and peak immunoglobulin G levels were similar in the second and third dose. Conversely, baseline immunoglobulin G levels after the fourth dose increased over fivefold over the second and third dose; however, peak immunoglobulin G levels after fourth dose decreased to 60% of those after the third dose. Baseline immunoglobulin G levels after the sixth dose increased 1.4-fold over the fifth dose; however, peak immunoglobulin G levels after the sixth dose decreased to 56% of those after the fifth dose. Dynamics of T-cell responses differed from those of immunoglobulin G antibodies. T cell responses increased gradually; however, their peak level was difficult to determine.

Conclusions: Ceiling effect or downregulation of peak immunoglobulin G levels was clearly observed after messenger RNA booster vaccination. After peaking, the IgG level declined exponentially, and the rate of decay decreased with each subsequent booster. Although this was a single-case study, this data may provide a generalized mathematical decay model for humoral immunity in healthy older adults. Moreover, our study provides insights into the immunogenicity after booster vaccination with messenger RNA vaccines.

背景:疫苗对严重急性呼吸综合征冠状病毒2感染的保护作用随着时间的推移逐渐减弱,需要接种更新的增强剂。然而,信使RNA疫苗第六剂后的长期免疫反应尚未得到很好的研究。病例介绍:我们纵向测定抗刺突蛋白免疫球蛋白G抗体水平在一个69岁的日本男子76次(第一次至第六次剂量),以调查其动态。对于信使RNA BNT162b2疫苗,第一至第四剂为相同单价疫苗,第五和第六剂为相同二价疫苗。使用T-SPOT研究第四和第五次给药后的t细胞反应。免疫球蛋白G水平在第二次至第六次给药后1-2周达到峰值,每次给药后呈指数下降。使用公式f (t) = Ae-t/τ + C来近似计算下降量。时间常数τ随每次加强疫苗接种而增加,表明抗体滴度衰减率随剂量增加而降低。基线和峰值免疫球蛋白G水平在第二次和第三次剂量相似。相反,第四次剂量后的基线免疫球蛋白G水平比第二次和第三次剂量增加了五倍以上;然而,第四次给药后免疫球蛋白G的峰值水平下降到第三次给药后的60%。第六剂后基线免疫球蛋白G水平比第五剂增加1.4倍;然而,第六次注射后免疫球蛋白G的峰值水平下降到第五次注射后的56%。t细胞反应的动力学不同于免疫球蛋白G抗体。T细胞反应逐渐增强;然而,它们的峰值水平很难确定。结论:信使RNA加强疫苗接种后,免疫球蛋白G峰值水平出现明显的天花板效应或下调。达到峰值后,IgG水平呈指数级下降,随着后续的每一次增强,IgG的衰减率下降。虽然这是一项单例研究,但该数据可能为健康老年人体液免疫提供一个广义的数学衰减模型。此外,我们的研究为信使RNA疫苗加强接种后的免疫原性提供了见解。
{"title":"Exponential decline, ceiling effect, downregulation, and T-cell response in immunoglobulin G antibody levels after messenger RNA vaccine boosters: a case report.","authors":"Harukazu Hirano, Hiroshi Asada","doi":"10.1186/s13256-024-04889-2","DOIUrl":"10.1186/s13256-024-04889-2","url":null,"abstract":"<p><strong>Background: </strong>Vaccine protection against severe acute respiratory syndrome coronavirus 2 infection reduces gradually over time, requiring administration of updated boosters. However, long-term immune response following up to the sixth dose of the messenger RNA vaccine has not been well studied.</p><p><strong>Case presentation: </strong>We longitudinally determined anti-spike protein immunoglobulin G antibody levels in a 69-year-old Japanese man 76 times (first to sixth dose) to investigate their dynamics. Regarding the messenger RNA BNT162b2 vaccine, first to fourth doses were identical monovalent vaccines, and fifth and sixth doses were identical bivalent vaccines. T-cell responses after fourth and fifth doses were studied using T-SPOT. Immunoglobulin G levels peaked at 1-2 weeks after second to sixth dose, declining exponentially after each dose. The decline was approximated using the formula f (t) = Ae<sup>-t/τ</sup> + C. Time constant τ increased with each booster vaccination, indicating a decreasing rate of antibody titer decay with increasing number of doses. Baseline and peak immunoglobulin G levels were similar in the second and third dose. Conversely, baseline immunoglobulin G levels after the fourth dose increased over fivefold over the second and third dose; however, peak immunoglobulin G levels after fourth dose decreased to 60% of those after the third dose. Baseline immunoglobulin G levels after the sixth dose increased 1.4-fold over the fifth dose; however, peak immunoglobulin G levels after the sixth dose decreased to 56% of those after the fifth dose. Dynamics of T-cell responses differed from those of immunoglobulin G antibodies. T cell responses increased gradually; however, their peak level was difficult to determine.</p><p><strong>Conclusions: </strong>Ceiling effect or downregulation of peak immunoglobulin G levels was clearly observed after messenger RNA booster vaccination. After peaking, the IgG level declined exponentially, and the rate of decay decreased with each subsequent booster. Although this was a single-case study, this data may provide a generalized mathematical decay model for humoral immunity in healthy older adults. Moreover, our study provides insights into the immunogenicity after booster vaccination with messenger RNA vaccines.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"631"},"PeriodicalIF":0.9,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660962/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872273","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Atypical Kawasaki disease with giant coronary artery aneurysms in a 2-month-old boy: a case report. 非典型川崎病伴巨大冠状动脉瘤1例2月大男婴。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-20 DOI: 10.1186/s13256-024-04987-1
Najmeh Navidi, Babak Najibi, Negar Dinarvand, Amirmohammad Zamani, Mohammad Reza Fathi

