Enterohepatic fistula in a Crohn's disease patient: A case report.
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ABSTRACT:
SUBMITTER: Van Backer JT
PROVIDER: S-EPMC5558617 | biostudies-other | 2017 Aug
REPOSITORIES: biostudies-other
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Project description:BackgroundMobile valvular masses are often considered pathognomonic for infective endocarditis. We present a case of a young patient with mobile valvular masses in the context of myocarditis likely secondary to active ileal Crohn's disease. Cardiac magnetic resonance (CMR) imaging was crucial in diagnosing and monitoring our patient.Case summaryA 27-year-old woman presented with pleuritic chest pain, dyspnoea, and a 3-day history of fevers. She also reported a 2-month history of intermittent visual loss in her right eye. She had a history of histologically proven ileal Crohn's disease, diagnosed 5 months prior. She was haemodynamically unstable on presentation. Abnormalities on a transthoracic echocardiogram necessitated a transoesophageal echocardiogram. After blood cultures were sent, the patient was commenced on empirical treatment for infective endocarditis with gentamicin and flucloxacillin. Eight days after her initial presentation, all blood cultures remained negative and she was changed to empirical treatment for culture negative endocarditis with ceftriaxone and vancomycin, according to local protocol. Despite 8 days of treatment for infective endocarditis she remained febrile. A CMR was organized on Day 9 and this showed myocarditis, which changed the treatment paradigm. She responded swiftly to steroids and anti-coagulation.DiscussionIn this case, echo-dense valvular lesions are not pathognomonic for infective endocarditis and a careful diagnostic process involving multi-modality imaging, including CMR, occurred to arrive at a diagnosis of myocarditis likely secondary to Crohn's disease.
| S-EPMC7501932 | biostudies-literature
Project description:Introduction and importanceReports of enterosalpingeal fistulae complicating Crohn's disease are scarce. They involve the last ileal loop and lead to a progressive destruction of the salpinx. Usually, no genital symptoms are found. In all the cases reported in the literature, the fistula was diagnosed intra-operatively and resection of the right salpinx was performed without the patient's pre-operative consent.Case presentationWe describe 2 cases of women presenting with an Enterosalpingeal fistulae complicating Crohn's disease. Radiological findings allowed a pre-operative diagnosis. Thus, the patients were warned of the right salpinx resection and consent was obtained.Clinical discussionEnterosalpingeal fistulae complicating Crohn's disease are exceptional. Indeed, to the best of our knowledge, only five cases have been reported till now. In all the reported cases, no genital signs were present. As for our patients who didn't experience such symptoms. Moreover, no radiological evidence of the enterosalpingeal fistula was found in the literature. Consequently, the fistula was always diagnosed intra-operatively. For our patients, radiological findings allowed a pre-operative diagnosis. This permitted to warn them of a possible resection of the fallopian tube. Intra-operative findings were unfortunately conflicting with its preservation.ConclusionEnterosalpingeal fistula is an exceptional complication of the Crohn's disease. No clinical findings are present. The diagnosis should be evoked when the CT-scan or the MRI show an abnormal apposition between the fallopian tube, the last loop and the cecum. Surgical resection of the involved salpinx with the diseased intestinal segment is unfortunately usually needed in a young patient population.
| S-EPMC8384770 | biostudies-literature
Project description:Introduction:Holt-Oram syndrome (HOS) is an uncommon autosomal dominant disorder defined by congenital cardiac defects, some anatomical deformities in the upper limb and conduction abnormalities. Sequence alteration of TBX5 gene located on chromosome 12 has associated with HOS. Case report:We present the case of a 26-year-old female with known upper limb alteration and ventricular septal defect who later in life developed Crohn's disease. Conclusion:To the best of our knowledge association of Holt-Oram syndrome with Crohn's disease has not been reported in literature before. Therefore, a possible genetic connection between Holt-Oram syndrome and Crohn's disease remains to be determined.
