A Case of Urinary Bladder Malakoplakia in a Young French Bulldog: Diagnostic and Therapeutic Issues.

A 3-month-old female French Bulldog presented with hematuria, severe pollakiuria, and urinary incontinence lasting for 1.5 months. Broad-spectrum empirical antibiotic therapy and nonsteroidal anti-inflammatory drugs were initiated by the referring veterinarian. Due to a lack of improvement, the dog was referred. At referral examination, urinary clinical signs persisted (hematuria, severe pollakiuria) and a firm bladder was noted. Abdominal ultrasonography revealed severe, diffuse bladder wall thickening with a significant reduction in the bladder lumen. Urinary tract endoscopy showed whitish exophytic proliferations throughout the entire bladder wall. Histological bladder wall analysis led to a diagnosis of bladder malakoplakia. Prolonged antibiotic therapy with fluoroquinolones was prescribed and resulted in clinical remission despite persistent bacteria in the bladder wall. This report describes a case of successfully medically managed bladder malakoplakia, a very rare condition in veterinary medicine, well documented in humans.

Cutaneous malakoplakia.

Malakoplakia is a form of chronic, granulomatous, inflammatory condition which usually affects the genitourinary tract or other internal organs of immunocompromised patients. It is usually caused by acquired bactericidal incapacity of macrophages in connection to Eschericia coli infection. This case report presents an extremely rare case of cutaneous malakoplakia in the left axilla of a 48-year-old male patient, who had undergone kidney transplant one year earlier. The clinical presentation of cutaneous malakoplakia varies from nodules to plaques and moist wounds. The primary treatment is long-term antibiotic therapy.

Malakoplakia of the urinary bladder in a young French Bulldog.

CASE DESCRIPTION: A 4-month-old 5.9-kg sexually intact female French Bulldog was presented because of recurrent urinary tract infections in combination with pollakiuria, hematuria, and urinary incontinence. CLINICAL FINDINGS: A diagnosis of malakoplakia was made on the basis of results of hematologic and serum biochemical testing, abdominal ultrasonography, bacterial culture, and cystoscopic biopsies of the urinary bladder wall. Biopsy samples were sent for routine histologic examination and fluorescence in situ hybridization to confirm the presence of intracellular and subendothelial bacteria. TREATMENT AND OUTCOME: Treatment with enrofloxacin was started after the diagnosis of malakoplakia was confirmed. During treatment, polypoid changes in the urinary bladder decreased dramatically but did not disappear. On follow-up ultrasonography after 12 weeks of treatment, marked improvement was visible and results of repeated bacterial culture and fluorescence in situ hybridization of bladder wall samples were negative. The patient was free from clinical signs and had an ultrasonographically normal urinary bladder 59 weeks after antimicrobial treatment was discontinued. CLINICAL RELEVANCE: Malakoplakia, a granulomatous disease characterized by impaired histiocytes that are unable to completely digest phagocytized bacteria, is a very rare disease in dogs, but early suspicion of the condition is essential to allow timely diagnosis and avoid disease progression and the need for prolonged treatment. Malakoplakia should be considered in young dogs with chronic urinary tract infections; the diagnosis can be made through a combination of histologic examination and fluorescence in situ hybridization of bladder wall biopsy samples.

Successful use of azithromycin for Escherichia coli-associated renal allograft malakoplakia: a report of two cases.

Malakoplakia is a chronic granulomatous disease associated with incomplete clearance of bacterial pathogens. A multimodal approach to therapy includes antimicrobials with intracellular activity, reduction in immunosuppression, and debulking of lesions. Azithromycin has an intracellular mechanism of action and enhanced Gram-negative activity compared to other macrolides. Despite some in vitro data to support its use, there are no clinical breakpoints or epidemiological cut-off values for most Enterobacterales from the European Committee on Antimicrobial Susceptibility Testing (EUCAST) or the Clinical and Laboratory Standards Institute (CLSI). We present two cases, previously unreported, of Escherichia coli associated renal allograft malakoplakia successfully treated with azithromycin.

Malakoplakia of the gastrointestinal tract: clinicopathologic analysis of 23 cases.

