Background: Dyskeratosis congenita (DKC), also known as Zinsser-Cole-Engman syndrome, is a progressive genetic disease with a triad of reticulate skin pigmentation, nail dystrophy, and leukoplakia. Approximately 8-10% of patients with DKC develop malignancies, and cases of colorectal cancer with DKC in young people have been reported previously.
Case presentation: A 25-year-old man with DKC since approximately 10 years of age developed fever and lower abdominal discomfort. Diagnostic imaging revealed locally advanced rectal cancer with lymph node metastasis, direct invasion of the prostate, and pelvic abscess due to tumor microperforation (cT4bN2M0 cStage IIIC). Biopsy showed well to moderately differentiated ductal adenocarcinoma. Genetic testing was negative for RAS and BRAF gene mutations, and microsatellite instability (MSI) testing was also negative. After sigmoid colostomy, the patient was treated with total neoadjuvant therapy (TNT) with systemic chemotherapy (six courses of FOLFOX + panitumumab) followed by chemoradiation therapy (50.4 Gy with capecitabine). After TNT, the primary tumor and metastatic lymph nodes shrank. According to the findings of colonoscopy and magnetic resonance image (MRI), we diagnosed near complete response (near-CR) and decided to follow the patient without surgery by every 3 months re-evaluation. However, 5 months after TNT, tumor regrowth was detected on colonoscopy and imaging, and the patient underwent total pelvic exenteration. He developed paralytic ileus as a postoperative complication, and was discharged on the 38th postoperative day. Pathological examination revealed a residual tumor with invasion of the periprostatic tissue. There was no metastasis in the pararectal and lateral pelvic lymph nodes, but one extramural non-contiguous cancerous extension (tumor deposit) was observed (ypT4bN1cM0 ypStage IIIC). The patient has been free of recurrence for one year after surgery.
Conclusions: DKC often develops into various tumors in the digestive system at an early age; therefore, appropriate surveillance may be required. In addition, considering that cancers in patients with DKC occur at a young age, fertility preservation and survivorship are also important, and adequate explanations and care should be provided to patients before and after treatment.
{"title":"Locally advanced rectal cancer in a young adult affected with dyskeratosis congenita (Zinsser-Cole-Engman syndrome): a case report.","authors":"Kosuke Ono, Yukiharu Hiyoshi, Asuka Ono, Mayuko Ouchi, Keisuke Kosumi, Kojiro Eto, Satoshi Ida, Masaaki Iwatsuki, Yoshifumi Baba, Yuji Miyamoto, Ikko Kajihara, Kazuhito Tanaka, Yuko Miyasato, Hideo Baba","doi":"10.1186/s40792-024-01985-9","DOIUrl":"10.1186/s40792-024-01985-9","url":null,"abstract":"<p><strong>Background: </strong>Dyskeratosis congenita (DKC), also known as Zinsser-Cole-Engman syndrome, is a progressive genetic disease with a triad of reticulate skin pigmentation, nail dystrophy, and leukoplakia. Approximately 8-10% of patients with DKC develop malignancies, and cases of colorectal cancer with DKC in young people have been reported previously.</p><p><strong>Case presentation: </strong>A 25-year-old man with DKC since approximately 10 years of age developed fever and lower abdominal discomfort. Diagnostic imaging revealed locally advanced rectal cancer with lymph node metastasis, direct invasion of the prostate, and pelvic abscess due to tumor microperforation (cT4bN2M0 cStage IIIC). Biopsy showed well to moderately differentiated ductal adenocarcinoma. Genetic testing was negative for RAS and BRAF gene mutations, and microsatellite instability (MSI) testing was also negative. After sigmoid colostomy, the patient was treated with total neoadjuvant therapy (TNT) with systemic chemotherapy (six courses of FOLFOX + panitumumab) followed by chemoradiation therapy (50.4 Gy with capecitabine). After TNT, the primary tumor and metastatic lymph nodes shrank. According to the findings of colonoscopy and magnetic resonance image (MRI), we diagnosed near complete response (near-CR) and decided to follow the patient without surgery by every 3 months re-evaluation. However, 5 months after TNT, tumor regrowth was detected on colonoscopy and imaging, and the patient underwent total pelvic exenteration. He developed paralytic ileus as a postoperative complication, and was discharged on the 38th postoperative day. Pathological examination revealed a residual tumor with invasion of the periprostatic tissue. There was no metastasis in the pararectal and lateral pelvic lymph nodes, but one extramural non-contiguous cancerous extension (tumor deposit) was observed (ypT4bN1cM0 ypStage IIIC). The patient has been free of recurrence for one year after surgery.</p><p><strong>Conclusions: </strong>DKC often develops into various tumors in the digestive system at an early age; therefore, appropriate surveillance may be required. In addition, considering that cancers in patients with DKC occur at a young age, fertility preservation and survivorship are also important, and adequate explanations and care should be provided to patients before and after treatment.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-09-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11377380/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142141122","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-09-06DOI: 10.1186/s40792-024-02013-6
Qin-Ming Zhao, Zhong-You Xu, Hui Wang
Purpose: Chronic mesenteric ischemia (CMI) is a rare disease that progresses with acute mesenteric ischemia, along with high mortality. How to choose the appropriate surgical method and the artery which should be opened first is the key to the treatment.
