scholarly journals MON-313 Thyrotropin-Secreting Pituitary Tumors: Clinical Features, Diagnostic Criteria and Treatment Outcomes in 28 Patients

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Diana A Dimitrova ◽  
Evgeniy T Abdullin ◽  
Elena G Przhiyalkovskaya ◽  
Andrey Yu. Grigoriev ◽  
Vilen N Azizyan ◽  
...  

Abstract Introduction Thyrotropin-secreting pituitary adenoma (TSH-oma) is a rare cause of thyrotoxicosis and represents 0.5-3% of all pituitary adenomas. Given the rarity of the disease and the lack of extensive clinical experience in world practice, the analysis of each case of TSH-secreting pituitary tumors is extremely relevant. Materials and methods: We enrolled consecutive patients with TSH secreting pituitary adenoma. TSH (0,25-3,5 mIU/L), FT4 (9-20 pmol/l) FT3 (2,5-5,5 pmol/l) were measured by Architect i2000SR (Abbott Laboratories, Abbott Park, Illinois, U.S.A). An octreotide test consisted of injecting short-acting somatostatin analogue (SSA) at a dose of 100 μg 3 times a day subcutaneously during 3 days or long-acting SSA 20 mg in 28 days minimum for two months and measuring thyroid profile blood tests before and at the end of the test. MRI was performed on GE Optima MR450w 1.5T. Results: We present 28 case series with TSH-omas followed in our center from 2010 to 2019, the median age - 46 (from 10 to 71 years) with a predominance of a female (21 cases) over a male (7 cases). Clinical manifestations included cardiac arrhythmias (82.1%), neurological changes (60.7%), visual impairment (39.3%), osteoporosis (28.6%), hypopituitarism (7.1%). At first admission, 17 patients had increased TSH, FT3 and FT4; in 7 patients - FT3 and FT4 only, and in 4 - an increase was in one of the hormones; mean value of TSH was 5,04 mIU/L, FT4 - 24,46 pmol/l, FT3 - 8,9 pmol/l. SSBG, CTx, osteocalcin were elevated in 60.9%, 57.9% and 31.6% of cases, respectively. In 88.5% of cases octreotide test led to thyroid hormones normalization. On MRI macroadenomas was registered in 67.9%. In 12 patients, diagnosis was confirmed by immunohistochemical examination. In 12 patients, the diagnosis verification was based on remission after surgical and medical treatment. In the remaining four cases, the diagnosis was made on clinical and laboratory data, however, confirmation was inconceivable due to inefficacy of medical treatment. Remission was achieved in 23 patients: 17 of 21 after neurosurgery (81%), 3 of 7 after primary administration of SSA, and 3 after administration of SSA after surgery (100% remission in cases of combined treatment and 42.9% of monotherapy); 3 out of 5 patients who did not achieve remission were lost for observation. Two patients died: in one case due to the brain herniation and cachexia in active thyrotoxicosis. In second case, the patient developed a mental disorder (catatonia) after the second transphenoidal adenomectomy, which led to death. Conclusion The diagnosis of TSH-oma should be based on all thyroid hormones assessment, since biochemical thyrotoxicosis can manifest itself in various ways. The most effective treatment is transphenoidal adenomectomy. If a neurosurgery is not effective or contraindicative, preoperative preparations SSA can be used.

2020 ◽  
Vol 10 (5) ◽  
pp. 93-99
Author(s):  
Ljudmila V. Tyrtova ◽  
Aleksej S. Olenev ◽  
Natalja V. Parshina ◽  
Christina V. Skobeleva

