Left Gland Research Articles

Analysis of the Parotid Glands on an Energy Spectrum CT Iodine Map to Evaluate Irradiation-Induced Acute Xerostomia in Patients With Nasopharyngeal Carcinoma.

Objective: This prospective study aims to evaluate acute irradiation-induced xerostomia during radiotherapy by utilizing the normalized iodine concentration (NIC) derived from energy spectrum computed tomography (CT) iodine maps. Methods: In this prospective study, we evaluated 28 patients diagnosed with nasopharyngeal carcinoma. At 4 distinct stages of radiotherapy (0, 10, 20, and 30 fractions), each patient underwent CT scans to generate iodine maps. The NIC of both the left and right parotid glands was obtained, with the NIC at the 0-fraction stage serving as the baseline measurement. After statistically comparing the NIC obtained in the arterial phase, early venous phase, late venous phase, and delayed phase, we chose the late venous iodine concentration as the NIC and proceeded to analyze the variations in NIC at each radiotherapy interval. Using the series of NIC values, we conducted hypothesis tests to evaluate the extent of change in NIC within the parotid gland across different stages. Furthermore, we identified the specific time point at which the NIC decay exhibited the most statistically significant results. In addition, we evaluated the xerostomia grades of the patients at these 4 stages, following the radiation therapy oncology group (RTOG) xerostomia evaluation standard, to draw comparisons with the changes observed in NIC. Results: The NIC in the late venous phase exhibited the highest level of statistical significance (P < .001). There was a noticeable attenuation in NIC as the RTOG dry mouth grade increased. Particularly, at the 20 fraction, the NIC experienced the most substantial attenuation (P < .001), a significant negative correlation was observed between the NIC of the left, right, and both parotid glands, and the RTOG evaluation grade of acute irradiation-induced xerostomia (P < .001, r = -0.46; P < .001, r = -0.45; P < .001, r = -0.47). The critical NIC values for the left, right, and both parotid glands when acute xerostomia occurred were 0.175, 0.185, and 0.345 mg/ml, respectively, with AUC = 0.73, AUC = 0.75, and AUC = 0.75. Conclusion: The NIC may be used to evaluate changes in parotid gland function during radiotherapy and acute irradiation-induced xerostomia.

Mixed Canine Mammary Gland Tumour in a 10 Year Old Lhasa Apso Bitch Histopathological Evaluation and Surgical Management

Mixed mammary gland tumors are the most frequent canine mammary gland neoplasms with a high incidence rate among females, however, there is paucity of studies that describe their clinic-pathological presentations and management approaches. This case evaluated the clinic-pathological data and management of mixed mammary gland tumors. A 10-year-old Lhasa Apso bitch weighing 11.4kg was referred to the University of Jos Veterinary Teaching Hospital with a ventral abdominal mass. Clinical examination revealed an enlarged left inguinal mammary gland with two palpable masses. The inguinal region was aseptically prepared for surgery and anesthesia achieved by the administration of 1% atropine sulphate (Atropine® - Shanxi Shuguang Pharmaceutical Co., Ltd., Qixian, China) and Chlorpromazine as preanesthetics and 50mg/mL Ketamine hydrochloride (Jawa ketamine® - Swiss Parenterals Ltd, Gujarat, India) + 2% Xylazine (Alfasan, Woerden-Holland) as induction and maintenance anesthetics. The glandular, connective/fibrous and fatty tissues of the mammary gland were carefully dissected to achieve the complete excision of the encapsulated mammary tumour masses before the reconstruction of the resulting defect. Postoperatively, penicillin and streptomycin, piroxicam, vitamin B complex, dexamethasone, vincristine sulphate and ivermectin were administered. The excised masses histologically presented numerous tubules with some containing mucin. Also there were the presence of acini with neoplastic epithelial cells and myoepithelial cells, cartilage and bony trabeculae. Surgical debulking with adjunctive immuno-chemotherapy was curative for the mixed mammary gland tumours in this bitch.

Out-of-field dosimetry using a validated PHITS model and computational phantom in clinical BNCT.

The out-of-field radiation dose for boron neutron capture therapy (BNCT), which results from both neutrons and γ-rays, has not been extensively evaluated. To safely perform BNCT, the neutron and γ-ray distributions inside the treatment room and the whole-body dose should be evaluated during commissioning. Although, certain previous studies have evaluated the whole-body dose in the clinical research phase, no institution providing BNCT covered by health insurance has yet validated the neutron distribution inside the room and the whole-body dose. To validate the Monte Carlo model of the BNCT irradiation room extended for the whole-body region and evaluate organ-at-risk (OAR) doses using the validated model with a human-body phantom. First, thermal neutron distribution inside the entire treatment room was measured by placing Au samples on the walls of the treatment room. Second, neutron and gamma-ray dose-rate distributions inside a human-body water phantom were measured. Both lying and sitting positions were considered. Bare Au, Au covered by Cd (Au+Cd), In, Al, and thermoluminescent dosimeters were arranged at 11 points corresponding to locations of the OARs inside the phantom. After the irradiation, γ-ray peaks emitted from the samples were measured by a high-purity germanium detector. The measured counts were converted to the reaction rate per unit charge of the sample. These measurements were compared with results of simulations performed with the Particle and Heavy Ion Transport code System (PHITS). A male adult mesh-type reference computational phantom was used to evaluate OAR doses in the whole-body region. The relative biological effectiveness (RBE)-weighted doses and dose-volume histograms (DVHs) for each OAR were evaluated. The median dose (D50% ) and near-maximum dose (D2% ) were evaluated for 14 OARs in a 1-h-irradiation process. The evaluated RBE-weighted doses were converted to equivalent doses in 2Gy fractions. Experimental results within 60cm from the irradiation center agreed with simulation results within the error bars except at±20, 30cm, and those over 70cm corresponded within one digit. The experimental results of reaction rates or γ-ray dose rate for lying and sitting positions agreed well with the simulation results within the error bars at 8, 4, 11, 7 and 7, 4, 7, 6, 5, 6 out of 11 points, respectively, for Au, Au+Cd, In, Al, and TLD. Among the detectors, the discrepancies in reaction rates between experiment and simulation were most common for Au+Cd, but were observed randomly for measurement points (brain, lung, etc.). The experimental results of γ-ray dose rates were systematically lower than simulation results at abdomen and waist regions for both positions. Extending the PHITS model to the whole-body region resulted in higher doses for all OARs, especially 0.13Gy-eq increase for D50% of the left salivary gland. The PHITS model for clinical BNCT for the whole-body region was validated, and the OAR doses were then evaluated. Clinicians and medical physicists should know that the out-of-field radiation increases the OAR dose in the whole-body region.

