Collagenous gastrointestinal (GI) disease is a rare inflammatory condition characterized by subepithelial collagen deposition and inflammatory cell infiltrates of the GI mucosa, which typically occurs in the stomach in children. There are few published reports of more extensive involvement in children, and descriptions of familial involvement are rare, with no previous reported cases of affected siblings in early childhood. We describe two siblings with contrasting medical backgrounds, who were simultaneously diagnosed with collagenous GI disease in early childhood. Both children demonstrated gastric and colonic involvement on serial endoscopies, however, had distinct patterns of clinical presentation, disease course, and histological progression, providing new insights into the pediatric phenotype of collagenous GI disease and further, its relationship to microscopic colitis. Although rare, this condition should be considered as a differential in children presenting with severe or refractory iron deficiency anemia, chronic non‐bloody watery diarrhea, or unexplained nonspecific chronic abdominal pain.
{"title":"Co‐occurrence of collagenous gastrointestinal disease in siblings in early childhood: New insight into a rare condition","authors":"Taryn L. Luitingh, Jessica Ng, K. McGrath","doi":"10.1002/jpr3.12097","DOIUrl":"https://doi.org/10.1002/jpr3.12097","url":null,"abstract":"Collagenous gastrointestinal (GI) disease is a rare inflammatory condition characterized by subepithelial collagen deposition and inflammatory cell infiltrates of the GI mucosa, which typically occurs in the stomach in children. There are few published reports of more extensive involvement in children, and descriptions of familial involvement are rare, with no previous reported cases of affected siblings in early childhood. We describe two siblings with contrasting medical backgrounds, who were simultaneously diagnosed with collagenous GI disease in early childhood. Both children demonstrated gastric and colonic involvement on serial endoscopies, however, had distinct patterns of clinical presentation, disease course, and histological progression, providing new insights into the pediatric phenotype of collagenous GI disease and further, its relationship to microscopic colitis. Although rare, this condition should be considered as a differential in children presenting with severe or refractory iron deficiency anemia, chronic non‐bloody watery diarrhea, or unexplained nonspecific chronic abdominal pain.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"38 18","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141339962","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
We report one of the first cases of eosinophilic gastritis (EoG) in a child under 12 years treated with benralizumab. At 7 years, our patient was started on benralizumab after failing to respond to various combinations of high‐dose omeprazole, milk elimination diet, oral viscous budesonide, and oral systemic steroids. He had a complete depletion of gastrointestinal tissue eosinophils with improved symptoms but had symptomatic flares with tapering of background therapy. However, after 4 years on benralizumab he became symptomatic again. Benralizumab may be a viable option for EoG refractory to systemic steroids but only as a short‐term adjunct therapy. More robust studies with long‐term data are needed, especially in this younger population.
{"title":"Pediatric eosinophilic gastritis treated with benralizumab: A case report","authors":"D. Cheung, Dawn Ebach, Benjamin Davis","doi":"10.1002/jpr3.12088","DOIUrl":"https://doi.org/10.1002/jpr3.12088","url":null,"abstract":"We report one of the first cases of eosinophilic gastritis (EoG) in a child under 12 years treated with benralizumab. At 7 years, our patient was started on benralizumab after failing to respond to various combinations of high‐dose omeprazole, milk elimination diet, oral viscous budesonide, and oral systemic steroids. He had a complete depletion of gastrointestinal tissue eosinophils with improved symptoms but had symptomatic flares with tapering of background therapy. However, after 4 years on benralizumab he became symptomatic again. Benralizumab may be a viable option for EoG refractory to systemic steroids but only as a short‐term adjunct therapy. More robust studies with long‐term data are needed, especially in this younger population.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"7 7","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141343408","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric autoimmune hepatitis has an incidence of 0.23/100.000 children in North America, with a bleak prognosis if left untreated. Steroids are the therapy of choice but are not always effective. B cell depletion is a safe and effective therapy that allows for a steroid‐sparing protocol, especially in patients who do not tolerate side effects.We retrospectively reviewed rituximab‐treated patients between 2017 and 2022. Demographics, previous treatments, reasons for B cell depletion, response, and adverse effects were noted.Six patients with a mean age of 10.2 years were included. All patients had comorbidities that rendered treatment with steroids unsuccessful or undesirable. Rituximab was started at a mean follow‐up of 8 months. After 6 months, the mean alanine transaminase and aspartate transaminase levels decreased from 575 IU/L and 342 IU/L, respectively, to 28 IU/L (p = 0.02) and 36 IU/L (p = 0.008), respectively. Mean γGT decreased from 105 to 25 IU/L (p = 0.01). Immunoglobulin G levels were normalized in all patients (p = 0.01). No severe adverse events were observed. One patient had persistent hypogammaglobulinemia, and another had lymphopenia.B‐cell depletion is an effective and safe treatment for autoimmune liver diseases and should be included as an option, particularly for relapsing patients in whom steroids are undesirable or have shown nonadherence.
