A Case of Hypothalamic Panhypopituitarism with Empty Sella Syndrome: Case Report and Review of the Literature

Komada, Hisako; Yamamoto, Masaaki; Okubo, Saki; Nagai, Kazuo; Iida, Keiji; Nakamura, Takehiro; Hirota, Yushi; Sakaguchi, Kazuhiko; Kasuga, Masato; Takahashi, Yutaka

doi:10.1507/endocrj.k08e-214

Cited by 12 publications

(6 citation statements)

References 15 publications

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“…Empty sella is also an expected frequent abnormality found by imaging, with an incidence in autopsy series that approximates 20 % [ 24 ] . However, despite the fact that the reported rate of endocrine dysfunction (complete or partial hypopituitarism and/or hyperprolactinemia) can be as high 20-50 % in such cases [ 25 ] , none of the patients from the present series was found to have any pituitary functional derangement. On the other hand, the most common hormonal abnormality seen in AI is SCS accounting for a mean of 8 % in diff erent studies [ 26 ] .…”

contrasting

confidence: 64%

Incidence of Pituitary Incidentalomas in Patients with Adrenal Adenomas

Chrisoulidou

Alexandraki

Kita

et al. 2014

Exp Clin Endocrinol Diabetes

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With the advent of modern imaging modalities, endocrine incidentalomas are increasingly being discovered. We aimed to investigate the presence of pituitary incidentalomas (PI) in patients with adrenal incidentalomas (AI), and identify potential metabolic correlates in this cohort. 26 patients (18 females) with AI discovered on abdominal computerized tomography were studied. All patients underwent pituitary magnetic resonance imaging (MRI) and endocrine investigations to evaluate functional adrenal pathology, anterior pituitary hormonal status, insulin-resistance indices and presence of metabolic syndrome. Pituitary MRI revealed a microadenoma and a 4×5 mm cyst in 1 patient respectively, and an empty sella in 4 (2 partial) patients. Overall, 6/26 (23%) patients with an AI had evidence of pituitary imaging pathology but only 8% had a PI; none had any evidence of abnormalities in pituitary function. Subclinical hypercortisolism was the only hyperfunctional status detected in 4 patients with AI but was unrelated to the pituitary findings. No abnormality of insulin secretion and action was found between patients with or without pituitary pathology. In the present study 23% of patients with AI had some alteration in pituitary morphology, and 2 a PI without accompanying pituitary hormonal deficit or metabolic derangement. Further studies are required to address this issue and identify a potential pathogenetic mechanism.

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confidence: 64%

Incidence of Pituitary Incidentalomas in Patients with Adrenal Adenomas

Chrisoulidou

Alexandraki

Kita

et al. 2014

Exp Clin Endocrinol Diabetes

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“…The first case was reported in 1986 by Okada et al (22) in a 28-yr-old woman with clinically suspected hypophysitis and pituitary antibodies who developed empty sella 2 yr after diagnosis. Empty sella was present at diagnosis in two patients (23,24) and reported at some time during follow-up in the remaining 25 cases. Follow-up, which ranged from 0.25 (25) to 24 (26) years, was short in cases where empty sella developed after pituitary surgery and more prolonged in those where no mass-reducing treatment (either surgical or with high-dose steroids) was used (Supplemental Table 1).…”

Section: Discussionmentioning

confidence: 97%

From Pituitary Expansion to Empty Sella: Disease Progression in a Mouse Model of Autoimmune Hypophysitis

et al. 2011

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Lymphocytic hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volume then decreased gradually in the majority of cases (14 of 18, 78%), reaching empty sella values by d 300 after immunization. In a minority of cases (four of 18, 22%), the decrease was so rapid and marked to induce a central area of necrosis accompanied by hemorrhages, mimicking the condition known in patients as pituitary apoplexy. No radiological or pathological changes were observed in controls. Overall, these findings indicate that the evolution of hypophysitis is complex but can lead, through different routes, to the development of empty sella.

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“…Radiologically, empty sella is defined as partial when less than 50% of the sella is filled with cerebrospinal fluid (CSF) and pituitary gland thickness is ≥ 3 mm ( Figure 1A,B), or total when more than 50% of the sella is filled with CSF and the gland thickness is ≤2 mm in diameter (1,3). Although PES is often found incidentally during neuroradiological studies, it could be associated with serious clinical conditions including different degrees of hypopituitarism and neurological deficits (7,14). However, most of the studies evaluating endocrinological aspects of the PES are retrospective and up to date no study has compared the pituitary functions between total and partial PES, despite the opinion that there is no relationship between the pituitary gland thickness and pituitary hormone deficiency (7).…”

Section: Zuhur Ss Et Al: Pituitary Deficiency In Primary Empty Sellamentioning

confidence: 99%

Anterior pituitary hormone deficiency in subjects with total and partial primary empty sella: do all cases need endocrinological evaluation?

