Case Of Adenoma Research Articles

Invasion of the Cavernous Sinus Medial Wall by Pituitary Adenomas: Intraoperative Evaluation and the Clinical Efficacy of the Cavernous Sinus Medial Wall Resection in the Treatment of Somatotroph Adenomas.

Pituitary adenomas are common benign intracranial tumors. This study aimed to explore the relevant factors influencing the cavernous sinus medial wall invasion by Pituitary adenomas based on intraoperative evaluation. Meanwhile, the authors introduced a safe technique for the resection of the medial wall of the cavernous sinus and conducted a preliminary evaluation of the efficacy of this technique. The clinical data of 108 patients with pituitary adenomas who underwent surgery at our institution were collected. Binary logistic regression analysis was utilized to identify independent predictors of medial wall invasion. Furthermore, the authors performed medial wall resection on 12 cases of somatotroph adenomas with medial wall invasion to initially assess the surgical efficacy of this technique for somatotroph adenomas. Multivariate Logistic regression analysis showed that tumor Knosp grade and somatotroph adenomas were independent predictors of medial wall invasion (P<0.05). For somatotroph adenomas, medial wall resection resulted in a high biochemical remission rate (83%), and all patients were free of recurrence at the last follow-up. Higher knosp grade and somatotroph adenomas will increase the risk of medial wall invasion of the cavernous sinus. Relying solely on the Knosp grade system to evaluate tumor invasion of the medial wall without differentiating between tumor subtypes is inaccurate. Invasion of the medial wall may be a cause of low biochemical remission rates and recurrence after surgery for somatotroph adenomas. The technique of medial wall resection is safe and effective, and holds significant importance in the treatment of somatotroph adenomas.

Esophageal submucosal gland duct adenoma: An unrecognised esophageal counterpart of minor salivary gland tumours with frequent BRAF V600E mutations.

Esophageal submucosal gland duct adenoma is an extremely rare benign tumour, with only a few reported cases. We conducted the largest single-centre clinical study of esophageal submucosal gland duct adenoma, examining its molecular mechanisms and clinicopathological features. Between 2018 and 2023, seven cases of esophageal submucosal gland duct adenoma were identified at a tertiary medical centre; two were female and five were male, aged between 51 and 75 years (mean = 63.8 years). Comprehensive evaluations of clinicopathological, immunohistochemical and molecular characteristics were conducted. Histologically, tumours showed papillotubular and cystic patterns lined with double layers of cells arranged in ducts, papillary folds and microcysts. The inner luminal tall columnar cells had eosinophilic cytoplasm and did not show mucin production and the basal cells showed myoepithelial differentiation. Immunohistochemically, inner luminal layer cells were positive for CK7, CK19 and CK5/6 and outer basal layer cells were positive for SMA and P40. Both layers were negative for CK20, CDX2, MUC5AC, MUC6, MUC2, GCDFP15 and Alcian blue-periodic acid-Schiff (AB-PAS). Genomic analyses revealed the presence of BRAF V600E mutations in five of seven tumours (71.43%). This study delineates a distinct subtype of benign adenoma arising from the esophageal submucosal gland duct, characterised by multiple lobulated cystic proliferation of benign epithelial layers within the submucosa. BRAF V600E mutations were present, similar to in sialadenoma papilliferum. We determined the genetic mutation present in esophageal submucosal gland duct adenoma, providing further evidence that it is an esophageal counterpart of minor salivary gland tumours.

Tubular adenoma at the hepatico-jejunal anastomosis in familial adenomatous polyposis (FAP) following pancreaticoduodenectomy: challenges in adenoma surveillance and management.

Upper gastrointestinal tumors, including ampullary adenomas, occur frequently in patients with familial adenomatous polyposis (FAP). Guidelines recommend upper gastrointestinal endoscopy in FAP for surveillance of gastric and duodenal adenomas. However, adenomas can rarely arise from biliary epithelium in patients with FAP. Here, we describe a case of tubular adenoma at the hepatico-jejunal anastomosis with intraductal extension in a patient with FAP and previous pancreaticoduodenectomy. This report illustrates a unique case and emphasizes the need for data on postoperative surveillance in patients with FAP, particularly following pancreaticoduodenectomy.

