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Ectopic TSH-secreting pituitary tumor: a case report and review of prior cases.

Song M, Wang H, Song L, Tian H, Ge Q, Li J, Zhu Y, Li J, Zhao R, Ji HL - BMC Cancer (2014)

Bottom Line: TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%.Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI).The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrinology, Weihai Municipal Hospital, 70 Heping Road, Weihai, Shandong 264200, China. whsmq1201@hotmail.com.

ABSTRACT

Background: Ectopic TSH-secreting pituitary adenoma (TSH-oma) is a very unusual disorder. To date, there are only four cases reported. It is difficult to distinguish ectopic cases from both regular TSH-omas and resistance to thyroid hormone (RTH).

Case presentation: A newly identified case of ectopic TSH-oma arising from the nasal pharynx was described, and reports of four prior cases were reviewed. The patient was a 41-year-old male who developed what appeared to be typical hyperthyroidism and atrial fibrillation in 2009. Thyroid function tests showed elevated basal levels of free T3 (FT3, 24.08 pmol/L), free T4 (FT4, 75.73 pmol/L), and serum TSH (7.26 μIU/ml). Both TSH-oma and resistance to thyroid hormone syndrome were considered. TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%. Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI). A normal pituitary was visualized. Ectopic TSH-oma was preliminarily established. Using an endoscopic endonasal approach, the tumor was resected. Histological features and immunophenotypes were consistent with those of TSH-secreting tumor. The levels of both free thyroxine and TSH returned to normal ranges the day after surgery and remained within normal range for 48 months.

Conclusions: Although exceedingly rare, ectopic TSH-oma should be considered for patients with inappropriate secretion of TSH with hyperthyroidism and pituitary tumor undetectable by computed tomography and MRI. To our knowledge, this is the first case followed up more than 4 years. The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

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Related in: MedlinePlus

TRH stimulating and octreotide inhibition tests. The unit for FT3 and FT4 is pmol/L, for PRL is ng/ml, and for sTSH is μIU/ml. A, TRH stimulating test. B, Octreotide inhibition test.
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Fig2: TRH stimulating and octreotide inhibition tests. The unit for FT3 and FT4 is pmol/L, for PRL is ng/ml, and for sTSH is μIU/ml. A, TRH stimulating test. B, Octreotide inhibition test.

Mentions: Over the course of the disease, the patient lost 6 kg of body weight. He had no symptoms of headache, nausea, dizziness, subnormal vision, impaired visual field, and obvious nasal obstruction. His physical examination was normal (T 36.7°C, P 80/min, R 17/min, BP120/80 mmHg, Ht 173 cm, Wt 65 Kg). He had a symmetrical figure, normal hair distribution, sweaty skin, normal superficial lymph nodes, and normal degree of convexity of eyeballs. Palpation revealed swelling of the thyroid gland, no nodules, medium texture, and no haphalgesia. Vascular murmur was not heard on auscultation. The patient had uneven cardiac sounds and arrhythmia with a heart rate of 100/min. No abdominal abnormalities were found. The proximal muscles did not show signs of atrophy. Mild tremor was observed when he raised his hands. The patellar tendon reflex was normal, and the pathological reflex was not observed. Lab and imaging results showed normal liver and kidney. TG-Ab <30%, TM-Ab <15%, GH 0.7 (normal <5.0 ng/ml), FSH 16.8 (normal 1.5-12 mIU/ml), LH 13.72 (normal 1.7-8.6 mIU/ml), PRL 14.9 (normal 4.1-18.4 ng/ml), and T 13.20 (normal 2.8-8.0 ng/ml). The blood electrolytes were within normal ranges: Ca 2.27 mmol/L, P 1.23 mmol/L, and K 3.87 mmol/L. Tumor biomarkers were analyzed, including carbohydrate antigen-199 12.26 (normal <37 U/ml), carcinoembryonic antigen 1.90 (normal <10 ng/ml), and neuron specific enolase (NSE) 22.31 (normal <16.3 ng/ml). Given a normal pituitary gland as pictured by magnetic resonance imaging (MRI) with gadolinium contrast, abnormal TSH level, and a large space-occupying lesion within the nasal cavity and the nasopharynx, with a maximum cross-section area of 1.9 × 1.7 cm (Figure 1C & D), as detected by CT scan, an ectopic TSH-secreting pituitary tumor was suspected. Emission computed tomography (ECT) demonstrated strong technetium-uptake by the thyroid. However, as shown in Figure 2A, the stimulating test was negative for TSH level was not up-regulated by TRH (Shanghai Lizhudongfeng Biologic Technology Inc., China). The amount of TSH was increased <2 μIU/ml. In comparison, the octreotide inhibition test was positive; a decrease of up to 30.8% in TSH level was observed (Figure 2B). Taken together, a diagnosis of ectopic TSH-secreting tumor in the nasopharynx was tentatively established. The strategy to combine resection and quick pathological examination was proposed to release patient’s financial burden and surgical stress. Nevertheless, his surgery was postponed due to treatment of the atrial fibrillation.


