Pub Date : 2024-12-21DOI: 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.
{"title":"Successful treatment of tumor lysis syndrome associated with hepatic artery infusion chemotherapy in a patient with hepatocellular carcinoma: a case report.","authors":"Miao Li, Ying-Ting Zhou, Bi-Wei Yang","doi":"10.1186/s13256-024-04928-y","DOIUrl":"10.1186/s13256-024-04928-y","url":null,"abstract":"<p><strong>Background: </strong>Tumor lysis syndrome is a life-threatening complication in the treatment of cancer. However, it rarely occurs in solid tumors, especially in hepatocellular carcinoma.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusions: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"634"},"PeriodicalIF":0.9,"publicationDate":"2024-12-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11662854/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872299","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}
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.
{"title":"Polyarteritis nodosa presenting with severe upper gastrointestinal bleeding: a case report.","authors":"Xin-Yue Xiao, Yun Niu, Ping-Ping Liu, Yu-Ming Zheng, Ming-Gang Zhang, Guo-Chun Wang, Xiao-Di Wang, Fang Liu","doi":"10.1186/s13256-024-04892-7","DOIUrl":"10.1186/s13256-024-04892-7","url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"616"},"PeriodicalIF":0.9,"publicationDate":"2024-12-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11662699/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872289","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}
Pub Date : 2024-12-21DOI: 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.
{"title":"Spinal schwannoma presenting with intraspinal hematoma: a case report and review of the literature.","authors":"Zhe Shen, Haoyang Chen, Haosheng Wang, Yaoqi Wang, Tao Li","doi":"10.1186/s13256-024-04968-4","DOIUrl":"10.1186/s13256-024-04968-4","url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"633"},"PeriodicalIF":0.9,"publicationDate":"2024-12-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11662569/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872295","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}
Pub Date : 2024-12-21DOI: 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.
{"title":"Postorgasmic illness syndrome benefiting from omalizumab and antidepressant: a case report.","authors":"Kasım Okan, Ragıp Fatih Kural, Meryem Demir, Furkan Cot, Mustafa Dinckal, Sevgi Altay, Nihal Mete Gökmen","doi":"10.1186/s13256-024-04986-2","DOIUrl":"10.1186/s13256-024-04986-2","url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"632"},"PeriodicalIF":0.9,"publicationDate":"2024-12-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11662807/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872293","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}
Pub Date : 2024-12-20DOI: 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.
{"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}
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.
{"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}
Pub Date : 2024-12-20DOI: 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.
{"title":"Acute kidney injury and tacrolimus toxicity in a kidney transplant recipient treated with nirmaltrevir/ritonavir: a case report.","authors":"Jack Rycen, Julia Jefferis, David Mudge","doi":"10.1186/s13256-024-04990-6","DOIUrl":"10.1186/s13256-024-04990-6","url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"629"},"PeriodicalIF":0.9,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660612/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142872269","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}
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.
{"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}
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.
{"title":"Intermuscular hydatid cyst in the thigh musculature: a case report.","authors":"Aderaw Getie, Bahiru Atinafu, Misganaw Alemu, Genetu Belay, Dereje Bedane Hunde","doi":"10.1186/s13256-024-04861-0","DOIUrl":"10.1186/s13256-024-04861-0","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"627"},"PeriodicalIF":0.9,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11661042/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142864548","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}
Pub Date : 2024-12-20DOI: 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.
{"title":"Management of patient with acute lymphocytic myocarditis and congenital long QT syndrome presenting with electrical storm and incessant Torsade de Pointes: a case report.","authors":"Giky Karwiky, Raymond Pranata, Alberta Claudia Undarsa, Mohamad Iqbal, Hawani Sasmaya Prameswari, Mohammad Rizki Akbar","doi":"10.1186/s13256-024-04919-z","DOIUrl":"10.1186/s13256-024-04919-z","url":null,"abstract":"<p><strong>Background: </strong>This case highlights the management of concomitant acute myocarditis and congenital long QT syndrome with electrical storm and incessant Torsade de Pointes.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":16236,"journal":{"name":"Journal of Medical Case Reports","volume":"18 1","pages":"603"},"PeriodicalIF":0.9,"publicationDate":"2024-12-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660443/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142864555","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}