Double pituitary adenomas in a large surgical series

Abstract Purpose To explore the incidence of double pituitary adenomas in a tertiary center for pituitary surgery and asses their clinical, imaging and histopathological features. Methods The medical records of the patients operated on for pituitary tumors at the Department of Neurosurgery of Military Institute of Medicine in Warsaw, Poland between the years 2003 and 2018 were retrospectively analyzed. Among the 3270 treated patients, the diagnosis of double pituitary adenoma was established in 22 patients. Clinical, laboratory, detailed histopathological and diagnostics imaging data were collected and analyzed. Results There were 21 cases of synchronous and one case of asynchronous double pituitary adenoma. The main clinical finding was acromegaly (12/22) followed by Cushing’s disease (3/22). The diagnosis of synchronous double pituitary adenoma was suspected in the preoperative MRI in 11 patients. In the remaining patients the diagnosis of contiguous double pituitary adenoma was confirmed in the histopathological examination. There was no predilection for gender and the mean observation time was 74.2 months. In one case of Cushing’s disease the occurrence of double pituitary adenoma led to the initial failure of achieving hormonal remission. One patient presented with double pituitary adenomas as a manifestation of Carney complex. Conclusions Double pituitary adenoma is a rare entity that can pose a significant challenge especially in the setting of Cushing’s disease. Careful inspection of preoperative MRI and diagnostic work-up before transsphenoidal surgery and thorough histopathological microscopic examinations with immunohistochemical staining for all pituitary hormones is essential for establishing the diagnosis of double pituitary adenoma.

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Diagnosis and Multimodality Management of Cushing’s Disease: A Practical Review

International Journal of Endocrinology ◽

10.1155/2013/893781 ◽

2013 ◽

Vol 2013 ◽

pp. 1-7 ◽

Cited By ~ 10

Author(s):

Gabriel Zada

Keyword(s):

Pituitary Adenoma ◽

Cushing’S Disease ◽

Patient Survival ◽

Clinical Laboratory ◽

Management Strategies ◽

Serum Cortisol ◽

Cushing's Disease ◽

First Line ◽

Stepwise Approach

Cushing’s Disease is caused by oversecretion of ACTH from a pituitary adenoma and results in subsequent elevations of systemic cortisol, ultimately contributing to reduced patient survival. The diagnosis of Cushing’s Disease frequently involves a stepwise approach including clinical, laboratory, neuroimaging, and sometimes interventional radiology techniques, often mandating multidisciplinary collaboration from numerous specialty practitioners. Pituitary microadenomas that do not appear on designated pituitary MRI or dynamic contrast protocols may pose a particularly challenging subset of this disease. The treatment of Cushing’s Disease typically involves transsphenoidal surgical resection of the pituitary adenoma as a first-line option, yet may require the addition of adjunctive measures such as stereotactic radiosurgery or medical management to achieve normalization of serum cortisol levels. Vigilant long-term serial endocrine monitoring of patients is imperative in order to detect any recurrence that may occur, even years following initial remission. In this paper, a stepwise approach to the diagnosis, and various management strategies and associated outcomes in patients with Cushing’s Disease are discussed.

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Pituitary Cushing's disease without adenoma

Acta Endocrinologica ◽

10.1530/acta.0.0940297 ◽

1980 ◽

Vol 94 (3) ◽

pp. 297-303 ◽

Cited By ~ 40

Author(s):

Adrian M. Schnall ◽

Kalman Kovacs ◽

Jerald S. Brodkey ◽

Olof H. Pearson

Keyword(s):

Pituitary Adenoma ◽

Cushing’S Disease ◽

Pituitary Adenomas ◽

Great Majority ◽

Serum Levels ◽

Cushing's Disease ◽

Serial Sections ◽

Cell Hyperplasia ◽

Biochemical Remission ◽

Lower Limits

Abstract. Recent reports of patients with Cushing's disease who have been explored via the transsphenoidal route indicate that the great majority has pituitary adenomas. We report a patient with biochemically documented pituitary-based hypercortisolism who had a clinical and biochemical remission following hypophysectomy. Serial sections of the pituitary tissue removed showed hyperplasia of corticotroph cells but no adenoma. Hypophysectomy was complete as documented by serum levels of FSH, LH, TSH, prolactin, hGH and ACTH at the lower limits of the respective assays, with no response to appropriate stimuli. This case demonstrates that a minority of patients with Cushing's disease has corticotroph cell hyperplasia without a pituitary adenoma.

