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Nine cases of symptomatic Rathke's cleft cyst are reported. Their most frequent signs and symptoms included headache, chiasmal syndrome and hypopituitarism, while one of the cases developed a sudden onset of headache and vomiting following diabetes insipidus. Endocrinological findings showed a decreased ACTH, gonadotropin and growth hormone more frequently while there were 2 cases of hyperprolactinemia and 1 case of diabetes insipidus. In a neuroradiological examination, a plain skull X-ray showed 5 cases of ballooning of the sella turcica, and a CT scan demonstrated a low to high density of the cyst and 2 cases of marginal enhancement of the cyst. MRI mostly demonstrated a well delineated mass at the sella extending mostly into the suprasellar region and a low to high intensity of the cyst in the T1-weighted image. Two cases were marginally enhanced after gadolinium DTPA administration. The pathological examination, done on 6 cases, showed either single or multiple layers of the epithelium which were mostly ciliated. The epithelium was positive in PAS and Alcian blue in all cases and a histochemical examination showed 3 cases to be positive in EMA and 2 cases positive in CEA. A resection of the cyst wall and an opening of the cyst is thus recommended in symptomatic cases. Therefore, the transsphenoidal approach should be the choice of treatment in an intra- and suprasellar extension of the cysts with sellar enlargement.(ABSTRACT TRUNCATED AT 250 WORDS)
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PMID:[Symptomatic Rathke's cleft cyst: a clinicopathologic study of 9 cases]. 816 47

A case of lymphocytic hypophysitis is described in a patient presenting with panhypopituitarism 8 years after her last childbirth. The patient developed headache, vomiting, and diplopia (due to palsy of the right lateral rectus muscle) 7 months after delivery of her last baby. The diplopia disappeared after a few days with symptomatic treatment, and the headache and vomiting decreased in intensity with analgesic therapy. Eight years later the patient developed symptoms suggestive of hypoadrenalism, hypothyroidism, and amenorrhea. Investigations revealed panhypopituitarism with a pituitary mass lesion. Repeat evaluation 1 year later demonstrated no change in the size of the pituitary gland. The patient underwent transsphenoidal surgery with a provisional diagnosis of pituitary adenoma. Histological examination of the resected gland revealed evidence of lymphocytic hypophysitis. Symptoms suggestive of a pituitary mass lesion were noted during the peripartum period, but features of hypopituitarism developed much later. Such a long latent period has not been reported before. This report also highlights the fact that glandular enlargement may persist for many years after the onset of lymphocytic hypophysitis.
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PMID:Lymphocytic hypophysitis. Case report. 816 36

A 21-year-old man was hospitalized with complaints of headache, nausea, polyuria, reduced body hair and reduced libido. Plain CT scan and MRI revealed multiple tumors in the pineal and suprasellar regions, and in the dorsal aspect of the medulla oblongata. Endocrinological examination showed hypopituitarism due to hypothalamic dysfunction. Replacement therapy with hydrocortisone was started preoperatively. Suprasellar tumor was explored. Postoperative CT scan and MRI showed marked diminishment of not only suprasellar tumor, but also tumors in the two other regions. These tumors were supposed to be sensitive to corticosteroid hormone. Histopathological diagnosis was T cell type malignant lymphoma. The patient was followed up for 10 months post-operatively with no recurrence on CT scan. Primary intracranial malignant lymphoma is not a rare disease today. However, primary intracranial T cell type lymphoma is extremely rare. Hypopituitarism due to suprasellar malignant lymphoma is also rare, and only 3 such cases have been reported previously. In our case, the tumor was located in the pineal and suprasellar regions, and the dorsal aspect of the medulla oblongata. Such disseminated malignant lymphoma as ours shows radiological resemblance to germ cell tumor. No such diseminated malignant lymphoma has been reported previously. We think that, in its radiological and clinical features, our case is very suggestive of primary intracranial malignant lymphoma.
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PMID:[A case of primary intracranial T cell type malignant lymphoma, radiologically resembling germ cell tumor and presenting hypopituitarism]. 839 52

Granular cell tumours of neurohypophysis are rare. These tumours are more often encountered as incidental autopsy findings seen in up to 17% of unselected adult autopsy cases. There are few reports of parasellar granular cell tumours large enough to cause symptoms. We present three cases of neurohypophysis granular cell tumour and a review of the literature. In one patient, the asymptomatic granular cell tumour was incidentally discovered at surgical removal of a corticotroph microadenoma. The remaining 2 patients had a symptomatic tumour which caused neurological symptoms such as visual disturbance and headaches and endocrine disorders such as hypopituitarism or hyperprolactinaemia. In these 2 cases, computerized tomography showed a well-circumscribed, contrast-enhanced, intrasellar and suprasellar mass. Magnetic resonance imaging demonstrated an isointense gadolinium-enhanced mass in T1-weighted images. Transsphenoidal partial resection was performed and histology was interpreted as a granular cell tumour. The immunohistochemical study was positive for glial fibrillary acidic protein (GFAP) and neuron specific enolase (NSE) in 1 of the 2 tumours and positive for S100 protein and vimentin in both tumours but negative for CD68. The histogenesis of neurohypophysis granular cell tumours is still controversial but ultrastructural and immunohistochemical studies support the theory that they may arise from pituicytes, the glial cells of neurohypophysis. Management of these benign, slow-growing, tumours is based mainly on neurosurgical resection. Data from the literature do not support a beneficial effect of postoperative radiation therapy on postoperative recurrences.
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PMID:[Granular cell tumors. Rare tumors of the neurohypophysis]. 854 14

