American Journal of Case Reports最新文献_第5页

Bilateral Knee Synovial Chondromatosis in a 2-Year-Old Girl: First Reported Case and Literature Review.

IF 1

American Journal of Case Reports Pub Date : 2025-03-19 DOI: 10.12659/AJCR.945921

Ana Paula Fernandes Barbosa, Paulyana Fernandes Barbosa Quintino, Hermann Fernandes Motta Camara, Pedro Henrique Laurindo Dos Anjos, Arthur Castro Guimaraes, Eloisio Barbosa Lopes Neto, Maria Eugenia Leite Duarte

{"title":"Bilateral Knee Synovial Chondromatosis in a 2-Year-Old Girl: First Reported Case and Literature Review.","authors":"Ana Paula Fernandes Barbosa, Paulyana Fernandes Barbosa Quintino, Hermann Fernandes Motta Camara, Pedro Henrique Laurindo Dos Anjos, Arthur Castro Guimaraes, Eloisio Barbosa Lopes Neto, Maria Eugenia Leite Duarte","doi":"10.12659/AJCR.945921","DOIUrl":"10.12659/AJCR.945921","url":null,"abstract":"BACKGROUND Synovial chondromatosis (SC) is a rare monoarticular benign condition characterized by the development of intra- or periarticular cartilage-like nodules or loose bodies. Typically affecting middle-aged men, SC is exceptionally rare in female pediatric patients. Symptoms include diffuse pain, swelling, and limited joint movement. The loose bodies are typically numerous, small, rounded, and uniform in size. Histologically, they consist of hyaline cartilage-like nodules containing clusters of chondrocytes. Bilateral SC in the knee of young children is even rarer and has not been previously documented in the medical literature. CASE REPORT A 2-year-old girl presented with recent pain in her right knee without preceding trauma. Physical examination revealed joint effusion, a limping gait, and limited knee flexion, with no signs of local inflammation. Radiographs showed normal joint structures, and laboratory test results, except for an elevated CRP, were normal. Joint aspiration ruled out septic arthritis. The worsening of symptoms within a relatively short period prompted further investigation, leading to the identification of numerous cartilaginous nodules within the joint during exploratory arthrotomy. Microscopic examination revealed unossified cartilage-like nodules containing disorganized chondrocytes. Four months after surgery, she was diagnosed with SC in the contralateral knee. CONCLUSIONS Given the rarity of synovial chondromatosis in young children, and the even rarer bilateral presentation in a large joint, this case report provides an opportunity to explore the challenges in diagnosing and managing such conditions in pediatric patients. To the best of our knowledge, this is the youngest reported case of bilateral SC of the knee in a pediatric patient.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945921"},"PeriodicalIF":1.0,"publicationDate":"2025-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11932548/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143664885","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

Uncommon Presentation of Perforated Appendicitis: Abdominal Wall Abscess and Fistula Formation.

IF 1

American Journal of Case Reports Pub Date : 2025-03-18 DOI: 10.12659/AJCR.946543

Zachary S Kauffman, David L Stuart

{"title":"Uncommon Presentation of Perforated Appendicitis: Abdominal Wall Abscess and Fistula Formation.","authors":"Zachary S Kauffman, David L Stuart","doi":"10.12659/AJCR.946543","DOIUrl":"10.12659/AJCR.946543","url":null,"abstract":"BACKGROUND Acute appendicitis is a common surgical emergency, and perforated appendix is one potential complication. Acute appendicitis can be complicated by perforation and peritonitis, but chronic abscess formation is less common. This report presents the case of a 45-year-old woman with a 7-day history of right lower-abdominal pain and swelling due to perforated acute appendicitis and abdominal wall abscess that required laparotomy and drainage. The presentation of this particular case is unique in that a fistulous tract formed subsequent to perforation of the appendix, with the resultant abscess forming in the abdominal wall. The current case study serves to showcase the diagnostic challenges associated with such a presentation. CASE REPORT A 45-year-old woman presented to the emergency department with a 7-day history of right lower-quadrant and midline lower-abdominal pain and swelling. Computed tomography (CT) scans with intravenous (IV) and rectal contrast showed an abdominal wall abscess with no signs of obstruction, perforation, or appendicitis. Incision and drainage of the abdominal wall abscess with debridement of the abdominal wall was complicated by peritoneal adhesions, and open laparotomy was thus performed. Upon entry into the abdominal cavity, the appendix was found to be adherent to the abdominal wall. It was noted that the appendix had perforated, allowing for fistula formation with the abdominal wall. CONCLUSIONS Clinicians should maintain a high index of suspicion for perforated appendicitis in cases of abdominal wall abscesses with leukocytosis and right lower-quadrant pain, even when initial imaging does not show obvious appendicitis.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946543"},"PeriodicalIF":1.0,"publicationDate":"2025-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11928066/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143658969","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

