Neoplastic Etiology and Natural Course of Pituitary Stalk Thickening: A Systematic Review and Meta-analysis

Author(s):  
Dong Yeong Kim ◽  
Pyeong Hwa Kim ◽  
Ah Young Jung ◽  
Jin-Ho Choi ◽  
Young Ah Cho ◽  
...  

Abstract Context Pituitary stalk thickening (PST) is often identified on magnetic resonance imaging (MRI), either incidentally or during diagnostic work-up of hypopituitarism. However, the neoplastic etiology and natural course of PST are not fully understood, although this knowledge is required to establish diagnostic and surveillance strategies. Objectives To investigate the neoplastic etiology and natural course of PST. Methods MEDLINE/PubMed and EMBASE databases were searched up to February 2021 to identify original research investigating the etiologies of PST. The proportion of neoplastic etiology in patients with PST was meta-analytically pooled. Supplementary analysis exploring factors suggesting neoplasm was also performed. For initially indeterminate cases without confirmed diagnosis, the proportion of patients showing progression of PST during follow-up was evaluated. Results Eighteen studies covering 1368 patients with PST were included. The pooled proportion of neoplasm was 45.2% (95% CI, 33.3–57.8%), with substantial heterogeneity across studies (I 2=93%). The most common neoplasm was germ cell tumor (14.0% of study population), followed by Langerhans cell histiocytosis (10.2%) and metastasis (4.7%). The studies on pediatric populations and those with >50% of patients having at least one pituitary hormone deficiency tended to show a higher proportion of neoplasm. The pituitary stalk was thicker in neoplasms, but the difference was not significant (pooled mean difference, 2.08 mm; P=0.08). In initially indeterminate cases, 18.5% (95% CI, 7.6–38.3%) showed progression of PST during follow-up. Conclusion PST was commonly confirmed to be neoplastic, especially in pediatric populations. As isolated PST frequently progresses, follow-up imaging is essential in initially indeterminate cases.

2016 ◽  
Vol 2016 ◽  
pp. 1-7 ◽  
Author(s):  
Wendong Liu ◽  
Limin Wang ◽  
Minghua Liu ◽  
Guimei Li

Objective. In pediatric central diabetes insipidus (CDI), etiology diagnosis and pituitary function monitoring are usually delayed. This study aimed to illustrate the importance of regular follow-up and pituitary function monitoring in pediatric CDI.Methods. The clinical, hormonal, and neuroradiological characteristics of children with CDI at diagnosis and during 1.5–2-year follow-up were collected and analyzed.Results. The study included 43 CDI patients. The mean interval between initial manifestation and diagnosis was 22.29 ± 3.67 months (range: 2–108 months). The most common complaint was polyuria/polydipsia. Causes included Langerhans cell histiocytosis, germinoma, and craniopharyngioma in 2, 5, and 4 patients; the remaining were idiopathic. No significant changes were found during the 1.5–2 years after CDI diagnosis. Twenty-three of the 43 cases (53.5%) had ≥1 anterior pituitary hormone deficiency. Isolated growth hormone deficiency was the most frequent abnormality (37.5%) and was not associated with pituitary stalk diameter. Multiple pituitary hormone deficiencies were found in 8 cases with pituitary stalk diameter > 4.5 mm.Conclusion. Diagnosis of CDI is usually delayed. CDI with a pituitary stalk diameter > 4.5 mm carries a higher risk of multiple pituitary hormone deficiencies. Long-term MRI and pituitary function follow-ups are necessary for children with idiopathic CDI.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
kehinde Matilda folawewo ◽  
MaKenzie Hodge ◽  
Nada Osman ◽  
Anteneh Woldetensay Zenebe ◽  
Vijaya A Ganta ◽  
...  

Abstract Pituitary stalk interruption syndrome (PSIS) is a congenital disorder of the pituitary gland. Symptoms at presentation may vary widely as this disease presents along a spectrum which includes; ectopic posterior pituitary, interrupted pituitary stalk or aplasia and hypoplasia of the pituitary gland. It is a heterogeneous disorder in terms of radiologic and clinical presentation. It can present clinically as an isolated pituitary hormone deficiency (most common being growth hormone deficiency) or as multihormonal deficiencies. CASE PRESENTATION Patient is a 34-year-old woman with history of primary amenorrhea who was evaluated by a gynecologist and was prescribed oral contraceptive pills which lead to her having a menstrual bleed for the first time in her life. She denied any difficulty with smell. She had undergone normal psychomotor milestones and highest level of education was high school. She had normal puberty with normal pubic and axillary hair growth, normal breast development but no menarche. Of note, patient has a short stature, height is 4 feet and 11 inches, and her biological parents are of normal adult height On evaluation, patient had normal am cortisol, prolactin and thyroid function tests. IGF-1 was significantly low for her age, FSH and LH were inappropriately low for her low estradiol level suggesting hypogonadotropic hypogonadism. Patient subsequently had an MRI of the pituitary and DXA scan. MRI findings were suggestive of ectopic neurohypophysis. DXA scan showed significant reduction in bone mineral density for age. Patient is currently being treated with hormonal replacement which is the main modality of treatment for ectopic neurohyphysis. She will need long term follow up as disease progression to pan-hypopituitarism is common. CONCLUSION PSIS is a rare syndrome with different phenotypic presentation depending on when the diagnosis is made; therefore, adequate follow up is indicated as the disease can progress from a single hormonal deficiency to pan-hypopituitarism.