Double pituitary neuroendocrine tumors in a patient with normal growth hormone level acromegaly: A case report and review of the literature

Background: Acromegaly is a rare disease caused by growth hormone (GH) hypersecretion caused by a pituitary neuroendocrine tumor (PitNET). However, some acromegaly patients show normal GH levels, and they can be a pitfall in clinical diagnosis. Moreover, rarely, synchronous true double or multiple PitNETs are encountered. Moreover, these PitNETs increase the risk of a left lesion during surgical exploration. Case Description: The patient, who was a 73-year-old female, was referred to our hospital with a chief complaint of headache. Assessment of basal anterior pituitary function revealed a slightly high level of insulin-like growth factor-1 (IGF-1) (standard deviation, 2.4), and her physical findings exhibited mild acromegalic features. The endocrine evaluation confirmed acromegaly and magnetic resonance imaging (MRI) showed a macro PitNET with suprasellar extension. Endoscopic endonasal surgery (EES) was performed to remove the macro PitNET. Although postoperative MRI showed complete removal of the macro PitNET, endocrinological testing indicated no improvement in GH or IGF-1 excess. Pathological examination of the surgical specimen revealed a gonadotropic PitNET. Therefore, we repeated the MRI scan and found a micro PitNET in the thin left normal pituitary gland. A second EES was successfully performed to remove the micro PitNET completely, and both endocrinological and pathological examinations confirmed that the disease was cured. Conclusion: Diagnosing acromegaly with low GH levels requires close monitoring. Double PitNETs are relatively rare and can cause incomplete remission of functional PitNETs.

Accuracy of Four-Dimensional Computed Tomography and Different Imaging Modalities in Primary Hyperparathyroidism.

Background This study aimed to compare the accuracy of different imaging modalities in the preoperative localization of parathyroid pathology in primary hyperparathyroidism. Methodology This prospective study enrolled 70 patients who were biochemically diagnosed with primary hyperparathyroidism between 2021 and 2022 at our center. Patients underwent scanning using three imaging modalities, namely, Tc99m sestamibi scan (sestamibi), parathyroid ultrasonography, and four-dimensional computed tomography (4DCT). A descriptive analysis was performed to determine and compare the respective localizing sensitivities. Results The most common site of parathyroid adenoma (PA) was the left inferior parathyroid gland, seen in 28 (40%) patients. Three patients had false-positive imaging studies with no parathyroid pathology identified surgically or on histological examination. The median levels of parathyroid hormone decreased significantly (p < 0.001) after the surgery, with a median of 24.3 (1.90-121). Furthermore, 4DCT accomplished a sensitivity of 97.14% for diagnosing the side and 94.03% for overall localization of PA. This sensitivity was superior to the sensitivity of ultrasonography and sestamibi scan to detect the side and quadrant of the adenoma. 4DCT was significantly higher in sensitivity when compared to the combination of ultrasound and sestamibi (p < 0.001). Conclusions 4DCT yielded the highest sensitivity in localizing parathyroid pathology from the imaging modalities studied with the lowest false-negative rate. Using ultrasound with 4DCT could be the most cost-effective combination for detecting primary hyperparathyroidism.

Minimally Invasive Parathyroidectomy using Intraoperative Ultrasonography in Parathyroid Adenoma Patients with a History of Total Thyroidectomy

A 65-year-old patient who underwent total thyroidectomy 10 years ago was suspected of having a parathyroid adenoma, and minimally invasive parathyroidectomy was planned. Preoperative ultrasonography(USG) and 99mTc MIBI scan indicated a left lower parathyroid lesion. In the first operation, intraoperative parathyroid hormone monitoring (IOPTH) was not possible due to hospital circumstances. Although no adenomatous lesion was found in the expected surgical field, surgery was completed after removing lesions around the left lower parathyroid gland. However, post-surgery, parathyroid hormone did not decrease at all, so a second operation was performed with IOPTH preparation. In the second operation, intraoperative ultrasonography was performed, and a suspected adenoma lesion was removed from the left upper lesion. He has been under follow-up for 3 years without complications. Surgeon-peformed intraoperative USG and preoperative scintigraphy had advantages in determining the localization of parathyroid lesion even withiout IOPTH.

Effectiveness of low tube voltage scan in the exposure dose for lenses during paediatric thoracic CT examination: anthropomorphic phantoms study.

