Outcomes after hemispherectomy in adult patients with intractable epilepsy: institutional experience and systematic review of the literature

2018 ◽  
Vol 128 (3) ◽  
pp. 853-861 ◽  
Author(s):  
Courtney M. Schusse ◽  
Kris Smith ◽  
Cornelia Drees
Keyword(s):  
Neuropsychological Testing ◽  
Case Series ◽  
Intractable Epilepsy ◽  
Adult Patients ◽  
Morbidity Rate ◽  
Seizure Outcome ◽  
Seizure Freedom ◽  
Surgical Tool ◽  
Drug Resistant Epilepsy

OBJECTIVEHemispherectomy is a surgical technique that is established as a standard treatment in appropriately selected patients with drug-resistant epilepsy. It has proven to be successful in pediatric patients with unilateral hemispheric lesions but is underutilized in adults. This study retrospectively evaluated the clinical outcomes after hemispherectomy in adult patients with refractory epilepsy.METHODSThis study examined 6 cases of hemispherectomy in adult patients at Barrow Neurological Institute. In addition, all case series of hemispherectomy in adult patients were identified through a literature review using MEDLINE and PubMed. Case series of patients older than 18 years were included; reports of patients without clear follow-up duration or method of validated seizure outcome quantification were excluded. Seizure outcome was based on the Engel classification.RESULTSA total of 90 cases of adult hemispherectomy were identified, including 6 newly added by Barrow Neurological Institute. Sixty-five patients underwent functional hemispherectomy; 25 patients had anatomical hemispherectomy. Length of follow-up ranged from 9 to 456 months. Seizure freedom was achieved in 80% of patients. The overall morbidity rate was low, with 9 patients (10%) having new or additional postoperative speech or language dysfunction, and 19 patients (21%) reporting some worsening of hemiparesis. No patients lost ambulatory or significant functional ability, and 2 patients had objective ambulatory improvement. Among the 41 patients who underwent additional formal neuropsychological testing postoperatively, overall stability or improvement was seen.CONCLUSIONSHemispherectomy is a valuable surgical tool for properly selected adult patients with pre-existing hemiparesis and intractable epilepsy. In published cases, as well as in this series, the procedure has overall been well tolerated without significant morbidity, and the majority of patients have been rendered free of seizures.

Stereoelectroencephalography in Children and Adolescents With Difficult-to-Localize Refractory Focal Epilepsy

Neurosurgery ◽  
2014 ◽  
Vol 75 (3) ◽  
pp. 258-268 ◽  
Author(s):  
Jorge Gonzalez-Martinez ◽  
Jeffrey Mullin ◽  
Juan Bulacio ◽  
Ajay Gupta ◽  
Rei Enatsu ◽  
...  
Keyword(s):  
Children And Adolescents ◽  
Pediatric Patients ◽  
Focal Epilepsy ◽  
Early Experience ◽  
Morbidity Rate ◽  
Seizure Outcome ◽  
Seizure Freedom ◽  
Epileptogenic Zone ◽  
Eloquent Cortex

Abstract BACKGROUND: Although stereoelectroencephalography (SEEG) has been shown to be a valuable tool for preoperative decision making in focal epilepsy, there are few reports addressing the utility and safety of SEEG methodology applied to children and adolescents. OBJECTIVE: To present the results of our early experience using SEEG in pediatric patients with difficult-to-localize epilepsy who were not considered candidates for subdural grid evaluation. METHODS: Thirty children and adolescents with the diagnosis of medically refractory focal epilepsy (not considered ideal candidates for subdural grids and strip placement) underwent SEEG implantation. Demographics, electrophysiological localization of the hypothetical epileptogenic zone, complications, and seizure outcome after resections were analyzed. RESULTS: Eighteen patients (60%) underwent resections after SEEG implantations. In patients who did not undergo resections (12 patients), reasons included failure to localize the epileptogenic zone (4 patients); multifocal epileptogenic zone (4 patients); epileptogenic zone located in eloquent cortex, preventing resection (3 patients); and improvement in seizures after the implantation (1 patient). In patients who subsequently underwent resections, 10 patients (55.5%) were seizure free (Engel class I) and 5 patients (27.7%) experienced seizure improvement (Engel class II or III) at the end of the follow-up period (mean, 25.9 months; range, 12 to 47 months). The complication rate in SEEG implantations was 3%. CONCLUSION: The SEEG methodology is safe and should be considered in children/adolescents with difficult-to-localize epilepsy. When applied to highly complex and difficult-to-localize pediatric patients, SEEG may provide an additional opportunity for seizure freedom in association with a low morbidity rate.

