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.

Malakoplakia Involving the Maxilla: A Case Report and a Review of the Literature.

Malakoplakia is a rare inflammatory disorder which typically occurs in immunocompromised patients secondary to impaired bactericidal activity of macrophages. While this entity commonly arises in the genitourinary and gastrointestinal tracts, lesions of the head and neck have been reported only rarely, with oral cavity involvement reported in 3 cases. The most common presentation of head and neck malakoplakia is that of a cutaneous flesh-colored papule or nodule. This case report, however, illustrates the first time malakoplakia is identified affecting the maxilla and maxillary alveolar ridge mucosa. Histochemical and immunohistochemical stains are presented and include positivity for PAS, von Kossa stain, iron stain, and CD68 and negativity for GMS and Gram stains, indicating an inability to demonstrate microbial infection. Thus, clinicians and pathologists alike should be aware of malakoplakia as a pathologic entity when forming differential diagnoses, particularly in immunosuppressed individuals.

Malakoplakia of the Endometrium: A Rare Unexpected Disease Raising Clinical Concern for Malignancy.

Malakoplakia is a rare disease that manifests as a histiocytic inflammatory process and most often occurs in the urinary bladder. It is caused by an impaired capacity of histiocytes to kill and digest bacteria. The typical histopathologic findings are sheets of histiocytes with granular eosinophilic cytoplasm and characteristic Michaelis-Gutmann bodies, spherical bodies with a targetoid appearance. Malakoplakia is even rarer in the gynecologic tract, and our literature search found only 21 published patients of malakoplakia involving the endometrium. Here we report a 60-year-old female patient who presented with recurrent pelvic infections and postmenopausal bleeding, which raised concern for an endometrial malignancy. Hysterectomy with salpingo-oophorectomy revealed malakoplakia involving the endometrium and also the right ovary. Michaelis-Gutmann bodies were visible on the intraoperative frozen section that was performed to rule out an endometrial malignancy. We summarize the clinicopathologic findings of the published patients of endometrial malakoplakia.

A case of extremely rare pathology: Renal malakoplakia.

Malakoplakia is a granulomatous tissue inflammation with a characteristic histological appearance, mainly affecting the urogenital system and morphologically reflecting a macrophage disease. If bladder involvement is the most common, renal involvement is very rare and may be responsible for a differential diagnosis problem with renal cell carcinoma. We present a clinical case of renal malacoplakia mimicking malignant renal cell carcinoma diagnosed after partial nephrectomy in a 58-year-old woman with no history of recurrent urinary infections.

Case report: Prostatic malakoplakia: a rare disease that has a profile mimicking prostate cancer.

Prostatic malakoplakia (PMP) is a rare inflammatory disease, and misdiagnosis on imaging is a major reason for unnecessary punctures; however, information on imaging is even rarer. Five patients with PMP between May 2022 and February 2023 were enrolled in this study to summarize the imaging manifestations. All patients underwent ultrasound (US)-guided prostate biopsy and were confirmed by pathology, and the presence of prostate cancer was also excluded by pathology. The five patients, with a median age of 71 years (range = 58-74 years), had a median total prostate-specific antigen (T-PSA) of 10.40 ng/mL (range = 1.74-63.42 ng/mL). In two patients, chest computed tomography showed pulmonary infections. All patients underwent magnetic resonance imaging (MRI). Of these patients, four had a Prostate Imaging-Reporting and Data System (PIRADS) score of 5, while one had a score of 4. The lesions were mostly distributed in the peripheral zone of the prostate and appeared as a high signal on T1-weighted imaging (T1WI) and a low signal on T2-weighted imaging (T2WI). In the US examination, four patients had abnormal prostate morphology, with an unsmooth envelope and non-uniform parenchymal echogenicity. Four patients had increased prostate volume. US showed a hypoechoic nodule with non-uniform internal echogenicity, and an abundant internal blood flow signal was detected by color Doppler US. PSA, MRI, and US were not specific for PMP in our study, but we found that a history of co-infection may be helpful in an accurate diagnosis and to avoid unnecessary biopsy.

Colonic malakoplakia in a pediatric renal transplant recipient case report.