To determine whether using lower-tube voltage reduces the scattered dose for the lens during paediatric thoracic computed tomography (CT). Two paediatric anthropomorphic phantoms (ATOM Phantom, CIRS, Norfolk, Virginia, USA) representing a newborn and 5-year-old were placed on the gantry of CT scanner, and optically stimulated luminescence dosemeters were placed on the left and right lenses, in front of the left and right thyroid glands, in front of the left and right mammary glands, and in front of and behind the mammary gland level and we measured scattered dose of the optically stimulated luminescence dosemeter was compared for each phantom between 80 and 120kVp. Significant differences were observed in the scatter doses for the lens between 80 and 120kVp (p<0.01). Compared with the 120kVp scan, the scatter doses for the lens were ~15-40% lower in newborn and 5-year-olds using the 80kVp scan during paediatric CT.

Parathyroid carcinoma case-report in a patient with multinodular goiter and chronic kidney disease: difficulties in diagnosis and treatment (observation from practice)

We would like to present a clinical case of severe primary hyperparathyroidism due to a parathyroid carcinoma of atypical location in a patient with chronic kidney disease of complex etiology and multinodular goiter. Patient S., 59 years old, was followed-up for a long time in tertiary referral hospitals for “chronic tubulointerstitial nephritis with nephrosclerosis”, secondary hyperparathyroidism due to chronic kidney disease (CKD) G3–4, osteoporosis, and a multinodular euthyroid colloid goiter. In July 2021 she was referred to the Endocrinology Research Centre in order to clarify the diagnosis because of the persistence of an extremely high level of parathyroid hormone (PTH) despite cinacalcet treatment. During examination, primary hyperparathyroidism, a left parathyroid gland lesion, multinodular goiter with subclinical thyrotoxicosis, and vitamin D deficiency were diagnosed. After the removal of the left parathyroid gland lesion (histologically confirmed parathyroid carcinoma) and a left-sided hemithyroidectomy, hypocalcemia («hungry bone syndrome») developed, but the level of parathyroid hormone remained elevated. After 3–18 months after surgery, no data for relapse of primary hyperparathyroidism was obtained. The persistent moderate increase in PTH was regarded as secondary hyperparathyroidism in CKD and hypocalcemia. Complex therapy of osteoporosis with the antiresorptive drug denosumab, vitamin D and its active metabolite, calcium preparations, and parathyroidectomy led to a significant increase in bone mineral density (BMD) and no repeated fractures 18 months after surgery.Conclusion. In patients with pre-dialysis CKD and high PTH levels, it is necessary to make a differential diagnosis between primary (PHPT) and secondary hyperparathyroidism (SHPT). Severe manifestations of primary hyperparathyroidism can be suspicious for parathyroid carcinoma.

Corticotropin-producing pheochromocytoma in multiple endocrine neoplasia type 1

A clinical case of a man 66 y.o. who was diagnosed with hormone-inactive pituitary macroadenoma complicated by corneal erosion and partial atrophy of the optic nerve of the left eye due to exophthalmos. The increase in prolactin level was regarded due to a «stalk-effect». The patient underwent a transnasal pituitary adenomectomy with subsequent regression of symptoms. After 4 years, against the background of a new coronavirus infection, increasing general weakness, headaches, a crisis increase in blood pressure and tachycardia attacks appeared. Computed tomography (CT) accidentally revealed an adrenal incidentaloma, in laboratory tests - hypercortisolism, elevated ACTH levels, hypokalemia, hyperglycemia, increased levels of metanephrine and normetanephrine. The patient developed acute steroid psychosis, after which an adrenalectomy with a tumor was performed, a pheochromocytoma was histologically confirmed. After surgery, there was a regression of symptoms, the development of adrenal insufficiency with reduced levels of ACTH and cortisol. Upon further examination, a polynodose euthyroid goiter was established, the biopsy of the nodes - Hashimoto's thyroiditis (Bethesda II). Meanwhile, primary hyperparathyroidism was detected. According to ultrasound, scintigraphy with Ts99m-Technetril and CT revealed an increase of left parathyroid gland. A bilateral revision of the neck, removal of the right upper and left upper parathyroid adenomas were performed. In the postoperative period, the levels of calcium and parathyroid hormone were normalized. Given the presence of a combination of multiple tumors of the endocrine system (primary hyperparathyroidism, corticotropin-producing pheochromocytoma, hormone-inactive pituitary macroadenoma, polynodose euthyroid goiter), the MEN1 syndrome was clinically established. The study of 2 and 10 exons of the MEN1 gene revealed no mutations, which does not exclude the presence of a hereditary syndrome. The patient continues observation. In the available literature in Russian and English languages the case of ACTH pheochromocytoma as part of the MEN type 1 syndrome have not been found. Therefore, we consider the presented case to be the first one.

Unilateral adrenalectomy for a drug-resistant bilateral primary aldosteronism with heart failure: pathophysiology and surgical indication