Background: Kawasaki disease is an acute inflammatory disorder primarily affecting medium-sized blood vessels, especially in infants and young children under 5 years old, characterized by inflammation of the arterial walls, including coronary arteries. While predominantly affecting young children, rarely Kawasaki disease is seen in adults. Reporting this case is crucial due to the patient's very young age and the disease's unusual presentation following vaccination, contributing to the limited literature on Kawasaki disease in very young infants.

Case presentation: A full-term, 2-month-and-10-day-old Iranian boy presented with persistent fever unresponsive to treatment, beginning 5 days after receiving the 5-in-1 vaccine. Although physical examination results were normal, his medical history included maternal hypothyroidism during pregnancy and neonatal jaundice that did not require hospitalization. Initial laboratory tests revealed bicytopenia, thrombocytosis on the complete blood count, and elevated erythrocyte sedimentation rate and C-reactive protein levels. Echocardiography identified giant coronary artery aneurysms. The patient was diagnosed with atypical Kawasaki disease and treated with intravenous immunoglobulin, methylprednisolone, infliximab, and aspirin, which resolved his fever.

Conclusions: This case emphasizes the importance of considering Kawasaki disease as a differential diagnosis in infants with persistent, treatment-resistant fever and abnormal complete blood count findings. Early diagnosis and timely intervention are essential to prevent severe complications, such as coronary artery aneurysms and possible mortality. It underscores the need for heightened awareness and vigilance among healthcare providers in similar clinical scenarios.

