Primary cerebral cystic echinococcosis in a child from Roman countryside: Source attribution and scoping review of cases from the literature.

BACKGROUND: Human cystic echinococcosis (CE) is a zoonotic parasitic infection caused by the larval stage of the species belonging to the Echinococcus granulosus sensu lato (s.l.) complex. Parasitic cysts causing human CE are mainly localized in the liver and in the lungs. In a smaller number of cases, larvae may establish in any organ or tissue, including the central nervous system (CNS). Cerebral CE (CCE) is rare but poses serious clinical challenges. METHODS: This study presents a case of CCE in a child living in the countryside near Rome (Italy), along with a comparative molecular analysis of the isolated cyst specimens from the patient and sheep of local farms. We also systematically searched the literature to summarize the most relevant epidemiological and clinical aspects of this uncommon localization. FINDINGS: The comparative molecular analysis confirmed that the infection was caused by E. granulosus sensu stricto (s.s.) (G3 genotype), and most likely acquired in the family farm. The literature search identified 2,238 cases of CCE. In 80.51% of cases, brain was the only localization and single CCE cysts were present in 84.07% of cases. Mean patients' age was 20 years and 70.46% were children. Cyst rupture was reported in 12.96% and recurrence of CCE after treatment in 9.61% of cases. Permanent disability was reported in 7.86% of cases, while death occurred in 6.21%. In case series reporting all CE localization, CCE represented 1.5% of all CE cases. In the few reports that identified at molecular level the CCE cyst, E. granulosus s.s. was found in 40% and E. canadensis in 60% of cases. CONCLUSIONS: We report a rare case of CCE and evidenced the probable local origin of infection. The proportions of CE cases with uncommon localizations and with high impact on patients' lives have been globally neglected and should be included in the computation of the global burden of CE.

A clinical case report of Balamuthia granulomatous amoebic encephalitis in a non-immunocompromised patient and literature review.

BACKGROUND: Balamuthia granulomatous amoebic encephalitis (GAE) is a peculiar parasitic infectious disease of the central nervous system, about 39% of the infected Balamuthia GAE patients were found to be immunocompromised and is extremely rare clinically. The presence of trophozoites in diseased tissue is an important basis for pathological diagnosis of GAE. Balamuthia GAE is a rare and highly fatal infection for which there is no effective treatment plan in clinical practice. CASE PRESENTATION: This paper reports clinical data from a patient with Balamuthia GAE to improve physician understanding of the disease and diagnostic accuracy of imaging and reduce misdiagnosis. A 61-year-old male poultry farmer presented with moderate swelling pain in the right frontoparietal region without obvious inducement three weeks ago. Head computed tomography(CT) and magnetic resonance imaging(MRI) revealed a space-occupying lesion in the right frontal lobe. Intially clinical imaging diagnosed it as a high-grade astrocytoma. The pathological diagnosis of the lesion was inflammatory granulomatous lesions with extensive necrosis, suggesting amoeba infection. The pathogen detected by metagenomic next-generation sequencing (mNGS) is Balamuthia mandrillaris, the final pathological diagnosis was Balamuthia GAE. CONCLUSION: When a head MRI shows irregular or annular enhancement, clinicians should not blindly diagnose common diseases such as brain tumors. Although Balamuthia GAE accounts for only a small proportion of intracranial infections, it should be considered in the differential diagnosis.

Higher Concentration of Taenia Antigens in the CSF is Related to Slight Ventricle Enlargement and Periventricular Neuronal Decrease in Young Rats