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Pleurihormonal Pituitary Adenomas Presenting as Acromegaly

Journal of the Endocrine Society ◽

10.1210/jendso/bvab048.1227 ◽

2021 ◽

Vol 5 (Supplement_1) ◽

pp. A602-A602

Author(s):

Megan Schwehr ◽

Mayumi Endo

Keyword(s):

Pituitary Adenoma ◽

Cushing’S Syndrome ◽

Cushing’S Disease ◽

Pituitary Adenomas ◽

Brain Mri ◽

Fatty Infiltration ◽

Cushing's Syndrome ◽

Cushing's Disease ◽

Peripheral Muscle ◽

Suppression Test

Abstract Background: Pleurihormonal pituitary adenomas represent 10-15% of all functioning pituitary adenomas1. One of the rarest combinations is ACTH-GH co-secretion with concomitant acromegaly and Cushing’s disease. Clinical Case: 30-year-old female with type 2 diabetes (A1c 11.4%) developed rapidly progressive peripheral neuropathy. Labs revealed a transaminitis. Hepatic ultrasound revealed fatty infiltration of the liver consistent with steatosis. Patient endorsed life-long “chicken legs”, buffalo hump, and easy bruising. She continued to have worsening paresthesias and stated her teeth were “more translucent”. At an endocrinology visit peripheral muscle wasting, atraumatic bruising at the ankles and toes,and striae on the abdomen, shoulders, breasts, and armpits were noted. She had also noted teeth separation and thinning over time and worsening diffuse joint aches. Lab results showed elevated cortisol value of 8.4 mcg/dl after 1 mg dexamethasone suppression test. 24-hour urine cortisol was elevated (87 ug/24 hrs, nl < 45 ug/24 hrs), confirming the diagnosis of Cushing’s syndrome. ACTH was inappropriately normal (56 pg/mL, nl 7.2 – 63 pg/mL), suggesting ACTH dependency. IGF-1 was elevated twice (328 ng/dL and 391 ng/dL, nl <303 ng/dL), and a GH suppression test was abnormal (GH 1.14 ng/mL at 90 minutes) confirming the diagnosis of acromegaly. Brain MRI revealed a cystic pituitary microadenoma (0.5 x 0.5 x 0.6 cm), and the patient underwent surgical resection via transnasal transsphenoidal pituitary surgery with total tumor removal. Immunostaining was positive for only synaptophysin and ACTH. Postoperative diagnosis of Cushing’s disease was made, with formal diagnosis of acromegaly pending improvement in follow-up IGF-1 levels. Literature reviews have revealed very few cases of pleurihormonal pituitary adenomas presenting with concomitant Cushing’s Syndrome and acromegaly. Roca et. al examined 17 studies which described 20 patients with this condition; only 4 presented with symptoms of both acromegaly and Cushing’s disease. In many cases previously reported, the clinical presentation of Cushing’s syndrome was subtle, often with subclinical Cushing’s syndrome, while patients had florid symptoms of acromegaly.1. Conclusion: This Cushing’s predominant case of a GH-ACTH co-secreting tumor is an incredibly rare presentation for this pleurihormonal pituitary adenoma, which is already a rare diagnosis. References: 1.Roca E, Mattogno PP, Porcelli T, Poliani L, Belotti F, Schreiber A, Maffezzoni F, Fontanella MM, Doglietto F. Plurihormonal ACTH-GH Pituitary Adenoma: Case Report and Systematic Literature Review. World Neurosurg. 2018 Jun;114:e158-e164. PMID: 29501516.

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Comparison of Constructive Interference in Steady-State and T1-Weighted MRI Sequence at Detecting Pituitary Adenomas in Cushing's Disease Patients

Journal of Neurological Surgery Part B Skull Base ◽

10.1055/s-0038-1642032 ◽

2018 ◽

Vol 79 (06) ◽

pp. 593-598 ◽

Cited By ~ 3

Author(s):

Min Lang ◽

Ghaith Habboub ◽

Doksu Moon ◽

Abin Bandyopadhyay ◽

Danilo Silva ◽

...