背景:川崎病是一种主要影响中等血管的急性炎症性疾病,特别是在5岁以下的婴儿和幼儿中,其特征是动脉壁炎症,包括冠状动脉。虽然川崎病主要影响幼儿,但很少见于成人。由于患者年龄非常小,且疫苗接种后该病的表现不寻常,因此报告本病例至关重要,这导致关于幼小婴儿川崎病的文献有限。病例介绍:一名2个月零10天的伊朗足月男孩在接种5合1疫苗5天后出现持续发热,对治疗无反应。虽然体格检查结果正常,但他的病史包括妊娠期间母亲甲状腺功能减退和新生儿黄疸,不需要住院治疗。最初的实验室检查显示双氧体减少症,全血细胞计数的血小板增多,红细胞沉降率和c反应蛋白水平升高。超声心动图发现巨大冠状动脉动脉瘤。患者被诊断为不典型川崎病,经静脉注射免疫球蛋白、甲基强的松龙、英夫利昔单抗和阿司匹林治疗,发热消退。结论:本病例强调了将川崎病作为鉴别诊断的重要性,这些婴儿有持续性、治疗抵抗性发热和全血细胞计数异常。早期诊断和及时干预对于预防冠状动脉瘤等严重并发症和可能的死亡至关重要。它强调了在类似的临床情况下,医疗保健提供者需要提高认识和警惕。
{"title":"Atypical Kawasaki disease with giant coronary artery aneurysms in a 2-month-old boy: a case report.","authors":"Najmeh Navidi, Babak Najibi, Negar Dinarvand, Amirmohammad Zamani, Mohammad Reza Fathi","doi":"10.1186/s13256-024-04987-1","DOIUrl":"10.1186/s13256-024-04987-1","url":null,"abstract":"<p><strong>Background: </strong>Kawasaki disease is an acute inflammatory disorder primarily affecting medium-sized blood vessels, especially in infants and young children under 5 years old, characterized by inflammation of the arterial walls, including coronary arteries. While predominantly affecting young children, rarely Kawasaki disease is seen in adults. Reporting this case is crucial due to the patient's very young age and the disease's unusual presentation following vaccination, contributing to the limited literature on Kawasaki disease in very young infants.</p><p><strong>Case presentation: </strong>A full-term, 2-month-and-10-day-old Iranian boy presented with persistent fever unresponsive to treatment, beginning 5 days after receiving the 5-in-1 vaccine. Although physical examination results were normal, his medical history included maternal hypothyroidism during pregnancy and neonatal jaundice that did not require hospitalization. Initial laboratory tests revealed bicytopenia, thrombocytosis on the complete blood count, and elevated erythrocyte sedimentation rate and C-reactive protein levels. Echocardiography identified giant coronary artery aneurysms. The patient was diagnosed with atypical Kawasaki disease and treated with intravenous immunoglobulin, methylprednisolone, infliximab, and aspirin, which resolved his fever.</p><p><strong>Conclusions: </strong>This case emphasizes the importance of considering Kawasaki disease as a differential diagnosis in infants with persistent, treatment-resistant fever and abnormal complete blood count findings. Early diagnosis and timely intervention are essential to prevent severe complications, such as coronary artery aneurysms and possible mortality. It underscores the need for heightened awareness and vigilance among healthcare providers in similar clinical scenarios.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"630"},"PeriodicalIF":0.9,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660496/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872271","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Acute kidney injury and tacrolimus toxicity in a kidney transplant recipient treated with nirmaltrevir/ritonavir: a case report. 尼马尔替韦/利托那韦治疗肾移植受者的急性肾损伤和他克莫司毒性:一例报告。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-20 DOI: 10.1186/s13256-024-04990-6
Jack Rycen, Julia Jefferis, David Mudge

Background: Kidney transplant recipients with severe acute respiratory syndrome-coronavirus-2 infection have an increased risk of severe disease and mortality. Nirmaltrevir/ritonavir (Paxlovid) is an effective oral disease-modifying therapy that has been shown to reduce risk of progression to severe disease in high-risk, nonhospitalized adults. However, owing to the potential for serious drug-drug interactions owing to ritonavir-induced inhibition of the CYP3A enzyme, this drug is not suitable option for transplant recipients with mild-moderate severe acute respiratory syndrome-coronavirus-2 infection.

Case presentation: A 57-year-old Caucasian man presented to the emergency department with 48 hours of nausea, vomiting, headaches, and lethargy. At 5 days earlier, he was diagnosed with a mild severe acute respiratory syndrome-coronavirus-2 infection by his general practitioner, who commenced treatment with Paxlovid at 300 mg/100 mg twice daily. Past medical history included kidney transplantation in 2018 for end-stage kidney secondary to hypertensive nephrosclerosis, managed with prednisone, tacrolimus, and mycophenolate. Vaccination status was up-to-date and prophylactic tixagevimab/cilgavimab (Evusheld) had been given > 6 months prior owing to lack of seroconversion. Examination showed a blood pressure of 176/94 mmHg and normal respiratory parameters. Investigations demonstrated a serum creatinine of 213 µmol/L (baseline 130 µmol/L) and tacrolimus trough level of 118 µg/L (baseline 6.9-8.7 µg/L). Treatment included intravenous rehydration, Evusheld and tacrolimus were withheld for 7 days, with recommencement guided by regular therapeutic drug monitoring.

