Gland Involvement Research Articles

A Rare Case of Adenoid Cystic Carcinoma of the External Auditory Canal

Malignancy of the external auditory canal (EAC) is rare, and adenoid cystic carcinoma (ACC) of EAC is even rarer. It is generally managed by radical resection with a clear oncological margin, followed by radiotherapy. However, local recurrence and distant metastasis are still the major concerns during follow-up. The authors present a case in a 32-year-old female, presenting with a mass in the EAC which was endoscopically removed. Histopathological examination revealed an ACC. Further imaging confirmed absence of distant metastasis and parotid gland involvement. The patient was treated with right modified lateral temporal bone resection and achieved clear surgical margins and thus planned for a long-term follow-up.

Orbital and Ocular Adnexal Lymphomas: A Retrospective Single Center Study

Aim: To evaluate and present the clinical and histopathologic features of patients with orbital and adnexal lymphoma.Methods: Eight patients who had histologically proven orbital and adnexal lymphoma between 2011 and 2021 were evaluated retrospectively. The clinical appearance, age, sex, imaging, tumor location, treatment methods, pathologic diagnosis, and systemic features of the patients were obtained from patient files.Results: The mean age of the patients was 59.1 (range, 42-79) years. Six of the patients were male and two were female. Painless mass and orbital swelling were the most common first signs and symptoms in the patients. The magnetic resonance imaging findings demonstrated unilateral involvement in six patients and bilateral involvement in two patients. Intraorbital location in three patients (behind the orbital septum), lid location in two patients (eyelids in front of the orbital septum), conjunctival involvement in one patient, and lacrimal gland involvement in two patients were detected. All cases were reported as non-Hodgkin B-cell lymphoma [primary extranodal marginal lymphoma of the mucosa-associated lymphoid tissue (n=5), diffuse large B-cell lymphoma (n=2), follicular lymphoma (n=1)].Conclusion: Orbital lymphomas can be seen more frequently among orbital tumors and clinical findings vary according to the location in the orbit. Ophthalmologists should be attentive to orbital swellings and consider orbital and ocular adnexal lymphomas among differential diagnoses.

KIMURA’S DISEASE IN AN ASIAN MALE: A VERY RARE CAUSE OF LYMPHADENOPATHY

The present case study highlights Kimura’s Disease in an Asian Male. Kimura disease (KD) is a rare chronic inflammatory disorder of unknown etiology. It usually presents as subcutaneous mass in the head and neck region and is frequently associated with regional lymphadenopathy or salivary gland involvement. A study done at Dept of Internal Medicine ,American Institute Of Medical sciences, GBH general hospital, Udaipur, Rajasthan, India. A case report of a 13 year old male, admitted to GBH general Hospital on 1st January, 2023 with chief complains of swelling (painless) Left side of the face below eye since 4 months and swelling under Rt. lobe of the ear since 3 months.On radiological examination, Kimura’s disease mimics other chronic and malignant diseases such as tuberculosis or lymphoma. This is an important study since it is a rare case and further research should be done in this area for further management and prompt diagnosis of such rare causes of upper body lymphadenopathy(frequently confused with malignancy),unless histopathology is clearly demonstrated.

Bacteremia and adrenal gland abscess due to Nocardia cyriacigeorgica: a case report and review

BackgroundNocardia cyriacigeorgica is one of the most common Nocardia species found in human infections, recently reclassified. Even though Nocardia may affect all organs by hematogenous dissemination, bacteremia are uncommon. Among all possible dissemination sites, the involvement of the adrenal glands is particularly rare.Case presentationWe report here a rare case of Nocardia disseminated infection with notably bacteremia and adrenal gland abscess, in a 77-years-old immunocompetent man. Adrenal gland abscess diagnosis was made by imaging (computerized tomography, magnetic resonance and positron emission tomography scan). A complete regression of all lesions including the left adrenal gland was obtained after 6 months of antibiotics. A review of literature was also performed.ConclusionNocardia bacteremia is a rare event but blood cultures may help to improve detection of Nocardia spp. in a non-invasive way. Adrenal abscess due to Nocardia spp. is very rare with only fourteen cases reported in the literature, but it is a true cause of adrenal masses. Our report suggests that clinician should be aware of this rare location and prioritize a non-invasive diagnosis strategy.

Kimura's disease: A clinicopathological study of 23 cases.

