A rare case of prostatic malakoplakia with multidrug-resistant Escherichia coli: a case report.

Prostatic malakoplakia is an uncommon chronic inflammatory disorder, tumor-like but non-cancerous, the diagnosis of which pivots crucially on the identification of characteristic Michaelis-Gutmann bodies within the pathological tissue. We hereby present an inaugural case report of prostatic malakoplakia concurrent with sepsis caused by multidrug-resistant Escherichia coli, verified through blood culture and metagenomic next-generation sequencing (mNGS). The pathogenesis might be associated with infections by Escherichia coli, immune system irregularities, or lysosomal dysfunction. Although the patient had no chronic underlying diseases, he presented early with sepsis and multi-organ dysfunction. This case emphasizes the imperative to further investigate the association between malakoplakia and Escherichia coli, the necessity for prompt diagnosis, and the supportive role of mNGS, and the treatment strategy focuses on rapid control of infection and systemic inflammatory response.

Bifocal malakoplakia in a patient living with HIV: case report.

BACKGROUND: Malakoplakia is a rare chronic granulomatous disease characterized by the presence of Michaelis-Gutmann bodies (MGBs) within histiocytic aggregates. It predominantly affects immunocompromised individuals, including those living with Human Immunodeficiency Virus (HIV). CASE PRESENTATION: We present a unique case of bifocal malakoplakia in a 49-year-old man, previously with Coronavirus disease 2019 (COVID-19) and HIV positive, presented with respiratory symptoms, weight loss, and lymphadenopathy. He had various infections including Non-Tuberculous Mycobacteria (NTM), Cytomegalovirus (CMV), and Candida, with evolving lung and gastrointestinal issues. Despite treatment attempts, he deteriorated due to respiratory distress, multi-organ failure, and coagulopathy, leading to his unfortunate demise. CONCLUSION: This report presents a distinctive and complex case of malakoplakia in an HIV-positive patient, a rare inflammatory disorder originally described by Michaelis and Gutmann in 1902. The hallmark Michaelis-Gutmann organisms were observed, confirming the diagnosis. While typically affecting the urinary tract, this case demonstrates the exceptional ability of malakoplakia to manifest in various organ systems, including pulmonary, gastrointestinal, and more. Although Escherichia coli is a prevalent associated pathogen, the exact cause remains elusive. Treatment, often involving surgical excision and antibiotic therapy, underscores the challenging nature of managing this condition in immunocompromised individuals.

A very complicated UTI: malakoplakia following E. coli urinary tract infection.

Malakoplakia is a rare inflammatory disorder believed to result from a defect in macrophage phagocytic function triggering a granulomatous reaction. It can present with genitourinary, gastrointestinal, or cutaneous manifestations in immunocompromised or, less commonly, immunocompetent hosts. We describe a case of renal malakoplakia in a young, otherwise healthy patient presenting with nephromegaly and sepsis following an E. coli urinary tract infection. We discuss diagnosis and management, including antibiotic selection and the decision to pursue nephrectomy. This case highlights the potential for kidney recovery with prolonged antibiotic therapy in conjunction with adjunct immunomodulatory therapies and source control.

Clinical Evaluation and Surgical Management of Ocular Malakoplakia of the Caruncle and Periorbital Region: A Report of Two Case and Literature Review.

Ocular malakoplakia, a rare inflammatory disorder characterized by Michaelis-Gutmann bodies, is presented in 2 unique cases involving uncommon anatomical sites-the orbit and caruncle. The first case describes a 10-year-old girl with localized subconjunctival swelling near the caruncle, prompting surgical excision, and revealing characteristic malakoplakia features. Despite medical management, surgical intervention provided symptom relief. The second case involves a painless swelling below the lower lid in a 23-year-old female, initially suggestive of lymphoma. Excision biopsy confirmed malakoplakia, emphasizing the potential for clinical misdiagnosis. Histopathological examination showcased Michaelis-Gutmann bodies, von Hansemann cells, and chronic inflammation, confirming the ocular malakoplakia diagnosis. These cases underscore the rarity of ocular malakoplakia, particularly in pediatric patients, and highlight the importance of accurate diagnosis and appropriate management.

Successful treatment of malakoplakia of the liver and skin in a pediatric liver transplant patient.