Conclusion: This acute kidney injury was attributed to tacrolimus toxicity resulting from a drug-drug interaction with Paxlovid. While transplant recipients have an increased risk of severe disease, current Australian guidelines recommend against Paxlovid use in adults taking medications that are heavily dependent on CYP3A4 for clearance, including calcineurin and mammalian target of rapamycin inhibitors.

背景:严重急性呼吸综合征-冠状病毒-2感染肾移植受者发生严重疾病和死亡的风险增加。Nirmaltrevir/ritonavir (Paxlovid)是一种有效的口腔疾病改善疗法,已被证明可降低高风险非住院成人进展为严重疾病的风险。然而,由于利托那韦诱导的CYP3A酶抑制可能导致严重的药物-药物相互作用,该药物不适合用于轻中度严重急性呼吸综合征-冠状病毒-2感染的移植受者。病例介绍:一名57岁的白人男性因48小时恶心、呕吐、头痛和嗜睡而被送往急诊室。5天前,他的全科医生诊断他患有轻度严重急性呼吸综合征-冠状病毒-2感染,并开始使用Paxlovid治疗,剂量为300 mg/100 mg,每日两次。既往病史包括2018年因继发于高血压肾硬化的终末期肾脏进行肾移植,使用强的松、他克莫司和霉酚酸盐治疗。疫苗接种状况是最新的,由于缺乏血清转化,已在2010年6个月前接种了预防性替沙吉维单抗/西gavimab (Evusheld)。检查显示血压176/94 mmHg,呼吸参数正常。调查显示血清肌酐为213µmol/L(基线130µmol/L),他克莫司谷水平为118µg/L(基线6.9-8.7µg/L)。治疗包括静脉补液,停用Evusheld和他克莫司7天,在定期治疗药物监测指导下重新开始。结论:急性肾损伤是由于他克莫司与Paxlovid药物相互作用引起的毒性所致。虽然移植受者发生严重疾病的风险增加,但目前澳大利亚的指南建议,在服用严重依赖CYP3A4清除药物的成人中,包括钙调磷酸酶和哺乳动物雷帕霉素靶抑制剂,不要使用Paxlovid。
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引用次数: 0
Adult-onset hypophosphatasia diagnosed after consecutive tooth loss during orthodontic treatment: a case report. 在正畸治疗期间连续牙齿脱落后诊断为成人发作的磷酸酶缺失:1例报告。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-20 DOI: 10.1186/s13256-024-04948-8
Shusuke Tokuchi, Toshihiro Kawano, Edward Hosea Ntege, Makoto Murahashi, Kentaro Ide, Nobuyuki Maruyama, Risako Suzuki, Mirei Takai-Nabeta, Tsuyoshi Nabeta, Hideo Tanaka, Yusuke Shimizu, Hiroyuki Nakamura

Background: Adult hypophosphatasia is an uncommon inherited disorder of mineral homeostasis affecting bone. It arises from mutations within the Alkaline Phosphatase, Biomineralization Associated (ALPL) gene, which encodes tissue-nonspecific alkaline phosphatase. Because of its low prevalence and non-specific clinical manifestations, underdiagnosis and misdiagnosis are frequent, particularly in Asian populations.

Case presentation: We present a case of a 38-year-old Japanese male diagnosed with adult hypophosphatasia following consecutive tooth loss during orthodontic treatment. Genetic analysis revealed a compound heterozygous mutation within the ALPL gene. The patient remained asymptomatic until orthodontic treatment, suggesting that increased mechanical stress overwhelmed residual enzyme activity, triggering the hypophosphatasia symptoms. Asfotase Alfa enzyme replacement therapy improved healing following tooth extraction.

Conclusion: This case highlights the significance of including adult hypophosphatasia in the differential diagnosis for obscure dental complications arising during orthodontic procedures, particularly in Asian patients where certain ALPL variants may be more prevalent. Effective diagnosis and management of adult hypophosphatasia necessitate collaboration between orthodontic practitioners and medical specialists. Improved awareness and a multidisciplinary approach are crucial for timely diagnosis and successful intervention.

