Case Reports in Endocrinology最新文献_第4页

DUOXA2 Variants as an Underrecognized Cause of Diffuse Goiter: A Euthyroid Adult Case Responsive to Levothyroxine. DUOXA2变异是弥漫性甲状腺肿的一个未被认识的原因：一个对左甲状腺素有反应的甲状腺功能正常的成人病例。

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Case Reports in Endocrinology Pub Date : 2026-05-29 eCollection Date: 2026-01-01 DOI: 10.1155/crie/9337816

Hanna Deguchi-Horiuchi, Shuji Fukata, Takahiko Kogai, Yuka Ito, Takashi Akamizu

{"title":"DUOXA2 Variants as an Underrecognized Cause of Diffuse Goiter: A Euthyroid Adult Case Responsive to Levothyroxine.","authors":"Hanna Deguchi-Horiuchi, Shuji Fukata, Takahiko Kogai, Yuka Ito, Takashi Akamizu","doi":"10.1155/crie/9337816","DOIUrl":"10.1155/crie/9337816","url":null,"abstract":"Hydrogen peroxide (H2O2) is an essential substrate for thyroid peroxidase (TPO) and plays a key role in iodine oxidation, organification, and coupling reactions that generate thyroid hormones. Dual oxidase 2 (DUOX2) and its maturation factor dual oxidase maturation factor 2 (DUOXA2) are critical for H2O2 production in thyroid follicular cells. Although DUOXA2 variants typically present with congenital hypothyroidism, phenotypic variability has been reported. We present an adult woman with a diffuse thyroid goiter and normal thyroid function who was found to have compound heterozygous DUOXA2 variants (p.Tyr138 ∗/p.Tyr246 ∗). Despite the patient being euthyroid, levothyroxine (LT4) administration led to a reduction in thyroid size. This case highlights that DUOXA2 variants may be present in euthyroid adults with thyroid goiter and that LT4 therapy can lead to a reduction in thyroid volume. Long-term follow-up may clarify the natural history and therapeutic response in euthyroid adults with DUOXA2 variants.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"9337816"},"PeriodicalIF":0.8,"publicationDate":"2026-05-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13239268/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148197543","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Triple Trouble: Early Clues in the Diagnosis of Allgrove Syndrome: A Case Report. 三重麻烦：诊断Allgrove综合征的早期线索：1例报告。

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Case Reports in Endocrinology Pub Date : 2026-05-16 eCollection Date: 2026-01-01 DOI: 10.1155/crie/3771629

Titilope Majiyagbe, Anitha Kumaran, Lawrence Gnanaraj, Claire Wood

{"title":"Triple Trouble: Early Clues in the Diagnosis of Allgrove Syndrome: A Case Report.","authors":"Titilope Majiyagbe, Anitha Kumaran, Lawrence Gnanaraj, Claire Wood","doi":"10.1155/crie/3771629","DOIUrl":"10.1155/crie/3771629","url":null,"abstract":"Introduction: Allgrove syndrome (also known as Triple A syndrome) is an inherited autosomal recessive condition, characterised by achalasia, alacrima and adrenal insufficiency. Its rarity and high phenotypic heterogeneity mean that it often poses a diagnostic challenge.Case presentation: We discuss the case of a previously well 5-year-old who presented with adrenal crisis. The R150 genetic panel showed a mutation in the achalasia-addisonianism-alacrima syndrome (AAAS) gene, confirming a diagnosis of Allgrove syndrome. It was noted in retrospect that his earliest presenting feature was alacrima, which was noted by parents in the neonatal period but not investigated. He was started on glucocorticoid replacement and is under regular paediatric endocrine outpatient follow-up.Conclusion: If our patient's alacrima had come to medical attention and Allgrove syndrome been diagnosed earlier, then the subsequent adrenal crisis could probably have been avoided, as regular monitoring of the adrenal axis would have been undertaken.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"3771629"},"PeriodicalIF":0.8,"publicationDate":"2026-05-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13179715/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147962211","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A Modern-Day Captain Charles Martell: Catastrophic Skeletal Manifestations of Primary Hyperparathyroidism. 现代查尔斯·马泰尔船长：原发性甲状旁腺功能亢进的灾难性骨骼表现。