Malakoplakia is a rare, chronic granulomatous disease that mainly affects the genitourinary system of immunocompromised adults. It is caused by a bactericidal deficit in macrophages and, therefore, the treatment includes antimicrobials that reach high concentrations in macrophages. To our knowledge, we present the first case of malakoplakia in a pediatric solid organ transplant recipient. Our patient is a 15-year-old male renal transplant recipient who presented with recurrent diarrhea. Blood, urine, and gastrointestinal pathogen panel testing were positive for enteroaggregative Escherichia coli. A colonoscopy revealed diffuse malakoplakia. He had a complete resolution of symptoms with trimethoprim-sulfamethoxazole therapy. Unfortunately, his malakoplakia recurred after 9 months prompting the transition of therapy to oral gentamicin with subsequent remission. Malakoplakia should be considered in the differential of solid organ transplant recipients with recurrent gastrointestinal infections.

[Xanthogranulomatous adrenalitis : A rare and difficult differential diagnosis of adrenal gland tumors]. / Xanthogranulomatöse Adrenalitis : Seltene und schwierige Differentialdiagnose von Nebennierentumoren.

A radiologically diagnosed tumor in a 29-year-old woman with a fever of around 39 °C was operated on under the suspicion of cholecystitis or a liver abscess. A solid tumor was found in the adrenal gland and resected. The frozen section findings did not reveal a clear diagnosis of entity and assignment. Histologically, the tumor was found to consist of densely clustered large histiocyte-like cells with expression of vimentin, CD68, and CD163 as well as negativity for keratin, langerin, and SMA. We diagnosed xanthogranulomatous adrenalitis and discussed the differential diagnoses (Langerhans cell histiocytosis, Rosai-Dorfman disease, malakoplakia, Erdheim-Chester disease).

Severe Ulcerative Colitis Complicated by Malakoplakia Resulting in Proctocolectomy.

We report a 45-year-old man with medically refractory ulcerative colitis with superimposed colonic malakoplakia, presumed related to chronic use of azathioprine and biologics. This is the first reported case of malakoplakia in a patient requiring high doses of combination therapy. Treatment of malakoplakia is not standardized, but can involve systemic antibiotics, or surgical resection, which in this case resulted in proctocolectomy.

Malakoplakia of urinary bladde: 3 cases reports and review.

OBJECTIVE: This study aims to explore the clinical diagnosis and treatment methods of bladder malakoplakia (MUB) to enhance the understanding of the disease. METHODS: A retrospective analysis of the diagnosis and treatment processes of three cases of MUB treated in our department was conducted. Relevant literature from both domestic and international sources was reviewed to provide a comprehensive analysis. RESULTS: All three patients underwent transurethral resection of bladder lesions combined with antibiotic therapy, and two of them received transurethral instillation of gemcitabine. There were two cases with two recurrences each, and one case with four recurrences, with the latter also concurrently presenting with unilateral ureteral malakoplakia. Postoperative pathology confirmed MUB in all three cases. Close follow-up revealed no significant recurrence in the patients. CONCLUSION: The effective diagnosis rate is increased by conducting multiple deep, repetitive, and randomly selected live tissue examinations. The definitive diagnosis of MUB relies on pathological histological examination. Treatment involving a combination of antibiotics and transurethral resection of bladder lesions proves to be effective. Exploring the use of bladder instillation of gemcitabine widens the spectrum of MUB treatment methods.

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.

Malakoplakia mimicking malignant ovarian tumor: A case report and literature review.

INTRODUCTION AND IMPORTANCE: Malakoplakia is a rare inflammatory condition that generally occurs in immunocompromised individuals and is thought to be secondary to a bactericidal defect in macrophages. CASE PRESENTATION: In this report, we present the case of a 50-year-old multiparous patient who presented with chronic pelvic pain. Ultrasonography revealed a suspicious left lateral-uterine mass. Laparoscopic exploration showed an inflammatory mass in the left adnexa adherent to the uterus, peritoneum, and meso-sigmoid. The patient underwent a total hysterectomy with bilateral salpingo-oophorectomy and peritoneal biopsy. Histological findings were consistent with a left adnexal location of malacopakia, and Bacteriological analysis revealed Escherichia coli infection sensitive to ciprofloxacin. At the 6-month follow-up, no recurrence was observed. The patient's condition improved following surgery and antibiotic treatment. CLINICAL DISCUSSION: Genitourinary malakoplakia is more common in women and has no specific clinical, biological, or radiological features. Diagnosis is based on histological criteria, notably the presence of Michaelis-Gutmann bodies. Advances in our understanding of the pathophysiology of malakoplakia have made it possible to consider medical treatment options, mainly through the use of antibiotics. However, in cases where the organ is severely affected, surgical excision is recommended. CONCLUSIONS: To summarize, adnexal malacoplakia is a highly uncommon disease that may be mistaken as a malignant tumor. The diagnosis is established through histological examination. The usual treatment is a combination of surgical excision followed by targeted antibiotic therapy, as the diagnosis is often not made until after surgery.