| S-EPMC6194948 | biostudies-literature
Project description:Mesenchymal stem cell therapy is a promising treatment for perianal Crohn's fistulas refractory to conventional therapy, which are an extremely morbid complication and a true therapeutic challenge. Autologous adipose-derived stromal vascular fraction (ADSVF) is an easily accessible source of cells with angiogenic, anti-inflammatory, immunomodulatory, and regenerative properties. Here, we describe a case involving a patient with severe perianal Crohn's fistulas refractory to the best medical and surgical practices who received local treatment with ADSVF and microfat. This patient was first examined under anesthesia with drainage via seton placement; 1 week later, on a single day, he underwent adipose tissue extraction, ADSVF and microfat preparation, and the local injection of 14 ml of microfat and approximately 20 million viable ADSVF cells into the soft tissue around the fistulas. No serious adverse events were observed. At the first endpoint at 12 weeks, the fistula had clinically healed with complete re-epithelialization of all external openings; no fistula tract was detected on magnetic resonance imaging, confirming this finding. This good clinical outcome was sustained at 48 weeks and was associated with a reduction in the severity of perianal disease and an improvement in quality of life. The current case highlights the therapeutic potential of a new cellular treatment for Crohn's patients with refractory perianal fistulas based on the innovative hypothesis that the combined action of ADSVF in association with the trophic characteristics of a microfat graft could be beneficial for this condition. TRIAL REGISTRATION:EudraCT number 201325, NCT02520843 . Registered on 5 August 2015.
| S-EPMC5764003 | biostudies-literature
Project description:IntroductionAzathioprine (AZA) has been widely used for the treatment of various immune-related diseases and has become a mainstay in the treatment of inflammatory bowel disease. However, patients with genetic mutations may experience severe adverse events when treated with azathioprine. Most of the previous literature focused on the TPMP gene-related adverse reactions, herein, we report a case of Crohn's disease patient with nucleoside diphosphate-linked moiety X motif 15 gene (NUDT15) variation and wild-type TPMP gene who developed toxoplasma gondii infection after azathioprine treatment.Patient concernsA 56-year-old Crohn's disease patient developed toxoplasma gondii infection within 2 months after the administration of azathioprine; however, he had no relevant high-risk factors.DiagnosisSubsequent genetic testing revealed that the patient was heterozygous for NUDT15. Therefore, it was reasonable to consider that the patient's genetic mutation resulted in reduced tolerance to azathioprine, leading to low immunity and eventually toxoplasma infection.InterventionsAZA was then discontinued; after anti-infection, antipyretic and other supportive treatments were administered, the patient's condition gradually improved.OutcomesThe patient was followed up at 1, 3, and 6 months after discharge; fortunately, he was in good health.ConclusionWe report a case of Crohn's disease in a patient who developed severe pneumonia caused by toxoplasma gondii infection due to the administration of AZA, with normal TPMP gene but NUDT15 gene mutation. This indicates that NUDT15 variation may contribute to severe adverse events in patients treated with azathioprine, and we suggest that NUDT15 genotype be detected before the use of azathioprine in order to provide personalized therapy and reduce side effects.
| S-EPMC8104275 | biostudies-literature
Project description:IntroductionHypophosphatemia occurs commonly in inflammatory bowel disease (IBD) patients and can cause considerable morbidity. The differential diagnoses in IBD include nutritional causes and hypophosphatemia induced by some formulations of intravenous iron infusions.Case presentationWe present the case of a 37-year-old man with active Crohn's disease, presenting with difficulty walking and fractures of the vertebrae and calcaneus. He had long-standing hypophosphatemia. Nutritional causes for hypophosphatemia were considered in the first instance given the presence of chronic diarrhea and vitamin D deficiency; however, there was minimal response to appropriate supplementation with oral phosphorous and vitamin D. Iron infusion-induced hypophosphatemia was then considered, but the nadir phosphate level preceded any iron infusion. Therefore, work-up was undertaken for less common causes. He was ultimately diagnosed with tumor-induced osteomalacia, caused by excess fibroblast growth factor 23 (FGF23) secretion from a phosphaturic mesenchymal tumor about the knee. He had complete resolution of symptoms and biochemical abnormalities following successful resection of the tumor.ConclusionThis case illustrates the approach to investigation of hypophosphatemia in IBD patients. If the time course and response to phosphate supplementation are not as expected for nutritional or iron infusion-induced hypophosphatemia, less common causes should be considered.
| S-EPMC10896610 | biostudies-literature
Project description:We have cultured Mycobacteria avium subspecies hominissuis (MAH) from the blood of a patient with Crohn's disease. The patient is a 21 year-old-female with a diagnosis of Crohn's disease for two years. She had been treated with corticosteroids and Humira for six months. A blood specimen was cultured in a specialized medium, and there was visible bacterial growth present in the liquid culture medium after eight weeks. PCR analysis of the bacterial growth and subsequent direct sequencing of the PCR amplicon confirmed the presence of MAH. The significance of this finding is discussed.