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Case Reports in Endocrinology Pub Date : 2026-05-14 eCollection Date: 2026-01-01 DOI: 10.1155/crie/1352595

Rafyat Ara, Ahmad Alam, Dileep Raja Kuraku, Mohd Salman, Vijayakumar Karthik

{"title":"A Modern-Day Captain Charles Martell: Catastrophic Skeletal Manifestations of Primary Hyperparathyroidism.","authors":"Rafyat Ara, Ahmad Alam, Dileep Raja Kuraku, Mohd Salman, Vijayakumar Karthik","doi":"10.1155/crie/1352595","DOIUrl":"10.1155/crie/1352595","url":null,"abstract":"Primary hyperparathyroidism (PHPT) is characterised by autonomous overproduction of parathyroid hormone (PTH), resulting in hypercalcemia and multisystem complications. Although routine calcium screening has reduced advanced skeletal disease in many regions, severe manifestations such as osteitis fibrosa cystica (OFC) still occur in resource-limited settings. We report a case of a woman in her 30s who presented with profound disability after 4-5 years of progressive bone pain, fatigue, proximal myopathy and eventual loss of independent ambulation, requiring her to drag herself on the ground. Imaging revealed generalised osteopenia, subperiosteal bone resorption, a 'salt and pepper' appearance of the skull, codfish vertebrae, anterior bowing of long bones and multiple lytic lesions consistent with advanced OFC. Laboratory investigations revealed marked hypercalcemia (14.3 mg/dL), hypophosphataemia (1.49 mg/dL), elevated alkaline phosphatase (781 U/L) and elevated intact PTH (iPTH) (1110 pg/mL), confirming PTH-dependent hypercalcemia. A 4D-CT scan localised a left inferior parathyroid adenoma (LIPA), and a renal ultrasound showed bilateral nephrocalcinosis. Following preoperative stabilisation with saline diuresis and intravenous zoledronic acid, she underwent minimally invasive parathyroidectomy (MIP) with an immediate postoperative fall in PTH, indicating biochemical cure. However, she developed hungry bone syndrome (HBS), requiring intensive intravenous and oral calcium supplementation along with active vitamin D therapy. This case highlights the catastrophic skeletal consequences of delayed PHPT diagnosis. It underscores the importance of early recognition of metabolic bone disease to prevent irreversible disability and systemic complications, particularly in low-resource settings.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"1352595"},"PeriodicalIF":0.8,"publicationDate":"2026-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13175074/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147962160","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Severe Transient Central Diabetes Insipidus After Pituitary Adenoma Removal With Peak Urine Output of 33.5 L in 24 h. 垂体腺瘤切除后出现严重一过性中枢性尿崩症，24小时尿量峰值33.5 L。

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Case Reports in Endocrinology Pub Date : 2026-05-07 eCollection Date: 2026-01-01 DOI: 10.1155/crie/7516452

