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Background: Allied disorders of Hirschsprung's disease (ADHD) exhibit symptoms akin to those of Hirschsprung's disease, primarily characterized by intestinal obstruction, bowel dilatation, and chronic constipation. The occurrence of amyloid complications in patients with ADHD is infrequent. In this report, we present a case of ADHD with intestinal ulcers as the initial gastrointestinal manifestation, and subsequent pathological examination revealed the presence of amyloid deposits in the colonic mucosa. Case Report: A male patient, aged 20, exhibited recurring abdominal distension and intestinal obstruction for a duration of three years. Multiple colonoscopies revealed the presence of recurrent colonic ulcers, with pathological examination indicating the existence of amyloid deposits within the mucosal layer of the colon. Abdominal CT scans suggested colonic dilatation. Following a multidisciplinary consultation, a subtotal resection of the colon was performed, and subsequent postoperative pathology confirmed a decrease and absence of myenteric plexus ganglion cells. Considering the patient's symptoms and the findings from the postoperative pathology, a diagnosis of ADHD was made. The patient's symptoms resolved postoperatively and he was discharged from the hospital and followed up for 1 year in stable condition. Conclusion: Our study highlights the potential association between ADHD and the initial presentation of recurrent colonic ulcers, accompanied by amyloid deposition in the intestinal mucosa. This finding suggests a possible pathogenic mechanism for ADHD and offers a novel perspective on its diagnosis.
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BACKGROUND: Ischemic colitis (IC) is also known as colon ischemia and is caused by colon vascular occlusion or nonocclusion, which results in a reduced blood supply to the colon and is not significant enough to maintain the metabolic function of cells, leading to intestinal wall ischemia. Its main symptoms include abdominal pain, diarrhea, and bloody stool. In severe cases, intestinal gangrene, peritonitis, intestinal stenosis and even intestinal obstruction may occur. IC induced by long-term use of certain special drugs is relatively rare in clinical practice. This article describes the clinical diagnosis and treatment of a typical case and provides a new treatment idea for the treatment of IC. CASE SUMMARY: The patient was admitted to the hospital with "abdominal pain for half a month and bloody stool with mucous and pus for 3 d" and was diagnosed with "IC". Symptomatic and supportive treatment, such as antibiotics (levofloxacin), acid inhibition and stomach protection, fluid replenishment, and intravenous nutrition, was given. The patient's colonic ulcers were considered to be related to the oral administration of platelet (PLT)-raising capsules; the patient was asked to stop PLT-raising drugs for selective review via colonoscopy, and antibiotics and mesalazine enteric-coated tablets were stopped. Under the guidance of hematology consultation, 60 mg of methylprednisolone was given in combination with PLT infusion to increase the PLT. After treatment, the patient's condition stabilized, the patient's stool turned yellow, the patient's symptoms improved, and the patient was allowed to leave the hospital. CONCLUSION: PLT-raising capsules can lead to IC, so clinicians should have a full understanding of the application of these drugs in the treatment of various causes of thrombocytopenia, weigh the advantages and disadvantages, and observe patients closely.
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BACKGROUND: Amyloidosis, an systemic disorder featuring an accumulation of misfolded proteins, is a significant diagnostic challenge because of its broad range of symptoms. The symptoms of amyloidosis vary depending on the affected organs. Amyloid accumulation in the kidney generally manifests as proteinuria or impaired kidney function, whereas cases with gastrointestinal (GI) involvement present as abdominal pain, weight loss, or GI bleeding. CASE DESCRIPTION: We report a case of systemic immunoglobulin light chain (AL) amyloidosis involving the colon and kidney in a 75yearold female who presented with intermittent lower abdominal pain and hematochezia. A colonoscopy revealed multiple ulcerations and a submucosal hematoma with κ light chain deposition confirmed by biopsy. The patient had many comorbidities, including renal tuberculosis, chronic kidney disease, diabetes, coronary heart disease (CHD), and paroxysmal atrial fibrillation, which rendered her clinical manifestations confusing. Her condition was relatively stable during treatment with bortezomib and dexamethasone for 4 cycles. CONCLUSIONS: Systemic amyloidosis usually has a poor prognosis since most cases are detected in the late disease phase. Early disease detection depends on a comprehensive understanding of the disease and a keen recognition of the lesion. We suggest that in patients with hematochezia, colonic ulcer, and submucosa hematoma, amyloidosis with colonic involvement should be considered when other diseases are excluded.