Rathke's cleft cysts (RCCs) are considered to arise from the remnants of Rathke's pouch, an invagination of the stomodeum. They are classically described as benign epithelium lined intrasellar cysts containing mucoid material, and also found in 2-33% of routine autopsy series. The most common presenting symptoms are visual impairment, hypothalamic dysfunction, hypopituitarism and headache. Diabetes insipidus has been described in patients with RCC. Very few cases presented with only diabetes insipidus in adults. To our knowledge, our patient is the first case of RCC presenting with only diabetes insipidus in childhood. A 9-year-old girl presented with diabetes insipidus. The physical, neurological and endocrinological examinations were normal, except for diabetes insipidus. Magnetic resonance imaging scan revealed a hyperintense lesion with supra sellar extension in the posterior pituitary both on T1 and T2 weighted images. Subtotal excision of RCC was performed via transsphenoidal surgery. However, diabetes insipidus persisted after the surgery.
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PMID:A case of Rathke's cleft cyst presenting with diabetes insipidus. 859 99

A 16-year-old male complained of a headache and a high fever followed by polyuria. The endocrinological studies showed he had hypopituitarism and central diabetes insipidus, and magnetic resonance imaging (MRI) revealed a pituitary mass. Diabetes insipidus gradually improved and hydrocortisone treatment was begun at three months after onset, but a month later painless thyroiditis developed. An MRI demonstrated a spontaneous shrinkage of the pituitary mass nine months after onset. Lymphocytic hypophysitis followed by painless thyroiditis was the most probable diagnosis, although it is very uncommon especially among men.
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PMID:Hypopituitarism associated with transient diabetes insipidus followed by an episode of painless thyroiditis in a young man. 868 Jan 2

Thyrotropin (TSH)-secreting pituitary tumors may be found in two opposite clinical situations: the hyperthyroidism secondary to thyrotroph adenomas, also called central hyperthyroidism, and the long-standing primary hypothyroidism which can be accompanied by a compensatory pituitary enlargement. TSH-secreting pituitary adenomas belong to the syndromes of "inappropriate secretion of TSH" (IST). The adjective "inappropriate" indicates the lack of the expected suppression of TSH secretion when free thyroid hormone levels are actually elevated, as in the other forms of thyrotoxicosis. Moreover, TSH-omas have to be differentiated from the non-neoplastic form of IST which is due to resistance to thyroid hormone. Differently, pituitary hyperplasia, which is reversible on thyroid hormone replacement, is the more frequent cause of a pituitary mass occurring in the context of untreated primary hypothyroidism. Failure or delay in the recognition of the above clinical situations may cause dramatic consequences, such as unnecessary pituitary surgery in hypothyroid patients or improper thyroid ablation in those with central hyperthyroidism. In contrast, early diagnosis and proper treatment of TSH-secreting pituitary tumors prevents the appearance of signs and symptoms of mechanical compression of the adjacent structures by the expanding tumor mass (visual field defects, headache and hypopituitarism).
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PMID:Thyrotropin-secreting pituitary tumors in hyper- and hypothyroidism. 876 13

Pituitary metastasis are rare but represent an important differential diagnosis of intrasellar tumorous syndromes. We report one case of intrasellar metastasis of a bronchial adenocarcinoma in a 50 year old woman. Clinical syndrome associated a tumorous syndrome (cephalalgia, bitemporal hemianopsia) and an anterior and posterior hypopituitarism biologically proven. A large intra and suprasellar mass which compressed the optic chiasma and highly enhanced after gadolinium injection was found by nuclear magnetic resonance imaging. The surgical biopsy displayed a pituitary metastasis of a right inferior lobar bronchial adenocarcinoma with bone secondary localizations. Cerebral radiotherapy and corticotherapy allowed recovery of visual loss but did not prevent rapid death. The clinical and radiological features which may evoke an intrasellar metastasis were: the tumorous syndrome associated with or revealed by diabetes insipidus, loss of spontaneous hypersignal of the neurohypophysis in nuclear magnetic resonance imaging, bulking pituitary stalk, bilobar character of the mass which is in favour of rapid cell proliferation, postero-superior extension, lowering of the V3 floor and very strong and homogeneous signal after gadolinium injection.
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PMID:[Diabetes insipidus revealing pituitary metastasis of bronchial carcinoma]. 899 Nov 5

