Abstract #912: Nine Year Follow Up of a 45 Year Old Female with Idiopathic Granulomatous Hypophysitis Maintaining Normal Pituitary Function

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.

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Pituitary function after high-dose 177Lu-DOTATATE therapy and long-term follow-up

Neuroendocrinology ◽

10.1159/000507761 ◽

2020 ◽

Author(s):

Anna Sundlöv ◽

Katarina Sjögreen-Gleisner ◽

Jan Tennvall ◽

Ludvig Dahl ◽

Johanna Svensson ◽

...

Keyword(s):

Pituitary Function ◽

High Dose ◽

Long Term Follow Up

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Idiopathic granulomatous hypophysitis presenting with galactorrhea, headache, and nausea in a woman: a case report and review of the literature

Journal of Medical Case Reports ◽

10.1186/s13256-019-2276-4 ◽

2019 ◽

Vol 13 (1) ◽

Author(s):

Guive Sharifi ◽

Mohammad Reza Mohajeri-Tehrani ◽

Behrouz Navabakhsh ◽

Bagher Larijani ◽

Touraj Valeh

Keyword(s):

Pituitary Gland ◽

Inflammatory Disease ◽

Transsphenoidal Surgery ◽

Brain Magnetic Resonance Imaging ◽

Serum Prolactin ◽

Granulomatous Hypophysitis ◽

History Of ◽

Work Up ◽

Visual Complaints

Abstract Background Inflammation of the pituitary gland can occur in a variety of primary or secondary disorders. Idiopathic granulomatous hypophysitis is a rare inflammatory disease of the pituitary gland that can closely mimic a pituitary adenoma clinicoradiologically. Most authorities agree on minimally invasive transsphenoidal surgery as the mainstay in diagnosis and treatment of this disorder. There is still some controversy regarding pure medical management of idiopathic granulomatous hypophysitis in the literature. Case presentation A 47-year-old Iranian woman of Azeri ethnicity with a history of benign breast cysts with a chief complaint of galactorrhea presented to our endocrinology clinic. Her past medical history was negative for any menstrual irregularities, hirsutism, visual complaints, diplopia, polyuria and polydipsia or seizures. She was taking 100 mcg of levothyroxine daily. Her familial history and physical examination were unremarkable. Her initial laboratory work-up revealed hyperprolactinemia (82.4 ng/mL) with otherwise normal pituitary axes. Brain magnetic resonance imaging showed a pituitary macroadenoma for which she was treated with 0.5 mg of cabergoline weekly. Although her serum prolactin level dropped to 1.7 ng/mL and her galactorrhea was resolved, she continued to complain of headaches and nausea. Repeated imaging showed no decrease in size of the macroadenoma. Therefore, she underwent transsphenoidal surgery of the macroadenoma which was reported as chronic granulomatous hypophysitis by expert pathologists. Tuberculosis, sarcoidosis, Wegener’s granulomatosis, Langerhans cell histiocytosis, and syphilis were ruled out by appropriate tests and she was diagnosed as having idiopathic granulomatous hypophysitis. Fortunately, her condition was not complicated by hypopituitarism and she was symptom free 9 months after transsphenoidal surgery. Conclusions Idiopathic granulomatous hypophysitis, a rare inflammatory disease of the pituitary gland, is a diagnosis of exclusion for which both medical and surgical management are reported in the literature. We present a case of idiopathic granulomatous hypophysitis who was symptom free with no complications of hypopituitarism following its transsphenoidal resection after 9 months of follow-up.

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Normal final height and apparent cure after pituitary irradiation for Cushing's disease in childhood: long-term follow-up of anterior pituitary function

Clinical Endocrinology ◽

10.1046/j.1365-2265.1998.00385.x ◽

1998 ◽

Vol 48 (5) ◽

pp. 663-667 ◽

Cited By ~ 4

Author(s):

L. B. Johnston ◽

A. B. Grossmann ◽

P. N. Plowman ◽

G. M. Besser ◽

M. O. Savage

Keyword(s):

Cushing’S Disease ◽

Anterior Pituitary ◽

Final Height ◽

Pituitary Function ◽

Cushing's Disease ◽

Long Term Follow Up ◽

Anterior Pituitary Function ◽

Pituitary Irradiation

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Residual Pituitary Function after Brain Injury-Induced Hypopituitarism: A Prospective 12-Month Study

The Journal of Clinical Endocrinology & Metabolism ◽

10.1210/jc.2005-0504 ◽

2005 ◽

Vol 90 (11) ◽

pp. 6085-6092 ◽

Cited By ~ 224

Author(s):

Gianluca Aimaretti ◽

Maria Rosaria Ambrosio ◽

Carolina Di Somma ◽

Maurizio Gasperi ◽

Salvatore Cannavò ◽

...