{"title":"B cell depletion for autoimmune liver diseases: A retrospective review of indications and outcomes","authors":"Guillermo A. Costaguta, Fernando Álvarez","doi":"10.1002/jpr3.12098","DOIUrl":"https://doi.org/10.1002/jpr3.12098","url":null,"abstract":"Pediatric autoimmune hepatitis has an incidence of 0.23/100.000 children in North America, with a bleak prognosis if left untreated. Steroids are the therapy of choice but are not always effective. B cell depletion is a safe and effective therapy that allows for a steroid‐sparing protocol, especially in patients who do not tolerate side effects.We retrospectively reviewed rituximab‐treated patients between 2017 and 2022. Demographics, previous treatments, reasons for B cell depletion, response, and adverse effects were noted.Six patients with a mean age of 10.2 years were included. All patients had comorbidities that rendered treatment with steroids unsuccessful or undesirable. Rituximab was started at a mean follow‐up of 8 months. After 6 months, the mean alanine transaminase and aspartate transaminase levels decreased from 575 IU/L and 342 IU/L, respectively, to 28 IU/L (p = 0.02) and 36 IU/L (p = 0.008), respectively. Mean γGT decreased from 105 to 25 IU/L (p = 0.01). Immunoglobulin G levels were normalized in all patients (p = 0.01). No severe adverse events were observed. One patient had persistent hypogammaglobulinemia, and another had lymphopenia.B‐cell depletion is an effective and safe treatment for autoimmune liver diseases and should be included as an option, particularly for relapsing patients in whom steroids are undesirable or have shown nonadherence.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"58 34","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141344502","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Button batteries pose a significant threat to young children, and parents and caregivers play an important role in keeping children safe from button batteries, especially in the home environment. However, little is known regarding parent or caregiver perspectives on button battery safety, in particular, the threat they pose to children. The aim of this study was to examine perspectives on button battery safety to enable tailored prevention strategies and to examine parent and caregiver perspectives on button battery safety in the home.One‐hundred and seventy‐four parents and caregivers of children aged 0–5 completed a cross‐sectional online survey. Distribution occurred via social media. The survey contained multiple‐choice questions pertaining to button battery knowledge and attitudes.Most respondents recognised the dangers of button battery ingestion, yet only 37% knew of button battery ingestion symptoms. While 68% knew of household items containing button batteries, 21% were aware of product‐recall information sources. Approximately 64% understood immediate post‐ingestion steps, but only 31% were familiar with first‐aid procedures. From an attitudinal standpoint, 95% understood the importance of child supervision around button batteries, 78% prioritised battery safety in toy purchases, and 17% found current safety labels unsatisfactory. This study provides a foundation for ongoing investigation of parent and caregiver perspectives on button battery home safety.Ongoing, comprehensive education and public‐safety campaigns are needed to rectify gaps in knowledge regarding button battery safety, and to reduce the risk of button battery ingestion in children.
{"title":"‘You can be as vigilant as you can, yet they make their way in’: A descriptive study of parent and caregiver perspectives towards keeping children safe from button batteries","authors":"Anna M. Girardi, David N. Long, Gavin Austin","doi":"10.1002/jpr3.12096","DOIUrl":"https://doi.org/10.1002/jpr3.12096","url":null,"abstract":"Button batteries pose a significant threat to young children, and parents and caregivers play an important role in keeping children safe from button batteries, especially in the home environment. However, little is known regarding parent or caregiver perspectives on button battery safety, in particular, the threat they pose to children. The aim of this study was to examine perspectives on button battery safety to enable tailored prevention strategies and to examine parent and caregiver perspectives on button battery safety in the home.One‐hundred and seventy‐four parents and caregivers of children aged 0–5 completed a cross‐sectional online survey. Distribution occurred via social media. The survey contained multiple‐choice questions pertaining to button battery knowledge and attitudes.Most respondents recognised the dangers of button battery ingestion, yet only 37% knew of button battery ingestion symptoms. While 68% knew of household items containing button batteries, 21% were aware of product‐recall information sources. Approximately 64% understood immediate post‐ingestion steps, but only 31% were familiar with first‐aid procedures. From an attitudinal standpoint, 95% understood the importance of child supervision around button batteries, 78% prioritised battery safety in toy purchases, and 17% found current safety labels unsatisfactory. This study provides a foundation for ongoing investigation of parent and caregiver perspectives on button battery home safety.Ongoing, comprehensive education and public‐safety campaigns are needed to rectify gaps in knowledge regarding button battery safety, and to reduce the risk of button battery ingestion in children.