RESUMO
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.
Assuntos
Transplante de Rim , Malacoplasia , Masculino , Humanos , Pessoa de Meia-Idade , Malacoplasia/tratamento farmacológico , Malacoplasia/etiologia , Pele , Transplante de Rim/efeitos adversos , Antibacterianos/uso terapêuticoRESUMO
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.
Assuntos
Malacoplasia , Neuropatias Fibulares , Feminino , Humanos , Malacoplasia/diagnóstico , Malacoplasia/etiologia , Malacoplasia/patologia , Neuropatias Fibulares/complicações , Neuropatias Fibulares/tratamento farmacológico , Reinfecção/complicações , Reinfecção/tratamento farmacológico , Antibacterianos/uso terapêutico , Ácido Ascórbico/uso terapêuticoRESUMO
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.
Assuntos
Neoplasias Renais , Transplante de Rim , Malacoplasia , Adulto , Feminino , Humanos , Transplante de Rim/efeitos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiologia , Malacoplasia/patologia , Rim/patologia , Aloenxertos/patologiaRESUMO
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.
Assuntos
Transplante de Rim , Malacoplasia , Neoplasias , Antibacterianos/uso terapêutico , Humanos , Hospedeiro Imunocomprometido , Transplante de Rim/efeitos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiologia , Malacoplasia/patologia , Neoplasias/tratamento farmacológicoRESUMO
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.
Assuntos
Infecções por Escherichia coli , Transplante de Pulmão , Malacoplasia , Humanos , Pulmão/diagnóstico por imagem , Transplante de Pulmão/efeitos adversos , Malacoplasia/diagnóstico , Malacoplasia/etiologia , TransplantadosRESUMO
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.
Assuntos
Transplante de Rim , Transtornos Linfoproliferativos , Malacoplasia , Infecções Urinárias , Feminino , Humanos , Transplante de Rim/efeitos adversos , Transtornos Linfoproliferativos/diagnóstico , Transtornos Linfoproliferativos/etiologia , Malacoplasia/diagnóstico , Malacoplasia/etiologia , Pessoa de Meia-Idade , Tomografia por Emissão de Pósitrons combinada à Tomografia ComputadorizadaRESUMO
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.
Assuntos
Aloenxertos/microbiologia , Antibacterianos/administração & dosagem , Azitromicina/administração & dosagem , Infecções por Escherichia coli/tratamento farmacológico , Transplante de Rim/efeitos adversos , Malacoplasia/tratamento farmacológico , Complicações Pós-Operatórias/tratamento farmacológico , Escherichia coli/efeitos dos fármacos , Escherichia coli/genética , Escherichia coli/fisiologia , Infecções por Escherichia coli/microbiologia , Feminino , Humanos , Malacoplasia/etiologia , Malacoplasia/microbiologia , Testes de Sensibilidade Microbiana , Pessoa de Meia-Idade , Complicações Pós-Operatórias/etiologia , Complicações Pós-Operatórias/microbiologiaRESUMO
BACKGROUND: Malacoplakia is a rare chronic inflammatory disease. The name derives from the Greek "µαλακÏς" 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.
Assuntos
Aborto Induzido/efeitos adversos , Dilatação e Curetagem/efeitos adversos , Endométrio/patologia , Malacoplasia/etiologia , Malacoplasia/patologia , Adulto , Feminino , Humanos , GravidezAssuntos
Infecções por Actinomycetales/patologia , Pneumopatias/microbiologia , Pneumopatias/patologia , Rhodococcus equi , Infecções por Actinomycetales/complicações , Idoso , Diagnóstico Diferencial , Feminino , Humanos , Pneumopatias/complicações , Neoplasias Pulmonares/patologia , Malacoplasia/etiologiaRESUMO
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.
Assuntos
Infecções por Actinomycetales/complicações , Pneumopatias/diagnóstico , Neoplasias Pulmonares/diagnóstico , Transplante de Pulmão/efeitos adversos , Malacoplasia/diagnóstico , Complicações Pós-Operatórias , Rhodococcus equi/isolamento & purificação , Infecções por Actinomycetales/epidemiologia , Infecções por Actinomycetales/microbiologia , Idoso , Diagnóstico Diferencial , Humanos , Pneumopatias/etiologia , Neoplasias Pulmonares/etiologia , Malacoplasia/etiologia , Masculino , PrognósticoRESUMO
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.
Assuntos
Transplante de Rim/efeitos adversos , Malacoplasia/etiologia , Malacoplasia/patologia , Feminino , Humanos , Pessoa de Meia-Idade , Doenças da Bexiga Urinária/etiologia , Doenças da Bexiga Urinária/patologiaRESUMO
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.
