scholarly journals A Case Series of Patients with Isolated IgG4-related Hypophysitis Treated with Rituximab

2020 ◽  
Vol 4 (6) ◽  
Author(s):  
Hessa Boharoon ◽  
James Tomlinson ◽  
Clara Limback-Stanic ◽  
Anastasia Gontsorova ◽  
Niamh Martin ◽  
...  
Keyword(s):  
Case Series ◽  
Disease Process ◽  
High Recurrence Rate ◽  
Sustained Remission ◽  
Mri Findings ◽  
Immunoglobulin G4 ◽  
Igg4 Related Disease ◽  
Anterior Pituitary Function ◽  
Pituitary Enlargement

Abstract Context The acute presentation of immunoglobulin G4 (IgG4)-related hypophysitis can be indistinguishable from other forms of acute hypophysitis, and histology remains the diagnostic gold standard. The high recurrence rate necessitates long-term immunosuppressive therapy. Rituximab (RTX) has been shown to be effective in systemic IgG4-related disease (IgG4-RD), but experience with isolated pituitary involvement remains limited. Case Description We report 3 female patients with MRI findings suggestive of hypophysitis. All patients underwent transsphenoidal biopsy and fulfilled diagnostic criteria for IgG4-related hypophysitis. Treatment with glucocorticoids (GCs) resulted in good therapeutic response in Patients 1 and 2, but the disease recurred on tapering doses of GCs. GC treatment led to emotional lability in Patient 3, necessitating a dose reduction. All 3 patients received RTX and Patients 2 and 3 received further courses of treatment when symptoms returned and B-cells repopulated. Patient 3 did not receive RTX until 12 months from the onset of symptoms. Patient 1 was not able to have further RTX treatments due to an allergic reaction when receiving the second dose. Rituximab treatment resulted in sustained remission and full recovery of anterior pituitary function in Patients 1 and 2, with complete resolution of pituitary enlargement. By contrast, Patient 3 only showed a symptomatic response following RTX treatment, but pituitary enlargement and hypofunction persisted. Conclusion Rituximab treatment for IgG4-related hypophysitis resulted in sustained remission in 2 patients treated early in the disease process but only achieved partial response in a patient with chronic disease, suggesting that early therapeutic intervention may be crucial in order to avoid irreversible changes.

scholarly journals Salvage endoscopic resection in patients with esophageal adenocarcinoma after chemoradiotherapy

2018 ◽  
Vol 06 (09) ◽  
pp. E1126-E1129 ◽  
Author(s):  
Irma C. Noordzij ◽  
Wouter L. Curvers ◽  
Clément J. Huysentruyt ◽  
Grard A.P. Nieuwenhuijzen ◽  
Geert-Jan Creemers ◽  
...  
Keyword(s):  
Esophageal Adenocarcinoma ◽  
Endoscopic Resection ◽  
Argon Plasma Coagulation ◽  
Case Series ◽  
Argon Plasma ◽  
Residual Tumor ◽  
Esophageal Resection ◽  
Sustained Remission ◽  
Long Term Prognosis

Abstract Background and study aims For early esophageal adenocarcinoma, endoscopic resection is an accepted curative treatment with an excellent long-term prognosis. Case series from Japan have reported endoscopic resection of residual esophageal squamous cell carcinoma after chemoradiotherapy. This is the first report describing endoscopic resection of residual esophageal adenocarcinoma after chemoradiotherapy. Two patients with advanced esophageal adenocarcinoma had been treated with chemoradiotherapy because comorbidity precluded esophageal resection. When residual tumor was observed endoscopically, complete remission was achieved by salvage endoscopic therapy alone or in combination with argon plasma coagulation (APC). Both patients achieved long-term sustained remission and died of non-tumor-related causes.

scholarly journals Immunoglobulin G4-Related Pancreatic and Biliary Diseases

2013 ◽  
Vol 27 (9) ◽  
pp. 523-530 ◽  
Author(s):  
Hisham Al-Dhahab ◽  
Julia McNabb-Baltar ◽  
Said Al-Busafi ◽  
Alan N Barkun
Keyword(s):  
Autoimmune Pancreatitis ◽  
Clinical Importance ◽  
Clinical Aspects ◽  
Autoimmune Cholangitis ◽  
Immunoglobulin G4 ◽  
Related Disease ◽  
Igg4 Related Disease ◽  
Biliary Diseases ◽  
Pubmed Database

