Xanthomatous hypophysitis

Niyazoğlu, Mutlu; Celik, O.; Bakkaloglu, Dogu Vuralli; Öz, Büğe; Tanrıöver, Necmettin; Gazioğlu, Nurperi; Kadıoğlu, Pınar

doi:10.1016/j.jocn.2011.08.041

Cited by 10 publications

(8 citation statements)

References 17 publications

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“…In addition to our patient, a review of the literature revealed a total of 17 previously reported patients with XH (Table 1) [1][2][3][4][5][6][7][8][9]. The review revealed a predominance of female patients with XH with a female to male ratio of approximately 3:1, similar to that reported by Gutenberg et al [5].…”

Section: Discussionsupporting

confidence: 85%

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Xanthomatous hypophysitis

Hanna¹,

Li²,

Beutler³

et al. 2015

Journal of Clinical Neuroscience

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We present a 69-year-old woman who presented with chronic headaches and was found to have a pituitary mass on MRI, which was biopsied and said to be lymphocytic hypophysitis. The woman was placed on prednisone and followed with routine eye examinations. Two years later, the lesion gradually increased in size and the woman developed a decrease in peripheral vision in the right eye. An MRI showed abutment of the right optic nerve by the mass. A repeat endoscopic transsphenoidal biopsy/resection of the pituitary lesion was performed. Histopathological analysis of the specimen was consistent with diagnosis of xanthomatous hypophysitis (XH). XH is an inflammatory disorder of the pituitary gland characterized by an infiltration of lipid-laden histiocytes, also known as xanthoma cells. The mass was biopsied and a diagnosis of lymphocytic hypophysitis was made. The woman reported improved visual acuity and peripheral vision postoperatively. One year after the second resection, her visual symptoms worsened. Repeat MRI revealed expansion of the residual pituitary tissue. She was referred to the radiation oncology department for external beam radiation therapy and was placed on a maintenance dose of steroids. Since undergoing radiation therapy, her vision has improved slightly and her 3month MRI revealed stable lesion size. This woman illustrates a rare pituitary pathology presented with a literature review of published patients describing xanthomatous hypophysitis. A discussion of the clinical presentation, epidemiology, etiology, diagnosis, histology and treatment is provided.

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Section: Discussionsupporting

confidence: 85%

“…Only one case report considered Erdheim-Chester in their patient workup [9]. Erdheim-Chester is a systemic disease with long bone and joint pain in over 95% of patients.…”

Section: Discussionmentioning

confidence: 99%

Xanthomatous hypophysitis

Hanna¹,

Li²,

Beutler³

et al. 2015

Journal of Clinical Neuroscience

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“…There are not enough data reflecting the effects of medical treatment such as glucocorticoids, because it is extremely rare and most cases of xanthomatous hypophysitis have been diagnosed only after surgical resection. Furthermore, no apparent benefit was reported after either pre- or postsurgical therapeutic trials with corticosteroids to date [1, 10]. To our knowledge, this is the first xanthomatous hypophysitis case with steroid responsiveness.…”

Section: Discussionmentioning

confidence: 97%

Steroid Responsive Xanthomatous Hypophysitis Associated with Autoimmune Thyroiditis: A Case Report

et al. 2013

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We report the case of a 36-year-old woman who presented with headache, fever, and amenorrhea. Laboratory analysis revealed hypopituitarism and autoimmune thyroiditis, while a cerebrospinal fluid study suggested concurrent aseptic meningitis. A magnetic resonance image (MRI) scan revealed a 1.0×0.9 cm cystic mass enlarging the sella turcica. Surgical resection via an endoscopic transsphenoidal route was performed. The histological finding of the excised tissue revealed foamy histiocytes with vacuolated cytoplasm, supporting the diagnosis of xanthomatous hypophysitis. Although a residual soft lesion was observed on the MRI image postoperatively, the patient's headache and fever improved. Ten months after surgery, the patient complained of visual impairment and headache, and the residual mass had enlarged into the suprasellar area. High dose (500 mg intravenous) methylprednisolone was administered for 3 days. During the methylprednisolone pulse therapy, the patient's visual acuity and headache improved. A follow-up MRI taken after methylprednisolone therapy showed a marked mass reduction. Our case supports an autoimmune pathophysiology for xanthomatous hypophysitis and suggests that high dose glucocorticoid therapy as a treatment option.

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“…Following this seminal report in 1998, case reports followed . Tashiro et al further subdivided xanthomatous hypophyseal lesions into xanthomatous (XH) and xanthogranulomatous (XGH) hypophysitis, with the latter containing multinucleated giant cells, epithelioid histiocytes and occasional neutrophilic infiltrates in addition to the foamy xanthoma cells and lymphocytic infiltrates of XH.…”

Section: Introductionmentioning

confidence: 99%

Review of xanthomatous lesions of the sella

Kleinschmidt‐DeMasters

Lillehei

Hankinson

2017

Brain Pathology

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Xanthomatous lesions of the sellar region have traditionally been divided into two separate categories, xanthomatous hypophysitis (XH) and xanthogranuloma (XG) of the sellar region. The seminal article on XH, a condition typified by foamy histiocytes and lymphoplasmacytic infiltrates in the pituitary gland/sellar region, but usually little or no hemosiderin pigment, detailed three patients. However, most reports since that time have been single cases, making understanding of the entity difficult. In contrast, the seminal report on XG, characterized by sellar region cholesterol clefts, lymphoplasmacytic infiltrates, marked hemosiderin deposits, fibrosis, multinucleated giant cells around cholesterol clefts, eosinophilic granular necrotic debris, and accumulation of macrophages, included 37 patients, allowing more insights into etiology. Few examples could be linked to adamantinomatous craniopharyngioma, and although ciliated epithelium similar to that of Rathke cleft cyst (RCC) was identified up to 35% of the 37 cases, it could not be proven that XG was related to hemorrhage into RCC. Case reports since that time, however, occasionally linked XG to RCC when an etiology could be identified at all, and a few recognized that a spectrum exists in xanthomatous lesions of the sella. They review literature, adding 23 cases from our own experience, to confirm that overlap occurs between XH and XG, and that the majority-but not all-can be linked to RCC leakage/rupture/hemorrhage. It was suggested that progressive accumulation of hemosiderin pigment in the lesion, possibly caused by the multiple episodes of bleeding, could account for the transition of at least some cases of XH to XG.

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Xanthomatous hypophysitis

Cited by 10 publications

References 17 publications

Xanthomatous hypophysitis

Xanthomatous hypophysitis

Steroid Responsive Xanthomatous Hypophysitis Associated with Autoimmune Thyroiditis: A Case Report

Review of xanthomatous lesions of the sella

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