Assuntos
Hospedeiro Imunocomprometido , Malacoplasia/tratamento farmacológico , Malacoplasia/etiologia , Pioderma Gangrenoso/complicações , Pioderma Gangrenoso/tratamento farmacológico , Administração Cutânea , Idoso , Humanos , Masculino , Resultado do TratamentoRESUMO
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.
Assuntos
Neoplasias Renais/etiologia , Malacoplasia/etiologia , Idoso , Humanos , MasculinoRESUMO
A 32-year-old woman named A.C. was diagnosed five years ago with stage IIIB nodular sclerosing Hodgkin lymphoma (HL). Following initial chemotherapy, she had refractory disease as evidenced by hypermetabolic activity on positron-emission tomography (PET) scan. A.C. was treated with a short course of salvage chemotherapy, stem cell collection, and autologous HSCT about 11 months after initial diagnosis. Her post-transplantation course was complicated by pneumonia and interstitial lung disease secondary to chemotherapy that rapidly improved with high-dose steroidsâ©.
Assuntos
Transplante de Células-Tronco Hematopoéticas/efeitos adversos , Malacoplasia/etiologia , Adulto , Feminino , Humanos , Malacoplasia/enfermagem , Transplante HomólogoRESUMO
Malakoplakia is a rare chronic granulomatous inflammatory disease, related to immune deficiency and impaired bactericidal macrophage activity. Common sites of involvement include urinary bladder and kidney followed by gastrointestinal tract. We present an unusual case of abdominal malakoplakia diagnosed preoperatively on ultrasound-guided fine-needle aspiration cytology (FNAC). It presented as a large locally aggressive mass with extensive involvement of multiple bowel loops in an 18-year-old boy following blunt trauma to the abdomen. The demonstration of calcified laminated intracytoplasmic Michaelis-Gutman bodies in histiocytes is a reliable diagnostic feature of malakoplakia on FNAC smears, which can guide an appropriate medical management.
Assuntos
Traumatismos Abdominais/complicações , Malacoplasia/diagnóstico , Adolescente , Biópsia por Agulha Fina , Humanos , Malacoplasia/etiologia , Malacoplasia/patologia , MasculinoAssuntos
Transplante de Rim , Rim/patologia , Malacoplasia/patologia , Adolescente , Biópsia , Diagnóstico Diferencial , Infecções por Escherichia coli/tratamento farmacológico , Infecções por Escherichia coli/etiologia , Feminino , Humanos , Imunossupressores/efeitos adversos , Falência Renal Crônica/diagnóstico por imagem , Falência Renal Crônica/patologia , Falência Renal Crônica/cirurgia , Imageamento por Ressonância Magnética , Malacoplasia/diagnóstico , Malacoplasia/etiologia , UltrassonografiaRESUMO
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.
Assuntos
Infecções por Escherichia coli/etiologia , Transplante de Rim/efeitos adversos , Malacoplasia/etiologia , Infecções Urinárias/etiologia , Antibacterianos/uso terapêutico , Biópsia , Infecções por Escherichia coli/diagnóstico , Infecções por Escherichia coli/terapia , Feminino , Histocompatibilidade , Humanos , Imunossupressores/efeitos adversos , Doadores Vivos , Malacoplasia/diagnóstico , Malacoplasia/terapia , Diálise Renal , Infecções Urinárias/diagnóstico , Infecções Urinárias/terapia , Adulto JovemRESUMO
Malakoplakia is a rare acquired disease that can affect many systems but is more common in the urogenital tract. Cutaneous malakoplakia is even rarer. It is far more frequent in immunodeficient patients. We report a case of cutaneous malakoplakia in a kidney transplant patient who had recently stopped receiving immunosuppressive therapy to illustrate a review of the relevant recent literature.
Malacoplaquia é uma doença adquirida rara que pode afetar diversos órgãos e sistemas, mas é mais comum no trato urogenital. O acometimento cutâneo é ainda menos frequente. Atinge principalmente imunodeficientes. Relatamos caso de malacoplaquia cutânea em um paciente transplantado renal que havia recentemente deixado de receber a terapia imunossupressora, a fim de ilustrar uma revisão da literatura recente relevante.
Assuntos
Humanos , Masculino , Pessoa de Meia-Idade , Transplante de Rim/efeitos adversos , Malacoplasia/patologia , Dermatopatias/patologia , Diagnóstico Diferencial , Hospedeiro Imunocomprometido , Malacoplasia/etiologia , Dermatopatias/etiologiaRESUMO
Malakoplakia is a rare granulomatous disorder of unknown aetiology and usually affects patients with underlying immunosuppression. This disorder usually involves the genitourinary tract but has been reported in a wide array of anatomical sites. We are presenting, what is to our knowledge, the first case in the literature in which a patient was diagnosed with malakoplakia and after his successful management; he was diagnosed with chronic meylomoncytic leukaemia. All cases of malakoplakia reported in the literature were either preceded or accompanied by an immunosuppressive state.