Kimura's disease (KD) is an uncommon lymphoproliferative fibroinflammatory disorder. Patients present with head and neck subcutaneous nodules with or without lymphadenopathy. Peripheral blood eosinophilia and elevated serum immunoglobulin E (IgE) levels are typical. This study was designed to delineate the clinicopathological features, pattern of care, and disease course of 23 Taiwanese patients with KD. We retrospectively analyzed the clinical data of 23 consecutive cases (16 male and 7 female; age at diagnosis: 12-77 years) of KD diagnosed at our institution from 2015 to 2020. The median time from presentation to diagnosis was 1 month. Twenty-one patients presented with unilateral or bilateral head and neck masses. The remaining two presented with right flank and right arm lesions, respectively. Peripheral blood eosinophilia was observed in nine, and elevated IgE levels were observed in four. All were diagnosed using either excisional or core-needle biopsy. Seven patients underwent fine needle aspiration without a diagnostic yield. Salivary gland and lymph node involvement was observed in three and seven patients, respectively. Most lesions showed tissue eosinophilia (100%) and florid follicular hyperplasia (78.26%). Three cases had histological KD-IgG4-RD overlap and three had comorbid IgG4-RD were recognized. Thirteen patients underwent surgical resection, one received adjuvant therapy, and two received prednisolone monotherapy. KD should be considered in patients with subcutaneous masses, eosinophilia, and elevated IgE levels. Biopsy remains the gold standard of diagnosis. Increased recruitment of IgG4+ plasma cells is a common feature. Consideration of IgG4-RD in all KD patients may be prudent.

Features of the diagnosis of primary hyperparathyroidism in children

Background. Primary hyperparathyroidism (PHPT) is a rare pathology in pediatric and adolescent patients. Collection, analysis and generalization of the literature data and experience of the leading clinics allow to develop unified, statistically substantiated approaches to diagnostics and surgical treatment of this group of patients.Material and methods. The article presents a retrospective analysis of 17 cases of PHPT in children and adolescents aged from 6 to 18 years operated on in the department of Surgery of St.-Petersburg State Pediatric Medical University in the period from 1973 till 2021. Among those operated there were 10 girls and 7 boys, the M:F ratio was 1:1.4. The mean age of the patients was 12,9±0,71 years.Results and discussion. The main criteria of the disease diagnosis were elevated blood calcium and parathormone levels, excessive urinary calcium secretion. Manifest forms of the disease were diagnosed in 10 (58,8%) of 17 children. In 3 (17,6%) cases the parathyroid neoplasms were accompanied neither by clinical, nor laboratory manifestations of the disease and were regarded as incidentalomas. Another 4 (23.5%) patients had only laboratory changes (hypercalcemia and hyperparathyrinaemia) that manifested themselves preoperatively. These observations were referred to the asymptomatic form of PHPT.In 7 (41.2%) cases parathyroid adenomas were found and removed during surgeries for various thyroid diseases (thyroid cancer in 5 cases, diffuse toxic goiter in 1 case). An incidental finding of parathyroid adenoma was during prophylactic thyroidectomy for Sipple syndrome.A radioisotope method proved to be the most informative way to localize parathyroid tumors.Conclusion. The diagnosis of the disease in manifest sporadic cases does not differ from that in adults. Genetic study is indicated in the presence of a family history of multiple involvement of the parathyroid glands. Surgical treatment with removal of parathyroid tumor is the main method, which allows to achieve complete recovery.

Hyperfunctioning parathyroid gland and skeletal involvement on [18F]fluorocholine PET/CT: one look with two views

Hyperparathyroidism is an endocrine disorder that may be associated with other metabolic diseases. Non-invasive imaging techniques including [99mTc]Tc-sestamibi single-photon emission computed tomography (SPECT) and [18F]fluorocholine positron emission tomography (PET)/computed tomography (CT) play a key role on management of patients with hyperparathyroidism. We report for the first time a case of a patient with evidence of both hyperfunctioning parathyroid tissue and multiple lytic bone lesions on [18F]fluorocholine PET/CT imaging. The present case report highlights the potential role of whole-body [18F]fluorocholine PET/CT for the identification of both parathyroid adenoma and multiple bone lesions in a single diagnostic setting.

