BACKGROUND

Central diabetes insipidus (CDI) is a common complication following transsphenoidal surgery for pituitary adenomas, but CDI as an initial presentation in pituitary adenomas is extremely rare. We report a case of a 67-year-old Filipino male with pituitary macroadenoma presenting as central diabetes insipidus, manifesting as a two-month history of severe frontotemporal headache, increased thirst, and polyuria, which was managed with desmopressin followed by transsphenoidal surgery. Three months postoperatively, the thyroid and adrenocorticotropic axis remained intact, and pituitary bright spot recovery was observed. He was clinically stable; hence, desmopressin was gradually tapered and discontinued. This case report presents a unique case of a pituitary adenoma that initially presented with central DI but later showed a complete resolution of symptoms along with the normalization of the "bright spot" seen on MRI, a hallmark of the posterior pituitary. Treatment options for preoperative CDI may include surgical or medical management, with some cases reported as self-limiting. However, the rarity of such cases underscores the urgent need for more clinical studies to fully understand the course of this condition. This case highlights a unique presentation of central diabetes insipidus in a pituitary macroadenoma and the possibility of complete resolution of symptoms coinciding with pituitary bright spot recovery post operatively.

Adenoma ; Diabetes Insipidus ; Diabetes Insipidus, Neurogenic ; Neoplasms ; Pituitary Neoplasms ; Research Report

Introduction: Functioning gonadotroph adenomas, also known as follicle-stimulating hormone (FSH)-secreting pituitary adenomas (FSH-omas), are rare pituitary tumors characterized by the secretion of biologically active FSH. Unlike the more common clinically nonfunctioning gonadotroph adenomas, these tumors produce endocrine manifestations due to excessive FSH secretion. Clinical presentation may result from local mass effects such as headache, visual disturbances due to optic chiasmal compression, or from hormonal activity affecting reproductive function. The endocrine manifestations vary between males and females and can lead to diagnostic challenges. Cases: We report two patients with functioning gonadotroph adenomas presenting with distinct clinical manifestations. The first case involved a 23-year-old female who presented with secondary amenorrhea for 4 years, accompanied by personality changes. Abdominal ultrasound demonstrated bilaterally enlarged multicystic ovaries suggestive of ovarian hyperstimulation, which subsequently regressed postoperatively. Biochemical evaluation revealed inappropriately elevated FSH levels (3.5-fold increase), with suppressed luteinizing hormone (LH) and markedly elevated estradiol levels (11-fold increase). Pituitary magnetic resonance imaging (MRI) identified a large sellar–suprasellar mass with mass effect, consistent with a pituitary macroadenoma. The second case involved a 68-year-old male who presented with progressively worsening vision over 2 years. Hormonal evaluation demonstrated elevated FSH levels (4.5-fold increase), with relatively normal LH and low testosterone levels. Pituitary MRI revealed a sellar mass consistent with a pituitary adenoma. Both patients subsequently underwent pterional craniotomy with tumor debulking. Histopathological examination confirmed gonadotroph adenomas, with positive immunohistochemical staining for FSH. Conclusion Functioning gonadotroph adenomas are rare and may present with diverse clinical features related to gonadal hyperstimulation or mass effects. Early recognition of the characteristic hormonal profile and radiological findings is crucial for diagnosis and appropriate management. These cases highlight the importance of considering functioning gonadotroph adenoma in patients presenting with unexplained gonadal hyperstimulation or atypical reproductive hormonal profiles.
Gonadotrophs ; Adenoma

OBJECTIVES

To report a case of a giant pleomorphic adenoma in a 64-year-old Filipino woman, its management and surgical outcome.

METHODS

Design:Case Report

Setting:Tertiary Government Training Hospital

Patient: One

RESULTS

A 64-year-old woman presented with a 50-year history of a slow growing, painless, left infra-auricular mass, not associated with facial weakness, xerostomia, or hearing loss. Computed tomography revealed a 14 x 15 x 19 cm large lobulated complex enhancing mass with calcifications and septations, with no enlarged lymph nodes identified in the neck. Fine needle aspiration cytomorphology was consistent with pleomorphic adenoma. The patient underwent superficial parotidectomy with facial nerve preservation. The facial nerve was identified using standard landmarks. Final histopathological findings were consistent with pleomorphic adenoma measuring 23.5 cm x 11.5 cm x 15 cm and weighing 2177 grams

CONCLUSION

Pleomorphic adenoma can grow to a gigantic size if left untreated. It often presents as a chronic, slow growing and painless swelling. The approach to its diagnosis is mainly clinical and can be confirmed by fine needle aspiration biopsy and computed tomography scan. In our case, the standard landmarks for facial nerve identification were still reliable despite the size of the mass, producing good post-surgical outcomes.