BACKGROUND: Autoimmune pancreatitis and autoimmune cholangitis are new clinical entities that are now recognized as the pancreaticobiliary manifestations of immunoglobulin (Ig) G4-related disease.OBJECTIVE: To summarize important clinical aspects of IgG4-related pancreatic and biliary diseases, and to review the role of IgG4 in the diagnosis of autoimmune pancreatitis (AIP) and autoimmune cholangitis (AIC).METHODS: A narrative review was performed using the PubMed database and the following keywords: “IgG4”, “IgG4 related disease”, “autoimmune pancreatitis”, “sclerosing cholangitis” and “autoimmune cholangitis”. A total of 955 articles were retrieved; of these, 381 contained relevant data regarding the IgG4 molecule, pathogenesis of IgG-related diseases, and diagnosis, management and long-term follow-up for patients with AIP and AIC. Of these 381 articles, 66 of the most pertinent were selected.RESULTS: The selected studies demonstrated the increasing clinical importance of both AIP and AIC, which can mimic pancreatic cancer and cholangiocarcinoma, respectively. IgG4 titration in tissue or blood cannot be used alone to diagnose all IgG4-related diseases; however, it is often a useful adjunct to clinical, radiological and histological features. AIP and AIC respond to steroids; however, relapse is common and long-term maintenance treatment often required.CONCLUSIONS: A review of the diagnosis and management of both AIC and AIP is timely and pertinent to clinical practice because the amount of information regarding these conditions has increased substantially in the past few years, resulting in significant impact on the clinical management of affected patients.

scholarly journals Long-Term MRI Findings of Muslin-Induced Foreign Body Granulomas after Aneurysm Wrapping

2014 ◽  
Vol 20 (1) ◽  
pp. 67-73 ◽  
Author(s):  
Lee-Anne Slater ◽  
Ronil V. Chandra ◽  
Michael Holt ◽  
Andrew Danks ◽  
Winston Chong
Keyword(s):  
Foreign Body ◽  
Case Series ◽  
Mri Findings ◽  
Invasive Treatment ◽  
Mri Features ◽  
Apparent Diffusion ◽  
Rim Enhancement ◽  
History Of ◽  
Arterial Aneurysms

Muslin-induced foreign body granulomas are rare delayed complications after wrapping of intracranial aneurysms. Few small case series have been reported, with a paucity of documented MRI findings. In addition, there are no reports on long-term radiological appearances or temporal evolution of conservatively managed patients. We thus report on the long-term radiological and clinical follow-up of two patients with asymptomatic muslin-induced foreign body granulomas after wrapping of recurrent middle cerebral arterial aneurysms. Both patients were successfully managed conservatively and remain asymptomatic three and six years after diagnosis of their granulomas. A literature review confirms that MRI features of muslin-induced foreign body granuloma are typical. Features include focal areas of elevated T2 signal with increased diffusion-weighted signal and thin rim enhancement. To the best of our knowledge, this is the first report to confirm that there is a corresponding reduction in apparent diffusion coefficient, as typical in an intracranial abscess. Thus a history of aneurysm wrapping is critical for diagnosis. Accurate clinical recognition of this exuberant inflammatory response will avoid misdiagnosis as pyogenic abscess or tumor and prevent unnecessary or invasive treatment.

scholarly journals Long-term outcomes of primarily metastatic juvenile pilocytic astrocytoma in children

2018 ◽  
Vol 21 (1) ◽  
pp. 49-53 ◽  
Author(s):  
Derek Yecies ◽  
Paul Graham Fisher ◽  
Samuel Cheshier ◽  
Michael Edwards ◽  
Gerald Grant
Keyword(s):  
Stable Disease ◽  
Pilocytic Astrocytoma ◽  
Radiation Treatment ◽  
Small Sample Size ◽  
Case Series ◽  
Disease Process ◽  
Small Sample ◽  
Long Term Outcomes ◽  
Juvenile Pilocytic Astrocytoma

OBJECTIVEPrimarily metastatic juvenile pilocytic astrocytoma (JPA) is rare, likely representing 2%–3% of all cases of JPA. Due to the rarity of primarily metastatic JPA, there is currently no standard treatment paradigm and the long-term outcomes are not fully known. The goal of this case series was to add to the current understanding of this disease process.METHODSThe authors searched a comprehensive database of pediatric patients with brain and spinal cord tumors treated at Lucile Packard Children’s Hospital from 1997 to 2016 and identified 5 patients with primarily metastatic JPA. A retrospective chart review was performed and details of the patients’ treatment and clinical course were recorded for further analysis.RESULTSFor the 5 patients with primarily metastatic JPA, the mean follow-up period was 12.3 years. All patients in our series had biopsies or subtotal resections and upfront treatment. Three patients were treated with chemotherapy alone, one was treated with chemotherapy and radiotherapy, and one was treated with radiotherapy alone. Four patients had stable disease after initial treatment, and one patient had multiple episodes of progressive disease but underwent successful salvage therapy and has had stable disease for 19 years. One patient died of an intracerebral hemorrhage 10 years following initial radiation treatment believed to be secondary to radiation vasculopathy.CONCLUSIONSEvaluation of the entire neuraxis should be performed in all instances of initial JPA diagnosis to properly assess for primarily metastatic disease. Many patients with primarily metastatic JPA will have stable disease after upfront treatment, although the higher rate of stable disease found in this series relative to other reports is likely secondary to the small sample size.