Functional Outcomes of Humeral Diaphyseal Hip Spacer in Tumor Resection: A Case Report.

IF 1

American Journal of Case Reports Pub Date : 2025-03-18 DOI: 10.12659/AJCR.946571

Javier Gutierrez-Pereira, Antonio Garcia-Lopez

{"title":"Functional Outcomes of Humeral Diaphyseal Hip Spacer in Tumor Resection: A Case Report.","authors":"Javier Gutierrez-Pereira, Antonio Garcia-Lopez","doi":"10.12659/AJCR.946571","DOIUrl":"10.12659/AJCR.946571","url":null,"abstract":"BACKGROUND Aggressive surgical resection is essential for managing malignant tumors involving the proximal humerus and scapula. Reconstruction of these defects presents a substantial therapeutic challenge, with functional preservation as the primary objective. Traditionally, a simple suspension technique connecting the humeral remnant to the clavicle has been utilized. While allografts and prosthetic replacements offer potential benefits, they are often associated with serious complications, such as infection, resorption or dislocation. This study aimed to assess the outcomes of a novel surgical technique for reconstructing the scapular and proximal humeral regions following sarcoma resection. CASE REPORT We present 2 cases involving patients diagnosed with dedifferentiated chondrosarcoma and fibrosarcoma in the scapulohumeral region who underwent radical tumor excision followed by a novel joint reconstruction technique. In each case, a humeral diaphyseal hip spacer with dual antibiotic-loaded cement was used. A new joint capsule was constructed with Trevira mesh, affixed to both the clavicle and the second costal arch to anchor the remaining structures. Functional outcomes were evaluated using the Musculoskeletal Tumor Society (MSTS) score and the Toronto Extremity Salvage Score (TESS). Both patients achieved favorable clinical outcomes, with disease-free resection margins, satisfactory aesthetic outcomes, and acceptable postoperative shoulder contour and function. CONCLUSIONS Joint reconstruction using a cemented humeral diaphyseal hip spacer anchored to the clavicle with Trevira mesh restores structural integrity and partially recovers glenohumeral function. This technique also addresses aesthetic deficits associated with extensive scapulohumeral tumor resection, presenting a promising alternative for functional and cosmetic rehabilitation due to the structural offset provided by the hip spacer. However, larger studies are necessary to validate these results.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946571"},"PeriodicalIF":1.0,"publicationDate":"2025-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11927925/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143650188","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

Self-Assembling Hemostatic Peptide Gel Used to Successfully Treat Rectal Ulcer in Radiation-Associated Vascular Ectasia: A Case Report.

IF 1

American Journal of Case Reports Pub Date : 2025-03-17 DOI: 10.12659/AJCR.946886

Rishi Bolla, Shaffer Mok, Mark Friedman, Aamir Dam, Anjuli K Luthra, Saraswathi Cappelle, Luis R Peña