Pituitary gigantism is a disease caused by an excess of growth hormone and characterized by tallness with a proportional increase in all parts of the body. Almost always in patients with pituitary gigantism found pituitary adenoma, producing growth hormone (somatotropin). In rare cases, there is excess production of somatoliberin by the hypothalamus or tumors outside the brain. Somatotropinoma can be sporadic or caused by a disease with genetic defects: multiple endocrine neoplasia type 1, McCuneAlbright syndrome, Carney complex, X-LAG syndrome, AIP-mutation. All associated with genetic defects somatotropinoma more invasive and less amenable to drug therapy than sporadic. Clinical recommendations (protocols) for the treatment of pituitary gigantism in children currently does not exist. The issue of preliminary medical treatment to improve the outcome of neurosurgical interventions has not been resolved, and further methodologically based studies are needed to clarify this point. The article presents a clinical case of pituitarygigantism caused by pituitary adenoma, which produces growth hormone in a 12-year-old boy. The diagnosis was established on the basis of clinical and anamnestic, laboratory data and magnetic resonance imaging. Clinical and laboratory manifestations of hypopituitarism and diabetes insipidus, visual field disorders, neurological symptoms, as well as signs of genetic diseases in the patient were not noted. Treatment with bromocriptin gave a partial positive effect: the size of the formation in the pituitary gland decreased, but the target hormonal parameters were not achieved. A trial administration of octreotide subcutaneously was carried out, as a result of which the level of growth hormone decreased to the target values, no side effects of the drug were noted, which led to the choice of a conservative method as the first line of therapy. Thedecision to treat with bromocryptine in combination with octreotide extended action. The dynamics of tumor sizeonthebackgroundofconservative therapy will answer the question of the need for subsequent neurosurgical treatment.


2020 ◽  
Vol 33 (1) ◽  
pp. 157-163 ◽  
Author(s):  
Melis Demir Köse ◽  
Mehtap Kagnici ◽  
Taha Reşit Özdemir ◽  
Cahit Barış Erdur ◽  
Gülin Erdemir ◽  
...  

AbstractBackgroundCitrin deficiency (CD) is an autosomal recessive genetic disorder caused by a defect in the mitochondrial aspartate/glutamate antiporter, citrin. Three clinical manifestations have been described until today.Case presentationWe reported 5 CD patients from two families. Four patients were male and one patient was female. Two of them have NICCD (neonatal intrahepatic cholestasis caused by citrin deficiency); three of them have CTLN2 (adult-onset type II citrullinemia). Both NICCD patients showed typical clinical and biochemical changes with a diagnosis confirmed by mutations in the SLC25A13 gene. We detected a previously unreported homozygous novel mutation c.478delC (L160Wfs*36 ) on the SLC25A13 gene. All of the CTLN2 patients were siblings. Proband was a 15-year-old mentally retarded and autistic male who had admitted to our emergency with disorientation. Laboratory data showed hyperammonemia and citrullinemia.ConclusionsTwo different profiles of age-related CD have been depicted with this article. It has been aimed to underline that the CD can be observed in different forms not only in neonatals or little infants but also in adolescents. This article is the first case series that covers both NICCD and CTLN2 cases together and that has been published in Turkey. Considering the fact that especially the majority of CTLN2 cases have been identified in Asian countries, our article has vital importance in terms of defining phenotypic features of the disease.


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A967-A968
Author(s):  
Aisha R Saand ◽  
Monica Flores ◽  
Sura Alqaisi ◽  
Ossama Lashin

Abstract Background: Thyrotropin secreting pituitary adenomas (TSH-oma) account for less than 1% of pituitary adenomas and are a rare cause of hyperthyroidism. In this case, we describe the safe use of cabergoline with the subsequent achievement of euthyroid levels and the resolution of symptoms. Clinical Case: A 67-year-old female with a presumed history of hypothyroidism for the last 3 years managed with Armour thyroid was referred for fatigue associated with palpitations, intermittent diarrhea, insomnia, and anxiety. Her TSH was noted to be suppressed at that time, and her Amour thyroid was changed to Levothyroxine 25 mcg daily, which was eventually stopped as her symptoms persisted. Follow-up thyroid function tests (TFTs) showed elevated TSH (8.60 µU/mL, reference range 0.340 - 5.600 µU/mL), and elevated free T3 (10.8 pg/mL, reference range 2.7-4.7 pg/mL) and free T4 (2.0 ng/dL, reference range 0.8-1.5 ng/dL) levels. Further laboratory workup including basal prolactin, LH, FSH, hCG α-subunit, IGF-1, and ACTH levels were within the normal range. Physical examination showed diffuse thyroid enlargement. An I-123 thyroid uptake and scan showed uniform uptake throughout the gland with no focal hyperintense areas. A dedicated pituitary MRI was performed and revealed an 8 mm microadenoma in the posterior left adenohypophysis. The patient wanted to avoid any invasive treatment and refused trans-sphenoidal surgery and medical therapy was chosen. Unfortunately, using a somatostatin analog as part of the standard medical treatment was not an option due to the high cost and lack of insurance coverage. For these reasons, a trial of cabergoline was started to suppress TSH secretion. Atenolol 25 mg daily was added temporarily for the management of her sympathetic symptoms. On periodic follow-up, her symptoms gradually subsided, and TFTs remained within normal limits. Treatment was continued for one year. Conclusion: Clinical manifestations of hyperthyroidism with biochemical evidence of elevated thyroid hormone levels and TSH levels should raise a strong suspicion for central or secondary hyperthyroidism due to TSH secreting pituitary adenoma. Cabergoline might represent an effective alternative form of medical treatment for TSH-oma in certain situations where the first line therapeutic options are refractory, contraindicated, or declined.