BackgroundPatients with bilateral primary aldosteronism (PA) generally are treated with antihypertensive drugs, but optimal treatment for patients with complications due to refractory hypertension has not been established. In this report, we present a case with bilateral PA who presented with persistent hypertension, despite treatment with 6 drugs, and left-dominant heart failure, which was improved after unilateral adrenalectomy.Case presentationA 61-year-old man was admitted to our hospital because of severe left-dominant heart failure. His heart rhythm was atrial fibrillation and the left ventricle was diffusely hypertrophic and hypokinetic. Coronary arteries were normal on coronary arteriogram. Primary aldosteronism was suspected based on severe hypokalemia (2.5 mEq/L) and plasma aldosterone concentration (PAC; 1,410 pg/mL). Although computed tomography (CT) showed a single left cortical nodule, adrenal vein sampling (AVS) indicated bilateral PA. Early in the case, heart failure and hyperkalemia in this patient were improved by treatment with a combination of 6 antihypertensive drugs (spironolactone 25 mg/day, eplerenone 100 mg/day, azosemide 60 mg/day, tolvaptan 7.5 mg/day, enalapril 5 mg/day, and bisoprolol fumarate 10 mg/day); however, heart failure relapsed after four months of treatment. We hypothesized that hypertension caused by excess aldosterone was inducing the patient’s heart failure. In order to reduce aldosterone secretory tissue, a laparoscopic adrenalectomy was performed for the left adrenal gland, given the higher level of aldosterone from the left gland compared to the right. Following surgery, the patient’s heart failure was successfully controlled despite the persistence of high PAC. Treatment with anti-hypertensive medications was reduced to two drugs (eplerenone 100 mg/day and bisoprolol fumarate 10 mg/day). In order to elucidate the mechanism of drug resistance, immunohistochemistry (IHC) and real time-polymerase chain reaction (RT-PCR) assays were performed to assess the expression of steroidogenic factor 1 (SF-1), a regulator of steroid synthesis in adrenal tissue. IHC and RT-PCR demonstrated that the expression of SF-1 in this patient (at both the protein and mRNA levels) was higher than that observed in unilateral PA cases that showed good responsivity to drug treatment.ConclusionsUnilateral adrenalectomy to reduce aldosterone secretory tissue may be useful for patients with drug-refractory, bilateral PA. Elevated expression of SF-1 may be involved in drug resistance in PA.

Brown tumor due to primary hyperparathyroidism in a familial case: a case report

BackgroundPrimary hyperparathyroidism (PHPT) is an uncommon disorder characterised by hypercalcemia with an increased parathyroid hormone level. We reported a PHPT familial case with two subjects, a father and a daughter, and both of them had suffered from the brown tumor.Case presentationThe proband, a 43-year-old patient, developed parathyroid adenomas at the age of 15; a histologically confirmed right parathyroid adenoma was removed by parathyroidectomy; and after six months follow-up, the serum calcium level was normalised. At the age of thirty-three, a CT scan of his head and neck revealed a mass in the right maxilla, as well as PHPT (i.e., left inferior parathyroid adenoma). Then, he underwent a biopsy of an exophytic lesion in the right maxilla and was diagnosed by pathology as a brown tumor, with the serum calcium and PTH levels at 2.78 mmol/L and 221 pg/mL, respectively. Subsequently, the patient took a left inferior parathyroid microwave ablation with ultrasound guidance. After three months of follow-up, the serum calcium and PTH levels returned to normal, and the brown tumor was resolved. After three years, it mineralised as revealed in a CT scan. By the time he was 43 years old, during the 28-year follow-up period, the serum calcium and PTH levels were still within the normal range, and there was no discomfort reported. He has consistently taken calcium supplements throughout the 28 years. Since the initial diagnosis, his blood indicators of kidney function have been normal, and ultrasound showed renal calculus in the right kidney and a normal left kidney. The proband’s daughter, a 15-year-old girl, experienced left upper extremity pain for ten months. CT scan revealed a mass in the distal left radius, and a giant cell tumor was suspected. A surgical internal fixation was performed, and the pathology showed a brown tumor. Laboratory tests revealed a serum parathyroid hormone (PTH) level of 1554pg/mL, calcium level of 3.14 mmol/L, phosphorus level of 0.72 mmol/L, and alkaline phosphatase level of 1892 U/L. Given the osteitic changes and elevated levels of calcium and PTH, ultrasonography was performed, after which a mass was detected measuring 19 × 9 × 7 mm mixed with solid components and cystic fluid in the right thyroid gland. The results of 99mTc-MIBI scintigraphy confirmed the abnormal accumulation of 99mTc-MIBI in the right thyroid gland but not seen in the bilateral parathyroid glands. The patient underwent thyroidectomy, and the postoperative pathology report indicated an intra-thyroid ectopic parathyroid adenoma. The serum calcium and PTH levels became normal at 4 h after surgery. One to three months after operation, the serum calcium level was low, while the serum PTH level was high. Then, the patient was advised to take calcium supplements. Until the sixth month after the operation, the serum calcium level and serum PTH level returned to normal, and the bone pain was relieved. The patient’s blood tests for kidney function remained normal. There was no evidence of bilateral kidney disease (such as nephrolithiasis or nephrocalcinosis) detected by ultrasound scan. There were several similarities in the state of illness between these two subjects. Both the father and the daughter developed parathyroid adenomas at the age of 15, and there was no lesion in other endocrine glands. And genetic testing revealed mutations in the CDC73 genes in both father and daughter. On the other hand, there were also a few differences. The father’s first signs of brown tumor were in the right maxilla, while the daughter’s appeared in the distal left radius. The father presented pathological changes in the left and right parathyroid glands, whereas the daughter presented with an ectopic parathyroid adenoma in the right thyroid gland.ConclusionWe report a familial case in which father and daughter were diagnosed to have brown tumors due to parathyroid adenoma and ectopic parathyroid adenoma, and genetic testing revealed CDC73 gene mutations in both. Therefore, in the diagnostic and differential process of young patients having bone disease, clinicians should not only focus on the clinical manifestations of the skeleton, but also implement a comprehensive analysis of systemic symptoms, considering the possibility that the patient has familial PHPT.

