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Deep medullary vein involvement in neonatal cerebral venous thrombosis: the Padua experience in the last 20 years
Get PDFopenIntroduzione: La trombosi venosa cerebrale è un disturbo cerebrovascolare riconosciuto come causa di ictus perinatale nel 20% dei casi. La sua incidenza varia da 0,67 a 1-12 per 100.000 neonati, mentre per quanto riguarda le trombosi delle vene midollari non può essere stimata con precisione.Inoltre, a causa della presenza di pazienti asintomatici o paucisintomatici, si tratta di una patologia spesso sottodiagnosticata. L’eziologia il più delle volte ha un’origine multifattoriale.
Scopo dello studio: lo scopo di questo studio è stato quello di analizzare la casistica della trombosi venosa cerebrale in epoca neonatale, con particolare attenzione al coinvolgimento delle vene midollari profonde. In particolare, sono state definite le caratteristiche epidemiologiche, eziopatogenetiche, neuroradiologiche, terapeutiche e di disabilità a breve termine, successivamente confrontate tra i sottogruppi della nostra popolazione e con i dati presenti in letteratura.
Materiali e Metodi: La raccolta dei dati è stata condotta utilizzando il R.I.T.I. (Registro Italiano Trombosi Infantili). L’estrazione e l’analisi dei dati hanno riguardato i pazienti con trombosi venosa cerebrale con e senza coinvolgimento delle vene midollari, seguiti presso il Dipartimento di Salute della Donna e del Bambino nel periodo che va dal gennaio 2002 ad aprile 2023. I pazienti sono stati suddivisi in sottogruppi.Dapprima è stata eseguita un’analisi descrittiva dei dati, quindi, sono state analizzate e confrontate le variabili categoriche mediante test χ2di Pearson o test di esatto di Fisher, le variabili continue con il test di Wilcoxon-Kruskal-Wallis tra i sottogruppi. È stato inoltre eseguito un confronto dal punto di vista descrittivo dei soli casi di “trombosi delle vene midollari isolate” con la letteratura.
Risultati: Sono stati individuati 42 pazienti con trombosi venosa cerebrale. Di questi, 27/42 (64%) hanno presentato un episodio di trombosi venosa cerebrale senza coinvolgimento delle vene midollari (trombosi venosa cerebrale senza DMVT), mentre i restanti 15/42 (36%) hanno presentato trombosi delle vene midollari: in 9/42 (21%) la trombosi delle vene midollari profonde era isolata (DMVT isolata), in 6/42 (14%) l’evento “DMVT” era associato a un’altra trombosi venosa cerebrale. L’evento si presenta dal punto di vista clinico intorno alla prima settimana di vita (mediana 8 giorni, IQR, 4-14) con una prevalenza del sesso maschile nel 59%. I principali fattori di rischio risultano essere il parto complicato nel 38% dei casi, la prematurità nel 43 % di tutti i pazienti con DMVT, la presenza di cardiopatie nel 48% del totale e nel 53% dei pazienti con DMVT e le infezioni nel 40% dei casi con DMVT. Il principale sintomo d’esordio sono le crisi epilettiche nel 52% dei casi totali, seguite dal sopore nel 36%. Alla RM cerebrale eseguita nel 93% del totale sono state identificate lesioni cerebrali nell’86% dei pazienti con DMVT e nel 77% era presente una componente emorragica rispetto ai pazienti senza coinvolgimento delle DMV (p=0,013). Il trattamento antitrombotico è stato somministrato nel 30% del totale e il ricovero in TIN si è reso necessario nell’87% dei pazienti con DMVT. Almeno un deficit neurologico è stato riscontrato nel 48% dei casi alle dimissioni. L’ultimo follow up ha una mediana di 1,5 anni nei pazienti con DMVT.
Conclusioni: l’analisi della casistica padovana della “trombosi delle vene midollari” ha consentito di identificare elementi di sovrapposizioni in termini di anamnesi, fattori di rischio e presentazione clinica rispetto all’evento “trombosi venosa cerebrale”. Ciò ha permesso di sottolineare come la trombosi/ingorgo delle vene midollari profonde anche in assenza di trombosi in altre sedi venose intracerebrali sia un’entità clinico patologica di consistente impatto clinico sia in fase acuta che in termini di disabilità permanente.Background: the cerebral venous thrombosis (CVT) is a cerebrovascular disorder recognised as a cause of perinatal stroke in 20% of cases. The incidence of cerebral venous thrombosis rises from 0,67 to 1-12 per 100,000 newborns, while the incidence of “thrombosis of the deep medullary veins” (DMVT) cannot be accurately estimated despite growing interest in recent years. Moreover, due to the presence of asymptomatic patients or those with non-specific clinical manifestations, the disease is often under-diagnosed. The aetiology is often multifactorial.
