BACKGROUND Acalculous cholecystitis is a rare form of gallbladder inflammation that occurs without the presence of gallstones. It primarily affects critically ill patients and warrants prompt treatment given its association with high mortality. Pericarditis, an inflammation of the pericardium, typically arises from viral infections but can also be secondary to rheumatological, malignant, or bacterial causes. The concurrent presentation of both these conditions is rare and should prompt clinicians to investigate for a potential underlying cause. Previous case reports have shown that systemic lupus erythematosus, malignancies, and parasitic or bacterial infections are possible underlying causes of this dual presentation. CASE REPORT We describe a unique case in which acute viral pericarditis initially presented with clinical and imaging findings consistent with acute acalculous cholecystitis. A man in his 40s presented with epigastric pain and community ultrasound findings consistent with acalculous cholecystitis. As part of the workup, a CT scan showed an incidental finding of a pericardial effusion, and transthoracic echocardiogram revealed no evidence of cardiac tamponade. Despite treatment, the patient continued to spike fevers. Consequently, an extensive workup was performed, including pericardial and pleural biopsies, which were negative for rheumatological, bacterial, and malignant causes. After ongoing treatment with non-steroidal anti-inflammatory medications, the patient made a full recovery. CONCLUSIONS This case highlights the importance of recognizing acalculous cholecystitis as an atypical manifestation of acute viral pericarditis. When these 2 conditions occur together, identifying a possible underlying cause is paramount, as the management differs greatly. This case report is one of the few describing the presence of both conditions not due to a primary bacterial, malignant, or rheumatological cause. Viral illnesses can cause inflammatory responses leading to both conditions.
{"title":"Acalculous Cholecystitis as an Atypical Presentation of Viral Pericarditis: A Case Report.","authors":"Andrew Chen, Omar Salehi, Jevan Cevik","doi":"10.12659/AJCR.946029","DOIUrl":"10.12659/AJCR.946029","url":null,"abstract":"<p><p>BACKGROUND Acalculous cholecystitis is a rare form of gallbladder inflammation that occurs without the presence of gallstones. It primarily affects critically ill patients and warrants prompt treatment given its association with high mortality. Pericarditis, an inflammation of the pericardium, typically arises from viral infections but can also be secondary to rheumatological, malignant, or bacterial causes. The concurrent presentation of both these conditions is rare and should prompt clinicians to investigate for a potential underlying cause. Previous case reports have shown that systemic lupus erythematosus, malignancies, and parasitic or bacterial infections are possible underlying causes of this dual presentation. CASE REPORT We describe a unique case in which acute viral pericarditis initially presented with clinical and imaging findings consistent with acute acalculous cholecystitis. A man in his 40s presented with epigastric pain and community ultrasound findings consistent with acalculous cholecystitis. As part of the workup, a CT scan showed an incidental finding of a pericardial effusion, and transthoracic echocardiogram revealed no evidence of cardiac tamponade. Despite treatment, the patient continued to spike fevers. Consequently, an extensive workup was performed, including pericardial and pleural biopsies, which were negative for rheumatological, bacterial, and malignant causes. After ongoing treatment with non-steroidal anti-inflammatory medications, the patient made a full recovery. CONCLUSIONS This case highlights the importance of recognizing acalculous cholecystitis as an atypical manifestation of acute viral pericarditis. When these 2 conditions occur together, identifying a possible underlying cause is paramount, as the management differs greatly. This case report is one of the few describing the presence of both conditions not due to a primary bacterial, malignant, or rheumatological cause. Viral illnesses can cause inflammatory responses leading to both conditions.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946029"},"PeriodicalIF":1.0,"publicationDate":"2025-01-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11751735/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143013482","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 Neuroendocrine carcinoma (NEC) of the cervix is rare and has high mortality and recurrence rates. The clinical symptoms of cervical NEC, such as abnormal vaginal bleeding and discharge, are similar to those of other cervical cancers. Here, we describe a case involving a 42-year-old woman with cervical NEC accompanied by an isolated large ovarian metastasis. CASE REPORT A 42-year-old woman had experienced abdominal discomfort for the past 4 months, along with a larger abdominal circumference. Physical examination revealed a 15-cm, solid, mobile, abdominal mass and a smooth cervix. Abdominal computed tomography revealed a hypoattenuating solid mass with a calcified component and indistinct borders, measuring 16.6×15.5 cm. Tumor marker levels were as follows: cancer antigen 125, 803.9 U/mL; carcinoembryonic antigen, 241.9 ng/mL. Preoperatively, we suspected a malignant ovarian tumor without any suspicion of cervical cancer. Intraoperatively, a 25×20-cm solid mass was found on the left adnexa with peritoneal wall and rectosigmoid adhesions. We performed a total abdominal hysterectomy with bilateral salpingo-oophorectomy, followed by peritoneal biopsy and omentectomy. Histopathological examination showed a 2.5-cm endocervical mass and a normal ectocervical epithelium. Immunohistochemistry revealed a small-cell cervical NEC with metastasis to the left ovary. The final diagnosis was a stage IB2 cervical NEC with ovarian metastasis. For treatment, we administered an etoposide-cisplatin adjuvant chemotherapy regimen. CONCLUSIONS NEC of the cervix can manifest as a large ovarian tumor, lack the usual indications for cervical cancer, and spread to the ovaries without metastasis to other organs.
