Fatal inflammatory hypophysitis

McIntyre, Emily; Perros, Petros

doi:10.1007/s11102-007-0016-z

Cited by 26 publications

(20 citation statements)

References 24 publications

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“…Laboratory screening every 3-4 weeks during treatment seems reasonable and should include a.m. cortisol, free thyroxine, TSH and serum sodium. ACTH [39], cyclophosphamide [36,40] or cyclosporin-A [41] will also play a role in these cases of hypophysitis.…”

Section: Discussionmentioning

confidence: 99%

Anti-CTLA-4 antibody therapy associated autoimmune hypophysitis: serious immune related adverse events across a spectrum of cancer subtypes

et al. 2009

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Anti-cytotoxic T-lymphocyte antigen-4 (CTLA-4) therapies represent a novel approach to cancer treatment via disruption of immune tolerance to antigens located on tumor cells. Disruption of immune tolerance, however, may occur at a cost. A host of immune related adverse events (IRAEs) are associated with anti-CTLA-4 therapy. Autoimmune hypophysitis has been reported in up to 17% of patients with melanoma and renal cell carcinoma treated with this therapy. Familiarity with the spectrum of IRAEs connected to these therapies is paramount for endocrinologists, oncologists and those involved in the care of these subjects. We review here key aspects of diagnosis and treatment of anti-CTLA-4 antibody therapy resultant IRAEs. We describe the first two cases of hypopituitarism in prostate cancer subjects undergoing experimental therapy with ipilimumab. The clinical evidence strongly suggests that the prostate cancer subjects developed autoimmune hypophysitis as a consequence of anti-CTLA-4 treatment. High dose glucocorticoid treatment resulted in markedly improved symptoms, and resolution of focal symptoms and diabetes insipidus. One subject recovered pituitary-thyroid axis function after 9 months; however, both continue to require GC replacement. These cases highlight the importance of early screening and treatment for hypopituitarism in all subjects undergoing treatment with anti-CTLA-4 therapy to prevent a potentially fatal outcome from secondary adrenal insufficiency, a readily treatable disease. We recommend mandatory long term follow-up to monitor the development of other hormonal deficits.

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Section: Discussionmentioning

confidence: 99%

Anti-CTLA-4 antibody therapy associated autoimmune hypophysitis: serious immune related adverse events across a spectrum of cancer subtypes

et al. 2009

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“…As for pituitary involvement, although there is consensus that it may affect less than 1% of the patients with WG (8), there are series that report pituitary dysfunction in approximately 8% of them (10). Furthermore, only as many as 24 cases have been reported in the literature from 1966 to 2011 (6,7). The majority of the patients suffered from CDI combined with partial involvement of the anterior pituitary (11)(12)(13)(14)(15)(16).…”

Section: Discussionmentioning

confidence: 99%

“…Approximately 24 cases of pituitary involvement in WG have been reported in the literature so far (6,7). The aim of this article is to describe a case of pituitary involvement in a patient with previous diagnose of WG, and to make a brief discussion of pituitary involvement in WG.…”

Section: Sumáriomentioning

confidence: 99%

Panhypopituitarism due to Wegener's granulomatosis

Santoro

Guida

Furioso

et al. 2011

Arq Bras Endocrinol Metab

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SUMMARYWegener's granulomatosis (WG) is a multi-system necrotizing granulomatous vasculitis which classically affects the upper respiratory tract, lungs and kidneys. Pituitary participation has been described in 24 patients in the literature to date. The aim of this article is to report a case of pituitary involvement in WG, and to present a literature review on this association. We present a female patient with WG who evolved with central diabetes insipidus (CDI), panhypopituitarism, and mild hyperprolactinemia. MRI showed an infiltrative pattern. Pituitary involvement has been reported in around 1% of patients with WG, mostly in women. It is represented by CDI and hypopituitarism. MRI generally shows pituitary enlargement, stalk thickening and loss of hyperintensity of the neurohypophysis. Permanent endocrine therapy is generally needed. WG should be considered in cases of CDI and hypopituitarism, essentially if a vasculitis is suspected and more common sellar disorders have been ruled out. Arq Bras Endocrinol Metab. 2011;55(7):481-5 SUMáRio A granulomatose de Wegener (GW) é uma vasculite necrotizante multissistêmica que afeta classicamente o trato respiratório superior, pulmões e rins. O envolvimento da hipófise foi descrita em 24 pacientes na literatura, até hoje. O objetivo deste artigo é relatar um caso de GW com envolvimento pituitário, e apresentar uma revisão da literatura sobre esta associação. Apresentamos uma paciente com GW que evoluiu para diabetes insipidus central (DIC), panhipopituitarismo e leve hiperprolactinemia. A RM mostrou um padrão infiltrativo. O envolvimento da hipó-fise foi relatado em cerca de 1% dos pacientes cm GW, na sua maioria mulheres. A desordem é representada por DIC e hipopituitarismo. A RM geralmente mostra o aumento da hipófise, aumento da espessura da haste, e perda da hiperintensidade da neurohipofise. Normalmente, é necessária terapia endócrina permanente. A GW é geralmente considerada nos casos de DIC e hipopituitarismo, essencialmente se há suspeita de vasculite e quando desordens selares mais comuns foram descartadas. Arq Bras Endocrinol Metab. 2011;55(7):481-5

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“…2). Once the histological diagnosis was clear Although it is generally thought no gender predilection in PGH [6,24], the real ratio of male to female among these 38 reviewed cases was closely to 1:2 (12:26). The mean age of these patients at presentation was 46.1 years old (range from 16 to 76 years old), and the female was 44.7 years, younger than the 49.2 years old mean age of male.…”

Section: Case Reportsmentioning

confidence: 93%