Endoscopic Endonasal Approach for Calcified Sellar/Parasellar Region Pathologies: Report of 11 Pituitary Adenoma Cases

Calcification in pituitary adenomas is a rare occurence and its differential diagnosis typically includes other sellar masses.Common calcifications in pituitary adenomas are classified into two morphological forms: capsular(eggshell-like)and multiple small nodular calcifications located within the adenoma.Also,there is a pituitary stone term.This study aims to present the results of calcified pituitary adenoma case series who underwent endoscopic endonasal approach(EEA)and the clinical,histopathological characteristics and surgical outcomes of these cases. This study conducted a retrospective cohort analysis of patients with calcified pituitary adenoma operated on by EEA between August 1997 and February 2024.The inclusion criteria were as follows:proven radiological calcification on pre-operative neuroimaging,intraoperative findings of calcification and definitive histopathological diagnosis of calcification.Among these patients,11 cases were included. The mean follow-up duration was 51.45±37.24 (6-118)months.Based on the pre-operative paranasal sinus computed tomography(CT)scans of patients,nine patients(81.8%)had intratumoral calcification,one(9.1%)had capsular(eggshell like)calcification and one(9.1%)had both intratumoral and capsular calcification.Moreover,stone-like calcifications were observed in four patients(36.4%),soft type calcifications in three(27.3%),hard-type calcifications in three(27.3%)and soft and hard-type calcifications in one(9%).Gross total resection(GTR)was achieved in nine patients(81.8%).Pathologic subtypes included non-functioning(n = 4),prolactin secreting(n = 3),growth hormone secreting adenoma(n = 2)and pituitary apoplexy(n = 2).Ten patients had psammomatous type calcifications and one had extensive ossification and osteoid metaplasia. Pre-operative radiological evaluation,intraoperative classification of calcification,and post-operative histopathological assessments are crucial in the treatment of calcified adenomas.Bases on these findings,the EEA,with its advantages,is an approach that can be effectively used in the management of these calcified adenomas.

TMOD-06. ESTABLISHMENT OF PITUITARY NEUROENDOCRINE TUMOR (PITNET) ORGANOID MODELS

Abstract Pituitary neuroendocrine tumors (PitNETs) are tumors that originate from the anterior pituitary gland. While they are mostly benign, they can cause various clinical symptoms due to hormonal secretion abnormalities. PitNET can be broadly categorized into three lineages, each of which has its differentiation controlled by specific transcription factors. Treatment options for PitNET include surgical resection and pharmacotherapy, but available drugs are limited, and their efficacy markers remain unclear. To develop novel drugs for PitNET and identify their efficacy markers, it is necessary to develop new research models. In recent years, organoid models have garnered attention as novel research models for various cancer types. In this study, we established novel organoid models derived from PitNET patients and analyzed their histological and endocrinological characteristics. Surgical specimens from 4 cases of somatotroph adenoma, 1 case of double adenoma, 1 case of gonadotroph adenoma, and 1 case of corticotroph adenoma were minced in dissection medium and embedded in Matrigel basement membrane matrix respectively. The organoids were cultured under 5% CO2 at 37°C using media supplemented with various growth factors. After 28 days of culture, formalin-fixed paraffin-embedded sections were prepared to examine histological homology with patient tumors. Hematoxylin and eosin staining results showed that all organoids maintained cell morphology and tissue structure similar to the original tumors. Immunohistochemical staining for markers specific to each PitNET revealed similar staining patterns between the organoids and the original tumors. Furthermore, analysis of hormone levels in the culture medium showed that hormone production capabilities, such as growth hormone and follicle stimulating hormone, were maintained even after long-term culture. In this study, we successfully established PitNET organoid models that retain histological and endocrinological characteristics. These models are expected to serve as useful research tools for elucidating the molecular mechanisms of PitNET and developing novel therapies targeting critical molecular abnormalities.

Diagnostic Performance of Faecal Immunochemical Testing (FIT) in Patients with Lynch Syndrome Scheduled for Colonoscopic Surveillance.

