ABSTRACT
Cerebral feohifomycosis are severe infections caused by dematiaceous fungi. Cladophialophora bantiana is one of the most commonly isolated species; it has central nervous system tropism and it often manifests as a brain abscess in immunocompetent patients. In immunocompromised patients, it can lead to brain abscesses and disseminated infections. Despite the availability of broad-spectrum antifungal drugs, it is a must to perform surgical management, in addition to drug therapy. However, mortality is high. The diagnostic approach must be invasive to establish a timely diagnosis and direct treatment based on culture and susceptibility tests. We report a case of brain abscess caused by C. bantiana in an immunosuppressed patient who was treated with surgical resection and voriconazole with an adequate response to therapy and without neurological sequels.
Las feohifomicosis cerebrales son infecciones graves causadas por mohos dematiáceos, entre los cuales Cladophialophora bantiana es una de las especies más comúnmente aislada. Esta tiene tropismo por el sistema nervioso central y frecuentemente produce abscesos cerebrales en pacientes inmunocompetentes; además, en los inmunocomprometidos también puede ocasionar infección diseminada. Pese a la disponibilidad de medicamentos antifúngicos de amplio espectro, a menudo se requiere también la intervención quirúrgica; de todas maneras, la mortalidad es elevada. El diagnóstico debe hacerse interviniendo para tomar la muestra y hacer el cultivo y las pruebas de sensibilidad. Se presenta aquí el caso de un paciente con trasplante renal que presentó un absceso cerebral por C. bantiana, el cual se extrajo mediante resección quirúrgica. El paciente recibió tratamiento con voriconazol, con adecuada respuesta, mejoría y sin secuelas neurológicas.
Subject(s)
Brain Abscess/microbiology , Cerebral Phaeohyphomycosis/microbiology , Kidney Transplantation , Postoperative Complications/microbiology , Saccharomycetales/isolation & purification , Amphotericin B/therapeutic use , Antifungal Agents/therapeutic use , Brain Abscess/drug therapy , Brain Abscess/etiology , Brain Abscess/surgery , Cerebral Phaeohyphomycosis/drug therapy , Cerebral Phaeohyphomycosis/etiology , Cerebral Phaeohyphomycosis/surgery , Combined Modality Therapy , Craniotomy , Graft Rejection/drug therapy , Humans , Hyperoxaluria, Primary/complications , Hyperoxaluria, Primary/diagnosis , Hyperoxaluria, Primary/genetics , Immunocompromised Host , Immunosuppressive Agents/adverse effects , Immunosuppressive Agents/therapeutic use , Male , Middle Aged , Nephrolithiasis/etiology , Postoperative Complications/drug therapy , Postoperative Complications/etiology , Postoperative Complications/surgery , Recurrence , Renal DialysisABSTRACT
Resumen Las feohifomicosis cerebrales son infecciones graves causadas por mohos dematiáceos, entre los cuales Cladophialophora bantiana es una de las especies más comúnmente aislada. Esta tiene tropismo por el sistema nervioso central y frecuentemente produce abscesos cerebrales en pacientes inmunocompetentes; además, en los inmunocomprometidos también puede ocasionar infección diseminada. Pese a la disponibilidad de medicamentos antifúngicos de amplio espectro, a menudo se requiere también la intervención quirúrgica; de todas maneras, la mortalidad es elevada. El diagnóstico debe hacerse interviniendo para tomar la muestra y hacer el cultivo y las pruebas de sensibilidad. Se presenta aquí el caso de un paciente con trasplante renal que presentó un absceso cerebral por C. bantiana, el cual se extrajo mediante resección quirúrgica. El paciente recibió tratamiento con voriconazol, con adecuada respuesta, mejoría y sin secuelas neurológicas.
Abstract Cerebral feohifomycosis are severe infections caused by dematiaceous fungi. Cladophialophora bantiana is one of the most commonly isolated species; it has central nervous system tropism and it often manifests as a brain abscess in immunocompetent patients. In immunocompromised patients, it can lead to brain abscesses and disseminated infections. Despite the availability of broad-spectrum antifungal drugs, it is a must to perform surgical management, in addition to drug therapy. However, mortality is high. The diagnostic approach must be invasive to establish a timely diagnosis and direct treatment based on culture and susceptibility tests. We report a case of brain abscess caused by C. bantiana in an immunosuppressed patient who was treated with surgical resection and voriconazole with an adequate response to therapy and without neurological sequels.