BACKGROUND: Malakoplakia occurs uncommonly at any age, but pediatric reports are exceptionally limited. Malakoplakia appears primarily in the urinary tract, although involvement of essentially all organs has been reported, cutaneous malakoplakia is very uncommon and liver involvement is the rarest. METHOD: We report the first pediatric case of concurrent hepatic and cutaneous malakoplakia in a pediatric liver transplant recipient. We also provide a literature review for cutaneous malakoplakia cases in children. RESULT: A 16-year-old male received a deceased-donor liver transplant for autoimmune hepatitis, present with the persistence of the liver mass of unknown etiology and cutaneous plaque-like lesions around the surgical scar. Core biopsies taken from the skin and abdominal wall lesions demonstrated histiocytes containing Michaelis-Gutmann bodies (MGB) revealing the diagnosis. The patient successfully was treated with antibiotics alone for 9 months without surgical intervention or a decrease in immunosuppressive therapy. CONCLUSION: This case demonstrates the need to include malakoplakia in the differential diagnosis of mass-forming lesions after solid transplantation and increase awareness of this very rare entity in pediatrics.

A Surgical Challenge Generated by Colonic Malakoplakia in Disguise as a Locally Advanced Colonic Malignancy-A Case Report.

Colonic malakoplakia is an uncommon granulomatous development of cells resulting from the impaired capacity of the mononuclear cells to eliminate the phagocytosed bacteria, and in rare cases it can also affect the gastrointestinal tract. We report the case of a 78-year-old female patient that was admitted to hospital by The Emergency Department with the diagnosis of bowel obstruction, confirmed by the clinical and paraclinical investigations. We decided to surgically manage the case for suspicious symptomatic colonic neoplasm. The histological examination of the surgical specimens revealed colonic malakoplakia, characterized by the presence of the aggregated granular histiocytes and Michaelis-Gutmann bodies. Through this paper, we want to raise awareness for Malakoplakia, which remains an extremely rare disease that may affect multiple organs, and because it does not present specific symptoms or clinical manifestations, the final diagnosis remains the histopathological study. The clinical conduct should be decided after taking into consideration all the aspects of this pathology along with the benefits and risks for the patient.

A Rare Case of Prostatic Malakoplakia Associated with Prostate Adenocarcinoma: A Case Report and Review of Literature.

Malakoplakia is a chronic granulomatous condition that has been rarely seen affecting the prostate. Isolated malakoplakia of the prostate occurring together with prostatic adenocarcinoma is rarer still with only 9 previously recorded cases. We present a case diagnosed through needle biopsy with prostatic adenocarcinoma and then on subsequent prostatectomy was diagnosed with extensive malakoplakia occurring with the carcinoma. Patient was noted to have a urinary tract infection (UTI) 2 weeks after needle biopsy and notably, 4 of the 9 previously reported cases also presented with UTI following their biopsies. The theory that prostatic malakoplakia may be a complication of the prostate needle biopsy is logically possible, but due to the paucity in cases, it is difficult to infer causality.

[Colonic malakoplakia: Rare disease that can mimic a consuntive syndrome]. / Malacoplaquia del colon: Rara enfermedad que puede presentarse como un sindrome consuntivo.

Colonic malacoplakia is an unusual cause of chronic diarrhea, and it may present as a consumptive disease. At the colon, it can induce ulcerative and erosive nodular lesions, that mimic other common granulomatous or infectious diseases. Diagnosis is support in biopsies showing groups of histiocytes, with typical Michaelis-Gutmann inclusions, which are positive with the Von Kossa stain. We present the case of a 55-year-old male, without associated diseases, who presented with diarrhea, weight loss and anemia, showing a very good clinical response to antibiotics.

[A Case of Malacoplakia of the Prostate which was Difficult to Distinguish from Prostate Cancer].

A 70-year-old-man was referred with urination pain and pyuria. Prostate specific antigen was 10.6 ng/ml, and urine culture was Escherichia coli. The abdominal ultrasonography showed irregular low echo at the right lobe of prostate. Prostate magnetic resonance imaging (MRI) showed an extracapsular infiltrated prostate tumor in the right lobe. Levofloxacin was administered and prostate biopsy was performed. The pathological examination revealed that the prostate tissue was filled with inflammatory cells and had lost its glandular duct structure. The patient was diagnosed with malacoplakia of the prostate. Four months after prostate biopsy, prostate MRI imaging showed disappearance of the extracapsular infiltration in right peripheral zone.

Prostatic malakoplakia: clinicopathological assessment of a multi-institutional series of 49 patients.