In 1992, we reported a lymphocytic adenohypophysitis (LIH) (Neurol Med Chir). We considered this case unusual in that the case was that of a menopausal female and that it was accompanied with diabetes insipidus as classical lymphocytic adenohypohysitis (LAH). Subsequently, Ahmed reported two cases which presented a similar pathological manifestation, except for necrosis, as did our case and named them "necrotizing infundibulo-hypophysitis." Recently we encountered another similar case, which is reported hereunder. A female, 34 years of age, had suffered from headache, polyuria, and amenorrhea. CT scan showed a pituitary mass, and pituitary tumor was surgically removed transcranially at a local hospital. The pathological examination revealed the findings of chronic inflammation and necrosis. One month after the operation, however, she was an in-patient again under the suspicion of meningitis for fever and, when antibiotic therapy at the local hospital resulted in no improvement, she was referred to our hospital. Endocrinological studies showed low FSH, LH, ACTH and plasma cortisol level. Antibodies of serum to RNP, Sm, mitochondria, nucleus, AChR, and DNA were all negative. Because of an intrasellar mass with suprasellar extension on MRI, transsphenoidal operation was conducted four months after the initial operation. The pathological examination revealed the infiltration of lymphocytes, plasma cells, and foamy macrophages, and necrosis. After this operation, the headache was cured and the patient was discharged. Two months subsequent to the second operation, headache recurred and temporal upper quadrantic anopsia was noted. An enlarged tumor was found, but prednisolone worked to cure the pain and the visual field defect was found to have been remedied. The patient's diabetes insipidus is presently persisting, and she still relies on the use of desmopressin acetate and is still in need of cortisol replacement therapy. Including our cases, ten cases of lymphocytic hypophysitis, not related to pregnancy or delivery but with diabetes insipidus, have been reported. Several clinical and anatomical features distinguish these 10 cases from classical LAH. The classical LAH was predominantly related to pregnancy or delivery. However 6 of 10 cases were male in LIH. LAH related to pregnancy or delivery does not accompany diabetes insipidus, but all reported cases of LIH had a diabetes insipidus. Visual field and/or ocular movement disturbance are LAH's chief complaints (15 out of 25 cases) but visual field disturbance seldom occurs in LIH (1 out of 10 cases). Hypopituitarism is more serious in LAH, and 4 cases became fatal from an adrenal crisis. Anatomically, inflammatory change of LIH is located anterior and posterior to the pituitary gland and extends to the pituitary stalk and, at times, hypothalamus. On the other hand, LAH relates to pregnancy or delivery, the inflammatory change localizes to the adenohypophysis. Ahmed emphasized necrosis, while necrosis was not a prominent histological finding in LIH. Necrosis was noted only in 3 of 10 cases. To be stressed, rather, are the inflammatory changes seen on the neurohypophysis and the pituitary stalk, together with the characteristic diabetes insipidus. We believe, in view of the above, that what Ahmed named necrotizing infundibulo-hypophysitis should be named "LIH with diabetes insipidus." Whereas differential diagnosis is necessary between this said new disorder and the conventional LAH, we advocate that the latter, which is related to pregnancy or parturition but is free from neurohypophysitis be identified as "LAH related to pregnancy or delivery." With respect to treatment, steroid therapy is essential. If the symptoms do not improve, a transsphenoidal operation for diagnosis (LIH and LAH) and decompression (the case of LAH with visual or external ocular movement disturbance) is advisable. However, extensive surgery is not recommended, because per
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PMID:[Lymphocytic infundibulo-hypophysitis with diabetes insipidus as a new clinical entity: a case report and review of the literature]. 902 95

We reported a case of Rathke's cleft cyst (RCC) with a moving mass in the cyst. A fifty-eight-year-old woman complaining of headache was admitted to our hospital. She suffered from hyponatremia, hypothalamic hypopituitarism, but did not show any neurological deficits, nor visual field nor visual acuity disturbance. MRI revealed an intrasellar cyst including a mass shadow and the cyst did not compress the hypothalamus. Interestingly, this mass moved gradually from the floor to the posterior wall of the sella turcica according to changes in position from standing to supine. We operated to remove the cyst partially by transsphenoidal approach. Xanthochromic fluid flowed out from the cyst. In addition, a brownish globular mass, 6 mm in diameter, existed within the cyst without connection to the surrounding tissue. Pathological findings showed that the capsule consisted of cuboid epithelium and was partially stratified, which confirmed the diagnosis of Rathke's cleft cyst. However, the mass was homogeneously stained with H.E., containing no cells, cellular debris, nor connective tissue. PAS stain was negative. Though we could not clarify the material in the cyst, this case is very rarely one of Rathke's cleft cyst. This case demonstrated the possibility of hypothalamic hypopituitarism in spite of the cyst being almost located in the sella turcica. We surmise that a cyst near the stalk of the pituitary gland, even if it is not so large, might compress the portal vein, and thus the function of hypothalamus-pituitary axis.
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PMID:[A Rathke's cleft cyst with a moving mass in the cyst]. 902 96


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