Seizure outcome and complications following hypothalamic hamartoma treatment in adults: endoscopic, open, and Gamma Knife procedures

2012 ◽  
Vol 117 (2) ◽  
pp. 255-261 ◽  
Author(s):  
Cornelia Drees ◽  
Kevin Chapman ◽  
Erin Prenger ◽  
Leslie Baxter ◽  
Rama Maganti ◽  
...  
Keyword(s):  
Mental Retardation ◽  
Weight Gain ◽  
Gamma Knife ◽  
Third Ventricle ◽  
Intractable Epilepsy ◽  
Adult Patients ◽  
Neurological Deficits ◽  
Hypothalamic Hamartoma ◽  
Seizure Outcome ◽  
Seizure Freedom

Object This study aimed at identifying outcomes with respect to seizures, morbidity, and mortality in adult patients undergoing resective or Gamma Knife surgery (GKS) to treat intractable epilepsy associated with hypothalamic hamartoma (HH). Methods Adult patients undergoing surgical treatment for HH-related epilepsy were prospectively monitored at a single center for complications and seizure outcome by using a proprietary database. Preintervention and postintervention data for patients 18 years of age and older, and with at least 1 year of follow-up, were analyzed, with specific attention to seizure control, complications, hormonal status, and death. Results Forty adult patients were found in the database (21 were women). The median HH volume was 0.54 cm3. In 70% of patients, it was located inside the third ventricle, attached unilaterally and vertically to the hypothalamus (Delalande Type II). Most patients (26) underwent an endoscopic resection, 10 patients had a transcallosal or other type of open (pterional or orbitozygomatic) resection, and 4 patients chose GKS. Twenty-nine percent became seizure free in the long term, and overall a majority of patients (55%) reported at least > 90% seizure improvement. Only 3 patients were ultimately able to discontinue anticonvulsants, whereas most patients were taking an average of 2 antiepileptic drugs pre- and postoperatively. The only factor significantly correlated with seizure-free outcome was the absence of mental retardation. The HH volume, HH type, and amount of resection or disconnection were not correlated to seizure freedom. A total of 4 patients (10%) died, 2 immediately after surgery and 2 later. All of them had undergone a resection, as opposed to GKS, and still had seizures. Postoperatively, persistent neurological deficits were seen in 1 patient; 34% of patients had mild hormonal problems; and 59% experienced weight gain of at least 6.8 kg (average gain 12.7 kg). Conclusions Surgical or GKS procedures in adults with HH provided seizure freedom in one-third of patients. The only significant favorable prognostic factor was the absence of mental retardation. The overall mortality rate was high, at 10%. Other important morbidities were persistent hormonal disturbances and weight gain.

Seizure Outcomes in Patients With Surgically Treated Cerebral Arteriovenous Malformations

Neurosurgery ◽  
2015 ◽  
Vol 77 (5) ◽  
pp. 762-768 ◽  
Author(s):  
Christian von der Brelie ◽  
Matthias Simon ◽  
Jonas Esche ◽  
Johannes Schramm ◽  
Azize Boström
Keyword(s):  
Life Quality ◽  
Seizure Outcome ◽  
Seizure Freedom ◽  
Common Symptom ◽  
Extensive Resection ◽  
Chronic Epilepsy ◽  
Epilepsy Classification ◽  
Drug Resistant Epilepsy ◽  
Postoperative Seizure

Abstract BACKGROUND: Epilepsy is the second most common symptom in cerebral arteriovenous malformation (AVM) patients. The consecutive reduction of life quality is a clinically underrated problem because treatment usually focuses on the prevention of intracerebral hemorrhage. OBJECTIVE: To evaluate postoperative seizure outcome with the aim of more accurate counseling for postoperative seizure outcome. METHODS: From 1985 to 2012, 293 patients with an AVM were surgically treated by J.S. One hundred twenty-six patients with preoperative seizures or epilepsy could be identified; 103 of 126 had a follow-up of at least 12 months and were included in the analysis. The different epilepsy subtypes were categorized (sporadic seizures, chronic epilepsy, drug-resistant epilepsy [DRE]). Preoperative workup and surgical technique were evaluated. Seizure outcome was analyzed by using International League Against Epilepsy classification. RESULTS: Sporadic seizures were identified in 41% of patients (chronic epilepsy and DRE were identified in 36% and 23%, respectively). Detailed preoperative epileptological workup was done in 13%. Seizure freedom was achieved in 77% (79% at 5 years, 84% at 10 years). Outcome was significantly poorer in DRE cases. More extensive resection was performed in 11 cases with longstanding symptoms (>24 months) and resulted in better seizure outcome as well as the short duration of preoperative seizure history. CONCLUSION: Patients presenting with AVM-associated epilepsy have a favorable seizure outcome after surgical treatment. Long-standing epilepsy and the progress into DRE markedly deteriorate the chances to obtain seizure freedom and should be considered an early factor in establishing the indication for AVM removal.

