Indian Journal of Endocrine Surgery and Research

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2024 | January-June | Volume 19 | Issue 1

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LETTER TO THE EDITOR

Rajni K Sah, Sabaretnam Mayilvaganan

Thyroid Malignancy and Grave's Disease

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:1] [Pages No:1 - 1]

   DOI: 10.5005/jp-journals-10088-11234  |  Open Access |  How to cite  | 

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ORIGINAL RESEARCH

Tanjum Kamboj, Ankur Bhatnagar, Anupama Singh, Sabaretnam Mayilvaganan

Gynecomastia: Evaluation and Management with Modified Intra-areolar Incision at Tertiary Care Center

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:4] [Pages No:2 - 5]

Keywords: Benign breast disease, Gynecomastia, Klinefelter syndrome, Lipomastia, Mammoplasty, Tamoxifen

   DOI: 10.5005/jp-journals-10088-11224  |  Open Access |  How to cite  | 

Abstract

Objectives: To study the hormonal profile of patients of gynecomastia, preoperative evaluation management, and outcome. Materials and methods: Retrospective study of all male patients presenting to the outpatient in plastic surgery clinic with gynecomastia. Complete endocrinology workup was done for each patient. Patient were treated by either liposuction alone or combined with gland excision with modified intra-areolar incision. Patients were followed up at 2-week, 1-month, and 6-month interval for complications, recurrence, and esthetic outcome. Results: Overall complication and recurrence rate was low with early and long-term use of postoperative pressure garment. Modified intra-areolar incision showed better esthetic outcomes in patients who underwent gland excision. Conclusion: Gynecomastia patient shows least recurrence and complications when treated with combination of liposuction and gland excision with superior esthetic results with intra-areolar incision.

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CASE REPORT

Anandh Saravanan Moorthy, Raju Palanigoundar, Karthikeyan Raju, Archana Harikrishnan, Senthil Kumar, Sabarigirivasan Kumba Santharam

Poorly Differentiated Thyroid Cancer—Still a Clinical Dilemma: A Case Study

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:3] [Pages No:6 - 8]

Keywords: Case report, Non-anaplastic follicular cell-derived thyroid cancer, Poorly differentiated thyroid carcinoma, Thyroid carcinoma, Turin criteria

   DOI: 10.5005/jp-journals-10088-11227  |  Open Access |  How to cite  | 

Abstract

Poorly differentiated thyroid cancer (PDTC) lies in the intermediate zone in the thyroid carcinoma spectrum. It is the most common cause of non-ATC cancers-related mortality. We happened to encounter such a rare disease in one of our patients and are here to discuss the entire details about the course of the patient in our hospital. This was a 73-year-old female patient who was operated on for goiter and postoperative histopathology revealed poorly differentiated thyroid cancer. Though it is an important cause of mortality in 15% of cases of thyroid cancer, very little is understood about the PDTC.

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CASE REPORT

Kishore Abuji, Akanksha Gautam, Manoj K Thumma, Sanjay K Bhadada, Divya Dahiya

Giant Cell Epulis as a Presenting Manifestation of Primary Hyperparathyroidism: A Case Series from a Tertiary Care Center

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:4] [Pages No:9 - 12]

Keywords: Case report, Epulis, Giant cell granuloma, Hyperparathyroidism, Primary hyperparathyroidism

   DOI: 10.5005/jp-journals-10088-11228  |  Open Access |  How to cite  | 

Abstract

Brown tumors are sequelae of abnormal bone metabolism in primary hyperparathyroidism (PHPT) when there is increase in osteoclastic activity and fibroblastic proliferation. They can be located in any part of the skeleton, but commonly affect the bony thorax (ribs and clavicle), pelvic rim, and long bones (tibia and femur). Clinically, significant lesions in the craniofacial bones are rare and usually involve the mandible. Oral manifestation may be the only presenting feature of PHPT and patient generally report first to a maxillofacial surgeon with a diagnosis of a giant cell tumor which is very similar in clinical, radiological, and histological features to brown tumor. Therefore, we aim to share our experience of giant cell epulis as a presenting manifestation in PHPT patients in a tertiary care center in North India.