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"10 2","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141339100","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kyle McKeown, Justin Pritchett, Annette Carlisle, Jie Zhang, Jay Lieberman, Dong Xi
To investigate differences in symptoms, allergy comorbidities, and eosinophilic inflammation at the time of diagnosis for patients with eosinophilic esophagitis (EoE) based on gender, race, and age of onset.A retrospective study was conducted at a multidisciplinary EoE clinic; the correlation between histological findings, previously identified symptoms, associated comorbidities, and demographics including gender, race, as well as age of onset was examined. Chi‐squared and Student's T‐tests were utilized for statistical analysis.A total of 91 patients were enrolled in this study, with 70% being male and 67% identifying as White. Among the patients, 45% had an early onset of EoE (defined as ≤6 years old). We revealed that White patients and females were significantly more likely to report dysphagia, while non‐White patients experienced significantly more vomiting symptoms and had a higher prevalence of asthma as a comorbidity. Early‐onset patients exhibited a significantly higher rate of vomiting and had elevated eosinophilic counts compared to patients with EoE onset at a regular age. We also revealed that abdominal pain is associated with a lower average proximal eosinophilic counts.Our study revealed the significant impact of gender, race, and age of onset on the phenotype and comorbidities of EoE, suggesting these factors should be considered when caring for these patients.
{"title":"Impact of gender, race, and age of onset on the phenotype and comorbidities of pediatric eosinophilic esophagitis","authors":"Kyle McKeown, Justin Pritchett, Annette Carlisle, Jie Zhang, Jay Lieberman, Dong Xi","doi":"10.1002/jpr3.12093","DOIUrl":"https://doi.org/10.1002/jpr3.12093","url":null,"abstract":"To investigate differences in symptoms, allergy comorbidities, and eosinophilic inflammation at the time of diagnosis for patients with eosinophilic esophagitis (EoE) based on gender, race, and age of onset.A retrospective study was conducted at a multidisciplinary EoE clinic; the correlation between histological findings, previously identified symptoms, associated comorbidities, and demographics including gender, race, as well as age of onset was examined. Chi‐squared and Student's T‐tests were utilized for statistical analysis.A total of 91 patients were enrolled in this study, with 70% being male and 67% identifying as White. Among the patients, 45% had an early onset of EoE (defined as ≤6 years old). We revealed that White patients and females were significantly more likely to report dysphagia, while non‐White patients experienced significantly more vomiting symptoms and had a higher prevalence of asthma as a comorbidity. Early‐onset patients exhibited a significantly higher rate of vomiting and had elevated eosinophilic counts compared to patients with EoE onset at a regular age. We also revealed that abdominal pain is associated with a lower average proximal eosinophilic counts.Our study revealed the significant impact of gender, race, and age of onset on the phenotype and comorbidities of EoE, suggesting these factors should be considered when caring for these patients.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"13 8","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141341696","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Jack J. Hachem, Sara Javadi, Milissa McKee, James Noel, Robert A. Noel
Adenocarcinoma of the colon is a rare diagnosis in pediatric patients. We present a previously healthy 15‐year‐old female who began experiencing escalating colicky abdominal pain and associated vomiting over 2 weeks in the setting of presumed acute gastroenteritis. A computed tomography scan revealed an obstruction in her descending colon. A multidisciplinary decision was made to perform a colonoscopy upon which a large, circumferential, friable lesion was discovered 40 cm from the anus. A colon decompression catheter was successfully inserted following CRE Balloon dilation to 13.5 mm beyond the mass, resulting in a significant discharge of fluid and gas. The patient underwent hemicolectomy with mass resection and colostomy. Biopsies confirmed poorly differentiated adenocarcinoma with “napkin‐ring” morphology and positive lymph node metastasis with extranodal extension.