Tailored multilobar disconnective epilepsy surgery in the posterior quadrant

2020 ◽  
Vol 132 (5) ◽  
pp. 1345-1357 ◽  
Author(s):  
Michele Rizzi ◽  
Martina Revay ◽  
Piergiorgio d’Orio ◽  
Pina Scarpa ◽  
Valeria Mariani ◽  
...  
Keyword(s):  
Safety Profile ◽  
Class I ◽  
Seizure Outcome ◽  
Seizure Freedom ◽  
Type I ◽  
Drug Resistant ◽  
Presurgical Evaluation ◽  
Posterior Quadrant ◽  
Drug Resistant Epilepsy

OBJECTIVESurgical treatment of drug-resistant epilepsy originating from the posterior quadrant (PQ) of the brain often requires large multilobar resections, and disconnective techniques have been advocated to limit the risks associated with extensive tissue removal. Few previous studies have described a tailored temporoparietooccipital (TPO) disconnective approach; only small series with short postoperative follow-ups have been reported. The aim of the present study was to present a tailored approach to multilobar PQ disconnections (MPQDs) for epilepsy and to provide details about selection of patients, presurgical investigations, surgical technique, treatment safety profile, and seizure and cognitive outcome in a large, single-center series of patients with a long-term follow-up.METHODSIn this retrospective longitudinal study, the authors searched their prospectively collected database for patients who underwent MPQD for drug-resistant epilepsy in the period of 2005–2017. Tailored MPQDs were a posteriori grouped as follows: type I (classic full TPO disconnection), type II (partial TPO disconnection), type III (full temporooccipital [TO] disconnection), and type IV (partial TO disconnection), according to the disconnection plane in the occipitoparietal area. A bivariate statistical analysis was carried out to identify possible predictors of seizure outcome (Engel class I vs classes II–IV) among several presurgical, surgical, and postsurgical variables. Preoperative and postoperative cognitive profiles were also collected and evaluated.RESULTSForty-two consecutive patients (29 males, 24 children) met the inclusion criteria. According to the presurgical evaluation (including stereo-electroencephalography in 13 cases), 12 (28.6%), 24 (57.1%), 2 (4.8%), and 4 (9.5%) patients received a type I, II, III, or IV MPQD, respectively. After a mean follow-up of 80.6 months, 76.2% patients were in Engel class I at last contact; at 6 months and 2 and 5 years postoperatively, Engel class I was recorded in 80.9%, 74.5%, and 73.5% of cases, respectively. Factors significantly associated with seizure freedom were the occipital pattern of seizure semiology and the absence of bilateral interictal epileptiform abnormalities at the EEG (p = 0.02). Severe complications occurred in 4.8% of the patients. The available neuropsychological data revealed postsurgical improvement in verbal domains, whereas nonunivocal outcomes were recorded in the other functions.CONCLUSIONSThe presented data indicate that the use of careful anatomo-electro-clinical criteria in the presurgical evaluation allows for customizing the extent of surgical disconnections in PQ epilepsies, with excellent results on seizures and an acceptable safety profile.

scholarly journals Magnesium as an Effective Adjunct Therapy for Drug Resistant Seizures

2012 ◽  
Vol 39 (3) ◽  
pp. 323-327 ◽  
Author(s):  
Peter A. Abdelmalik ◽  
Nina Politzer ◽  
Peter L. Carlen
Keyword(s):  
Intractable Epilepsy ◽  
Retrospective Chart Review ◽  
Epileptic Seizures ◽  
Controlled Study ◽  
Seizure Freedom ◽  
Drug Resistant ◽  
Cns Depressant ◽  
Drug Resistant Epilepsy ◽  
Double Blinded