SAT567 Collision Tumor Of The Thyroid With Papillary Thyroid Carcinoma And Metastatic Renal Clear Cell Carcinoma With Concomitant Parathyroid Adenoma

Abstract Disclosure: C.M. Mirano: None. R.C. Mirasol: None. INTRODUCTION Collision tumors of the thyroid are rare diseases that have two or more histologically and morphologically distinct tumors that occur simultaneously within the thyroid. There are only a few reported cases of collision tumors involving a metastasis from a distant tumor and a papillary thyroid cancer. In addition, the coexistence of Primary Hyperparathyroidism (PHPT) with nonmedullary thyroid cancers is unusual with an incidence range between 2 to 24%. Here, we describe an unusual case of a patient with Papillary Thyroid Carcinoma and Metastatic Renal Clear Cell Carcinoma complicated with a Parathyroid Adenoma. CASE The patient is a 65 year-old female, known Renal Clear Cell Carcinoma Stage IV who underwent left nephrectomy, distal pancreatectomy splenectomy, and adjuvant chemotherapy. She then developed subclinical hyperthyroidism with bilateral thyroid nodules on ultrasound 13 years post nephrectomy. Thyroid Scintigraphy showed hypofunctioning nodules on both lobes. Biopsy of the thyroid nodules was done on showing a Metastatic Renal Cell Carcinoma. Laboratory tests also showed hypercalcemia and an elevated iPTH. SPECT-CT images showed uptake in the left thyroid suggestive of a parathyroid adenoma. She underwent four gland exploration with parathyroidectomy. Intraoperatively, pre-excision PTH assay extracted peripherally was 386.10 pg/mL. The right superior, right inferior, and left superior parathyroid gland were normal in size. The left inferior parathyroid gland was found to be enlarged measuring 2.5 x 2.0 cm, intrathyroidal in location and was encapsulated. The left inferior parathyroid was dissected and sent for histopathology. 10 minutes post excision PTH levels declined by 57%. After which, total thyroidectomy was done with note of a 2.5 x 2 cm firm mass at the right thyroid lobe slightly adherent to strap muscles. The left thyroid lobe had no palpable nodules, Histopathology of the thyroid isthmus showed one focus of follicular cells exhibiting nuclear changes consistent with papillary thyroid carcinoma. Microsections of the right thyroid lobe show a malignant neoplasm composed of atypical cells arranged in sheets that have enlarged, hyperchromatic to vesicular, pleomorphic, prominent nucleoli, irregular nuclear border, and clear cytoplasm consistent with metastatic clear cell carcinoma. Histopathology of the Left inferior parathyroid gland revealed findings consistent with Parathyroid Adenoma. She was discharged stable with normal calcium levels. CONCLUSION Collision tumors are rare and an adequate histologic assessment of these tumors are important for decisions regarding management. Differentials with patients with nodular goiter should always include metastatic disease in the background of an underlying carcinoma. It is equally important to be aware of concomitant PHPT and thyroid diseases in the management of patients with hypercalcemia. Presentation: Saturday, June 17, 2023

FRI206 A Rare Case Of Coexisting Acromegaly And Adrenal Cushing’s Syndrome

Abstract Disclosure: T. Chamakkala: None. T. Gallagher: None. Introduction: Acromegaly has been associated with higher risk of cancer morbidity and mortality. Acromegaly coexisting with adrenal Cushing’s disease has been rarely described in literature. We present a patient who was found to have acromegaly during evaluation for Cushing’s syndrome. Clinical Case: A 50-year-old woman presented to the office for endocrinology evaluation at the referral of PCP for elevated 24h urine cortisol level 176ug/24h. She had noticed facial plethora over several years, fatigue, and easy bruising. She had recently been diagnosed with hypertension, poorly controlled on two medications at the time of evaluation. Repeat 24h urine cortisol and midnight salivary cortisol levels were elevated; dexamethasone suppression test was positive. ACTH was &amp;lt;1.5pg/mL, consistent with ACTH independent hypercortisolemia. Adrenal CT showed 2.6x3cm left adrenal adenoma. She underwent left adrenalectomy and pathology showed adrenal cortical adenoma. During evaluation for Cushing’s disease and new onset hypertension, IGF1 was evaluated and found to be elevated at 354ng/mL. She additionally endorsed symptoms of worsening carpal tunnel syndrome and increasing ring size. After adrenalectomy, IGF1 was repeated and remained elevated at 429ng/mL. Growth hormone suppression test showed nadir of 0.56ng/mL which is higher than &amp;lt;0.3ng/mL expected of high sensitivity GH assays. Pituitary MRI showed three suspected pituitary microadenomas, 4mm and 5mm in left gland, 7mm in right gland. Given difficulty with surgical approach and higher risk of hypopituitarism, she was started on lanreotide for treatment without surgery. She underwent genetic counseling and was offered genetic testing. Conclusion: This case highlights the rare coexistence of acromegaly and adrenal Cushing’s disease in a 50 year old woman. Though the relationship between acromegaly and cancer has been historically controversial, more recent studies have shown an increased risk of both benign and malignant tumors with acromegaly. Untreated Cushing’s Disease has been associated with mildly elevated IGF1 which then decreases after remission; it is unclear if the same pathological process is involved in other etiologies of hypercortisolemia like adrenal Cushing’s syndrome, but re-evaluation of IGF1 after treatment of Cushing’s should be considered. As Cushing’s and acromegaly share similar consequences such as worsening HTN, changing facial appearance, and worsening diabetes, it is prudent to consider each as differential diagnoses when signs do not remit after treatment. Acromegaly has additionally been associated with other endocrinopathies in syndromes including MEN1 syndrome, McCune Albright Syndrome, and Carney Complex. Evaluation for these syndromes and consideration for genetic testing should be considered in the evaluation of a patient with acromegaly and additional associated endocrinopathies. Presentation: Friday, June 16, 2023

SAT192 Non-syndromal Primary Hyperparathyroidism Presenting As Asymptomatic Hypercalcemia In A Teenage Patient.

