Intestinal Behçet disease (BD), associated with myelodysplastic syndrome (MDS), is often refractory to treatment. An 80-year-old man with trisomy 8 MDS (refractory anemia) developed intermittent fever. Despite investigations to exclude infectious disease, autoimmune disease, and malignancy as the cause of the fever, the etiology could not be determined. A colonoscopy revealed several shallow round ulcers in the ileocecal region and ascending colon, and the biopsy specimens showed nonspecific inflammation. Thereafter, the patient experienced abdominal pain and diarrhea. Other than an oral aphthous ulcer, the patient did not show symptoms to meet the diagnostic criteria for BD. The patient was diagnosed with intestinal ulcers (intestinal BD-like disease) with MDS and trisomy 8. After treatment failure with 5-aminosalicylic acid, steroid, colchicine, and azacitidine, cerebral infarction occurred. Eating was difficult because of the patient's impaired consciousness; hence, total parenteral nutrition (TPN) was commenced. The fever and abdominal symptoms improved with bowel rest over approximately 1 month. Small amounts of food were orally administered to the patient following recovery from the after-effects of the cerebral infarction, but diarrhea and fever repeatedly flared up. Therefore, TPN was continued at home. The patient has not experienced any further intestinal BD symptoms for approximately 1 year with bowel rest. Nutritional therapy, including bowel rest, may be an effective treatment option for intestinal BD with MDS, and might be used as an induction therapy of remission or a supportive therapy for other treatments.
{"title":"Refractory Intestinal Behçet-Like Disease Associated with Trisomy 8 Myelodysplastic Syndrome Resolved by Parenteral Nutrition.","authors":"Ryo Takahashi, Yasuo Matsubara, Satoshi Takahashi, Kazuaki Yokoyama, Lim Lay Ahyoung, Michiko Koga, Hiroyuki Sakamoto, Narikazu Boku, Dai Shida, Hiroshi Yotsuyanagi","doi":"10.1159/000533578","DOIUrl":"https://doi.org/10.1159/000533578","url":null,"abstract":"<p><p>Intestinal Behçet disease (BD), associated with myelodysplastic syndrome (MDS), is often refractory to treatment. An 80-year-old man with trisomy 8 MDS (refractory anemia) developed intermittent fever. Despite investigations to exclude infectious disease, autoimmune disease, and malignancy as the cause of the fever, the etiology could not be determined. A colonoscopy revealed several shallow round ulcers in the ileocecal region and ascending colon, and the biopsy specimens showed nonspecific inflammation. Thereafter, the patient experienced abdominal pain and diarrhea. Other than an oral aphthous ulcer, the patient did not show symptoms to meet the diagnostic criteria for BD. The patient was diagnosed with intestinal ulcers (intestinal BD-like disease) with MDS and trisomy 8. After treatment failure with 5-aminosalicylic acid, steroid, colchicine, and azacitidine, cerebral infarction occurred. Eating was difficult because of the patient's impaired consciousness; hence, total parenteral nutrition (TPN) was commenced. The fever and abdominal symptoms improved with bowel rest over approximately 1 month. Small amounts of food were orally administered to the patient following recovery from the after-effects of the cerebral infarction, but diarrhea and fever repeatedly flared up. Therefore, TPN was continued at home. The patient has not experienced any further intestinal BD symptoms for approximately 1 year with bowel rest. Nutritional therapy, including bowel rest, may be an effective treatment option for intestinal BD with MDS, and might be used as an induction therapy of remission or a supportive therapy for other treatments.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"287-293"},"PeriodicalIF":0.6,"publicationDate":"2023-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624938/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71478343","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}
Visceral artery pseudoaneurysms is a known vascular complication of pancreatitis that can lead to life-threatening hemorrhages with a high mortality rate if left untreated. We present a case of ruptured gastroduodenal artery pseudoaneurysm in a 68-year-old male with acute pancreatitis presenting with fatal gastrointestinal and retroperitoneal bleeding that was successfully managed with endovascular coil embolization of the involved vasculature. Patients with hemorrhagic pancreatitis or those presenting with unexplained retroperitoneal or gastrointestinal bleeding in the setting of pancreatitis with an unexplained drop in hematocrit or sudden expansion of pancreatic fluid collection should be screened in a timely manner for pseudoaneurysm using CT angiogram of the abdomen, which is the gold standard imaging modality to identify pseudoaneurysms. Once pseudoaneurysm is diagnosed, it should be treated immediately. Endovascular treatment options are now favored over surgical options in most cases.