Case report: In this study, we successively used vascular bypass and endovascular therapy to treat a case of complex chronic mesenteric ischemia.
Conclusion: For mesenteric ischemic disease, the superior mesenteric artery (SMA) should be opened preferentially. Arterial bypass or interventional therapy can be used, or both can be combined, to finally achieve the purpose of treatment.
{"title":"Mesenteric ischemia caused by chronic occlusion of multiple mesenteric arteries: a case report.","authors":"Qin-Ming Zhao, Zhong-You Xu, Hui Wang","doi":"10.1186/s40792-024-02013-6","DOIUrl":"10.1186/s40792-024-02013-6","url":null,"abstract":"<p><strong>Purpose: </strong>Chronic mesenteric ischemia (CMI) is a rare disease that progresses with acute mesenteric ischemia, along with high mortality. How to choose the appropriate surgical method and the artery which should be opened first is the key to the treatment.</p><p><strong>Case report: </strong>In this study, we successively used vascular bypass and endovascular therapy to treat a case of complex chronic mesenteric ischemia.</p><p><strong>Conclusion: </strong>For mesenteric ischemic disease, the superior mesenteric artery (SMA) should be opened preferentially. Arterial bypass or interventional therapy can be used, or both can be combined, to finally achieve the purpose of treatment.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-09-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11379834/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142141123","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: Granulocyte colony-stimulating factor (G-CSF)-producing tumors have been reported in various organs, and the prognosis of patients with G-CSF-producing pancreatic cancers is particularly dismal. In this report, we present a case of G-CSF-producing anaplastic carcinoma of the pancreas (ACP), characterized by early postoperative recurrence and rapid, uncontrolled growth.
Case presentation: A 74-year-old man presented to our hospital with complaints of abdominal fullness and pain after eating. On admission, it was observed that the peripheral leukocyte counts and serum G-CSF levels were significantly elevated (23,770/µL and 251 pg/mL, respectively). Computed tomography of the abdomen revealed a pancreatic head tumor involving the superior mesenteric vein. Pathologically, ultrasound-guided fine-needle aspiration confirmed ACP. Subsequently, we performed a subtotal stomach-preserving pancreaticoduodenectomy with portal vein reconstruction and partial transverse colon resection. On postoperative day (POD) 7, the leukocyte count decreased from 21,180/μL to 8490/μL; moreover, computed tomography revealed liver metastasis. Therefore, mFOLFILINOX chemotherapy was initiated on POD 30. However, the tumor exhibited rapid progression, and the patient died on POD 45.
Conclusions: G-CSF-producing ACP is rare, and the prognosis of patients is extremely poor. Basic research is required to develop effective drugs against G-CSF-producing tumors, and large-scale studies using national databases are needed to develop multidisciplinary treatment methods.