Lynch syndrome (LS) carries a substantial lifetime risk of colorectal cancer which is currently mitigated by biennial colonoscopy surveillance. Paramount to the surveillance programme is the removal of adenomas before malignant transformation but there is an associated service burden and morbidity of repeated endoscopy. We investigated if faecal immunochemical testing (FIT) for faecal haemoglobin has the diagnostic performance to replace colonoscopy. In this retrospective cohort study, patients due to undergo planned surveillance for LS between November 2020 and April 2022 were sent two FIT kits prior to colonoscopy. Test diagnostic performance of colorectal cancer (CRC), advanced and non-advanced adenoma detection was calculated for single and double FIT strategies. A faecal-Hb of 10 µg Hb/g was considered positive. In total, 78 patients, with 45 (57.7%) female, median age 52 years (IQR 41-63), completed at least one FIT and colonoscopy. The median time from FIT to colonoscopy was 47 days. A single FIT was positive in 7/30 cases of adenoma (2/3 advanced, 5/27 non-advanced). A total of 64 (82.1% of FIT1T returners) completed a second FIT. Using the greatest of the two FITs (FIT2TMAX) 8/26 (2/3 advanced, 4/23 non-advanced), patients with adenomas were identified. There were no cases of CRC. The sensitivity for adenoma detection was 23.3% and 23.1%, respectively. In patients with LS awaiting colonoscopy, FIT has a low sensitivity for detecting adenomas and advanced adenomas. This is not improved by the addition of a second FIT test.

A rare case of a pleomorphic adenoma of the submandibular gland

We report the case of a 27-year-old female with a painless slowly growing mass that had developed over 2 years in the right submandibular region. Physical examination revealed a firm painless submandibular mass. Fine-needle aspiration and computed tomography (CT) scan concluded a pleomorphic adenoma of the submandibular gland. The patient underwent a right total submandibular sialoadenectomy. Histopathological examination confirmed the diagnosis of pleomorphic adenoma arising from the submandibular gland. Transient palsy of the marginal mandibular division of the facial nerve was noted post-operatively, which recovered with supportive management. Pleomorphic adenoma of the submandibular gland represents a diagnostic and therapeutic dilemma in young adults. Preoperative evaluation, including MRI/CT scan and fine-needle aspiration, is recommended in all cases. Total submandibular sialoadenectomy is the standard recommended treatment to minimise recurrences and avoid malignant transformation.

NanoString nCounter-Based Assay for Detection of Fusion-Associated Salivary Gland Tumors.

Salivary gland tumors include numerous subtypes that vary from benign to highly aggressive, with many showing overlapping histopathological features that can make diagnosis challenging. Most subtypes express driver fusion genes that are tumor specific, and detection of such fusions is useful for differentiating amongst specific diagnoses, determining appropriate tumor grading, and guiding effective treatment. Currently, fusions can be detected by FISH, RT-PCR or through next-generation sequencing approaches, all of which are highly effective methodologies but can be costly or time consuming. We developed a rapid NanoString nCounter platform-based assay to detect salivary gland tumor fusions using a combination of fusion junction-specific probes and an approach through differential exon expression analysis. The assay includes 68 junction-specific probes and analysis of exon expression across 9 fusion-associated genes in a single multiplex assay. Out of 55 retrospective and 171 prospective cases assayed, we accurately detected the majority of cases of pleomorphic adenoma, adenoid cystic carcinoma, cribriform adenocarcinoma, clear cell carcinoma, secretory carcinoma and NUT-rearranged carcinoma, including cases of these tumor types arising in non-salivary gland sites, with the major drawback being an inability to detect MAML2-containing mucoepidermoid samples. With mucoepidermoid carcinoma excluded, the assay shows an overall sensitivity of 96.1% and specificity of 100%. We show that the majority of salivary gland tumor fusions can be effectively detected with a single rapid NanoString based assay, which can serve as a useful adjunctive tool for routine diagnostic head and neck pathology. The assay is low cost with a rapid turnaround time (30h total assay time per sample batch, with minimal technician input required) compared to alternate detection methods.

Management of a labial pleomorphic adenoma in the odontostomatology department of the Idrissa Pouye general hospital in Dakar

Pleomorphic adenoma of the salivary glands (formerly called mixed tumor) represents 60% of oral salivary tumors. The accessory salivary glands have their preferential location at the palate, the lips of the cheek and the oropharynx. At the level of the lips, this tumor most often affects the right upper lip.We report a case of pleomorphic adenoma of the right upper lip evolving for more than 4 years in a 70-year-old melanoderm patient who was referred to the odontostomatology department of the Idrissa Pouye General Hospital (HOGIP).The swelling was large, slightly exceeding the midline of the upper lip. The swelling measured approximately 20 cm from its long axis. It was firm, painless and did not bleed on contact and covered with a healthy-looking mucosa. The management was surgical under local anesthesia and the postoperative course was simple.Histological analysis specified that it was a well-limited nodular lesion, corresponding to a mixed epithelial and mesenchymal proliferation with a thin capsule in favor of a pleomorphic adenoma. No recurrence was observed 4 months after the operation.