Zuhur

Kuzu²,

Öztürk³

et al. 2014

Turkish Neurosurgery

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AIm:To compare anterior pituitary functions between subjects with total and partial primary empty sella (PES) and to assess whether all cases with PES need endocrinological evaluation. mATERIAL and mETHods: Eighty-one subjects with PES (34 total and 47 partial) were included in the study. Basal anterior pituitary and its target hormones were assessed and those with low insulin like growth factor-1 and/or low basal cortisol levels underwent insulin tolerance test (ITT).REsuLTs: 67.4% of the subjects with total and 14.9% of those with partial PES had different degrees of hypopituitarism. However, the frequency of hypopituitarism was significantly higher in cases with total PES. The odds ratio (OR) and 95% confidence interval (CI) of secondary hypothyroidism, secondary adrenal, growth hormone and gonadotropin deficiency in subjects with total compared to those with partial PES were as follows: OR = 20.0, 95% CI 4.16 -95.9, OR = 2.4, 95% CI 1.34 -5.7, OR = 15.3, 95% CI 4.48 -52.6 and OR = 10.6, 95% CI 3.37 -33.5, respectively. CoNCLusIoN:A substantial number of subjects with PES, particularly those with total PES, have pituitary hormone deficiency, so regardless of the type of PES, all subjects must be promptly and carefully evaluated for anterior pituitary hormone deficiency.KEywoRds: Primary empty sella, Hypopituitarism, Clinical evaluation, MRI ÖZ AmAÇ: Bu çalışmada parsiyel ve total primer boş sella (PBS) olguları arasında ön hipofiz fonksiyonları bakımından karşılaştırma yapılması ve tüm olgularda endokrinolojik değerlendirme gerekliliğinin saptanması amaçlanmıştır. yÖNTEm ve GEREÇLER: PBS tanısı konan 81 olgu (34 total ve 47 parsiyel) çalışmaya dahil edildi. Tüm olguların bazal ön hipofiz ve bunların hedef hormonlarının düzeylerine bakıldı, insülin benzeri büyüme faktörü-1 ve/veya bazal kortizol düzeyi düşük olan olgulara insülin tolerans testi (İTT) uygulandı.BuLGuLAR: Total PBS'sı olan olguların % 67,4'ü ve parsiyel PBS'sı olan olguların %14,9'unda değişik düzeylerde hipopituitarizm saptandı. Ancak total PBS'sı olan olgularda hipopituitarizm belirgin olarak daha yüksek bulundu. Sekonder hipotiroidizm, sekonder adrenal yetmezlik, büyüme hormonu ve gonadotropin eksikliklerinin total PBS'sı olan olgularda parsiyel PBS'sı olan olgulara kıyasla odds oranları (OR) ve %95 güven aralıkları (%95 GA) sırasıyla OR = 20,0; %95 GA 4,9;OR = 2,4; %95 GA 1,7;OR = 15,3; %95 GA 4,[48][49][50][51][52]6 ve OR = 10,6; %95 GA 3,37 -33,5 bulundu. soNuÇ: Bu çalışmanın sonuçları özellikle total PBS'sı olan olgular olmak üzere PBS'sı olan olguların önemli bir kısmında değişik düzeylerde hipopituitarizmin varlığını göstermektedir. Bu nedenle PBS'nın tipine bakılmaksızın tüm olgular ön hipofiz hormon yetmezliği bakımından en kısa sürede uygun bir şekilde değerlendirilmelidirler.ANAHTAR sÖZCÜKLER: Primer boş sella, Hipopituitarizm, Klinik değerlendirme, MRG

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A Case of Hypothalamic Panhypopituitarism with Empty Sella Syndrome: Case Report and Review of the Literature

Cited by 12 publications

References 15 publications

Incidence of Pituitary Incidentalomas in Patients with Adrenal Adenomas

Incidence of Pituitary Incidentalomas in Patients with Adrenal Adenomas

From Pituitary Expansion to Empty Sella: Disease Progression in a Mouse Model of Autoimmune Hypophysitis

Anterior pituitary hormone deficiency in subjects with total and partial primary empty sella: do all cases need endocrinological evaluation?

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