{"title":"Ruptured Gastroduodenal Artery Pseudoaneurysms as a Complication of Pancreatitis.","authors":"Haider Ghazanfar, Abhilasha Jyala, Sameer Datta Kandhi, Dongmin Shin, Kazi Samsuddoha, Harish Patel","doi":"10.1159/000533617","DOIUrl":"https://doi.org/10.1159/000533617","url":null,"abstract":"<p><p>Visceral artery pseudoaneurysms is a known vascular complication of pancreatitis that can lead to life-threatening hemorrhages with a high mortality rate if left untreated. We present a case of ruptured gastroduodenal artery pseudoaneurysm in a 68-year-old male with acute pancreatitis presenting with fatal gastrointestinal and retroperitoneal bleeding that was successfully managed with endovascular coil embolization of the involved vasculature. Patients with hemorrhagic pancreatitis or those presenting with unexplained retroperitoneal or gastrointestinal bleeding in the setting of pancreatitis with an unexplained drop in hematocrit or sudden expansion of pancreatic fluid collection should be screened in a timely manner for pseudoaneurysm using CT angiogram of the abdomen, which is the gold standard imaging modality to identify pseudoaneurysms. Once pseudoaneurysm is diagnosed, it should be treated immediately. Endovascular treatment options are now favored over surgical options in most cases.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"294-301"},"PeriodicalIF":0.6,"publicationDate":"2023-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10623810/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71478344","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}
Autoimmune hepatitis (AIH) is a common and debilitating pathology that has acute, subacute, and chronic presentation, requiring prompt diagnosis and early intervention. Several serologic markers are found to be associated with the pathogenesis and progression of autoimmune hepatitis, most notably antinuclear antibodies and anti-smooth muscle antibodies [Front Immunol. 2018;9:609]. In addition, AIH is also characterized by the elevation of gamma globulin levels, mainly immunoglobulin G (IgG) [World J Gastroenterol. 2015;21(1):60-83]. Although the literature has well established the presence of increased IgG levels in AIH, few studies have evaluated the subtypes of IgG and their differential levels associated with AIH. Here, we present a rare case of AIH that lacks the common serologic markers but instead reveals an elevation in IgG1 level. Our patient was subsequently placed on corticosteroids, and her symptoms quickly resolved. We intend to introduce this case to the medical community in the hope of aiding in the proper diagnosis and timely intervention of subsequent cases with similar presentations.
{"title":"A Novel Presentation of Autoimmune Hepatitis with IgG1 Elevation.","authors":"Yujiao Zhang, Bilal Niazi, Auda Auda, Angel Ann Chacko, Amer Jarri, Abdifatah Mohamed, Saad Ali, Hongfa Zhu, Syed Sirajuddin","doi":"10.1159/000530517","DOIUrl":"https://doi.org/10.1159/000530517","url":null,"abstract":"<p><p>Autoimmune hepatitis (AIH) is a common and debilitating pathology that has acute, subacute, and chronic presentation, requiring prompt diagnosis and early intervention. Several serologic markers are found to be associated with the pathogenesis and progression of autoimmune hepatitis, most notably antinuclear antibodies and anti-smooth muscle antibodies [Front Immunol. 2018;9:609]. In addition, AIH is also characterized by the elevation of gamma globulin levels, mainly immunoglobulin G (IgG) [World J Gastroenterol. 2015;21(1):60-83]. Although the literature has well established the presence of increased IgG levels in AIH, few studies have evaluated the subtypes of IgG and their differential levels associated with AIH. Here, we present a rare case of AIH that lacks the common serologic markers but instead reveals an elevation in IgG1 level. Our patient was subsequently placed on corticosteroids, and her symptoms quickly resolved. We intend to introduce this case to the medical community in the hope of aiding in the proper diagnosis and timely intervention of subsequent cases with similar presentations.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"281-286"},"PeriodicalIF":0.6,"publicationDate":"2023-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624941/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71478339","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}
We describe an autopsied case of systemic AA amyloidosis secondary to metastatic renal cell carcinoma presenting intractable diarrhea. Severe diarrhea was the major symptom for the diagnosis of AA amyloidosis. No renal symptoms which are common in AA amyloidosis secondary to renal cell carcinoma were shown because hemodialysis following bilateral nephrectomy had already been started 9 years before. Treatment against metastatic tumors as a solution of AA amyloidosis could not be performed because of bad performance status and the patient died 5 months after the diagnosis. Autopsy findings revealed that AA amyloid deposition was seen in multi-organs including the intestine. The metastatic tumors were histologically compatible as metastasis of renal cell carcinoma. There was no other cause of chronic inflammation such as inflammatory arthritis. We concluded that chronic inflammation provoked by the metastatic tumors of renal cell carcinoma was a major cause of systemic AA amyloidosis. Intestinal AA amyloidosis with malabsorption was the cause of death. Clinicians should keep it in mind that solid organ malignancy can be a cause of AA amyloidosis and renal cell carcinoma is the most common carcinomatous cause. This case is particularly instructive in that progression of amyloidosis may be missed in hemodialysis patients with anuria and that gastrointestinal symptoms can be the primary indicators of systemic amyloidosis. Endoscopic examination including biopsy is important for the diagnosis and early treatment of amyloidosis.
