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 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.

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.

Presacral malakoplakia presenting as foot drop: a case report.

BACKGROUND: Malakoplakia is a rare condition characterized by inflammatory masses with specific histological characteristics. These soft tissue masses can mimic tumors and tend to develop in association with chronic or recurrent infections, typically of the urinary tract. A specific defect in innate immunity has been described. In the absence of randomized controlled trials, management is based on an understanding of the biology and on case reports. CASE PRESENTATION: Here we describe a case of presacral malakoplakia in a British Indian woman in her late 30s, presenting with complex unilateral foot drop. Four years earlier, she had suffered a protracted episode of intrapelvic sepsis following a caesarean delivery. Resection of her presacral soft tissue mass was not possible. She received empiric antibiotics, a cholinergic agonist, and ascorbic acid. She responded well to medical management both when first treated and following a recurrence of symptoms after completing an initial 8 months of therapy. Whole exome sequencing of the patient and her parents was undertaken but no clear causal variant was identified. CONCLUSIONS: Malakoplakia is uncommon but the diagnosis should be considered where soft tissue masses develop at the site of chronic or recurrent infections. Obtaining tissue for histological examination is key to making the diagnosis. This case suggests that surgical resection is not always needed to achieve a good clinical and radiological outcome.

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 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.

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.

CT and pathological features in renal parenchymal malakoplakia: a case report and review of the literature.

Renal malakoplakia is a rare form of chronic inflammatory granulomatous disease in the kidney. It occurs in adult patients with immunocompromised status or debilitating disease. In the present study, we reported a case of a 50-year-old woman with no underlying disease. This report describes the CT and pathological features of renal malakoplakia in a 50-year-old woman. Plain CT scan showed a large soft tissue mass at the middle and upper pole of the left kidney. Enhanced CT scan showed delayed enhancement in the solid part of the mass. Our results might provide some useful information for the diagnosis of renal parenchymal malakoplakia.

Renal Allograft Malakoplakia Presenting as a Pseudotumoral Lesion.

Malakoplakia is an uncommon inflammatory disease that can involve many organ systems but is often encountered in the urogenital tract. Kidney allograft malakoplakia is even rarer and can have a diffuse parenchymal or a pseudotumoral presentation. We describe a case of grafi malakoplakia in an adult female, who presented with dull aching pain in the right loin, fever, and vomiting. Ultrasonography of the kidney graft showed a heterogeneous lesion (2.6 cm × 2.9 cm), raising suspicion of primary or metastatic renal tumors. The diagnosis was established after a histopathological examination of the kidney biopsy. This pseudotumoral presentation of malakoplakia can mimic renal cell carcinoma, lymphoma, fungal infections, or tuberculosis. It is essential to perform a biopsy for establishing the diagnosis.

Renal malakoplakia with invasion of the liver and diaphragm: a patient case and literature review.

Renal malakoplakia, a seldom seen chronic inflammatory condition, continues to elude medical, surgical, radiological and pathological specialists due to its mimicry of other renal pathologies and low incidence. The variable clinical manifestations and non-specific radiological findings of malakoplakia can be misleading, and ultimately require a pathological diagnosis. A literature review reveals an extremely low prevalence of renal malakoplakia, a handful of invasive renal malakoplakia cases and no reports of liver and diaphragmatic invasion. We present a case of a renal mass with liver and diaphragmatic invasion in a 59-year-old woman that deceived clinicians and radiologists until a pathological diagnosis of renal malakoplakia was performed. This case highlights the need of awareness for malakoplakia in the differential diagnosis for renal invasive and non-invasive masses. The need to await a surgical biopsy and pathological diagnosis is critical to ensure a correct diagnosis and avoid unnecessary surgery of the kidney.

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.

Case study: Malakoplakia of the bladder.