Govinda Bhandari, Anupam Katuwal Chhetri, Rajan Gyawali, Durlab Thapa

{"title":"Severe Transient Central Diabetes Insipidus After Pituitary Adenoma Removal With Peak Urine Output of 33.5 L in 24 h.","authors":"Govinda Bhandari, Anupam Katuwal Chhetri, Rajan Gyawali, Durlab Thapa","doi":"10.1155/crie/7516452","DOIUrl":"10.1155/crie/7516452","url":null,"abstract":"Background: Postoperative central diabetes insipidus (CDI) is a well-documented but often transient complication following transsphenoidal surgery (TSS) for pituitary adenomas. CDI is characterized by excessive urine output due to impaired antidiuretic hormone (ADH) secretion following damage to the neurohypophyseal stalk or hypothalamus. While most cases resolve within a few days, severe cases with extremely high urine output are rare. This case is notable due to an exceptionally high urine output of 33.5 L in a single day, far exceeding typical postoperative presentations and highlighting the need for vigilant postoperative monitoring.Case presentation: A 47-year-old female with hypertension and hypothyroidism underwent endoscopic endonasal TSS at Tribhuvan University Teaching Hospital (TUTH) for a pituitary macroadenoma compressing the optic chiasm. Within hours postoperatively, she developed significant polyuria exceeding 1000 mL/h, with urine osmolality of 213.3 mOsm/kg and hypernatremia (serum sodium 149 mmol/L), consistent with CDI. Urine output increased to 30.8 L on postoperative day 2 and peaked at 33.5 L on day 3, requiring aggressive fluid replacement and desmopressin therapy. Despite escalating doses of desmopressin, polyuria persisted before gradually resolving by postoperative day 5. Careful monitoring of electrolytes and stepwise adjustment of desmopressin led to stabilization, confirming transient rather than permanent CDI.Conclusion: This case highlights that postoperative CDI can present with extreme polyuria and rapid shifts in sodium and osmolality, making early recognition essential. It reinforces that individualized desmopressin titration and close fluid-electrolyte monitoring are critical to prevent complications and support full recovery, especially when urine output reaches unusually high levels.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"7516452"},"PeriodicalIF":0.8,"publicationDate":"2026-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13150337/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147866908","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Primary Aldosteronism Presenting as Dropped Head Syndrome With Hypokalemic Rhabdomyolysis: A Case Report. 原发性醛固酮增多症表现为低钾性横纹肌溶解伴头下垂综合征1例报告。

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Case Reports in Endocrinology Pub Date : 2026-04-29 eCollection Date: 2026-01-01 DOI: 10.1155/crie/6424516

Ya-Chen Kao, Yu-Cheng Liang, Ye-Fong Du, Horng-Yih Ou

{"title":"Primary Aldosteronism Presenting as Dropped Head Syndrome With Hypokalemic Rhabdomyolysis: A Case Report.","authors":"Ya-Chen Kao, Yu-Cheng Liang, Ye-Fong Du, Horng-Yih Ou","doi":"10.1155/crie/6424516","DOIUrl":"10.1155/crie/6424516","url":null,"abstract":"Background: Primary aldosteronism (PA) is a common cause of secondary hypertension and hypokalemia, but it rarely presents with neuromuscular symptoms. Although only a few cases of hypokalemia-induced rhabdomyolysis have been reported, dropped head syndrome (DHS) due to cervical axial muscle weakness has not previously been described in the context of PA.Case presentation: A 65-year-old woman with a history of hypertension presented with progressive posterior neck weakness, followed by proximal limb soreness and weakness. Cervical spine disease was initially suspected, but CT imaging revealed only degenerative changes without cord compression. Laboratory tests showed severe hypokalemia (1.5 mmol/L), metabolic alkalosis, and markedly elevated creatine kinase and myoglobin levels, consistent with rhabdomyolysis. Her symptoms resolved after potassium supplementation. Endocrine workup revealed an elevated aldosterone-to-renin ratio (ARR) and a positive captopril suppression test. CT and NP-59 SPECT/CT identified a left adrenal functioning adenoma. Laparoscopic adrenalectomy confirmed an aldosterone-producing adrenal cortical adenoma. Postoperatively, the patient achieved complete biochemical remission with normalized potassium levels, improved blood pressure, and no recurrence of muscle weakness or neck symptoms.Conclusion: This case highlights a rare presentation of PA as hypokalemia-induced rhabdomyolysis and DHS mimicking cervical spine pathology. In patients with unexplained focal muscle weakness and persistent hypokalemia, particularly in the setting of hypertension, PA should be considered. Clinicians should maintain a high index of suspicion and pursue comprehensive diagnostic evaluations. Early diagnosis and definitive treatment can prevent long-term complications and enable full metabolic recovery.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"6424516"},"PeriodicalIF":0.8,"publicationDate":"2026-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13126241/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147811322","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Congenital Adrenal Hyperplasia due to 11β-Hydroxylase Deficiency Presented With Leydig Cell Tumor and Testicular Adrenal Rest Tumors: A Case Report. 11β-羟化酶缺乏所致先天性肾上腺增生伴间质细胞瘤和睾丸肾上腺休息瘤1例。