1997 ◽  
Vol 82 (5) ◽  
pp. 1362-1367
Author(s):  
Sudha L. Mootha ◽  
Anthony J. Barkovich ◽  
Melvin M. Grumbach ◽  
Michael S. Edwards ◽  
Stephen E. Gitelman ◽  
...  

Abstract We report nine consecutive children and adolescents [five females and four males; aged 2 yr 8 months (m) to 18 yr 1 m] studied over the last 5 yr with idiopathic central diabetes insipidus. In addition to vasopressin deficiency, anterior pituitary hormone deficiencies were detected, either on evaluation at presentation or during follow-up studies over the following 3 yr. Four patients had an increased concentration of plasma PRL. One patient had multiple pituitary hormone deficiencies at diagnosis, and two others developed the same by 21 m of follow-up. Brain magnestic resonance imaging scans, performed at presentation, were originally interpreted as normal in four of nine patients, except for absence of the bright posterior pituitary signal; after retrospective review, two of nine were considered normal. All of the brain magnetic resonance imaging (MRI) scans showed positive findings by 14 m of follow-up. The first abnormal finding in all patients was isolated pituitary stalk thickening. Evaluation of cerebrospinal fluid (CSF) for hCG was positive in three of eight evaluated patients; the three positive CSF values were found at presentation and 3 and 9 m after presentation. All eight patients assessed were negative for CSF α-fetoprotein and cytology, and no patient had serum tumor markers. Transsphenoidal biopsy of the lesion in seven of nine patients showed a germinoma in six patients and inflammatory cells in one. The six patients with documented germinoma comprise 31% of the intracranial germinomas diagnosed in this age group at the University of California-San Francisco during the last 5 yr. The patient with mononuclear inflammatory cells on biopsy along with one other patient have had spontaneous resolution of their stalk thickening. So-called “idiopathic” central diabetes insipidus warrants close follow-up to determine the etiology, especially if anterior pituitary hormone deficiencies are detected. Normal brain MRI scans or scans that show isolated pituitary stalk thickening merit follow-up with serial contrast enhanced brain MRI for the early detection of an evolving occult hypothalamic-stalk lesion. CSF evaluation is recommended at presentation because elevated CSF hCG may precede MRI abnormalities.


2022 ◽  
Author(s):  
Steinunn Arnardóttir ◽  
Jacob Järås ◽  
Pia Burman ◽  
Katarina Berinder ◽  
Per Dahlqvist ◽  
...  

Objective: To describe treatment and long-term outcomes of patients with acromegaly from all health-care regions in Sweden. Design and Methods: Analysis of prospectively reported data from the Swedish Pituitary Register of 698 patients (51% females) with acromegaly diagnosed from 1991-2011. The latest clinical follow-up date was December, 2012, while mortality data were collected for 28.5 years until June, 2019. Results: The annual incidence was 3.7/million; 71% of patients had a macroadenoma, 18% had visual field defects, and 25% had at least one pituitary hormone deficiency. Eighty-two percent had pituitary surgery, 10% radiotherapy and 39% medical treatment. At the 5- and 10-year follow-ups, IGF-I levels were within the reference range in 69% and 78% of patients, respectively. In linear regression the proportion of patients with biochemical control including adjuvant therapy at 10 year follow-up increased over time with 1.23 % per year. The SMR (95% CI) for all patients was 1.29 (1.11-1.49). For patients with biochemical control at the latest follow-up, SMR was not increased, neither among patients diagnosed 1991-2000, SMR 1.06 (0.85-1.33) or 2001-2011, SMR 0.87 (0.61-1.24). In contrast, non- controlled patients at the latest follow up from both decades had elevated SMR, 1.90 (1.33-2.72) and 1.98 (1.24-3.14), respectively. Conclusions: The proportion of patients with biochemical control increased over time. Patients with biochemically controlled acromegaly have normal life expectancy while non-controlled patients still have increased mortality. The high rate of macroadenomas and unchanged age at diagnosis illustrates the need for improvements in the management of patients with acromegaly.


