Idiopathic hypereosinophilic syndrome revealed by encephalopathy

Lian, Wang; Wei, Ling; Wang, Jin-Cun; Yong-hong, Liu; Deng, Yanchun

doi:10.1016/j.jocn.2011.12.036

Cited by 10 publications

(11 citation statements)

References 9 publications

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“…Table 1). [2][3][4][5][13][14][15] The overall prognosis for XH is good, but improvement of pituitary function after transsphenoidal surgery has been reported in less than 50% of patients. 4,13 Transsphenoidal surgery is both diagnostic and therapeutic, and therefore should be performed in patients with progressive compression or those in whom radiological and/or clinical progression occurs during conservative medical management.…”

Section: Discussionmentioning

confidence: 99%

“…4,13 Transsphenoidal surgery is both diagnostic and therapeutic, and therefore should be performed in patients with progressive compression or those in whom radiological and/or clinical progression occurs during conservative medical management. 15 Glucocorticoid therapy has been advocated to reduce inflammation and has been temporarily effective in some patients with primary hypophysitis. 4,15 However, Gutenberg et al demonstrated that methylprednisolone therapy, administered to one of four patients with XH, did not yield an efficient outcome.…”

Section: Discussionmentioning

confidence: 99%

“…15 Glucocorticoid therapy has been advocated to reduce inflammation and has been temporarily effective in some patients with primary hypophysitis. 4,15 However, Gutenberg et al demonstrated that methylprednisolone therapy, administered to one of four patients with XH, did not yield an efficient outcome. 4 In conclusion, XH and ECD have similar IHC characteristics in pituitary gland tissue.…”

Section: Discussionmentioning

confidence: 99%

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Xanthomatous hypophysitis

Niyazoğlu¹,

Celik²,

Bakkaloglu

et al. 2012

Journal of Clinical Neuroscience

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Xanthomatous hypophysitis (XH) is the rarely seen primary form of hypophysitis. The histological differential diagnosis includes other causes of hypophysitis, Erdheim-Chester disease (ECD), Langerhans cell histiocytosis, Rosai-Dorfman disease and plasma cell granulomas. We present a 39-year-old woman admitted to our department with headache, menstrual irregularity and galactorrhea. The MRI revealed a lesion with a central cystic/necrotic region and a diameter of almost 1cm. Histologic examination showed an inflammatory infiltrate of numerous foamy histiocytes, surrounding the necrotic tissue. On immunohistochemical sections, infiltrating foamy cells stained strongly positive for CD68, and negative for CD1a and S100. After establishing the diagnosis of XH, the patient underwent glucocorticoid treatment. XH should be considered in the differential diagnosis of pituitary lesions. Since XH is rare, it is difficult to assess the efficacy of medical/surgical treatment of this entity accurately.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Xanthomatous hypophysitis

Niyazoğlu¹,

Celik²,

Bakkaloglu

et al. 2012

Journal of Clinical Neuroscience

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“…9 In addition to the confluent hemorrhage, which can also be seen on conventional T1-weighted and T2-weighted MRI, SWI reveals non-displaced medullary veins with concomitant petechial hemorrhage as a unique diagnostic clue of AHL. These findings on SWI are consistent with the pathological hallmarks of AHL, perivascular inflammation and hemorrhage.…”

Section: Discussionmentioning

confidence: 96%

Value of susceptibility-weighted imaging in acute hemorrhagic leukoencephalitis

Kao

Alexandru

Kim

et al. 2012

Journal of Clinical Neuroscience

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“…Various neurological complications have been reported [9][10][11][12][13][14][15], but there are extremely few accounts of CNS pathological changes (and none showing vasculitis) [16][17][18][19][20].…”

Section: Introductionmentioning

confidence: 99%

Idiopathic hypereosinophilic syndrome: a new cause of vasculitis of the central nervous system

et al. 2015

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Idiopathic hypereosinophilic syndrome (IHES) is a primary haematological condition characterised by persistent, otherwise unexplained hypereosinophilia sufficient to cause organ damage. Various neurological complications are reported, but very few have mentioned CNS pathology and none has included CNS vasculitis. Our objective here is to report IHES as a new cause of histopathologically confirmed CNS vasculitis. A 39-year-old man presented with a relapsing sub-acute encephalopathy, with severe headaches, confusion and drowsiness, myoclonus, ataxia and papilloedema. He had a history of nephrotic syndrome 18 years earlier, stable for the past 5 years on low-dose corticosteroids and low-dose tacrolimus (2 mg bd); lichen planus, and (15 years previously) aloplecia totalis. On admission, he had a marked peripheral eosinophilia (up to 9.1 × 10(9)/dL), which—it subsequently became clear—had been intermittently present for 16 years. After extensive investigation, biopsies of brain and bone marrow confirmed diagnoses of cerebral vasculitis, with lymphocytic and macrophage (but not eosinophilic) cellular infiltration of blood vessel walls, and IHES. CNS vasculitis can therefore now be added to the list of neurological complications of IHES. A dramatic and sustained neurological improvement, and likewise of the eosinophilia, following treatment with corticosteroids and cyclophosphamide, emphasises the tractability of this newly described form of CNS vasculitis.

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Idiopathic hypereosinophilic syndrome revealed by encephalopathy

Cited by 10 publications

References 9 publications

Xanthomatous hypophysitis

Xanthomatous hypophysitis

Value of susceptibility-weighted imaging in acute hemorrhagic leukoencephalitis

Idiopathic hypereosinophilic syndrome: a new cause of vasculitis of the central nervous system

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