Mixed Gangliocytoma-Pituitary Adenoma: Incidental Discovery During Eye Injury Evaluation: A Case Study

Abstract Introduction/Objective Mixed gangliocytoma-adenoma is exceedingly rare, representing less than 0.5% of sellar tumors. We present a unique case of mixed gangliocytoma-pituitary adenoma incidentally diagnosed during the evaluation of traumatic eye injury. We aim to clarify the diagnostic process using real clinical data, stressing how such cases might be missed without careful histopathological examination Methods/Case Report A 32-year-old male presented for elective transsphenoidal resection (TSR) due to a suspected growth hormone (GH) secreting pituitary tumor. Surprisingly, the adenoma was discovered incidentally during an eye injury evaluation. Referred from an ophthalmology clinic, initial concerns were a possible blood clot or foreign body in the left eye, following a forceful encounter with a tree branch Results (if a Case Study enter NA) Imaging incidentally identified a 2 cm pituitary mass. Further evaluation showed elevated levels of insulin-like growth factor 1 (IGF1) at 1162 ng/ml and GH at 10.2 ng/ml. Remarkably, the patient had not observed signs of acromegaly, until photographic comparison between past and present appearances was made. TSR of the pituitary mass was done. Microscopic examination revealed monomorphic cells with round nuclei with the typical speckled chromatin pattern of neuroendocrine cells with cytoplasm ranging from chromophobic to eosinophilic, consistent with pituitary adenoma. An additional unexpected finding was the presence of large gangliocytic cells (large neurons), dispersed throughout the tumor as single or clustered cells. In some regions, distinct patches of fine neuropil stroma were associated with the clustered ganglion cells. IHC staining with synaptophysin and neurofilament highlighted the gangliocytic component. CAM 5.2 IHC stain showed perinuclear dot-like expression in the adenoma cells, GH IHC showed patchy expression, and prolactin IHC was expressed in scattered cells. Ki67 index was ~ 2%. There was no evidence of necrosis nor mitosis. The coexistence of sellar gangliocytoma with pituitary adenoma, as observed in this case, represents a rare entity known as mixed gangliocytoma-pituitary adenoma Conclusion The adenomatous component of mixed gangliocytoma-pituitary adenoma often secrete GH, leading to its classification as pituitary somatotroph adenoma. This poses a diagnostic challenge due to the lack of specific symptoms or unique brain imaging features, necessitating thorough histological evaluation for precise diagnosis and to ensure the gangliocytoma component is not overlooked

A Decade of Insights into Bilateral Adrenal Masses: Epidemiology, Clinical Profiles, and Therapeutic Approaches

Bilateral adrenal masses (BAMs) are rare, accounting for 10-15% of adrenal masses. Their diagnostic and therapeutic approaches differ from those of unilateral masses. This study aimed to analyze the epidemiological, clinical, paraclinical, and therapeutic profiles of BAMs. It is based on a retrospective analysis of 30 patient records from the Endocrinology, Diabetology, and Nutrition Department at CHU Hassan II in Fez, over a 12-year period. The average age of the patients was 53 years, with a male predominance (53.3%). BAMs were symptomatically revealed in 46.7% of cases, incidentally discovered in 23.3%, identified during tumor staging in 20%, and associated with a genetic syndrome in 10% of cases. Clinical examination revealed hypertension in 23.3% of patients, an abdominal mass in 10%, signs of adrenal insufficiency in 10%, hypercortisolism in 6.67%, and hyperandrogenism in 16% of cases. Biological assessment showed elevated urinary or plasma metanephrine derivatives in 9 patients, hypercortisolism in 3 patients, adrenal insufficiency in 3 patients, 11-β-hydroxylase enzyme block in 3 patients, and primary hyperaldosteronism in 1 patient. Adrenal computed tomography (CT) was performed in 29 patients. A staging workup, including PET scan, was requested in 20% of cases. MIBG scintigraphy was performed in 23.3% of patients. The investigations revealed 53% of BAMs were secreting and 47% non-secreting. There were 9 cases of pheochromocytomas, including 5 associated with a syndrome (3 MEN2a, 1 MEN2b, 1 VHL), 8 cases of non-secreting adenomas, 3 cases of congenital adrenal hyperplasia due to 11-β-hydroxylase enzyme block, 2 cases of adrenal metastases, 2 cases of adrenocortical carcinoma, 2 cases of adrenal cysts, 1 case of Conn’s adenoma, 1 case of cortisol-secreting adenoma, 1 case of primary adrenal lymphoma, and 1 case of adrenal hematoma. Treatment included the management of emergencies and preoperative preparation for patients eligible for surgery. Bilateral adrenalectomy was performed in 33.3% of patients, and unilateral adenectomy in 1 case of cortisol-secreting adenoma. Medical treatment was initiated in 16.6% of cases, and palliative treatment in 13.33% of cases. Surveillance alone was implemented in the remaining 33.3%. Prognosis depends on the type of adrenal mass, with outcomes marked by the death of one case of malignant adrenocortical carcinoma and one case of primary adrenal lymphoma. Our study, involving 30 cases of BAMs, highlights their clinical, biological, radiological, therapeutic, and prognostic characteristics. Data in the literature are limited compared to unilateral adrenal masses. Future larger studies are desirable for optimal management.

