American Journal of Case Reports最新文献

{"title":"Imatinib-Induced Clinical Response in ETV6::ACSL6 Myeloid Neoplasm with Eosinophilic Pneumonitis: A Case Report.","authors":"Fieke W Hoff, Sharon Germans, Olga K Weinberg, Robert H Collins, Rolando García, Weina Chen, Miguel D Cantu, Mingyi Chen, Prasad Koduru, Jeffrey SoRelle, Yazan F Madanat, Stephen S Chung","doi":"10.12659/AJCR.946517","DOIUrl":"https://doi.org/10.12659/AJCR.946517","url":null,"abstract":"<p><p>BACKGROUND Myeloid neoplasms with [em]ETV6::ACSL6[/em] fusions are extremely rare entities that are characterized by eosinophilic and/or basophilic leukocytosis. While they clinically mimic myeloid neoplasms with eosinophilia and tyrosine kinase fusions such as [em]ETV6::PDGFRB[/em], they have not been shown to be responsive to imatinib. There are currently no effective treatments available and clinical outcomes are poor. CASE REPORT We report a rare case of a 71-year-old man with a history of myelodysplastic syndrome/neoplasms (MDS) with mutated [em]SF3B1[/em] and multilineage dysplasia treated with luspatercept followed by azacitidine. However, he developed clonal evolution of disease to MDS with hypereosinophilia. Chromosome analysis identified t(5;12)(q31;p13). Fluorescence in situ hybridization was negative for [em]FIP1L1/PGFFRA[/em] or [em]PDGFRB[/em] gene rearrangement, but RNA-sequencing identified the [em]ETV6::ACSL6[/em] fusion. He received a hematopoietic cell transplantation with achievement of complete remission but subsequently relapsed, with chromosome analysis again revealing t(5;12)(q31;p13) [[em]ETV6::ACSL6[/em]]. He rapidly clinically deteriorated and developed refractory respiratory failure due to acute eosinophilic pneumonitis. He received a prolonged course of high-dose steroids without adequate improvement of the eosinophilia. Based on reports showing good response to tyrosine kinase inhibitors in patients with the [em]ETV6::PDGFRB[/em] fusion, treatment was switched to imatinib, leading to rapid normalization of absolute eosinophil counts, with clinical improvement. CONCLUSIONS Our findings suggest that imatinib should be considered for patients with a myeloid neoplasm with an [em]ETV6::ACSL6[/em] fusion who are refractory to corticosteroids. Further molecular investigations are needed to elucidate the underlying mechanism of imatinib sensitivity in [em]ETV6::ASCL6[/em]-associated disease, given the absence of genetic involvement of a tyrosine kinase.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946517"},"PeriodicalIF":1.0,"publicationDate":"2025-04-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143812638","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Acremonium strictum Sphenoid Sinusitis in an Immunocompetent Patient.","authors":"Yuting Wang, Qingzhong Liu, Huiming Sheng, Shuohui Yang","doi":"10.12659/AJCR.946501","DOIUrl":"https://doi.org/10.12659/AJCR.946501","url":null,"abstract":"<p><p>BACKGROUND Acremonium strictum (A. strictum) is an opportunistic fungus that is a rare environmental and saprophytic pathogen. Because of its rarity and inadequate mastery of clinicians, A. strictum sphenoid sinusitis becomes a refractory disease in some immunocompetent patients. Moreover, its etiologic and radiological diagnoses are challenging, leading to inappropriate treatment and a protracted course. CASE REPORT We report a case of A. strictum sphenoid sinusitis in an immunocompetent 74-year-old female patient. She had dizziness and head distension accompanied by intermittent vomiting for 3 months. Computed tomography (CT) suggested a chronic noninvasive fungal sphenoid sinusitis. She underwent a resection of the lesion and postoperative anti-infection therapy. Two months later, the lesions reappeared and were confirmed by CT again. Microbiological culture of purulent tissue revealed a growth of filamentous fungus identified as A. strictum by 26S rRNA sequencing. After 4 weeks of oral voriconazole treatment, the patient recovered and had not relapsed at 2-year follow-up. CONCLUSIONS The rare occurrence of A. strictum as a causative agent of sphenoid sinusitis underscores the importance of utilizing radiological imaging, microbiological culture, and molecular diagnostic techniques to enhance diagnostic accuracy. This case emphasizes the need for heightened clinical suspicion and targeted antifungal therapy to manage fungal infections effectively.