Purpose Experimental models might help understand the pathophysiology of neurocysticercosis-associated hydrocephalus. The present study aimed to compare the extent of hydrocephalus and tissue damage in rats with subarachnoid inoculation of different concentrations of Taenia crassiceps cyst proteins. Methods Sixty young rats were divided into two groups: low- and high-concentration groups. The animals in the low concentration group received 0.02ml of 2.4mg/ml T. crassiceps cyst proteins while those in the high concentration group received 0.02 ml of 11.6mg/ml T. crassiceps cyst proteins. The animals underwent magnetic resonance imaging at 1, 3, and 6 months postinoculation to assess the ventricle volume. Morphological assessment was performed at the end of the observation period. Results Repeated measures of ventricle volumes at 1, 3, and 6 months showed progressive enlargement of the ventricles. At 1 and 3 months, we observed no differences in ventricle volumes between the 2 groups. However, at 6 months, the ventricles were larger in the high concentration group (median » 3.86mm3, range: 2.37­12.68) compared with the low concentration group (median » 2.00mm3, range: 0.37­11.57), p » 0.003. The morphological assessment revealed a few inflammatory features in both groups. However, the density of oligodendrocytes and neurons within the periventricular region was lower in the high concentration group (5.18 versus 9.72 for oligodendrocytes and 15.69 versus 21.00 for neurons; p < 0.001 for both). Conclusion Our results suggest that, in rats, a higher concentration of T. crassiceps cyst proteins in the subarachnoid space could induce ventricle enlargement and reduce the number of neurons within the periventricular area.

Cerebrospinal Fluid Analysis.

Cerebrospinal fluid (CSF) analysis is a diagnostic tool for many conditions affecting the central nervous system. Urgent indications for lumbar puncture include suspected central nervous system infection or subarachnoid hemorrhage. CSF analysis is not necessarily diagnostic but can be useful in the evaluation of other neurologic conditions, such as spontaneous intracranial hypotension, idiopathic intracranial hypertension, multiple sclerosis, Guillain-Barré syndrome, and malignancy. Bacterial meningitis has a high mortality rate and characteristic effects on CSF white blood cell counts, CSF protein levels, and the CSF:serum glucose ratio. CSF culture can identify causative organisms and antibiotic sensitivities. Viral meningitis can present similarly to bacterial meningitis but usually has a low mortality rate. Adjunctive tests such as CSF lactate measurement, latex agglutination, and polymerase chain reaction testing can help differentiate between bacterial and viral causes of meningitis. Immunocompromised patients may have meningitis caused by tuberculosis, neurosyphilis, or fungal or parasitic infections. Subarachnoid hemorrhage has a high mortality rate, and rapid diagnosis is key to improve outcomes. Computed tomography of the head is nearly 100% sensitive for subarachnoid hemorrhage in the first six hours after symptom onset, but CSF analysis may be required if there is a delay in presentation or if imaging findings are equivocal. Xanthochromia and an elevated red blood cell count are characteristic CSF findings in patients with subarachnoid hemorrhage. Leptomeningeal carcinomatosis can mimic central nervous system infection. It has a poor prognosis, and large-volume CSF cytology is diagnostic.

Case report: multiple and atypical amoebic cerebral abscesses resistant to treatment.

BACKGROUND: The parasite Entamoeba histolytica is the causal agent of amoebiasis, a worldwide emerging disease. Amebic brain abscess is a form of invasive amebiasis that is both rare and frequently lethal. This condition always begins with the infection of the colon by E. histolytica trophozoites, which subsequently travel through the bloodstream to extraintestinal tissues. CASE PRESENTATION: We report a case of a 71-year-old female who reported an altered state of consciousness, disorientation, sleepiness and memory loss. She had no history of hepatic or intestinal amoebiasis. A preliminary diagnosis of colloidal vesicular phase neurocysticercosis was made based on nuclear magnetic resonance imaging (NMRI). A postsurgery immunofluorescence study was positive for the 140 kDa fibronectin receptor of E. histolytica, although a serum analysis by ELISA was negative for IgG antibodies against this parasite. A specific E. histolytica 128 bp rRNA gene was identified by PCR in biopsy tissue. The final diagnosis was cerebral amoebiasis. The patient underwent neurosurgery to eliminate amoebic abscesses and was then given a regimen of metronidazole, ceftriaxone and dexamethasone for 4 weeks after the neurosurgery. However, a rapid decline in her condition led to death. CONCLUSIONS: The present case of an individual with a rare form of cerebral amoebiasis highlights the importance of performing immunofluorescence, NMRI and PCR if a patient has brain abscess and a poorly defined diagnosis. Moreover, the administration of corticosteroids to such patients can often lead to a rapid decline in their condition.

Molecular and Histologic Diagnosis of Central Nervous System Infections.