6629 Pure Androgen Secreting Adrenal Adenoma In A Premenopausal Woman

Abstract Disclosure: S. Syed: None. Background: Pure androgen secreting adrenal adenomas are extremely rare; most of the reported cases occur in post-menopausal women, with significant virilization of acute onset and are usually malignant. Here we present a case of an androgen secreting adrenal adenoma found in a young female with regular menstrual cycles and hirsutism. Clinical Case: A 27-year-old female with no significant past medical history was referred to Endocrinology clinic for evaluation of elevated testosterone level noted on a routine OBGYN exam. Patient had miscarriage six months prior to initial endocrine clinic visit. She had a routine follow up visit with her gynecologist after miscarriage and noted to have mild male pattern hair growth on her chin, worse after stopping birth control pills two years ago. Patient had menarche at 14 years of age and had regular menstrual cycle until started birth control pills. The skin examination revealed coarse male pattern hair on chin. Scattered acne was present on back. No signs of hypercortisolism were present. Labs done at time of initial consultation showed unremarkable comprehensive metabolic panel, thyroid and gonadotropic hormones. Testosterone was elevated at 122ng/dl (0-75ng/dl). Pelvic ultrasound done in the year prior showed normal uterus and adnexal structures. Further work up showed elevated DHEAS 781 µg/dL (18-391 µg/dL) and normal 17 OH progesterone and androstenedione levels. MRI abdomen with and without contrast done at outside facility showed a 4 cm x 3.3 cm x 4.1 cm well-circumscribed round shaped, mildly heterogeneously enhancing mass arising from right adrenal gland, with restricting diffusion, no signal dropout in out-of-phase imaging. Normal ovaries were noted. Given the large size of lesion, adrenalectomy was recommended. Patient underwent laparoscopic right adrenalectomy. The histologic features on pathology specimen were consistent with an adrenal cortical adenoma. No evidence of malignancy was seen (based on modified Weiss criteria). 3 weeks post op labs showed Testosterone level reduced to 19 ng/dl (0-75ng/dl) and DHEAS 138 ug/dl (18-391 µg/dL). Patient is recovering from recent childbirth. Conclusion: In adults, pure androgen secreting adrenal tumors are extremely rare. Diagnosis of malignancy is made with histopathological examination and Weiss score calculation. Our patient has unique presentation of having mild hirsutism in setting of regular menstrual cycles with no other symptoms of virilization and is noted to have unexpectedly large adrenal adenoma. Given the size of adenoma, there was concern for adrenal cortical carcinoma, however histological examination was consistent with a benign lesion. Presentation: 6/1/2024

