Parasellar Chronic Inflammatory Disease Presenting Tolosa-Hunt Syndrome, Hypopituitarism and Diabetes Insipidus: A Case Report.

Hama, Seiji; Arita, Kazunori; Kurisu, Kaoru; Sumida, Masayuki; Kurihara, Kanji

doi:10.1507/endocrj.43.503

Cited by 11 publications

(5 citation statements)

References 27 publications

(18 reference statements)

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“…Chronic parasellar inflammation such as those in hypertrophic pachymeningitis, cavernous sinusitis or Tolosa-Hunt syndrome have been reported to be accompanied by hypopituitarism and/or diabetes insipidus [40]. We have previously reported three such cases but did not measure the subjects' IgG or IgG4 levels [41].…”

Section: ) Effect Of Glucocorticoid Therapymentioning

confidence: 99%

Pituitary and Stalk Lesions (Infundibulo-hypophysitis) Associated with Immunoglobulin G4-related Systemic Disease: an Emerging Clinical Entity

et al. 2009

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Abstract. Inflammatory lesions of the pituitary gland are rarely encountered. Recently, the concept of immunoglobulin G4 (IgG4)-related systemic disease was proposed in Japan, and more than 20 cases have been reported as possibly associated with infundibulo-hypophysitis since 2000. We herein review such case reports in the published literature and in the abstracts of scientific meetings. Almost all cases involved middle-aged to elderly men presenting with various degrees of hypopituitarism and diabetes insipidus and demonstrating a thickened pituitary stalk and/or pituitary mass. These structures shrank remarkably in response to glucocorticoid therapy, even in a lower dose range similar to that prescribed as a replacement for adrenocortical insufficiency. Some of the anterior pituitary insufficiencies were also resolved by glucocorticoid administration. The presence of IgG4-related systemic disease and an elevated serum IgG4 level before glucocorticoid therapy were the main clues to a correct diagnosis of IgG4-related infundibulo-hypophysitis. Autoimmunity is suggested but not yet established to play a role in the pathogenesis for IgG4-related systemic disease. The fact that hypertrophic pachymeningitis and para-sinusitis accompanied some cases suggested that both sellar and parasellar structures are involved in the chronic inflammation. We therefore classify this disorder not as a variant form of primary autoimmune hypophysitis but as a secondary form of infundibulo-hypophysitis associated with IgG4-related systemic disease.

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Section: ) Effect Of Glucocorticoid Therapymentioning

confidence: 99%

Pituitary and Stalk Lesions (Infundibulo-hypophysitis) Associated with Immunoglobulin G4-related Systemic Disease: an Emerging Clinical Entity

et al. 2009

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“…Evidence of pituitary dysfunction in association with CS inflammation has been ascribed to spreading of inflammation from one site to another and termed parasellar chronic inflammatory disease. 72 …”

Section: R Esultsmentioning

confidence: 99%

Tolosa–Hunt Syndrome

Dutta

Anand

2021

Journal of Current Ophthalmology

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Purpose: To review the diagnostic criteria for Tolosa–Hunt syndrome (THS) and utility of recent modifications. Methods: We searched PubMed for keywords Tolosa Hunt and magnetic resonance imaging. We compared the three editions of International Classification of Headache Disorders and isolated case reports and case series with the assessment of cavernous internal carotid artery (ICA) caliber to find the prevalence of vascular anomalies. We also evaluated cases of THS with the involvement of extracavernous structures and the possible role of idiopathic hypertrophic pachymeningitis (HP). Cases diagnosed falsely as THS were also reviewed for the presence of atypical features and relevance of criterion D. We assessed nonconforming cases (those with normal neuroimaging benign THS) and idiopathic inflammatory orbital pseudotumor (IIPO). Results: Vascular abnormalities were found in 36.36% of THS cases. Benign THS may also show changes in ICA caliber. Evidence suggestive of idiopathic HP could be found in 57% of cases with the involvement of extracavernous structures, such as facial nerve and pituitary gland. Both THS and IIPO are steroid-responsive pathologies with similar clinical and radiological features. False-positive diagnosis of THS results from early labeling, based solely on clinical features and symptom resolution after steroid therapy. Conclusions: Benign THS may be a result of limitation of resolution of available neuroimaging technique or early testing. Early and late vascular changes can be seen in both THS and its benign variant; some of them are not innocuous. THS may be considered a type of focal idiopathic HP. IIPO may represent an anterior variant of THS. In the absence of histopathological diagnosis, steroid-induced resolution of symptoms should be confirmed radiologically and followed-up.

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“…1) Histological studies of the abnormal lesions reveal non-specific inflammation in meninges, compatible with pachymeningitis. 3,4,8,9) Tolosa-Hunt syndrome is considered to represent some of the symptoms of idiopathic cranial pachymeningitis involving the cavernous sinuses and surrounding cranial nerves. 9) The differential diagnosis of Tolosa-Hunt syndrome should include other paracavernous diseases, such as pituitary macroadenoma with cavernous sinus infiltration, paracavernous meningioma, Meckel's cave neurinoma, suprasellar epidermoid tumor, leptomeningeal metastases, granulomatous pachymeningitis sequelae, and aneurysm causing compression of the cavernous sinus.…”

Section: Discussionmentioning

confidence: 99%

“…3,5,8) All patients presented with severe panhypopituitarism. Histological examinations of the pituitary gland showed marked increases of collagen fibers and inflammatory cells in the dura mater and the entire pituitary gland.…”

Section: Discussionmentioning

confidence: 99%

Granulomatous Pachymeningitis Around the Sella Turcica (Tolosa-Hunt Syndrome) Involving the Hypophysis-Case Report-

Kita

Tachibana

Nagai

et al. 2007

Neurol. Med. Chir.(Tokyo)

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A 50-year-old female presented with right painful abducens nerve palsy persisting for 4 months and mild panhypopituitarism with diabetes insipidus for 6 months. T 1 -weighted magnetic resonance (MR) imaging of the sellar region showed a homogeneously enhanced mass lesion in the right cavernous sinus which seemed to extend from the swollen pituitary gland. T 2 -weighted MR imaging clearly showed the mass in the right cavernous sinus and the thickened dura mater of the sellar floor as hypointense, and the enlarged pituitary gland as isointense. Biopsy of the thickened dura mater and swollen pituitary gland was performed via the transsphenoidal approach. Histological examination revealed inflammation and collagen fiber formation in these regions. The diagnosis was secondary panhypophysitis resulting from granulomatous pachymeningitis involving the cavernous sinus (Tolosa-Hunt syndrome). Corticosteroid therapy was begun after the biopsy. Her periorbital pain and diplopia were relieved, but diabetes insipidus persisted. Follow-up MR imaging showed a decrease in the volumes of the pituitary gland and the mass in the cavernous sinus.

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Parasellar Chronic Inflammatory Disease Presenting Tolosa-Hunt Syndrome, Hypopituitarism and Diabetes Insipidus: A Case Report.

Cited by 11 publications

References 27 publications

Pituitary and Stalk Lesions (Infundibulo-hypophysitis) Associated with Immunoglobulin G4-related Systemic Disease: an Emerging Clinical Entity

Pituitary and Stalk Lesions (Infundibulo-hypophysitis) Associated with Immunoglobulin G4-related Systemic Disease: an Emerging Clinical Entity

Tolosa–Hunt Syndrome

Granulomatous Pachymeningitis Around the Sella Turcica (Tolosa-Hunt Syndrome) Involving the Hypophysis-Case Report-

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