RESUMEN
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.
Asunto(s)
Infecciones por Escherichia coli , Malacoplasia , Infecciones Urinarias , Humanos , Malacoplasia/complicaciones , Malacoplasia/etiología , Infecciones Urinarias/complicaciones , Infecciones Urinarias/tratamiento farmacológico , Infecciones por Escherichia coli/complicaciones , Masculino , Antibacterianos/uso terapéutico , Adulto , Femenino , Escherichia coli/aislamiento & purificaciónRESUMEN
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.
Asunto(s)
Trasplante de Hígado , Malacoplasia , Masculino , Humanos , Niño , Adolescente , Trasplante de Hígado/efectos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiología , Malacoplasia/patología , Donadores Vivos , Piel/patología , Hígado/patologíaRESUMEN
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.
Asunto(s)
Aloinjertos/microbiología , Antibacterianos/administración & dosificación , Azitromicina/administración & dosificación , Infecciones por Escherichia coli/tratamiento farmacológico , Trasplante de Riñón/efectos adversos , Malacoplasia/tratamiento farmacológico , Complicaciones Posoperatorias/tratamiento farmacológico , Escherichia coli/efectos de los fármacos , Escherichia coli/genética , Escherichia coli/fisiología , Infecciones por Escherichia coli/microbiología , Femenino , Humanos , Malacoplasia/etiología , Malacoplasia/microbiología , Pruebas de Sensibilidad Microbiana , Persona de Mediana Edad , Complicaciones Posoperatorias/etiología , Complicaciones Posoperatorias/microbiologíaRESUMEN
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.
Asunto(s)
Infecciones por Actinomycetales/complicaciones , Enfermedades Pulmonares/diagnóstico , Neoplasias Pulmonares/diagnóstico , Trasplante de Pulmón/efectos adversos , Malacoplasia/diagnóstico , Complicaciones Posoperatorias , Rhodococcus equi/aislamiento & purificación , Infecciones por Actinomycetales/epidemiología , Infecciones por Actinomycetales/microbiología , Anciano , Diagnóstico Diferencial , Humanos , Enfermedades Pulmonares/etiología , Neoplasias Pulmonares/etiología , Malacoplasia/etiología , Masculino , PronósticoAsunto(s)
Hidronefrosis , Trasplante de Riñón , Malacoplasia , Humanos , Trasplante de Riñón/efectos adversos , Malacoplasia/tratamiento farmacológico , Malacoplasia/etiología , Antibacterianos/uso terapéutico , Terapia de Inmunosupresión/efectos adversos , Hidronefrosis/complicaciones , Hidronefrosis/tratamiento farmacológicoRESUMEN
Malakoplakia is a granulomatous disease associated with an infectious etiology, usually involving the urinary tract. It reveals itself as a recurrent urinary tract infection (r-UTI), and in some cases, it is associated with impairment of renal function. Immunosuppression is one of its main associated factors, and it has been increasingly described in patients with solid organ transplantation (SOT), mainly kidney transplantation. Macroscopically, it can form masses and sometimes it may be confused with neoplasia, which is why histological findings are fundamental for the diagnosis. Here, we present a case of bladder malakoplakia, manifested by r-UTI from Escherichia coli in a patient with renal transplantation, refractory to long-term antibiotic treatment and reduction in immunosuppression, which resolved after surgical management. We also summarize the clinical characteristics of malakoplakia and compare them with previous reports in the literature on SOT.
Asunto(s)
Trasplante de Riñón/efectos adversos , Malacoplasia/etiología , Malacoplasia/patología , Femenino , Humanos , Persona de Mediana Edad , Enfermedades de la Vejiga Urinaria/etiología , Enfermedades de la Vejiga Urinaria/patologíaRESUMEN
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.
Asunto(s)
Huésped Inmunocomprometido , Malacoplasia/tratamiento farmacológico , Malacoplasia/etiología , Piodermia Gangrenosa/complicaciones , Piodermia Gangrenosa/tratamiento farmacológico , Administración Cutánea , Anciano , Humanos , Masculino , Resultado del TratamientoRESUMEN
Nephrolithic non-functioning kidney and malakoplakia are major health problems. Kidney function cannot be fulfilled and also this leads to a high risk of development of urothelial neoplasm. We report herein a case of urothelial carcinoma concomitant with malakoplakia in non-functioning nephrolithic kidneys.