PECULIARITIES OF CYSTIC FIBROSIS IN NEWBORN ON THE EXAMPLE OF A CLINICAL CASE

Cystic fibrosis is a severe genetic disease, which is inherited in an autosomal recessive manner and is characterized by a polyorganic clinical presentation with predominantly exocrine, respiratory and digestive gland involvement. Every year more than 45 000 children are born worldwide with this genetic disease. According to official statistics, 674 children with cystic fibrosis are registered in Ukraine, 25 of them in the Vinnytsia region. The disease is caused by a mutation in the gene encoding the cystic fibrosis transmembrane conductance regulator (CFTR). The F508del mutation is most common in the paediatric population (19-34%). The most common clinical manifestations of cystic fibrosis in newborns are disorders of the gastrointestinal tract, pancreas, hepatobiliary system and respiratory system. In 20% of newborns, one of the earliest manifestations of cystic fibrosis is meconial ileus, which may pose a risk of complications such as meconium peritonitis, sputum, gangrene or intestinal necrosis. Early diagnosis with neonatal screening, improved care for the neonatal manifestations of meconial ileus and adequate medical follow-up by a multidisciplinary team of specialists, and improved lifelong replacement fermentation therapy have a significant impact on the life expectancy of patients with cystic fibrosis.This article presents a clinical case of early diagnosis of cystic fibrosis in a newborn. When meconial ileus is present and cysticfibrosis is suspected in a newborn, early genetic diagnosis is mandatory, in addition to timely and correctly performed neonatal screening. Comprehensive and timely treatment leads to improvement in cystic fibrosis patients and slows down the development of pathological changes in the bronchopulmonary and hepatobiliary systems.Conclusions. Prevention of a severe course of cystic fibrosis is in early detection of the disease during neonatal screening, indiagnosis at the first signs of the disease and early start of pathogenetic treatment, which improves the quality of life of patients.

“CERVICAL LYMPHADENOPATHY AND ITS ETIOLOGY IN CHILDREN: A STUDY IN A TERTIARY HEALTH CARE INSTITUTE.”

Background: Lymphadenopathy occurs in a number of cases presenting in ENT out-patient department. Most cases are found to have enlarged cervical lymph glands. Lymphadenopathy is a condition affecting the lymph nodes of the body as a result of infection, benign or malignant conditions. As a result, lymph nodes become enlarged and abnormal in size, shape, consistency and they may show xity, abnormal mobility or symptoms that differ from normal. This is due to uid accumulation and action of lymphocytes. The most common form is lymphadenitis when it is due to inammation. Inammation may spread from surrounding soft tissue via lymphatic vessels or direct involvement of the lymph glands. A study was conducted to have an idea about the most common cause of cervical lymphadenopathy in children as most common region of lymph node in children is cervical groups of lymph nodes. A hospital-based prospective study was Methods and Materials: conducted in the department of otorhinolaryngology and head and neck surgery, from a time period of October 2021 to March 2022 and results analysed from the collected data. Results: Reactive lymphadenopathy due to viral or bacterial infections is the leading cause followed by systemic and malignant diseases. Discussion: Inammatory response is the leading cause of cervical lymphadenopathy in children.

Long-Term Follow-Up in IgG4-Related Ophthalmic Disease: Serum IgG4 Levels and Their Clinical Relevance.

(1) Background: To analyze the association between long-term changes in serum IgG4 levels and the clinical course of patients with IgG4-related ophthalmic disease (IgG4-ROD). (2) Methods: Retrospective analysis of 25 patients with IgG4-ROD. (3) Results: Mean age at diagnosis was 60.68 years. Fifty-six percent of patients had bilateral ocular involvement and 32% had systemic associations. The ocular structures involved were the lacrimal gland (76%), orbital soft tissue (36%), extraocular muscle (20%) and infraorbital nerve (20%). According to last follow-up, 9 (36%) patients had normalized IgG4 levels, and 16 (64%) patients had elevated IgG4 levels. Patients with normalized IgG4 levels had better response to initial steroid treatment and attained a significantly lower IgG4 level after treatment (p = 0.002). The highest IgG4 levels were at baseline and disease recurrence, and lowest after initial treatment. At final follow-up, IgG4 levels differed in patients with remission (mean 326.25 mg/dL) and stable disease (mean 699.55 mg/dL). Subgroup analysis was performed in patients with remission, categorized according to whether IgG4 levels were normalized (9 patients) or elevated (10 patients) on last follow up. The elevated group had a higher percentage of bilateral disease, lacrimal gland involvement and recurrence. (4) Conclusions: IgG4-ROD patients with a greater response to initial steroid therapy were more inclined to have normalized IgG4 levels in the long term. Some patients remained in remission despite persistently elevated IgG4 levels, and had regular follow-up without treatment.