2021 ◽  
Author(s):  
Carlien de Herdt ◽  
Eva Philipse ◽  
Christophe De Block

Background and Aims: Thyrotropin-stimulating pituitary adenoma (TSHoma) are a rare entity, occurring in 1 per million people. We performed a systematic review of 535 adult cases summarizing the clinical, biochemical, hormonal and radiological characteristics of TSHoma. Furthermore, we discuss the current guidelines for diagnosis and treatment. Methods A structured research was conducted using Pubmed with the following MeSH terms: “thyrotropin secreting pituitary adenoma” OR “TSHoma” OR “thyrotropinoma”. Results Our analysis included 535 cases originating from 18 case series, 5 cohort studies and 91 case reports. Mean age at diagnosis was 46 years. At presentation 75% had symptoms of hyperthyroidism, 55.5% presented with a goitre and 24.9% had visual field defects. Median TSH at diagnosis was 5.16 (3.20-7.43) mU/L with a mean fT4 of 41.5±15.3 pmol/L. The majority (76.9%) of the TSHoma were macroadenoma. Plurihormonality was seen in 37.4% of the adenoma with a higher incidence in macroadenoma. Surgical resection of the adenoma was performed in 87.7% patients of which 33.5% had residual pituitary adenoma. Postoperative treatment with a somatostatin analogue led to a stable disease in 81.3% of the cases with residual tumour. We noticed a significant correlation between the diameter of the adenoma and residual pituitary adenoma (r=0.490, p<0.001). However, in patients pre-operatively treated with somatostatin analogue this correlation was absent. Conclusion TSHoma are a rare cause of hyperthyroidism and are frequently misdiagnosed. Based on our structured analysis of case series, cohort studies and case reports, we conclude that the majority of TSHoma are macroadenoma being diagnosed in the 5-6th decade of life and presenting with symptoms of hyperthyroidism. Plurihormonalitiy is observed in one third of TSHoma. Treatment consists of neurosurgical resection and SSA in case of surgical failure.


2021 ◽  
Vol 104 (12) ◽  
pp. 1992-1999

Background: Cryptococcal infection, especially cryptococcal meningitis, is the most common cause of central nervous system (CNS) infection with a high mortality rate in patients with systemic lupus erythematosus (SLE). The clinical features of cryptococcal meningitis may be non-specific, which may lead to miss or delay diagnosis and treatment. Objective: To collect the case series of SLE patients with cryptococcosis treated in Nongkhai Hospital between 2013 and 2021 and compared it with other studies. Materials and Methods: The medical records of SLE patients (ICD-10 M320-M329) with cryptococcal infection (ICD-10 B450-B459) treated in Nongkhai Hospital between 2013 and 2021 were reviewed and collected onto a medical record form. The following information were obtained, gender, occupation, age at SLE diagnosis, age of onset, duration of disease, comorbid or risks, previous infection, SLE disease activity, glucocorticoids, and immunosuppressors administered before or at infection diagnosis, cryptococcosis clinical manifestations, laboratory data, Cerebrospinal fluid (CSF) findings, antifungal agents used, and outcomes. Results: Six hundred thirty-six patients with SLE were identified and six patients developed cryptococcosis. Five patients had cryptococcal meningitis and one patient had cryptococcocemia. Fever and headache were the symptoms of all patients. CSF cryptococcal antigen was positive in five patients. Antifungal therapy was initiated as soon as the diagnosis was confirmed in all patients. Five patients (83.3%) recovered completely, and one patient was against the advice. Conclusion: The present study suggested that SLE patients presenting with fever and headache along with a history of moderate to high dose steroids and immunosuppressants administration should always be suspected of cryptococcal infection and cryptococcal meningitis. Meanwhile, CSF cryptococcal antigens are the effective screening tools to establish an early diagnosis. Accordingly, early appropriate treatment is crucial for a favorable outcome. Keywords: Cryptococcal infection; Cryptococcosis; Cryptococcal meningitis; SLE; Lupus


2021 ◽  
pp. 1-5
Author(s):  
Antonio Piras ◽  
Luca Boldrini ◽  
Sebastiano Menna ◽  
Antonella Sanfratello ◽  
Andrea D’Aviero ◽  
...  