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Case Reports in Endocrinology Pub Date : 2026-04-27 eCollection Date: 2026-01-01 DOI: 10.1155/crie/3675251

Shervin Mossavarali, Faezeh Sehatpour, Shahrzad Mohseni, Mahnaz Pejman Sani

{"title":"Congenital Adrenal Hyperplasia due to 11β-Hydroxylase Deficiency Presented With Leydig Cell Tumor and Testicular Adrenal Rest Tumors: A Case Report.","authors":"Shervin Mossavarali, Faezeh Sehatpour, Shahrzad Mohseni, Mahnaz Pejman Sani","doi":"10.1155/crie/3675251","DOIUrl":"10.1155/crie/3675251","url":null,"abstract":"Congenital adrenal hyperplasia (CAH) due to 11β-hydroxylase deficiency is an uncommon disorder characterized by impaired cortisol synthesis, hyperandrogenism, and mineralocorticoid excess. The coexistence of Leydig cell tumors (LCTs) and testicular adrenal rest tumors (TARTs) is rarely reported, highlighting the diagnostic and therapeutic challenges in such cases. A 35-year-old man with a history of hypertension and infertility presented with left testicular masses. His medical history was significant for a prior right orchiectomy, with pathology confirming LCT. Subsequent evaluations revealed azoospermia, elevated adrenal androgen levels, adrenocorticotropic hormone (ACTH) and 17-OH progesterone levels indicative of CAH due to 11β-hydroxylase deficiency. Imaging studies identified left testicular masses and bilateral adrenal myelolipomas. The patient was managed with oral dexamethasone and eplerenone, resulting in normalization of blood pressure and electrolytes. This case highlights the complexity of CAH presentations with overlapping testicular and adrenal pathologies. Patients with such conditions should be closely monitored and regularly checked for common complications to ensure timely intervention and optimal management.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"3675251"},"PeriodicalIF":0.8,"publicationDate":"2026-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13115910/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147763416","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Acute Kidney Injury and Nephrotic-Range Proteinuria as Initial Presentation of Pheochromocytoma: A Case Report. 急性肾损伤和肾范围蛋白尿作为嗜铬细胞瘤的初始表现：1例报告。

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Case Reports in Endocrinology Pub Date : 2026-04-26 eCollection Date: 2026-01-01 DOI: 10.1155/crie/8510972

Anum Rizwan, Sajid Islam Bhatti, Huda Raja, Sidra German, Tajammul Waqar, Abdullah Nadeem, Nahid Raufi

{"title":"Acute Kidney Injury and Nephrotic-Range Proteinuria as Initial Presentation of Pheochromocytoma: A Case Report.","authors":"Anum Rizwan, Sajid Islam Bhatti, Huda Raja, Sidra German, Tajammul Waqar, Abdullah Nadeem, Nahid Raufi","doi":"10.1155/crie/8510972","DOIUrl":"10.1155/crie/8510972","url":null,"abstract":"Introduction: Pheochromocytomas are rare catecholamine-producing neuroendocrine tumors that present with the classical triad of paroxysmal hypertension, palpitations, and sweating; however, this presentation is seen in less than 1/4 of pheochromocytoma patients. Hypertension is usually the predominant manifestation. Atypical manifestations, including renal involvement, are infrequent and may delay diagnosis.Case presentation: We report the case of a 37-year-old male with no prior comorbidities who presented with progressive abdominal pain, shortness of breath, and oliguria. Laboratory evaluation demonstrated acute kidney injury with nephrotic-range proteinuria, initially raising suspicion of a primary glomerular disorder. Chest imaging revealed pulmonary congestion, while abdominal imaging identified a right adrenal mass. Plasma-free metanephrine and normetanephrine levels were markedly elevated, confirming pheochromocytoma. The patient underwent preoperative optimization with alpha-adrenergic blockade and supportive measures, including temporary hemodialysis. A right adrenalectomy was subsequently performed, leading to complete resolution of renal dysfunction and normalization of blood pressure.Conclusion: This case emphasizes the diagnostic challenge of pheochromocytoma when presenting with renal complications. Clinicians should consider adrenal tumors in the differential diagnosis of patients with unexplained acute kidney injury, as timely recognition and surgical intervention can result in full recovery.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"8510972"},"PeriodicalIF":0.8,"publicationDate":"2026-04-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13110390/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147763370","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Denys-Drash Syndrome by WT1 Gene: Clinical Variability and Management Challenges in Two Saudi Infants. 由WT1基因引起的Denys-Drash综合征：两个沙特婴儿的临床变异性和管理挑战。

