| Background: Encrusted cystitis was first described by Francois in1914whoreported "an ulcerated inflammatory bladder with calcium deposits on its wall,causing intolerable functional consequences for the patient". It has been reportedsporadically during the past Century, and didn’t have an unified name. Some foreignscholars called it encrusted cystitis, others called it alkaline-encrusted cystitis, orincrusted cystitis. So, for the time being, we called this disease Encrusted Cystitis inthis article. It is a very rare disease. The etiology and pathogenesis of EC are stillControversial. EC have no characteristic symptoms, there are only some such asfrequency, urgency, dysuria and gross hematuria in the early stage, which are similarto the symptoms of common urinary infection. To later just presents suprapubic painand bladder mucosa encrustation. Because of its rare incidence, most doctors are notalert with EC, and then it’s difficult to make a early diagnosis of EC. So far, there areno standard therapeutic methods of EC. Treatment of encrusted cystitis consists of the4complementary elements of radical treatment of infection, acidification of the urineand chemolysis, elimination of calcified plaques containing microorganisms andsymptomatic therapy. Not only because the pathogen of EC is resist to a large numberof antibiotics, but also there will be residual encrustation that contains bacteria onbladder mucosa after surgery, the treatment of EC is very difficult. In this case report,the patient complained of frequency, urgency, dysuria, gross hematuria andsuprapubic pain after ureteroscopy lithotripsy, and was diagnosed EC two monthslater. Via the combined treatment of trans urethral resection of the crystals, bladderirrigation of sodium hyaluronate and detrusor injection of A-botulinum toxin, he wascured. Due to the rare incidence, the difficult diagnosis and therapy, and no reportsabout the treatment methods with bladder irrigation of sodium hyaluronate or detrusorinjection of A-botulinum toxin for EC, we make this case report and review relevantliterature.Objedtives: To report a case of encrusted cystiti（sEC）, and to review the relatedliteratures. In this paper, we will discuss the pathogenesis, clinical characteristics,diagnosis, treatment and prognosis of this rare disease. Methods: Describe the clinical symptoms and signs, laboratory findings,imaging examinations, stone composition, pathologic results, therapeutic methods andprognosis of this case. And also review the related literatures.Results: EC is a chronic inflammatory disease of urinary epithelium which hasulcerated and encrusted bladder mucosa. It is rare, and can occur in any age, and theincidence in renal transplant patients is highest. It occurs mainly inimmunocompromised or debilitated patients who have had urologic procedures. Theetiology and pathogenesis of EC are still Controversial, but most scholars considerthere is a relationship between EC and Corynebacterium urealuticum(CU). EC haveno characteristic symptoms, there are only some such as frequency, urgency, dysuria,gross hematuria, suprapubic pain, mucosa encrustation and ammonin sell in urine.The urine of EC patients contain erythrocytes, leukocytes, struvite, apatite crystals.Urine pH is always alkaline, urine culture is always positive for CU. Imageexamination show mucosa encrustation. Cystoscopy shows an inflammatory mucosawith ulcerations and superficial whitish plaques corresponding to encrustedcalcifications. Histologic examination shows three distinct layers: a superficial layerwith necrosis and microcalcifications; an intermediate layer with inflammatorychanges; and a third normal layer corresponding to bladder muscularis.Crystallographic analysis of wall encrustations and stones reveals predominantlystruvite. Only combine history, symptoms, physical examination and auxiliaryexamination above mentioned just can we make the diagnosis of EC. But alkalineurine and positive for CU is not obligatory. As long as image examination andcystoscopy revealed mucosa ulceration and encrustation, and schistosomiasis ofbladder, urinary tuberulosis, plaques of vesical leukoplakia, bladder tumour necrosis,encrustation after formaldehyde or cyclophosphamide injections, encrustation afterintravesical instillations of mitomycin C, encrustation after pelvic radiotherapy havebeen excluded, we can reach the diagnosis of EC. Vesical calcification is not peculiarfor EC, and can also be Observed in others diseases. Many kinds of diagnoses shoudbe excluded, such as schistosomiasis, tuberculosis, urinary tract leukoplakia, necroticbladder carcinoma, lesions after formaldehyde or cyclophosphamide injections,lesions after intravesical instillations of mitomycin C, lesions after pelvic irradiation.Treatment of encrusted cystitis consists of the4complementary elements of radicaltreatment of infection, acidification of the urine and chemolysis, elimination ofcalcified plaques containing microorganisms and symptomatic therapy. This case was a27-year-old man, who presented to our hospital after2months of recalcitrant grosshematuria, pyuria, frequency, urgency, dysuria and suprapubic pain. Two monthsbefore, he underwent a ureteroscopic procedure for ureter stone and the operation wassuccessful. Before surgery, ultrasonography revealed left ureteral calculus and normalbladder. Intravenous urography(IVU) showed left ureteral calculus, lefthydronephrosis, normal bladder and right kidney. The catheter was removed3daysafter the operation.He started to complain of frequency, urgency, dysuria in the4thday after operation, and gradually aggravate. These symptoms had no improvementafter the ureteral stent tube been removed in the10th day. He must go for micturitionevery5minutes, and also complained of gross hematuria, necrotic tissue expelledwith urine and suprapubic pain. On August13,2010(12days after the occurrence ofsymptoms), ultrasonography revealed thickening bladder wall with hyperechogenicmaterials on it. At cystoscopy under general anesthesia on September1,2010, thebladder showed a marked inflammatory appearance of all parts of the bladder mucosawith ulcerations and bladder lumen filled with minimal materials. Histopathologicalstudies of the necrotic tissue showed that the tissue was inflammatory granulationtissue and ulceronecrotic, with many crystals incrusted on it. Another cystoscopy onSeptember16,2010revealled a bladder with reduction in volume, a markedinflammatory appearance of all parts of the bladder mucosa with ulcerations andwhitish plaques corresponding to multiple encrusted calcifications, but there were nomiliary nodules on the mucosa. Another histopathological studies showed that thebladder mucosa was edematous and necrotic, with many crystals incrusted, fibroustissue proliferation, and neutrophils, eosinophils and lymphocyte infiltrating. From thetime of occurrence of symptoms to his visiting for our hospital, he was misdiagnosedurinary tract tuberculosis and had received anti-tuberculosis therapy, and had receiveda long time therapy of different kinds of antibiotics. After admission, ultrasonographyrevealed thickening bladder wall with hyperechogenic materials on it. IVU showedextensive thickening and calcifications outlining the bladder wall and mild lefthydronephrosis. Cystoscopy showed a marked inflammatory appearance of all parts ofthe bladder mucosa with ulcerations and whitish plaques corresponding to multipleencrusted calcifications. Infrared spectrophotometry analysis of the encrustedmaterials from the bladder wall confirmed the presence of struvite. Histopathologicalstudies on the biopsied bladder tissue showed that the bladder mucosa was edematousand necrotic, with many crystals encrusted and a polymorphonuclear infiltrate forming a thick conglomerate. After comprehensive analysis of medical history,symptoms, physical and auxiliary examination, he was diagnosed EC. Via thecombined treatment of trans urethral resection of the crystals, bladder irrigation ofsodium hyaluronate and detrusor injection of A-botulinum toxin, he was cured.Conclusions: Except for the conventional therapeutic methods of radicaltreatment of infection, acidification of the urine and chemolysis, elimination ofcalcified plaques, we can also use the method of bladder irrigation of sodiumhyaluronate and/or detrusor injection of A-botulinum toxin to treat EC. |
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