Lacrimal Gland involvement in systemic Sarcoidosis

Lacrimal gland lesions generallypresent as palpable masses in thesuperolateral aspects of the orbits. Approximately 50% of lacrimalgland masses are inflammatory lesions, 25% are lymphoid lesions or lymphoma, and the other 25% are salivary gland type tumors. Although there are overlaps and exceptions, features such as laterality, portion of gland involvement, presence or absence of bony findings, enhancement pattern, andclinical presentation are valuable in differentiating among lacrimal gland lesions. The 3D ultrasound has been used to define lacrimal gland shape, size, density, structural features the pattern of blood supply, as well as the anatomic and topographic position in the orbit. The study was conducted in the B- and 3D-modes of ultrasonography with color and energy Doppler mapping on both sides.

Proteomic characteristics of saliva in patients with different subgroups of IgG4-RD.

Immunoglobulin G4-related disease (IgG4-RD) is a newly defined disease entity, with great heterogeneity among IgG4-RD subgroups with different organ involvement patterns. Identification of the proteomic characteristics of IgG4-RD subgroups will be critical for the understanding of the pathogenic mechanisms of IgG4-RD. In this study, we performed proteomic analysis using Tandem Mass Tags (TMT) technology with "high field" mass analyzer with improved resolution and sequencing speed to investigate the proteomic profile of saliva and plasma samples from ten untreated IgG4-RD patients and five healthy controls (HCs). Differentially expressed proteins (DEPs) were identified by "t test" function in R package. Functional enrichment analysis was used to investigate pathways enriched in IgG4-RD samples. Most salivary DEPs identified in IgG4-RD patients compared with HCs were mainly enriched in neutrophil mediated GO bioprocess. Within the comparisons between four IgG4-RD subgroups, more DEPs were identified in the comparison of Mikulicz group and Head and neck group. Among four subgroups of IgG4-RD, Head and neck group showed the most distinctive proteomic expression pattern when compared with HCs. Moreover, "Neutrophil mediated process" related GO bioprocess was commonly identified between comparisons of Mikulicz group and Head and neck group, Head and neck group and Retroperitoneal aorta group, Head and neck group and HCs, IgG4-RD patients with saliva gland involvement and those without saliva gland involvement. Key DEPs that involved in this GO bioprocess were identified. Besides, we performed proteomic analysis for plasma samples between ten IgG4-RD and five HCs and there were several DEPs identified overlapped in saliva and plasma. We identified multiple processes/factors and several signaling pathways in saliva that may be involved in the IgG4-RD pathogenesis.

Adenoid Basal Carcinoma of the Cervix with Squamous Differentiation: A Case Report and Literature Review

Adenoid basal carcinoma (ABC) of the cervix is a rare and low-incidence low-grade cervical cancer. In our practice, we encountered a case of cervical ABC with squamous differentiation and high-grade squamous intraepithelial lesion (HSIL) with gland involvement in the peripheral cervix. Reviewing relevant literature and analyzing its clinical manifestations, pathological morphology, and immunohistochemical characteristics would help deepen the understanding of this malignant tumor, so as to make a comprehensive diagnosis with differential diagnosis and prevent misdiagnosis.

Intraepithelial involvement of non-neoplastic glands in papillary preinvasive neoplasms of the biliary tract: a potential diagnostic pitfall

Intracholecystic papillary neoplasms of the gallbladder (ICPN) and intraductal papillary neoplasms of the bile duct (IPNB) show intramural neoplastic growths in addition to intraluminal papillary or polypoid neoplastic growth. Such intramural growths include intraepithelial involvement of non-neoplastic glands by preinvasive neoplastic epithelia (glandular involvement) as well as stromal invasive carcinoma. A total of 29 ICPN cases and 84 IPNB cases were pathologically examined for their glandular involvement. Glandular involvement was characterized by intramural neoplastic glands (1) showing cytological and phenotypical similarities to intraluminal preinvasive papillary neoplasms and (2) showing reminiscent configurations of non-neoplastic glands, such as (i) a mixture of preinvasive neoplastic epithelia and non-neoplastic epithelia within the same glands, (ii) neoplastic glands close to or within clustered non-neoplastic glands, or (iii) continuous growth of intraluminal preinvasive neoplastic glands into the walls. Such glandular involvement was found in 16 of 29 ICPN and 48 of 84 IPNB, and 15 of the former and 28 of the latter were not associated with invasive carcinoma. Non-invasive ICPN and IPNB with glandular involvement showed a favorable postoperative overall survival (OS). Glandular involvement by preinvasive neoplastic epithelia was frequently found in ICPN and IPNB. Such lesions may be diagnostic pitfalls in ICPN and IPNB referring to invasion. Glandular involvement without invasive carcinoma was not associated with an unfavorable postoperative OS in ICPN and IPNB. Recognition of glandular involvement may thus prevent overestimation of invasive carcinoma in ICPN and IPNB.