{"title":"A case of resected anaplastic carcinoma of the pancreas producing granulocyte-colony stimulating factor with literature review.","authors":"Norio Kubo, Shigemasa Suzuki, Takahiro Seki, Shunsaku Furuke, Naoki Yagi, Takashi Ooki, Ryusuke Aihara, Akira Mogi, Yuka Yoshida, Kenji Kashiwabara, Yasuo Hosouchi, Ken Shirabe","doi":"10.1186/s40792-024-02008-3","DOIUrl":"10.1186/s40792-024-02008-3","url":null,"abstract":"<p><strong>Background: </strong>Granulocyte colony-stimulating factor (G-CSF)-producing tumors have been reported in various organs, and the prognosis of patients with G-CSF-producing pancreatic cancers is particularly dismal. In this report, we present a case of G-CSF-producing anaplastic carcinoma of the pancreas (ACP), characterized by early postoperative recurrence and rapid, uncontrolled growth.</p><p><strong>Case presentation: </strong>A 74-year-old man presented to our hospital with complaints of abdominal fullness and pain after eating. On admission, it was observed that the peripheral leukocyte counts and serum G-CSF levels were significantly elevated (23,770/µL and 251 pg/mL, respectively). Computed tomography of the abdomen revealed a pancreatic head tumor involving the superior mesenteric vein. Pathologically, ultrasound-guided fine-needle aspiration confirmed ACP. Subsequently, we performed a subtotal stomach-preserving pancreaticoduodenectomy with portal vein reconstruction and partial transverse colon resection. On postoperative day (POD) 7, the leukocyte count decreased from 21,180/μL to 8490/μL; moreover, computed tomography revealed liver metastasis. Therefore, mFOLFILINOX chemotherapy was initiated on POD 30. However, the tumor exhibited rapid progression, and the patient died on POD 45.</p><p><strong>Conclusions: </strong>G-CSF-producing ACP is rare, and the prognosis of patients is extremely poor. Basic research is required to develop effective drugs against G-CSF-producing tumors, and large-scale studies using national databases are needed to develop multidisciplinary treatment methods.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-09-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11374941/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142133827","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: An internal thoracic artery aneurysm (ITAA) is an exceedingly rare condition, with approximately two-thirds of reported cases being iatrogenic pseudoaneurysms. The remainder are attributed to various causes, including vasculitis, connective tissue disease, and neurofibromatosis type 1 (NF-1). NF-1 is an autosomal dominant disorder characterized by distinct clinical manifestations that occasionally include life-threatening vascular complications. Although NF-1 patients may develop various vascular abnormalities, ruptured ITAA is rarely reported, with only seven published cases.
Case presentation: A 32-year-old man with NF-1 consulted for a three-day history of persistent left back and upper arm pain. Initial chest radiography indicated left pleural effusion and an opacity at the left lung apex. Computed tomography scan revealed a mass in the left upper mediastinum that was initially suspected to be a tumor. Subsequent contrast-enhanced computed tomography revealed the mass to be a subclavian artery aneurysm. Detailed contrast-enhanced computed tomography with 1-mm slices was performed for surgical planning, identifying the mass as a left ITAA with contained rupture. Given the risk of re-rupture, emergency angiography was performed, which confirmed rupture of the left ITAA without extravasation. The ITAA was successfully treated with multiple microcoils at the proximal and distal ends. The patient had an uneventful recovery and was discharged on the fourth postoperative day.
Conclusions: This case highlights the importance of considering vascular lesions in NF-1 patients who present with pleural effusion. It also emphasizes the challenges in diagnosing ITAA and the effectiveness of thin-slice contrast-enhanced computed tomography scans and endovascular treatment.