{"title":"Systemic AA Amyloidosis Secondary to Metastatic Renal Cell Carcinoma in a Hemodialysis Patient with Intractable Diarrhea.","authors":"Hiroyuki Endo, Noriyuki Obara, Shinichi Mizuno, Yasuhiro Nakamura","doi":"10.1159/000531066","DOIUrl":"https://doi.org/10.1159/000531066","url":null,"abstract":"<p><p>We describe an autopsied case of systemic AA amyloidosis secondary to metastatic renal cell carcinoma presenting intractable diarrhea. Severe diarrhea was the major symptom for the diagnosis of AA amyloidosis. No renal symptoms which are common in AA amyloidosis secondary to renal cell carcinoma were shown because hemodialysis following bilateral nephrectomy had already been started 9 years before. Treatment against metastatic tumors as a solution of AA amyloidosis could not be performed because of bad performance status and the patient died 5 months after the diagnosis. Autopsy findings revealed that AA amyloid deposition was seen in multi-organs including the intestine. The metastatic tumors were histologically compatible as metastasis of renal cell carcinoma. There was no other cause of chronic inflammation such as inflammatory arthritis. We concluded that chronic inflammation provoked by the metastatic tumors of renal cell carcinoma was a major cause of systemic AA amyloidosis. Intestinal AA amyloidosis with malabsorption was the cause of death. Clinicians should keep it in mind that solid organ malignancy can be a cause of AA amyloidosis and renal cell carcinoma is the most common carcinomatous cause. This case is particularly instructive in that progression of amyloidosis may be missed in hemodialysis patients with anuria and that gastrointestinal symptoms can be the primary indicators of systemic amyloidosis. Endoscopic examination including biopsy is important for the diagnosis and early treatment of amyloidosis.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"275-280"},"PeriodicalIF":0.6,"publicationDate":"2023-09-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624937/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71478345","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}
Subepithelial lesions (SELs) originating from muscularis mucosae of the colon are very rare findings on endoscopy. Appropriate management of SELs involves making a correct diagnosis and estimating their malignant potential. In this case study, a 58-year-old Saudi man presented with a small, 8-mm sigmoid polyp during screening colonoscopy. The polyp was removed by hot snare polypectomy and sent to pathology laboratory. Report showed an unremarkable colonic mucosa and underlying well-circumscribed submucosal lesion composed of monotonous spindle cells. Immunohistochemical (IHC) analysis ruled out CD117-/DOG1-positive GIST and confirmed the lesion as leiomyomatous polyp. Colonic leiomyomas are usually benign and often asymptomatic and discovered during CRC screening procedures. Diagnosis is made on histology/IHC analysis since endoscopically they might be indistinguishable from other SELs. Conventional polypectomy is an appropriate treatment for small colonic leiomyoma and these benign lesions typically do not recur.