{"title":"Cervical Neuroendocrine Carcinoma Presenting as Isolated Large Ovarian Metastasis: A Case Report.","authors":"Ach Salman Faridzi, Grace Ariani Sugianto, Khanisyah Erza Gumilar, Brahmana Askandar Tjokroprawiro","doi":"10.12659/AJCR.945078","DOIUrl":"10.12659/AJCR.945078","url":null,"abstract":"<p><p>BACKGROUND Neuroendocrine carcinoma (NEC) of the cervix is rare and has high mortality and recurrence rates. The clinical symptoms of cervical NEC, such as abnormal vaginal bleeding and discharge, are similar to those of other cervical cancers. Here, we describe a case involving a 42-year-old woman with cervical NEC accompanied by an isolated large ovarian metastasis. CASE REPORT A 42-year-old woman had experienced abdominal discomfort for the past 4 months, along with a larger abdominal circumference. Physical examination revealed a 15-cm, solid, mobile, abdominal mass and a smooth cervix. Abdominal computed tomography revealed a hypoattenuating solid mass with a calcified component and indistinct borders, measuring 16.6×15.5 cm. Tumor marker levels were as follows: cancer antigen 125, 803.9 U/mL; carcinoembryonic antigen, 241.9 ng/mL. Preoperatively, we suspected a malignant ovarian tumor without any suspicion of cervical cancer. Intraoperatively, a 25×20-cm solid mass was found on the left adnexa with peritoneal wall and rectosigmoid adhesions. We performed a total abdominal hysterectomy with bilateral salpingo-oophorectomy, followed by peritoneal biopsy and omentectomy. Histopathological examination showed a 2.5-cm endocervical mass and a normal ectocervical epithelium. Immunohistochemistry revealed a small-cell cervical NEC with metastasis to the left ovary. The final diagnosis was a stage IB2 cervical NEC with ovarian metastasis. For treatment, we administered an etoposide-cisplatin adjuvant chemotherapy regimen. CONCLUSIONS NEC of the cervix can manifest as a large ovarian tumor, lack the usual indications for cervical cancer, and spread to the ovaries without metastasis to other organs.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945078"},"PeriodicalIF":1.0,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11751734/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143013485","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}
Qingfei Xing, Li He, Tingshuai Cao, Chunhai Hu, Xiaoteng Liu
BACKGROUND Surgery involving the right retroperitoneum can result in lymphatic (chylous) leakage from the cisterna chyli located anterior to the L1 and L2 vertebra or from lymph node dissection. This report describes a 46-year-old woman with retroperitoneal lymphatic (chylous) leak following right adrenalectomy for a nonfunctional adrenal adenoma. CASE REPORT A 46-year-old woman presented with a medical history of hypertension. An adrenal tumor (3.2×2.0 cm) was identified by computed tomography (CT). She was admitted for right retroperitoneoscopic adrenalectomy. The drainage volume of the drainage tube increased on the second day after surgery. The fluid had a milky and turbid discharge. She was started on a high-protein fat-restricted diet. In addition, 3 mg somatostatin acetate was administered daily. The chylous discharge dramatically decreased. After confirming that there was no increase in discharge, the drainage tube was removed on the 11th postoperative day. There was no recurrence of chylous fluid in 5 months. CONCLUSIONS This report shows that lymphatic (chylous) leak can be a complication of retroperitoneal surgery. The most important factor is the prevention of chylous complications. Even if no lymphatic leakage is found, it is necessary for the laparoscopic surgeon to fully coagulate the lymphatic channels. In most cases, it can be managed with conservative treatment.