Asunto(s)
Neoplasias Renales/etiología , Malacoplasia/etiología , Anciano , Humanos , MasculinoRESUMEN
Malakoplakia is an unusual chronic inflammatory disease related to prior urinary tract infection. It is characterized by the presence of macrophages with foamy cytoplasm exhibiting larger PAS positive inclusions that stain for calcium and iron. Malakoplakia affects renal allograft and is associated with severe morbidity. Herein, the authors report a new case of renal graft malakoplakia in a 23-year-old female patient. The patient received a living-related donor renal transplantation with a high immunological risk. Plasmapheresis and intravenous immunoglobulin (i.v. Ig) treatment, pre- and post-transplant, and induction with rabbit anti-thymocyte globulins were used due to presence of donor specific antibodies and positive B cross match by flow cytometry. The patient had an early urinary tract infection with a good outcome. On Day 36 post-transplant (PO), the patient returned to the clinic with fever, graft pain and acute renal dysfunction leading to hemodialysis. Escherichia coli (E. coli) was present in the blood and urine culture. At the time, the renal biopsy revealed numerous sheets of macrophages with foamy, eosinophilic cytoplasm showing several PAS positive granules and large inclusions that stained strongly with hematoxylin, calcium (von Kossa method) and iron (Prussian blue). The patient was diagnosed with malakoplakia related to a kidney transplant. Despite prolonged treatment with antibiotics, determined by a susceptibility test, the patient did not recover renal function and remained on dialysis.
Asunto(s)
Infecciones por Escherichia coli/etiología , Trasplante de Riñón/efectos adversos , Malacoplasia/etiología , Infecciones Urinarias/etiología , Antibacterianos/uso terapéutico , Biopsia , Infecciones por Escherichia coli/diagnóstico , Infecciones por Escherichia coli/terapia , Femenino , Histocompatibilidad , Humanos , Inmunosupresores/efectos adversos , Donadores Vivos , Malacoplasia/diagnóstico , Malacoplasia/terapia , Diálisis Renal , Infecciones Urinarias/diagnóstico , Infecciones Urinarias/terapia , Adulto JovenAsunto(s)
Infecciones por Actinomycetales/patología , Enfermedades Pulmonares/microbiología , Enfermedades Pulmonares/patología , Rhodococcus equi , Infecciones por Actinomycetales/complicaciones , Anciano , Diagnóstico Diferencial , Femenino , Humanos , Enfermedades Pulmonares/complicaciones , Neoplasias Pulmonares/patología , Malacoplasia/etiologíaRESUMEN
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.
Asunto(s)
Trasplante de Riñón , Malacoplasia , Masculino , Humanos , Persona de Mediana Edad , Malacoplasia/tratamiento farmacológico , Malacoplasia/etiología , Piel , Trasplante de Riñón/efectos adversos , Antibacterianos/uso terapéuticoRESUMEN
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.
Asunto(s)
Neoplasias Renales , Trasplante de Riñón , Malacoplasia , Adulto , Femenino , Humanos , Trasplante de Riñón/efectos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiología , Malacoplasia/patología , Riñón/patología , Aloinjertos/patologíaRESUMEN
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.
Asunto(s)
Malacoplasia , Neuropatías Peroneas , Femenino , Humanos , Malacoplasia/diagnóstico , Malacoplasia/etiología , Malacoplasia/patología , Neuropatías Peroneas/complicaciones , Neuropatías Peroneas/tratamiento farmacológico , Reinfección/complicaciones , Reinfección/tratamiento farmacológico , Antibacterianos/uso terapéutico , Ácido Ascórbico/uso terapéuticoRESUMEN
Malakoplakia is a rare chronic granulomatous disease of unknown cause. It is thought to be caused by an acquired bactericidal defect of macrophages. Malakoplakia is associated with chronic infections and immunosuppression. Although it occurs mainly in the urinary tract, it has already been reported in almost every organ system. The isolation of bacteria, especially Escherichia coli, is common in malakoplakia patients. Here, we present a case of primary cutaneous malakoplakia in a kidney transplant recipient who had been taking prednisone, tacrolimus, and mycophenolate. Culture of a lesion grew Burkholderia cepacia complex. Treatment with high doses of trimethoprim-sulfamethoxazole was successful. We also present a systematic review of the literature, identifying 4 previously reported cases of malakoplakia after renal transplantation under similar immunosuppressive therapy, most occurring in the urinary tract or perineum and following benign courses to cure. Data in the literature suggest that malakoplakia has become even rarer since changes were made in the immunosuppressive therapy employed after kidney transplantation.
Asunto(s)
Inmunosupresores/uso terapéutico , Trasplante de Riñón/efectos adversos , Malacoplasia/prevención & control , Ácido Micofenólico/análogos & derivados , Adulto , Humanos , Huésped Inmunocomprometido , Malacoplasia/etiología , Masculino , Ácido Micofenólico/uso terapéuticoRESUMEN
We report a case of a 72-year-old male that underwent two sets of transrectal prostatic needle biopsy (TPNB) within 9 month period. Pathology showed unremarkable benign prostatic tissues in the first group of biopsies while extensive diffuse inflammation with the characteristic features of malakoplakia in the second set. Three cores in the repeat biopsy contained foci of prostatic adenocarcinoma as well. Occurrence of malakoplakia several months after TPNB in our case suggests that microorganisms may have been inoculated to the prostate during the biopsy procedure. We believe that malakoplakia must be added to the list of complications after TPNB.