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CASE REPORT

Basavaraju Santosh, Narasimha Anilkumar, Balachandra Bhat

A Rare Association of Adrenal Lipoma and Primary Hyperaldosteronism: A Case Report

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:4] [Pages No:13 - 16]

Keywords: Adrenal incidentaloma, Adrenal lipoma, Case report, Laparoscopic adrenalectomy, Primary hyperaldosteronism

   DOI: 10.5005/jp-journals-10088-11229  |  Open Access |  How to cite  | 

Abstract

Aim and background: Adrenal lipomas are very rare benign tumors of adrenal gland. Most of these are non-functional and asymptomatic, with most of them being diagnosed on autopsy or as an incidentaloma on imaging. There are no cases being reported of adrenal lipoma associated with hormonal secretion in the literature until now. Primary hyperaldosteronism is one of the common causes of resistant hypertension which can increase the cardiovascular morbidity and mortality. It is usually caused by an adrenal adenoma, unilateral or bilateral adrenal hyperplasia (BAH), or in rare cases adrenal carcinoma or inherited conditions of familial hyperaldosteronism. It is important to diagnose it appropriately as it can be treated effectively with either medical or surgical modalities. We report a rare case, probably the first case, of association of adrenal lipoma with primary hyperaldosteronism, who was treated successfully with surgery. Case description: A 57 years old female, with uncontrolled type 2 diabetes mellitus and hypertension, was admitted with right lower limb cellulitis and lower respiratory tract infection. She was on three antihypertensive medications and yet the blood pressure was high (180/100 mm Hg). Her initial blood investigations during admission showed: HbA1c–9.5%, serum potassium–4.1 mmoL/L, serum creatinine–0.8 mg/dL. Computed tomography of the chest, which was done for her lung infection, revealed a lipid rich right adrenal adenoma (incidentaloma), of size 21 × 17 mm. Clinically, she did not have any specific symptoms or signs of Cushing syndrome, pheochromocytoma or primary hyperaldosteronism, except for resistant hypertension. There was no clinical evidence of adrenal insufficiency either. Baseline 8 am cortisol was found to be normal-22.93 μg/dL. The 2D echocardiogram was normal except for grade I diastolic dysfunction. She was treated for her infection, following which she recovered and discharged. Hormonal evaluation for right adrenal adenoma was done later during follow-up. The investigations showed: 8 am cortisol–14.9 μg/dL, potassium–4.1 mmoL/L; post over-night dexamethasone suppression test cortisol–1.7 μg/dL, plasma free metanephrines–89.3 pg/mL (<100), plasma free normetanephrines–472 pg/mL (216), 24 hours urinary normetanephrines–91.26 μg, plasma renin activity (PRA)–0.32 ng/mL/hour, and serum aldosterone–12.1 ng/dL. The aldosterone renin ratio (ARR) was 37.81, which was found to be elevated and suggestive of primary hyperaldosteronism. Then she underwent confirmatory test in the form of saline infusion test (SIT). Post-infusion serum aldosterone level was 6.99 ng/dL. A diagnosis of primary hyperaldosteronism secondary to right adrenal adenoma was considered. She was started on spironolactone along with prazosin and verapamil. Following spironolactone treatment, her blood pressure normalized. After adequate glycemic and blood pressure control, she underwent laparoscopic right adrenalectomy. The histopathological report of operative specimen was suggestive of adrenal lipoma. There were also foci of adrenocortical hyperplastic nodules in the rest of the gland. Following surgery, her blood pressure and glucose levels were maintained with lesser dose of medications. Postoperative PRA was 1.17 ng/mL/hour, serum aldosterone was 9.93 ng/dL, and ARR was 8.48, which showed biochemical normalization. Conclusion: Although very rare, adrenal lipomas should always be considered when evaluating adrenal tumors. They are usually non-functional and asymptomatic, but can be associated with symptoms related to local tumor per se and hormone hypersecretion. Hence, it is important to subject all these patients for biochemical evaluation before surgery to avoid untoward complications. Whether these tumors are completely non-functional or are associated with hormone hypersecretion, needs to be evaluated. The pathogenesis of hormone hypersecretion, if at all, by these tumors, needs to be looked for. Clinical significance: We present a very rare case of adrenal lipoma with surrounding foci of adrenocortical hyperplastic nodules in the rest of the gland; and probably the first case of its kind in association with primary hyperaldosteronism.