The effect of local photodynamic therapy with 5-aminolevulinic acid in treating different grades of cervical intraepithelial neoplasia

BackgroundCervical intraepithelial neoplasia (CIN) is a precursor lesion of cervical cancer. Traditional treatments for CIN might have negative effects on cervical anatomical structure and physiological function. Topical 5-aminolevulinic acid photodynamic therapy (5-ALA PDT) is a novel, non-invasive targeted therapy for intraepithelial lesions. This study aims to evaluate and compare the efficacy and safety of 5-ALA PDT for different grades of CIN. MethodsA retrospective study of 183 patients aged 19–50 with histologically confirmed CIN and receiving ALA-PDT was conducted. ALA-PDT was performed with 20% ALA thermosensitive gel and irradiation at a wavelength of 635 nm and density of 80–100 J/cm2. ALA-PDT was conducted every 7–10 days for 4–6 times. Patients were followed up three, six, nine, and twelve months after treatment. The effect was evaluated through HPV genotyping, ThinPrep cytology test (TCT), and colposcopy-directed biopsy. ResultsThe HPV clearance rate was 71.0% (130/183) at the six-month follow-up and 84.5% (147/174) at the 12-month follow-up. The complete lesion remission (CR) rate was 90.2% (165/183). No statistically significant differences concerning the CR rate (P>0.05) or HPV clearance rates (P>0.05) were observed in CIN I, CIN II, and CIN III. In women with CIN III, gland involvement was revealed to be associated with a significantly lower HPV clearance rate (63.16% vs. 92.60%, P= 0.036) at the 12-month follow-up. Our study showed that the atypical vessels seemed to be a risk factor for HPV clearance rate in the CIN II group at six-month follow-up, although the difference was not statistically significant (P= 0.089). During the follow-up, 13 cases had persistent lesions (7.1%), four cases recurred (2.3%), and none of the patients progressed. The study also showed that the efficacy of PDT in the treatment of patients with CIN III involving glands was comparable to that of CKC (P>0.05). ConclusionsALA-PDT is an effective andsafe treatment for CIN, and responseis unaffected by the grade of lesions. However, for patients with atypical vessels and glandular involvement, the effect of PDT seems to be poorer.

Multiple Cutaneous Features Associated with Primary Sjogren’s Syndrome

Sjogren’s syndrome is a systemic autoimmune disorder that mainly affects exocrine glands, although the clinical manifestations include both exocrine gland involvement and extraglandular disease features. In addition to dryness, musculoskeletal pain and fatigue are the hallmarks of this disease and constitute the classic symptom triad presented by the vast majority of patients. The authors present the case of a 59-year-old woman with achalasia, sicca syndrome, subcutaneous nodules affecting the knees, purpuric plaques on the palms and vitiligo. Serological tests were consistent with Sjögren′s syndrome, therefore the diagnosis of primary Sjögren’s syndrome with extensive cutaneous manifestations as extraglandular organ involvement was made.