Ectopic TSH-secreting pituitary tumor: a case report and review of prior cases.

Song M, Wang H, Song L, Tian H, Ge Q, Li J, Zhu Y, Li J, Zhao R, Ji HL - BMC Cancer (2014)

TRH stimulating and octreotide inhibition tests. The unit for FT3 and FT4 is pmol/L, for PRL is ng/ml, and for sTSH is μIU/ml. A, TRH stimulating test. B, Octreotide inhibition test.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4125694&req=5

Fig2: TRH stimulating and octreotide inhibition tests. The unit for FT3 and FT4 is pmol/L, for PRL is ng/ml, and for sTSH is μIU/ml. A, TRH stimulating test. B, Octreotide inhibition test.
Mentions: Over the course of the disease, the patient lost 6 kg of body weight. He had no symptoms of headache, nausea, dizziness, subnormal vision, impaired visual field, and obvious nasal obstruction. His physical examination was normal (T 36.7°C, P 80/min, R 17/min, BP120/80 mmHg, Ht 173 cm, Wt 65 Kg). He had a symmetrical figure, normal hair distribution, sweaty skin, normal superficial lymph nodes, and normal degree of convexity of eyeballs. Palpation revealed swelling of the thyroid gland, no nodules, medium texture, and no haphalgesia. Vascular murmur was not heard on auscultation. The patient had uneven cardiac sounds and arrhythmia with a heart rate of 100/min. No abdominal abnormalities were found. The proximal muscles did not show signs of atrophy. Mild tremor was observed when he raised his hands. The patellar tendon reflex was normal, and the pathological reflex was not observed. Lab and imaging results showed normal liver and kidney. TG-Ab <30%, TM-Ab <15%, GH 0.7 (normal <5.0 ng/ml), FSH 16.8 (normal 1.5-12 mIU/ml), LH 13.72 (normal 1.7-8.6 mIU/ml), PRL 14.9 (normal 4.1-18.4 ng/ml), and T 13.20 (normal 2.8-8.0 ng/ml). The blood electrolytes were within normal ranges: Ca 2.27 mmol/L, P 1.23 mmol/L, and K 3.87 mmol/L. Tumor biomarkers were analyzed, including carbohydrate antigen-199 12.26 (normal <37 U/ml), carcinoembryonic antigen 1.90 (normal <10 ng/ml), and neuron specific enolase (NSE) 22.31 (normal <16.3 ng/ml). Given a normal pituitary gland as pictured by magnetic resonance imaging (MRI) with gadolinium contrast, abnormal TSH level, and a large space-occupying lesion within the nasal cavity and the nasopharynx, with a maximum cross-section area of 1.9 × 1.7 cm (Figure 1C & D), as detected by CT scan, an ectopic TSH-secreting pituitary tumor was suspected. Emission computed tomography (ECT) demonstrated strong technetium-uptake by the thyroid. However, as shown in Figure 2A, the stimulating test was negative for TSH level was not up-regulated by TRH (Shanghai Lizhudongfeng Biologic Technology Inc., China). The amount of TSH was increased <2 μIU/ml. In comparison, the octreotide inhibition test was positive; a decrease of up to 30.8% in TSH level was observed (Figure 2B). Taken together, a diagnosis of ectopic TSH-secreting tumor in the nasopharynx was tentatively established. The strategy to combine resection and quick pathological examination was proposed to release patient’s financial burden and surgical stress. Nevertheless, his surgery was postponed due to treatment of the atrial fibrillation.

Bottom Line: TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%.Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI).The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrinology, Weihai Municipal Hospital, 70 Heping Road, Weihai, Shandong 264200, China. whsmq1201@hotmail.com.

ABSTRACT

Background: Ectopic TSH-secreting pituitary adenoma (TSH-oma) is a very unusual disorder. To date, there are only four cases reported. It is difficult to distinguish ectopic cases from both regular TSH-omas and resistance to thyroid hormone (RTH).

Case presentation: A newly identified case of ectopic TSH-oma arising from the nasal pharynx was described, and reports of four prior cases were reviewed. The patient was a 41-year-old male who developed what appeared to be typical hyperthyroidism and atrial fibrillation in 2009. Thyroid function tests showed elevated basal levels of free T3 (FT3, 24.08 pmol/L), free T4 (FT4, 75.73 pmol/L), and serum TSH (7.26 μIU/ml). Both TSH-oma and resistance to thyroid hormone syndrome were considered. TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%. Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI). A normal pituitary was visualized. Ectopic TSH-oma was preliminarily established. Using an endoscopic endonasal approach, the tumor was resected. Histological features and immunophenotypes were consistent with those of TSH-secreting tumor. The levels of both free thyroxine and TSH returned to normal ranges the day after surgery and remained within normal range for 48 months.

Conclusions: Although exceedingly rare, ectopic TSH-oma should be considered for patients with inappropriate secretion of TSH with hyperthyroidism and pituitary tumor undetectable by computed tomography and MRI. To our knowledge, this is the first case followed up more than 4 years. The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

Show MeSH
Related in: MedlinePlus