Aim of the study: The aim of this study was to analyse the case history of “cerebral venous thrombosis” in the neonatal period, with a focus on the involvement of the deep medullary veins. In particular, the epidemiological, etiopathogenetic, neuroradiological, therapeutic and short-term disability characteristics were defined and compared between our population subgroups and with the literature about “isolated DMVT”.
Materials and methods: Data collection was conducted using R.I.T.I., a registry aimed at collecting data on patients with episodes of cerebral and systemic thromboembolism, both arterial or venous, in paediatric, neonatal or presumed perinatal age. Data extraction and analysis covered cases of cerebral venous thrombosis with and without medullary vein involvement, collected at the Dipartimento di Salute della Donna e del Bambino in Padua from January 2002 to April 2023. The patients were divided into subgroups: “all subjects with medullary vein thrombosis (DMVT)”, subjects with “isolated DMVT', subjects with “DMVT with involvement of other intracerebral venous sites” and subjects with “cerebral venous thrombosis without MVT'.
First, a descriptive analysis of the data was performed, then, categorical variables were analysed and compared by means of Pearson's χ2 test or Fisher's exact test, and continuous variables by means of the Wilcoxon-Kruskal-Wallis test between subgroups. A descriptive comparison of only the cases with “isolated DMVT” with the relevant literature was also performed.
Results: 42 patients with cerebral venous thrombosis were identified. 27/42 (64%) presented with “cerebral venous thrombosis without DMVT”, while the remaining 15/42 (36%) presented with “medullary vein thrombosis”: in 9/42 (21%) the “deep medullary vein thrombosis was isolated2, in 6/42 (14%) the “deep medullary vein thrombosis event was associated with the at least another cerebral venous thrombosis”. In all cases the event showed up clinically around the first week of life (median 8 days, IQR, 4-14) with a male prevalence in 59%. The most common risk factors were complicated delivery in 38% of cases, prematurity in 43% of all DMVT patients, heart diseases in 48% of all and 53% of DMVT patients, and infections in 40% of DMVT cases. The main onset symptom was seizures in 52% of all cases, followed by drowsiness in 36%. Brain MRI has been performed in 93% of the total showed brain lesions in 86% of patients with DMVT and a haemorrhagic component was present in 77% compared to patients without DMVT involvement (p=0.013). MRA has been conducted in 71% of cases. Antithrombotic treatment was administered in 30% of the total and admission to the NICU was necessary in 87% of patients with DMVT. At least one neurological deficit was found in 48% of cases at discharge. Follow-up has a median of 1.5 years in DMVT patients.
Conclusions: the analysis of the Paduan case history of DMVT over the last 20 years allowed the identification of overlapping elements (personal history, risk factors and clinical presentation) with “cerebral venous thrombosis” and makes it possible to emphatize how thrombosis/engorgement of the deep medullary veins even in the absence of thrombosis in other intracerebral venous sites represents a pathological entity with a clinical impact both in the acute phase and in terms of permanent disability
Update on the Italian cohort of patients with neonatal arterial ischemic stroke collected in the Italian Registry of Infantile Thrombosis (R.I.T.I.)
No full textopenIntroduzione. Lo stroke ischemico arterioso neonatale (NAIS) è uno evento acuto che si presenta nei primi 28 giorni di vita, confermato dalla presenza al neuroimaging di una recente area focale ischemica. Ha un’incidenza di 1:2500/1:4000 nati vivi e presenta un’eziopatogenesi multifattoriale, con fattori di rischio materni/placentari e fetali/neonatali. Le manifestazioni più comuni all’esordio sono convulsioni, alterazioni dello stato mentale e del tono muscolare, con esiti neurologici permanenti in una proporzione significativa, tra cui epilessia, disabilità motorie o cognitive.
Scopo dello studio. Obiettivi dello studio sono descrivere i casi di NAIS in Italia utilizzando i dati dal Registro Italiano Trombosi Infantile (R.I.T.I.) e identificare i fattori associati a outcome neurologico e rischio di crisi epilettiche al follow-up.