Adrenal malakoplakia a rare lesion that mimics a neoplasm.

Background: Malakoplakia is a rare disorder 75% of the reported cases affect mainly the genitourinary tract, its occurrence in the adrenal gland is extremely rare. Case presentation: A 65-year-old female patient presented to the emergency department for chronic abdominal pain. Radiographic and biochemical studies revealed a left adrenal incidentaloma and left adrenalectomy was performed. Histological examination showed the presence of Michaelis-Gutmann bodies, compatible with a malakoplakia of the adrenal gland. Conclusions: Malakoplakia is a rare disorder, with non-standardized treatment, medical and surgical therapies appear to be effective in treating the condition.

Rectal malakoplakia mimicking advanced rectal cancer: A case report.

Background: Malakoplakia is a rare acquired chronic infectious granulomatous condition, that is characterized by the accumulation of large granular macrophages containing basophilic inclusion bodies in the cytoplasm termed Michaelis-Gutmann (MG) bodies. Malakoplakia most commonly involves the genitourinary system, and the second most commonly affected site is the gastrointestinal tract. Rectal malakoplakia is an unusual entity that is difficult to diagnose due to its diverse clinical manifestations and radiological findings that are similar to different diseases and advanced cancers. Case description: A 61-year-old male patient presented with difficulty in urination and defecation that started 4 months prior, along with a weight loss of 10 kg. Abdominal computerized tomography (CT) scanning revealed diffuse lesions of the perirectal region with multiple lymphadenopathies and involvement of the bladder, prostate, bilateral seminal vesicles, and left ureter. 18F-FDG PET/CT MIP showed intense FDG uptake in the rectal region, and a diagnosis of an occupying lesion was proposed. Colonoscopy and histological examination of rectal lesion biopsies showed the characteristic features of malakoplakia. Conclusion: Malakoplakia of the rectum with lymph node involvement and adjacent organ extension has been extensively misdiagnosed in clinical practice, and mimics malignancy radiologically. It is of great importance for radiologists to be aware of malakoplakia when making the differential diagnosis of benign and malignant mass lesions of the rectum, although the radiologic findings are nonspecific. Endoscopic evaluation and pathologic examination of a biopsy should be recommended to make the correct diagnosis, which may prevent unnecessary surgical resection.

Malakoplakia with aberrant ALK expression by immunohistochemistry: a case report.

BACKGROUND: Malakoplakia is a rare inflammatory disease of the urogenital tract. There have been no reports of malakoplakia expressing anaplastic lymphoma kinase (ALK) to date. Here, we present one case of malakoplakia with aberrant ALK expression by immunohistochemistry and discuss the clinical significance. CASE PRESENTATION: A 65-year-old Chinese woman with a history of diabetes presented with solid masses in the liver and kidney and elevated lesions on the mucosal surface of the colon. Right nephrectomy and partial liver resection were performed. Microscopically, sheets of histiocytes with poor intercellular adhesion were seen, with Michaelis-Gutmann bodies present in both the intracellular and extracellular interstitium. CD10-, CD68-, and CD163-positive cells were present, with Michaelis-Gutmann bodies confirmed by staining with Alcian blue, periodic acid-Schiff (PAS), periodic acid-Schiff with diastase, Von Kossa, and Prussian blue. Aberrant ALK1 and ALK (D5F3) expression was observed in the cytoplasm and nucleus of cells. However, ALK gene mutation was not detected by fluorescence in situ hybridization or whole exome next-generation sequencing. NGS revealed nine individual somatic gene mutations: GOT1L1, GLIS2, SPOUT1, TMEM97, MUC3A, NSD2, SFXN5, ADAD1 and RAD50. The significance of the somatic gene mutations detected in this study is not clear, and the relationship between them and malakoplakia cannot be clarified by existing scientific studies. The pathological diagnosis was malakoplakia with aberrant ALK expression by immunohistochemistry. The antibiotics imipenem and vancomycin were started based on the results of drug sensitivity analysis and the patient was subsequently discharged. She experienced no discomfort during 30 months of follow-up. CONCLUSION: This is the first reported case of malakoplakia with aberrant ALK expression, it should be differentiated from ALK-positive histiocytosis to avoid misdiagnosis.