Novel Application of Steroid Eluting Stents in Choanal Atresia Repair

2016 ◽  
Vol 126 (1) ◽  
pp. 79-82 ◽  
Author(s):  
John N. Bangiyev ◽  
Nandini Govil ◽  
Anthony Sheyn ◽  
Michael Haupert ◽  
Prasad John Thottam
Keyword(s):  
Pediatric Population ◽  
Choanal Atresia ◽  
Case Series ◽  
Disease Process ◽  
Mometasone Furoate ◽  
Pediatric Hospital ◽  
Routine Surveillance ◽  
Surveillance Endoscopy

Purpose: To describe the application of mometasone furoate eluting sinus stent technology in the treatment of choanal atresia (CA) in the hopes of preventing postsurgical stenosis. Methods: We analyzed 3 consecutive patients aged 4 days to 16 years undergoing repair of CA at a tertiary pediatric hospital. Mometasone furoate eluting sinus stents were placed intraoperatively. Postoperative need for revision surgery as well as routine surveillance endoscopy were used to determine success of surgery. Results: Three patients of varying age and etiology underwent successful repair of choanal atresia/stenosis. The steroid eluting sinus stent was deployed successfully in all 3 cases. There was no identifiable restenosis in any of the 3 patients with 12-month follow-up. There were no complications noted throughout the follow-up period. Conclusions: Choanal atresia is a rare disorder that can prove difficult in postsurgical management. In our case series, mometasone furoate eluting stents were effective and safe for the management of this disease process. Further prospective studies are needed to determine the exact safety profile, long-term consequences, and efficacy of steroid eluting sinus stents in the pediatric population.

scholarly journals Late Relapses of Membranous Nephropathy: A Case Series

Kidney360 ◽  
2021 ◽  
pp. 10.34067/KID.0007712020
Author(s):  
Yonatan Peleg ◽  
Andrew S. Bomback ◽  
Pietro A. Canetta ◽  
Jai Radhakrishnan ◽  
Gerald B. Appel ◽  
...  
Keyword(s):  
Nephrotic Syndrome ◽  
Membranous Nephropathy ◽  
Clinical Symptoms ◽  
Case Series ◽  
Renal Outcome ◽  
Late Relapse ◽  
Sustained Remission ◽  
Glomerular Diseases ◽  
Disease Remission

Background: Relapse of the nephrotic syndrome is common among primary membranous nephropathy (MN) patients. Relapses of MN typically occur within a few years of achieving disease remission. There is limited description to date regarding MN patients who have late relapse of MN, i.e. after more than five years of sustained disease remission. The objective of this case series was to report the clinical course of MN patients with late relapse. Methods: We analyzed the patient database of the Center for Glomerular Diseases at Columbia University to identify patients seen at our center who had relapse of biopsy-proven MN at least five years after achieving sustained disease remission. Results: We identified 16 patients with late MN relapse. The median time in sustained remission prior to relapse was 10.2 (range 7-29.0) years. Ten (62.5%) patients were diagnosed with late relapse based on laboratory monitoring alone without clinical symptoms of the nephrotic syndrome. Fourteen (87.5%) patients received immunosuppression during their initial presentation and late relapse. Patients had favorable long term renal outcomes over a median 21 (range 12-56) year follow-up period with 14 (87.5%) patients in remission at study conclusion and median decline in eGFR per year -0.63 (range -6.3 - 17.5) ml/min/1.73m2/year. Conclusions: This case series highlights a previously under-appreciated and likely rare outcome of MN, namely late relapse. Late relapse patients, having a longer time in sustained remission, may have a more favorable long-term renal outcome.

Force Plate Analysis of Dogs with Bilateral Hip Dysplasia Treated with a Unilateral Triple Pelvic Osteotomy: A Long-term Review of Cases

1998 ◽  
Vol 11 (02) ◽  
pp. 85-93 ◽  
Author(s):  
Joanne R. Cockshutt ◽  
H. Dobson ◽  
C. W. Miller ◽  
D. L. Holmberg ◽  
Connie L. Taves ◽  
...  
Keyword(s):  
Gait Analysis ◽  
Hip Dysplasia ◽  
Weight Bearing ◽  
Force Plate ◽  
Case Series ◽  
Pelvic Osteotomy ◽  
Vertical Force ◽  
Long Term Outcome ◽  
Case Series Study