{"title":"Endovascular treatment of contained ruptured internal thoracic artery aneurysm mimicking a tumor in a patient with neurofibromatosis type 1: a case report.","authors":"Ryoma Oda, Daisuke Endo, Takeshi Udagawa, Shingo Okada, Ryohei Kuwatsuru, Minoru Tabata","doi":"10.1186/s40792-024-02002-9","DOIUrl":"https://doi.org/10.1186/s40792-024-02002-9","url":null,"abstract":"<p><strong>Background: </strong>An internal thoracic artery aneurysm (ITAA) is an exceedingly rare condition, with approximately two-thirds of reported cases being iatrogenic pseudoaneurysms. The remainder are attributed to various causes, including vasculitis, connective tissue disease, and neurofibromatosis type 1 (NF-1). NF-1 is an autosomal dominant disorder characterized by distinct clinical manifestations that occasionally include life-threatening vascular complications. Although NF-1 patients may develop various vascular abnormalities, ruptured ITAA is rarely reported, with only seven published cases.</p><p><strong>Case presentation: </strong>A 32-year-old man with NF-1 consulted for a three-day history of persistent left back and upper arm pain. Initial chest radiography indicated left pleural effusion and an opacity at the left lung apex. Computed tomography scan revealed a mass in the left upper mediastinum that was initially suspected to be a tumor. Subsequent contrast-enhanced computed tomography revealed the mass to be a subclavian artery aneurysm. Detailed contrast-enhanced computed tomography with 1-mm slices was performed for surgical planning, identifying the mass as a left ITAA with contained rupture. Given the risk of re-rupture, emergency angiography was performed, which confirmed rupture of the left ITAA without extravasation. The ITAA was successfully treated with multiple microcoils at the proximal and distal ends. The patient had an uneventful recovery and was discharged on the fourth postoperative day.</p><p><strong>Conclusions: </strong>This case highlights the importance of considering vascular lesions in NF-1 patients who present with pleural effusion. It also emphasizes the challenges in diagnosing ITAA and the effectiveness of thin-slice contrast-enhanced computed tomography scans and endovascular treatment.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11362441/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142112261","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: Heart transplantation in patients with situs inversus is challenging, especially in terms of reconstruction of the systemic venous return. Several rerouting techniques have been presented but are associated with vulnerability to external compression, which might cause hemodynamic instability, especially in the presence of chest deformity. In this study, we report a rare case of successful heart transplantation in the presence of situs inversus and pectus excavatum.
Case presentation: A 55-year-old man, with a history of surgeries for corrected transposition of the great arteries with ventricular septal defect, was registered for heart transplantation owing to progression of heart failure. Subsequently, he had undergone a left ventricular assist device implantation; 14 years after registration, he underwent transplantation of the heart with normal anatomy. The inferior vena cava was reconstructed by anastomosing the left atria with a counterclockwise rotation of the donor heart and by lengthening the recipient inferior vena cava with a conduit made of the residual right atrial tissue. The superior vena cava was reconstructed using a donor innominate vein harvested with sufficient length. After successful weaning from cardiopulmonary bypass, the chest could not be closed because the heart was compressed owing to chest deformity, resulting in hemodynamic instability. Therefore, to exclude the left lung, a left pericardial screen was created using a bovine pericardium, allowing the chest to be closed with acceptable hemodynamics. The patient suffered postoperatively from a higher venous pressure, suggesting an obstruction of venous return early after surgery. The obstruction gradually resolved, and the patient was transferred for rehabilitation.
Conclusions: Heart transplantation in the presence of situs inversus is challenging; moreover, the presence of pectus excavatum further complicates the procedure. The paradoxically larger left lung and chest deformity compressed and impaired reconstructed systemic venous return. Although intrathoracic exclusion of the left lung was effective, an intraoperative or early postoperative thoracoplasty for pectus excavatum was also a viable option. Patient-specific management is mandatory, depending on the anatomy.
