Spontaneous Resolution Of Invasive Cerebral Aspergillosis Following Partial Resection In A Medically Untreated Infant
Thakar, S., Chickabasaviah, Y. T., & Hegde, A. S. (2012). spontaneous resolution of invasive cerebral aspergillosis following partial resection in a medically untreated infant. Journal of neurosurgery. Pediatrics, 10(1), 71–74. https://doi.org/10.3171/2012.4.PEDS1275
View Original Source →Abstract
Invasive craniocerebral aspergillosis, often encountered in an immunocompromised setting, is almost uniformly fatal despite radical surgical and medical management, and is frequently a necropsy finding. The authors report a unique, self-resolving clinical course of this aggressive infection in a 10-month-old infant. The infant was brought to the emergency services in altered sensorium with a 1-week history of left-sided hemiparesis, excessive irritability, and vomiting. An MRI study of the brain revealed multiple, heterogeneously enhancing lesions in the right cerebral hemisphere with mass effect. The largest lesion in the frontotemporal cortical and subcortical regions was decompressed on an emergent basis. Histopathological findings were suggestive of invasive aspergillosis, although there was no evidence of the infection in the lungs or paranasal sinuses. Computed tomography-guided aspiration of the remaining lesions and follow-up antifungal therapy were recommended. The parents, however, requested discharge without further treatment. The child was seen at a follow-up visit 3 years later without having received any antifungal treatment. Imaging showed resolution of the infection and features of Dyke-Davidoff-Masson syndrome (cerebral hemiatrophy). This report of invasive cerebral aspergillosis resolving without medical therapy is the first of its kind. Its clinicoradiological aspects are discussed in light of previously reported cases.
Case Details
Disease Location
Brain
Personal Characteristics
10- months-old male infant was brought to our institution’s emergency service during the winter season with a 1-week history of progressive left-sided hemiparesis, excessive irritability, and vomiting
Clinical Characteristics
Appeared well nourished and had no signs of any nutritional deficiency vital signs were stable and the findings of a general examination were normal. Glasgow coma scale score was 12/15 (e3, m5, v4) positive findings included papilledema and a partial right third cranial nerve paresis. Examination of the remaining cranial nerves showed no other abnormality an MRI study of the brain done at the onset of symptoms showed multiple, heterogeneously contrast enhancing lesions in the right cerebral hemisphere. The largest lesion measured 5 × 6.5 × 7 cm and spanned the cortical and subcritical areas in the right frontotemporal region there were 2 smaller lesions superior to the large one: a 2.5 × 3 × 3–cm lesion in the frontal region and a 2 × 2 × 2.5–cm lesion in the parietal parasagittal region in view of the clinical findings and the mass effect caused by the large lesion, an emergent right frontotemporal craniotomy and decompression procedure was performed after initiation of appropriate an_x0002_tiedema measures. At surgery, the lesion was found to contain frank pus and necrotic brain within a cyst cavity lined by a wall of variable thickness histopathological examination revealed the wall of a chronic abscess with necrotic material in the lumen. The vessels at the periphery had dense perivascular lym_x0002_phocytic inflammation, and some were thrombosed. Colonies of septemberate and acutely branching fungal hyphae were seen invading and migrating through the vessel walls, rupturing the internal elastic lamina and spilling into the brain parenchyma. Special staining with gms and pas stains highlighted the numerous fungal hyphae. The hyphae were not pigmented. The findings were consistent with the diagnosis of invasive aspergillosis. Fungal cultures showed no signs of growth
Remission Characteristics
The child’s sensorium improved to normal in the immediate postoperative period the results of workup for a primary source of aspergillosis, including imaging of the paranasal sinuses and chest, were normal the child was seen 3 years later at a routine follow-up visit. There was no history of seizures or of any other neurological symptoms. His development had been normal. He had not received any antifungal medications after he was discharged from the hospital. Neurological examination revealed a mild left-sided hemiparesis (grade 4 strength). The rest of his examination was normal
Treatment & Mechanisms
Proposed Remission Mechanisms
An extraordinarily strong immune response
Clinical Treatment
Frontotemporal craniotomy, decompression procedure