A case of canine intestinal malakoplakia.

Malakoplakia is a rare chronic granulomatous disease usually affecting the urinary bladder and other locations. In humans, the gastrointestinal tract is the second most common location but there are no reports of intestinal malakoplakia in animals. A 10-month-old female French Bulldog was presented with chronic haemorrhagic diarrhoea and anorexia with normochromic-normocytic anaemia and hypoalbuminaemia. Grossly, there was mucosal thickening and ulceration of the caecum, colon and rectum. Microscopically, transmural sheets of foamy macrophages were seen in these tissues. Macrophages were periodic acid-Schiff, vimentin and ionized calcium-binding adaptor molecule 1 positive and contained von Kossa- and Prussian blue-positive Michaelis-Gutmann bodies. Giemsa staining revealed rod-shaped bacterial colonies and fluorescence in-situ hybridization demonstrated Escherichia coli within macrophages. This is the first reported case of intestinal malakoplakia in domestic animals. Pathological features of intestinal malakoplakia share many similarities with ulcerative histiocytic colitis in dogs but it is unclear if they are different forms of the same pathological process or distinct entities.

Intestinal Lesions Due to Rhodococcus equi in a Patient With Advanced Retroviral Disease and Pulmonary Infection: A Case of Colonic Malakoplakia.

In humans, Rhodococcus equi (R. equi) is a zoonotic infection usually involving immunocompromised subjects, only rarely affecting immunocompetent subjects. Herein, we describe an R. equi infection in a 50-year-old Russian man with acquired immune deficiency syndrome (AIDS) who presented with pulmonary cavitary lesions and clinical manifestation of colonic malakoplakia. A colonoscopy examination showed ulceration and mucosal erosion, and the histological findings confirmed the colonic malakoplakia. The patient recovered from pulmonary and gastrointestinal disease after four weeks of antibiotic treatment with intravenous ciprofloxacin and oral azithromycin and also underwent subsequent long-term oral antibiotic treatment to achieve clinical and immune restoration after antiretroviral therapy. Infectious disease pathology subspecialties should always consider R. equi chronic infection as a cause of malakoplakia in patients with AIDS. As only a few cases of colonic malakoplakia associated with R. equi are reported in the literature, these cases are important to describe, especially for clinical and treatment management.

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 arising on the ankle of a patient with pyoderma gangrenosum.

Malakoplakia is a rare chronic inflammatory condition that most commonly involves the urogenital tract. Cutaneous malakoplakia is extremely rare and many patients diagnosed with skin involvement are immunosuppressed. While the clinical presentation of cutaneous malakoplakia is variable, the histopathologic features are quite distinct and include sheets of closely packed dermal histiocytes with foamy-appearing cytoplasm and Michaelis-Gutmann bodies that are positive with certain immunohistochemical stains. While the exact pathogenesis of malakoplakia is unknown, it has been associated with certain bacterial infections. Treatment generally involves a combination of surgery and antimicrobial agents and/or modulation of immunosuppressant therapy if appropriate. Herein, the authors report a unique case of cutaneous malakoplakia arising in a patient on chronic immunosuppressive therapy for the management of pyoderma gangrenosum.

Malakoplakia of the Gallbladder: A Case Report.

Histiocytes are cells that are involved in the immune responses of the body. They are unable to properly break down the bacterial material in malakoplakia, a chronic granulomatous histiocytic disease that occurs in immunocompromised patients and autoimmune conditions. Very few reports of these lesions exist, as those that occur in the gallbladder. It typically affects the urinary bladder, alimentary tract, cutaneous, hepato-biliary, and male and female genital systems. These lesions are usually incidental findings that result in patients being misdiagnosed. A 70-year-old female presented with right lower quadrant abdominal pain, and malakoplakia of the gallbladder was diagnosed. Histopathology findings revealed malakoplakia of the gallbladder, and the same was confirmed with special stains such as periodic acid-Schiff (PAS). This case highlights the role of gross and histopathology findings as a clue to the diagnosis, which helps the surgeon with further management.