{"title":"An Asymptomatic Patient with Colonic Leiomyoma.","authors":"Saad Alkhowaiter, Abdulmalik Alsheikh, Ammar Alotaibi","doi":"10.1159/000533550","DOIUrl":"https://doi.org/10.1159/000533550","url":null,"abstract":"<p><p>Subepithelial lesions (SELs) originating from muscularis mucosae of the colon are very rare findings on endoscopy. Appropriate management of SELs involves making a correct diagnosis and estimating their malignant potential. In this case study, a 58-year-old Saudi man presented with a small, 8-mm sigmoid polyp during screening colonoscopy. The polyp was removed by hot snare polypectomy and sent to pathology laboratory. Report showed an unremarkable colonic mucosa and underlying well-circumscribed submucosal lesion composed of monotonous spindle cells. Immunohistochemical (IHC) analysis ruled out CD117-/DOG1-positive GIST and confirmed the lesion as leiomyomatous polyp. Colonic leiomyomas are usually benign and often asymptomatic and discovered during CRC screening procedures. Diagnosis is made on histology/IHC analysis since endoscopically they might be indistinguishable from other SELs. Conventional polypectomy is an appropriate treatment for small colonic leiomyoma and these benign lesions typically do not recur.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"269-274"},"PeriodicalIF":0.6,"publicationDate":"2023-09-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624942/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71478340","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 : 2023-09-06eCollection Date: 2023-01-01DOI: 10.1159/000531892
Junghyun Lim, Catherine Zaw, Simon Abramson, Paola N Lichtenberger, Binu V John, Lorena Cuebas-Rosado
Extracolonic manifestations of Clostridium difficile have been rarely reported. We herein report a case of a 60-year-old immunocompetent man presenting with fever, nausea, abdominal pain, and loose stools for 2 weeks. Triple-phase liver computed tomography demonstrated pyogenic liver abscesses and portal pylephlebitis. Blood cultures grew C. difficile and Bacteroides fragilis, and liver abscess cultures grew Proteus mirabilis, Escherichia coli, and the viridans group Streptococci. Antibiotics coverage was selected to direct at all identified organisms. This demonstrates an unusual case of C. difficile bacteremia in a patient with polymicrobial pyogenic liver abscesses and pylephlebitis.
{"title":"<i>Clostridium difficile</i> Bacteremia as a Rare Presentation of Polymicrobial Pyogenic Liver Abscesses and Its Management Challenges.","authors":"Junghyun Lim, Catherine Zaw, Simon Abramson, Paola N Lichtenberger, Binu V John, Lorena Cuebas-Rosado","doi":"10.1159/000531892","DOIUrl":"https://doi.org/10.1159/000531892","url":null,"abstract":"<p><p>Extracolonic manifestations of <i>Clostridium difficile</i> have been rarely reported. We herein report a case of a 60-year-old immunocompetent man presenting with fever, nausea, abdominal pain, and loose stools for 2 weeks. Triple-phase liver computed tomography demonstrated pyogenic liver abscesses and portal pylephlebitis. Blood cultures grew <i>C. difficile</i> and <i>Bacteroides fragilis</i>, and liver abscess cultures grew <i>Proteus mirabilis</i>, <i>Escherichia coli</i>, and the viridans group <i>Streptococci</i>. Antibiotics coverage was selected to direct at all identified organisms. This demonstrates an unusual case of <i>C. difficile</i> bacteremia in a patient with polymicrobial pyogenic liver abscesses and pylephlebitis.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"264-268"},"PeriodicalIF":0.6,"publicationDate":"2023-09-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624945/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71478338","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}
The patient was a woman in her 40s who was diagnosed with Crohn's disease (CD) of the large and small intestines in 1996. In 2005, she was referred to our hospital for treatment. We treated her for 17 years with corticosteroids, biologics, immunosuppressive agents, 5-aminosalicylic acid, and nutrition care. However, her Crohn's Disease Activity Index remained between 200 and 250, indicating refractory CD. During her medical treatment, the patient also underwent 3 operations. One year ago, the patient became pregnant through in vitro fertilization. Even after pregnancy was confirmed, the patient continued her treatment for refractory CD with ustekinumab, granulocyte apheresis, and budesonide. Nonetheless, her CD was highly active during pregnancy, and she experienced various complications: sigmoid volvulus at gestational week 15, venous thrombosis at gestational week 17, nontraumatic rib fracture due to fetal movement at gestational week 32, and sepsis from central venous catheter infection at gestational week 37. At gestational week 38, the patient gave birth by emergency cesarian delivery. This paper reports details of the case in which delivery was achieved after various complications were overcome and discusses previous relevant reports.