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946501"},"PeriodicalIF":1.0,"publicationDate":"2025-04-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143804333","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Co-Occurrence of Systemic Sclerosis, Lupus, and Celiac Disease in Multiple Autoimmune Syndrome: A Case Report.","authors":"Jawaher Saleh Enani, Abdulaziz Aljasser, Nuha N Alrajhi, Salman Saad Alsaleh, Mohammed A Omair","doi":"10.12659/AJCR.945176","DOIUrl":"https://doi.org/10.12659/AJCR.945176","url":null,"abstract":"<p><p>BACKGROUND Multiple autoimmune syndromes are caused by immune dysregulation pathways and shared genetic polymorphisms, resulting in the coexistence of multiple autoimmune disorders. Systemic sclerosis (SSc) and systemic lupus erythematosus (SLE) are connective tissue diseases with distinct pathogenesis. Celiac disease (CD) is an immune-mediated small intestine pathology. Previous case reports have documented the coexistence of SLE and CD, SSc and CD, and SSc and SLE (overlap syndrome). CASE REPORT A 21-year-old woman with juvenile SSc and interstitial lung disease was admitted with fever, cough, and ongoing lower abdominal pain, diarrhea, and weight loss for the previous 3 months. Laboratory investigations revealed leukopenia, normocytic normochromic anemia, and thrombocytopenia, with positive antinuclear antibody and anti-double-stranded DNA. SLE was diagnosed, and the patient was started on a steroid and hydroxychloroquine. Celiac serology was ordered, followed by an upper gastrointestinal endoscopy, with biopsy. The results of both tests indicated CD. The patient was advised to follow a gluten-free diet and was started on hydroxychloroquine, mycophenolate mofetil, and prednisolone. CONCLUSIONS Our patient's presentation of CD and SLE occurring 9 years after SSc onset is unique. Individuals with one autoimmune disease have approximately a 25% chance of developing another. Limited case reports discuss CD in patients with SSc and the association between CD and SLE. To the best of our knowledge, no prior reports documented the coexistence of SSc, SLE, and CD. This case report underscores the importance of investigating autoimmune syndromes based on clinical presentation, as rare associations can occur.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945176"},"PeriodicalIF":1.0,"publicationDate":"2025-04-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143812612","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Diagnostic Challenges in Ectopic Pregnancy: Identifying Pseudo Sacs in Suspected Heterotopic Cases.","authors":"Aditya Utomo, Aditiyono Aditiyono, Dina Marlina, Yuda Putra Disastra, Artha Falentin Putri Susilo, Putri Nadhira Adinda Adriansyah","doi":"10.12659/AJCR.944924","DOIUrl":"https://doi.org/10.12659/AJCR.944924","url":null,"abstract":"<p><p>BACKGROUND A pseudo-gestational sac (pseudo sac), or intracavitary fluid, occurs in up to 10% of ectopic tubal pregnancies and can mimic an intrauterine gestational sac, posing a diagnostic challenge. Distinguishing a pseudo sac from a true gestational sac or an ovarian cyst is critical to avoid misdiagnosis. This report describes the case of a 28-year-old woman presenting at 6 weeks of pregnancy with hydrosalpinx, hemosalpinx, and a pseudo sac due to tubal ectopic pregnancy. CASE REPORT A 28-year-old woman, G3P1A1 at 6 weeks of pregnancy, presented with right lower quadrant abdominal pain lasting 4 days and vaginal bleeding that began 10 h before admission. Physical examination revealed cervical motion tenderness and bulging of the Douglas pouch. Ultrasonography showed a suspected heterotopic pregnancy, with a gestational sac in the right adnexa measuring 1.0×7.4×1.9 cm and a \"ring of fire\" sign, alongside free fluid in the Douglas and Morrison pouches. Intraoperative findings confirmed a ruptured ectopic pregnancy in the interstitial portion of the fallopian tube, with approximately 400 cc of blood clots. Bilateral salpingectomy was performed, owing to the ruptured ectopic pregnancy and contralateral hydrosalpinx. Histopathological analysis confirmed the intrauterine finding as a pseudo sac with no conception tissue. CONCLUSIONS This report highlights the importance of distinguishing between true intrauterine pregnancy and a pseudo-gestational sac, to avoid unnecessary interventions. It emphasizes the role of early and precise transvaginal ultrasonography combined with histopathological confirmation in diagnosing ectopic pregnancies, particularly in cases mimicking heterotopic pregnancy.