{"title":"Orthotopic heart transplantation in patient with situs inversus and pectus excavatum: a case report.","authors":"Satoru Wakasa, Tomonori Ooka, Takuma Sato, Yasushige Shingu, Nobuyasu Kato, Toshiyuki Nagai, Toshihisa Anzai, Minoru Ono, Yoshiro Matsui","doi":"10.1186/s40792-024-02006-5","DOIUrl":"https://doi.org/10.1186/s40792-024-02006-5","url":null,"abstract":"<p><strong>Background: </strong>Heart transplantation in patients with situs inversus is challenging, especially in terms of reconstruction of the systemic venous return. Several rerouting techniques have been presented but are associated with vulnerability to external compression, which might cause hemodynamic instability, especially in the presence of chest deformity. In this study, we report a rare case of successful heart transplantation in the presence of situs inversus and pectus excavatum.</p><p><strong>Case presentation: </strong>A 55-year-old man, with a history of surgeries for corrected transposition of the great arteries with ventricular septal defect, was registered for heart transplantation owing to progression of heart failure. Subsequently, he had undergone a left ventricular assist device implantation; 14 years after registration, he underwent transplantation of the heart with normal anatomy. The inferior vena cava was reconstructed by anastomosing the left atria with a counterclockwise rotation of the donor heart and by lengthening the recipient inferior vena cava with a conduit made of the residual right atrial tissue. The superior vena cava was reconstructed using a donor innominate vein harvested with sufficient length. After successful weaning from cardiopulmonary bypass, the chest could not be closed because the heart was compressed owing to chest deformity, resulting in hemodynamic instability. Therefore, to exclude the left lung, a left pericardial screen was created using a bovine pericardium, allowing the chest to be closed with acceptable hemodynamics. The patient suffered postoperatively from a higher venous pressure, suggesting an obstruction of venous return early after surgery. The obstruction gradually resolved, and the patient was transferred for rehabilitation.</p><p><strong>Conclusions: </strong>Heart transplantation in the presence of situs inversus is challenging; moreover, the presence of pectus excavatum further complicates the procedure. The paradoxically larger left lung and chest deformity compressed and impaired reconstructed systemic venous return. Although intrathoracic exclusion of the left lung was effective, an intraoperative or early postoperative thoracoplasty for pectus excavatum was also a viable option. Patient-specific management is mandatory, depending on the anatomy.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11362432/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142112271","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: Intestinal malrotation is a condition in which the process of counterclockwise rotation and fixation to the peritoneum and retroperitoneum during fetal life is incomplete. In adults, it is generally asymptomatic and is often discovered incidentally. We report a case of laparoscopic partial resection of the descending colon for a tumor of the descending colon with a rare form of intestinal malrotation in which the inferior mesenteric artery ran symmetrically and the sigmoid colon was fixed to the dorsal cecum and right-sided retroperitoneum.
Case presentation: A 75-year-old man was referred to our department of internal medicine due to a positive fecal occult blood test. Lower endoscopy revealed a laterally spreading tumor in the descending colon, and endoscopic submucosal dissection was attempted; however, this procedure was difficult, and the patient was referred to our department for surgical treatment. Contrast-enhanced computed tomography revealed that the endoscopic clip was located in the descending colon on the right side, the inferior mesenteric artery was symmetrical, and the sigmoid colon was located on both the right and dorsal sides of the cecum. Laparoscopic ileocecum and sigmoid colon mobilization was performed from the left side of the patient. After the completion of sigmoid colon mobilization, which returned the sigmoid colon and descending colon to anatomical normalcy, laparoscopic partial resection of the descending colon was performed. Based on the results of a histopathological examination, a granular type of laterally spreading tumor was diagnosed. The patient was discharged uneventfully on postoperative day 8.
Conclusions: Detailed preoperative imaging and surgical simulation are necessary for abdominal surgery involving intestinal malrotation.
{"title":"Laparoscopic resection of a descending colon tumor with right-sided fixation of the sigmoid colon: a case report.","authors":"Shinya Ohno, Yukimasa Nagata, Tatsuki Kawahara, Yusuke Nonomura, Reo Tachikawa, Tomohito Shinoda, Kakeru Tawada, Aiko Ikawa, Bun Sano","doi":"10.1186/s40792-024-02004-7","DOIUrl":"https://doi.org/10.1186/s40792-024-02004-7","url":null,"abstract":"<p><strong>Background: </strong>Intestinal malrotation is a condition in which the process of counterclockwise rotation and fixation to the peritoneum and retroperitoneum during fetal life is incomplete. In adults, it is generally asymptomatic and is often discovered incidentally. We report a case of laparoscopic partial resection of the descending colon for a tumor of the descending colon with a rare form of intestinal malrotation in which the inferior mesenteric artery ran symmetrically and the sigmoid colon was fixed to the dorsal cecum and right-sided retroperitoneum.</p><p><strong>Case presentation: </strong>A 75-year-old man was referred to our department of internal medicine due to a positive fecal occult blood test. Lower endoscopy revealed a laterally spreading tumor in the descending colon, and endoscopic submucosal dissection was attempted; however, this procedure was difficult, and the patient was referred to our department for surgical treatment. Contrast-enhanced computed tomography revealed that the endoscopic clip was located in the descending colon on the right side, the inferior mesenteric artery was symmetrical, and the sigmoid colon was located on both the right and dorsal sides of the cecum. Laparoscopic ileocecum and sigmoid colon mobilization was performed from the left side of the patient. After the completion of sigmoid colon mobilization, which returned the sigmoid colon and descending colon to anatomical normalcy, laparoscopic partial resection of the descending colon was performed. Based on the results of a histopathological examination, a granular type of laterally spreading tumor was diagnosed. The patient was discharged uneventfully on postoperative day 8.</p><p><strong>Conclusions: </strong>Detailed preoperative imaging and surgical simulation are necessary for abdominal surgery involving intestinal malrotation.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11362403/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142112262","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: Post-colectomy pan-enteritis in ulcerative colitis (UC) is very rare, but it is often severe and fatal. We present a case of massive bleeding and perforation due to post-colectomy pan-enteritis, which showed a significant response to biologics in a UC patient.