{"title":"A Patient with Crohn's Disease Who Gave Birth Despite Sigmoid Volvulus, Venous Thrombosis, Nontraumatic Fracture of the Rib, and Sepsis during Pregnancy.","authors":"Ayumi Ito, Maria Yonezawa, Shun Murasugi, Teppei Omori, Shinichi Nakamura, Katsutoshi Tokushige","doi":"10.1159/000531705","DOIUrl":"10.1159/000531705","url":null,"abstract":"<p><p>The patient was a woman in her 40s who was diagnosed with Crohn's disease (CD) of the large and small intestines in 1996. In 2005, she was referred to our hospital for treatment. We treated her for 17 years with corticosteroids, biologics, immunosuppressive agents, 5-aminosalicylic acid, and nutrition care. However, her Crohn's Disease Activity Index remained between 200 and 250, indicating refractory CD. During her medical treatment, the patient also underwent 3 operations. One year ago, the patient became pregnant through in vitro fertilization. Even after pregnancy was confirmed, the patient continued her treatment for refractory CD with ustekinumab, granulocyte apheresis, and budesonide. Nonetheless, her CD was highly active during pregnancy, and she experienced various complications: sigmoid volvulus at gestational week 15, venous thrombosis at gestational week 17, nontraumatic rib fracture due to fetal movement at gestational week 32, and sepsis from central venous catheter infection at gestational week 37. At gestational week 38, the patient gave birth by emergency cesarian delivery. This paper reports details of the case in which delivery was achieved after various complications were overcome and discusses previous relevant reports.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"1 1","pages":"255-263"},"PeriodicalIF":0.6,"publicationDate":"2023-08-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624936/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48774448","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}
Eosinophilic granulomatosis with polyangiitis (EGPA), historically named Churg-Strauss syndrome, is a rare vasculitis affecting small- and medium-sized blood vessels. The disease has a predilection for numerous organs including the lungs, sinuses, kidneys, heart, nerves, and gastrointestinal tract but is prominently associated with asthma, rhinosinusitis, and eosinophilia. Gastrointestinal involvement is common; however, a gastrointestinal manifestation as the cardinal symptom following an infection is atypical. Here, we present a case of a 61-year-old male who presented with persistent diarrhea following a toxigenic Clostridium difficile infection despite multiple antibiotic courses. Repeat testing confirmed eradication of the infection, and further evaluation with colon biopsy revealed small and medium-sized vasculitis with eosinophilic infiltration and granulomas. Treatment with prednisone and cyclophosphamide resulted in rapid improvement of his diarrhea. Gastrointestinal symptoms in EGPA are associated with worse prognosis, so prompt identification and treatment of the disease is crucial. EGPA is rarely documented in histopathological samples from the gastrointestinal tract as endoscopic biopsies are typically too superficial to sample the submucosal layer containing the affected vessels. Additionally, the link between EGPA and infections as a potential trigger has not been clearly established, but gastrointestinal EGPA manifesting after a colonic infection raises concerns that this may have been a triggering event. Ultimately, further study is needed to understand, diagnose, and treat gastrointestinal and postinfection EGPA.
{"title":"Gastrointestinal Eosinophilic Granulomatosis with Polyangiitis following a <i>Clostridium difficile</i> Infection.","authors":"Haseeb Mohideen, Wegahta Weldemichael, Hafsa Hussain, Dushyant Singh Dahiya, Andrea Shin","doi":"10.1159/000530373","DOIUrl":"10.1159/000530373","url":null,"abstract":"<p><p>Eosinophilic granulomatosis with polyangiitis (EGPA), historically named Churg-Strauss syndrome, is a rare vasculitis affecting small- and medium-sized blood vessels. The disease has a predilection for numerous organs including the lungs, sinuses, kidneys, heart, nerves, and gastrointestinal tract but is prominently associated with asthma, rhinosinusitis, and eosinophilia. Gastrointestinal involvement is common; however, a gastrointestinal manifestation as the cardinal symptom following an infection is atypical. Here, we present a case of a 61-year-old male who presented with persistent diarrhea following a toxigenic <i>Clostridium difficile</i> infection despite multiple antibiotic courses. Repeat testing confirmed eradication of the infection, and further evaluation with colon biopsy revealed small and medium-sized vasculitis with eosinophilic infiltration and granulomas. Treatment with prednisone and cyclophosphamide resulted in rapid improvement of his diarrhea. Gastrointestinal symptoms in EGPA are associated with worse prognosis, so prompt identification and treatment of the disease is crucial. EGPA is rarely documented in histopathological samples from the gastrointestinal tract as endoscopic biopsies are typically too superficial to sample the submucosal layer containing the affected vessels. Additionally, the link between EGPA and infections as a potential trigger has not been clearly established, but gastrointestinal EGPA manifesting after a colonic infection raises concerns that this may have been a triggering event. Ultimately, further study is needed to understand, diagnose, and treat gastrointestinal and postinfection EGPA.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"228-234"},"PeriodicalIF":0.6,"publicationDate":"2023-06-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10294265/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9728056","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}
We treated a 39-year-old Japanese man who was admitted for an abdominal mass. He had had neurofibroma-like skin lesions since childhood. Computed tomography and endoscopic ultrasound results were consistent with a tumor in the small intestine. Although the tumor was undetectable by single-balloon endoscopy, the patient's background and imaging results led us to suspect a gastrointestinal stromal tumor (GIST). He also met the diagnostic criteria for neurofibroma type 1 (NF1). We performed a surgical removal of the tumor, and the biopsy results led to a definitive diagnosis of GIST. Small bowel GISTs should be considered in cases of NF1.