Objective:To explore the use of magnesium (Mg), an endogenous ion and enzymatic co-factor used in a variety of medical applications, for the treatment of epileptic seizures resistant to traditional medical therapy.Background:For almost a century, Mg has been used as prophylaxis and treatment of seizures associated with eclampsia. Mg is a CNS depressant, with numerous functions intracellularly and extracellularly. However, because of the availability of well studied anticonvulsant drugs, Mg has not been tested widely in the treatment of epileptic seizures.Methods:A retrospective chart review of 22 cases of drug resistant epilepsy, where a trial of empiric oral Mg supplementation (mainly in the form of Mg-oxide) was conducted.Results:Oral Mg supplementation was associated with a significant decrease in the number of seizure days per month, from 15.3 ± 13.2 (mean ± SD) to 10.2 ± 12.6 at first follow up (3-6 months, p=0.021), and to 7.8 ± 10.0 seizure days/month at second follow up (6-12 months, p=0.004). Thirty-six percent had a response rate of 75% or greater at second follow-up. Two patients reported seizure freedom. Most patients were well maintained on MgO 420mg twice a day, or in 2 cases, Mg Lactate, without significant adverse effects, the most frequent being diarrhea (4/22).Discussion:These results suggest that oral Mg supplementation may prove to be a worthwhile adjunctive medication in treating drug intractable epilepsy.Conclusions:A prospective, double-blinded, placebo controlled study is warranted to evaluate the potential of Mg for the treatment of drug-resistant seizures.

Selective posterior cerebral artery amobarbital test: a predictor of memory following subtemporal selective amygdalohippocampectomy

2019 ◽  
Vol 12 (2) ◽  
pp. 165-169 ◽  
Author(s):  
Joshua S Catapano ◽  
Alexander C Whiting ◽  
Derrick J Wang ◽  
Randall J Hlubek ◽  
Mohamed A Labib ◽  
...  
Keyword(s):  
Cerebral Artery ◽  
Posterior Cerebral Artery ◽  
Hippocampal Sclerosis ◽  
Neuropsychological Testing ◽  
Intractable Epilepsy ◽  
Seizure Freedom ◽  
Wada Test ◽  
Selective Amygdalohippocampectomy ◽  
Stable Memory

BackgroundThe selective posterior cerebral artery (PCA) amobarbital test, or PCA Wada test, is used to predict memory impairment after epilepsy surgery in patients who have previously had a failed internal carotid artery (ICA) amobarbital test.MethodsMedical records from 2012 to 2018 were retrospectively reviewed for all patients with seizures who underwent a selective PCA Wada test at our institution following a failed or inconclusive ICA Wada test. Standardized neuropsychological testing was performed before and during the Wada procedure and postoperatively in patients who underwent resection.ResultsThirty-three patients underwent a selective PCA Wada test, with no complications. Twenty-six patients with medically refractory epilepsy had a seizure focus amenable to selective amygdalohippocampectomy (AHE). Six patients (23%, n=26) had a failed PCA Wada test and did not undergo selective AHE, seven (27%) declined surgical resection, leaving 13 patients who underwent subtemporal selective AHE. Hippocampal sclerosis was found in all 13 patients (100%). Twelve patients (92%) subsequently underwent formal neuropsychological testing and all were found to have stable memory. Ten patients (77%) were seizure-free (Engel Class I), with average follow-up of 13 months.ConclusionThe selective PCA Wada test is predictive of memory outcomes after subtemporal selective AHE in patients with a failed or inconclusive ICA Wada test. Furthermore, given the low risk of complications and potential benefit of seizure freedom, a selective PCA Wada test may be warranted in patients with medically intractable epilepsy who are candidates for a selective AHE and who have a prior failed or inconclusive ICA Wada test.

scholarly journals Utility of diffusion tensor imaging studies linked to neuronavigation and other modalities in repeat hemispherotomy for intractable epilepsy

2016 ◽  
Vol 17 (4) ◽  
pp. 483-490 ◽  
Author(s):  
Erin N. Kiehna ◽  
Elysa Widjaja ◽  
Stephanie Holowka ◽  
O. Carter Snead ◽  
James Drake ◽  
...  
Keyword(s):  
Diffusion Tensor Imaging ◽  
Diffusion Tensor ◽  
Intractable Epilepsy ◽  
Seizure Freedom ◽  
Rasmussen’S Encephalitis ◽  
Initial Procedure ◽  
Surgical Options ◽  
Failed Surgery ◽  
Association Fibers

