scholarly journals Tissue is the issue: when a second biopsy reveals the true diagnosis

2019 ◽  
Author(s):  
Anne-Marie Bogaert ◽  
Anne Hoorens ◽  
Marleen Praet ◽  
Jo Van Dorpe ◽  
Bruce Poppe ◽  
...  
Keyword(s):  
Kidney Biopsy ◽  
Late Onset ◽  
Immunosuppressive Treatment ◽  
Treatment Strategies ◽  
Nephrotic Proteinuria ◽  
Long Term Follow Up ◽  
Generation Sequencing ◽  
Genetic Form

Abstract We describe the case of a woman with minimal glomerular changes on initial kidney biopsy. On long-term follow-up, the patient developed nephrotic proteinuria and a second kidney biopsy was performed, which revealed focal segmental glomerulosclerosis (FSGS). Findings from electron microscopy (EM) examination suggested a genetic form of FSGS. Next-generation sequencing showed heterozygosity for a mutation in COL4A3. Collagen IV nephropathies can be linked to late-onset FSGS. By establishing a genetic cause of FSGS, immunosuppressive treatment can be avoided. This case emphasizes the importance of re-biopsy in cases of a non-explained rise in proteinuria. EM can be helpful in differentiating between primary and secondary FSGS and informing treatment strategies. In cases of adult-onset FSGS that cannot be categorized by clinical–pathological assessment, genetic testing should be considered.

Late onset hypothyroidism after subtotal thyroidectomy for hyperthyroidism: Implications for long term follow-up

1983 ◽  
Vol 70 (12) ◽  
pp. 740-743 ◽  
Author(s):  
A. J. Hedley ◽  
P. D. Bewsher ◽  
S. J. Jones ◽  
A. S. M. Khir ◽  
P. Clements ◽  
...  
Keyword(s):  
Late Onset ◽  
Subtotal Thyroidectomy ◽  
Long Term Follow Up

scholarly journals Does immunological remission, defined as disappearance of autoantibodies, occur with current treatment strategies? A long-term follow-up study in rheumatoid arthritis patients who achieved sustained DMARD-free status

2019 ◽  
Vol 78 (11) ◽  
pp. 1497-1504 ◽  
Author(s):  
Debbie M Boeters ◽  
Leonie E Burgers ◽  
René EM Toes ◽  
Annette van der Helm-van Mil
Keyword(s):  
Rheumatoid Arthritis ◽  
Treatment Strategies ◽  
Signs And Symptoms ◽  
Current Treatment ◽  
Dmard Therapy ◽  
Before And After ◽  
Long Term Follow Up ◽  
Free Status

ObjectivesSustained disease-modifying antirheumatic drug (DMARD)-free status, the sustained absence of synovitis after cessation of DMARD therapy, is infrequent in autoantibody-positive rheumatoid arthritis (RA), but approximates cure (ie, disappearance of signs and symptoms). It was recently suggested that immunological remission, defined as disappearance of anti-citrullinated protein antibodies (ACPA) and rheumatoid factor (RF), underlies this outcome. Therefore, this long-term observational study determined if autoantibodies disappear in RA patients who achieved sustained DMARD-free remission.MethodsWe studied 95 ACPA-positive and/or RF-positive RA patients who achieved DMARD-free remission after median 4.8 years and kept this status for the remaining follow-up (median 4.2 years). Additionally, 21 autoantibody-positive RA patients with a late flare, defined as recurrence of clinical synovitis after a DMARD-free status of ≥1 year, and 45 autoantibody-positive RA patients who were unable to stop DMARD therapy (during median 10 years) were studied. Anti-cyclic citrullinated peptide 2 (anti-CCP2) IgG, IgM and RF IgM levels were measured in 587 samples obtained at diagnosis, before and after achieving DMARD-free remission.Results13% of anti-CCP2 IgG-positive RA patients had seroreverted when achieving remission. In RA patients with a flare and persistent disease this was 8% and 6%, respectively (p=0.63). For anti-CCP2 IgM and RF IgM, similar results were observed. Evaluating the estimated slope of serially measured levels revealed that RF levels decreased more in patients with than without remission (p<0.001); the course of anti-CCP2 levels was not different (p=0.66).ConclusionsSustained DMARD-free status in autoantibody-positive RA was not paralleled by an increased frequency of reversion to autoantibody negativity. This form of immunological remission may therefore not be a treatment target in patients with classified RA.

scholarly journals Algorithm approach for revision surgery following late-onset bleb complications after trabeculectomy: long-term follow-up

2017 ◽  
Vol 80 (1) ◽  
Author(s):  
Claudio I. Perez ◽  
María J. Oportus ◽  
Felipe Mellado ◽  
Felipe Valenzuela ◽  
Cristián Cartes ◽  
...  
Keyword(s):  
Revision Surgery ◽  
Late Onset ◽  
Long Term Follow Up

scholarly journals Why is AAV FVIII gene therapy not approved by the US Food and Drug Administration yet?