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CASE REPORT

Ajay Mohan, Rijuta Aphale, Akanksha Dingoriya, Yashwant S Rathore, Sunil Chumber, Shipra Aggarwal

Atypical Parathyroid Adenoma with Multiple Brown Tumors: A Rare Case

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:4] [Pages No:17 - 20]

Keywords: Atypical parathyroid adenoma, Browns tumor, Case report, Hypercalcemia, Hyperparathyroidism, Hypercalcemic crisis

   DOI: 10.5005/jp-journals-10088-11232  |  Open Access |  How to cite  | 

Abstract

Objective: Present a case of an atypical parathyroid adenoma with extreme clinical features like multiple brown tumors and discuss the uncertainties in differentiating between a benign and malignant entity. Background: The atypical parathyroid adenoma is purely a histological diagnosis. It is a parathyroid tumor with atypical histological features different from an adenoma but not similar enough to be considered as a carcinoma. It has an uncertain malignant potential. Case description: We report the case of a 48-year-old woman, who presented with multiple swellings all over the bony areas of her body. Positron emission tomography computed tomography (PET CT) showed a fluorodeoxyglucose (FDG) avid lesion in the right thyroid lobe with multiple skeletal lesions. Further evaluation revealed hypercalcemia following which a Sestamibi scan was done. Diagnosis of primary hyperparathyroidism (PHPT) with right inferior parathyroid adenoma and associated multiple brown tumors in hypercalcemic crisis was made. The patient underwent focused right inferior parathyroidectomy and postoperatively, iPTH and calcium levels dropped significantly. The histopathological examination of the removed tissue concluded to be an “atypical” parathyroid adenoma. The atypical parathyroid adenoma is a very rare tumor, and the diagnosis is still a challenge, the outcome of patients is not well known yet, therefore close surveillance is important and must be done regularly.

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CASE REPORT

Rohit Singh, Chanchal Rana, M Shreyamsa, Pooja Ramakant, Anand Kumar Mishra

Exploring the Extraordinary: Unusual Hematopoietic Malignancies of Thyroid Gland

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:5] [Pages No:21 - 25]

Keywords: Case report, Langerhans cell histiocytosis, Plasmacytoma, T-cell lymphoma, Thyroid diseases, Thyroid neoplasms

   DOI: 10.5005/jp-journals-10088-11235  |  Open Access |  How to cite  | 

Abstract

Thyroid disorders encompass a broad spectrum of pathological conditions, the majority of which are benign and also include autoimmune thyroiditis, multinodular goiter, and papillary thyroid carcinoma. However, some thyroid pathologies are unusual and pose diagnostic and management challenges. This study discusses the presentation, diagnostic challenges, and management of these rare presentations and tumors, as seen in our institution. First patient with primary T-cell lymphoma of the thyroid, an exceedingly rare malignancy accounting for a minute fraction of thyroid neoplasms. Second patient highlights an uncommon case of solitary extramedullary plasmacytoma of the thyroid, constituting less than 1% of all extramedullary plasmacytomas. Third patient portrays Langerhans cell histiocytosis (LCH) of the thyroid, an unusual entity often presenting diagnostic complexities due to its rarity and association with other thyroid pathologies. These cases emphasize the rarity of these thyroid pathologies and the need for a comprehensive approach to diagnosis and management. Tailored treatment strategies are necessary, taking into account the clinical presentation and extent of the disease. Due to the limited data available for these rare pathologies, further research and comprehensive reporting are crucial for refining our understanding and optimizing treatment strategies. In conclusion, these cases exemplify the diagnostic and management challenges posed by unusual thyroid pathologies and underscore the importance of accurate diagnosis and a multidisciplinary approach to manage these rare conditions effectively.

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CASE REPORT

Swarna Azaria, Shawn Sam Thomas, Kripa Elizabeth Cherian, Vinay Murahari Rao, Elanthenral Sigamani, Mazhuvanchary Jacob Paul

The Mediastinal Parathyroid-peeping through the Window

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:4] [Pages No:26 - 29]

Keywords: Case report, Hyperparathyroidism, Parathyroid adenoma, Primary hyperparathyroidism