<b><i>Introduction:</i></b> Bone metastases (BMs) are the common cause of cancer-related pain, as approximately 45% of cancer patients suffer from bone pain (BP). Radiotherapy (RT) is well established as BP treatment strategy; also, other approaches have been shown to be effective in this setting. Radiofrequency thermoablation (RFA) in a combined strategy with RT appears to be feasible and effective in the treatment of metastatic BP ensuring a better quality of life. Aim of this retrospective study was to describe a case series of patients with painful osteolytic lesions at risk of fracture treated with the RFA-RT combined approach, analyzing local control and pain control as outcomes. <b><i>Methods:</i></b> Data of all patients with BM treated with combined approach in our center from April 2016 to June 2020 were retrospectively analyzed. Patients underwent RFA followed by cementoplasty on the same day and RT in a second phase. RT dose ranged between 30 and 37.5 Gy in 5/10 fractions. BP was evaluated according to the numeric rating scale (NRS), at the beginning of treatment and at 1, 2, 3, 6, 9, and 12 months from the end of combined treatment. <b><i>Results:</i></b> A total of 27 patients were treated from April 2016 to June 2020 with RFA-RT combined approach. The large majority of patients underwent stereotactic body radiotherapy (SBRT) (23/27). All patients experienced an NRS value decrease &#x3e;2 at 1 month and between the first and second months. NRS mean value reached 0 at 3, 6, 9, and 12 months’ evaluations. <b><i>Discussion/Conclusion:</i></b> The results of this retrospective analysis of patients treated with RFA-RT combined approach for BP support its safety and efficacy in terms of pain reduction. SBRT role in this combined approach has to be investigated in randomized trials.


2012 ◽  
Vol 58 (4) ◽  
pp. 8-13
Author(s):  
I B Zhurtova ◽  
N A Strebkova ◽  
V A Peterkova ◽  
A V Vorontsov ◽  
A V Il'in

Scarce clinical manifestations of prolactin-secreting pituitary tumors in children and adolescents make it difficult to timely diagnosis. Girls have more prevalence of microprolactinomas, manifests as a rule, menstrual irregularities. In males, the greater incidence of macroadenomas results in the presence of neuroophthalmologic signs. The diagnosis of prolactinomas requires both radiographic evidence of pituitary adenoma and laboratory analysis of sustained hyperprolactinemia.


2021 ◽  
Vol 10 (4) ◽  
pp. 825
Author(s):  
Carlos Nicolás Pérez-García ◽  
Daniel Enríquez-Vázquez ◽  
Manuel Méndez-Bailón ◽  
Carmen Olmos ◽  
Juan Carlos Gómez-Polo ◽  
...  

Introduction: The worldwide pandemic, coronavirus disease 2019 (COVID-19) is a novel infection with serious clinical manifestations, including death. Our aim is to describe the first non-ICU Spanish deceased series with COVID-19, comparing specifically between unexpected and expected deaths. Methods: In this single-centre study, all deceased inpatients with laboratory-confirmed COVID-19 who had died from March 4 to April 16, 2020 were consecutively included. Demographic, clinical, treatment, and laboratory data, were analyzed and compared between groups. Factors associated with unexpected death were identified by multivariable logistic regression methods. Results: In total, 324 deceased patients were included. Median age was 82 years (IQR 76–87); 55.9% males. The most common cardiovascular risk factors were hypertension (78.4%), hyperlipidemia (57.7%), and diabetes (34.3%). Other common comorbidities were chronic kidney disease (40.1%), chronic pulmonary disease (30.3%), active cancer (13%), and immunosuppression (13%). The Confusion, BUN, Respiratory Rate, Systolic BP and age ≥65 (CURB-65) score at admission was >2 in 40.7% of patients. During hospitalization, 77.8% of patients received antivirals, 43.3% systemic corticosteroids, and 22.2% full anticoagulation. The rate of bacterial co-infection was 5.5%, and 105 (32.4%) patients had an increased level of troponin I. The median time from initiation of therapy to death was 5 days (IQR 3.0–8.0). In 45 patients (13.9%), the death was exclusively attributed to COVID-19, and in 254 patients (78.4%), both COVID-19 and the clinical status before admission contributed to death. Progressive respiratory failure was the most frequent cause of death (92.0%). Twenty-five patients (7.7%) had an unexpected death. Factors independently associated with unexpected death were male sex, chronic kidney disease, insulin-treated diabetes, and functional independence. Conclusions: This case series provides in-depth characterization of hospitalized non-ICU COVID-19 patients who died in Madrid. Male sex, insulin-treated diabetes, chronic kidney disease, and independency for activities of daily living are predictors of unexpected death.