2021 ◽  
Vol 5 (20) ◽  
pp. 4313-4313
Author(s):  
Valder R. Arruda
Keyword(s):  
Late Onset ◽  
Ectopic Expression ◽  
Large Animal ◽  
Large Animal Models ◽  
The Us ◽  
Long Term Follow Up ◽  
Manufacturing Platform ◽  
Underlying Mechanisms

Abstract The prospect of a clinical strategy using an adeno-associated virus (AAV) vector for expression of therapeutic levels of factor VIII (FVIII) has been highly desirable. This was initially anticipated by promising data from clinical studies on AAV5-FVIII in men with severe hemophilia A. However, long-term follow-up showed a unique efficacy concern on the sustainability and durability derived from a continuous decline in the FVIII transgene levels starting 1 year after vector injection through year 5. Additional follow-up of early-phase studies and outcomes of an ongoing phase 3 study will likely provide evidence on the feasibility of this approach. Here, the potential underlying mechanisms of the FVIII declining levels, together with the revision of several unique early and late onset findings, are discussed. The lack of long-term preclinical studies in large animal models prevents the firm conclusion that FVIII levels decline was unexpected. It is possible that the combination of vector manufacturing platform and dose, accompanied with ectopic expression of supraphysiologic levels of FVIII at short-term follow-up, may all contribute to the sustainability and durability of the transgene levels. Notably, vector readministration to further improve the FVIII levels is not feasible at this time. Thus, the need of a one-and-done AAV strategy to achieve sustain FVIII levels of expression is sine qua non to impact favorably the disease phenotype.

Long-term follow-up of patients (pts) with relapsed or refractory (r/r) follicular lymphoma (FL) treated with copanlisib.

2019 ◽  
Vol 37 (15_suppl) ◽  
pp. 7553-7553
Author(s):  
Sirpa Leppä ◽  
Armando Santoro ◽  
Judit Demeter ◽  
George Follows ◽  
Georg Lenz ◽  
...  
Keyword(s):  
Adverse Events ◽  
Median Duration ◽  
Kinase Inhibitor ◽  
Late Onset ◽  
Progression Free Survival ◽  
Duration Of Treatment ◽  
Primary Analysis ◽  
Long Term Follow Up

7553 Background: The pan-class I phosphatidylinositol 3-kinase inhibitor copanlisib was approved by the FDA in September 2017 for treatment of relapsed FL based on results from the CHRONOS-1 study in pts with indolent non-Hodgkin lymphoma. We report efficacy and safety results of a 2-year (yr) follow-up of FL pts. Methods: Pts with indolent FL (grade [G] 1-3a) r/r to ≥2 prior lines of treatment received copanlisib (60 mg i.v. infusion) on days 1, 8, and 15 of a 28-day cycle. The primary endpoint was objective tumor response rate (ORR) per independent radiologic review (Cheson et al. 2007). Adverse events were reported using MedDRA (v20.1). Data cut-off: February 20, 2018. Results: 104 FL pts were enrolled. Median age was 62 yr (39% >65 yr), the median number of prior lines of anti-cancer therapy was 3 (range 2-8), and 27 pts (26%) were classified as having G3a disease. The ORR was 59%, with complete responses (CR) in 20% ( n=21); 14 pts had a CR at the primary analysis in June 2016. The median duration of response (mDoR) was 12.2 months (mo) (range 0.03-43 mo). Stable disease (SD) was observed in 33% of pts; median duration of SD was 7.8 mo (range 1.3-23 mo). Median progression-free survival (mPFS) was 11.2 mo (range 0.03-44 mo) with 33% alive and progression-free at 2 yrs. Median overall survival (mOS) was 3.2 yr (range 0.06-4.2 yr) with 67% alive at 2 yrs. Median duration of treatment was 26 weeks (wk) (range 1-192 wk); median duration of safety follow-up was 29 wk. In the G3a subset, the ORR was 67% (26% CR), mDoR was 10.9 mo, mPFS was 12.5 mo, and mOS was 2.5 yr. The most common treatment-emergent adverse events occurring in >25% of pts included (all grade/G3+): diarrhea (37%/9%), neutropenia (26%/23%), and pyrexia (28%/5%). Hyperglycemia (49%/40%) and hypertension (29%/23%) were transient. Incidences of pneumonitis (6.7%/1.9%) and colitis (1.0% G4) were low. Conclusions: Long-term follow-up of r/r FL pts treated with copanlisib revealed robust and durable responses with CRs exceeding 20%, including in pts with higher grade disease. The safety profile continues to be both manageable and favorable, with no evidence of late-onset severe toxicities. Clinical trial information: NCT01660451.

Late Complete Heart Block after Surgical Repair of an Atrial Septal Defect

2021 ◽  
Author(s):  
Bert Zwaenepoel ◽  
Raf Roelandt ◽  
Julie De Backer ◽  
Jan De Pooter
Keyword(s):  
Heart Block ◽  
Late Onset ◽  
Complete Heart Block ◽  
Later Life ◽  
Interesting Case ◽  
Septal Defects ◽  
Long Term Follow Up ◽  
Asd Closure

ABSTRACT Atrial septal defects (ASD) are among the most common forms of congenital heart disease. Although surgical correction was the only available therapy for decades, its long-term complications remain unknown and many patients do not have structured medical follow-up in later life. However, increasing evidences suggest that late-onset cardiac problems, such as complete heart block (CHB), can arise after surgery and therefore, long-term follow-up should be advised in these patients. We hereby present an interesting case of CHB occurring in a 30-year-old patient who had undergone surgical secundum ASD closure approximately 21 years prior to this event and now presented with episodes of dizziness and pre-syncope. Seven-day Holter reported seven episodes of CHB, corresponding to the presenting complaints. The patient was successfully managed with conduction system pacing and he remained asymptomatic on further follow-up. The case description is followed by a brief overview of the available literature.

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