2019 ◽  
Vol 105 (3) ◽  
pp. e23-e31 ◽  
Author(s):  
Matteo Parolin ◽  
Francesca Dassie ◽  
Luigi Alessio ◽  
Alexandra Wennberg ◽  
Marco Rossato ◽  
...  

Abstract Background Obstructive sleep apnea (OSA) is a common disorder characterized by upper airway collapse requiring nocturnal ventilatory assistance. Multiple studies have investigated the relationship between acromegaly and OSA, reporting discordant results. Aim To conduct a meta-analysis on the risk for OSA in acromegaly, and in particular to assess the role of disease activity and the effect of treatments. Methods and Study Selection A search through literature databases retrieved 21 articles for a total of 24 studies (n = 734). Selected outcomes were OSA prevalence and apnea-hypopnea index (AHI) in studies comparing acromegalic patients with active (ACT) vs inactive (INACT) disease and pretreatment and posttreatment measures. Factors used for moderator and meta-regression analysis included the percentage of patients with severe OSA, patient sex, age, body mass index, levels of insulin-like growth factor 1, disease duration and follow-up, and therapy. Results OSA prevalence was similar in patients with acromegaly who had ACT and INACT disease (ES = −0.16; 95% CI, −0.47 to 0.15; number of studies [k] = 10; P = 0.32). In addition, AHI was similar in ACT and INACT acromegaly patients (ES = −0.03; 95% CI, −0.49 to 0.43; k = 6; P = 0.89). When AHI was compared before and after treatment in patients with acromegaly (median follow-up of 6 months), a significant improvement was observed after treatment (ES = −0.36; 95% CI, −0.49 to −0.23; k = 10; P < 0.0001). In moderator analysis, the percentage of patients with severe OSA in the populations significantly influenced the difference in OSA prevalence (P = 0.038) and AHI (P = 0.04) in ACT vs INACT patients. Conclusion Prevalence of OSA and AHI is similar in ACT and INACT patients in cross-sectional studies. However, when AHI was measured longitudinally before and after treatment, a significant improvement was observed after treatment.


2017 ◽  
Vol 35 (15_suppl) ◽  
pp. e21058-e21058
Author(s):  
Lee D. Cranmer

e21058 Background: Commercial cabin and cockpit aircrews are at increased cutaneous MEL risk. Occupational and social factors have been invoked to explain this. However, the two groups work in on-board environments with different types/levels of potential exposures. Meta-analysis is conducted to address the hypothesis that differences exist in aircrew MEL risk based on location of in-aircraft job assignment, reflecting differential etiologic occupational exposures. Methods: Medline was searched with the keywords “Cancer,” “Aviation,” and “Melanoma.” Included studies: (1) primarily focused on cutaneous MEL; (2) were in commercial aircrew; (3) measured risk vs a general population; (4) measured risk by Standardized Incidence Ratio (SIR) with 95% confidence intervals (CI); (5) defined the gender distribution; and (7) defined cabin vs cockpit job assignment. Summary SIRs and CIs were calculated using maximum-likelihood, random effects meta-analysis. Values obtained were compared to one another using a z-test, with p < 0.05 pre-specified for null hypothesis rejection. Results: 6 primary studies were identified in the initial search. These were screened for other pertinent references. The secondary search yielded an additional 9 primary studies. For cabin crew, 10 study groups from 6 studies were included. 411,561 person-years of follow-up among 28,093 subjects (86% female) were included (SIR 2.18, 95% CI 1.83-2.60). For cockpit crew, data from 9 studies representing 830,670 person-years of follow-up among 45,986 subjects (1% female) were included (SIR 2.44, 95% CI 1.94-3.08). z-test for the difference indicated that the two SIR values were not statistically different ( z= 0.77, p = 0.441). Conclusions: Though cockpit crew demonstrate an increased SIR vs cabin crew, this is not statistically significant. These results suggest that occupational exposures in different locations within commercial aircraft does not influence MEL risk. Other factors, such as cosmic radiation, non-aircraft occupational exposures and avocational exposures, should be the targets of further assessment.


Author(s):  
Shunsuke Nakagawa ◽  
Yuichi Shinkoda ◽  
Daisuke Hazeki ◽  
Mari Imamura ◽  
Yasuhiro Okamoto ◽  
...  