BACKGROUND: Malakoplakia is an uncommon, tumor-like inflammatory disease characterized by impaired histiocytes that are unable to completely digest phagocytized bacteria. The genitourinary tract is the most common site of involvement, however, cases have also been described in the gastrointestinal tract, suggesting that it is the second most common site of involvement. This study investigates the clinical and histologic features of malakoplakia in the gastrointestinal tract. CASE PRESENTATION: For 23 gastrointestinal specimens (biopsies and resections) from patients with a pathologic diagnosis of malakoplakia, we recorded the gender, age, location, primary diagnosis, endoscopic or surgical indication, endoscopic/gross impression and immune status (immunocompromised vs. immunocompetent). CONCLUSION: Malakoplakia occurred throughout the length of the gastrointestinal tract with most of the cases located in the sigmoid colon and rectum (n = 10); other sites included the transverse and descending colon (n = 4), stomach/gastroesophageal junction (n = 4), appendix (n = 2), cecum (n = 1), small bowel (n = 1), and the peri-anal area (n = 1). Endoscopically, these lesions most commonly appeared as polyps (n = 10) or masses (n = 5), other clinical endoscopic impressions varied from a thickened area/fibrosis to mucosal erythema. Most patients were immunocompromised due to a disease state (e.g. organ transplantation, cancer diagnosis, autoimmune condition) and/or medication effect. Eight patients with malakoplakia were on immunosuppressive medications (8/23, 35%). Common immunosuppressed disease states included cancer (n = 9), autoimmune disease (n = 5), status post organ transplantation (n = 4), diabetes (n = 5), infection/sepsis (n = 3), and HIV/AIDS (n = 1). Some patients had multiple co-morbidities (i.e. diabetes and organ transplant). Twenty-one patients with malakoplakia were in an immunosuppressive state (21/23, 91%).

Renal malakoplakia mimicking a malignancy and diagnosed by fine-needle aspiration: A case report.

Malakoplakia is a rare, granulomatous disease that affects a wide variety of organs and can have a clinical and radiographic presentation resembling that of malignancy. The genitourinary tract is the most commonly involved site. There are scant reported cases presenting as a locally advanced renal mass and even rarer, diagnosed by fine-needle aspiration (FNA) cytology. We report clinical, imaging, cytologic, and histological findings of an interesting case of malakoplakia initially diagnosed by FNA cytology. We also briefly review the literature and emphasize the importance of recognizing this entity when encountered in an aspirate material, which can help mitigate the diagnostic confusion of malakoplakia for clinicians.

Malakoplakia of the urinary system.

Malakoplakia is regarded as a chronic granulomatous inflammatory disease with a good prognosis. It usually affects the urinary system, especially the urinary bladder. Bacterial infections, including E. coli are thought to be the main factor in pathogenesis. It frequently coexists with chronic diseases and immunosuppression state. Histopathological examination of affected tissue samples is thought to be the best diagnostic method. The basic microscopic feature is mixed inflammatory infiltration containing foamy histiocytes known as von Hansemann cells, frequently with basophilic inclusions known as Michaelis-Gutmann bodies. Symptoms and the clinical course of malakoplakia depend on location and the extent of the lesions. The lesion is treated successfully with antibiotic therapy and surgical excision.

Malakoplakia of the Urinary Bladder and Unilateral Ureter.

Malakoplakia is an unusual acquired granulomatous disease that can affect many systems including urogenital tract. It presents a huge diagnostic challenge as it can mimic malignancy. We report a 55-year diabetic woman who presented with history of macroscopic hematuria and right flank pain. On investigations, ultrasound of kidney, ureter and bladder (KUB) showed right hydronephroureter, and CT KUB showed right moderate hydronephroureter and right ureteric stone. Endoscopic examination revealed multiple white plaques involving urinary bladder and right ureter. The diagnosis of malakoplakia was based on microscopic findings that are specific for its diagnosis.

Malakoplakia of the colon following renal transplantation in a 73 year old woman: report of a case presenting as intestinal perforation.