Materiali e metodi. Lo studio è di tipo osservazionale, longitudinale retrospettivo, non sponsorizzato, no-profit. Dal R.I.T.I. sono stati estratti i casi di NAIS, ed è stata effettuata un’analisi descrittiva della popolazione. I dati sono stati elaborati in variabili categoriche e continue, e confrontati con la letteratura pertinente. Per la valutazione dei fattori di rischio associati ad outcome al follow-up sono stati utilizzati il test di Wilcoxon, di chi-quadrato e di Fisher. Per valutare l'associazione tra covariate ed esito di interesse è stato utilizzato un modello di regressione logistica univariata.
Risultati. Sono stati individuati 181 pazienti con NAIS (56,2% maschi). Tra i fattori di rischio materni si segnalano: tampone vaginale positivo nel 29,3%, rottura prematura delle membrane nel 14%, infezioni materne in gravidanza nel 16,1% e peripartum nel 10,1%, altre patologie in gravidanza nel 24,8% e disordini placentari nel 7,9%. Tra i fattori di rischio fetali e neonatali sono stati individuati: parto cesareo nel 50%, indice Apgar al 5° minuto < 7 nel 10,6%, alterazioni specifiche dell’epoca neonatale nel 46,5% dei casi (60/129, di cui rianimazione alla nascita in 35/60 e necessità di ventilazione assistita in 25/60), positività allo screening trombofilico nel 46,4%, disordini cardiaci nel 32,4% ed infezioni nel 18,2%. Nell’87,6% lo stroke ha avuto un esordio sintomatico, con crisi epilettiche nel 79,4%. Nell’85,5% si è ricorsi al ricovero in terapia intensiva. La RM cerebrale è stata eseguita nel 93,6%, l’ecografia transfontanellare nel 77% e l’EEG nel 94,7%. Nel 62% la lesione si è presentata a sinistra e il vaso più coinvolto è stata l’arteria cerebrale media sinistra (56,2%). Nel 67% è stato descritto un infarto singolo. Terapie antitrombotiche sono state somministrate nel 16%. Il ricovero in media è durato 25 giorni. Un follow-up a distanza è disponibile per il 69,1% dei casi, con durata media di 35 mesi. Deficit neurologici e crisi epilettiche all’ultimo follow-up sono stati segnalati rispettivamente nel 38,8%, e nel 12%. In 1 paziente si è verificata recidiva di evento trombotico durante il ricovero (non in sede cerebrale), e in 2 al follow-up. L'indagine sulle variabili associate a deficit neurologici al follow-up ha evidenziato come significative: parto cesareo d'urgenza, età materna ≥32 anni, minor età gestazionale, presenza di deficit neurologi alla dimissione e presenza di crisi epilettiche al follow-up. Per lo sviluppo di crisi epilettiche al follow-up sono risultate significative: necessità di ventilazione assistita, lesioni del tronco encefalico e ricovero ≥19 giorni.
Conclusioni Abbiamo descritto le caratteristiche eziologiche, cliniche, radiologiche e di outcome della popolazione del R.I.T.I con NAIS e alcuni fattori di rischio associati a peggior outcome a lungo termine. Il Registro R.I.T.I., grazie ad un’ampia popolazione e una vasta raccolta di dati, rappresenta un'importante risorsa per possibili futuri progetti volti ad individuare strategie preventive e di diagnosi per le trombosi infantili.Introduction. Neonatal Arterial Ischemic Stroke (NAIS) is an acute event that occurs within the first 28 days of life and is confirmed by the presence of a recent focal ischemic area in neuroimaging. It has an incidence of 1:2500/1:4000 live births and has a multifactorial etiopathogenesis involving maternal/placental and fetal/neonatal risk factors. Common clinical manifestations include seizures, alterations in mental state and muscle tone, and a significant proportion of patients has permanent neurological sequelae, such as epilepsy or motor and cognitive disabilities.
Aim of the study. The aims of this study are to describe NAIS cases in Italy using data from the Italian Infant Thrombosis Registry (R.I.T.I.) and identify factors that influence neurological outcomes and the risk of epileptic seizures in the follow-up period.
Materials and Methods The study is observational, retrospective, non-sponsored, nonprofit. NAIS cases were extracted from the R.I.T.I., and a descriptive analysis of the population was performed. Data were categorized and analyzed as categorical and continuous variables and compared with relevant literature. To evaluate risk factors associated with outcomes in the follow-up, statistical tests such as the Wilcoxon test, chi-square, and Fisher's test were used. Univariate logistic regression models were employed to assess the association between covariates and the outcome of interest.