AbstractCentral diabetes insipidus (CDI) and relapse are frequently seen in multifocal Langerhans cell histiocytosis (LCH). We present two females with multifocal LCH who developed CDI 9 and 5 years after the initial diagnosis, respectively, as a relapse limited to the pituitary stalk. Combination chemotherapy with cytarabine reduced the mass in the pituitary stalk. Although CDI did not improve, there has been no anterior pituitary hormone deficiency (APHD), neurodegenerative disease in the central nervous system (ND-CNS) or additional relapse for 2 years after therapy. It was difficult to predict the development of CDI in these cases. CDI might develop very late in patients with multifocal LCH, and therefore strict follow-up is necessary, especially with regard to symptoms of CDI such as polydipsia and polyuria. For new-onset CDI with LCH, chemotherapy with cytarabine might be useful for preventing APHD and ND-CNS.


2013 ◽  
Vol 202 (1) ◽  
pp. 22-27 ◽  
Author(s):  
Pirn Cuijpers ◽  
Nicole Vogelzangs ◽  
Jos Twisk ◽  
Annet Kleiboer ◽  
Juan Li ◽  
...  

BackgroundAlthough the association between depression and excess mortality has been well established, it is not clear whether this is greater in major depression than in subthreshold depression.AimsTo compare excess mortality in major depression with that in subthreshold depression.MethodWe searched bibliographic databases and included prospective studies in which both major and subthreshold depression were examined at baseline and mortality was measured at follow-up.ResultsA total of 22 studies were included. People with major depression had a somewhat increased chance of dying earlier than people with subthreshold depression but this difference was not significant, although there was a trend (relative risk 1.13, 95% CI 0.98-1.30, P=0.1). The population attributable fraction was 7% for major depression and an additional 7% for subthreshold depression.ConclusionsAlthough excess mortality may be somewhat higher in major than in subthreshold depression, the difference is small and the overall impact on excess mortality is comparable.


2013 ◽  
Vol 98 (4) ◽  
pp. E779-E784 ◽  
Author(s):  
Christina Tatsi ◽  
Amalia Sertedaki ◽  
Antonis Voutetakis ◽  
Eleni Valavani ◽  
Maria-Alexandra Magiakou ◽  
...  

Context: Holoprosencephaly (HPE) is a developmental defect characterized by wide phenotypic variability, ranging from minor midline malformations (eg, single central incisor) to severe deformities. In 10–15% of HPE patients, mutations in specific genes have been identified (eg, SHH, TGIF, SIX3). Pituitary stalk interruption syndrome (PSIS) constitutes a distinct abnormality of unknown pathogenesis, whereas isolated pituitary hypoplasia (IPH) has been linked to various developmental genes. Objective: Three of our patients with PSIS had a single central incisor, a malformation encountered in some HPE cases. Based on this observation, we initiated a search for mutations in HPE-associated genes in 30 patients with PSIS or IPH. Design and Participants: The entire coding region of the TGIF, SHH, and SIX3 genes was sequenced in patients with combined pituitary hormone deficiency associated with either PSIS or IPH and in healthy controls. Results: Two novel mutations in the HPE-related genes were detected (ie, c.799 C&gt;T, p.Q267X in the TGIF gene, and c.1279G&gt;A, p.G427R in the SHH gene) in 2 of our patients. The overall incidence of HPE-related gene mutations in our nonsyndromic and nonchromosomal patients was 6.6%. No molecular defect in the SIX3 gene was detected in our cohort. Conclusions: The data suggest that HPE-related gene mutations are implicated in the etiology of isolated pituitary defects (PSIS or IPH). Alternatively, PSIS or IPH may constitute mild forms of an expanded HPE spectrum.


2019 ◽  
Author(s):  
Liu Daqian ◽  
Wang Yonghua ◽  
Zhao Yang ◽  
Yang Xuecheng ◽  
wang xinsheng ◽  
...  

Abstract Introduction: This was a meta-analysis of studies on conjoined and separate anastomosis for urinary diversion after radical cystectomy due to bladder cancer. Methods: We searched databases (PubMed, Embase and the Cochrane Central Register of Controlled Trials) for studies regarding anastomosis for urinary diversion due to bladder cancer. The meta-analysis was designed to determine the difference between conjoined and separate anastomosis. Six studies including 1601 patients were considered in the meta-analysis (654 in the separate group and 947 in the conjoined group). Results: There were no significant differences with respect to age, gender or duration of follow-up. There were seven studies that compared the rate of stricture and the rate of hydronephrosis. For overall stricture, there were 186 of 1163 ureters in the separate group and 64 of 1718 ureters in the conjoined group (odds ratio, 4.53; 95% CI, 2.01-10.22; P = 0.0003). For overall hydronephrosis patients, there were 55 of 205 ureters in the separate group and 51 of 254 ureters in the conjoined group (odds ratio, 1.48; 95% CI, 0.95-2.29; P = 0.08). Conclusion: The separate anastomosis had a higher ratio of strictures than did the conjoined group. The separate anastomosis group experienced hydronephrosis more often than did the conjoined anastomosis group. The separate anastomosis is the better choice for surgery during the operation.


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