BACKGROUND: Malakoplakia is a chronic inflammatory disease characterized by tissue infiltrates of large granular macrophages containing distinctive intracytoplasmic inclusions termed Michaelis-Gutmann (MG) bodies. The genitourinary system is the most commonly involved site, followed by the gastrointestinal tract. Malakoplakia may occur as a complication of primary or secondary immunosuppression and, therefore, renal transplant recipients are at risk. The graft itself or extra-renal sites may be involved. Regarding the latter, six cases of colorectal malakoplakia have been reported following renal transplantation, with all but one patient experiencing significant morbidity. We describe a further example of colorectal malakoplakia following renal transplantation. The other previously reported cases are reviewed. CASE PRESENTATION: A 72 year old female presented with left lower quadrant abdominal pain and vaginal bleeding. She had received a cadaveric renal transplant for chronic renal failure ten months previously. Abdomino-pelvic computerized tomography (CT) scanning demonstrated two lesions in the mesocolon: the first adjacent to the descending colon and the second involving the sigmoid colon. A diagnosis of sub-acute perforated diverticulitis with two phlegmons was proposed. The sigmoid lesion was resected. The descending colon lesion was treated by creation of a cutaneous fistula. Microscopy of the sigmoid lesion showed the typical features of malakoplakia. She was discharged on sulfamethoxazole-trimethoprim. Nine months later, no longer receiving antibiotic therapy, the patient reported lower left quadrant discomfort. CT scanning showed para-rectal and pelvic abdominal masses with cutaneous and intestinal fistulas. Treatment with tazobactam-piperacillin was begun and sulfamethoxazole-trimethoprim was reinstated, with subsequent slow clinical improvement. Subsequent abdominal CT scans have shown persistence of the lesions. CONCLUSIONS: Physicians caring for renal transplant recipients should be aware of colorectal malakoplakia as a rare but serious complication. The onset may be within months or as long as a decade or more following transplantation. The clinical presentation is varied, nonspecific, and will likely suggest more common diseases. Although radiologic imaging is also nonspecific, awareness of malakoplakia is of importance to radiologists when formulating the differential diagnosis of mass lesions of the colorectum in this clinical setting. Definitive diagnosis remains dependent on pathologic examination of a biopsy or surgical resection specimen.

Malakoplakia of the large intestine: an incidental extremely rare finding.

Malakoplakia is a rare inflammatory disease, most commonly found in the urinary tract. It appears be related to a functional deficiency of macrophages, resulting in an inability to destroy digested bacteria and it is associated with various conditions that cause immunodeficiency. A rare case of malakoplakia of the colon in a healthy 68-year old male is presented. The patient underwent emergency surgery with colon resection and an end stoma with closure of the distal bowel (Hartmann's procedure), due to incarcerated ventral hernia and sigmoid-colon rupture. He underwent reversal of the Hartmann's procedure four months after the initial operation. The histological examination from the anastomotic rings revealed Michaelis-Gutmann bodies that are pathognomonic of malakoplakia. He received per os ciprofloxacin, bethanecol and ascorbic acid for 12 months. Follow-up endoscopy did not exhibit any signs of the disease. A case of a healthy patient presenting with malakoplakia without any underlying disease that causes immunodeficiency is extremely rare. Treatment of malakoplakia involves the eradication of microorganisms. Cholinergic agonists, such as bethanechol and ascorbic acid, as well as antimicrobial treatment with trimpethoprim/sulphamethoxazol and rifampicin are most commonly being used. Long-term antimicrobial treatment has been reported (6 months to 3 years).

Rectosigmoid malakoplakia.

We present a rare case of rectosigmoid malakoplakia treated with fluoroquinolone.

Cutaneous malakoplakia masquerading as pyoderma gangrenosum.

Cutaneous malakoplakia is a rare infection-related granulomatous disease frequently associated with immunocompromised states. Foamy macrophages containing basophilic granules, called the Michaelis-Gutman bodies, are pathognomonic. We report a case of cutaneous malakoplakia in a 77-year-old male with pyoderma gangrenosum and a 2-year history of a non-healing malleolar ulcer treated successfully with cotrimoxazole.

[Malakoplakia of the submandibular gland in a renal transplant patient]. / Malakoplakie de la glande sous-mandibulaire chez un patient transplanté rénal.