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INTRODUCTION: Solitary ulcers in the colon are rare and infrequent; little over 200 cases have been reported in medical literature. We present a case of a patient presenting with a solitary colonic ulcer associated with NSAIDs intake, mimicking a malignant lesion. A review of the literature is also revised. PRESENTATION OF CASE: 68- year-old female patient with past history of nonsteroidal anti-inflammatory drugs (NSAID) intake for chronic pain, complaining of severe abdominal pain was admitted to our teaching hospital. The diagnosis of a low-grade dysplasia was made with colonoscopy and biopsy, a malignant lesion could not be ruled out. A laparoscopy right colectomy was performed without complications. The final diagnosis resulted in a solitary cecal ulcer. DISCUSSION: The majority of the cases of solitary colonic ulcers occur in the ascending colon, at the cecum, which has been attributed mostly to the intake of NSAIDs. There could be solitary colonic ulcers in other portions of the large intestine, caused by different etiologies: ischemia, inflammatory disease, sterocoraceus ulcers, ulcers caused by infections, among other more uncommon causes. The diagnosis is often made through a biopsy of the tissue during a colonoscopy, with either surgical or conservative care. CONCLUSION: The diagnosis of solitary cecal ulcer should be considered in patients presenting with RLQ abdominal pain and with history of NSAIDs consumption. Recognition of this diagnosis by surgeons, ruling out malignancies, understanding the morphologic features, and carefully taking the patient's history are essential for the diagnosis and treatment of this uncommon disease.
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AIM: To investigate management of patients who develop ipilimumab-mediated enterocolitis, including association of endoscopic findings with steroid-refractory symptoms and utility of infliximab as second-line therapy. METHODS: We retrospectively reviewed all patients at our center with metastatic melanoma who were treated with ipilimumab between March 2011 and May 2014. All patients received a standard regimen of intravenous ipilimumab 3 mg/kg every 3 wk for four doses or until therapy was stopped due to toxicity or disease progression. Basic demographic and clinical data were collected on all patients. For patients who developed grade 2 or worse diarrhea (increase of 4 bowel movements per day), additional data were collected regarding details of gastrointestinal symptoms, endoscopic findings and treatment course. Descriptive statistics were used. RESULTS: A total of 114 patients were treated with ipilimumab during the study period and all were included. Sixteen patients (14%) developed ≥ grade 2 diarrhea. All patients were treated with high-dose corticosteroids (1-2 mg/kg prednisone daily or equivalent). Nine of 16 patients (56%) had ongoing diarrhea despite high-dose steroids. Steroid-refractory patients received one dose of intravenous infliximab at 5 mg/kg, and all but one had brisk resolution of diarrhea. Fourteen of the patients underwent either colonoscopy or sigmoidoscopy with variable endoscopic findings, ranging from mild erythema to colonic ulcers. Among 8 patients with ulcers demonstrated by sigmoidoscopy or colonoscopy, 7 patients (88%) developed steroid-refractory symptoms requiring infliximab. With a median follow-up of 264 d, no major adverse events associated with prednisone or infliximab were reported. CONCLUSION: In patients with ipilimumab-mediated enterocolitis, the presence of colonic ulcers on endoscopy was associated with a steroid-refractory course.
Assuntos
Corticosteroides/farmacologia , Anticorpos Monoclonais/efeitos adversos , Antineoplásicos/efeitos adversos , Doenças do Colo/tratamento farmacológico , Resistência a Medicamentos , Enterocolite/tratamento farmacológico , Melanoma/tratamento farmacológico , Neoplasias Cutâneas/tratamento farmacológico , Administração Intravenosa , Corticosteroides/efeitos adversos , Corticosteroides/uso terapêutico , Anticorpos Monoclonais/administração & dosagem , Anticorpos Monoclonais/uso terapêutico , Antineoplásicos/administração & dosagem , Antineoplásicos/uso terapêutico , Antígeno CTLA-4/antagonistas & inibidores , Antígeno CTLA-4/imunologia , Doenças do Colo/induzido quimicamente , Doenças do Colo/diagnóstico , Doenças do Colo/imunologia , Colonoscopia , Diarreia/tratamento farmacológico , Diarreia/etiologia , Enterocolite/induzido quimicamente , Enterocolite/diagnóstico , Enterocolite/imunologia , Feminino , Fármacos Gastrointestinais/uso terapêutico , Humanos , Infliximab/uso terapêutico , Ipilimumab , Masculino , Pessoa de Meia-Idade , Prednisona/efeitos adversos , Prednisona/farmacologia , Prednisona/uso terapêutico , Estudos Retrospectivos , Úlcera/induzido quimicamente , Úlcera/diagnóstico , Úlcera/imunologiaRESUMO
A 94-year-old female with end-stage renal disease presents with fever, fatigue, and hematochezia. She had previously resided in Hunan Province, China, and Myanmar, and she immigrated to Taiwan 30 years ago. Colonoscopy revealed a colonic ulcer. Biopsy of the colonic ulcer showed ulceration of the colonic mucosa, and many Paragonimus westermani-like eggs were noted. Serum IgG antibody levels showed strong reactivity with P. westermani excretory-secretory antigens by ELISA. Intestinal paragonimiasis was thus diagnosed according to the morphology of the eggs and serologic finding. After treatment with praziquantel, hematochezia resolved. The present case illustrates the extreme manifestations encountered in severe intestinal paragonimiasis.