Abstract Disclosure: S. Humayon: None. M. Shah: None. P.N. Kabir: None. C.A. Penaherrera: None. Introduction: Primary hyperparathyroidism is the most common cause of hypercalcemia in the general population, and is often discovered incidentally. The condition rarely presents in patients under age 18, particularly when it is not associated with genetic syndromes such as MEN 1. Case presentation: A previously healthy 17-year-old female was referred to Endocrinology clinic for evaluation of positive thyroid peroxidase antibodies with normal TSH and free T4. Review of laboratory results from 6 months prior, revealed an elevated total calcium level of 12.3 mg/dl. Patient at the time denied any symptoms of hypercalcemia. Repeat testing revealed a calcium level of 13.1 mg/dl, PTH level of 183 pg/ml, alkaline phosphatase 232 IU/L. Her vitamin D level was 28 ng/ml, with elevated calcitriol level of 362 ng/ml. The patient denied a history of kidney stones or other endocrine abnormalities in the past. She did mention a family history of hypercalcemia in her father requiring neck surgery. Patient was admitted to the hospital for management of hypercalcemia with intravenous fluid hydration. Subsequent work-up included a sestamibi parathyroid scan which revealed an asymmetric area of delayed uptake in the left thyroid gland, suggestive of a single parathyroid adenoma. The patient underwent single-gland parathyroidectomy, during which PTH levels decreased from 134 pg/ml to 15 pg/ml. She recovered uneventfully from the procedure and was started on oral calcium supplementation, which was tapered two months after surgery since there was no evidence of post-operative hypoparathyroidism. Conclusion: Primary hyperparathyroidism, when presenting prior to the age of 30, is most commonly associated with genetic syndromes such as MEN1 and typically presents as multi-glandular hyperplasia. Isolated primary hyperparathyroidism, secondary to a single parathyroid adenoma is seen less frequently in this younger subset of patients. This can be found in the absence of other endocrine abnormalities, with or without the presence of a family history. Prompt identification of hypercalcemia followed by evaluation of the same, may prevent delays in diagnosis and subsequent management of primary hyperparathyroidism. This can help prevent morbidity associated with hypercalcemia. Presentation: Saturday, June 17, 2023

SAT197 A Rare Case Of Infected Parathyroid Cyst Associated With Hyperparathyroidism

Abstract Disclosure: B. Ataallah: None. M. Al Tameemi: None. P.R. Orlander: None. Introduction: Parathyroid cysts are considered rare lesions compromising 1-5 % of neck masses. Clinical pictures can range from asymptomatic to symptoms and signs of hyperparathyroidism, and it may lead to compressive symptoms as well. Infection of the parathyroid cyst is not reported in literature. We are presenting a patient with parathyroid cyst and was found to be infected with E-coli. Case presentation: A 59-year-old woman presented with neck swelling and ear pain. She was initially diagnosed with otitis externa and was treated with antibiotics. Her neck swelling worsens over a brief period with associated ear drainage and shortness of breath. Computed tomography (CT) scan of the neck revealed a large cystic lesion up to 10 cm in largest diameter in the region of the thyroid gland with inflammatory changes extending to the prevertebral soft tissues. CT scan of the chest showed a mass effect on the trachea and adjacent structures with possible hemorrhage. Eventually the patient was intubated. Laboratory evaluation revealed parathyroid hormone (PTH) at 455 pg/ml, corrected calcium of 11.3 mg/dl, Ionized calcium of 1.41 mMol/L, and phosphorus at 3.0 mg/dl. Aspiration of the cyst was done prior to surgery and the culture grew E-coli. She started on antibiotics, and eventually underwent a total thyroidectomy with excision of the parathyroid cyst and adenoma. Pathology showed a ruptured cyst with marked inflammation and granulation tissue with hypercellular left superior parathyroid gland. Postoperatively, corrected calcium dropped to 10.1mg/dl and PTH to 38.5 pg/ml. Discussion: Parathyroid cyst (PC) is a relatively rare neck or mediastinal lesion, representing less than 0.5% of parathyroid gland disorders. Most PCs are nonfunctional, but some may produce excess parathyroid hormone or can cause local compressive symptoms like dyspnea, dysphagia, or hoarseness of voice, depending on the size and location of the lesion. There are no distinct clinical or radiological features to differentiate parathyroid cysts, and that makes preoperative diagnosis challenging, and most cases are diagnosed during surgery or postoperatively. Aspiration and fluid analysis can confirm the diagnosis preoperatively, and the treatment can be achieved either by cyst aspiration for small lesions with less than 2.5 cm in diameter, through sclerosing agent injection, or by surgical resection. In this patient, the culture of the aspirated parathyroid cyst surprisingly grew E coli. It is unusual to have an infection with E coli in the neck tissues or the surrounding structures, and it is unknown whether the cyst infection is related to the ear infection that our patient has. Parathyroid cyst infection is not reported in literature reviews, and it is important to consider fluid analysis and culture to confirm the diagnosis preoperatively and to rule out cyst infection Presentation: Saturday, June 17, 2023

SAT194 A Rare Case Of Untreated Primary Hyperparathyroidism With Extremely High PTH And Lytic Bone Lesions