Results A total of 181 NAIS patients were identified (56.2% males). Maternal risk factors include: positive vaginal swab tests in 29.3%, premature rupture of membranes in 14%, maternal infections during pregnancy in 16.1%, and peripartum infections in 10.1%; other pregnancy-related pathologies were present in 24.8%, and placental disorders in 7.9%. Foetal and neonatal risk factors include: cesarean section delivery in 50%, and Apgar score < 7 at the 5th minute in 10.6%, specific neonatal period alterations were found in 46.5% of cases (60/129), including resuscitation at birth in 35/60 and the need for assisted ventilation in 25/60 cases; additionally, 46.4% had positive thrombophilia screening, 32.4% had cardiac disorders, and 18.2% had infections. In 87.6% of cases, onset was symptomatic, with epileptic seizures in 79.4%. 85.5% required admission to the intensive care unit. Brain MRI was performed in 93.6% of cases, transfontanelle ultrasound in 77%, and EEG in 94.7%. In 62% of cases, the lesion was located on the left side, with the left middle cerebral artery being the most affected (56.2%). A single infarct was described in 67% of cases, and antithrombotic therapies were given in 16% of cases. The average hospitalization was 25 days long. Follow-up data were available for 69.1% of patients, with an average length of 35 months. Neurological deficits were reported in 38.8% of cases, and 12% experienced epileptic seizures during follow-up. One stroke recurrence occurred during hospitalization, and two more were observed during follow-up. The analysis of variables associated with neurological deficits in the follow-up revealed as significant factors: emergency c-section, maternal age ≥32 years, lower gestational age, presence of neurological deficits at discharge, and development of epileptic seizures during follow-up. Significant factors for the development of epileptic seizures during follow-up included: need for assisted ventilation, lesions in the brainstem and hospitalization for ≥19 days.
Conclusions This study described the etiological, clinical, radiological, and outcome characteristics of the NAIS population in the R.I.T.I. and identified risk factors associated with poorer long-term outcomes. It also highlighted that the R.I.T.I., with its extensive population and data collection, represents an important resource for potential future projects aimed at identifying strategies for preventing and diagnosing infantile thrombosis
Neonatal Arterial Ischemic Stroke Secondary to Carotid Artery Dissection: A Case Report and Systematic Literature Review
No full textBackground: Carotid artery (CA) dissection is a rare etiology of neonatal arterial ischemic stroke (NAIS).
Methods: We describe one novel case and conduct a systematic literature review on NAIS attributed to CA dissection, to collect data on its clinical-radiological presentation, treatment, and outcome.
Results: Eight published cases of NAIS attributed to CA dissection were identified and analyzed with our case. All patients (nine of nine) were born at term, and eight of nine experienced instrumental/traumatic delivery or urgent Caesarean section. None had fetal problems during pregnancy or thrombophilia. Signs and symptoms at presentation (between days of life 0 and 6) included seizures (eight of nine), respiratory distress or irregular breathing (five of nine), hyporeactivity, decreased consciousness or irritability (four of nine), and focal neurological signs (two of nine). At magnetic resonance imaging (MRI), stroke was unilateral in seven of nine and extensive in five of nine. CA dissection was documented by neuroimaging or at postmortem studies (seven of nine), and hypothesized by the treating physicians based on delivery and neuroradiology characteristics (in the remaining two of nine). Antithrombotic treatment was used in two of nine. According to available follow-up, one of eight died at age seven days, seven of eight had neurological/epileptic sequelae, and CA recanalization occurred in three of four.