8141 A Rare Case of Follicular Adenoma with Osseous Metaplasia and Mature Bone Formation- Is Surveillance Important?

Abstract Disclosure: K. Madani: None. Y. Al-Khazraji: None. D.H. Sacoto: None. G.K. Rai: None. R. Belokovskaya: None. A.A. Franco-Akel, MD: None. Osseous metaplasia and mature bone formation (MBF) are common in malignant thyroid and parathyroid tumors. It is very unusual to have MBF in a benign tumor-like follicular adenoma, as in the case we describe. We found only twelve such cases reported in the literature, and none addressed the cancer risk or need for surveillance for the development of potential malignancy. Thus, further studies are required to investigate the need for surveillance in patients not undergoing excision and in those with remnant thyroid tissue post-removal of the adenoma for recurrence. A 43-year-old female with history of hyperlipidemia and hypothyroidism had an incidental finding of midline neck mass during the evaluation of a left upper extremity lipoma. Ultrasound of the neck showed two nodules in the Left (L) thyroid lobe: a complex nodule measured 2.2 x 1.2 x 2.1 cm and a calcified nodule measuring 1.5 x 0.8 x 0.6 cm. MRI of the neck revealed a 1.9 cm heterogeneously enhancing nodule in the mid to lower pole of the L thyroid lobe. The patient had symptoms suggestive of compression, such as dysphagia, odynophagia, and hoarseness; thus, left hemothyroidectomy was performed. Tissue biopsy was notable for follicular adenoma with osseous metaplasia and mature bone formation. The patient remained clinically and biochemically euthyroid, with only mildly elevated thyrotropin level, and treatment with levothyroxine 25 mcg once daily was initiated. On post-surgical follow-up, there was a lack of data to support close monitoring modality and strategy for the remnant thyroid lobe and neck nodal compartments. We concluded with the patient to follow up in a clinical visit for a physical exam of the neck and to obtain a follow-up sonogram at an interval of six months, in addition to monitoring TSH levels to guide hypothyroidism therapy. Osseous metaplasia has not been established as a premalignant condition, but in certain tissues like the esophagus, metaplasia is a risk factor for malignant conversion, for instance, in Barrett’s esophagus leading to esophageal adenocarcinoma. Moreover, MBF in thyroid tissue has been attributed to the activity of basic fibroblast growth factor (bFGF) and bone morphogenetic protein-2 (BMP-2), which are linked with mitogenesis and tumorigenesis. Thus, the question arises if post-excision surveillance is required in patients with a history of osseous metaplasia in remnant thyroid tissue post-lobectomy or partial thyroidectomy. At this point, more epidemiological data on similar cases may help us understand the true risk of this clinical scenario, in order to develop a reasonable approach for surveillance. Presentation: 6/3/2024

8267 A Case of Pineal Gland Cyst and Adrenal Adenoma- Is this a New Syndrome?

Abstract Disclosure: M. Siddiqui: None. U. Rafat: None. S. Shaik: None. J.L. Gilden: None. Background: The Pineal Gland receives information through a complex multi-neuronal pathway and is responsible for the secretion of melatonin, a hormone regulated by circadian rhythm and suppressed by light stimuli. Melatonin is involved in biological rhythms and regulates immunomodulation, temperature homeostasis and maturation of hypothalamic-pituitary and gonadal axis, and can also modulate thyroid, prolactin, growth hormone, cortisol, and ACTH activity. Other effects include downregulation of thyroid activity. In addition, studies have shown that pineal body extracts can cause a minor increase in the zona glomerulosa and fasciculata of the adrenal gland. Case: A 54-year-old male with recurrent nephrolithiasis, obstructive sleep apnea and chronic rhinosinusitis, first presented to Endocrinology Clinic in 2018 for evaluation of an incidental adrenal adenoma (CT abdomen -2.8 cm, less than 10 Hounsfield units, left sided lipid rich adrenal adenoma). He had new onset erectile dysfunction, decreased libido, and fatigue. Initial lab work including LH, FSH, TSH, midnight salivary cortisol, ACTH, plasma cortisol AM, free and total testosterone, aldosterone, plasma and urine metanephrines/normetanephrines were all normal. MRI brain-1 cm thin-walled pineal cyst with internal characteristics compatible with proteinaceous benign pineal cyst. He was then lost to follow-up for 5 yrs due to the COVID pandemic. He had tried taking “testosterone protein builders”, but his symptoms remained. In addition, he now has twice weekly dull occipital headaches, relieved by ibuprofen. Lab tests-elevation in 1of 2 MN salivary cortisol samples at 0.58 mcg/dL (nl≤0.09), ACTH 12pg/mL(0-47), IGF1 261 ng/mL(50-317), and low total testosterone 222 ng/dl (250-1100) Free testosterone 36 pg/mL(35-155), borderline low TSH 0.386 µIU/ml(0.55-4.78), Free T3 and Free T4 were normal, thyroid antibodies-negative . Rest of the hormone evaluation was otherwise normal, LH 4.90 mIU/mL(1.5-9.3), FSH 11.10 mIU/mL (1.4-18.1), Prolactin 6.8 ng/mL(2.1-17.7). Thyroid Ultrasound-homogeneous echogenicity of the thyroid gland without discrete thyroid nodules. Repeat MRI brain- benign pineal cyst which was stable with no midline shift or mass effect. Repeat CT adrenal gland-lipid rich adrenal adenoma slightly reduced in size from 2.8 to 2.4 cm. Lab tests returned to normal one month later. Conclusion: Although it is unclear whether there is an exact association between this patient’s pineal cyst and adrenal adenoma, there have been intermittent hormonal abnormalities and symptoms consistent with hypogonadism and/or adrenal, also possibly related to the pineal abnormality. Although more research needs to be conducted explaining the relationship between the pineal and pituitary, and the adrenal gland, this clinical case suggests that there may be a relationship between these two abnormalities and symptoms. Presentation: 6/2/2024

