Lung destruction secondary to intrapulmonary migration of a ventriculoperitoneal shunt catheter: report of an unusual case and literature review.

Ventriculoperitoneal shunt placement for the treatment of hydrocephalus is one of the most common pediatric neurosurgical procedures. Complications, including infections, catheter obstruction, shunt breakdown, and hemorrhage, have been described in the literature. Occasionally, however, uncommon and devastating complications occur. We report a case of a 10-year-old female patient who at birth underwent surgical closure of lumbar myelomeningocele and placement of a CSF shunt at another center. Her neurosurgical follow-up was poor. She presented at our institution with a history of recurrent pneumonia. Control chest X-rays showed a right pulmonary infiltrate with lung retraction and mediastinal shift. Chest and brain CT scans confirmed the intrapulmonary location of the distal catheter tip and ventricular dilation. Surgical shunt revision was performed with removal of the intrapulmonary catheter and placement of a new intraperitoneal catheter. Subsequently, right pneumonectomy was performed with good postoperative recovery of the patient. Intrathoracic migration of the distal catheter of the CSF shunt is an extremely rare complication that may produce severe morbidity. To our knowledge, there have been no previous reports on extensive lung destruction secondary to intrathoracic and intrapulmonary ventriculoperitoneal shunt migration. In patients with CSF shunts and pulmonary symptoms, intrapulmonary catheter migration should be considered.

Melanotic neuroectodermal tumor of infancy arising from the skull: report of an unusual case, review of the literature, and a diagnostic approach.

INTRODUCTION: Melanotic neuroectodermal tumor of infancy is a rare neoplasm mainly seen in children under 1 year of life. The most common location of the tumor is the maxilla followed by the cranial vault. Surgery is the treatment of choice and outcome mainly depends on extent of resection. OBJECTIVES: To report an atypical case of an 8-year-old patient with a melanotic neuroectodermal tumor of infancy, to review the cases with melanotic neuroectodermal tumor of infancy arising from the skull published over the last 13 years, and to provide a diagnostic approach that may allow recognition of a pattern in these rare neoplastic lesions. METHODS: A case is reported with a description of the clinical, radiological, surgical, and histopathological features. Additionally, the literature was reviewed to identify reports of patients with melanotic neuroectodermal tumor of infancy arising from the cranial vault and all cases published in PubMed over the last 13 years were included. Only studies that evaluated clinical, radiological, surgical, and histopathological findings were included. CONCLUSION: Melanotic neuroectodermal tumor of infancy is a rare entity that may present with unusual features, but nevertheless has an identifiable pattern that allows the tumor to be considered in the differential diagnosis of intracranial space-occupying lesions in children.

New proteins configure a brain drug resistance map in tuberous sclerosis.

Epileptogenic cortical tubers, characterized by dysplastic neurons and balloon cells, is a frequent feature of tuberous sclerosis. In severe tuberous sclerosis-affected individuals, seizures are refractory to medication. Multidrug resistance proteins (multidrug resistance protein-1 [MDR-1] and multidrug resistance-associated protein-1 [MRP-1]) have been found to be highly expressed in epileptogenic cortical tubers. However, two new proteins related to refractoriness in cancer (breast cancer resistance protein and major vault protein) have not been investigated in tuberous sclerosis and refractory epilepsy. On the same brain specimens previously describing the MDR-1 and MRP-1 expression, we investigated retrospectively breast cancer resistance protein and major vault protein by specific monoclonal antibodies and routine immunohistochemistry methods. Breast cancer resistance protein was present in vascular endothelial cells from all the vessels examined in 3 of 3 cases. Major vault protein was detected in only one case, and selectively expressed in several but not all ballooned cells. In epileptogenic cortical tubers, the additional expression of breast cancer resistance protein on vessels and major vault protein in some ballooned cells to the previously demonstrated expression of MDR-1 and MRP-1 (in vessels, astroglia, microglia, neurons, and ballooned cells) configures a brain protein pharmacoresistance map from patients with tuberous sclerosis and refractory epilepsy.