Infections of the central nervous system cause significant morbidity and mortality in immunocompetent and immunocompromised individuals. A wide variety of microorganisms can cause infections, including bacteria, mycobacteria, fungi, viruses, and parasites. Although less invasive testing is preferred, surgical biopsy may be necessary to collect diagnostic tissue. Histologic findings, including special stains and immunohistochemistry, can provide a morphologic diagnosis in many cases, which can be further classified by molecular testing. Correlation of molecular, culture, and other laboratory results with histologic findings is essential for an accurate diagnosis, and to minimize false positives from microbial contamination.

Pathogenesis of cerebral toxocariasis and neurodegenerative diseases.

Toxocara canis belongs to one of zoonotic parasites that commonly infects canines worldwide, and its eggs in host faeces may contaminate the food, water, soil and their fur as well as the larvae entrapped in the granuloma can infect paratenic hosts including mice and humans. Survivability of T. canis embryonated eggs under moist, cool conditions may be as long as 2-4 years or more. In paratenic hosts such as mice and humans, T. canis L3 larvae neither moult, grow, nor replicate and will wander through a number of internal organs in humans so as to cause Th2-dominant pathology in various internal organs as leading to neurotoxocariasis (NT), ocular toxocariasis (OT), or visceral larva migrans (VLM). Although the systemic immune response to T. canis has been widely reported, the immune response in the brain has received little attention. Differential cytokine expression and other brain injury-associated biomarkers or neurodegeneration-associated factors have been observed in infected versus uninfected outbred and inbred mice. Preliminary data have also suggested a possible link between significant memory impairment and cytokine production associated with T. canis infection in the hippocampus which has been long recognised as being responsible for learning and memory functions. Notably, it remains an enigma concerning cerebral invasion by T. canis larvae rarely induces a recognisable neurological syndrome or its involvement in neuropathological disorders in humans. Exploration of the relationship between host and parasite in the brain may elucidate the cryptic symptoms of human cerebral toxocariasis, with patients presenting with mental retardation, epilepsy, neurodegeneration and other central nervous system (CNS) disorders.

Nerve growth factor in the psychiatric brain.

The nerve growth factor (NGF) belongs to a family of proteins named neurotrophins, consisting of NGF, brain-derived neurotrophic factor (BDNF), neurotrophin-3 (NT-3), NT-4/5 and NT-6. NGF regulates a large number of physiological mechanisms that result in neurotrophic, metabotrophic and/or immunotrophic effects. Neurodegenerative diseases, including Alzheimer disease, psychiatric disorders (e.g. depression and schizophrenia) and brain parasitic infection have in common the effect of changing the brain levels of neurotrophins, in particular NGF. The contribution of both NGF and its receptor TrkA in such events and the recent promising results of NGF based therapies are here presented and discussed.

Parasitic Infections of the Nervous System.

Parasitic infections of the central nervous system are much more common than suspected, although most infections are asymptomatic. For example, parasites like the ubiquitous protozoa Toxoplasma gondii or the nematode larvae Toxocara canis infect significant proportions of the human population. Other parasitic infections such as malaria and neurocysticercosis are widespread in developing countries and become major causes of neurological morbidity in these regions as well in immigrants and travelers. This article reviews parasitic pathogens causing neurological morbidity and mortality, including an extensive list of less common parasitic infections of the human nervous system.

Simultaneous occurrence of two new myxosporean species infecting the central nervous system of Hypopygus lepturus from Brazil.

This paper describes 2 new myxosporean species, Henneguya lepturus sp. nov. and Thelohanellus lepturus sp. nov., simultaneously infecting the brain and spinal cord of Hypopygus lepturus Hoedeman, 1962 (Teleostei, Hypopomidae) from the Brazilian Amazon (Roraima State). Several spherical cysts of varying dimensions (up to 135 µm) were microscopically observed. The myxospores of H. lepturus sp. nov. measured 25.8 µm in total length, having an ellipsoidal body (12.4 × 6.4 × 2.2 µm) and 2 equal tapering tails (13.4 µm in length). Each of the 2 pyriform polar capsules measured 4.4 × 1.6 µm and possessed a polar filament coiled in 8-9 turns. The myxospores of T. lepturus sp. nov. were pyriform, formed by 2 equal valves (17.7 × 9.1 × 4.3 µm) surrounding a single polar capsule (10.9 × 3.5 µm) that had a coiled polar filament with 13-16 turns and a binucleated sporoplasm that contained several circular sporoplasmosomes. Molecular analysis of the small subunit (SSU) rRNA gene sequences of these 2 species were in agreement with the taxonomic classification derived from the ultrastructure of the myxospores. Histopathology of the host tissue showed degradation of the myelinated axons surrounding the cysts of both species, with the hosts displaying behavioural changes and erratic movements when observed in an aquarium.