Asunto(s)
Biopsia con Aguja/efectos adversos , Malacoplasia/etiología , Siembra Neoplásica , Enfermedades de la Próstata/etiología , Adenocarcinoma/diagnóstico , Adenocarcinoma/epidemiología , Adenocarcinoma/patología , Anciano , Comorbilidad , Humanos , Malacoplasia/epidemiología , Malacoplasia/patología , Masculino , Clasificación del Tumor , Próstata/patología , Enfermedades de la Próstata/epidemiología , Enfermedades de la Próstata/patología , Neoplasias de la Próstata/diagnóstico , Neoplasias de la Próstata/epidemiología , Neoplasias de la Próstata/patologíaRESUMEN
Malacoplakia is a benign and uncommon inflammatory response that develops most frequently in the genitourinary tract but has been reported at other sites. We report the case of 67-year-old man who presented with a lesion at the base of the tongue 7 months after chemoradiation for biopsy-proven invasive squamous cell carcinoma of the tongue. The lesion showed intense avidity by positron emission tomography and was biopsied with the putative clinical diagnosis of recurrent malignancy. Histologically, the lesion showed the characteristics of malacoplakia as well as abundant intracellular gram-negative bacilli. Ultrastructural analysis revealed giant lysosomes, intracytoplasmic calcific concretions (Michaelis-Gutman bodies), and partially digested gram-negative bacilli within vacuolated lysosomes of macrophages. To our knowledge, this is the seventh reported case of malacoplakia of the tongue and the first to develop at the site of prior radiation treatment of a carcinoma. The clinical features of this case, a review of previously reported cases, and a consideration of pathogenesis are presented.
Asunto(s)
Carcinoma de Células Escamosas/radioterapia , Malacoplasia/etiología , Malacoplasia/patología , Radioterapia/efectos adversos , Neoplasias de la Lengua/radioterapia , Anciano , Candidiasis/etiología , Infecciones por Bacterias Grampositivas , Humanos , Masculino , Estomatitis/etiologíaRESUMEN
This is a case report of a 65-year-old female with malakoplakia in the urinary bladder. An ultrasound scan showed many tumor-like polyp lesions, where a transurethral resection was performed. Since the lesions revealed gross papillary neoplasms, malignancy was suspected. However, histpathological examination showed an aggregation of many infiltrating histiocytes containing characteristic Michaelis-Gutmann bodies positively reactive with von Kossa staining, indicating a typical case of malakoplakia of the urinary bladder. Immunohistochemical study demonstrated that the cytoplasms of the histiocytes were focally positive for an anti-Escherichia coli antibody. One of the etiologies of malakoplakia is infection by Escherichia coli, which is supported by our data.
Asunto(s)
Malacoplasia/patología , Enfermedades de la Vejiga Urinaria/patología , Anciano , Anticuerpos Antibacterianos/análisis , Escherichia coli/inmunología , Infecciones por Escherichia coli/complicaciones , Femenino , Humanos , Inmunohistoquímica , Malacoplasia/etiología , Enfermedades de la Vejiga Urinaria/etiologíaRESUMEN
Lung nodules or masses due to a variety of malignant or benign conditions such as opportunistic infections are observed after lung transplant. Malakoplakia is a rare complication in immunocompromised patients. Here we describe the clinical course and management of a lung transplant recipient with pulmonary malakoplakia and provide a review of the literature. To our knowledge, this is the first report of a case of pulmonary malakoplakia due to Escherichia coli infection in a lung allograft.
Asunto(s)
Infecciones por Escherichia coli , Trasplante de Pulmón , Malacoplasia , Humanos , Pulmón/diagnóstico por imagen , Trasplante de Pulmón/efectos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiología , Receptores de TrasplantesRESUMEN
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.
Asunto(s)
Trasplante de Riñón , Malacoplasia , Neoplasias , Antibacterianos/uso terapéutico , Humanos , Huésped Inmunocomprometido , Trasplante de Riñón/efectos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiología , Malacoplasia/patología , Neoplasias/tratamiento farmacológicoRESUMEN
A 45-year-old woman presented to us in March 2019 with complaints of fever and right lower quadrant abdominal pain for 1 month. She had undergone renal transplantation in 2017 for end-stage renal disease and developed four episodes of urinary tract infection in the next 16 months post transplantation, which were treated based on culture reports. She was subsequently kept on long-term prophylaxis with trimethoprim and sulfamethoxazole. Her present laboratory parameters showed a normal blood picture and elevated creatinine. Urine culture grew Escherichia coli Non-contrast CT of the abdomen-pelvis revealed an endo-exophytic hyperdense mass in the graft kidney showing local infiltration and associated few regional lymph nodes. PET-CT revealed the soft-tissue mass and regional lymph nodes to be hypermetabolic, raising the possibility of lymphoma. However, biopsy showed features of malakoplakia. She was subsequently initiated on long-term antibiotic therapy and her immunosuppression decreased.