Pseudotumoral Lymphocytic Hypophysitis Successfully Treated by Corticosteroid Alone

Beressi, Nathalie; Cohen, R.; Beressi, J.P.; Dumas, Jean-Luc; Legrand, Maggy; Iba-Zizen, M. T.; Modigliani, E

doi:10.1227/00006123-199409000-00020

Cited by 76 publications

(42 citation statements)

References 12 publications

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“…It is generally believed that conservative management is appropriate in the majority of patients with lymphocytic hypophysitis since spontaneous resolution of both the mass and the hypopituitarism has been reported [20][21][22][23]. However, the experience of this case implies that a diagnosis of lymphocytic hypophysitis from PPL is challenging.…”

Section: Discussionmentioning

confidence: 88%

Primary Pituitary Lymphoma Presenting as Hypophysitis

et al. 2005

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Abstract.A 47-year-old man had suffered from prolonged fever for two months without clinical evidence of infection. Blood biochemistry and endocrine dysfunction indicated that he had pituitary insufficiency. Thorough whole body imaging studies merely identified a 22 × 14 mm mass lesion in the sella turcica. Tumor pathology and special cell marker study revealed the infiltration of atypical T-lymphoid cells and concomitant presence of some B-lymphoid cells. The fever subsided gradually following subtotal tumor resection and steroid supplementation. However, the mass lesion had invaded the cavernous sinus and optic chiasma shortly after surgery. Six months after his initial visit, metastasis lesions in the liver, the left adrenal gland, and retroperitoneal lymph nodes were discovered. In contrast to cells in the pituitary, the pathological investigation of the liver mass confirmed it to be exclusively of T-cell origin. Therefore, it is plausible that the pituitary dysfunction was related to an inflammatory process, namely hypophysitis, as well as the T-cell lymphoma. This case exemplifies the rarely noted condition of primary pituitary lymphoma with concomitant hypophysitis. Clinical diagnosis is indiscernible until the occurrence of systemic tumor metastasis.

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Section: Discussionmentioning

confidence: 88%

Primary Pituitary Lymphoma Presenting as Hypophysitis

et al. 2005

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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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“…For this reason, it has been referred to as pseudotumoural LH [14]. A subtle difference between an adenoma and LH is the hierarchy of endocrine loss: in LH there is a predilection for destruction of ACTH and TSH secreting cells unlike that in an adenoma where ACTH secretion is last to be lost.…”

Section: Discussionmentioning

confidence: 99%

Lymphocytic Hypophysitis Occurring Simultaneously with a Functioning Pituitary Adenoma

et al. 2008

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Abstract. Lymphocytic Hypophysitis (LH) is a rare and previously under-recognised disorder, most commonly affecting young females in the post-partum period. It presents clinically with symptoms and signs related to either a pituitary mass or hypopituitarism, frequently mimicking a pituitary adenoma; the diagnosis of LH can only be made histologically with the presence of a dense lymphocytic infiltration usually confined to the anterior pituitary. We present two case histories of patients who presented with symptoms suggestive of a functioning pituitary adenoma who also had concomitant LH confirmed histologically. The first case was a 39 year old lady, with a history of primary hypothyroidism, who presented with weight gain and hirsutism and clinical and biochemical features of Cushing's syndrome. The second case was a 61 year old male, also with a history of primary hypothyroidism, who presented with visual field loss and biochemically with hyperprolactinaemia. In both patients, magnetic resonance (MR) imaging of the pituitary demonstrated an enlarged partially cystic pituitary mass with slight suprasellar extension. Both patients were treated surgically with transphenoidal drainage and excision and histological examination of the surgical specimens demonstrated a mixture of pathologies with fragments of adenohypophyseal tissue (staining positive for ACTH and prolactin respectively) with a dense chronic inflammatory cell infiltrate suggestive of LH in nearby normal anterior pituitary. In both cases a joint diagnosis of a functioning pituitary adenoma with LH was made. There have been only several reported cases of this combination of pathologies but LH even in isolation is becoming increasingly recognised.Key words: Lymphocytic hypophysitis, Pituitary adenoma, Prolactinoma, Cushing's disease (Endocrine Journal 55: 729-735, 2008) Case 1A 39 year old lady presented with a two year history of general lethargy, weight gain and hirsutism of the face and breasts. She had a history of hypothyroidism, assumed to be auto-immune although the general practitioner had not tested thyroid antibodies. She had complained of irregular periods since a transcervical endometrial resection for endometriosis and subsequently had commenced hormone replacement therapy because of hot flushes and sweating. She had had 2 children with no obstetric complications.On clinical examination she had a round plethoric face with hirsutism affecting the chin, cheeks and breasts with thinning of her skin. Blood pressure was 140/92 mmHg with no evidence of hypertensive retinopathy on fundoscopy. Systemic examination, including visual field testing on confrontation, was normal.

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Pseudotumoral Lymphocytic Hypophysitis Successfully Treated by Corticosteroid Alone

Cited by 76 publications

References 12 publications

Primary Pituitary Lymphoma Presenting as Hypophysitis

Primary Pituitary Lymphoma Presenting as Hypophysitis

Primary hypophysitis: clinical-pathological correlations

Lymphocytic Hypophysitis Occurring Simultaneously with a Functioning Pituitary Adenoma

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