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Case Reports in Endocrinology Pub Date : 2026-04-24 eCollection Date: 2026-01-01 DOI: 10.1155/crie/8875066

Waleed Al-Amoudi, Raghad Alhuthil, Abdulrahman Alnwiji, Megren S Alqarni, Sarah Murad, Afaf Alsagheir

{"title":"Denys-Drash Syndrome by WT1 Gene: Clinical Variability and Management Challenges in Two Saudi Infants.","authors":"Waleed Al-Amoudi, Raghad Alhuthil, Abdulrahman Alnwiji, Megren S Alqarni, Sarah Murad, Afaf Alsagheir","doi":"10.1155/crie/8875066","DOIUrl":"10.1155/crie/8875066","url":null,"abstract":"Background: Denys-Drash syndrome (DDS) is a rare genetic disorder characterized by mutations in the Wilms tumor suppressor gene (WT1), leading to a triad of conditions including nephrotic syndrome progressing to end-stage renal disease (ESRD), Wilms tumor, and ambiguous genitalia. We present two pediatric cases illustrating the complexity and variability of DDS.Case presentation: The first case is a 7-month-old male presenting with ambiguous genitalia, nephropathy, and intussusception. Genetic analysis identified a likely pathogenic heterozygous WT1 variant: c.1384C >T (p.Gln462Ter). Despite surgical interventions, treatment was delayed due to COVID-19 restrictions, and the patient unfortunately passed away at 16 months during the lockdown period. The second case involves an 8-month-old female with normal external genitalia, a horseshoe kidney, bilateral renal masses, and recurrent hypotensive episodes. Genetic testing revealed a pathogenic heterozygous WT1 variant: c.453G >A (p.Trp151Ter). She was diagnosed with DDS-associated Wilms tumor and, despite aggressive management, passed away at 21 months.Conclusion: These cases underscore the importance of early diagnosis, multidisciplinary management, and personalized therapeutic approaches in DDS patients. Bilateral nephrectomy, renal transplantation, and monitoring for Wilms tumor are pivotal in improving prognosis, though variability in clinical presentations often complicates decision-making.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"8875066"},"PeriodicalIF":0.8,"publicationDate":"2026-04-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13108425/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147763440","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Partial Horner's Syndrome Following Thyroidectomy Without Lateral Neck Dissection: A Rare Case Report and Literature Review. 甲状腺切除术后无侧颈清扫的部分霍纳综合征：罕见病例报告及文献复习。

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Case Reports in Endocrinology Pub Date : 2026-04-23 eCollection Date: 2026-01-01 DOI: 10.1155/crie/1253094