OBJECT Hemispherectomy for unilateral, medically refractory epilepsy is associated with excellent long-term seizure control. However, for patients with recurrent seizures following disconnection, workup and investigation can be challenging, and surgical options may be limited. Few studies have examined the role of repeat hemispherotomy in these patients. The authors hypothesized that residual fiber connections between the hemispheres could be the underlying cause of recurrent epilepsy in these patients. Diffusion tensor imaging (DTI) was used to test this hypothesis, and to target residual connections at reoperation using neuronavigation. METHODS The authors identified 8 patients with recurrent seizures following hemispherectomy who underwent surgery between 1995 and 2012. Prolonged video electroencephalography recordings documented persistent seizures arising from the affected hemisphere. In all patients, DTI demonstrated residual white matter association fibers connecting the hemispheres. A repeat craniotomy and neuronavigation-guided targeted disconnection of these residual fibers was performed. Engel class was used to determine outcome after surgery at a minimum of 2 years of follow-up. RESULTS Two patients underwent initial hemidecortication and 6 had periinsular hemispherotomy as their first procedures at a median age of 9.7 months. Initial pathologies included hemimegalencephaly (n = 4), multilobar cortical dysplasia (n = 3), and Rasmussen's encephalitis (n = 1). The mean duration of seizure freedom for the group after the initial procedure was 32.5 months (range 6–77 months). In all patients, DTI showed limited but definite residual connections between the 2 hemispheres, primarily across the rostrum/genu of the corpus callosum. The median age at reoperation was 6.8 years (range 1.3–14 years). The average time taken for reoperation was 3 hours (range 1.8–4.3 hours), with a mean blood loss of 150 ml (range 50–250 ml). One patient required a blood transfusion. Five patients are seizure free, and the remaining 3 patients are Engel Class II, with a minimum follow-up of 24 months for the group. CONCLUSIONS Repeat hemispherotomy is an option for consideration in patients with recurrent intractable epilepsy following failed surgery for catastrophic epilepsy. In conjunction with other modalities to establish seizure onset zones, advanced MRI and DTI sequences may be of value in identifying patients with residual connectivity between the affected and unaffected hemispheres. Targeted disconnection of these residual areas of connectivity using neuronavigation may result in improved seizure outcomes, with minimal and acceptable morbidity.

scholarly journals Epicardial Box Lesion Using Bipolar Biparietal Radiofrequency and Multimodality Scar Evaluation - a case series

2021 ◽  
Author(s):  
G P Bijvoet ◽  
S M Chaldoupi ◽  
E Bidar ◽  
R J Holtackers ◽  
J G L M Luermans ◽  
...  
Keyword(s):  
Pulmonary Veins ◽  
Case Series ◽  
Small Population ◽  
Surgical Ablation ◽  
Af Ablation ◽  
Surgical Tool ◽  
Atrial Remodelling ◽  
Magnetic Resonance Imaging Mri ◽  
Thoracoscopic Procedure

Abstract Background Surgical epicardial AF ablation can be performed as a stand-alone (thoracoscopic) procedure or concomitant to other cardiac surgery. In hybrid AF ablation thoracoscopic surgical epicardial ablation is combined with a percutaneous endocardial ablation. The Medtronic Gemini-S clamp is a surgical tool that uses irrigated bipolar biparietal RF energy applied with two clamp lesions that overlap to create one epicardial box lesion including the posterior LA wall and the pulmonary veins. Case summary We describe three patients with therapy-refractory persistent AF and different stages of atrial remodelling in whom the Medtronic Cardioblate Gemini-S Irrigated RF Surgical Ablation System was used for hybrid AF ablation. Acute endocardial validation at the end of the hybrid ablation revealed a complete box lesion in all three cases. At 2-year follow-up, two out of three patients had recurrence of atrial arrhythmias. Invasive electro-anatomical mapping confirmed persistence of the box lesion, and the mechanism of arrhythmia recurrence in both patients was unrelated to posterior left atrium or the pulmonary veins. The third patient has been without arrhythmia symptoms since the ablation procedure. A 3D late gadolinium enhancement (LGE) magnetic resonance imaging (MRI) illustrates the ablation scar non-invasively in two cases. Discussion Thoracoscopic biparietal RF AF ablation with the Medtronic Cardioblate Gemini-S Irrigated RF Surgical Ablation System results in permanent transmural scar formation, irrespective of the stage of atrial remodelling, as shown in this small population by means of multimodality scar evaluation.