PSAT202 A Rare Case of Four-Gland Parathyroid Adenomas

Abstract Background Amongst patients diagnosed with primary hyperparathyroidism, 80% have been identified to have a single parathyroid gland involvement, with 10% involving two glands or more. Case A 71-year-old female with medical history of hypothyroidism, multinodular goiter, CKD stage 3a, recurrent nephrolithiasis and hyperparathyroidism presented for evaluation of hypercalcemia. She was diagnosed with hyperparathyroidism 9 years prior to presentation however she preferred medical management with cinacalcet. For the past 5 months, she had been experiencing worsening fatigue, constipation, body aches and appetite loss. Further blood analysis revealed PTH 1176 pg/mL (14-64 pg/mL), TSH 2.69 uU/mL (0.35-4.94 uU/mL), free T4 1.12 ng/dL (0.60-1.89 ng/dL), vitamin D, 25-Hydroxy 24 ng/mL (30-80 ng/mL), serum calcium 12.6 mg/dl (8.9-10.4 mg/dL), ionized calcium 1.65 mmol/L (1.14-1.33 mmol/L), phosphorus 1.9 mg/dl (2.4-4.8 mg/dl), magnesium 1.0 mg/dl (1.8-3.0 mg/dL). Thyroid ultrasound revealed a left isthmus heterogenous solid nodule measuring up to 2.0 cm and a separate nodule superior to the isthmus measuring up to 0.6 cm. Subsequent NM parathyroid scan was limited due to diffuse uptake of enlarged nodular thyroid. Although limited, imaging did reveal a small focus of uptake lateral to the right lower pole concerning for parathyroid adenoma. A CT 4D neck with and without contrast demonstrated a suspicious parathyroid adenoma posterior to the inferior aspect of the left thyroid measuring up to 1.7 cm, an additional 0.8 cm questionable node, and a 4 cm multicystic mass inferior to the right thyroid lobe concerning for exophytic thyroid nodule or multi cystic parathyroid adenoma. Due to persistence of symptoms and significantly elevated PTH levels concerning for carcinoma, decision was made to undergo a four-gland parathyroid exploration and a right hemithyroidectomy. A right superior parathyroid adenoma was identified; after resection, intraoperative PTH levels failed to improve which prompted further exploration. Left superior and inferior parathyroid adenomas were identified and resected. An ectopic nodule located in the right superior mediastinum with surrounding normal parathyroid tissue was identified and dissected, leaving parathyroid tissue behind. Post operatively, PTH levels appropriately decreased to 54 pg/ml. Pathology of identified parathyroid glands had hypercellular parathyroid tissue with fibrous capsule, consistent with adenomas. Interestingly, the pathology of the presumed ectopic nodule was consistent with atypical parathyroid adenoma. Immunostatins with parafibromin demonstrated nuclear immunoreactivity in the lesional cells, consistent with a benign neoplasm. Patient developed symptomatic hypocalcemia following surgery. She was gradually weaned off calcitriol and is currently normocalcemic. Conclusion It is extremely rare to have adenoma of 4 parathyroid glands simultaneously in the absence of genetic conditions associated with hyperparathyroidism. Age of onset of hyperparathyroidism and absence of other endocrine abnormalities clinically was reassuring, however; further biochemical and genetic testing will be considered in this patient. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

PSAT278 A Curious Case of Thyroglossal Duct Cancer

Abstract Introduction Thyroglossal duct (TGD) cyst is a common congenital anomaly. Thyroid cancer rarely occurs in TGD cysts, affecting less than 1% of cysts. Here we present a case of TGD cancer presenting as a central neck mass. Case Presentation 60 y.o female presented to her primary care physician with a neck mass without any compressive symptoms. She had no history of head/neck radiation exposure and no family history of thyroid cancer. She was noted to have a non-tender central neck mass with palpable cervical lymphadenopathy. Neck ultrasound revealed a 5.7 cm multilobulated cystic and solid mass anterior to the thyroid gland. CT scan of the neck revealed a lobulated mixed solid cystic mass at the level of the thyroid cartilage projecting anteriorly, with an abnormal right submandibular lymph node measuring 2.2×1.0 cm. She was evaluated by ENT and underwent resection of the neck mass and the hyoid bone (Sistrunk's procedure). Level 1b lymph node was also removed. Operative findings were concerning for a malignant process. This was confirmed on pathology with findings of TGD papillary thyroid cancer (PTC) and level 1b lymph node demonstrating metastatic PTC with cystic features. She was then referred to endocrine. Thyroid ultrasound revealed multiple bilateral intermediate suspicion nodules. Left inferior thyroid nodule was noted to have high suspicion sonographic features with abnormal right sided level 2 and 3 lymph nodes. She was referred for total thyroidectomy, pathology revealed multifocal PTC with lymph node involvement with extra nodal extension and metastasis to the left sternohyiod muscle, pT2, pN1b, AJCC stage 2. 6-week post-surgical labs revealed TSH of 1.69 mIU/L (ref range 0.40-4.50), thyroglobulin level of 0.8 ng/ml (ref range athyrotic <0.1), with undetectable thyroglobulin antibodies <1 IU/ml (ref range < or = 1 IU/mL). She underwent RAI with 104.6 mci. I-131 Metastatic survey revealed increased uptake in the thyroid bed, otherwise no increased activity elsewhere. Stimulated TSH and Thyroglobulin levels were done but not collected appropriately and therefore not mentioned here. Repeat labs 12 weeks after RAI revealed a TSH of 2.86 mIU/L, Thyroglobulin level of 0.2 ng/ml, and undetectable Thyroglobulin antibody. Conclusion There are no standardized guidelines for management of TGD cancer given the rarity of this condition. FNAB (Fine need aspiration biopsy) may be associated with high false negatives due to inadequate sampling, given the cystic nature of these lesions. In majority of these cases definitive diagnosis of cancer is made after excision of the TGD cyst. Some experts favor a conservative approach via Sistrunk's procedure and pursuing total thyroidectomy if there is high suspicion of thyroid gland involvement as well. Whereas other authors have suggested a more aggressive approach with total thyroidectomy even without evidence of concomitant involvement of the thyroid gland. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

