Radiology
Dr. Flaig: Dr. McKinney, would you please review this patient’s radiographic findings?
Dr. Kristin McKinney: This patient’s past medical history and surgical interventions would make his imaging difficult to interpret. Differentiating cystitis glandularis from bladder carcinoma would not have been feasible with imaging alone. On traditional excretory urography or computed tomography (CT), both entities may present as irregular bladder thickening with or without nodular masses.[6,7] In addition, both entities have a predilection for the bladder neck and trigone regions and both may present with hydronephrosis.
A variety of infective, inflammatory, and fibrotic conditions can result in bladder wall thickening on imaging.[8] To differentiate between these possibilities, a biopsy is necessary for definitive diagnosis. Pelvic lipomatosis is associated with adenocarcinoma of the bladder and cystitis glandularis, speculating that the chronic inflammatory changes in the bladder may be the result of lymphatic obstruction created by the pelvic fat proliferation. However, its absence in this case is not helpful.[9]
CT Scan—Contrast-enhanced computed tomography (CT) scan demonstrating prominent bilateral external iliac lymph nodesAn outside CT report from 1 year ago did not mention pelvic adenopathy, but mildly enlarged lymph nodes measuring from 1.5 to 2 cm were demonstrated in CT imaging in the last 3 months and confirmed with follow-up imaging (Figure 4). These nodes were in the para-aortic and retroperitoneal regions as well as in the bilateral external iliac and inguinal distributions. Typically, lymph nodes greater than 1 cm in short axis are considered suspicious on CT. However, benign entities such as hyperplasia, infection, or inflammation may also result in lymph node enlargement, confounding the diagnosis. If necessary, nodal involvement may be confirmed with needle biopsy.
Consideration of Findings
Dr. Flaig: Considering this patient’s radiographic findings, can you discuss the clinical entity of cystitis glandularis and the implications of this finding in this case?
Dr. Shandra Wilson: Patients who undergo urinary diversion are not only at risk for pyocystitis and recurrent urinary tract infections, they are also at risk for glandular metaplasia as we see here, and even adenocarcinoma of the bladder. Thus, as urologists, we generally recommend removing the bladder when diverting the urine away from it. Many patients with multiple sclerosis were treated with ileal conduits in the past. However, with the advance of urodynamics to monitor bladder pressure, we now know that these patients with high bladder pressure can do very well with bladder augmentation or even Botox, avoiding the potential complications seen in this patient.
Dr. Flaig: What is known about cystitis glandularis and its potential to progress to an invasive cancer?
Dr. La Rosa: The extent of cystitis glandularis in this case appears to correlate with the duration of urinary stasis. Diverting the urine, but leaving the bladder in place likely produced recurrent infection of the urinary bladder due to inadequate drainage. Lin et al postulate that the intensity of the infection may be responsible for the development of adenocarcinoma in such circumstances.[2] Cystitis glandularis, especially the diffuse type, can undergo malignant degeneration under constant irritation, but this is a rare and long-term process.
Differential Diagnosis
Dr. Flaig: What would be the differential diagnostic and treatment considerations with respect to the pelvic lymphadenopathy at this time?
Dr. Paul Maroni: The differential diagnosis would include regional metastasis from a pelvic malignancy, adenitis from a local inflammatory event, and, less commonly, other diseases causing enlargement of lymph nodes, such as lymphoma. A percutaneous biopsy would be indicated to determine the nature of the nodal enlargement, especially if this is a new finding. If a percutaneous biopsy would not be feasible, either a laparoscopic or open biopsy may be entertained. Given this patient’s other abdominal operations, a laparoscopic procedure may not be the best choice. Certainly, if these lymph nodes harbor metastatic adenocarcinoma, the prognosis would be quite grim, and chemotherapy of some sort—preferably on an experimental protocol—would be the best choice.
Dr. Flaig: Returning to this case, we have a patient status post cystectomy for cystitis glandularis with intestinal metaplasia. He now has lymphadenopathy of the pelvis, which is a new finding in the past year. At this point, we are planning to discuss the merits of a lymph node biopsy with the patient.
Summary
Dr. Flaig: Cystitis glandularis with this degree of intestinal metaplasia is a rare finding in the bladder. At the outside institution, the original biopsy was diagnosed as an invasive adenocarcinoma, and the patient proceeded to cystectomy. Upon our review of the cystectomy sample, the case appears most consistent with florid cystitis glandularis with intestinal metaplasia. The pathologic discrimination between invasive adenocarcinoma and cystitis glandularis with intestinal or glandular metaplasia may be difficult.[3,10] There is a long-standing debate over whether cystitis glandularis is a precancerous lesion,[11] but individual case reports describe this transition, and the risk of malignant transformation may be associated with the degree of replacement of normal urothelium by intestinal features.[12]
While cystitis glandularis has been associated with chronic inflammation as seen with recurrent infections, outflow obstruction, indwelling catheterization, pelvic lipomatosis, and urolithiasis,[13,14] the potential predisposing condition in our current case may be attributed to an approximately 30-year history of a blind, nonirrigated bladder.
Considering this history, the putative link between cystitis glandularis with intestinal metaplasia and adenocarcinoma, and the new appearance of pelvic lymphadenopathy, we will now discuss the merits of lymph node biopsy with the patient. This case, and the multidisciplinary input exchanged, highlights the benefit of a multidisciplinary review, especially in cases with uncommon features and diagnostic dilemmas.
Clinical Follow-up
A percutaneous biopsy was pursued after discussion with the patient, revealing a heterogeneous population of lymphoid cells. Given this information, we will continue to follow the patient radiographically, with consideration of surgical lymph node sampling at the time of any significant growth or systemic symptoms of disease.
Financial Disclosure: The participants in this conference have no significant financial interest or other relationship with the manufacturers of any products or providers of any service mentioned in this article.