{"title":"Managing Chyle Leakage Following Right Retroperitoneoscopic Adrenalectomy: A Case Study.","authors":"Qingfei Xing, Li He, Tingshuai Cao, Chunhai Hu, Xiaoteng Liu","doi":"10.12659/AJCR.945469","DOIUrl":"10.12659/AJCR.945469","url":null,"abstract":"<p><p>BACKGROUND Surgery involving the right retroperitoneum can result in lymphatic (chylous) leakage from the cisterna chyli located anterior to the L1 and L2 vertebra or from lymph node dissection. This report describes a 46-year-old woman with retroperitoneal lymphatic (chylous) leak following right adrenalectomy for a nonfunctional adrenal adenoma. CASE REPORT A 46-year-old woman presented with a medical history of hypertension. An adrenal tumor (3.2×2.0 cm) was identified by computed tomography (CT). She was admitted for right retroperitoneoscopic adrenalectomy. The drainage volume of the drainage tube increased on the second day after surgery. The fluid had a milky and turbid discharge. She was started on a high-protein fat-restricted diet. In addition, 3 mg somatostatin acetate was administered daily. The chylous discharge dramatically decreased. After confirming that there was no increase in discharge, the drainage tube was removed on the 11th postoperative day. There was no recurrence of chylous fluid in 5 months. CONCLUSIONS This report shows that lymphatic (chylous) leak can be a complication of retroperitoneal surgery. The most important factor is the prevention of chylous complications. Even if no lymphatic leakage is found, it is necessary for the laparoscopic surgeon to fully coagulate the lymphatic channels. In most cases, it can be managed with conservative treatment.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945469"},"PeriodicalIF":1.0,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11751733/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143013524","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 Gestational trophoblastic diseases (GTDs) are a group of benign and malignant tumors that arise from placental tissue. Ectopic pregnancies most commonly occur within the fallopian tubes. The estimated incidence of ectopic gestational trophoblastic diseases (GTDs) is approximated at 1.5 per 1 000 000 pregnancies, which makes it a very rare condition. The diagnosis is challenging since the clinical presentation often mimics conventional ectopic pregnancy, but management approaches are significantly different. CASE REPORT A 41-year-old woman (G7 T4 P0 A2 L4) presented to the Emergency Department with a period of amenorrhea followed by vaginal bleeding and abdominal pain. Right-side abdominal tenderness was noted upon physical examination. Laboratory investigations showed elevated serum ß-hCG level. Ultrasound examination showed a normal-size uterus. No intrauterine gestational sac was identified. An echogenic mass with central lucency was noted in the right adnexa, measuring 2.1×1.8 cm, with minimal free fluid present in the pouch of Douglas. The findings were suggestive of a ruptured right-side ectopic pregnancy. The patient underwent laparoscopic salpingectomy. Histopathological examination of the specimen revealed a complete hydatidiform mole with vascular invasion. The patient was treated conservatively and was followed up by a serial ß-hCG level until she achieved 3 consecutive negative ß-hCG titers. CONCLUSIONS Invasive moles located in the fallopian tube are very rare in the literature, and further research is needed to ascertain the proper management of such cases. Histopathological examination of the salpingectomy specimen has an essential role in confirming the diagnosis of ectopic GTDs and guiding the management.