SummaryA retrospective case series study was done to determine the long-term outcome of operations upon dogs treated for canine hip dysplasia by means of a triple pelvic osteotomy (TPO). Twentyfour dogs with bilateral hip dysplasia, that received a unilateral TPO between January 1988 and June 1995, were re-examined at the Ontario Veterinary College. The assessment included physical, orthopedic and lameness examinations, standard blood work, pelvic radiographs and force plate gait analysis. They were compared to bilaterally dysplastic dogs that had not been treated, and also to normal dogs. Force plate data analysis demonstrated a significant increase in peak vertical force (PVF) and mean vertical force over stance (MVF) in the limb that underwent surgical correction by means of a TPO, when compared to the unoperated hip. It was determined that performing a unilateral TPO on a young dysplastic dog resulted in greater forces and weight bearing being projected through the TPO corrected limb when compared to the unoperated limb.Dogs with bilateral hip dysplasia treated with a unilateral triple pelvic osteotomy (TPO) were assessed by force plate gait analysis, radiographs and orthopedic examination. There was a significant increase in hip Norberg angles over time, although degenerative changes did progress. Limbs that had been operated upon had significantly greater peak and mean ground reaction forces than limbs that had not received an operation.

Mechanical failure of the Mobi-C implant for artificial cervical disc replacement: report of 4 cases

2020 ◽  
Vol 33 (6) ◽  
pp. 727-733
Author(s):  
Jasmine A. T. DiCesare ◽  
Alexander M. Tucker ◽  
Irene Say ◽  
Kunal Patel ◽  
Todd H. Lanman ◽  
...  
Keyword(s):  
Cervical Spondylosis ◽  
Case Series ◽  
Dynamic Imaging ◽  
Disc Replacement ◽  
Cervical Disc ◽  
Artificial Disc ◽  
Younger Patients ◽  
Cervical Disc Replacement ◽  
Short And Long Term

Cervical spondylosis is one of the most commonly treated conditions in neurosurgery. Increasingly, cervical disc replacement (CDR) has become an alternative to traditional arthrodesis, particularly when treating younger patients. Thus, surgeons continue to gain a greater understanding of short- and long-term complications of arthroplasty. Here, the authors present a series of 4 patients initially treated with Mobi-C artificial disc implants who developed postoperative neck pain. Dynamic imaging revealed segmental kyphosis at the level of the implant. All implants were locked in the flexion position, and all patients required reoperation. This is the first reported case series of symptomatic segmental kyphosis after CDR.

Long-term outcomes of lumbar microdiscectomy in the pediatric population: a large single-institution case series

2019 ◽  
Vol 24 (5) ◽  
pp. 549-557
Author(s):  
Malia McAvoy ◽  
Heather J. McCrea ◽  
Vamsidhar Chavakula ◽  
Hoon Choi ◽  
Wenya Linda Bi ◽  
...  
Keyword(s):  
Conservative Management ◽  
Functional Outcomes ◽  
Pediatric Patients ◽  
Clinical Presentation ◽  
Case Series ◽  
Csf Leak ◽  
Single Institution ◽  
The Mean

OBJECTIVEFew studies describe long-term functional outcomes of pediatric patients who have undergone lumbar microdiscectomy (LMD) because of the rarity of pediatric disc herniation and the short follow-up periods. The authors analyzed risk factors, clinical presentation, complications, and functional outcomes of a single-institution series of LMD patients over a 19-year period.METHODSA retrospective case series was conducted of pediatric LMD patients at a large pediatric academic hospital from 1998 to 2017. The authors examined premorbid risk factors, clinical presentation, physical examination findings, type and duration of conservative management, indications for surgical intervention, complications, and postoperative outcomes.RESULTSOver the 19-year study period, 199 patients underwent LMD at the authors’ institution. The mean age at presentation was 16.0 years (range 12–18 years), and 55.8% were female. Of these patients, 70.9% participated in competitive sports, and among those who did not play sports, 65.0% had a body mass index greater than 25 kg/m2. Prior to surgery, conservative management had failed in 98.0% of the patients. Only 3 patients (1.5%) presented with cauda equina syndrome requiring emergent microdiscectomy. Complications included 4 cases of postoperative CSF leak (2.0%), 1 case of a noted intraoperative CSF leak, and 3 cases of wound infection (1.5%). At the first postoperative follow-up appointment, minimal or no pain was reported by 93.3% of patients. The mean time to return to sports was 9.8 weeks. During a mean follow-up duration of 8.2 years, 72.9% of patients did not present again after routine postoperative appointments. The total risk of reoperation was a rate of 7.5% (3.5% of patients underwent reoperation for the same level; 4.5% underwent adjacent-level decompression, and one patient [0.5%] ultimately underwent a fusion).CONCLUSIONSMicrodiscectomy is a safe and effective treatment for long-term relief of pain and return to daily activities among pediatric patients with symptomatic lumbar disc disease in whom conservative management has failed.

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