Abstract Disclosure: M. Shah: None. K. Akinola: None. M. Bollineni: None. S.A. Milan: None. S. Challagulla: None. Background: Primary hyperparathyroidism (PHPT) typically presents with PTH levels two times above the upper limit of normal. Given the increase in routine calcium measurements, PHPT is usually diagnosed and treated early and skeletal complications are not frequent at presentation. We present a case of PHPT with severely elevated PTH levels and bone lesions mimicking metastatic disease. Clinical Case: A 30-year-old male with a history of untreated primary hyperparathyroidism presented with severe chest wall pain and abdominal pain. Initial laboratory tests revealed a calcium level of 13.3 mg/dL (normal 8.6 to 10.5 mg/dL), alkaline phosphatase (AP) of 747 IU/L (normal 24 to 130 IU/L), and PTH of 2485 pg/mL (normal 9-77 pg/mL). The patient was diagnosed with primary hyperparathyroidism years before this presentation, but was lost to follow up due to lack of insurance. His PTH was first noted to be elevated in 2013 at 266 pg/mL. CT of the chest, abdomen, pelvis showed a 2.8 x 3.0 x 3.3 cm anterior mediastinal mass and diffuse mixed lytic and sclerotic bone lesions consistent with metastatic disease. The patient underwent further imaging with CT 4D parathyroid which revealed a 1.1 cm left inferior thyroid nodule and an anterior mediastinal mass of unclear etiology. Given the high concern for malignancy, a biopsy of the mediastinal mass was performed and returned as hypercellular parathyroid tissue with no atypia. Bone biopsies of the sclerotic lesions of the iliac crest were consistent with osseous tissue with prominent bone remodeling and fibrous tissue; no neoplasm was seen. He was treated with IV fluids and zoledronic acid during this work up which only temporarily improved his high calcium levels. He was then placed on cinacalcet prior to surgery. He ultimately underwent resection of the anterior mediastinal mass and left inferior parathyroid gland via a cervical approach. The patient’s preoperative PTH level was 3455 pg/mL and decreased to 209 pg/mL immediately after resection. Resection of the other parathyroid glands was not done due to the high risk of postoperative hypocalcemia. Pathology of both lesions returned as intrathymic parathyroid adenoma with focal fibrosis. Postoperatively, the patient developed symptomatic hypocalcemia as low as 6.4 mg/dL. He required IV and oral calcium for approximately 3 weeks postoperatively, consistent with hungry bone syndrome. He was discharged on an oral regimen of calcium supplementation. Discussion: Long standing untreated PHPT can present with severely elevated PTH levels and skeletal manifestations commonly mistaken for metastatic lesions. As in this case, parathyroidectomy was the only effective treatment. However, it can be complicated by hungry bone syndrome, especially in a patient with significant bone remodeling as evidenced by high AP and lytic bone lesions. Presentation: Saturday, June 17, 2023

SAT215 Sagliker Syndrome In A Filipino Female With End Stage Renal Disease

Abstract Disclosure: M.R. Pajanel: None. E. Clarito: None. K. Uytiepo: None. P. De La Peña: None. This is a case of a 29-year-old Filipina, with chronic kidney disease secondary to chronic glomerulonephritis, undergoing hemodialysis for 10 years. She presented with maxillary mass, associated with other craniofacial deformities such as mandibular enlargement and widening of teeth spaces. Other associated symptoms include difficulty in swallowing solid food, difficulty in ambulation due to bone pains, and with noted shortening of stature. Workup revealed elevated intact parathyroid hormone (iPTH) 1638.8pg/mL(20x ULN), low Vitamin D, elevated ALP, but with normal levels of calcium and phosphorus. Sestamibi scan showed focal uptake and retention along medial aspect of the right thyroid bed and inferior to both thyroid beds suggestive of parathyroid adenoma or hyperplasia. Parathyroid ultrasound was consistent with findings of Sestamibi scan which also showed parathyroid nodules in the middle segment right thyroid lobe, and inferior to right thyroid lobe. Bone mineral density is below the expected range for age. Right parathyroidectomy was done and she was discharged well. She came in for follow-up with persistent elevated PTH 4765pg/ml(60x ULN), still with symptoms. Repeat imaging on ultrasound showed a parathyroid nodule posterior to the superior aspect of left thyroid 0.9 x0.5cm. Sestamibi scan confirmed a functioning parathyroid adenoma left inferior and superior thyroid bed. Left parathyroidectomy was done with a histopathologic result of left superior parathyroid consistent with parathyroid hyperplasia. On follow up, repeat intact PTH still elevated at 3677pg/mL and persistently elevated 2 months after at 3626pg/mL. Parathyroid ultrasound showed Parathyroid nodule, left, measuring 1.5x1.7x0.9 cm, inferior and posterior to the inferior border of the left thyroid gland. 99mTc-Sestamibi scan was consistent with the ultrasound in the left thyroid lobe suggestive of parathyroid adenomas or hyperplasia. Completion Parathyroidectomy was done on November 2018. Pre-operative iPTH was 4205pg/mL, which decreased to 291.3pg/mL postoperatively. She was discharged improved. On followup in 2022, she reports of decrease bone pain and now able to ambulate without difficulty. Repeat bone mineral density has improved and now within expected range for age. CONCLUSION: Sagliker syndrome is an entity described as an “uglyfying face syndrome” seen in patients with chronic kidney disease, presenting with facial deformities. Patients usually present with elevated parathyroid hormone (PTH) levels, and has been refractory to medical therapy. Surgical treatment has been the course of approach to prevent further clinical decline of patients. Identification of the syndrome is important for clinicians to know when to maximize medical management, and to identify patients who will benefit with early surgical management. The patient’s quality of life improved even 4 years post-surgery. Presentation: Saturday, June 17, 2023

SAT534 A Rare Case of Poorly Differentiated Thyroid Cancer With Extraordinary Limitations to Treatment

