ABSTRACT
BACKGROUND: Nasal chondromesenchymal hamartomas (NCMHs) are benign, slow-growing lesions formed by mesenchymal and cartilaginous components. They occur predominantly in male infants at the nasopharynx and orbit. Rare cases have been reported in adults. Ectopic NCMHs occurring in other head regions without the typical nasopharyngeal or orbital involvement have not been previously described. OBSERVATIONS: The authors presented the case of a 40-year-old woman with a giant mass in the left frontoparietal region that started to enlarge progressively after the patient's first pregnancy at the age of 21 years. The tumor caused intense headaches, nausea, vomiting, asthenia, and syncope. On admission, the neurological examination revealed no abnormalities. Brain magnetic resonance imaging showed a solid homogeneous tumor without intraaxial involvement extending inferiorly to the left zygomatic arch, with a significant mass effect on the adjacent bones but no infiltration. Remarkably, digital subtraction angiography demonstrated that the tumor received blood supply from superficial as well as intracranial branches of the left vertebral artery. After tumor resection, histopathological analysis revealed characteristics indistinguishable from an NCMH. LESSONS: The authors described a rare NCMH of the scalp with intracranial blood supply in an adult patient. A case with similar characteristics had not been reported before.
ABSTRACT
Antecedentes. La mucormicosis pulmonar es una infección oportunista rara con alta mortalidad causada por hongos Mucorales. Los más frecuentes son Rhizopus, Mucor, Lichtheimia y Rhizomucor. Caso clínico. Se presenta el caso de una mujer de 56 años con diabetes mellitus tipo 2 y enfermedad renal crónica, receptora de un trasplante de riñón cadavérico dos años antes de su ingreso. Por este motivo recibió tratamiento inmunomodulador con timoglobulina, micofenolato de mofetilo, tacrolimus y prednisona. La paciente ingresó por un cuadro neumónico con tos, expectoración y disnea; una tomografía computarizada mostró una lesión cavitada en el lóbulo superior derecho. Con la sospecha de una aspergilosis pulmonar invasiva se comenzó un tratamiento antifúngico con voriconazol, sin mejoría. Se realizó una biopsia por aspiración con aguja fina, y en el cultivo de la misma creció Rhizomucor pusillus. La identificación se confirmó por PCR. A pesar del tratamiento con anfotericina B, la paciente presentó hemoptisis masiva incoercible, que provocó su fallecimiento. Conclusiones. La mucormicosis pulmonar es una infección rara que suele ser fatal en receptores de trasplante renal con terapia antirrechazo. Los mucorales producen fenómenos trombóticos, necrosis y destrucción tisular, que provocaron en nuestra paciente una hemoptisis incoercible. Es importante un diagnóstico preciso que permita instaurar un tratamiento quirúrgico adecuado y la administración de anfotericina B (AU)
Background. Pulmonary mucormycosis is a rare opportunistic infection with high mortality that is caused by species of Mucorales. The most common species involved are Rhizopus, Mucor, Lichtheimia, and Rhizomucor. Case report. A 56 year-old woman presented with a clinical history of diabetes mellitus type 2 and chronic renal disease. She underwent a cadaveric kidney transplantation two years before her admission, for which immunomodulating therapy with thymoglobulin, tacrolimus, mofetil-microphenolate and prednisone was established. The patient suffered a pneumonic process with cough, expectoration, and dyspnoea. The computed tomography scan showed a cavitation in the right upper lobe. With all these findings an invasive broncopulmonary aspergillosis was suspected and the patient began an antifungal treatment with voriconazole without improvement. Rhizomucor pusillus was isolated from a clinical specimen obtained by fine needle aspiration, and its identification was confirmed by PCR. After this finding amphotericin B was administered, but the patient had an uncontrolled haemoptysis and died. Conclusions. Pulmonary mucormycosis is a rare infection, usually fatal in kidney transplant recipients with anti-rejection therapy. Mucorales species usually produce thrombotic phenomena, associated with necrosis and parenchymal destruction that caused a fatal uncontrolled haemoptysis in our patient. Early diagnosis is important in order to perform any surgical treatment and to administer amphotericin B (AU)
Subject(s)
Humans , Male , Middle Aged , Hemoptysis/etiology , Kidney Transplantation , Mucormycosis/diagnosis , Lung Diseases, Fungal/diagnosis , Mucorales/isolation & purification , Diabetes Mellitus, Type 2/complications , Rhizomucor/pathogenicity , Antifungal Agents/therapeutic useABSTRACT
BACKGROUND: Pulmonary mucormycosis is a rare opportunistic infection with high mortality that is caused by species of Mucorales. The most common species involved are Rhizopus, Mucor, Lichtheimia, and Rhizomucor. CASE REPORT: A 56 year-old woman presented with a clinical history of diabetes mellitus type 2 and chronic renal disease. She underwent a cadaveric kidney transplantation two years before her admission, for which immunomodulating therapy with thymoglobulin, tacrolimus, mofetil-microphenolate and prednisone was established. The patient suffered a pneumonic process with cough, expectoration, and dyspnoea. The computed tomography scan showed a cavitation in the right upper lobe. With all these findings an invasive broncopulmonary aspergillosis was suspected and the patient began an antifungal treatment with voriconazole without improvement. Rhizomucor pusillus was isolated from a clinical specimen obtained by fine needle aspiration, and its identification was confirmed by PCR. After this finding amphotericin B was administered, but the patient had an uncontrolled haemoptysis and died. CONCLUSIONS: Pulmonary mucormycosis is a rare infection, usually fatal in kidney transplant recipients with anti-rejection therapy. Mucorales species usually produce thrombotic phenomena, associated with necrosis and parenchymal destruction that caused a fatal uncontrolled haemoptysis in our patient. Early diagnosis is important in order to perform any surgical treatment and to administer amphotericin B.