OBJECTIVES: Malakoplakia is a rare chronic inflammatory disease thought to be the result of defective bacterial phagocytosis and lysosome function, and there is difficulty in accurate diagnosis as a result of non-specific symptoms that mimic other diseases and cancers. This study presents a case of bladder malakoplakia associated with renal failure presenting as a tumor. METHODS: A 55-year-old woman with history of kidney disease who presented with general malaise and worsening renal failure was found to have a bladder mass and underwent transurethral resection of bladder tumor (TURBT), and subsequent histological examination. RESULTS: The bladder mass consisted of basophilic structures known as Michaelis-Gutmann bodies within clusters of macrophages on histological examination, and stained positive for CD68. Von Kossa stain highlights Michaelis-Gutmann bodies, consistent with the diagnosis of malakoplakia. CONCLUSIONS: Conservative treatment via antibiotics has been effective. Proper diagnosis of bladder malakoplakia is important, as the conditions it mimics often require surgery and resection. Additionally, it is important to recognize the implications bladder malakoplakia has on renal functioning, particularly regarding urinary obstruction.

[Cutaneous malakoplakia: A rare pseudo-tumor to know in immunocompromised patients]. / Malakoplakie cutanée : une pseudo-tumeur rare à ne pas méconnaître chez l'immunodéprimé.

Cutaneous malakoplakia is a rare pseudo-tumor that occurs in immunocompromised patients. It is a reaction to an infection caused by Gram negative bacteria. The clinical presentation is nonspecific and the diagnosis is histological. The evolution is recurrent and the combination of a surgical treatment, antibiotics and adaptation of immunosuppressive therapy is necessary to cure the disease. The emergence of antibiotic resistance in bacteria responsible for the pathology can complicate the treatment and require additional microbial sampling. We report a case that occurred in a renal transplant patient with a complex diagnostic and therapeutic management.

[An unusual association of malakoplakia and adenocarcinoma of the prostate]. / Asociación inusual de malacoplaquia y adenocarcinoma prostático.

Malakoplakia is a chronic inflammatory process caused by a lysosomal defect in bacterial digestion. Although rare, it occurs more frequently in the genitourinary tract and in patients with immune dysfunction. The bladder is the most commonly affected site, although cases have been reported in other organs, including the prostate gland. Clinically, this lesion can be confused with malignant tumours, both on physical examination and imagining techniques. This is particularly pronounced in the prostate, making the differential diagnosis challenging. Histologically, characteristic aggregates of histiocytes with basophilic intracytoplasmic inclusions composed of calcium and iron salts are found. We present a case diagnosed on transrectal biopsy as acinar adenocarcinoma with a Gleason 5 + 5 = 10 score. Prostatectomy revealed an unusual association of diffuse prostate malakoplakia and an area of acinar adenocarcinoma with a Gleason score of 3 + 4 = 7.

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.

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%).

Unexpected endometrial malacoplakia related to abortion and placental rests retention: a case report.

BACKGROUND: Malacoplakia is a rare chronic inflammatory disease. The name derives from the Greek "µαλακός" meaning "soft" and "πλάξ" meaning "plaque", describing its usual macroscopic presentation as a friable yellow soft plaque. It was first described by von Hansemann in 1901 and by Michaelis and Gutmann in 1902. The urinary system is the most commonly involved site. Female genital tract involvement is extremely rare. Treatment is prevalently based on antibiotics with surgical intervention sometimes necessary. Prognosis is usually good, but relapse may frequently occur. CASE PRESENTATION: This report illustrates the first case of endometrial malacoplakia in a 40 years-old patient who received endometrial curettage due to the retention of placental rests following an abortion. After conspicuous vaginal sero-hematic secretions, the patient received a further curettage. The histological examination did not show any retention of chorionic rests, but an endometrial and myometrial infiltration of histiocytes with large granular cytoplasm within a chronic inflammatory background. Immunoreactivity for CK-pool was negative, while CD68 immunostaining was strongly positive. CONCLUSIONS: Malacoplakia of endometrium is an extremely rare condition, with few cases reported in the whole international literature. In this paper, we present the first case associated to an abortion followed by endometrial curettage procedures. This rare disease should always be attentively examined, considering, among differential diagnoses, uterine neoplasms or physiological conditions such as cumulus of foamy macrophages in the endometrium.