Conclusions: NAIS attributed to CA dissection is rarely identified in the literature, often preceded by traumatic/instrumental delivery, presenting with seizures and systemic signs/symptoms, and often characterized by extensive MRI lesions and neurological sequelae. Definite evidence and recommendations on antithrombotic treatment are lacking
Going Beyond Counting First Authors in Author Co-citation Analysis
Get PDFThe present study examines one of the fundamental aspects of author co-citation analysis (ACA) - the way co-citation
counts are defined. Co-citation counting provides the data on which all subsequent statistical analyses and mappings
are based, and we compare ACA results based on two different types of co-citation counting - the traditional type that
only counts the first one among a cited work's authors on the one hand and a non-traditional type that takes into
account the first 5 authors of a cited work on the other hand. Results indicate that the picture produced through this non-traditional author co-citation counting contains more coherent author groups and is therefore considerably clearer. However, this picture represents fewer specialties in the research field being studied than that produced through the traditional first-author co-citation counting when the same number of top-ranked authors is selected and analyzed. Reasons for these effects are discussed
Variations on the Author
Get PDF“Variations on the Author” discusses two of Eduardo Coutinho’s recent films (Um Dia na Vida, from 2010, and Últimas Conversas, posthumously released in 2015) and their contribution to the general question of documentary authorship. The director’s filmography is characterized by a consistent yet self-effacing form of authorial self-inscription: Coutinho often features as an interviewer that rather than express opinions propels discourses; an interviewer that is good at listening. This mode of self-inscription characterizes him as an author who is not expressive but who is nonetheless markedly present on the screen. In Um Dia na Vida, however, Coutinho is completely absent form the image, while Últimas Conversas, on the contrary, includes a confessional prologue that moves the director from the margins to the center of his films. This article examines the ways in which these works stand out in the filmography of a director who offers new insights into the notion of cinematic authorship
Appropriate Similarity Measures for Author Cocitation Analysis
Get PDFWe provide a number of new insights into the methodological discussion about author cocitation analysis. We first argue that the use of the Pearson correlation for measuring the similarity between authors’ cocitation profiles is not very satisfactory. We then discuss what kind of similarity measures may be used as an alternative to the Pearson correlation. We consider three similarity measures in particular. One is the well-known cosine. The other two similarity measures have not been used before in the bibliometric literature. Finally, we show by means of an example that our findings have a high practical relevance.information science;Pearson correlation;cosine;similarity measure;author cocitation analysis
Dispelling the Myths Behind First-author Citation Counts
Get PDFWe conducted a full-scale evaluative citation analysis study of scholars in the XML research field to explore just how different from each other author rankings resulting from different citation counting methods actually are, and to demonstrate the capability of emerging data and tools on the Web in supporting more realistic citation counting methods. Our results contest some common arguments for the continued
use of first-author citation counts in the evaluation of scholars, such as high correlations between author rankings by first-author citation counts and other citation
counting methods, and high costs of using more realistic citation counting methods that are not well-supported by the ISI databases. It is argued that increasingly available digital full text research papers make it possible for citation analysis studies to go beyond what the ISI databases have directly supported and to employ more
sophisticated methods
A systematic review of surgical and interventional radiology procedures for pediatric idiopathic intracranial hypertension
Get PDFBackgroundIdiopathic intracranial hypertension (IIH) is defined as elevated intracranial pressure and consequent symptoms (mainly headache and visual deterioration) occurring in the absence of secondary causes. Surgical and interventional radiology procedures should be considered for refractory IIH and mainly include cerebrospinal fluid (CSF) diversion techniques, optic nerve sheath fenestration (ONSF), and venous sinus stenting (VSS). Our study aims to review the current literature on the application of these techniques in clinical practice.MethodsA systematic literature review on the surgical and interventional radiology treatment of IIH was conducted, focusing on ONSF, VSS, and CSF diversion techniques. According to PRISMA guidelines, all reports published in PubMed in the last 30 years (1993–2023) were considered, and among 722 papers, 48 were included in the present study, resulting in a total study population of 454 children or adolescents (11 months–17 years old).ResultsAmong 454 patients, 193 underwent an invasive approach, divided into CSF diversion (115/193), ONSF (65/193), VSS (11/193), cranial subtemporal decompression (8/193), and internal cranial expansion (9/193). Sixteen of the 193 patients (8%) required reintervention due to relapsing symptoms or surgical complications, particularly those who underwent CSF diversion. Furthermore, 9/115 required shunt revision due to shunt obstruction or malfunction. We extracted data on the outcome of each procedure: of the 193 patients, 71 experienced a positive outcome with symptom resolution or improvement, while 27 demonstrated a negative outcome.Discussion and conclusionsSevere and refractory cases of IIH are eligible for invasive treatments. CSF diversion is the most frequently used technique, despite its high failure risk and need for reintervention. ONSF has shown good results in terms of outcome and safety, particularly in children with visual symptoms. VSS is the most recent approach, indicated in children with stenosis of the venous sinus. In our study population, VSS demonstrated good results in terms of symptom resolution and need for reintervention, but its use remains limited to a few centers.Systematic Review Registrationhttps://www.crd.york.ac.uk/, PROSPERO (CRD42024504244)
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