6436 Adrenal Venous Congestion Clinically Mimicking Carcinoma In A Large Degenerated Adrenocortical Adenoma

Abstract Disclosure: K. Ishiwata: None. S. Suzuki: None. S. Watanabe: None. Y. Yamazaki: None. H. Sasano: None. K. Yokote: None. Introduction: A large heterogenous adrenal tumor is often malignant. Only eight reported cases of adrenal adenoma harboring hemorrhage or venous thrombosis have been reported but their details not. We encountered a case of large degenerated adrenocortical adenoma harboring adrenal venous congestion mimicking adrenocortical carcinoma. Case: Adrenal tumor was incidentally detected in a 71-year-old woman with a history of hypertension, dyslipidemia, and osteoporosis in her regular health check-up. Abdominal CT and MRI demonstrated a heterogenous mass measuring 7.6 cm with peripheral enhancement in the left adrenal gland. [1]3[1]I-adosterol scintigraphy revealed accumulation only in the left tumor margins with no accumulation in [1]8F-FDG-PET scan. Results of endocrinologic evaluation were consistent with subclinical Cushing syndrome. The large size and heterogeneous nature of the mass clinically suggested adrenocortical malignancy, and left laparoscopic adrenalectomy was subsequently performed. The resected left adrenal lesion was encapsulated and measured 6.5 x 6.3 x 5.8 cm appearing almost black and partially yellow on the cut surface. Microscopically, the nodule was composed of SF-1 positive, clear cortical cells with abundant hyaline stroma containing numerous dilated veins with CD31/34-positive endothelial cells, and no foci of necrosis or hemorrhage were detected. The tumor cells above were positive for steroidogenic enzymes including CTP17, HSD3β and CYP11B1 but negative for CYP11B2. Ki-67 labeling index was &amp;lt;2% in the tumor. The criteria of Weiss (0 out of 9) revealed that the tumor was cortisol-producing adrenocortical adenoma. Discussion: In this case, clusters of SF1-positive cortisol producing adrenocortical adenoma cells were present in the hyalinized stroma containing many dilated blood vessels. These findings indicated that those histological features were caused by ischemic degeneration due to intratumoral circulatory disturbance, which clinically simulated adrenocortical carcinoma. Conclusion: We have experienced a very rare case of cortisol-producing adrenocortical adenoma with adrenal venous congestion, resulting in marked enlargement of the lesion. Presentation: 6/3/2024