{"title":"Invasive Hydatidiform Mole Mimicking Ectopic Pregnancy: A Case Report and Literature Analysis.","authors":"Amany A Fathaddin","doi":"10.12659/AJCR.946388","DOIUrl":"10.12659/AJCR.946388","url":null,"abstract":"<p><p>BACKGROUND Gestational trophoblastic diseases (GTDs) are a group of benign and malignant tumors that arise from placental tissue. Ectopic pregnancies most commonly occur within the fallopian tubes. The estimated incidence of ectopic gestational trophoblastic diseases (GTDs) is approximated at 1.5 per 1 000 000 pregnancies, which makes it a very rare condition. The diagnosis is challenging since the clinical presentation often mimics conventional ectopic pregnancy, but management approaches are significantly different. CASE REPORT A 41-year-old woman (G7 T4 P0 A2 L4) presented to the Emergency Department with a period of amenorrhea followed by vaginal bleeding and abdominal pain. Right-side abdominal tenderness was noted upon physical examination. Laboratory investigations showed elevated serum ß-hCG level. Ultrasound examination showed a normal-size uterus. No intrauterine gestational sac was identified. An echogenic mass with central lucency was noted in the right adnexa, measuring 2.1×1.8 cm, with minimal free fluid present in the pouch of Douglas. The findings were suggestive of a ruptured right-side ectopic pregnancy. The patient underwent laparoscopic salpingectomy. Histopathological examination of the specimen revealed a complete hydatidiform mole with vascular invasion. The patient was treated conservatively and was followed up by a serial ß-hCG level until she achieved 3 consecutive negative ß-hCG titers. CONCLUSIONS Invasive moles located in the fallopian tube are very rare in the literature, and further research is needed to ascertain the proper management of such cases. Histopathological examination of the salpingectomy specimen has an essential role in confirming the diagnosis of ectopic GTDs and guiding the management.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946388"},"PeriodicalIF":1.0,"publicationDate":"2025-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11742276/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142985100","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}
Celine Scholin, Andrew D Calvin, F N U Shweta, Tiziano Tallarita
BACKGROUND The bacterial organism Capnocytophaga canimorsus is an oral commensal of cats and dogs and can cause life-threatening infections like mycotic aneurysm, meningitis, and sepsis. Mycotic aneurysms occur when microbial infections cause arterial wall degeneration. Difficulty in diagnosing Capnocytophaga canimorsus infection can occur due to the bacteria's fastidious nature and laboratory testing limitations, contributing to the infection's high morbidity and mortality. This report describes the case of a patient with an iliac artery mycotic aneurysm 2 months after a dog bite. Identification of Capnocytophaga canimorsus was achieved through polymerase chain reaction. CASE REPORT The 67-year-old female patient presented initially with nonspecific abdominal pain. Imaging revealed a right iliac artery abnormality suspicious for mycotic aneurysm. Capnocytophaga canimorsus was identified through broad-range bacterial polymerase chain reaction after standard culture failed to determine the infectious etiology. A history of dog bite was discovered after diagnosis. When standard culture cannot provide a diagnosis, 16s rRNA polymerase chain reaction is the preferred molecular-based test at our institution. CONCLUSIONS Through presentation of a case of Capnocytophaga canimorsus mycotic aneurysm in an immunocompetent woman, this report illustrates the importance of familiarity with Capnocytophaga canimorsus and molecular laboratory methods in achieving favorable outcomes when faced with Capnocytophaga canimorsus infection. In these difficult cases, 16s rRNA polymerase chain reaction and similar molecular technologies are becoming essential. This case also highlights thorough history-taking as essential for guiding correct diagnosis and reinforces that infection with Capnocytophaga canimorsus should be investigated when there is a history of dog bite.
{"title":"Capnocytophaga canimorsus in Iliac Artery Mycotic Aneurysm: The Role of Molecular Diagnostics.","authors":"Celine Scholin, Andrew D Calvin, F N U Shweta, Tiziano Tallarita","doi":"10.12659/AJCR.946054","DOIUrl":"10.12659/AJCR.946054","url":null,"abstract":"<p><p>BACKGROUND The bacterial organism Capnocytophaga canimorsus is an oral commensal of cats and dogs and can cause life-threatening infections like mycotic aneurysm, meningitis, and sepsis. Mycotic aneurysms occur when microbial infections cause arterial wall degeneration. Difficulty in diagnosing Capnocytophaga canimorsus infection can occur due to the bacteria's fastidious nature and laboratory testing limitations, contributing to the infection's high morbidity and mortality. This report describes the case of a patient with an iliac artery mycotic aneurysm 2 months after a dog bite. Identification of Capnocytophaga canimorsus was achieved through polymerase chain reaction. CASE REPORT The 67-year-old female patient presented initially with nonspecific abdominal pain. Imaging revealed a right iliac artery abnormality suspicious for mycotic aneurysm. Capnocytophaga canimorsus was identified through broad-range bacterial polymerase chain reaction after standard culture failed to determine the infectious etiology. A history of dog bite was discovered after diagnosis. When standard culture cannot provide a diagnosis, 16s rRNA polymerase chain reaction is the preferred molecular-based test at our institution. CONCLUSIONS Through presentation of a case of Capnocytophaga canimorsus mycotic aneurysm in an immunocompetent woman, this report illustrates the importance of familiarity with Capnocytophaga canimorsus and molecular laboratory methods in achieving favorable outcomes when faced with Capnocytophaga canimorsus infection. In these difficult cases, 16s rRNA polymerase chain reaction and similar molecular technologies are becoming essential. This case also highlights thorough history-taking as essential for guiding correct diagnosis and reinforces that infection with Capnocytophaga canimorsus should be investigated when there is a history of dog bite.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946054"},"PeriodicalIF":1.0,"publicationDate":"2025-01-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11742453/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142980248","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}