Eve Hopping, Femi E Ayeni, Senarath Edirimanne

{"title":"Partial Horner's Syndrome Following Thyroidectomy Without Lateral Neck Dissection: A Rare Case Report and Literature Review.","authors":"Eve Hopping, Femi E Ayeni, Senarath Edirimanne","doi":"10.1155/crie/1253094","DOIUrl":"10.1155/crie/1253094","url":null,"abstract":"Horner's syndrome is a rare complication of thyroid surgery and is most commonly reported as a complication associated with lateral neck dissection. We present the case of a 46-year-old woman who developed a partial Horner's syndrome following hemithyroidectomy and central neck dissection, without lateral neck dissection. The patient underwent right hemithyroidectomy and central neck dissection for a TIRADS-5 nodule and developed right sided facial anhidrosis and absence of right-sided facial erythema on exertion 6 weeks post-operatively. No ocular involvement was detected. Neurologist review confirmed partial Horner's syndrome, which eventually resolved. The literature regarding development of Horner's syndrome as a complication of thyroid surgery without lateral neck dissection was reviewed via PubMed. All English language publications of adult patients developing features of Horner's syndrome after open thyroid surgery (hemithyroidectomy or thyroidectomy, +/- central neck dissection) were included. Twenty one cases of Horner's syndrome following thyroid surgery without lateral neck dissection were identified. Interestingly, only four of these cases featured anhidrosis, and no case reported asymmetrical facial erythema. Four possible mechanisms of cervical sympathetic chain dysfunction resulting in Horner's syndrome post-thyroidectomy have been proposed: neuropraxia from retraction; mass effect from post-operative haematoma; ischaemia-induced nerve damage; damage to communicating fibres during repeated inspection of the recurrent laryngeal nerve. To our knowledge, this is the only reported case of a post-thyroidectomy partial Horner's syndrome with associated asymmetrical flushing.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"1253094"},"PeriodicalIF":0.8,"publicationDate":"2026-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13106947/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147763448","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

A Rare Primary Adrenal Malignancy Manifesting as a Hemorrhagic Mass: Case Report and Literature Review. 一例罕见的原发性肾上腺恶性肿瘤表现为出血性肿块：病例报告及文献复习。

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Case Reports in Endocrinology Pub Date : 2026-04-08 eCollection Date: 2026-01-01 DOI: 10.1155/crie/1260707

Salem Al Ghaithi, Ibrahim Alali, Ali Al Reesi, Ali Almadhani

{"title":"A Rare Primary Adrenal Malignancy Manifesting as a Hemorrhagic Mass: Case Report and Literature Review.","authors":"Salem Al Ghaithi, Ibrahim Alali, Ali Al Reesi, Ali Almadhani","doi":"10.1155/crie/1260707","DOIUrl":"10.1155/crie/1260707","url":null,"abstract":"Primary adrenal extranodal natural killer/T-cell lymphoma (ENKTL) is an exceptionally rare, aggressive malignancy with fewer than 10 cases worldwide. We describe a 55-year-old man with diabetes mellitus and benign prostatic hyperplasia who presented with progressive left-sided abdominal pain and a 17-kg unintentional weight loss over 4 months. Imaging showed a 4.9-cm left adrenal mass with hemorrhagic features, initially presumed to be spontaneous adrenal hemorrhage. Endocrine evaluation excluded functional adrenal tumors, and surgical management required conversion from laparoscopic adrenalectomy to open resection due to superior mesenteric vein (SMV) involvement. Histopathological and immunophenotypic confirmed non-nasal ENKTL (CD3+, CD56+, granzyme B+, perforin+, and Epstein-Barr virus-encoded RNA [EBER]+). Postoperative 18F-FDG PET/computed tomography (CT) demonstrated residual disease; steroid, methotrexate, ifosfamide, L-asparaginase, and etoposide (SMILE) chemotherapy achieved a marked complete metabolic response after six cycles-one of the most significant documented responses in primary adrenal NK/T-cell lymphoma. This case highlights diagnostic challenges of primary adrenal lymphoma (PAL), the need for histopathological confirmation in atypical masses, and suspicion for hematological malignancies with systemic symptoms and adrenal lesions.","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":"2026 ","pages":"1260707"},"PeriodicalIF":0.8,"publicationDate":"2026-04-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13058563/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147644004","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0