Efficacy comparison between long-term high-dose praziquantel and surgical therapy for cerebral sparganosis: A multicenter retrospective cohort study.

BACKGROUND: Sparganosis is a parasitic infection caused by the plerocercoid larvae of Spirometra mansoni in East and Southeast Asia. The plerocercoid larvae sometimes invade the encephalon, resulting in severe cerebral sparganosis. Surgical removal of the larvae is considered a standard therapy for cerebral sparganosis. In contrast, the efficacy and safety of long-term, high-dose praziquantel treatment for cerebral sparganosis have not been explored. METHODOLOGY/PRINCIPAL FINDINGS: In this multicenter retrospective study, we assessed the records of 96 patients with cerebral sparganosis who consulted at three medical centers from 2013 to 2017. Forty-two patients underwent surgical lesion removal, and the other 54 patients received long-term, high-dose praziquantel (50 mg/kg/day for 10 days, repeated at monthly intervals). The primary outcome was the complete disappearance of active lesions on cerebral magnetic resonance imaging. The secondary outcomes included the modified Rankin scale score at 90 days, incidence of seizure, eosinophil count, and serological Spirometra. mansoni antibody titer. The efficacy of praziquantel treatment was similar to that of surgical lesion removal for cerebral sparganosis with respect to both the primary outcome and secondary outcomes. Although binary logistic regression models also supported the primary outcome after adjustment for age, sex, lesion location, and loss to follow-up, some unavoidable confounders might have biased the statistical power. No significant clinical complications or laboratory side effects occurred in the praziquantel group with the exception of a relatively benign allergic reaction. CONCLUSIONS/SIGNIFICANCE: In this small-sample, nonrandomized, retrospective exploratory study, some patients with cerebral sparganosis were responsive to long-term, high-dose praziquantel with an efficacy similar to that of surgical lesion removal. These findings increase the treatment flexibility for this serious infection.

Cerebral cystic echinococcosis in Mongolian children caused by Echinococcus canadensis.

Recent molecular re-evaluation of Echinococcus granulosus, which causes cystic echinococcosis (CE), has revealed that it is not a single species, but instead consists of 5 cryptic species. Among them, E. granulosus (dog-sheep strain) is predominant (75%) followed by Echinococcus canadensis (22%). The major affected organs, in humans, are the liver (88%) and lungs (11%). Primary cerebral CE comprises less than 1% of all cases. As cerebral CE cases are rare, there are few reports with molecular confirmation of the causative species. This study reports mitochondrial gene analysis from 4 Mongolian pediatric cerebral CE cases. Molecular confirmation was obtained for 3 of the 4 cases, with all 3 cases determined to be due to E. canadensis (G6/G7) infection. None of the cases had other organ involvement. This is only the third report on the molecular identification of the Echinococcus species responsible for cerebral CE, and only the second report of E. canadensis (G6/G7) being the causative agent of cerebral CE.

Takotsubo cardiomyopathy associated with Paragonimiasis westermani.