Keyword(s):

Steady State ◽

Pituitary Adenoma ◽

Cushing’S Disease ◽

Pituitary Adenomas ◽

Cushing's Disease ◽

Breath Hold ◽

Mri Imaging ◽

Constructive Interference ◽

Ciss Sequence ◽

Magnetic Resonance Imaging Mri

Background T1-weighted magnetic resonance imaging (MRI) remains the standard pituitary imaging sequence. However, up to 50% of pituitary adenomas are missed with standard imaging in Cushing's disease (CD) patients. Utility of other imaging sequences needs be explored. Objective To compare a T2-gradient echo sequence, constructive interference in steady state (CISS), with a contrast-enhanced (CE) T1-weighted sequence, volume-interpolated breath-hold examination (VIBE), in the detection of pituitary adenomas in CD patients. Design Retrospective analysis of CD patients who underwent endoscopic transsphenoidal pituitary adenomectomy at our institution. Patients and Measurement Twelve patients were included in the study. Two neuroradiologists, who were blinded to the patients' clinical and surgical findings, independently reviewed the CE-VIBE and CISS images. Localization of pituitary adenoma from imaging was compared with intraoperative localization. Results The averaged sensitivity of detecting pituitary adenoma in CD patients was not significantly different between CE-VIBE sequence (63%) and CISS sequence (54%). The positive predictive value was 75% (Observer A) and 100% (Observer B) with CE-VIBE sequence, and 64% (Observer A) and 100% (Observer B) with CISS sequence. In two patients, pituitary adenoma was easily localized with CISS sequence but hard to detect with CE-VIBE sequence. In two other patients, the adenoma was much better delineated on CE-VIBE sequence. Conclusion In our series, the addition of CISS sequence to T1-weighted MRI imaging protocols improved the detection of pituitary adenomas in CD patients. CISS sequence may be a useful adjunct to T1-weighted pituitary MRI protocols and an appropriate alternative for patients with gadolinium contraindications.

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Double Pituitary Adenomas with Synchronous Somatotroph and Corticotroph Clinical Presentation of Acromegaly and Cushing's Disease

World Neurosurgery ◽

10.1016/j.wneu.2019.08.224 ◽

2019 ◽

Vol 132 ◽

pp. 161-164

Author(s):

Naomi Collazo-Gutiérrez ◽

Orlando de Jesús ◽

Maria Villamil-Jarauta ◽

Milliette Alvarado ◽

Loida González ◽

...

Keyword(s):

Cushing’S Disease ◽

Pituitary Adenomas ◽

Clinical Presentation ◽

Cushing's Disease ◽

Double Pituitary Adenomas

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Aseptic femur head necrosis revealing Cushing's disease: a rare presentation

Journal of Clinical Surgery and Research ◽

10.31579/2768-2757/002 ◽

2020 ◽

Vol 1 (1) ◽

pp. 01-04

Author(s):

Zeineb Alaya

Keyword(s):

Pituitary Adenoma ◽

Cushing’S Disease ◽

Histopathological Examination ◽

Hip Pain ◽

Cushing's Disease ◽

Rare Presentation ◽

Case Presentation ◽

Disease Case ◽

One Year

Background: Cushing's disease is an endocrine disorder and may be associated with non-traumatic osteonecrosis of the hip. We report the case of avascular necrosis of the femoral heads (AVNFH) as the presenting manifestation of Cushing's disease. Case presentation: A 29-year-old male patient was referred to our department because of non-traumatic bilateral hip pain evolving for two years. The pain was associated with lameness when walking. He noticed a drop in libido since one year with erectile dysfunction. Physical examination revealed facio-truncal obesity, abdominal violaceous striae, dorsal hyper-kyphosis, a pain and limitation of internal rotation of the left hip with a Lequesne index of 16. The standard radiography showed a loss of sphericity of the left femoral head with an aspect of eggshell. The MRI confirmed the diagnosis of a bilateral AVNFH. The baseline cortisol level was 287 ng/ml. AVNFH as the presenting manifestation of Cushing's disease was suspected and confirmed by a pituitary MRI which revealed a pituitary adenoma. A discharge of the hips was indicated. The patient underwent surgical resection of the adenoma and was referred to the endocrinology department. The histopathological examination confirmed the diagnosis of pituitary adenoma (corticotrope adenoma). The patient has also received an intravenous perfusion of 5 mg of Zoledronic acid. The follow up was marked by the regression of the symptoms of Cushing's disease and the hip pain. Conclusion: AVNFH as the presenting manifestation of Cushing's disease is rarely described. Hence, it’s needed to exclude endogenous hypercorticism in any patient with idiopathic osteonecrosis.

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