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e944924"},"PeriodicalIF":1.0,"publicationDate":"2025-04-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143804281","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Postpartum Superior Mesenteric Vein Thrombosis and Heparin-Induced Thrombocytopenia: Clinical Insights.","authors":"Xinye Cui, Yuqiang Tang, Guoxin Guan, Zhongtao Zhang, Fuwen Luo","doi":"10.12659/AJCR.947094","DOIUrl":"https://doi.org/10.12659/AJCR.947094","url":null,"abstract":"<p><p>BACKGROUND Women are at an increased risk of developing venous thrombosis during gestation and the postpartum period, due to physiological and anatomical changes. Superior mesenteric vein thrombosis (SMVT) is a rare and severe complication with a dangerous course and a high mortality rate. Heparin-induced thrombocytopenia (HIT) is also rare in patients treated with heparin and presents with thrombosis and thrombocytopenia. We report a patient with SMVT diagnosis who was treated with heparin anticoagulation after surgery and eventually developed HIT. CASE REPORT The patient was a 30-year-old woman with a diagnosis of SMVT 10 days after giving birth to her second child. She was treated with heparin anticoagulation and interventional catheter-based thrombolysis and thrombectomy, which was ineffective, and she showed peritonitis. We therefore performed an emergency laparotomy, during which we found partial small intestine necrosis and performed a partial small bowel resection. However, the systemic thrombosis worsened postoperatively, and after a multidisciplinary team consultation, HIT was diagnosed. After switching to non-heparin anticoagulation, the patient's condition improved, and she was discharged from the hospital. CONCLUSIONS This is the first case report of HIT secondary to heparin anticoagulation in a parturient who underwent surgery for intestinal necrosis caused by SMVT. Our case highlights the challenge of treating SMVT and the difficulty of diagnosis of HIT. SMVT is a serious condition that requires sufficient experience to judge the timing of surgery. HIT is a rare complication that occurs during heparin use, and its timely diagnosis and treatment have a significant impact on patient prognosis.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947094"},"PeriodicalIF":1.0,"publicationDate":"2025-04-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143796625","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Delayed Diagnosis of Peritoneal Mesothelioma in Recurrent Ascites: A Case Report.","authors":"Hafiz Javed, Arfa Ahmad, Abdul Rehman, Michael Wagner, John E McKnight","doi":"10.12659/AJCR.946777","DOIUrl":"https://doi.org/10.12659/AJCR.946777","url":null,"abstract":"<p><p>BACKGROUND Ascites is a common clinical sign noted in 3% to 4% of hospitalized medical patients. Nearly 94% of patients with ascites have non-malignant etiologies, with the most common being chronic liver disease. Peritoneal mesothelioma is an exceedingly rare malignancy associated with asbestos exposure. Here, we report the case of an elderly man who presented with recurrent loculated ascites and received a diagnosis of peritoneal mesothelioma after a delay of approximately 10 weeks. CASE REPORT A 67-year-old man with a history of asbestos exposure presented with abdominal distension and muscle wasting for 1 month. Initial abdominal CT was notable for smooth liver contour and massive ascites. Ascitic fluid analysis was consistent with low serum albumin-ascites gradient but showed negative cytology for malignant cells. Upper and lower endoscopies did not reveal lesions. Due to recurring ascites, serial abdominal paracenteses were performed, although cytology was negative for malignant cells. Repeat abdominal CT performed after 6 weeks showed worsening loculated ascites, with possible peritoneal carcinomatosis. At this point, percutaneous omental biopsy was performed, which established the diagnosis of peritoneal mesothelioma (75 days after initial presentation). CONCLUSIONS In patients presenting with recurrent ascites and a history of asbestos exposure, it is important to consider the diagnosis of peritoneal mesothelioma. While 94% of patients with ascites have non-malignant etiologies, a small proportion of patients can have malignant ascites. In patients in which the first set of ascitic fluid studies are inconclusive, repeat abdominal paracentesis and omental biopsy or diagnostic laparoscopy should be expedited to avoid delays in diagnosis.