INTRODUCTION: Malakoplakia (MP) is a rare granulomatous disease, usually occurring in immunocompromised patients, linked to Escherichia coli infection. The lesions are usually located in the genitourinary tract, but there is a great variability in the topography and the clinical presentation. CASE REPORT: A 70-year-old diabetic kidney transplant patient under immunosuppressive treatment presented with a voluminous submandibular chronic lesion, involving the skin, associated with a burgeoning lesion of the oral mucosa. Histological examination of biopsies concluded to MP and bacteriological samples were positive for E. coli. Antibiotic treatment allowed for the regression of the lesion before surgical removal. Histological examination of resected material confirmed the diagnosis of invasive MP of the submandibular gland. DISCUSSION: The diagnosis of MP relies on histological examination, showing the presence of von Hansemann's cells and Michaelis- Gutmann bodies. The treatment is based on active antibiotics targeted against intracellular bacteria, possibly associated with surgery. We report the first case of MP involving the submandibular gland.

An adult case of cerebral malakoplakia successfully cured by treatment with antibiotics, bethanechol and ascorbic acid.

Cerebral malakoplakia is a very rare chronic inflammatory disease. We herein report the case of a 49-year-old female who presented with a slowly progressive speech disturbance and right hemiparesis. Computed tomography and magnetic resonance imaging showed irregular enhanced mass lesions with numerous scattered areas of calcification in the left insula, thalamus and basal ganglia. Histopathologically, the biopsy specimen showed basophilic laminated inclusion bodies and intracellular and extracellular calculospherules, usually with a typical targetoid appearance (Michaelis-Gutmann bodies). Treatment with antibiotics, bethanechol and ascorbic acid improved her symptoms in association with a decrease in the abnormal calcification and enhancement. The cerebral malakoplakia mimicked a brain tumor in terms of the patient's clinical course and neuroradiological image findings; however, it was successfully cured with medical treatment. This case provides evidence that the pathogenesis of cerebral malakoplakia is deeply tied to bacterial infection and that medical treatment is effective in cases of this disease.

Successful treatment of renal allograft and bladder malakoplakia with minimization of immunosuppression and prolonged antibiotic therapy.

Malakoplakia is an unusual granulomatous inflammatory disorder associated with diminished bactericidal action of leucocytes that occurs in immunosuppressed hosts. Cases of renal allograft malakoplakia are generally associated with a poor graft and patient survival. We present the case of a 56-year-old female with allograft and bladder malakoplakia occurring two years after renal transplantation complicated by an early antibody mediated rejection. Following a number of symptomatic urinary tract infections caused by resistant Gram-negative bacilli, a diagnosis of malakoplakia was made by biopsy of a new mass lesion of the renal allograft. Cystoscopy also revealed malakoplakia of the bladder wall. Immunosuppressant regimen was modified. Mycophenolate mofetil was ceased, prednisolone reduced to 5 mg/day and tacrolimus concentrations were carefully monitored to maintain trough serum concentrations of 2-4 µg/L. Concurrently, she received a prolonged course of intravenous antibiotics followed by 13 months of dual oral antibiotic therapy with fosfomycin and faropenem. This joint approach resulted in almost complete resolution of allograft malakoplakia lesions and sustained regression of bladder lesions on cystoscopy with histological resolution in bladder lesions. Her renal function has remained stable throughout the illness. If treated with sustained antimicrobial therapy and reduction of immunosuppression, cases of allograft malakoplakia may not necessarily be associated with poor graft survival.

Cured malakoplakia of the renal allograft followed by long-term good function: a case report.

BACKGROUND: Malakoplakia is an unusual chronic inflammatory disease with distinctive histopathological features rarely involving the parenchyma of a transplanted kidney, and to date less than ten cases have been reported. METHODS AND RESULTS: We present a case of malakoplakia of a kidney graft in a 31 year old woman after simultaneous kidney and pancreas transplantation, which was successfully treated with quinolones. After the treatment of malakoplakia, she was monitored regularly, and her renal and pancreas grafts functioned well for the following 9 years, which is 12 years post transplantation. Moreover, 1 year after treatment of malakoplakia she became pregnant and gave birth to a healthy child. CONCLUSION: Evaluation of a kidney biopsy sample represents the key to diagnosis of malakoplakia which is important for correct patient management. Treatment with antibiotics with intracellular penetration (quinolone type) may result in curing the disease. According to our knowledge, this is the first case of allograft renal malakoplakia after combined kidney and pancreas transplantation.