Patients with Crohn's disease generally present with chronic diarrhoea and/or abdominal pain. However, it may be the extraintestinal manifestations as orofacial granulomatosis (OFG)--a rare syndrome with chronic swelling of the lips and the lower half of the face combined with oral ulcerations and hyperplastic gingivitis--that urge patients to seek medical advice. We report two rare cases in which swelling of the lips and cheeks were the initial symptoms that finally led to the diagnosis of Crohn's disease.
Patients with inflammatory bowel disease (IBD) often undergo several radiologic imaging studies, which -with the exception of magnetic resonance imaging (MRI) and B-scan ultrasound (US) -subject patients to ionizing radiation. With contrast enhanced ultrasound microvascular imaging of the bowel is possible. Hence, the aim of our study was to assess the perfusion pattern of inflamed bowel walls in Crohn's disease compared with healthy volunteers quantitatively using a specific quantification software.We evaluated 4 volunteers and 20 patients with proven Crohn's disease, who went through an active episode based on clinical symptoms and complementary imaging by MRI (19 patients) and computed tomography (1 patient), respectively, with dynamic contrast-enhanced ultrasound (CEUS) using a second generation contrast agent (SonoVue ® , Bracco, Germany). Retrospectively, we applied the quantification software Qontrast ® (Bracco, Italy) to obtain contrast-enhanced sonographic perfusion maps for each lesion.Patients had significant higher peak values (median 46.86, lower quartile 37.91, upper quartile 53.20) and significant higher regional blood volume (median 2133.65, lower quartile 1202.90, upper quartile 2820.44) than volunteers.Considering the very low peak value of the healthy, it is easy to understand that the time-to-peak was significantly shorter in the volunteers (median 4.45, lower quartile 1.82, upper quartile 6.88) than in the patients (median 12.15, lower quartile 9.18, upper quartile 15.74). Our study showed clear differences between inflamed and normal bowel wall vascularity regarding all perfusion parameters. These results show that a quantitative assessment of the bowel wall vascularisation and inflammation, respectively, is possible. The software used here enables us to collect data, not only in a semi-quantitative but also in a reproducible, quantitative manner which is comparable with the evaluation of CT or MRI generated data.
BackgroundTherapy-refractory persistent hypoparathyroidism after extensive neck surgery is a rare but severe complication. Parathyroid allotransplantation may represent a definitive treatment option.Case presentationA 32-year old female was referred to our hospital with intractable persistent hypocalcemia after neck surgery for papillary thyroid cancer. Despite optimal medical treatment including calcium and vitamin D supplementation and even hormonal replacement therapy hypocalcemic symptoms failed to improve. The quality of life was considered very low. In light of the unsuccessful medical therapy and the young age of the patient parathyroid allotransplantation seemed an attractive treatment option to restore normal calcium homeostasis despite of the need for immunosuppressive therapy after the procedure. Therefore, we performed living-donor allotransplantation of two healthy parathyroid glands to the recipient’s left forearm. The surgical intervention was successful. Neither the donor nor the recipient showed any complications. In the postoperative course clinical symptoms of hypocalcemia significantly improved whereas serum calcium and parathyroid hormone (PTH) levels progressively increased into the normal range. Former intense replacement therapy could be discontinued completely in a stepwise fashion. To date, nearly three years after transplantation, the patient remains asymptomatic with normal serum levels of calcium and PTH.ConclusionSuccessful living-donor parathyroid allotransplantation for postsurgical hypoparathyroidism represents an innovative therapeutic strategy that could provide the definitive treatment in those patients in which the disease is therapy-refractory. The procedure can be justified even in nontransplant recipients. Retrieval of parathyroid glands from healthy donors is feasible and safe.
Ultrasound as a widely available radiation-free imaging method would be preferable for assessing inflammatory activity of CD, particularly since the ultrasound findings correlate significantly with a histopathological scoring system.
Quantitative evaluation with CEUS, particularly the calculation of the ratio TTP (s)/Peak (%), provides a simple method for assessment of inflammatory activity in UC.
In our case, a 45-year-old male patient had multiple fractures accompanied by hypophosphatemia. FGF-23 levels were significantly increased, and total body magnetic resonance imaging (MRI) revealed a tumor mass located at the distal tibia leading to the diagnosis of tumor-induced osteomalacia (TIO). After resection of the tumor, hypophosphatemia and the increased levels of FGF-23 normalized within a few days. Subsequent microscopic examination and immunohistochemical analysis revealed a phosphaturic mesenchymal tumor mixed connective tissue variant (PMTMCT) showing a positive expression of somatostatin receptor 2A (SSTR2A), CD68, and Periostin. Electron microscopy demonstrated a poorly differentiated mesenchymal tumor with a multifocal giant cell component and evidence of neurosecretory-granules. However, the resected margins showed no tumor-free tissue, and therefore a subsequent postoperative radiotherapy was performed. The patient is still in complete remission after 34 months. Tumor resection of PMTMCTs is the therapy of choice. Subsequent radiotherapy in case of incompletely resected tumors can be an important option to avoid recurrence or metastasis even though this occurs rarely. The prognostic value of expression of Periostin has to be evaluated more precisely in a larger series of patients with TIO.
Introduction: Many reports, mainly from the US and Canada but also a recent report from a center in Europe, have documented the increasing impact of Clostridium difficile infections in patients with inflammatory bowel disease (IBD) during the last years. To determine the prevalence of C. difficile infections in hospitalized IBD patients in a tertiary referral center in Germany, we conducted this retrospective analysis. Methods: Data of all IBD in-patients treated due to an acute flare of their IBD at the Department of Internal Medicine I of the University of Regensburg between January 1, 2001, and June 30, 2008, were analyzed. In patients with a concomitant diagnosis of C. difficile infection, further variables such as IBD-related treatment at the time of infection or outcome were examined. Results: In total, 995 in-patients with IBD were treated in this hospital [638 patients with Crohn’s disease (CD), 357 with ulcerative colitis (UC)] during the study period. Of these, 279 patients with CD and 242 patients with UC were admitted with an acute flare and suffering from diarrhea and abdominal pain. Only 10 of those were diagnosed as having a concomitant infection with C. difficile. Six patients were female and the median age was 49 years (range: 15–80). Six patients with C. difficile infections suffered from UC and 4 patients from CD, all with previous colonic involvement. Eight patients used immunosuppressive therapies; only 2 patients were treated with antibiotics before infection. Conclusion: In contrast to recent reports from other countries, only a low percentage of hospitalized patients with acute flares of their IBD were identified as having an underlying C. difficile infection in this German tertiary referral center. However, in IBD patients with an acute flare, a concomitant C. difficile infection should be excluded, especially in patients with immunosuppressive treatment and colonic involvement of their disease. Further research is needed to evaluate if regions with different risks of C. difficile infections exist and to find out more about potential reasons for this observation.
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