{"title":"Self-Assembling Hemostatic Peptide Gel Used to Successfully Treat Rectal Ulcer in Radiation-Associated Vascular Ectasia: A Case Report.","authors":"Rishi Bolla, Shaffer Mok, Mark Friedman, Aamir Dam, Anjuli K Luthra, Saraswathi Cappelle, Luis R Peña","doi":"10.12659/AJCR.946886","DOIUrl":"10.12659/AJCR.946886","url":null,"abstract":"BACKGROUND Radiation-associated vascular ectasia, formerly known as radiation proctitis, is a known gastrointestinal complication of radiation therapy. Rectal ulcers in the setting of radiation-associated vascular ectasia are particularly challenging to treat. They are often refractory to conventional medical and endoscopic therapies and cause significant morbidity for patients. Self-assembling hemostatic peptide gel is an endoscopic tool used primarily for hemostasis but has also been hypothesized to have mucosal repair properties. CASE REPORT A 41-year-old woman with a history of cervical adenocarcinoma presented with abdominal pain and rectal bleeding several months after brachytherapy and external beam radiation therapy. Conservative therapy with topical pramoxine was ineffective, so she underwent colonoscopy, which revealed a non-bleeding rectal ulcer in the setting of bleeding radiation-associated vascular ectasia. Argon plasma coagulation was used to treat the bleeding vascular ectasia, but the ulcer was treated solely with self-assembling hemostatic peptide gel. A follow-up endoscopy two months later showed complete resolution of the ulcer. CONCLUSIONS The mucosal repair properties of the self-assembling hemostatic peptide gel, an endoscopic product initially designed for hemostasis, are highlighted by this case in which a non-hemorrhagic ulcer was successfully treated. Furthermore, this is a novel use of the self-assembling hemostatic peptide gel in the setting of radiation-associated vascular ectasia and may represent a useful addition to the endoscopist's toolkit for managing a challenging condition.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946886"},"PeriodicalIF":1.0,"publicationDate":"2025-03-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11927923/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143650211","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

Enzyme Replacement and Immunosuppression in Heart Transplant Recipients with Fabry Cardiomyopathy: A 7-Year Case Study.

IF 1

American Journal of Case Reports Pub Date : 2025-03-17 DOI: 10.12659/AJCR.945873

Katarzyna Muras-Szwedziak, Maciej Wójcik, Olga Wojtyczka, Krzysztof Kaczmarek

{"title":"Enzyme Replacement and Immunosuppression in Heart Transplant Recipients with Fabry Cardiomyopathy: A 7-Year Case Study.","authors":"Katarzyna Muras-Szwedziak, Maciej Wójcik, Olga Wojtyczka, Krzysztof Kaczmarek","doi":"10.12659/AJCR.945873","DOIUrl":"10.12659/AJCR.945873","url":null,"abstract":"BACKGROUND Fabry disease (FD) is a rare storage disorder caused by the absence or deficiency of alpha-galactosidase A, which leads to accumulation of glycosphingolipids in tissues, chronic inflammation, fibrosis, and multiple-organ dysfunction. There are only a few cases of orthotopic heart transplantation (OHT) in patients with FD in the literature. This report shows the manifestation of FD, its course in OHT recipient and outcome of 4-year enzyme replacement therapy (ERT) combined with post-transplant immunosuppression. CASE REPORT A 54-year-old female patient had cardiomegaly revealed by a chest X-ray. Subsequent diagnostics included transthoracic echocardiography (TTE), which showed left ventricular hypertrophy. Despite typical therapeutic management, she had progression of heart failure (HF) symptoms. After 6 years, cardiac magnetic resonance imaging (MRI) showed myocardial fibrosis. This finding and TTE analysis led to suspicion of storage disease with cardiac involvement. Further biochemical and genetic diagnostics showed low activity of alpha-galactosidase A and detected mutations of the GLA gene. Five years after the first cardiac symptoms, Fabry cardiomyopathy was diagnosed. Due to HF progression, she underwent OHT with subsequent standard immunosuppressive treatment. As soon as ERT was available in Poland, it was included to the treatment. Currently, there is no evidence of cardiac involvement of FD or graft rejection. CONCLUSIONS Severe HF not responding to medical therapy in patients with FD can be successfully treated with cardiac transplantation. The risk of disease recurrence in the graft appears to be low. The anti-inflammatory action of post-transplant immunosuppression may play a role in that phenomenon, but further research is needed to confirm this hypothesis.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945873"},"PeriodicalIF":1.0,"publicationDate":"2025-03-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11927924/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143651308","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

Pseudoaneurysm-Induced Pain Following Ankle Arthroscopy: A Case Report and Literature Review.

