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Cervicothoracic spinal cord compression caused by IgG4-related sclerosing pachymeningitis: a case report and literature review

Published on May 1, 2016in European Spine Journal2.51
· DOI :10.1007/s00586-015-4251-0
Rui Gu5
Estimated H-index: 5
(JLU: Jilin University),
Peng-yuan Hao1
Estimated H-index: 1
(JLU: Jilin University)
+ 2 AuthorsQingsan Zhu5
Estimated H-index: 5
(JLU: Jilin University)
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Abstract
Purpose To report a case of cervicothoracic spinal cord compression caused by IgG4-related sclerosing pachymeningitis (IgG4-RSP).
  • References (18)
  • Citations (6)
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References18
Newest
Published on Jan 1, 2014in Rinshō shinkeigaku Clinical neurology
坂井 利行1
Estimated H-index: 1
,
近藤 昌秀1
Estimated H-index: 1
+ 1 Authors冨本 秀和2
Estimated H-index: 2
症例は32歳の女性である.頭痛,視力低下で発症し,尿崩症,球後視神経炎,四肢腱反射亢進と軽度筋力低下を呈し,血清IgG4上昇,髄液細胞数軽度増多と蛋白増加をみとめ,中枢性IgG4関連疾患と診断した.頭部と脊髄造影MRIにて下垂体炎,および頭蓋,頸椎,腰椎と仙椎レベルの肥厚性硬膜炎を,胸部CTで左肺小結節をみとめた.硬膜生検ではリンパ球系細胞浸潤をみとめたがIgG4免疫染色陽性形質細胞はみられなかった.ステロイドパルス療法にて視力は回復し,血清IgG4は正常になり肥厚性硬膜炎と左肺小結節は軽快した.IgG4関連下垂体炎,および頭蓋,頸椎,腰椎と仙椎レベルの肥厚性硬膜炎を同時期に呈した最初の報告例である.
Published on Dec 1, 2013in British Journal of Neurosurgery1.48
Nitin Mukerji10
Estimated H-index: 10
,
Nicholas V. Todd4
Estimated H-index: 4
AbstractObjective. Spinal epidural haematoma (SEH) causing spinal cord compression is potentially a cause of long-term neurological disability. We reviewed the relevant literature with the intention of establishing the factors that influence long-term outcome and the timeframe within which operative intervention must be undertaken for optimal results. Methods. A total of 1177 papers were identified using PubMed among which 31 papers were selected and analysed. The grades of neurological deficit,...
Published on Feb 1, 2012in Modern Rheumatology1.97
Hisanori Umehara23
Estimated H-index: 23
(Kanazawa Medical University),
Kazuichi Okazaki62
Estimated H-index: 62
(Kansai Medical University)
+ 19 AuthorsYoshiya Tanaka51
Estimated H-index: 51
IgG4-related disease (IgG4RD) is a novel clinical disease entity characterized by elevated serum IgG4 concentration and tumefaction or tissue infiltration by IgG4-positive plasma cells. IgG4RD may be present in a certain proportion of patients with a wide variety of diseases, including Mikulicz’s disease, autoimmune pancreatitis, hypophysitis, Riedel thyroiditis, interstitial pneumonitis, interstitial nephritis, prostatitis, lymphadenopathy, retroperitoneal fibrosis, inflammatory aortic aneurysm...
Published on Aug 1, 2011in Journal of Neurotrauma3.75
Julio C. Furlan27
Estimated H-index: 27
(U of T: University of Toronto),
Vanessa K. Noonan35
Estimated H-index: 35
(UBC: University of British Columbia)
+ 1 AuthorsMichael G. Fehlings93
Estimated H-index: 93
(U of T: University of Toronto)
Abstract While the recommendations for spine surgery in specific cases of acute traumatic spinal cord injury (SCI) are well recognized, there is considerable uncertainty regarding the role of the timing of surgical decompression of the spinal cord in the management of patients with SCI. Given this, we sought to critically review the literature regarding the pre-clinical and clinical evidence on the potential impact of timing of surgical decompression of the spinal cord on outcomes after traumati...
Published on Mar 1, 2011in Journal of Gastroenterology5.13
Kazuichi Okazaki62
Estimated H-index: 62
(Kansai Medical University),
Kazushige Uchida34
Estimated H-index: 34
(Kansai Medical University)
+ 2 AuthorsMakoto Takaoka22
Estimated H-index: 22
(Kansai Medical University)
Recent studies have suggested the existence of two subtypes of autoimmune pancreatitis (AIP): type 1 AIP, related to IgG4 (lymphoplasmacytic sclerosing pancreatitis); and type 2 AIP, related to a granulocytic epithelial lesion (idiopathic duct-centric chronic pancreatitis). Compared with type 2 AIP, the clinicopathological features of type 1 AIP, with increased serum IgG4/IgE levels, abundant infiltration of IgG4 + plasmacytes and lymphocytes, autoantibodies, and steroid responsiveness, are more...