Case presentation: A 30-year-old woman with a 5-month history of pancolitis UC underwent subtotal colectomy with ileostomy and mucosal fistula for refractory UC. She was diagnosed with small bowel obstruction on postoperative day (POD) 8 and bowel bleeding was observed on POD18. Reoperation was performed for bowel obstruction and bleeding on POD20. Intraoperatively, adhesive small bowel obstruction in the ileum and multiple erosions and ulcers with perforation were observed throughout the small bowel. We diagnosed post-colectomy pan-enteritis, and jejunostomy, lavage, adhesiolysis, and a simple closure of the perforated ileum were performed. High-dose steroid therapy for pan-enteritis was administered immediately after reoperation, and infliximab was administered because of worsening bleeding on day 3 after reoperation. Bleeding decreased one day after biologic administration and bleeding completely disappeared on day 10 after biologic administration. Specimens obtained from the terminal ileum at colectomy showed a normal ileum without inflammation and villus atrophy, while specimens from the perforated ileum showed congestion, villous atrophy, epithelial erosion, and mononuclear cell infiltration. No cryptitis, crypt distortion, or basal plasmacytosis (common characteristics in UC) were observed in either specimen.
Conclusion: An early diagnosis and intervention are important for post-colectomy pan-enteritis, and infliximab may be effective. Post-colectomy pan-enteritis with a multiple ulcer phenotype has different histological characteristics from UC and may have a different pathogenesis.
{"title":"Massive bleeding and perforation due to post-colectomy pan-enteritis with a significant response to biologic in a patient with ulcerative colitis: a case report.","authors":"Kenichiro Toritani, Hideaki Kimura, Manabu Maebashi, Kazuki Kurimura, Serina Haruyama, Yoshinori Nakamori, Mao Matsubayashi, Reiko Kunisaki, Reiko Tanaka, Satoshi Fujii, Itaru Endo","doi":"10.1186/s40792-024-02003-8","DOIUrl":"10.1186/s40792-024-02003-8","url":null,"abstract":"<p><strong>Background: </strong>Post-colectomy pan-enteritis in ulcerative colitis (UC) is very rare, but it is often severe and fatal. We present a case of massive bleeding and perforation due to post-colectomy pan-enteritis, which showed a significant response to biologics in a UC patient.</p><p><strong>Case presentation: </strong>A 30-year-old woman with a 5-month history of pancolitis UC underwent subtotal colectomy with ileostomy and mucosal fistula for refractory UC. She was diagnosed with small bowel obstruction on postoperative day (POD) 8 and bowel bleeding was observed on POD18. Reoperation was performed for bowel obstruction and bleeding on POD20. Intraoperatively, adhesive small bowel obstruction in the ileum and multiple erosions and ulcers with perforation were observed throughout the small bowel. We diagnosed post-colectomy pan-enteritis, and jejunostomy, lavage, adhesiolysis, and a simple closure of the perforated ileum were performed. High-dose steroid therapy for pan-enteritis was administered immediately after reoperation, and infliximab was administered because of worsening bleeding on day 3 after reoperation. Bleeding decreased one day after biologic administration and bleeding completely disappeared on day 10 after biologic administration. Specimens obtained from the terminal ileum at colectomy showed a normal ileum without inflammation and villus atrophy, while specimens from the perforated ileum showed congestion, villous atrophy, epithelial erosion, and mononuclear cell infiltration. No cryptitis, crypt distortion, or basal plasmacytosis (common characteristics in UC) were observed in either specimen.</p><p><strong>Conclusion: </strong>An early diagnosis and intervention are important for post-colectomy pan-enteritis, and infliximab may be effective. Post-colectomy pan-enteritis with a multiple ulcer phenotype has different histological characteristics from UC and may have a different pathogenesis.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11358364/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142081592","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: Nivolumab with chemotherapy has been transformative for metastatic gastric cancer (GC). The potential of this regimen for local tumor control could be utilized for perioperative chemotherapy in locally advanced GC with bulky tumors or lymph node metastasis involving other organs.