Human ; Female ; Middle Aged: 45-64 Yrs Old ; Adenoma ; Lymph ; Needles ; Diagnosis ; Xerostomia ; Hearing Loss ; Neck ; Research Report ; Tomography ; Facial Nerve

OBJECTIVE: To evaluate the clinical outcomes and explore the application of partial sialoadenectomy for the treatment of benign tumors in the submandibular gland (SMG). METHODS: Patients with pleomorphic adenoma of the SMG who underwent surgical treatment in the Department of Oral and Maxillofacial Surgery, Peking University Shenzhen Hospital, from October 2017 to February 2021, were enrolled and assessed in the follow-up. Fifteen patients underwent partial sialoadenectomy (PS group), and 18 patients underwent total sialoadenectomy (TS group). Postoperative salivary secretion, degree of dry mouth, appearance changes of the face and neck, nerve damage, and tumor recurrence were compared between the groups. The volume of the glands on the operated and contralateral sides of the patients in the PS group, the saliva flow rate, and their correlations, were also analyzed. RESULTS: There was no recurrence during the follow-up period. The whole saliva flow rate at rest in the PS group was higher than that in the TS group [(2.15±1.10) g/5 min vs. (1.35±0.97) g/5 min, t=2.208, P=0.035)], while the stimulated saliva flow rate was not significantly different. The objective feeling of dry mouth, evaluated by visual analogue scale (VAS) score, was more obvious in the TS group than in the PS group (Z=-2.244, P=0.025). In the PS group, the resting saliva flow rate of the SMG on the operated side was lower than that on the contralateral side of the same patient [(0.92±0.40) g/5 min vs. (1.18±0.40) g/5 min, t=-2.821, P=0.014], however, in the cases whose remaining SMG was more than 80% of the contralateral side, the saliva flow rate of both sides was not significantly different (t=-0.027, P=0.980). There was no significant difference in the saliva flow rate per unit volume of the gland on either side (t=-0.015, P=0.989), and the saliva flow rate of the operated SMG was positively correlated with the volume of the remaining gland (r=0.750, P=0.012). The VAS scores for neck deformity were not significantly different between the two groups (t=-0.997, P=0.319). No symptoms of nerve injury occurred in either group. CONCLUSION Partial sialoadenectomy in the SMG can safely remove benign tumors while preserving glandular secretory function, with fewer complications and improved quality of life.
Humans ; Submandibular Gland/pathology* ; Male ; Female ; Middle Aged ; Adenoma, Pleomorphic/surgery* ; Adult ; Treatment Outcome ; Submandibular Gland Neoplasms/surgery* ; Saliva/metabolism* ; Aged