Zhiqin Lin, Yi Chen, Xiaofu Dai, Liangwan Chen, Heng Lu
BACKGROUND Acute intramural hematoma (IMH) of the ascending thoracic aorta and aortic arch is a life-threatening condition, particularly in elderly patients with comorbidities, due to its risk of progression and rupture. Unlike aortic dissection, IMH lacks an intimal tear, influencing both clinical presentation and treatment strategy. This report describes a 74-year-old hypertensive woman with type A IMH and a penetrating atherosclerotic ulcer (PAU), managed with a hybrid surgical approach that combines external Dacron wrapping of the ascending aorta and endovascular stenting of the aortic arch with in-situ fenestration of the supra-aortic arteries. CASE REPORT A 74-year-old woman with a history of hypertension, insulin-dependent diabetes, chronic kidney disease, coronary artery disease, and extracardiac arteriopathy presented with chest pain and was diagnosed with type A IMH. Cardiac assessment showed a moderate left ventricular ejection fraction (45%) and New York Heart Association class III functional status, indicating high surgical risk (EuroSCORE II: 11.66). A hybrid approach was chosen, involving Dacron wrapping of the ascending aorta to reduce its diameter, followed by endovascular stent grafting of the aortic arch with in-situ fenestration to preserve supra-aortic branch blood flow. The patient recovered without complications, and 5-month follow-up imaging confirmed stable stent position, PAU exclusion, and preserved branch patency. CONCLUSIONS This case illustrates the feasibility and safety of combining off-pump external wrapping of the ascending aorta with endovascular stent grafting using in-situ fenestration, offering a promising, less-invasive alternative for high-risk patients with favorable short-term outcomes.
{"title":"Hybrid Repair of Ascending Aortic Intramural Hematoma and Arch Ulcer in a 74-Year-Old Woman - A Case Report.","authors":"Zhiqin Lin, Yi Chen, Xiaofu Dai, Liangwan Chen, Heng Lu","doi":"10.12659/AJCR.946212","DOIUrl":"10.12659/AJCR.946212","url":null,"abstract":"<p><p>BACKGROUND Acute intramural hematoma (IMH) of the ascending thoracic aorta and aortic arch is a life-threatening condition, particularly in elderly patients with comorbidities, due to its risk of progression and rupture. Unlike aortic dissection, IMH lacks an intimal tear, influencing both clinical presentation and treatment strategy. This report describes a 74-year-old hypertensive woman with type A IMH and a penetrating atherosclerotic ulcer (PAU), managed with a hybrid surgical approach that combines external Dacron wrapping of the ascending aorta and endovascular stenting of the aortic arch with in-situ fenestration of the supra-aortic arteries. CASE REPORT A 74-year-old woman with a history of hypertension, insulin-dependent diabetes, chronic kidney disease, coronary artery disease, and extracardiac arteriopathy presented with chest pain and was diagnosed with type A IMH. Cardiac assessment showed a moderate left ventricular ejection fraction (45%) and New York Heart Association class III functional status, indicating high surgical risk (EuroSCORE II: 11.66). A hybrid approach was chosen, involving Dacron wrapping of the ascending aorta to reduce its diameter, followed by endovascular stent grafting of the aortic arch with in-situ fenestration to preserve supra-aortic branch blood flow. The patient recovered without complications, and 5-month follow-up imaging confirmed stable stent position, PAU exclusion, and preserved branch patency. CONCLUSIONS This case illustrates the feasibility and safety of combining off-pump external wrapping of the ascending aorta with endovascular stent grafting using in-situ fenestration, offering a promising, less-invasive alternative for high-risk patients with favorable short-term outcomes.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946212"},"PeriodicalIF":1.0,"publicationDate":"2025-01-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11742452/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142972547","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 Cowden syndrome is a genetic disorder that predisposes individuals to cancer and is characterized by hamartomas derived from 3 germ layers. Although the clinical signs can be pathognomonic, diagnosis is often aided by biopsies, histopathological examination of oral and cutaneous lesions, and genetic studies, including multiple ligation-dependent probe amplification (MLPA). CASE REPORT We report a case of a 35-year-old woman who manifested with multiple lesions in the buccal mucosa, dorsum of the tongue, and gums, along with papillomatous papules on her facial skin and the dorsal surfaces of her hands. These lesions were identified as hamartomas. Laboratory tests, including blood biometry, blood chemistry, and coagulation profiles, returned results within normal ranges. Her medical history revealed uterine fibroids, raising suspicion of Cowden syndrome. A genetic consultation confirmed the diagnosis, revealing a heterozygous PTEN deletion. CONCLUSIONS This case illustrates the importance of a multidisciplinary approach in diagnosing Cowden syndrome, especially the role of dental professionals in recognizing early clinical signs. Early diagnosis through genetic testing is crucial due to the patient's elevated risk of malignancies. Healthcare providers must remain vigilant to syndromes such as Cowden syndrome, particularly in patients with relevant family histories, to ensure timely intervention and comprehensive management.