Keyword(s):

Brain Injury ◽

High Risk ◽

Gh Deficiency ◽

Pituitary Function ◽

Brain Injured ◽

Injured Patients ◽

The Brain ◽

Over Time

Abstract Context: Traumatic brain injury (TBI) and subarachnoid hemorrhage (SAH) are conditions at high risk for the development of hypopituitarism. Objective: The objective of the study was to clarify whether pituitary deficiencies and normal pituitary function recorded at 3 months would improve or worsen at 12 months after the brain injury. Design and Patients: Pituitary function was tested at 3 and 12 months in patients who had TBI (n = 70) or SAH (n = 32). Results: In TBI, the 3-month evaluation had shown hypopituitarism (H) in 32.8%. Panhypopituitarism (PH), multiple (MH), and isolated (IH) hypopituitarism had been demonstrated in 5.7, 5.7, and 21.4%, respectively. The retesting demonstrated some degree of H in 22.7%. PH, MH, and IH were present in 5.7, 4.2, and 12.8%, respectively. PH was always confirmed at 12 months, whereas MH and IH were confirmed in 25% only. In 5.5% of TBI with no deficit at 3 months, IH was recorded at retesting. In 13.3% of TBI with IH at 3 months, MH was demonstrated at 12-month retesting. In SAH, the 3-month evaluation had shown H in 46.8%. MH and IH had been demonstrated in 6.2 and 40.6%, respectively. The retesting demonstrated H in 37.5%. MH and IH were present in 6.2 and 31.3%, respectively. Although no MH was confirmed at 12 months, two patients with IH at 3 months showed MH at retesting; 30.7% of SAH with IH at 3 months displayed normal pituitary function at retesting. In SAH, normal pituitary function was always confirmed. In TBI and SAH, the most common deficit was always severe GH deficiency. Conclusion: There is high risk for H in TBI and SAH patients. Early diagnosis of PH is always confirmed in the long term. Pituitary function in brain-injured patients may improve over time but, although rarely, may also worsen. Thus, brain-injured patients must undergo neuroendocrine follow-up over time.

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Anterior Pituitary Function in Patients with Nonfunctioning Pituitary Adenoma: Results of Longitudinal Follow-up.

Endocrine Journal ◽

10.1507/endocrj.42.421 ◽

1995 ◽

Vol 42 (3) ◽

pp. 421-427 ◽

Cited By ~ 24

Author(s):

ATSUSHI TOMINAGA ◽

TOHRU UOZUMI ◽

KAZUNORI ARITA ◽

KAORU KURISU ◽

TAKASHI YANO ◽

...

Keyword(s):

Pituitary Adenoma ◽

Anterior Pituitary ◽

Pituitary Function ◽

Nonfunctioning Pituitary Adenoma ◽

Anterior Pituitary Function

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A Longitudinal Study of Vasopressin Cell Antibodies, Posterior Pituitary Function, and Magnetic Resonance Imaging Evaluations in Subclinical Autoimmune Central Diabetes Insipidus

The Journal of Clinical Endocrinology & Metabolism ◽

10.1210/jcem.84.9.5945 ◽

1999 ◽

Vol 84 (9) ◽

pp. 3047-3051 ◽

Cited By ~ 20

Author(s):

A. De Bellis ◽

A. Colao ◽

F. Di Salle ◽

V. I. Muccitelli ◽

S. Iorio ◽

...

Keyword(s):

Magnetic Resonance Imaging ◽

Magnetic Resonance ◽

Autoimmune Diseases ◽

Diabetes Insipidus ◽

Central Diabetes Insipidus ◽

Pituitary Function ◽

Study Entry ◽

Posterior Pituitary ◽

Resonance Imaging

Cytoplasmic autoantibodies to vasopressin-cells (AVPcAb) have been detected not only in patients with overt central diabetes insipidus (CDI), but also in patients with endocrine autoimmune diseases without CDI. This suggests that complete CDI can be preceded by a preclinical stage. Among 878 patients with endocrine autoimmune diseases without CDI, 9 patients found to be AVPcAb positive and 139 AVPcAb-negative controls were enrolled in this open prospective study. They were evaluated for AVPcAb and posterior pituitary function at least yearly for about 4 yr (range, 37–48 months); during this span, magnetic resonance imaging (MRI) of posterior pituitary and stalk was performed only in the AVPcAb-positive patients. Five of the 9 AVPcAb-positive patients had normal posterior pituitary function at study entry. They were AVPcAb positive throughout the follow-up period. At later stages of the study, 3 of them developed partial CDI, and 1 developed complete CDI. The remaining 4 patients showed impaired response to the water deprivation test at study entry and were diagnosed as having partial CDI. Two of them agreed to receive desmopressin replacement for 1 yr. After this treatment, the patients became negative for AVPcAb and displayed normal posterior pituitary function until the end of the follow-up. Conversely, the 2 untreated patients with partial CDI remained AVPcAb positive. One of them developed overt CDI. None of the controls became AVPcAb positive or developed CDI. The normal hyperintense MRI signal of the posterior pituitary, present at study entry, persisted subsequently in all 9 AVPcAb-positive patients, including those developing overt CDI, only disappearing in the late phase of complete CDI. In asymptomatic subjects, the monitoring of AVPcAb, but not MRI, seems to be useful to predict a progression toward partial/overt CDI. Early desmopressin therapy in patients with partial CDI could interrupt or delay the autoimmune damage and the progression toward clinically overt CDI.