An 11-year-old boy collapsed during morning assembly at his junior high school. The automated external defibrillator detected ventricular fibrillation and provided shock delivery. He was successfully resuscitated and reverted to sinus rhythm. Electrocardiography showed ST-T elevation in the precordial leads. Echocardiography and angiography demonstrated akinesia of the apex and mid-wall of the left ventricle with preserved contraction of the basal segments, which suggested Takotsubo cardiomyopathy. The patient and his family had often eaten uncooked crab, and his father had a past history of infection with Paragonimiasis westermani. The patient had had a persistent cough and chest pain for several weeks. Chest radiograph showed cystic cavities in the left upper lung. Microbiological examination of the sputum demonstrated an egg of P. westermani and immunological assay showed a raised antibody titre to P. westermani. On the12th day of admission, he developed seizures, and magnetic resonance imaging demonstrated cerebral involvement. After the administration of praziquantel for 3 days, the clinical manifestations improved immediately, and echocardiography normalised within 3 weeks. The patient was discharged on the 32nd day + and follow-up was normal. Takotsubo cardiomyopathy following a potentially fatal arrhythmia is a rare cardiac complication associated with pulmonary and central nervous system infection by P. westermani.

Dumbbell-Shape Hydatid Cyst of Meckel Cave Extending to Cerebellopontine Angle and Middle Fossa; Surgical Technique and Outcome of Rare Case.

BACKGROUND: Cerebral hydatid cyst is among the rare presentations of echinococcosis. Almost all the reported cases of intracranial hydatid cysts are in the cortical regions (parietal lobe in the territory of the middle cerebral artery), and all have been removed surgically using the water jet dissection technique. However, in locations in which there are several neuronal structures, such as the cerebellopontine angle, this method might not be applicable. CASE DESCRIPTION: A 62-year-old lady presented to our outpatient clinic with left-sided facial deviation, inability to close the eyes, and diplopia since a month before. She was diagnosed with having a dumbbell-shape lesion in the Meckel cave extending to the left cerebellopontine angle and middle fossa just lateral to the cavernous sinus. As it was adherent to the surrounding structures, we evacuated the contents using a fine needle and then dissected the cyst totally. The patient's symptoms alleviated after the surgery, and there was no residue. After the 2-year follow-up, our patient does not have recurrence. CONCLUSIONS: The Meckel cave and cerebellopontine angle are rare locations for hydatid cysts, and the surgical technique is different due to adhesion of the cyst to several neuronal structures (lower cranial nerve, brainstem, and cerebellum). Needle aspiration along with dissection of the cyst microsurgically is recommended in similar cases.

Solitary Cysticercal Cyst Inside the Blake's Pouch Remnant of Mega Cisterna Magna with Associated Aqueductal Stenosis: Clinical and Management Implications.

BACKGROUND: Intraventricular and subarachnoid forms represent the relatively complex, albeit uncommon, manifestations of central nervous system involvement by cysticercal cysts. Cysticercal encystation inside the Blake's pouch remnant of mega cisterna magna (MCM) remains an extremely rare clinical scenario that, to the best of our knowledge, has not been reported previously. CASE PRESENTATION: A 12-year-old boy presented with acute worsening and subsequent spontaneous resolution of his compensated hydrocephalus. Neuroimaging revealed features consistent with a MCM with septum inside, indicating remnants of the embryologic fourth ventricular diverticulum, the Blake's pouch. It also revealed a free-floating intact cysticercal cyst inside the MCM limited by the septum with aqueductal stenosis. The spontaneous clinicoradiologic resolution of hydrocephalus raised the possibility of ball valve obstruction of the cerebrospinal fluid outflow, i.e., Bruns syndrome. We successfully treated this patient with endoscopic extraction of the viable cysticercal cyst through a suboccipital burrhole. CONCLUSIONS: The clinical scenario presented here has not been described previously. Endoscopic cyst removal in such a situation is an effective and low-risk procedure that obviates the further need for antihelminthic medications.

[Cerebral hydatid cyst: Clinical case and review of the literature]. / Quiste hidatídico cerebral: caso clínico y revisión de la literatura.

Hydatid disease is an accidental parasitosis, with brain location being rare. The case is reported of a 33year-old male, with no history of note, who was admitted to hospital with intracranial hypertension syndrome and right hemiparesis. Computed tomography showed a cystic lesion in the left frontal-parietal lobule. Surgery was performed by complete excision of the lesion, with a good outcome. Hydatid disease is a rare condition in the brain. Clinical suspicion is important for an early diagnosis. A review is presented on the pathogenesis, diagnosis and treatment of cerebral hydatid disease.