The oral glucose growth hormone suppression test is commonly used in the clinical diagnosis of acromegaly, but its results can be influenced by a variety of factors. This case report discusses a patient with a pituitary tumor and concurrent liver cirrhosis, highlighting the complexities in interpreting test results under such conditions. The patient, a 54-year-old male, presented with blurred vision as his primary complaint. Notably, the physical examination revealed no changes in facial features, no enlargement of hands or feet, and no other symptoms typically associated with acromegaly, which might otherwise suggest excessive growth hormone activity. Magnetic Resonance Imaging (MRI) of the pituitary gland indicated that the gland was within normal size parameters, but a small low-intensity lesion mea-suring approximately 3 mm×2 mm identified. This finding was consistent with a pituitary microadenoma. The patient's fasting growth hormone levels were significantly elevated at 8.470 μg/L, compared with the normal range of less than 2.47 μg/L. Conversely, fasting insulin-like growth factor-1 (IGF-1) levels were notably low, recorded at 41 and 52 μg/L, whereas the normal range for a person of his age was between 87 and 234 μg/L. Other pituitary hormones, including those regulating the thyroid, adrenal cortex, and sex hormones, were found to be within normal ranges. Despite this, during the glucose growth hormone suppression test, an abnormal elevation of growth hormone was observed. To investigate further, the patient was administered branched-chain amino acids, and the suppression test was repeated. However, the abnormal elevation of growth hormone persisted, indicating a failure to normalize the response. Given the patient's lack of clinical signs typically associated with elevated growth hormone secretion, the history of liver cirrhosis became a significant consideration. The disparity between elevated growth hormone levels and reduced IGF-1 levels suggested that the pituitary lesion was a non-functional adenoma rather than a source of excess hormone production. Consequently, it was concluded that the abnormal response of growth hormone to the glucose suppression test was likely related to the patient's liver cirrhosis. In addition to chronic liver disease, various other conditions could influence the results of the oral glucose tolerance growth hormone suppression test. According to the literature, factors such as puberty, diabetes, anorexia nervosa, and protein malnutrition could also affect test outcomes. These conditions could cause similar abnormalities in growth hormone dynamics, complicating the diagnosis. Therefore, clinicians must be vigilant and consider these potential influences when interpreting test results.For an accurate diagnosis of acromegaly, it is essential to combine clinical symptoms, detailed medical history, and imaging studies. The presence of conditions like liver cirrhosis should prompt careful interpretation of the test results, ensuring that other contributing factors are not overlooked. This comprehensive approach is crucial to avoid misdiagnosis and to ensure that appropriate treatment strategies are implemented based on a thorough understanding of the patient's overall health status.
Humans ; Male ; Middle Aged ; Pituitary Neoplasms/blood* ; Liver Cirrhosis/blood* ; Adenoma/blood* ; Human Growth Hormone/blood* ; Insulin-Like Growth Factor I/metabolism* ; Acromegaly/etiology* ; Magnetic Resonance Imaging

Objective:To assess the efficacy of "three-step" strategy for preparing autologous pedicled nasal mucosal flaps in repairing cerebrospinal fluid（CSF） leaks following transsphenoidal pituitary adenoma surgery. Methods:A retrospective study was conducted on the clinical data of 25 patients who developed CSF leaks after transsphenoidal pituitary adenoma surgery at the First Affiliated Hospital of Kunming Medical University between July 2012 and June 2022. Surgical repair was selected step by step using nasal septal mucosal flap with either the posterior septal artery or septal branch of the sphenopalatine artery as the pedicle, or a pedicled middle turbinate mucosal flap. All patients underwent ≥2-year endoscopic follow-up to assess flap viability and CSF leak recurrence. Results:The median postoperative hospital stay was 4 days. Five patients developed intracranial infections postoperatively. The follow-up period ranged from 2 to 12 years. Nasal endoscopic examinations showed good mucosal flap growth, with no recurrence of CSF leakage in any of the patients. Conclusion:High-flow cerebrospinal fluid（CSF） leaks following pituitary tumor surgery pose significant challenges for clinical repair. Based on intraoperative nasal septal mucosal preservation and the condition of sellar base CSF leakage, the "three-step" strategy for preparing autologous pedicled nasal mucosal flaps-utilizing posterior septal artery, ethmoidal artery-based, or pedicled middle turbinate mucosal flaps sequentially-is a safe and effective repair method.
Humans ; Retrospective Studies ; Pituitary Neoplasms/surgery* ; Surgical Flaps ; Nasal Mucosa/surgery* ; Cerebrospinal Fluid Leak/surgery* ; Adenoma/surgery* ; Postoperative Complications/surgery* ; Male ; Female ; Middle Aged ; Adult ; Aged

Pleomorphic adenoma arising from the torus tubarius of the nasopharynx is an extremely rare entity with limited epidemiological data and unclear etiological mechanisms. Its pathogenesis may be related to the eustachian tube salivary glands. Here we report an elderly female patient with a long history of snoring, hypernasal speech and epistaxis. Following comprehensive diagnostic evaluation, the patient underwent tumor resection under nasal endoscopy. There were no postoperative complications, the symptoms were significantly improved, and there was no obvious recurrence during the follow-up. We summarized the experience of diagnosis and treatment of giant pleomorphic adenoma of the tubal torus. The main treatment for tubal torus pleomorphic adenoma is complete surgical resection, with a good prognosis and a low recurrence rate.
Humans ; Female ; Adenoma, Pleomorphic/surgery* ; Aged ; Nasopharynx/pathology*