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Long-term anterior pituitary function in patients with paediatric Cushing’s disease treated with pituitary radiotherapy

Acta Endocrinologica ◽

10.1530/eje-06-0588 ◽

2007 ◽

Vol 156 (4) ◽

pp. 477-482 ◽

Cited By ~ 20

Author(s):

L F Chan ◽

H L Storr ◽

P N Plowman ◽

L A Perry ◽

G M Besser ◽

...

Keyword(s):

Cushing’S Disease ◽

Anterior Pituitary ◽

Serum Cortisol ◽

Pituitary Function ◽

Cushing's Disease ◽

Serum Prolactin ◽

Second Line ◽

Anterior Pituitary Function

Background/objective: Pituitary radiotherapy (RT) is an effective second-line treatment for paediatric Cushing’s disease (CD). Although the short-term effects of pituitary RT are well documented, there are less data on possible long-term sequelae. We report the long-term anterior pituitary function in a cohort of paediatric CD patients treated with pituitary RT. Patients and methods: Between 1983 and 2006, 12 paediatric CD patients (10 males and 2 females) of mean age 11.4 years at diagnosis (range 6.4–17.4) underwent second-line pituitary RT (45 Gy in 25 fractions), following unsuccessful transsphenoidal surgery. Out of 12, 11 patients were cured by RT (cure interval 0.13–2.86 years) defined by mean serum cortisol of <150 nmol/l on 5-point day curve and midnight sleeping cortisol of <50 nmol/l. Long-term data are available for six male patients, who received RT at the age of 7.0–17.6 years. The mean follow-up from the completion of RT was 10.5 years (6.6–16.5). Results: At a mean of 1.0 year (0.11–2.54) following RT, GH deficiency (peak GH <1–17.9 mU/l) was present in five out of six patients. On retesting at a mean of 9.3 years (7.6–11.3) after RT, three out of four patients were GH sufficient (peak GH 19.2–50.4 mU/l). Other anterior pituitary functions including serum prolactin in five out of six patients were normal on follow-up. All the six patients had testicular volumes of 20–25 ml at the age of 14.5–28.5 years. Conclusion: This series of patients illustrates the absence of serious long-term pituitary deficiency after RT and emphasises the importance of continued surveillance.

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Optimal Surgical Approaches for Rathke Cleft Cyst With Consideration of Endocrine Function

Operative Neurosurgery ◽

10.1227/neu.0b013e3182418034 ◽

2011 ◽

Vol 70 (suppl_2) ◽

pp. ons250-ons258 ◽

Cited By ~ 2

Author(s):

Jeong Kyung Park ◽

Eun Jig Lee ◽

Sun Ho Kim

Keyword(s):

Visual Field ◽

Transsphenoidal Surgery ◽

Endocrine Function ◽

Pituitary Function ◽

Female Ratio ◽

Rathke Cleft Cyst ◽

Function Test ◽

The Mean ◽

Pituitary Function Test

ABSTRACT BACKGROUND: Surgical indications for Rathke cleft cyst are not clear. OBJECTIVE: To evaluate postoperative outcomes in terms of endocrine function. METHODS: The study analyzed a total 73 patients who underwent transsphenoidal surgery. All patients underwent a visual field test, combined pituitary function test, and magnetic resonance imaging before and after surgery. A follow-up combined pituitary function test was performed at 1.5-year intervals. RESULTS: The mean age at the time of surgery was 35 ± 14 years, and the male/female ratio was 1:1.25 (33/40). The mean follow-up duration after surgery was 59 ± 39 months. The most common symptoms were headache (84%), visual disturbance (48%), and polyuria (38%). After transsphenoidal surgery, 75% of polyuria and 96% of visual field defects were resolved, and pituitary function improved in 42% of patients. The mean age of patients who exhibited worsened hypopituitarism was significantly higher than that of patients who exhibited unchanged or improved hypopituitarism (44 ± 15.7 vs 33 ± 13.5 years; P = .02). Twelve patients (16%) experienced recollection of cyst, but none required reoperation. Five of the recollected cysts presented with characteristics that were different from those of the initial lesions, and 2 recollected cysts underwent spontaneous regression. CONCLUSION: Minimal incision with radical removal of cyst content is reasonable to prevent the development of endocrine disturbances and other complications. Individualized risks and benefits must be assessed before a decision is reached regarding surgery and surgical method. Patients with recurrent Rathke cleft cyst require careful follow-up with special attention rather than a hasty operation.

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