Prostatic malakoplakia (MP) is rare, with only case reports and small series (< five patients) available in the literature. In this study we analysed an international multi-institutional series of 49 patients with prostatic MP to more clearly define its clinicopathological features. The median age was 67 years and the median serum prostate-specific antigen (PSA) was 7.5 ng/ml. MP was clinically manifest in most cases (28 of 45 patients with data available, 62%). Of 43 patients with detailed clinical history available, 21 (49%) had concurrent or metachronous malignancies (including prostate cancer). Diabetes or insulin resistance was present in 11 patients (26%). Additionally, three patients had a history of solid organ transplantation and one had HIV. Of note, six of 34 patients (18%) without concurrent prostate cancer had an abnormal digital rectal examination and/or lesions on magnetic resonance imaging (MRI) with prostate imaging reporting and data system (PIRADS) scores 4-5. The initial diagnosis was made on core biopsies (25 of 49, 51%), transurethal resection specimens (12 of 49, 24%), radical prostatectomies (10 of 49, 20%), Holmium-laser enucleation (one of 49, 2%) and cystoprostatectomy (one of 49, 2%). Tissue involvement was more commonly diffuse or multifocal (40 of 49, 82%). Von Kossa and periodic acid-Schiff stains were positive in 35 of 38 (92%) and 26 of 27 lesions (96%), respectively. Of note, two cases were received in consultation by the authors with a preliminary diagnosis of mesenchymal tumour/tumour of the specialised prostatic stroma. The present study suggests that prostatic MP is often associated with clinical findings that may mimic those of prostate cancer in a subset of patients. Moreover, MP may be found incidentally in patients with concurrent prostate cancer.

Cutaneous malakoplakia with verruciform xanthoma in the same lesion.

Cutaneous malakoplakia (CM) is a rare, chronic, granulomatous disease characterized histopathologically by Michaelis-Gutmann bodies (MGB). Verruciform xanthoma (VX) is a rare, benign lesion characterized histopathologically by epithelial papillomatous hyperplasia, local hyperkeratosis with incomplete keratosis, infiltration of foam cells and inflammatory cells in the papillary dermis. We present an elderly Chinese man with CM and coexisting VX with histological confirmation of MGB.

The necessity of transperineal ultrasound view in urethra malacoplakia.

This case illustrates the untypical presentation of primary bladder malacoplakia. The patient was in her mid-50s have impaired immunity by the long-term hyperglycemic condition. She presented with symptoms of urinary tract infection and dysuria, and had multiple nodulars in bladder and significantly mass in urethra. Although the diagnosis of bladder malacoplakia was established on bladder biopsy, transperineal ultrasound examination can find its distinct clinical presentation.

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.

Bilateral Ovarian Malakoplakia: Case Report and Review of the Literature With Clinical and Diagnostic Considerations.

Malakoplakia is a rare condition in which histiocytic cells accumulate within different organs and tissues, sometimes mimicking neoplasia. Gynecologic involvement is extremely rare and therefore may cause relevant diagnostic confusion for both clinicians and pathologists. In this paper, we described the seventh case of ovarian malakoplakia, and we reviewed the literature to compare it with the previously reported ones. Moreover, we investigated the histologic and molecular differential diagnosis of malakoplakia, with special attention to other histiocytic disorders of gynecologic interest. Finally, we discussed the most relevant points with regard to possible pathogenesis and management. Malakoplakia often represents a forgotten entity that should be remembered preoperatively, when approaching a possible gynecologic neoplasia. Moreover, it is of remarkable importance to differentiate malakoplakia from multisystem histiocytosis involving gynecologic organs. All this would prevent misdiagnosis and overtreatment of such a rare but benign condition.

Colonic malakoplakia in a dual stem cell and cardiac transplant recipient: A case report and literature review.

We describe the first published case of malakoplakia in a dual stem cell and cardiac transplant recipient. In the 2 months following cardiac transplantation, our patient developed persistent diarrhea and recurrent E coli bacteremia. Biopsies obtained from areas of colonic thickening revealed malakoplakia. Despite improvement in symptoms with prolonged antimicrobial therapy and reduction of his immunosuppression, he eventually died from sepsis. Our case highlights not only the importance of the timely diagnosis of this rare disorder, but also the difficulty in determining optimal treatment duration, particularly where excision of involved areas is not possible, as data on this disease are lacking. Here we describe our case and review the available literature published on malakoplakia in the cardiac transplant population.

Malakoplakia in the Urinary Bladder of 4 Puppies.