2020 ◽  
Vol 22 (Supplement_2) ◽  
pp. ii172-ii172
Author(s):  
Franco Rubino ◽  
Daniel Eichberg ◽  
Ashish shah ◽  
Evan Luther ◽  
Victor Lu ◽  
...  

Abstract BACKGROUND Tumors of the posterior pituitary are a distinct group of low-grade neoplasms arising from the sellar region. According to the most recent World Health Organization (WHO) guidelines, these tumors are grouped based on TTF-1 immunostain positivity. The coexistence of a posterior and anterior pituitary tumors is statistically extremely unlikely. OBJECTIVE In this study, we present a case of a 41-year-old woman suffering from Cushing syndrome with the presence of two pituitary masses on MRI. Also, the relevant literature is systematically reviewed. METHODS Two databases (PubMed and Google Scholar) and crossed references were systematically reviewed for TTF-1 pituitary tumors with coexisting pituitary adenoma or increased pituitary serum hormone levels. A literature review was performed according to PRISMA guidelines. Also, one patient from our institution is reported. Data regarding demographic features, type of anterior and posterior pituitary tumor, clinical features of the patients and lab results, MRI findings, and pathology results were extracted. RESULTS Seventeen retrospective case reports and case series were identified and included. Twenty-four patients were analyzed (including the reported case). The mean age was 46 ± 16 years, with a male-to-female ratio of 0.33:1. Pituicytomas and granular cell tumors were the only posterior pituitary tumors reported in 19 (79%) and 5 (21%) cases, respectively. There are only eight cases reported (including our patient) with a confirmed pituitary adenoma and a TTF-1 pituitary tumor (one GCT with GH-secreting adenoma, five pituicytoma with ACTH-secreting adenoma, one with GH-secreting adenoma and one with a null type adenoma). CONCLUSION We presented the eighth reported case of a TTF-1 pituitary tumor coexisting with a pituitary adenoma. We think that improving the amount of the tissue specimen available for pathology evaluation could increase the incidence of these very rare tumors.


2016 ◽  
Vol 13 (2) ◽  
pp. 63-67
Author(s):  
Prabin Shrestha ◽  
Anjana Rajbhandari

Pituitary tumors are one of the common brain tumors after gliomas, meningieomas and acoustic schwaanomas. It is commonly encountered in Nepal also as in other countries. Retrospective case series analytical study was done. The cases of pituitary adenoma from 2009 till 2015 were included. Types of pituitary adenoma, surgical technique, postoperative morbidities, mortalities and outcome were analyzed. Total 21 cases of pituitary adenoma were operated. All the cases were diagnosed by MRI, plain and contrast. In addition, thorough hormonal evaluation was done. All the cases were operated via transnasal transsphenoidal (TSS) approach. Microscope was used in all the cases whereas endoscope was used in selected cases. Preoperative lumbar puncture and CSF drain was not done in any case. Postoperative transient nasal CSF leak and diabetes insipidus were the most common surgery related morbidities in almost 50% cases which were totally cured with time. All the patients became asymptomatic and got better after surgery. Postoperative mortality and long term morbidity were nil. Recurrence was noted in one case after 3 years of surgery. Pituitary tumor surgery is safe and rewarding provided it is done carefully with skilled hands. TSS is a good approach with minimal postoperative complications. Better to have both microscope and endoscope for TSS.Nepal Journal of Neuroscience. Vol. 13, No. 2, 2016, Page:63-67


Sign in / Sign up

Export Citation Format

Share Document