Subject(s)
Humans , Male , Middle Aged , Postoperative Complications/microbiology , Brain Abscess/microbiology , Kidney Transplantation , Saccharomycetales/isolation & purification , Cerebral Phaeohyphomycosis/microbiology , Postoperative Complications/surgery , Postoperative Complications/etiology , Postoperative Complications/drug therapy , Recurrence , Hyperoxaluria, Primary/complications , Hyperoxaluria, Primary/diagnosis , Hyperoxaluria, Primary/genetics , Brain Abscess/surgery , Brain Abscess/etiology , Brain Abscess/drug therapy , Amphotericin B/therapeutic use , Renal Dialysis , Immunocompromised Host , Combined Modality Therapy , Craniotomy , Nephrolithiasis/etiology , Cerebral Phaeohyphomycosis/surgery , Cerebral Phaeohyphomycosis/etiology , Cerebral Phaeohyphomycosis/drug therapy , Graft Rejection/drug therapy , Immunosuppressive Agents/adverse effects , Immunosuppressive Agents/therapeutic use , Antifungal Agents/therapeutic useABSTRACT
Primary hyperoxaluria is a congenital innate error of the metabolism of the amino acids, that is transmitted like an autosomal recessive character. Two types of hyperoxaluria exist: the primary type I, that corresponds to the peroxisomal enzymatic deficit of the alanine glyoxylate aminotransferase in the liver (AGT) and type II, due to the deficit of the glyoxylate reductase/hydroxypyruvate reductase deficiency (GRHPR). The primary type I (AGT) is the most frequenty. We report the case of a female infant of one month of age, that on her first day post birth, presented myoclonic convulsions and tonic spasms, both during wakefullness and sleep periods, that became more frequent and did not respond to the use of anticonvulsants. The ictal Electroencephalogram presented an intermittent activity of spikes and spike-waves of high voltage in the right hemisphere. Eight minutes after the intravenous administration of 150 mg of pyridoxine, it was observed a diminution of the epileptic activity, as well as the clinical manifestations. The determination of organic acids in urine revealed an increase in the concentration levels of oxalic acid (3064 mmol/mol of creatinine). The molecular genetic study of the AGXT gene, showed the existence of a R197Q mutation in exón 5 of the patient and her father. She received treatment with pyridoxine at a dose of 50 mg/day. When she reached the age of three months both a normal electroencephalogram and biochemistry were obtained. Although it is a rare cause of neonatal convulsions, hyperoxaluria, due to new mutations is an underdiagnosed disease by neonatologists and paediatricias.
Subject(s)
Hyperoxaluria, Primary/genetics , Mutation/genetics , Transaminases/genetics , Female , Humans , Infant , Polymorphism, GeneticABSTRACT
We describe 4 patients, aged 3 months to 23 years, with end-stage renal disease and severe, symptomatic hypothyroidism. All 4 had primary hyperoxaluria type 1 (PH1) with diffuse tissue (kidneys, skeleton, eyes, heart) calcium-oxalate deposition, a condition known as oxalosis. The hypothyroidism responded to thyroid hormone replacement therapy. Clinical hypothyroidism within the framework of PH1/oxalosis was probably caused by thyroid tissue damage from an abundance of calcium oxalate. We recommend that thyroid function be monitored in patients with PH1 and oxalosis.
Subject(s)
Hyperoxaluria, Primary/complications , Hypothyroidism/etiology , Child, Preschool , Consanguinity , Female , Humans , Hyperoxaluria, Primary/genetics , Hypothyroidism/drug therapy , Infant , Male , Thyrotropin-Releasing Hormone/therapeutic use , Thyroxine/therapeutic useABSTRACT
Oxalosis, the systemic deposition of calcium oxalate crystals, may occur in several hyperoxaluric states due to increased production or absorption of oxalate. Type I primary hyperoxaluria (PH I) is a rare autosomal recessive disease caused by deficiency of the peroxisomal enzyme alanine:glyoxylate aminotransferase. Most patients with this disorder are noticed in mid-childhood or even later due to symptoms related to urinary stone disease. In this paper, we report a patient with PH I with a rapid downhill progression to renal failure and death. Oxalosis was detected by renal biopsy, and the diagnosis of PH I was confirmed by increased urinary oxalate and glycolate levels.