Bilateral Tolosa-Hunt syndrome mimicking pituitary adenoma

Świątkowska−Stodulska, Renata; Stodulski, Dominik; Babińska, Anna; Piskunowicz, Maciej; Sworczak, Krzysztof

doi:10.1007/s12020-017-1422-2

Cited by 11 publications

(4 citation statements)

References 18 publications

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“…Criterion 3 may help the diagnosis and clarify pathogenesis, but may not be necessarily obvious at the time of diagnosis based on endocrine abnormalities. Other rarer causes of SH are presented in Table 1 [36, 37].…”

Section: Resultsmentioning

confidence: 99%

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Hypophysitis (Including IgG4 and Immunotherapy)

et al. 2020

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Hypophysitis is characterized by inflammation of the pituitary gland that can be primary (PH) or secondary (SH) to other diseases or following drug administration. It may also be classified according to anatomical and histopathological criteria, leading to variable degrees of hypopituitarism and/or compressive symptoms to nearby structures. There has recently been an increase in the number of hypophysitis cases, raising the interest on the spectrum of its pathogenesis, clinical, biochemical/endocrinological, and imaging features. However, the use of conventional biomarkers, including currently utilized pituitary autoantibodies, has relatively limited diagnostic accuracy. Lymphocytic hypophysitis (LH) is the commonest cause of PH, whereas IgG4-related hypophysitis is increasingly being recognized. Histiocytosis and granulomatous diseases are the most frequent causes of SH, although infections and lymphoma have also been reported. The increasing use of immune checkpoint inhibitors in oncology is associated with a high incidence of hypophysitis, providing further understanding of its pathogenesis. Hy-pophysitis can occur silently and be easily missed, potentially leading to substantial morbidity or mortality due to adrenal insufficiency, requiring a high index of clinical suspicion and timely initiation of appropriate treatment. In most cases of LH or drug-induced hypophysitis, active surveillance along with replacement of established hormonal deficiencies is needed. In the presence of compressive and/or evolving symptoms, treatment with glucocorticoids either alone or in combination with other immunosuppressive agents can be used. Surgical decompression is reserved for nonresponsive cases with threatened vital structures. Timely diagnosis and intervention are important to minimize disease-related morbidity and mortality. We aimed to review current concepts and recent developments in the pathogenesis, diagnosis, and management of hypophysitis.

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Section: Resultsmentioning

confidence: 99%

“…Headache (50%) is the most frequent symptom in any type of PH, followed by adrenal insufficiency (10–40%) and hyperprolactinemia (20%) [35, 37]. In LH, adrenocorticotropic hormone followed by TSH deficiency are most commonly reported [1, 55].…”

Section: Resultsmentioning

confidence: 99%

Hypophysitis (Including IgG4 and Immunotherapy)

et al. 2020

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“… 4 The painful ophthalmoparesis is almost exclusively unilateral, with a small number of bilateral cases described. 6 Diagnostic criteria ( Table 1 ) require involvement of one or more of the third, fourth, or sixth CNs, which are most common, but involvement of CN V and CN VII has also been reported in adults. 7 – 9 In a review of 16 pediatric cases, 81% involved CN III, 56% CN VI, 13% CN IV, and 31% both CN III and CN VI.…”

Section: Introductionmentioning

confidence: 99%

Ophthalmoplegia and cranial nerve deficits in an adolescent with headache

Corredor

Holmberg

2021

SAGE Open Medical Case Reports

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Tolosa–Hunt syndrome is an idiopathic, inflammatory condition involving the cavernous sinus and is characterized by unilateral, painful ophthalmoparesis. The condition often begins with retro-orbital pain followed by select cranial nerve involvement. We report the case of a 17-year-old female whose presentation with progressive left-sided headache and ophthalmoparesis culminated in the diagnosis of Tolosa–Hunt syndrome. While many of her signs and symptoms have been previously reported in the rare pediatric cases of Tolosa–Hunt syndrome described in the literature, this case illustrates a unique presentation involving cranial nerves V and VII in addition to the more commonly reported cranial nerve III, IV, and VI palsies.

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“…The only other case reported in the literature has been bilateral THS with involvement of the pituitary, where spontaneous regression was documented. [ 4 ] While THS is considered innocuous and has been shown to resolve spontaneously within a few weeks, morbidity from residual cranial nerve palsies can be disabling, which warrants immunosuppressive treatment. [ 2 ] Hence, we instituted early treatment with intravenous steroids that led to complete remission.…”

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confidence: 99%