Published on Jan 1, 2011in Current Opinion in Rheumatology3.85
Arezou Khosroshahi25
Estimated H-index: 25
,
John H. Stone67
Estimated H-index: 67
Purpose of reviewTo summarize the existing knowledge of various clinical presentations of IgG4-related systemic disease (IgG4-RSD) and to review the evolving list of organs affected by IgG4-RSD.Recent findingsThe term IgG4-RSD encompasses a variety of clinical entities once regarded as being entirel
Published on Jan 1, 2011in Current Opinion in Rheumatology3.85
Raghuwansh P. Sah17
Estimated H-index: 17
(Mayo Clinic),
Suresh T. Chari73
Estimated H-index: 73
Purpose of review IgG4-related systemic disease (ISD) is a recently recognized syndrome affecting multiple organs. Autoimmune pancreatitis (AIP) is the pancreatic manifestation of ISD and mimics pancreatic cancer. Current data show frequent association with serum IgG4 elevation and other serologic abnormalities. Here we explore the diagnostic and possible prognostic utility and pathogenetic implications of serologic abnormalities in ISD. Recent findings Serum IgG4 elevations (>140 mg/dl) are see...
Published on Oct 12, 2010in Neurology8.69
Sang-Ho Choi33
Estimated H-index: 33
,
Sang Hoon Lee2
Estimated H-index: 2
+ 1 AuthorsSang Ryong Jeon1
Estimated H-index: 1
Immunoglobulin G (IgG)4-related sclerosing disease is an emerging clinicopathologic entity. It is defined as a systemic disease characterized histopathologically by extensive IgG4-positive plasma cell infiltration of various organs together with T-lymphocyte infiltration.1 Pancreas is the most frequently affected organ. This disease was included within the category of autoimmune pancreatitis before the concept of IgG4-related sclerosing disease was introduced.2 There have been several reports of...
Published on Jul 1, 2010in Autoimmunity Reviews7.72
Hiroki Takahashi45
Estimated H-index: 45
(Sapporo Medical University),
Motohisa YamamotoTetsuya32
Estimated H-index: 32
(Sapporo Medical University)
+ 3 AuthorsImaiKohzoh82
Estimated H-index: 82
(Sapporo Medical University)
Abstract IgG4-related disease is a distinct clinical entity, whose characteristic features are the following; Serum IgG4 is prominently elevated, IgG4-positive plasma cells infiltrate in involved tissues, various mass-forming lesions with fibrosis develop in a timely and spatial manner and the response to corticosteroids is prompt and good. IgG4-related diseases mainly target two organs. One is the pancreas (autoimmune pancreatitis; AIP), and the other comprises the lacrimal and salivary glands,...
Lizhi Zhang37
Estimated H-index: 37
,
Thomas Christopher Smyrk1
Estimated H-index: 1
Autoimmune pancreatitis (AIP) is a rare form of chronic pancreatitis that is characterized by lymphoplasmacytic infiltrate, storiform fibrosis, obliterative phlebitis, and increased IgG4+ plasma cells. Serum IgG4 levels usually are elevated. Patients with AIP frequently have disease affecting other organs or sites; these tissues show similar histologic changes, including increased IgG4+ plasma cell infiltrate and response to corticosteroid therapy. A new clinicopathologic concept of IgG4-related...
Cited By6
Newest
Published on Jun 1, 2019in World Neurosurgery1.72
Stephen J. Slade (Virginia Mason Medical Center), Erin M. Bauer (Virginia Mason Medical Center)+ 1 AuthorsAmish J. Dave (Virginia Mason Medical Center)
Abstract Background Back pain is a leading reason patients seek medical attention. While musculoskeletal causes are common, rarer etiologies can present. Case Description A 50-year-old male presented with 2 months of isolated upper back pain initially suspected to be secondary to overuse muscular strain. Over the next 3 months, his pain worsened, and he developed lower extremity dysesthesias and subjective weakness despite a normal neurological exam. Non-revealing laboratory workup included norm...