   DOI: 10.5005/jp-journals-10088-11237  |  Open Access |  How to cite  | 

Abstract

Introduction: Mediastinal parathyroid adenomas (MPAs) result from aberrant migration of the parathyroid during development, constituting 20% of ectopically located adenomas. Aortopulmonary window (APW) parathyroid adenomas are rare, accounting for only 1% of ectopic mediastinal adenomas and 0.24% of all parathyroid adenomas. We present here a case of an MPA in the APW which was managed surgically and a brief review of the literature. Case description: A man in his 30s presented with low-backaches and recurrent renal calculi for 7 years, and also a history of fatigue, increased irritability, and insomnia. He was detected to have hypercalcemia, with inappropriately elevated parathyroid hormone (PTH), with background chronic kidney disease probably due to nephrocalcinosis. The ultrasound thyroid showed small subcentimetric doubtful parathyroid lesions in their ectopic locations. Tc99SestaMIBI scan detected an MPA, which on CT neck and thorax revealed an ~ 2.1 × 2 cm lesion in the APW. He underwent a left posterolateral thoracotomy with excision of APW parathyroid + cervical exploration and excision of left superior parathyroid and left inferior parathyroid biopsy. His PTH and calcium levels normalized postoperatively. Discussion: Aortopulmonary window parathyroid adenomas are very rare, usually located in the middle mediastinum, and are postulated to be supernumerary glands in 58–60%. The blood supply is derived from the mediastinum, from the bronchial arteries or internal mammary artery. The cervical approach is not feasible for adenomas in this location, and video-assisted thoracoscopic surgery (VATS)/thoracotomy is required. Conclusion: Structural imaging is paramount when MPAs are suspected, for appropriate surgical planning and management.

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REVIEW ARTICLE

Upander Kumar, Nancy Raja, Rambhit C Dwivedi, Ashwinee Rahalkar, Mithun Raam, Kul R Singh, Pooja Ramakant, Anand Mishra

Targeted Therapy in Thyroid Cancer

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:17] [Pages No:30 - 46]

Keywords: Advanced thyroid cancer, Personalized medicine, Radioiodine refractory thyroid cancer, Redifferentiating agents, Tyrosine kinase inhibitors

   DOI: 10.5005/jp-journals-10088-11236  |  Open Access |  How to cite  | 

Abstract

Personalized medicine for thyroid cancer (TC) involves tailoring treatment plans based on a patient's specific genetic makeup and tumor characteristics. This effective treatment strategy is ultimately improving patient outcomes and quality of life. Various tyrosine kinase inhibitors, serine/threonine kinase or B-Raf proto-oncogene (BRAF) and mitogen-activated protein kinase (MAPK/MEK) inhibitors, neurotrophic tyrosine receptor kinase (NTRK) fusion inhibitors, rearranged during transfection (RET) inhibitors, vascular endothelial growth factor (VEGFR) inhibitors, redifferentiating agents, and immunotherapies have shown promising results in the last decade since their introduction for treating thyroid cancer (ThyCa). These agents may be used for radioiodine refractory (RAIR) differentiated TC (DTC), disseminated, recurrent, residual anaplastic TC (ATC), and medullary TC (MTC). They hardly have any contraindications. The knowledge of their safety profile has expanded with more and more use. They have tolerable side effects and occasionally may require treatment discontinuation or switching to other agents. The armamentarium of available drugs and new targets is rapidly increasing with the results of clinical trials. Their use in pregnancy, the risk of hematological malignancy, geographical variations in their availability and cost, definitive guidelines, and managing drug resistance are current challenges. Advances in molecular diagnosis, artificial intelligence, and molecular scissors may completely revolutionize targeted therapy in the future.

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Journal Watch

Rinelle Mascarenhas, Gaurav Agarwal

Primary Hyperparathyroidism in Multiple Endocrine Neoplasia Type 1: Less than Subtotal Parathyroidectomy is Still Relevant

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:1] [Pages No:47 - 47]

Keywords: Less than subtotal parathyroidectomy, Multiple endocrine neoplasia type 1, Primary hyperparathyroidism, Recurrent primary hyperparathyroidism, Subtotal parathyroidectomy

   DOI: 10.5005/jp-journals-10088-11231  |  Open Access |  How to cite  | 

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Journal Watch

Rajni K Sah, Sabaretnam Mayilvaganan

Litigation after Thyroid Surgery

[Year:2024] [Month:January-June] [Volume:19] [Number:1] [Pages:1] [Pages No:48 - 48]

Keywords: Thyroid, Thyroid cancer, Thyroid surgery

   DOI: 10.5005/jp-journals-10088-11233  |  Open Access |  How to cite  | 

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