Malakoplakia in humans most often affects the urinary bladder and is characterized by inflammation with von Hansemann-type macrophages, with or without Michaelis-Gutmann bodies, and is frequently associated with Escherichia coli infection. We describe the microscopic features of malakoplakia in the urinary bladder of 4 puppies. In all cases, the lamina propria of the urinary bladder was markedly expanded by sheets of large, round to polygonal macrophages with intracytoplasmic, periodic acid-Schiff-positive granules and granular inclusions, and rare Prussian blue-positive inclusions. Macrophages were positive for CD18 and Iba1. In 2 cases, Michaelis-Gutmann bodies were detected with hematoxylin and eosin stain and were best demonstrated with von Kossa stain. E. coli infection was confirmed in 2 cases with bacterial culture or polymerase chain reaction (PCR) and sequencing of the bacterial 16S ribosomal RNA gene. Transmission electron microscopy of one case demonstrated macrophages with abundant lysosomes, phagolysosomes, and rod-shaped bacteria. Microscopic features were similar to human cases of malakoplakia. In dogs, the light microscopic characteristics of malakoplakia closely resemble granular cell tumors and histiocytic ulcerative colitis.

Pulmonary malakoplakia secondary to Rhodococcus equi infection mimicking a lung neoplasm in a lung transplant recipient.

Pulmonary masses occasionally occur after lung transplantation and vary in etiology, which includes malignant and benign conditions, such as infection. Here, we report a case of a patient presenting with a lung mass 3 years after lung transplant. To our knowledge, this is the first described case of pulmonary malakoplakia due to Rhodococcus equi infection in an allograft post-lung transplantation. This case outlines the challenges of differentiating benign from malignant masses after transplantation.

Malakoplakia of the Bladder in a 4-Month-Old Puppy.

A 4 mo old female Staffordshire bull terrier puppy was presented with chronic Escherichia coli cystitis. Ultrasound and cystoscopic examination revealed innumerable, intraluminal, finger-like proliferations arising from the dorsal urinary bladder (UB) wall. Histological examination of mucosal biopsies obtained by cystoscopy was suggestive of granulomatous cystitis. The proliferative lesions were removed surgically and submitted for histological examination. The UB submucosa was heavily infiltrated by macrophages with periodic acid-Schiff-positive cytoplasm exhibiting rare Michaelis-Gutmann bodies, leading to the diagnosis of malakoplakia. The puppy was prescribed with sulfamethoxazole-trimethoprim. The urinary signs disappeared despite the persistent UB wall thickening revealed by abdominal ultrasound. Urine culture performed during the ninth week of treatment showed a persistent infection by E coli resistant to sulfamethoxazole-trimethoprim. The dog was switched to doxycycline but was then lost to follow-up. Malakoplakia is a chronic granulomatous inflammation well documented in humans. Its pathophysiology is not fully understood, but bacterial infection, immunodepression, and a defective lysosomal function may lead to the intracytoplasmic accumulation of partially degraded bacteria that can subsequently mineralize to form the Michaelis-Gutmann bodies. Malakoplakia should be suspected when UB mass lesions are identified in a young dog with bacterial cystitis.

Malacoplakia of the skin: overview of a rare clinical entity.

BACKGROUND: Malacoplakia is a rare acquired, infection-related granulomatous disorder, that may affect many systems, but typically occurs in the urinary tract. Cutaneous involvement is less prevalent, and most commonly presents with a perianal or genital region localization. Cutaneous malacoplakia is believed to be caused by an acquired bactericidal defect of macrophages in the setting of chronic infections and immunocompromised states. A diagnosis of cutaneous malacoplakia should be considered when encountering non-specific granulomatous lesions that are refractory to treatment. Histologic findings are marked by the presence of foamy macrophages containing the pathognomonic Michaelis-Gutman bodies. OBJECTIVES: The aim of this review is to discuss the current perspectives on the pathophysiology, clinical features, diagnosis, and treatment of this disease. We would also like to emphasize that the integration of clinical information, microscopic findings, and exclusion of other cutaneous granulomatous processes is necessary to accurately diagnose this exceedingly rare disease and provide opportunity for therapeutic intervention. PATIENTS/METHODS: Data for this work were collected from the published literature and textbooks. RESULTS: Combined surgical excision and protracted antibiotic courses appear to have the highest success rate. Antibiotics should be culture specific, but drugs that easily permeate the macrophages appear to be the best choice.