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946777"},"PeriodicalIF":1.0,"publicationDate":"2025-04-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143796622","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Co-Occurrence of Neurofibromatosis Type 1 and Polycystic Liver Disease: A Case of Hypertension with PKHD1 Variant.","authors":"Yotsapon Thewjitcharoen, Soontaree Nakasatien, Veekij Veerasomboonsin, Sandra T F Tsoi, Cadmon K P Lim, Thep Himathongkam","doi":"10.12659/AJCR.947141","DOIUrl":"10.12659/AJCR.947141","url":null,"abstract":"<p><p>BACKGROUND Neurofibromatosis type 1 (NF1) is a common genetic condition; 0.1-5.7% of patients with NF1 will develop pheochromocytomas in their lifetime. However, other causes of hypertension (HT) in young patients can be present, and polycystic liver disease is not a part of NF1 syndrome. Polycystic liver disease had been described among patients with heterozygotic polycystic kidney and hepatic disease 1 (PKHD1) variant. We report a rare case of a young patient with NF1 who presented with HT and polycystic liver disease. CASE REPORT A 37-year-old Thai woman with history of NF-1 (clinically diagnosed at the age of 20 years from presence of cafe-au-lait spots and neurofibromas) had HT for 2 years without other symptoms. Abdominal computed tomography revealed polycystic liver disease and a simple renal cyst with both adrenal glands normal. Laboratory studies showed normal results. Whole-exome sequencing (WES) confirmed the molecular diagnosis of NF1 with heterozygous pathogenic variants c.5268+1G>A of NF1 and heterozygous pathogenic variants c.7594_7597del of PKHD1 gene. Given the results of genetic testing and no other identified causes of HT, co-occurrence of NF1 and HT-associated heterozygotic PKHD1 variant was diagnosed. CONCLUSIONS Our case highlights the diagnostic challenges of atypical phenotypes among individuals with NF1, which can depend on the background of other genes. With increasing affordability of WES, its utility in uncovering the possibility of being affected by 2 inherited genetic conditions should be considered when findings are incompatible with the primary disease.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e947141"},"PeriodicalIF":1.0,"publicationDate":"2025-04-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143789122","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Airway Management for Tracheal Perforation After Left Hemi-Thyroid Lobectomy: A Case Report.","authors":"Masato Ryo, Shunsaku Goto, Keiko Hamada, Yasuko Nagasaka","doi":"10.12659/AJCR.946437","DOIUrl":"10.12659/AJCR.946437","url":null,"abstract":"<p><p>BACKGROUND Tracheal perforation, although rare, poses significant challenges for anesthesiologists following thyroid surgery, often necessitating complex airway management under general anesthesia. There is limited literature on effective strategies for managing airway complications in these patients. This report details a case of successful airway management using awake fiberoptic intubation in a patient with tracheal perforation and necrosis following a thyroidectomy. CASE REPORT A 65-year-old woman developed tracheal perforation 6 months after left hemi-thyroid lobectomy. She presented with symptoms of sore throat and neck swelling. Preoperative exams revealed an erosive tracheal perforation (3 cm) on the left side, immediately below the glottis. She was scheduled for an urgent tracheostomy under general anesthesia. A multidisciplinary team discussed various airway management strategies, including awake fiberoptic intubation, awake tracheostomy, and extracorporeal membrane oxygenation. Due to the adequate distance between the perforation and the tracheal bifurcation, awake fiberoptic intubation was selected. In the operating room, the bronchoscope was carefully advanced below the perforation site, and the endotracheal tube was then gently passed over the bronchoscope, ensuring the cuff did not disturb the perforation. The tube was positioned with its tip just above the tracheal bifurcation, and general anesthesia was subsequently initiated. Postoperative histological analysis of the tissue obtained during the tracheostomy revealed squamous cell carcinoma, suggesting that malignancy-related tracheal necrosis may have contributed to the perforation. CONCLUSIONS We report successful airway management in a patient with tracheal perforation following left hemi-thyroid lobectomy. Awake fiberoptic intubation proved to be an essential technique for managing such complex cases.