背景:成人低磷血症是一种罕见的影响骨骼矿物质平衡的遗传性疾病。它起源于碱性磷酸酶,生物矿化相关(ALPL)基因的突变,该基因编码组织非特异性碱性磷酸酶。由于其低患病率和非特异性临床表现,漏诊和误诊是常见的,特别是在亚洲人群中。病例介绍:我们报告一个38岁的日本男性,在正畸治疗期间连续牙齿脱落,被诊断为成人低磷症。遗传分析显示ALPL基因存在复合杂合突变。患者在正畸治疗前无症状,提示机械应力的增加压倒了残留的酶活性,引发磷酸酶减少症状。Asfotase - Alfa酶替代疗法改善拔牙后的愈合。结论:本病例强调了将成人低磷酸酶血症纳入正畸手术中出现的模糊牙齿并发症的鉴别诊断的重要性,特别是在某些ALPL变异可能更为普遍的亚洲患者中。成人低磷血症的有效诊断和管理需要正畸医生和医学专家之间的合作。提高认识和多学科方法对于及时诊断和成功干预至关重要。
{"title":"Adult-onset hypophosphatasia diagnosed after consecutive tooth loss during orthodontic treatment: a case report.","authors":"Shusuke Tokuchi, Toshihiro Kawano, Edward Hosea Ntege, Makoto Murahashi, Kentaro Ide, Nobuyuki Maruyama, Risako Suzuki, Mirei Takai-Nabeta, Tsuyoshi Nabeta, Hideo Tanaka, Yusuke Shimizu, Hiroyuki Nakamura","doi":"10.1186/s13256-024-04948-8","DOIUrl":"10.1186/s13256-024-04948-8","url":null,"abstract":"<p><strong>Background: </strong>Adult hypophosphatasia is an uncommon inherited disorder of mineral homeostasis affecting bone. It arises from mutations within the Alkaline Phosphatase, Biomineralization Associated (ALPL) gene, which encodes tissue-nonspecific alkaline phosphatase. Because of its low prevalence and non-specific clinical manifestations, underdiagnosis and misdiagnosis are frequent, particularly in Asian populations.</p><p><strong>Case presentation: </strong>We present a case of a 38-year-old Japanese male diagnosed with adult hypophosphatasia following consecutive tooth loss during orthodontic treatment. Genetic analysis revealed a compound heterozygous mutation within the ALPL gene. The patient remained asymptomatic until orthodontic treatment, suggesting that increased mechanical stress overwhelmed residual enzyme activity, triggering the hypophosphatasia symptoms. Asfotase Alfa enzyme replacement therapy improved healing following tooth extraction.</p><p><strong>Conclusion: </strong>This case highlights the significance of including adult hypophosphatasia in the differential diagnosis for obscure dental complications arising during orthodontic procedures, particularly in Asian patients where certain ALPL variants may be more prevalent. Effective diagnosis and management of adult hypophosphatasia necessitate collaboration between orthodontic practitioners and medical specialists. Improved awareness and a multidisciplinary approach are crucial for timely diagnosis and successful intervention.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"626"},"PeriodicalIF":0.9,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660432/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142864564","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Intermuscular hydatid cyst in the thigh musculature: a case report. 大腿肌肉间水瘤囊肿:病例报告。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-20 DOI: 10.1186/s13256-024-04861-0
Aderaw Getie, Bahiru Atinafu, Misganaw Alemu, Genetu Belay, Dereje Bedane Hunde

Introduction: Echinococcus granulosus, a tapeworm, is responsible for causing hydatid disease. Hydatid cysts rarely affect the musculoskeletal system, occurring in less than 5% of cases. This case report presents the clinical presentation, diagnosis, and management of muscle echinococcosis.

Case presentation: A 28-year-old Black African female presented with a 4-month history of left anteromedial thigh swelling. Despite mild burning pain, she remained functional. While pyomyositis was initially considered, ultrasound and magnetic resonance imaging suggested hydatid disease. Surgical excision was performed, and histopathology confirmed the diagnosis. The patient received albendazole and praziquantel for 4 and 2 weeks, respectively, as adjunctive therapy. A year-long follow-up revealed good progress and continued health.