Published on May 1, 2019in Seminars in Arthritis and Rheumatism5.07
Cléa Melenotte5
Estimated H-index: 5
,
Julie Seguier1
Estimated H-index: 1
+ 20 AuthorsLaurent Daniel30
Estimated H-index: 30
Abstract Pachymeningitis is rare, either idiopathic or secondary to inflammatory disorders, after tumoral, surgical or infectious causes have been excluded. The fibroinflammatory IgG4-related disease is one of the etiologies of pachymeningitis with only few cases reported yet. From a single referral regional center, we evaluated the frequency of IgG4-related disease as the cause of inflammatory pachymeningitis in 10% of cases. From a National case registry of IgG4-related disease the pachymening...
Published on Sep 27, 2018in Case Reports
We report the rare case of an 80-year-old male patient with hypertrophic pachymeningoencephalitis that may be associated with temporal arteritis. The patient presented to our neurological department with a 2-week history of latent paresis and ataxia affecting his right hand. He had been diagnosed with temporal arteritis 12 years earlier. Brain MRI showed an enhancement of the left-sided frontoparietal meninges with oedema of the adjacent tissue of the precentral and postcentral cortex. A leptome...
Published on Feb 1, 2018in Lancet Neurology28.75
Mahmoud A. AbdelRazek1
Estimated H-index: 1
(Harvard University),
Nagagopal Venna16
Estimated H-index: 16
(Harvard University),
John H. Stone67
Estimated H-index: 67
(Harvard University)
Summary IgG4-related disease can involve nearly any organ system, including the central and peripheral nervous systems. The pathology findings are consistent from organ to organ, but careful clinicopathological correlation is necessary to establish the diagnosis. Many non-neurological and neurological inflammatory conditions, previously regarded as idiopathic in nature, are now recognised to fall within the spectrum of IgG4-related disease. The condition is highly treatable, but probably remains...
Published on Nov 1, 2017in Rheumatic Diseases Clinics of North America3.53
Mahmoud A. AbdelRazek1
Estimated H-index: 1
(Mayo Clinic),
John H. Stone67
Estimated H-index: 67
(Harvard University)
Published on Nov 1, 2017in Clinical Imaging1.14
Mary Maher1
Estimated H-index: 1
(CUMC: Columbia University Medical Center),
George Zanazzi19
Estimated H-index: 19
(CUMC: Columbia University Medical Center)
+ 2 AuthorsTony T. Wong3
Estimated H-index: 3
(CUMC: Columbia University Medical Center)
Abstract IgG4-related disease is an immune-mediated fibro-inflammatory condition that affects multiple organ systems. When the pachymeninges are involved, it is termed IgG4-related hypertrophic pachymeningitis (IgG4-RHP). We present a 79-year-old woman with back pain found to have IgG4-RHP localized exclusively in the spine who was also seropositive for myeloperoxidase anti-neutrophil cytoplasmic antibodies (MPO-ANCA). This case is the first known report of IgG4-RHP in the spine with MPO-ANCA an...
Published on Sep 28, 2017in British Journal of Neurosurgery1.48
Kelly J. Bridges2
Estimated H-index: 2
(OHSU: Oregon Health & Science University),
Caitlin H. DeDeaux (OHSU: Oregon Health & Science University), Khoi D. Than5
Estimated H-index: 5
(OHSU: Oregon Health & Science University)
AbstractObject: IgG4-related disease (IgG4-RD) is a fibro-inflammatory disorder affecting various anatomical sites, and only recently was identified to affect the dura of the spine. The authors present the second reported case of an intradural extramedullary lesion consistent with IgG4-related spinal disease.Methods: A literature review was performed that identified 15 other cases of spinal disease, and common features of all known reported spinal IgG4-RD are discussed.Results: Spinal IgG4-RD ty...
Published on Apr 1, 2017in Current Treatment Options in Neurology3.57
Bernardo Baptista1
Estimated H-index: 1
,
Alina Casian6
Estimated H-index: 6
(Guy's and St Thomas' NHS Foundation Trust)
+ 2 AuthorsClaire M Rice2
Estimated H-index: 2
(Southmead Hospital)
IgG4-related disease (IgG4-RD) is a multisystem inflammatory disorder. Early recognition of IgG4-RD is important to avoid permanent organ dysfunction and disability. Neurological involvement by IgG4-RD is relatively uncommon, but well recognised—hypertrophic pachymeningitis and hypophysitis are the most frequent manifestations. Although the nervous system may be involved in isolation, this more frequently occurs in conjunction with involvement of other systems. Elevated circulating levels of IgG...
View next paperIgG4-Related Disease and Hypertrophic Pachymeningitis