PSAT342 Hypothyroidism After Use Of Immune Checkpoint Inhibitor Therapy In Patient With Graves’ Disease

Abstract Background Immune checkpoint inhibitors (ICIs) are commonly used for many cancers. Thyroid gland involvement is the commonest endocrine toxicity seen with this class of drugs. Most common thyroid dysfunction is thyroiditis followed by hypothyroidism with some cases of recovery. There are rare case reports of Graves’ disease induced by ICIs. However, we report a patient with pre-existing Graves’ disease who developed hypothyroidism after immunotherapy. Clinical Case 66-year-old man with metastatic squamous cell carcinoma of lung was referred to endocrine clinic for suppressed TSH. He was diagnosed with metastatic poorly differentiated lung cancer in September 2018. He received cheamotherapy with carboplatin and paclitaxel from October 2018 to January 2019. He was switched to immune checkpoint inhibitors with programmed cell death protein-1 (PD-1) inhibitor pembrolizumab every 3 weeks from February 2019. ICI therapy was stopped in until July 2019 due to immune mediated hepatitis. He was started on high dose steroids. Lung cancer findings remained stable even after stopping ICIs. Prior to starting ICIs his thyroid functions were consistent with hyperthyroidism. He continued to have suppressed TSH until his 6th cycle of pembrolizumab. TSH was found be significantly elevated prior to his 7th infusion. He was referred to endocrinology for further management. Clinically, patient was asymptomatic except for fatigue. Family history was positive for mother with Graves’ disease. Laboratory results on presentation showed significantly elevated TSH, low thyroxine (T4) and triiodothyronine (T3) consistent with diagnosis of primary hypothyroidism. Thyroid stimulating immunoglobulin (TSI) was positive suggesting autoimmune thyroid disease. Thyroid scan showed decreased 24-hour uptake (0.8%). A diagnosis of immune checkpoint inhibitor (ICI) related hypothyroidism was made. He was started on hormonal replacement with levothyroxine 50 mcg daily. Review of previous thyroid blood tests showed suppressed TSH since October 2016, but he was never treated. Thyroid peroxidase (TPO) antibodies were positive in the past. Thyroid sonogram in December 2016 showed a heterogeneous thyroid with no nodules. A radioactive scan in 1/2017 showed normal 24-hour uptake (12%) with no hot or cold nodules. (Fig 1). Since starting levothyroxine, the dose has been adjusted few times and patient has been doing well. Conclusion This is a unique case of patient with Graves’ disease who was cured because of immune related adverse event from use of immune-checkpoint inhibitor therapy. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

Molecular Mechanisms Linking Inflammation to Autoimmunity in Sjögren's Syndrome: Identification of New Targets.

Sjögren's syndrome (SS) is a systemic autoimmune rheumatic disorder characterized by the lymphocytic infiltration of exocrine glands and the production of autoantibodies to self-antigens. The involvement of the exocrine glands drives the pathognomonic manifestations of dry eyes (keratoconjunctivitis sicca) and dry mouth (xerostomia) that define sicca syndrome. To date, the molecular mechanisms mediating pathological salivary gland dysfunction in SS remain to be elucidated, despite extensive studies investigating the underlying cause of this disease, hampering the development of novel therapeutic strategies. Many researchers have identified a multifactorial pathogenesis of SS, including environmental, genetic, neuroendocrine, and immune factors. In this review, we explore the latest developments in understanding the molecular mechanisms involved in the pathogenesis of SS, which have attracted increasing interest in recent years.