Stereotactic radiotherapy for the treatment of lymphocytic hypophysitis

Selch, Michael T.; DeSalles, Antonio; Kelly, Daniel F.; Frighetto, Leonardo; Vinters, Harry V.; Cabatan-Awang, Cynthia; Wallace, Robert E.; Solberg, Timothy D.

doi:10.3171/jns.2003.99.3.0591

Cited by 54 publications

(34 citation statements)

References 25 publications

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“…Selch et al reported a good efficacy of stereotactic radiotherapy for two cases of recurrent lymphocytic hypophysitis [14]. Although we have no experience with stereotactic radiotherapy in the treatment in granulomatous hypophysitis, we believe this treatment modality may present a noninvasive treatment option for patients with granulomatous hypophysitis, particularly if the disease is recurrent after surgery or resistant to corticosteroids.…”

Section: Managementmentioning

confidence: 98%

“…While the standard management for granulomatous hypophysitis remains controversial, accepted regimens includes transsphenoidal removal of the intrasellar mass and administration of corticosteroid therapy. Stereotactic radiotherapy has also been successfully used [14]. As we known, the important reviews and case reports on granulomatous hypophysitis are only involved in one or two cases after since 1980s [2], however, only one single center in India reported six cases with granulomatous hypophysis diagnosed after biopsy in 2004 [2].…”

Section: Introductionmentioning

confidence: 99%

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Granulomatous hypophysitis: experience with eight surgical cases of a single center

et al. 2016

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Background: Although primary hypophysitis is not uncommon, a granulomatous etiology of hypophysitis is relatively rare. Radiological and diagnostic characteristics of several isolated cases have been described to date. However, to the best of our knowledge, surgical treatment of eight cases of granulomatous hypophysitis confirmed by histopathology has not yet been reported. Methods: A retrospective study was performed to review cases of granulomatous hypophysitis from November 2003 to November 2013 in our neurosurgical department. Only eight out of 1345 operations were diagnosed with granulomatous hypophysitis. Results: The cohort included two men and six women. The most common manifestations reported were headache, fatigue and polyuria. All six female patients presented with either menstrual disorders or amenorrhea. Seven of eight patients suffered polyuria. Two patients suffered bitemporal hemianopsia. One was accompanied with eosinophilic granuloma of the skull. Prolactin levels were increased in six patients, four of them were noted to have thyroid axis imbalances, one was noted to be deficient in the gonadotropin axis and two were deficient in the adrenal axis. The lesion demonstrated homogenous enhancement and an abnormally thickened pituitary stalk. A histological diagnosis of granulomatous hypophysitis was established in all eight patients, who were treated surgically. Characteristic granulomas formed by epithelioid histiocytes and multinucleated giant cells were observed. Conclusion: Pure glucocorticoid therapy is less effective than surgical intervention and has not been recommended for the treatment of granulomatous hypophysitis. Although replacement therapy can relieve associated symptoms, we conclude surgical intervention, particularly minimally invasive surgery via a transsphenoidal approach, to be mandatory in establishing a diagnosis and reducing the size of the inflammatory mass. Adjuvant corticosteroids therapy is recommended.

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Section: Managementmentioning

confidence: 98%

Section: Introductionmentioning

confidence: 99%

Granulomatous hypophysitis: experience with eight surgical cases of a single center

et al. 2016

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“…Its treatment at the moment is only symptomatic and aimed at reducing the size of the enlarged pituitary. Pituitary surgery (5,6), lympholytic drugs such as glucocorticoids (7,8), azathioprine (9), and methotrexate (10), or radiotherapy (11) are the current approaches.…”

Section: Introductionmentioning

confidence: 99%

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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“…This is a noninvasive option of treatment in recurrent LH and corticosteroid-resistant cases of LH. Unfortunately, there is no experience with stereotactic radiotherapy in the management of postsurgical recurrence of GH (3,13).…”

Section: Discussionmentioning

confidence: 99%

Primary Granulomatous Hypophysitis Presenting with Secondary Hypothyroidism

Antonios

Washington

et al. 2018

AACE Clinical Case Reports

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Objective: We present a case of a 45-year-old woman with history of Graves disease treated with thyroidectomy who developed panhypopituitarism due to granulomatous hypophysitis.Methods: The details of the case presentation, evaluation, diagnosis, and treatment are reviewed. On routine follow-up visits for thyroid condition, the patient complained of symptoms of hyperthyroidism, despite being on a previously adequate dose of levothyroxine. Work-up led to the diagnosis of hypophysitis.Results: The patient responded to corticosteroid therapy, but 5 months later, magnetic resonance imaging of the brain showed recurrence of the disease. Eventually, the patient underwent partial transsphenoidal hypophysectomy, later developed recurrence of disease, and had total hypophysectomy. The postoperative pathology report was consistent of granulomatous hypophysitis.Conclusion: Granulomatous hypophysitis is an uncommon inflammatory disease affecting the pituitary. It is commonly misdiagnosed as a macroadenoma because of its clinical presentation. (AACE Clinical Case Rep. 2018;4:e329-e333) Abbreviations: FT4 = free thyroxine; GH = granulomatous hypophysitis; LH = lymphocytic hypophysitis; MRI = magnetic resonance imaging; TSH = thyroid-stimulating hormone CASE REPORTHypophysitis is a rare inflammatory process of the pituitary gland that mimics pituitary macroadenomas. It can be idiopathic or manifest as a part of a systemic autoimmune disease. Granulomatous hypophysitis (GH) and lymphocytic hypophysitis (LH) are the most common subtypes reported in literature (1-3). We present a case of a 45-year-old female with past medical history of hypothyroidism after total thyroidectomy for Graves disease and surgical history of hysterectomy. On presentation, she complained of hyperthyroid symptoms (e.g., fatigue, excess sweating, hot flashes, presyncope, weight loss, and headaches), despite being on a previously adequate levothyroxine dose. Two months prior, her thyroid-stimulating hormone (TSH) and free thyroxine (FT4) were normal. Her physical exam was unremarkable except for a surgically absent thyroid. Laboratory evaluation revealed TSH <0.006 mU/L (normal range, 0.34 to 5.6 mU/L) and FT4 of 1.1 ng/dL (normal range, 0.58 to 1.64 ng/dL); thus, her levothyroxine dose was decreased. Despite this, she continued to have symptoms and low TSH. Secondary hypothyroidism was suspected, as TSH was undetectable and FT4 0.47 ng/dL off levothyroxine. Severe headache, insomnia, and worsening fatigue developed, so imaging was done and magnetic resonance imaging (MRI) revealed a large bilobed enhancing pituitary mass with suprasellar extension and thickening of the pituitary stalk (Fig. 1). Laboratory results showed low am cortisol, estrogen, follicle-stimulating hormone (FSH), insulin-like growth factor See accompanying article, p. e350.

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Stereotactic radiotherapy for the treatment of lymphocytic hypophysitis

Cited by 54 publications

References 25 publications

Granulomatous hypophysitis: experience with eight surgical cases of a single center

Granulomatous hypophysitis: experience with eight surgical cases of a single center

Primary hypophysitis: clinical-pathological correlations

Primary Granulomatous Hypophysitis Presenting with Secondary Hypothyroidism

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