IF 1

American Journal of Case Reports Pub Date : 2025-03-16 DOI: 10.12659/AJCR.945540

Young Uk Park, DaeHyun Han, Juho An, Young Wook Seo

{"title":"Pseudoaneurysm-Induced Pain Following Ankle Arthroscopy: A Case Report and Literature Review.","authors":"Young Uk Park, DaeHyun Han, Juho An, Young Wook Seo","doi":"10.12659/AJCR.945540","DOIUrl":"10.12659/AJCR.945540","url":null,"abstract":"BACKGROUND Arthroscopic surgery is esteemed for its arthroscopic lateral ligament repair to treating ankle ligament injuries, characterized by a low complication rate. Nevertheless, rare complications such as pseudoaneurysms require careful monitoring and a strategic approach to management during the postoperative follow-up period. This case report details a 42-year-old man with chronic instability in his left ankle. The aim of this paper is to present a case report and conduct a literature review on the rare complication of pseudoaneurysm following arthroscopic procedures. CASE REPORT A 42-year-old man with chronic instability in his left ankle was experiencing severe pain and limited motion following arthroscopic lateral ligament repair surgery. He was diagnosed with a pseudoaneurysm of the left anterior tibial artery and an intra-articular hematoma in the left ankle joint, indicated by MRI and Doppler ultrasound findings. Initial conservative management was followed by interventional procedures, including thrombin injection and balloon tamponade. After these interventions proved unsuccessful, surgical intervention for anterior tibial artery reconstruction and hematoma evacuation was performed. The surgical interventions resulted in the closure of the pseudoaneurysm and improvement in the patient's pain. CONCLUSIONS This case underscores the challenges involved in diagnosing and managing pseudoaneurysm and hematoma after ankle surgery, highlighting the necessity for thorough evaluation and a comprehensive treatment strategy.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945540"},"PeriodicalIF":1.0,"publicationDate":"2025-03-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11927926/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143634849","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

Incidental High-Grade Sellar Solitary Fibrous Tumor Mimicking Non-Functioning Pituitary Adenoma: A Case Report and Literature Review. 模仿无功能垂体腺瘤的偶发高级别ellar孤立性纤维瘤：病例报告和文献综述。

IF 1

American Journal of Case Reports Pub Date : 2025-03-15 DOI: 10.12659/AJCR.946592

Michele Persico, Brent D Weinberg, Stewart G Neill, Hui-Kuo Shu, Jim Zhong

{"title":"Incidental High-Grade Sellar Solitary Fibrous Tumor Mimicking Non-Functioning Pituitary Adenoma: A Case Report and Literature Review.","authors":"Michele Persico, Brent D Weinberg, Stewart G Neill, Hui-Kuo Shu, Jim Zhong","doi":"10.12659/AJCR.946592","DOIUrl":"10.12659/AJCR.946592","url":null,"abstract":"BACKGROUND Sellar solitary fibrous tumors (SFT) are uncommon brain tumors belonging to the fibroblastic and myofibroblastic category of the 2021 World Health Organization (WHO) Classification of Central Nervous System (CNS) Tumors. Twenty-two instances of sellar WHO grade II and III SFT (SFT II/III) have been reported since 1983. SFTs II/III clinical and imaging findings resemble non-functioning pituitary adenomas (NFPAs). We report a 62-year-old woman with encephalopathy and an incidental WHO grade III sellar SFT presenting as a non-functioning pituitary mass. CASE REPORT A 62-year-old woman with encephalopathy and urosepsis was transferred for evaluation of an incidentally detected sellar mass. An endocrine workup was unremarkable and a presumed diagnosis of NFPA was made. A partial adenectomy was then performed through a trans-nasal trans-sphenoidal approach. Pathological analysis revealed a mesenchymal neoplasm with haphazardly arranged spindled to epithelioid cells, collagenized background, tortuous vasculature, high mitotic index, and Signal Transducer and Activator of Transcription 6 (STAT6) positivity. According to the 2016 CNS WHO guidelines, the diagnosis was an anaplastic hemangiopericytoma WHO grade III, now termed an SFT under the 2021 CNS WHO guidelines. Proton therapy to the dose of 59.4 gray given in 33 fractions was implemented adjuvantly. The residual tumor demonstrated shrinkage 1.5 years after proton therapy, and absence of recurrent disease after 3 years. CONCLUSIONS Sellar SFTs II/III can present as incidentalomas and be considered in the differential diagnosis of sellar masses. While some cause symptoms by local expansion mimicking NFPA presentations, local invasion and metastasis are reported. Surgical excision and pathologic confirmation guide management.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946592"},"PeriodicalIF":1.0,"publicationDate":"2025-03-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11918454/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143634847","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