Surgical outcomes in children with bottom-of-sulcus dysplasia and drug-resistant epilepsy: a retrospective cohort study

2021 ◽  
pp. 1-11
Author(s):  
Puneet Jain ◽  
Ayako Ochi ◽  
Carter McInnis ◽  
Hiroshi Otsubo ◽  
O. Carter Snead ◽  
...  
Keyword(s):  
Surgical Treatment ◽  
Medical Records ◽  
Focal Cortical Dysplasia ◽  
Seizure Freedom ◽  
Language Mapping ◽  
Resective Surgery ◽  
Histological Data ◽  
Drug Resistant Epilepsy ◽  
Video Eeg

OBJECTIVE Bottom-of-sulcus dysplasia (BOSD) is challenging to identify radiologically. The aim of this study was to explore seizure outcomes after resective surgery or MR-guided laser interstitial thermal therapy (MRgLITT) in children with BOSD. METHODS Children with radiologically defined BOSD who underwent resective surgery or MRgLITT, with at least 1 year of follow-up were included. Clinical, radiological, neurophysiological, and histological data were extracted from medical records. Invasive video EEG (IVEEG) was used to evaluate the ictal onset zone or motor/language mapping, wherever appropriate. Histology of MRI-visible BOSD, including the overlying and adjacent cortex, was also evaluated. RESULTS Forty-one children with BOSD underwent surgical treatment. The lesion was initially overlooked on MRI in 20 patients (48.8%). Of 34 patients who underwent IVEEG and who had available ictal data, the ictal onset zone extended beyond the MRI-visible BOSD in 23 patients (67.6%). Surgical treatment included lesionectomy (24 patients), extended lesionectomy (12 patients), lobectomy (1 patient), and ablation of BOSD (4 patients). The pathology in 37 patients who underwent resection showed focal cortical dysplasia type IIB and type IIA in 21 (53.8%) and 16 patients (41%), respectively. Seizure freedom was achieved in 32 patients (78.1%) after a mean follow-up of 4.3 years. CONCLUSIONS Seizure outcomes after resective surgery or MRgLITT in children with BOSD were generally favorable. The authors found that the neurophysiological abnormality and pathology often extended beyond the MRI-visible BOSD.

MO291RITUXIMAB IS EFFECTIVE AND SAFE IN ADULTS WITH STEROID-DEPENDENT NEPHROTIC SYNDROME: A LONG-TERM, SINGLE-CENTER EXPERIENCE

2021 ◽  
Vol 36 (Supplement_1) ◽  
Author(s):  
Gemma Patella ◽  
Alessandro Comi ◽  
Giuseppe Coppolino ◽  
Nicolino Comi ◽  
Giorgio Fuiano ◽  
...  
Keyword(s):  
Nephrotic Syndrome ◽  
Case Series ◽  
Adult Patients ◽  
Single Center ◽  
Single Center Experience ◽  
Steroid Dependent ◽  
The Mean ◽  
Steroid Dependent Nephrotic Syndrome

Abstract Background and Aims Steroid-dependent nephrotic syndrome (SDNS) may require a prolonged multi-drug therapy with risk of drug toxicity and renal failure. Rituximab (RTX) treatment has been found to be helpful in reducing the steroid dosage and the need for immunosuppressants (ISs), but little data are currently available regarding very long-term outcomes in adults. We herein describe a long-term, single-center experience of RTX use in a large series of adults with SDNS. Method We studied 23 adult patients with SDNS (mean age 54.2±17.1 y; 65% male; BMI 28.5±4.7), mostly consequent to membranous (47.8%) or focal glomerulonephritis (30.2 %) who were eligible to start a RTX regimen. Before entering the RTX protocol, proteinuria and eGFR were 7.06±3.87 g/24h and 65.9±28.2 ml/min/1.73 m2, respectively; albumin and CD19/CD20 ratio were 2.9±0.9 g/L and 0.99±0.01 respectively; the mean number of ISs was 2.39±0.89 and the mean annual rate of relapses was 2.2±0.9. Results Patients were followed over a mean follow-up of 64 months (range: 12-144). After RTX (mean dose: 1202.1±372.4 mg) the rate of relapses was virtually nullified (p<0.001). eGFR remained roughly stable (62.1±19.8 ml/min/1.73 m2, p=NS), while proteinuria, albumin, CD19/CD20 and BMI all significantly improved (p ranging from 0.01 to 0.001). The mean number of additional ISs was also reduced (0.44±0.12; p<0.001) and RTX enabled discontinuation of steroids in 13/23 (56.5%) patients. No major adverse events related to therapy were recorded. Conclusion Findings from this large case-series with a remarkable very long follow-up reinforce the role of RTX as an efficient and safe weapon to improve outcomes in adult patients suffering from SDNS.

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