Objective:To summarize the clinical characteristics, diagnosis and treatment experience of salivary pleomorphic adenoma in children. Methods:Thirty patients with salivary pleomorphic adenomas treated in Beijing Childrens Hospital from January 2008 to December 2022 were retrospectively reviewed, including 11 boys and 19 girls, with the age ranging from 0.3 to 14.4 years（median age 10.4 years）. Initial presentation, medical history, imaging workups, surgical approaches, complications, rates of recurrence were evaluated. Results:Major salivary gland lesions were most common（n=24, 80%）; 53.3%（16 of 30） arising in the submandibular glands and 26.7%（8 of 30） in the parotid. Minor salivary gland lesions（n=6, 20%） were removed from the palate, tongue, face, trachea, nasopharynx, and upper mediastinumand. Preoperative imaging was reviewed in all patients and consisted of 26 ultrasound exams, 2 computerized tomography（CT） exams, and 15 magnetic resonance imaging（MRI） exams. Fine needle aspiration biopsy was performed in 12 patients. Surgical excision was performed in all patients. Postoperative complications included transient facial paresis（n=3）, Pneumonia and pleural effusion（n=1）. Average length of follow-up was 36.7 months; confirmed recurrence occurred in one patients. Conclusion:The symptoms of salivary gland pleomorphic adenoma in children are different according to the location of the tumor. The treatment is complete surgical resection, and a small amount of normal tissue around the tumor should be removed to reduce recurrence.
Humans ; Male ; Female ; Child ; Adenoma, Pleomorphic/diagnosis* ; Adolescent ; Retrospective Studies ; Salivary Gland Neoplasms/diagnosis* ; Child, Preschool ; Infant ; Neoplasm Recurrence, Local

A 30-year-old female patient with a benign tumor in the superficial lobe of the left parotid gland underwent tumor resection via a 5 cm intra-hairline incision, using the da Vinci Xi surgical robot combined with the NIM-Response 3.0 facial nerve monitoring system. During the operation, facial nerve branches were located and protected through facial nerve monitoring, and the robotic arms were used for precise tumor dissection. Postoperatively, the facial nerve function, incision healing, and tumor recurrence were observed. duration of the procedure was 120 minutes, and the tumor（2.0 cm×1.5 cm） was completely resected. Postoperative pathological examination indicated a pleomorphic adenoma. During the 3-month postoperative follow-up, the patient's facial nerve function remained normal, no salivary fistula occurred, the incision was hidden within the hairline, no tumor recurrence was found in the ultrasound reexamination, and the patient was highly satisfied with the appearance. The surgical approach of robot-assisted resection of benign parotid gland tumor via a hairline incision under facial nerve monitoring has significant advantages in facial nerve protection and cosmetic effect, and is suitable for patients with benign parotid gland tumors meeting specific conditions.
Humans ; Female ; Adult ; Parotid Neoplasms/surgery* ; Facial Nerve ; Robotic Surgical Procedures/methods* ; Adenoma, Pleomorphic/surgery* ; Parotid Gland/surgery* ; Monitoring, Intraoperative

The parathyroid glands play a crucial role in calcium regulation through parathyroid hormone (PTH) production. Indicators for those with hyperparathyroidism would be elevated serum calcium and PTH levels with further evaluation followed by imaging with neck ultrasonography and parathyroid scintigraphy. Limitations in the initial imaging modalities include operator-dependent sensitivity in neck ultrasonography, and poor spatial resolution, and poor sensitivity for smaller-sized adenomas in parathyroid scintigraphy.

This case report describes a 24-year-old female with elevated parathyroid hormone, and borderline elevated calcium levels with an initial diagnosis of primary hyperparathyroidism secondary to a suspected parathyroid adenoma. The dual tracer dual-phase scintigraphy accompanied by SPECT/CT and subtraction method was negative for a parathyroid adenoma, however, ultrasonography of the neck showed a suspicious nodular structure. Due to the high clinical suspicion, a subsequent ¹⁸F-Choline PET/CT was done and identified an ¹⁸F-Choline-avid lesion, highly suggestive of a parathyroid adenoma. This prompted parathyroidectomy in which histopathology as well as intraoperative reduction in parathyroid hormone levels, confirmed the presence of a parathyroid adenoma.

This is the first recorded ¹⁸F-Choline PET/CT usage for a parathyroid adenoma in the country and highlights the potential of its usage as a sensitive and specific imaging modality in cases where conventional imaging is inconclusive.

Human ; Female ; Young Adult: 19-24 Yrs Old ; Parathyroid Adenoma ; Parathyroid Neoplasms ; Hyperparathyroidism