Hemiparesia súbita secundaria a cisticercosis cerebral: presentación de un caso clínico / Sudden hemiparesis secondary to cerebral cysticercosis: presentation of a clinical case

Introducción: La neurocisticercosis es una infección parasitaria del sistema nervioso central, ocasionada por la etapa larval del parásito Taenia solium. Su prevalencia mundial es de 9,1%. Se presenta a continuación un caso clínico en el que destaca una clínica infrecuente y tamaño de la lesión poco común; caso que fue resuelto con éxito mediante neurocirugía. Presentación del caso: Hombre de 48 años, residente en localidad rural, consultó por cuadro súbito de hemiparesia derecha de predominio crural, posteriormente con aumento de paresia braquial ipsilateral. Al examen físico destacó síndrome piramidal derecho. Se estudió con Tomografía Computarizada que evidenció quiste frontal izquierdo con compresión del área motora, sugerente de lesión parasitaria. Resonancia Nuclear Magnética de Cerebro (RNMC) informó proceso expansivo quístico fronto-parietal parasagital izquierdo de 5 x 5,5 cm, con efecto de masa. Por tamaño, tipo de lesión y clínica se decidió realizar cirugía abierta con extirpación total y biopsia que informó cisticercosis cerebral. El paciente evolucionó con recuperación total de su déficit motor posterior a la cirugía. Discusión: La neurocisticercosis se encuentra dentro de los diagnósticos diferenciales de lesiones quísticas encefálicas. Su clínica es inespecífica, manifestándose comúnmente con convulsiones y cefalea. La RNMC permite el diagnóstico y localización de las lesiones. El tratamiento es médico, quirúrgico o combinado. Generalmente, el manejo se basa en antiepilépticos y antiparasitarios sistémicos, sin embargo en este caso, se decidió el manejo quirúrgico que llevó a una mejoría total del paciente, lo que avala la cirugía precoz como principal medida en lesiones de este tipo

Introduction: Neurocysticercosis is a parasitic infection of the central nervous system, caused by the larval stage of the parasite Taenia solium. Its worldwide prevalence is 9.1%. We present below a clinical case with an infrequent clinical and uncommon lesion size, with surgical resolved. Case report: A 48 year old man, who was a resident of a rural locality, consulted for sudden hemiparesis on the right side of the crural predominance, subsequently with increased ipsilateral brachial paresis. Physical examination highlighted right pyramidal syndrome. It was studied with Computed Axial Tomography that showed left frontal cyst with compression of the motor area, suggestive of parasite lesion. Brain Nuclear Magnetic Resonance (BNMR) reported left parasagittal fronto-parietal cystic expansive process of 5 x 5.5 cm, with mass effect. By size, type of lesion and clinic it was decided to perform open surgery with total extirpation and biopsy that reported cerebral cysticercosis. The patient evolved with complete recovery of motor deficit after surgery. Discussion: Neurocysticercosis is a differential diagnosis of brain cystic lesions. Its clinic is non-specific, commonly manifesting with seizures and headache. The RNMC allows the diagnosis and location of the lesions. The treatment is medical, surgical or combined. Usually, the management is based on antiepileptics and systemic antiparasitic, however in this case, it was decided the surgical management that led to the total improvement of the patient, which guarantees the early surgery as the main measure in lesions of this type

Clinical evolution of cerebral coenurosis from invasive to chronic infection in sheep and a goat.

This survey describes the evolution in vivo of Coenurus cerebralis in small ruminants. At presentation, neurological signs and cerebrospinal fluid (CSF) features were suggestive of multifocal or diffuse inflammatory reaction. Magnetic resonance imaging (MRI) captured the transition between the invasive and quiescent phase of the infection, revealing the concurrent presence of meningitis and small cysts. During the quiescent phase, in all animals, neurological symptoms disappeared, and cerebrospinal fluid was unremarkable while cysts grew progressively. Subsequently, the onset of neurological symptoms coincided with MRI signs of diffuse or localized increase of intracranial pressure, as confirmed by direct intracranial pressure measuring. All the animals had an excellent post-surgical recovery. This is the first report describing the evolution of coenurosis in vivo. Sequential imaging allowed describing interesting such as the death of some coenuri and different parasite growth rate in the same host.