Case presentation: Five patients with HER2-negative advanced GC were treated with nivolumab and oxaliplatin-based chemotherapy. All patients presented with clinical stage III or IVA GC with tumors in contact with either the pancreas or liver. Following chemotherapy, all tumors demonstrated shrinkage, allowing successful radical gastrectomies including four minimally invasive approach without postoperative complications. Four patients avoided combined resection of other organs.
Conclusions: Perioperative chemotherapy with nivolumab was effective for local disease control in this case series. This regimen could be a promising treatment approach for locally advanced GC; however, its survival benefits should be evaluated in clinical trials.
{"title":"Perioperative chemotherapy with nivolumab for HER2-negative locally advanced gastric cancer: a case series.","authors":"Yuta Toji, Shintaro Takeuchi, Yuma Ebihara, Yo Kurashima, Kazuaki Harada, Mariko Hayashi, Hirotake Abe, Hideyuki Wada, Satoko Yorinaga, Toshiaki Shichinohe, Utano Tomaru, Yoshito Komatsu, Satoshi Hirano","doi":"10.1186/s40792-024-02001-w","DOIUrl":"10.1186/s40792-024-02001-w","url":null,"abstract":"<p><strong>Background: </strong>Nivolumab with chemotherapy has been transformative for metastatic gastric cancer (GC). The potential of this regimen for local tumor control could be utilized for perioperative chemotherapy in locally advanced GC with bulky tumors or lymph node metastasis involving other organs.</p><p><strong>Case presentation: </strong>Five patients with HER2-negative advanced GC were treated with nivolumab and oxaliplatin-based chemotherapy. All patients presented with clinical stage III or IVA GC with tumors in contact with either the pancreas or liver. Following chemotherapy, all tumors demonstrated shrinkage, allowing successful radical gastrectomies including four minimally invasive approach without postoperative complications. Four patients avoided combined resection of other organs.</p><p><strong>Conclusions: </strong>Perioperative chemotherapy with nivolumab was effective for local disease control in this case series. This regimen could be a promising treatment approach for locally advanced GC; however, its survival benefits should be evaluated in clinical trials.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11349963/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142081593","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: Treating rectal cancer presents challenges due to postoperative complications and reduced quality of life (QOL). Recent evidence supports the watch-and-wait (WW) approach for patients with a clinical complete response (cCR) following preoperative treatment. In this report, we discuss a case of metastatic rectal cancer with deficient mismatch repair (dMMR) treated successfully with pembrolizumab.
Case presentation: A 47-year-old male with dMMR rectal cancer and a single liver metastasis underwent treatment with pembrolizumab as neoadjuvant therapy. After 10 courses, the rectal lesion achieved cCR, prompting the selection of the WW approach. The liver metastasis showed significant shrinkage; however, the presence of a residual tumor was suspected, leading to a metastasectomy. A pathological complete response (pCR) was confirmed via histological examination. During a 24-month follow-up, there was no evidence of tumor regrowth, local recurrence, or distant metastasis.
Conclusions: The WW strategy is increasingly accepted for patients achieving cCR after preoperative treatment. While pCR in dMMR rectal cancer patients treated with immune checkpoint inhibitors (ICIs) has been documented, accurately predicting pCR from imaging remains challenging. This case illustrates that integrating ICI therapy, surgical interventions, and the WW approach can effectively achieve both oncological safety and improved QOL in the treatment of dMMR metastatic rectal cancer.