| S-EPMC5714243 | biostudies-literature
Project description:Introduction and importanceTextiloma is a retained surgical item such as a sponge or gauze that is unintentionally left in the surgical field after the wound closure. Here, we present the first reported surgical gauze that penetrated the intestine, made a duodenal-ceca fistula and then stuck far away in the ileum. Mechanical obstruction didn't appear clinically or even in radiological investigations because of the fistula which provided the intestinal continuity.Case presentationWe report a 34-year-old man with a previous abdominal interventions complained of cramping, frequent vomiting and presence of undigested food in stool. The frequency of the bowel movement increased recently. Endoscopies, radiological investigations and pathological findings figure out a duodenal-ceca fistula with nonspecific inflammatory tissues in the intestinal biopsy. When we performed the abdomen surgery, retained gauze in the ileum was taken out and the duodenal-ceca fistula was fixed.Clinical discussionGauze or sponge that is forgotten in the surgical field called gossypiboma, textiloma, gauzoma or cottonoid. It could present with various complaints; as an acute or chronic problem, clear or ambiguous symptoms. It could reside in a space; extend across a gap, migrate through a tissue, or even make a fistula between lumina like in our case.ConclusionTextiloma could change pre-operative diagnosis, intra-operative techniques, postoperative follow-up plan and prognosis. This is the first report proves its ability to mimic inflammatory diseases that penetrate two different lumina and perform fistula. So it should be written in the list of any differential diagnosis when the patient has a previous procedure or surgery.
| S-EPMC7841073 | biostudies-literature
Project description:BackgroundAortopulmonary fistula is a rare complication of ascending aorta pathology. Presentation is most commonly dramatic with acute onset chest pain or heart failure secondary to left to right shunting. We describe a patient with acquired aortopulmonary fistula who had an insidious onset of heart failure as his presenting complaint. We also highlight the utility of multimodality cardiac imaging in establishing the diagnosis.Case summaryA 79-year-old male patient with a history of coronary artery bypass graft surgery and mechanical aortic valve replacement, 23 years prior, presented with exertional dyspnoea of 7 months duration. An initial workup that included transthoracic and transoesophageal echocardiography as well as coronary and bypass graft angiography failed to diagnose an acquired aortopulmonary fistula complicating an ascending aortic pseudoaneurysm. Upon referral to our institution, the correct diagnosis was suspected on off-axis transthoracic echocardiography. The fistula was subsequently confirmed, and the extent of ascending aorta pathology defined via a multimodality imaging approach that consisted of transoesophageal echocardiography and cardiac computed tomography. The patient underwent successful surgical repair and was discharged in a stable condition.DiscussionAcquired aortopulmonary fistula is a rare clinical entity. We describe a patient who had an insidious presentation of heart failure and found to have a large ascending aortic aneurysm that eroded into the main pulmonary artery creating a fistulous communication. The diagnosis was delayed and required a high index of suspicion and multimodality cardiac imaging.
| S-EPMC7501908 | biostudies-literature
Project description:IntroductionCrohn's disease (CD) is an idiopathic inflammatory disorder of unknown etiology with genetic, immunologic, and environmental influences. Infliximab is a treatment modality for fistulated Crohn's disease. Infliximab induced hemolysis is rare and very few cases reported before in Ulcerative colitis (UC) but not in Crohn's disease.Case presentationWe are reporting a 63 years old gentleman who was diagnosed as Crohn's disease and started on Tumor necrosis factor Inhibitor (TNF) -α i.e. (infliximab - Remsima) infusion. The course was complicated by Coomb's negative hemolytic anemia which is suggestive of non-immune drug induced hemolysis. Our patient was treated with steroid and conservative measures. Upon following up, his hemoglobin level as well as all hemolytic markers showed dramatic improvement. Adalimumab was used to treat this patient as an alternative choice without relapse of hemolysis.Clinical discussionDrug induced Hemolysis is not a well-known complication post receiving Tumor necrosis factor Inhibitor (TNF) -α infusion in patients with Crohn's disease. Coombs negative hemolysis keeps in favor of non-immune drug induced rather than other differentials in our case scenario.ConclusionAlthough cross-reactivity is expected between one biological agent to another, in our case the use of Adalimumab as alternative choice post Tumor necrosis factor Inhibitor (TNF) -α (Remsima infliximab) induced hemolysis did not cause hemolysis or any type of reaction.
| S-EPMC8403549 | biostudies-literature