{"title":"Multiple Small Bowel Gastrointestinal Stromal Tumors Associated with Neurofibromatosis Type 1 that Were Not Detected by Endoscopy: A Case Report.","authors":"Satomi Saito, Teppei Omori, Shun Murasugi, Maria Yonezawa, Yukiko Takayama, Takeshi Ohki, Hiromi Onizuka, Yoji Nagashima, Katsutoshi Tokushige","doi":"10.1159/000529340","DOIUrl":"10.1159/000529340","url":null,"abstract":"<p><p>We treated a 39-year-old Japanese man who was admitted for an abdominal mass. He had had neurofibroma-like skin lesions since childhood. Computed tomography and endoscopic ultrasound results were consistent with a tumor in the small intestine. Although the tumor was undetectable by single-balloon endoscopy, the patient's background and imaging results led us to suspect a gastrointestinal stromal tumor (GIST). He also met the diagnostic criteria for neurofibroma type 1 (NF1). We performed a surgical removal of the tumor, and the biopsy results led to a definitive diagnosis of GIST. Small bowel GISTs should be considered in cases of NF1.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"160-167"},"PeriodicalIF":0.5,"publicationDate":"2023-03-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/47/7f/crg-2022-0017-0001-529340.PMC9996247.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9109688","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}
Colonic metastasis from ovarian cancer is extremely rare, with only seven reported cases. A 77-year-old woman who had previously undergone surgery for ovarian cancer was admitted to a local hospital with anal bleeding. Histopathological analysis confirmed the presence of adenocarcinoma. Colonoscopy revealed a descending colon tumor. The patient was diagnosed with Union for International Cancer Control T3N0M0 descending colon cancer or colon metastasis of the ovarian cancer. Laparoscopic left colectomy was performed; intraoperative frozen section diagnosis confirmed metastasis from ovarian cancer, and the absence of invasion to the serosal surface suggested hematogenous metastasis. This is the first case of colonic metastasis from ovarian cancer that was diagnosed using an intraoperative frozen section and laparoscopically treated.
{"title":"Metastasis of Ovarian Cancer to the Descending Colon.","authors":"Kentaro Abe, Hiroyuki Anzai, Satoko Eguchi, Masako Ikemura, Aya Shinozaki-Ushiku, Takahide Shinagawa, Hirofumi Sonoda, Yuichiro Yoshioka, Yuzo Nagai, Shinya Abe, Hiroyuki Matsuzaki, Yuichiro Yokoyama, Shigenobu Emoto, Koji Murono, Kazuhito Sasaki, Hiroaki Nozawa, Tetsuo Ushiku, Soichiro Ishihara","doi":"10.1159/000529299","DOIUrl":"https://doi.org/10.1159/000529299","url":null,"abstract":"<p><p>Colonic metastasis from ovarian cancer is extremely rare, with only seven reported cases. A 77-year-old woman who had previously undergone surgery for ovarian cancer was admitted to a local hospital with anal bleeding. Histopathological analysis confirmed the presence of adenocarcinoma. Colonoscopy revealed a descending colon tumor. The patient was diagnosed with Union for International Cancer Control T3N0M0 descending colon cancer or colon metastasis of the ovarian cancer. Laparoscopic left colectomy was performed; intraoperative frozen section diagnosis confirmed metastasis from ovarian cancer, and the absence of invasion to the serosal surface suggested hematogenous metastasis. This is the first case of colonic metastasis from ovarian cancer that was diagnosed using an intraoperative frozen section and laparoscopically treated.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"17 1","pages":"129-136"},"PeriodicalIF":0.6,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/2d/e4/crg-2022-0017-0001-529299.PMC9971617.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9372647","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}