{"title":"Integrating surgical intervention and watch-and-wait approach in dMMR metastatic rectal cancer with pembrolizumab: a case report.","authors":"Yohei Ando, Tsubasa Sakurai, Kosuke Ozaki, Shimpei Matsui, Toshiki Mukai, Tomohiro Yamaguchi, Takashi Akiyoshi, Izuma Nakayama, Yasuyuki Shigematsu, Atsushi Oba, Akiko Chino, Yosuke Fukunaga","doi":"10.1186/s40792-024-01994-8","DOIUrl":"10.1186/s40792-024-01994-8","url":null,"abstract":"<p><strong>Background: </strong>Treating rectal cancer presents challenges due to postoperative complications and reduced quality of life (QOL). Recent evidence supports the watch-and-wait (WW) approach for patients with a clinical complete response (cCR) following preoperative treatment. In this report, we discuss a case of metastatic rectal cancer with deficient mismatch repair (dMMR) treated successfully with pembrolizumab.</p><p><strong>Case presentation: </strong>A 47-year-old male with dMMR rectal cancer and a single liver metastasis underwent treatment with pembrolizumab as neoadjuvant therapy. After 10 courses, the rectal lesion achieved cCR, prompting the selection of the WW approach. The liver metastasis showed significant shrinkage; however, the presence of a residual tumor was suspected, leading to a metastasectomy. A pathological complete response (pCR) was confirmed via histological examination. During a 24-month follow-up, there was no evidence of tumor regrowth, local recurrence, or distant metastasis.</p><p><strong>Conclusions: </strong>The WW strategy is increasingly accepted for patients achieving cCR after preoperative treatment. While pCR in dMMR rectal cancer patients treated with immune checkpoint inhibitors (ICIs) has been documented, accurately predicting pCR from imaging remains challenging. This case illustrates that integrating ICI therapy, surgical interventions, and the WW approach can effectively achieve both oncological safety and improved QOL in the treatment of dMMR metastatic rectal cancer.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11347510/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142056589","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-08-26DOI: 10.1186/s40792-024-01999-3
Shinichi Tanaka, Takahiro Ohmine, Takashi Maeda
Background: Distal embolization as the first manifestation of an abdominal aortic aneurysm (AAA) is relatively rare. AAAs presenting with multiple organs embolization are rarer and serious systemic conditions. There are no reports of life-saving outcomes in patients with AAAs presenting with multiple organs embolization prior to surgery.
Case presentation: A 78-year-old man presented with right acute lower limb ischemia (ALLI) and ischemic enteritis coexisting with an 83-mm AAA with a large mural thrombus. Although the patient underwent successful revascularization with endovascular treatment for right ALLI, the manifestation of ischemia of the calf muscles and infection forced right above-knee amputation. The patient also presented with ischemic colitis that resolved with conservative treatment. After receiving appropriate medical therapy and rehabilitation, the patient was successfully treated with open aortic repair for the AAA.
Conclusion: We successfully performed open aortic surgery for a rare case of AAA presenting with multiple organs embolization.
{"title":"A case of abdominal aortic aneurysm presenting with multiple organs embolization.","authors":"Shinichi Tanaka, Takahiro Ohmine, Takashi Maeda","doi":"10.1186/s40792-024-01999-3","DOIUrl":"10.1186/s40792-024-01999-3","url":null,"abstract":"<p><strong>Background: </strong>Distal embolization as the first manifestation of an abdominal aortic aneurysm (AAA) is relatively rare. AAAs presenting with multiple organs embolization are rarer and serious systemic conditions. There are no reports of life-saving outcomes in patients with AAAs presenting with multiple organs embolization prior to surgery.</p><p><strong>Case presentation: </strong>A 78-year-old man presented with right acute lower limb ischemia (ALLI) and ischemic enteritis coexisting with an 83-mm AAA with a large mural thrombus. Although the patient underwent successful revascularization with endovascular treatment for right ALLI, the manifestation of ischemia of the calf muscles and infection forced right above-knee amputation. The patient also presented with ischemic colitis that resolved with conservative treatment. After receiving appropriate medical therapy and rehabilitation, the patient was successfully treated with open aortic repair for the AAA.</p><p><strong>Conclusion: </strong>We successfully performed open aortic surgery for a rare case of AAA presenting with multiple organs embolization.</p>","PeriodicalId":22096,"journal":{"name":"Surgical Case Reports","volume":null,"pages":null},"PeriodicalIF":0.7,"publicationDate":"2024-08-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11347495/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142056588","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}