{"title":"Cowden Syndrome and Oral Lesions: A Case Report Using MLPA.","authors":"Mariana Cristina Barrón-Márquez, Rogelio González-González, Lucina Bobadilla-Morales, Victor Ulises Rodriguez-Machuca, Ronell Bologna-Molina, Nelly Molina-Frechero, Omar Alejandro Tremillo-Maldonado, Sandra López-Verdín","doi":"10.12659/AJCR.945876","DOIUrl":"10.12659/AJCR.945876","url":null,"abstract":"<p><p>BACKGROUND Cowden syndrome is a genetic disorder that predisposes individuals to cancer and is characterized by hamartomas derived from 3 germ layers. Although the clinical signs can be pathognomonic, diagnosis is often aided by biopsies, histopathological examination of oral and cutaneous lesions, and genetic studies, including multiple ligation-dependent probe amplification (MLPA). CASE REPORT We report a case of a 35-year-old woman who manifested with multiple lesions in the buccal mucosa, dorsum of the tongue, and gums, along with papillomatous papules on her facial skin and the dorsal surfaces of her hands. These lesions were identified as hamartomas. Laboratory tests, including blood biometry, blood chemistry, and coagulation profiles, returned results within normal ranges. Her medical history revealed uterine fibroids, raising suspicion of Cowden syndrome. A genetic consultation confirmed the diagnosis, revealing a heterozygous PTEN deletion. CONCLUSIONS This case illustrates the importance of a multidisciplinary approach in diagnosing Cowden syndrome, especially the role of dental professionals in recognizing early clinical signs. Early diagnosis through genetic testing is crucial due to the patient's elevated risk of malignancies. Healthcare providers must remain vigilant to syndromes such as Cowden syndrome, particularly in patients with relevant family histories, to ensure timely intervention and comprehensive management.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945876"},"PeriodicalIF":1.0,"publicationDate":"2025-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11742275/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142972546","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}
Aline Misar, Alexis Litchinko, Florence Bloget, Michael John Chilcott, Bernhard Egger
BACKGROUND Crohn disease is a chronic inflammatory bowel disease known for causing fistulous tracts, abscesses, and bowel perforation. Enterohepatic fistulas, a rare but significant complication, are scarcely reported. This article presents the case of a hepatic abscess due to an enterohepatic fistula in a patient with long-term Crohn disease and reviews the existing literature on this phenomenon. CASE REPORT A 59-year-old female patient with a known history of Crohn disease and previous ileocolic resection due to enteroenteric fistulas presented to our Emergency Department with right-sided abdominal pain persisting for 10 days. Diagnostic investigations, including imaging, revealed an enterohepatic fistula with a 3-4 cm hepatic abscess in segment V of the liver. Initial management involved conservative treatment with radiological drainage and antibiotics, leading to the patient's discharge. An elective laparotomy was scheduled 1 month later. The patient underwent resection of the ileocolic anastomosis with ileotransverse re-anastomosis and catheter removal. Postoperative management included treatment for paralytic ileus. She was discharged in good condition on postoperative day 11. CONCLUSIONS This report highlights the range of complications that can occur in patients with Crohn disease and presents the rare association between Crohn disease and enterohepatic fistula and abscess formation. Only 2 other case reports of enterohepatic fistula due to Crohn disease exist in the literature. Given the scarcity of evidence, no standardized guidelines are available, necessitating an individualized treatment approach. Initial conservative management can be effective; however, close monitoring is crucial to determine the need for subsequent surgical intervention.
