Xanthomatous hypophysitis: A novel entity of obscure etiology

Deodhare, Sanjeev; Bilbao, Juan M.; Kovács, K.; Horváth, Éva; Nomikos, P.; Buchfelder, Michael; Reschke, Kirsten; Lehnert, Hendrik

doi:10.1007/bf02738885

Cited by 44 publications

(39 citation statements)

References 10 publications

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“…1,12,21 Many authors have described the presentation of endocrine, visual, and neurological manifestations in parasellar xanthogranulomatous reactions, but a unique symptomatology has not yet been elucidated. 1,2,[6][7][8]12,15,16,20,21,23,[25][26][27] Furthermore, there is significant controversy regarding the etiology and development of these unusual lesions. One theory of the etiology of these parasellar lesions is that all xanthogranulomas arise from inflammatory processes occurring in preexisting CPs.…”

Section: Discussionmentioning

confidence: 99%

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Parasellar xanthogranulomas

Rahmani

Sukumaran

Donaldson

et al. 2015

JNS

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OBJECT Xanthogranulomas are rare inflammatory masses most often found in the skin and eye. The incidence of intracranial xanthogranulomas is 1.6%–7%, with those found in the sellar and parasellar region being exceedingly rare and their etiology controversial. Sellar and parasellar xanthogranulomas are rarely reported in the western hemisphere, and their incidence in Western countries is unknown. METHODS A prospectively acquired database of all endonasal endoscopic transsphenoidal surgeries performed at Weill Cornell Medical College was queried. Patients with histologically confirmed xanthogranulomas who were diagnosed and treated between 2003 and 2013 were included in the study. Patient history, demographic data, histological findings, and surgical approach were also evaluated. RESULTS A total of 643 endonasal endoscopic procedures had been performed at the time of this study. Four patients (0.6%) were identified as having a histologically confirmed xanthogranuloma of the parasellar region, compared with an incidence of 6.7% for craniopharyngioma (CP) and 2% for Rathke cleft cyst (RCC). The most common symptom was visual loss, followed by headache. Preoperative diagnosis was CP in all cases. All patients underwent extended endonasal endoscopic transsphenoidal surgery with gross-total resection. Two patients developed panhypopituitarism after surgery. There were no CSF leaks. The mean follow-up was 61 months, at which time there were no recurrences. The key histological features differentiating xanthogranulomas from CPs were accumulation of foamy macrophages, multinucleated foreign body giant cells, cholesterol clefts, and hemosiderin deposits without stratified squamous epithelium. These histological features appear commonly as part of the spectrum of a secondary inflammatory response in an RCC. CONCLUSIONS Parasellar xanthogranulomas most closely approximate CPs clinically but pathological evidence may suggest an RCC origin. Gross-total resection can be achieved through extended endonasal endoscopic transsphenoidal approaches, and is curative.

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Section: Discussionmentioning

confidence: 99%

“…Folkerth et al 8 and Deodhare et al 6 described patients with xanthomatous hypophysitis. Their histological findings showed foamy histiocytes and lymphocytes within a matrix of anterior pituitary acinar cells and intact vasculature.…”

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confidence: 99%

Parasellar xanthogranulomas

Rahmani

Sukumaran

Donaldson

et al. 2015

JNS

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“…Here, they did not considerably differ among the histological subtypes. But interestingly, visual compromise has, to date, not been described in cases with xanthomatous hypophysitis (13,15,16). This is most likely due to the relatively small size of the pituitary mass in most cases without affecting the optic Table 2 Postsurgical clinical features of 31 patients with lymphocytic, granulomatous or xanthomatous hypophysitis with follow-up time ranging from 1 to 133 months (median: 24 months).…”

Section: Discussionmentioning

confidence: 99%

“…Granulomatous ( Primary hypophysitis chiasm (16), although suprasellar expansion of such pituitary mass lesion was described (13,15,16). Most mentionable, at the time of transphenoidal biopsy, xanthomatous patients were about 10 years younger than patients suffering from lymphocytic and granulomatous hypophysitis.…”

Section: Lymphocytic (Nz21)mentioning

confidence: 99%

“…Additionally, in our study, only patients with lymphocytic hypophysitis showed other autoimmune diseases like juvenile diabetes mellitus, Wegener's granulomatosis, psoriasis or Hashimoto's thyroiditis. Associated autoimmune disorders are only incidentally reported in granulomatous subtype, such as individuals suffering from Crohn's disease (19), psoriasis (20) and Hashimoto's thyroiditis (13) and no association in xanthomatous cases (15,16,21).…”

Section: Lymphocytic (Nz21)mentioning

confidence: 99%

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Primary hypophysitis: clinical-pathological correlations

et al. 2006

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Objective: Primary hypophysitis comprises of three distinct histomorphological entities: lymphocytic, granulomatous and xanthomatous. Clinical features of the three subtypes for diagnostic and treatment strategies have yet not been well characterized. Methods: Endocrine function, visual fields and acuity as well as magnetic resonance imaging characteristics were assessed before and after transphenoidal surgery in the largest series of 31 patients with primary hypophysitis (21 lymphocytic, 6 granulomatous, and 4 xanthomatous cases). Results: Only lymphocytic hypophysitis occurred during pregnancy (30%) and was associated with other autoimmune diseases (24%). Visual fields and acuity abnormalities were not seen in xanthomatous hypophysitis. Lymphocytic and granulomatous hypophysitis most often resulted in severe dysfunction of the adrenal, gonadal and thyroidal axes as well as diabetes insipidus. For patients presenting with xanthomatous hypophysitis most often, mild anterior pituitary axis failure was documented and posterior pituitary involvement was hardly found. The outcome after transphenoidal biopsy was generally favorable. Pre-or postsurgical glucocorticoid treatment was very effective in 75% of the lymphocytic form in reducing the pituitary size. In contrast, glucocorticoid therapy was less effective in granulomatous or xanthomatous hypophysitis. Conclusion: Diffuse destruction of the complete pituitary gland including the infundibulum has to be considered in lymphocytic and granulomatous hypophysitis, whereas in xanthomatous, a circumscribed anterior pituitary lesion leading to compression of the pituitary gland without alteration of the pituitary stalk and optic chiasm can be assumed.

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