6630 Crooke Cell Pituitary Adenoma Without a Cushingoid Appearance

Abstract Disclosure: J. Hodge: None. S. Idriss: None. I. Jamal: None. R. Panta: None. Background: Crooke cell pituitary adenomas are corticotroph adenomas with Crooke’s cell changes, which are cytoplasmic accumulation of cytokeratin filaments in response to glucocorticoid excess. They are aggressive, invasive tumors, typically macroadenomas, with a high rate of Cushing’s disease and post -treatment (surgery and/or radiotherapy) recurrence. We present a case of Crooke cell pituitary adenoma without a Cushingoid appearance. Clinical Case: A 66-year-old Hispanic male with a 16-year history type II diabetes mellitus with retinopathy, presented with a pituitary macroadenoma at age 60. This was found incidentally when he had a brain CT for evaluation of vertigo. Subsequent brain MRI demonstrated a large 2.4 x 2.0 x 1.7 cm pituitary macroadenoma with extension into the suprasellar cistern and the left cavernous sinus. He was asymptomatic, without Cushingoid features. Hormonal evaluation revealed central hypogonadism and hypothyroidism, mildly elevated prolactin of 39.3 (4-15.2 ng/mL) and morning cortisol level of 12.7 (6-18.4 ug/dL). A 24-hour urine cortisol was normal. He was started on testosterone and thyroid hormone replacement therapy. His diabetes mellitus became progressively difficult to control with a peak HbA1C of 10.1%, about 7 years prior the macroadenoma finding. His HbA1C remained persistently elevated in the 8.1-9.5% range despite being on 6 anti-hyperglycemic agents, including basal insulin. A morning ACTH level 4 years after the initial visit, was 115 (6-50 pg/mL) with a cortisol level of 13.7 (6-18.4 ug/dL). Repeat morning ACTH and cortisol levels 7 months later were very similar. 5 years after the macroadenoma finding, he was noted to have worsening glaucoma, which was believed to be due to optic nerve compression. Therefore, he underwent endoscopic transphenoidal resection for tumor debulking. Pathology supported the diagnosis of Crooke cell corticotroph pituitary adenoma. Repeat brain MRI 3-months post-surgery demonstrated residual tumor, but normal optic chiasm and optic tracts. He had an abnormal dexamethasone suppression test prior to surgery (cortisol 6.4 ug/dL) and continued to do so post-operatively (cortisol 3.7 ug/dL with persistently elevated ACTH). Just prior to his surgery, testosterone replacement therapy was held. Total testosterone level after surgery was normal 309.6 (280-800 ng/dL) and free testosterone 7 (5-21 ng/dL); he remained off replacement therapy. Conclusion: Crooke cell pituitary adenomas are rare with limited data on prognosis and guidance for clinical management. Some patients may have subtle features of hypercortisolism, such as difficult to control diabetes mellitus, without an overt Cushingoid appearance. Presentation: 6/1/2024

S2460 Endoscopic Papillectomy for a Rare Case of Synchronous Major and Minor Papillary Adenoma

S2460 Endoscopic Papillectomy for a Rare Case of Synchronous Major and Minor Papillary Adenoma

Case report of a rare benign breast lesion: Unveiling nipple adenoma

Nipple adenoma is a rare and benign proliferative lesion originating from the lactiferous ducts of the nipple. It predominantly affects middle-aged women and can mimic the clinical and radiological features of more concerning breast malignancies. Due to its uncommon occurrence and overlapping characteristics with malignant lesions, a definitive diagnosis often requires histopathological examination. We present a case of nipple adenoma in a 30-year-old woman, detailing the clinical, radiological, and histopathological features, as well as the management and outcomes.

Submandibular gland pleomorphic adenoma masquerading as a multinodular goitre: a case report

Tumors of the salivary gland comprise of a small percentage of all head and neck tumors, of which nearly 80% are benign. The submandibular gland tumors constitute only 5-10% of this group, of which pleomorphic adenoma is the commonest type. They can weigh several kilograms and grow to ludicrous sizes. If left untreated, they may result in facial deformity and airway compromise. We describe a case of a big pleomorphic adenoma arising from submandibular salivary gland that developed in a 65 years old female presenting as a swelling in right side of neck since 15 years. The dimensions of the lesion were roughly around 8×12×11 cm. During surgical examination, a solid mass that originated from the right submandibular gland was discovered to be linked to both the skin and the soft tissue beneath it. Pleomorphic adenoma was confirmed by histopathological examination. Review and discussion of the relevant literature was conducted. This article's emphasis is on the fact that, although the pleomorphic adenoma is a well-reported entity, they can attain very large sizes involving the neck region and can misrepresent itself leading to diagnostic dilemma due to many possible differential diagnoses of neck swellings.

Giant adolescent tubular adenoma of the breast mimicking giant fibroadenoma: a case report

Tubular adenomas are a rare benign breast neoplasm, often clinically indistinguishable from fibroadenomas or phyllodes tumors. We present a case of the largest adolescent tubular adenoma reported in the literature, measuring 12.5 cm. Radiological evaluation and histopathological assessment of the core biopsy sample was consistent with a giant fibroadenoma. However, the final histopathological analysis of the excisional biopsy sample confirmed a tubular adenoma. This case highlights the clinical conundrum of reaching the definitive diagnosis in a patient with a tubular adenoma. A contemporary review of the clinical features, radiology, histopathology and management options for tubular adenoma of the breast was undertaken.