{"title":"Rare Enterohepatic Fistula in Crohn's Disease: Case Analysis and Literature Synthesis.","authors":"Aline Misar, Alexis Litchinko, Florence Bloget, Michael John Chilcott, Bernhard Egger","doi":"10.12659/AJCR.945701","DOIUrl":"10.12659/AJCR.945701","url":null,"abstract":"<p><p>BACKGROUND Crohn disease is a chronic inflammatory bowel disease known for causing fistulous tracts, abscesses, and bowel perforation. Enterohepatic fistulas, a rare but significant complication, are scarcely reported. This article presents the case of a hepatic abscess due to an enterohepatic fistula in a patient with long-term Crohn disease and reviews the existing literature on this phenomenon. CASE REPORT A 59-year-old female patient with a known history of Crohn disease and previous ileocolic resection due to enteroenteric fistulas presented to our Emergency Department with right-sided abdominal pain persisting for 10 days. Diagnostic investigations, including imaging, revealed an enterohepatic fistula with a 3-4 cm hepatic abscess in segment V of the liver. Initial management involved conservative treatment with radiological drainage and antibiotics, leading to the patient's discharge. An elective laparotomy was scheduled 1 month later. The patient underwent resection of the ileocolic anastomosis with ileotransverse re-anastomosis and catheter removal. Postoperative management included treatment for paralytic ileus. She was discharged in good condition on postoperative day 11. CONCLUSIONS This report highlights the range of complications that can occur in patients with Crohn disease and presents the rare association between Crohn disease and enterohepatic fistula and abscess formation. Only 2 other case reports of enterohepatic fistula due to Crohn disease exist in the literature. Given the scarcity of evidence, no standardized guidelines are available, necessitating an individualized treatment approach. Initial conservative management can be effective; however, close monitoring is crucial to determine the need for subsequent surgical intervention.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e945701"},"PeriodicalIF":1.0,"publicationDate":"2025-01-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11730555/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142967231","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}
Qifan Wang, Jie Zhong, Yi Wang, Jun Bao, Sheng Li, Liu Yang
BACKGROUND Programmed death 1 (PD-1) inhibitors have demonstrated limited effectiveness in patients with microsatellite instability-high (MSI-H) colorectal cancer (CRC). Recent studies suggest that their efficacy can be enhanced when combined with anti-angiogenic agents. CASE REPORT We present a case of a 25-year-old woman with CRC harboring a KRAS mutation and MSI-H status, along with initially unresectable liver metastases. Despite receiving first-line chemotherapy combined with bevacizumab, her disease progressed. Subsequently, she was treated with a combination of a PD-1 inhibitor and bevacizumab as second-line therapy. This approach resulted in a partial response, ultimately leading to a pathological complete response after resection of the liver metastases. The patient continued with the combination therapy for over a year and showed no serious treatment-related adverse events. Postoperative follow-up imaging confirmed the absence of tumor recurrence or metastasis, and the patient remained in remission. CONCLUSIONS This case highlights the potential of combining immune checkpoint inhibitors with anti-angiogenic agents in treating patients with MSI-H metastatic CRC, particularly those with initially unresectable liver metastases. Although further research is warranted to validate this therapeutic strategy, our findings support the use of this combination as a viable option for achieving pathological complete response and improving outcomes in this patient population. Comprehensive clinical studies are needed to optimize conversion therapy regimens and enhance the likelihood of success in treating patients with MSI-H CRC with advanced disease.