Conclusion: Primary muscle hydatid cysts should be considered in the differential diagnosis of cystic, painless, localized masses in the muscular system, especially in endemic regions. Serological tests and imaging techniques are essential for investigation. Complete surgical excision and anthelminthic therapy should be pursued when possible.

简介:细粒棘球绦虫是一种绦虫,可引起包虫病。包虫囊肿很少影响肌肉骨骼系统,发生在不到5%的病例。本病例报告介绍肌包虫病的临床表现、诊断和治疗。病例介绍:一名28岁的非洲黑人女性,左侧大腿前内侧肿胀4个月。尽管有轻微的灼痛,但她仍能正常工作。虽然最初认为是脓性肌炎,但超声和磁共振成像提示是包虫病。手术切除,组织病理学证实了诊断。患者分别给予阿苯达唑和吡喹酮辅助治疗4周和2周。一年的随访显示进展良好,健康状况持续。结论:原发性肌包虫病在肌肉系统的囊性、无痛性、局限性肿块鉴别诊断中应予以重视,特别是在流行地区。血清学测试和成像技术是必不可少的调查。如有可能,应进行完全手术切除和驱虫治疗。
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引用次数: 0
Management of patient with acute lymphocytic myocarditis and congenital long QT syndrome presenting with electrical storm and incessant Torsade de Pointes: a case report. 急性淋巴细胞性心肌炎合并先天性长QT综合征并发电风暴和不间断角扭转1例的处理。
IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-20 DOI: 10.1186/s13256-024-04919-z
Giky Karwiky, Raymond Pranata, Alberta Claudia Undarsa, Mohamad Iqbal, Hawani Sasmaya Prameswari, Mohammad Rizki Akbar

Background: This case highlights the management of concomitant acute myocarditis and congenital long QT syndrome with electrical storm and incessant Torsade de Pointes.

Case presentation: An 18 years-old Southeast Asian para 1 abortus 0 (P1A0) postpartum patient with cesarean section owing to severe preeclampsia, acute lymphocytic myocarditis, and prolonged QT interval owing to long QT syndrome. She has incessant Torsade de Pointes treated with beta-blocker, lidocaine, overdrive pacing with a temporary transvenous pacemaker, left cardiac sympathetic denervation per video-assisted thoracoscopic surgery, and implantable cardioverter-defibrillator implantation. We initially used bisoprolol, then switched to propranolol and finally to carvedilol, which reduced the Torsade de Pointes frequency. The longest QTc interval was 696 ms, and the shortest was 624 ms, 2 months after initial corticosteroid administration and left cardiac sympathetic denervation. Device interrogation at 9 months follow up showed three episodes of ventricular fibrillation, 2 spontaneously resolved and one necessitates shock.

Conclusion: Management of concomitant acute myocarditis and congenital long QT syndrome with incessant Torsade de Pointes requires beta-blockers, anti-inflammatory drugs, autonomic modulation, and short-term measures, such as overdrive pacing with deep sedation. Implantable cardioverter-defibrillator is vital to prevent sudden cardiac death.

背景:本病例强调了急性心肌炎和先天性长QT综合征合并电风暴和不间断角扭转的处理。病例介绍:一例18岁东南亚产妇,因严重子痫前期、急性淋巴细胞性心肌炎和长QT间期综合征导致的QT间期延长而行剖宫产。她持续接受β受体阻滞剂、利多卡因治疗,使用临时经静脉起搏器进行过度起搏,通过电视胸腔镜手术进行左心交感神经去神经,以及植入式心律转复除颤器植入。我们一开始用的是比索洛尔,后来改用心得安,最后改用卡维地洛,这减少了扭转角的频率。QTc间隔最长为696 ms,最短为624 ms,均发生在皮质类固醇初始给药和左心交感神经去支配2个月后。随访9个月时,器械检查显示3次心室颤动,2次自行消退,1次需要休克。结论:合并急性心肌炎和先天性长QT综合征合并持续性角扭转需要β受体阻滞剂、抗炎药物、自主调节和短期措施,如深度镇静的过度起搏。植入式心律转复除颤器对预防心源性猝死至关重要。
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Journal of Medical Case Reports
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