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946437"},"PeriodicalIF":1.0,"publicationDate":"2025-04-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143784525","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A 65-Year-Old Man with Coronary Artery Embolism and Acute Inferior Myocardial Infarction Following Cardioversion for Ventricular Tachycardia.","authors":"Heba Khalid Alkoheji, Shanei Ali Shanei, Mohammed Ali Zayed","doi":"10.12659/AJCR.945228","DOIUrl":"10.12659/AJCR.945228","url":null,"abstract":"<p><p>BACKGROUND Cardioversion is a medical procedure aimed to convert abnormal rhythm to normal rhythm in hemodynamically unstable patients in ventricular tachycardia. The complications of cardioversion are mainly thromboembolism, with stroke being the most common complication. However, emboli can travel anywhere, including to the coronary arteries. In the literature, the complications highlighted are usually tailored towards atrial fibrillation. Indeed, acute embolic myocardial infarction following direct-current cardioversion of ventricular tachycardia has not been identified in the literature. This report describes a 65-year-old man with coronary artery embolism and acute inferior myocardial infarction following cardioversion for ventricular tachycardia. CASE REPORT A 65-year-old man with medical history of hypertension, chronic kidney disease, diabetes mellitus, dyslipidemia, and monomorphic ventricular tachycardia presented to the Emergency Department with monomorphic ventricular tachycardia not responding to conservative management; thus, electrical cardioversion was performed. He then developed embolic inferior wall myocardial infarction later in the day, confirmed with coronary angiogram and was treated conservatively according to patient comorbidities. CONCLUSIONS Embolic myocardial infarction following electrical cardioversion is rare, with only few cases reported in patients with atrial fibrillation. Proper diagnosis of this condition can help improve patient outcomes and reduce the rate of reoccurrence. To the best of our knowledge, this is the first case showing acute embolic myocardial infarction in a patient who underwent direct-current cardioversion for ventricular tachycardia.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945228"},"PeriodicalIF":1.0,"publicationDate":"2025-04-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143781486","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A Rare Case of Ovarian Serous Borderline Tumor Recurrence with Muscle Metastasis.","authors":"Akitoshi Yamamura, Mana Taki, Sachiko Kitamura, Masumi Sunada, Koji Yamanoi, Ryusuke Murakami, Takayoshi Shimizu, Ken Yamaguchi, Junzo Hamanishi, Sachiko Minamiguchi, Masaki Mandai","doi":"10.12659/AJCR.946680","DOIUrl":"10.12659/AJCR.946680","url":null,"abstract":"<p><p>BACKGROUND Ovarian serous borderline tumors (SBTs) generally have a favorable prognosis, with a very low recurrence rate. However, in rare cases, they can recur as invasive low-grade serous carcinoma (LGSC) after a prolonged follow-up period. Here, we report a case of LGSC originating from SBT that recurred 23 years after the initial surgery, with metastasis to the quadratus lumborum muscle - an exceptionally rare site of metastasis. CASE REPORT A 50-year-old woman, initially diagnosed with stage IIIC SBT and treated with complete tumor resection 23 years prior, presented with an asymptomatic recurrence detected by an elevated serum cancer antigen 125 (CA125) level. Contrast-enhanced computed tomography (CT) revealed multiple nodules suspected of peritoneal dissemination and a tumor infiltrating the quadratus lumborum muscle, suggesting recurrent SBT. A CT-guided needle biopsy confirmed that the tumor within the quadratus lumborum was a recurrence of SBT. Complete cytoreductive surgery was performed with the assistance of an orthopedic surgeon. Histopathological examination revealed progression to LGSC with cytoplasmic expression of the BRAF proto-oncogene (BRAF) V600E, indicating the presence of the BRAF V600E mutation, which is a characteristic feature of both SBT and LGSC. A retrospective review of CT images taken 10 years prior to the recurrence diagnosis showed a peritoneal tumor with calcification attached to the ileocecum, suggesting that the patient had remained asymptomatic for more than a decade after the actual onset of recurrence. CONCLUSIONS This case illustrates a rare instance of recurrent SBT with metastasis to the quadratus lumborum muscle. Given the exceptionally slow progression of recurrent SBT, long-term follow-up with CT imaging and serum CA125 monitoring is crucial for timely intervention and appropriate management upon recurrence.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946680"},"PeriodicalIF":1.0,"publicationDate":"2025-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143781521","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}