Sustained Immunotherapy Response in Metastatic Brain Melanoma Through 2 Pregnancies.

IF 1

American Journal of Case Reports Pub Date : 2025-03-14 DOI: 10.12659/AJCR.945533

Marcin Rajczykowski, Magdalena Olbryt, Katarzyna Galwas, Adam Idasiak, Ewa Stobiecka, Rafał Suwiński

{"title":"Sustained Immunotherapy Response in Metastatic Brain Melanoma Through 2 Pregnancies.","authors":"Marcin Rajczykowski, Magdalena Olbryt, Katarzyna Galwas, Adam Idasiak, Ewa Stobiecka, Rafał Suwiński","doi":"10.12659/AJCR.945533","DOIUrl":"10.12659/AJCR.945533","url":null,"abstract":"BACKGROUND Metastatic brain melanoma is a deadly form of cancer with a high mortality rate and short overall survival. Immunotherapy with immune checkpoint inhibitors is the first treatment option for BRAF wild-type patients. Pregnancy is the exclusion criterion for immunotherapy and may promote the progression of melanoma. This report shows the long-lasting response of a patient with metastasis in multiple locations, including the brain, to immunotherapy and radiotherapy, who delivered 2 healthy boys during the disease. CASE REPORT A 39-year-old woman was diagnosed with BRAF(-)/NRAS(+) skin melanoma, pT2bN2aM0 (IIIB). Due to pregnancy, she did not receive adjuvant therapy. Upon delivery, the disease manifested with multiple extracranial and symptomatic brain metastasis. She was treated with whole-brain radiation and immunotherapy with ipilimumab and nivolumab followed by nivolumab. A partial response of the brain metastases and an extracranial complete response were observed. During the immunotherapy, she became pregnant and the therapy was discontinued. She was under regular medical surveillance, during which she delivered a healthy boy. The last CT scan and magnetic resonance brain examination showed a maintenance response for 43 months after initiation of immunotherapy and 31 months after therapy completion. CONCLUSIONS A long-lasting response to radiotherapy and interrupted immunotherapy is possible in the case of symptomatic metastatic brain melanoma developing during pregnancy, and healthy deliveries are possible despite the mother's progressive melanoma or exposure of the fetus to nivolumab (first trimester).","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945533"},"PeriodicalIF":1.0,"publicationDate":"2025-03-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11918453/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143626385","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

Dextromethorphan Overdose with Refractory Status Epilepticus and Reversible Cranial Nerve Reflex Loss: A Case Report.

IF 1

American Journal of Case Reports Pub Date : 2025-03-13 DOI: 10.12659/AJCR.946447

Akifumi Okamoto, Naoki Yonezawa, Kazuhiro Yoshizawa, Reiki Kumashiro, Shinya Suzuki

