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Journal of Comparative Pathology Aug 2023Malakoplakia is a rare chronic granulomatous disease usually affecting the urinary bladder and other locations. In humans, the gastrointestinal tract is the second most...
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.
Topics: Humans; Animals; Dogs; Female; Malacoplakia; Intestines; Colitis, Ulcerative; Dog Diseases
PubMed: 37647838
DOI: 10.1016/j.jcpa.2023.07.002 -
Diagnostic Pathology Aug 2023Malakoplakia is a rare inflammatory disease of the urogenital tract. There have been no reports of malakoplakia expressing anaplastic lymphoma kinase (ALK) to date....
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.
Topics: Female; Humans; Aged; Anaplastic Lymphoma Kinase; Immunohistochemistry; Malacoplakia; In Situ Hybridization, Fluorescence; Periodic Acid
PubMed: 37644531
DOI: 10.1186/s13000-023-01383-z -
A Case of Urinary Bladder Malakoplakia in a Young French Bulldog: Diagnostic and Therapeutic Issues.Topics in Companion Animal Medicine 2023A 3-month-old female French Bulldog presented with hematuria, severe pollakiuria, and urinary incontinence lasting for 1.5 months. Broad-spectrum empirical antibiotic...
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.
Topics: Humans; Dogs; Female; Animals; Urinary Bladder; Hematuria; Malacoplakia; Cystitis; Anti-Bacterial Agents; Dog Diseases
PubMed: 37597743
DOI: 10.1016/j.tcam.2023.100804 -
Clinical Nuclear Medicine Aug 2023Abdominal contrast-enhanced CT was performed in a 61-year-old man with difficulties of urination and defecation for 4 months, which revealed huge rectal masses involving...
Abdominal contrast-enhanced CT was performed in a 61-year-old man with difficulties of urination and defecation for 4 months, which revealed huge rectal masses involving multiple adjacent organs, suspected as malignant lesions. 18 F-FDG PET/CT was subsequently performed for staging. The images showed intense FDG uptake and slightly hyperdense masses involving rectum, bladder, prostate, left ureter, and the anterior abdominal wall at the level of the pelvic cavity. Histopathological examination confirmed the masses were due to malakoplakia, which displayed as abundant von Hansemann cells aggregated and infiltrated in lesions, with distinctive cytoplasmic inclusions termed Michaelis-Gutmann bodies.
Topics: Male; Humans; Middle Aged; Fluorodeoxyglucose F18; Positron Emission Tomography Computed Tomography; Malacoplakia; Positron-Emission Tomography; Neoplasms
PubMed: 37276495
DOI: 10.1097/RLU.0000000000004728 -
Ugeskrift For Laeger May 2023Malakoplakia is a form of chronic, granulomatous, inflammatory condition which usually affects the genitourinary tract or other internal organs of immunocompromised...
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.
Topics: Male; Humans; Middle Aged; Malacoplakia; Skin; Kidney Transplantation; Anti-Bacterial Agents
PubMed: 37264866
DOI: No ID Found -
Revista de Gastroenterologia Del Peru :... 2023Colonic 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...
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.
Topics: Male; Humans; Middle Aged; Rare Diseases; Malacoplakia; Colon; Biopsy; Diarrhea; Syndrome
PubMed: 37226073
DOI: No ID Found -
Zhonghua Bing Li Xue Za Zhi = Chinese... May 2023
Topics: Child; Humans; Malacoplakia; Intestines
PubMed: 37106292
DOI: 10.3760/cma.j.cn112151-20220801-00667 -
Journal of Medical Case Reports Apr 2023Malakoplakia is a rare condition characterized by inflammatory masses with specific histological characteristics. These soft tissue masses can mimic tumors and tend to...
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.
Topics: Female; Humans; Malacoplakia; Peroneal Neuropathies; Reinfection; Anti-Bacterial Agents; Ascorbic Acid
PubMed: 37024963
DOI: 10.1186/s13256-023-03883-4 -
Pediatric Transplantation Aug 2023Malakoplakia occurs uncommonly at any age, but pediatric reports are exceptionally limited. Malakoplakia appears primarily in the urinary tract, although involvement of... (Review)
Review
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.
Topics: Male; Humans; Child; Adolescent; Liver Transplantation; Malacoplakia; Living Donors; Skin; Liver
PubMed: 36860187
DOI: 10.1111/petr.14492 -
Frontiers in Surgery 2023The malacoplakia of the bladder is a rare chronic acquired infection- associated granulomatous disease and even less common in combination with urinary stones.
INTRODUCTION
The malacoplakia of the bladder is a rare chronic acquired infection- associated granulomatous disease and even less common in combination with urinary stones.
CASE PRESENTATION
We report the case of a 58-year-old female patient with malacoplakia of the bladder combined with renal calculi. The patient was admitted to the hospital with bilateral low back pain for one month and space-occupying lesions of the bladder for three days. Preoperative imaging suggested space-occupying lesions of the bladder: high probability of bladder cancer. Following the anti-infection treatment, the transurethral electrodesiccation was performed on the space-occupying lesions of the bladder. Pathological examination confirmed the diagnosis of malacoplakia of the bladder. Left-sided percutaneous nephrolithotomy was performed electively to remove the predisposing factors of infection. After the operation, the patient continued to receive anti-infection treatment for two months. The patient had a good prognosis in the six-month follow-up.
CONCLUSIONS
Malacoplakia of the bladder is easily misdiagnosed as bladder cancer before operation, and the diagnosis depends on pathological diagnosis. Complete removal of urinary calculi, infection and other inducing factors, is beneficial to the treatment of malacoplakia of the bladder.
PubMed: 36778649
DOI: 10.3389/fsurg.2023.1044963