Abstract Disclosure: N. Shahid: None. L. Rudman: None. H. Maldonado Rivera: None. J. Chambi-Torres: None. J.A. Gonzalez: None. S. Valepu: None. A. Barco: None. N.H. Shaaban: None. E. Angueira: None. Background: Poorly differentiated thyroid cancer (PDTC) is a rare and aggressive form of follicular epithelial cell derived thyroid carcinoma, representing 3-5% of all thyroid cancers. As a subtype, PDTC is in a category that is between well differentiated thyrocyte malignancies and undifferentiated (anaplastic) thyroid carcinomas. Current guidelines for treatment of PDTC are not well defined. Case presentation: A 56-year-old male with a history of PDTC was admitted to the hospital directly from a correctional facility due to concern for metastasis. Patient had a total thyroidectomy performed just two months ago that revealed PDTC on surgical pathology. Recommendations at that time were to complete a full body PET-CT and follow up with radiation oncology for radiation and chemotherapy. Patient was also discharged on Synthroid 175 mcg daily for TSH suppression. However, the patient did not have any follow up appointments scheduled while at the facility. PET-CT showed stable FDG avid 2.4 cm lower right neck mass and 2 cm focus of mildly increased activity in the right lobe of the liver. On admission, a triple phase MRI liver protocol was ordered and showed a 15 mm hepatic lesion that was arterially enhancing with persistent enhancement in venous and delayed phase without dropping signal on T2 fat and additional hyperintensity on T2. Bilateral 1.7 cm adrenal nodule was also identified with relative hypointensity on T2. FNA of right neck mass was performed but did not yield sufficient tissue for pathology. Subsequent right neck exploration and skin biopsies were obtained and revealed benign fibroadipose tissue with focal fat necrosis in the thyroid and lobular capillary hemangioma in right back, left chest, and right abdomen. Patient’s status as an inmate continued to delay radiation and chemotherapy for PDTC with possible metastasis. The decision was made with the hematology/oncology inpatient consult team to perform RAI treatment with 138 mCI of I-131. Synthroid was held in anticipation of the RAI and whole body scan was ordered for 1 week after administration of I-131. Post ablation whole body scan showed intense radiotracer uptake over the region of the left and right thyroid glands and normal uptake in the stomach, small bowel, and urinary bladder. No other areas of radiotracer uptake were found. Conclusion: Diagnosis and treatment of PDTC is challenging and the social constraints as an inmate make it even more difficult to initiate appropriate therapy with radiation oncology follow up. Our case illustrates the implications of RAI ablation after total thyroidectomy in staging and therapy of PDTC. Given the intermediate histologic characteristics of PDTC, further investigation is required in the setting of metastasis and exploration of the benefits of early aggressive therapy, such as external beam radiotherapy. Presentation Date: Saturday, June 17, 2023

Normal intellectual ability and hyperprolactinemia as unique clinical manifestations of congenital hypothyroidism: A case report and review of hypotheses.

Congenital hypothyroidism is the deficiency of thyroid hormone in infants and hyperprolactinemia is frequently observed. Previously reported cases typically involve intellectual disability, highlighting this particular unique case report to the first reported patient demonstrating normal intellectual ability despite experiencing growth and gonad dysfunction. This study aims to present a case and review medical hypotheses related to the patient's condition. A 19-year-old female presented with a chief complaint of irregular menstruation for up to 40 days or not occurring at all. The patient experienced the first menstruation at the age of 16 years old. The patient's height was 133 cm, body weight 40 kg, and body mass index 22.61 kg/m2; other family members were normal. Physical examination showed no abnormalities, and laboratory examination showed suppressed serum free T4 (FT4) level (6.41 pmol/L), elevated thyroid stimulating hormone (TSH) level (333.700 µIU/mL), and elevated prolactin hormone level (32.03 ng/mL). Ultrasound of the thyroid gland found hypoplasia of the left and right thyroid glands. The patient was a college student enrolled in a public national university and had never complained about academic performance throughout the patient's education. The patient was diagnosed with congenital hypothyroidism and hyperprolactinemia. The patient was administered up to 100 µg daily of oral levothyroxine, which improved the patient's menstrual cycles. The patient's delayed diagnosis may be attributed to central congenital hypothyroidism being underdiagnosed. We hypothesized that thyroid-releasing hormone receptor (TRHR) gene mutation might contribute to the underlying cause of hyperprolactinemia and normal intellectual ability of the patient. Further study on the significance of TRHR gene mutations in congenital hypothyroidism is required to improve diagnosis and treatment.

Multiple Bone Cysts Caused by Hyperparathyroidism: A Case Report and Review of the Literature.

Brown tumor, a skeletal complication of severe hyperparathyroidism, comprises reparative granulation tissue and proliferating fibrous tissue with hemosiderin deposition. Multiple brown tumors are extremely rare complications of primary hyperparathyroidism. A 41-year-old woman presented with pain in the left knee. Radiography showed multiple cystic lesions in both femurs and the left proximal tibia, and additional radiography showed multiple cystic lesions in the left humerus and ulna. Magnetic resonance imaging (MRI) revealed multiple cystic lesions in the bilateral femurs, left proximal tibia, and ilium. Laboratory tests revealed hypercalcemia (albumin-corrected calcium level, 13.9 mg/dl), hypophosphatemia (phosphate level, 1.6 mg/dl), elevated level of alkaline phosphatase level (614 U/l), and markedly elevated parathyroid hormone (PTH) level (1,070 pg/ml; normal range=10-65 ng/l). 99mTc-hexakis-2-methoxyisobutyl-isonitrile scintigraphy revealed tracer accumulation in the left upper parathyroid gland, which was consistent with parathyroid tumor. Although resection of the parathyroid tumor was planned, the patient developed parathyroid apoplexy before tumor excision. After the parathyroid apoplexy, serum calcium and PTH levels temporarily normalized. Resurgence of the PTH level was observed 2 years after the diagnosis, and the patient underwent left upper parathyroidectomy. One year after the tumor excision, the patient had no symptoms, and MRI showed shrinkage of the cystic bone lesions. This report emphasizes the importance of considering hyperparathy-roidism as a differential diagnosis for patients with multiple bone lesions.