{"title":"Dextromethorphan Overdose with Refractory Status Epilepticus and Reversible Cranial Nerve Reflex Loss: A Case Report.","authors":"Akifumi Okamoto, Naoki Yonezawa, Kazuhiro Yoshizawa, Reiki Kumashiro, Shinya Suzuki","doi":"10.12659/AJCR.946447","DOIUrl":"10.12659/AJCR.946447","url":null,"abstract":"BACKGROUND Dextromethorphan is a widely used over-the-counter antitussive medication. Generally safe within the recommended dosages, its misuse can lead to severe adverse effects, particularly in large amounts. However, comprehensive descriptions of severe overdose cases - including pharmacokinetic data of dextromethorphan and its active metabolite (dextrorphan) - are scarce. CASE REPORT A 21-year-old woman with schizophrenia ingested 20 280 mg of dextromethorphan along with other prescribed medications during a suicide attempt. She was discovered semiconscious and experienced a generalized seizure en route to the hospital. Despite intensive treatments - including endotracheal intubation and administration of propofol and midazolam - she continued to experience refractory status epilepticus. Subsequent neurological examination revealed cranial nerve reflex loss. The serum concentration of dextromethorphan was 5.8 mg/L on admission and decreased to 2.2 mg/L by day 1 and 1.1 mg/L by day 2, contrasting with that of dextrorphan, which remained within the therapeutic limits. By day 4, her condition stabilized; she became alert, responsive to commands, and was successfully extubated. She was discharged on day 9 without any sequelae. CONCLUSIONS This report describes the case of a patient who survived a massive dextromethorphan overdose, who presented with refractory status epilepticus followed by reversible suppression of cranial nerve reflexes. The pharmacokinetic profiles suggested that dextromethorphan, rather than dextrorphan, was responsible for the symptoms. High-dose dextromethorphan ingestion can lead to varied and potentially fatal outcomes, especially when compounded by metabolism-altering factors such as CYP2D6 inhibition, genetic variability, or co-ingested medications. This case underscores the importance of prompt, intensive supportive care in managing severe dextromethorphan toxicity.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946447"},"PeriodicalIF":1.0,"publicationDate":"2025-03-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11918451/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143617543","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

Thymoma-Induced Severe Biventricular Failure without Myasthenia Gravis: Investigating Tachycardia-Induced Cardiomyopathy.

IF 1

American Journal of Case Reports Pub Date : 2025-03-12 DOI: 10.12659/AJCR.945796

Roscoe Lim, Stephanie Wiltshire, Megan Barnet, Julia P Low, Samuel Bolitho, Alisa Kane, Andrew Jabbour, Eugene Kotlyar, Christopher Hayward

{"title":"Thymoma-Induced Severe Biventricular Failure without Myasthenia Gravis: Investigating Tachycardia-Induced Cardiomyopathy.","authors":"Roscoe Lim, Stephanie Wiltshire, Megan Barnet, Julia P Low, Samuel Bolitho, Alisa Kane, Andrew Jabbour, Eugene Kotlyar, Christopher Hayward","doi":"10.12659/AJCR.945796","DOIUrl":"10.12659/AJCR.945796","url":null,"abstract":"BACKGROUND Cardiomyopathy associated with thymoma is thought to be a cardiac manifestations of myasthenia gravis (MG). However, there are case reports of newly diagnosed thymoma presenting with cardiomyopathy without MG, and the mechanism remains unclear. The purpose of this report is to explore tachycardia-induced cardiomyopathy (TIC) as a potential mechanism for cardiomyopathy in thymoma without features of MG. CASE REPORT A 31-year-old man presented with atrial flutter with right bundle branch block and severe biventricular heart failure. Echocardiogram revealed severe left ventricle ejection fraction (LVEF) of 15% with biventricular dilation with impaired systolic function. Computer tomography coronary angiography demonstrated normal coronary artery disease. Cardiac magnetic resonance imaging showed normal T1 and T2 mapping, without inflammation or edema. A large anterior mediastinal mass was found on computer tomography chest. Mediastinal mass biopsy identified type B3 thymoma (WHO classification) with dual population of large, uniform epithelial thymic cells and immature T cell phenotype. Acetylcholine receptor antibody was positive without clinical features of MG and hypogammaglobulinemia indicating Good syndrome. He was treated with antiarrhythmic and heart failure pharmacotherapy, carboplatin and paclitaxel, and intravenous immunoglobulin. He demonstrated reversible heart failure following abolishment of tachyarrhythmia, consistent with tachycardia-induced cardiomyopathy. CONCLUSIONS We report a rare case of a newly diagnosed thymoma and Good syndrome without clinical features of MG presenting with tachyarrhythmia and severe biventricular failure. The reversibility of the cardiomyopathy following abortion of tachyarrhythmia with treatment highlights TIC as a potential cause.","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945796"},"PeriodicalIF":1.0,"publicationDate":"2025-03-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11918452/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143606606","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