{"title":"Pediatric colonic adenocarcinoma: A deceptive case of gastroenteritis and constipation","authors":"Jack J. Hachem, Sara Javadi, Milissa McKee, James Noel, Robert A. Noel","doi":"10.1002/jpr3.12080","DOIUrl":"https://doi.org/10.1002/jpr3.12080","url":null,"abstract":"Adenocarcinoma of the colon is a rare diagnosis in pediatric patients. We present a previously healthy 15‐year‐old female who began experiencing escalating colicky abdominal pain and associated vomiting over 2 weeks in the setting of presumed acute gastroenteritis. A computed tomography scan revealed an obstruction in her descending colon. A multidisciplinary decision was made to perform a colonoscopy upon which a large, circumferential, friable lesion was discovered 40 cm from the anus. A colon decompression catheter was successfully inserted following CRE Balloon dilation to 13.5 mm beyond the mass, resulting in a significant discharge of fluid and gas. The patient underwent hemicolectomy with mass resection and colostomy. Biopsies confirmed poorly differentiated adenocarcinoma with “napkin‐ring” morphology and positive lymph node metastasis with extranodal extension.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"57 2","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-05-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140974513","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Michelle M. Corrado, Melissa Wong, Laura Z. Fenton, Steven Moulton, Alexandra L. Kilgore
{"title":"Constipation: On knife edge","authors":"Michelle M. Corrado, Melissa Wong, Laura Z. Fenton, Steven Moulton, Alexandra L. Kilgore","doi":"10.1002/jpr3.12079","DOIUrl":"https://doi.org/10.1002/jpr3.12079","url":null,"abstract":"","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"7 8","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140980572","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Small bowel into small bowel intussusception can present with symptoms similar to those observed in patients with more common small bowel into large bowel intussusception. In most cases, intussusceptions isolated to the small bowel are self‐limited and less likely to result in bowel ischemia. Nonetheless, any patient with recurrent intussusception should be evaluated to assess location and for the presence of a pathologic lead point. We report a patient with recurrent small bowel into small bowel intussusception who underwent a comprehensive evaluation that revealed lymphoid hyperplasia in the absence of a pathologic lead point. His symptoms resolved after a dose of oral dexamethasone.
{"title":"Evaluation and novel treatment in a case of refractory small bowel‐small bowel intussusception","authors":"Danielle G. Rabinowitz, Paul A. Rufo","doi":"10.1002/jpr3.12078","DOIUrl":"https://doi.org/10.1002/jpr3.12078","url":null,"abstract":"Small bowel into small bowel intussusception can present with symptoms similar to those observed in patients with more common small bowel into large bowel intussusception. In most cases, intussusceptions isolated to the small bowel are self‐limited and less likely to result in bowel ischemia. Nonetheless, any patient with recurrent intussusception should be evaluated to assess location and for the presence of a pathologic lead point. We report a patient with recurrent small bowel into small bowel intussusception who underwent a comprehensive evaluation that revealed lymphoid hyperplasia in the absence of a pathologic lead point. His symptoms resolved after a dose of oral dexamethasone.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"9 17","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141005041","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Ariel Porto, Jonathan Lebowitz, Robert Byrd, Elaine Morgan, Joshua D. Prozialeck
Malakoplakia is a rare inflammatory condition characterized by impaired macrophages unable to completely digest and kill phagocytized bacteria, resulting in partially digested bacterial components accumulating within the phagolysosome. Malakoplakia typically presents in immunocompromised individuals due to underlying disease or to medication effects and is rarely diagnosed in the pediatric population. The urinary tract is the most commonly involved site, followed by the gastrointestinal (GI) tract, mainly affecting the descending colon, sigmoid colon, and rectum. Treatment focuses on the use of antibiotics that concentrate in macrophages such as quinolones and trimethoprim‐sulfamethoxazole as well as cholinergic agents such as bethanechol, which raise intracellular levels of cyclic guanosine monophosphate in macrophages to improve bactericidal activity. We report a rare case of GI tract malakoplakia in a pediatric patient undergoing treatment for leukemia.
{"title":"Malakoplakia: A rare cause of hematochezia in pediatric patients","authors":"Ariel Porto, Jonathan Lebowitz, Robert Byrd, Elaine Morgan, Joshua D. Prozialeck","doi":"10.1002/jpr3.12075","DOIUrl":"https://doi.org/10.1002/jpr3.12075","url":null,"abstract":"Malakoplakia is a rare inflammatory condition characterized by impaired macrophages unable to completely digest and kill phagocytized bacteria, resulting in partially digested bacterial components accumulating within the phagolysosome. Malakoplakia typically presents in immunocompromised individuals due to underlying disease or to medication effects and is rarely diagnosed in the pediatric population. The urinary tract is the most commonly involved site, followed by the gastrointestinal (GI) tract, mainly affecting the descending colon, sigmoid colon, and rectum. Treatment focuses on the use of antibiotics that concentrate in macrophages such as quinolones and trimethoprim‐sulfamethoxazole as well as cholinergic agents such as bethanechol, which raise intracellular levels of cyclic guanosine monophosphate in macrophages to improve bactericidal activity. We report a rare case of GI tract malakoplakia in a pediatric patient undergoing treatment for leukemia.","PeriodicalId":17618,"journal":{"name":"JPGN Reports","volume":"12 11","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141030726","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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