{"title":"Efficacy of Combined PD-1 Inhibitor and Bevacizumab in Unresectable Liver Metastasis of MSI-H Colorectal Cancer: A Case Report.","authors":"Qifan Wang, Jie Zhong, Yi Wang, Jun Bao, Sheng Li, Liu Yang","doi":"10.12659/AJCR.946757","DOIUrl":"10.12659/AJCR.946757","url":null,"abstract":"<p><p>BACKGROUND Programmed death 1 (PD-1) inhibitors have demonstrated limited effectiveness in patients with microsatellite instability-high (MSI-H) colorectal cancer (CRC). Recent studies suggest that their efficacy can be enhanced when combined with anti-angiogenic agents. CASE REPORT We present a case of a 25-year-old woman with CRC harboring a KRAS mutation and MSI-H status, along with initially unresectable liver metastases. Despite receiving first-line chemotherapy combined with bevacizumab, her disease progressed. Subsequently, she was treated with a combination of a PD-1 inhibitor and bevacizumab as second-line therapy. This approach resulted in a partial response, ultimately leading to a pathological complete response after resection of the liver metastases. The patient continued with the combination therapy for over a year and showed no serious treatment-related adverse events. Postoperative follow-up imaging confirmed the absence of tumor recurrence or metastasis, and the patient remained in remission. CONCLUSIONS This case highlights the potential of combining immune checkpoint inhibitors with anti-angiogenic agents in treating patients with MSI-H metastatic CRC, particularly those with initially unresectable liver metastases. Although further research is warranted to validate this therapeutic strategy, our findings support the use of this combination as a viable option for achieving pathological complete response and improving outcomes in this patient population. Comprehensive clinical studies are needed to optimize conversion therapy regimens and enhance the likelihood of success in treating patients with MSI-H CRC with advanced disease.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e946757"},"PeriodicalIF":1.0,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11730553/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956432","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 Vulvar melanoma during pregnancy is exceptionally rare. Hormonal and immunological changes in pregnancy have raised concerns about the potential for accelerated melanoma progression and poorer maternal outcomes. This case report describes an unusual presentation of vulvar melanoma in a pregnant patient, which rapidly progressed despite previous treatments, but resulted in a favorable fetal outcome. CASE REPORT A 40-year-old G3P2A0 woman at 28 weeks of gestation, with a history of vulvar malignant melanoma diagnosed 3 years prior, presented with sudden abdominal pain and hematuria. She had previously received 6 courses of chemotherapy. Physical examination revealed a 3-cm mass in the right vulva, while ultrasonography detected a hyperechoic solid mass in the cervix and elevated LDH levels. Given the advanced disease, the medical team proceeded with a cesarean hysterectomy, colpotomy for uterine corpus involvement, and bladder repair due to an iatrogenic laceration. Histopathological findings confirmed metastatic vulvar melanoma in the cervix and uterine corpus. The pregnancy was terminated at 27 weeks due to the progression of grade IV melanoma, but the neonate was delivered in stable condition. Unfortunately, the patient died 1 month after the operation. CONCLUSIONS This case underscores the potential for aggressive melanoma progression during pregnancy, likely exacerbated by physiological changes, yet highlights a successful fetal outcome. While chemotherapy can adversely affect the reproductive system and may lead to infertility, this patient was able to conceive, and the case illustrates the complex interplay of pregnancy and cancer progression.
{"title":"Pregnancy Complicated by Rapidly Progressing Vulvar Melanoma: A Case Study.","authors":"Bagas Satriyo Wicaksono, Aditiyono Aditiyono, Dina Marlina, Aditya Utomo, Putri Nadhira Adinda Adriansyah, Dody Novrial","doi":"10.12659/AJCR.944972","DOIUrl":"10.12659/AJCR.944972","url":null,"abstract":"<p><p>BACKGROUND Vulvar melanoma during pregnancy is exceptionally rare. Hormonal and immunological changes in pregnancy have raised concerns about the potential for accelerated melanoma progression and poorer maternal outcomes. This case report describes an unusual presentation of vulvar melanoma in a pregnant patient, which rapidly progressed despite previous treatments, but resulted in a favorable fetal outcome. CASE REPORT A 40-year-old G3P2A0 woman at 28 weeks of gestation, with a history of vulvar malignant melanoma diagnosed 3 years prior, presented with sudden abdominal pain and hematuria. She had previously received 6 courses of chemotherapy. Physical examination revealed a 3-cm mass in the right vulva, while ultrasonography detected a hyperechoic solid mass in the cervix and elevated LDH levels. Given the advanced disease, the medical team proceeded with a cesarean hysterectomy, colpotomy for uterine corpus involvement, and bladder repair due to an iatrogenic laceration. Histopathological findings confirmed metastatic vulvar melanoma in the cervix and uterine corpus. The pregnancy was terminated at 27 weeks due to the progression of grade IV melanoma, but the neonate was delivered in stable condition. Unfortunately, the patient died 1 month after the operation. CONCLUSIONS This case underscores the potential for aggressive melanoma progression during pregnancy, likely exacerbated by physiological changes, yet highlights a successful fetal outcome. While chemotherapy can adversely affect the reproductive system and may lead to infertility, this patient was able to conceive, and the case illustrates the complex interplay of pregnancy and cancer progression.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e944972"},"PeriodicalIF":1.0,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11730550/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142962337","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}