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Co-existence of two different types of pancreatic neuroendocrine tumors in a patient with multiple endocrine neoplasia type-1 多发性1型内分泌瘤患者两种不同类型胰腺神经内分泌肿瘤共存
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/j.jecr.2021.100088
B. Ozbas , Z. Karaca , I. Okcesiz , I. Karahan , N. Topaloglu , U. Abdulrezzak , F. Ozturk , K. Unluhizarci

Pancreatic neuroendocrine tumors (PNETs) are rare forms of pancreatic malignancies which are classified according to functionality. Coexistence of two functional PNETs is extremely rare, but case reports are encountered in the literature. We present a 23-year-old male patient who was referred for recurrent hypoglycemic symptoms while investigating for pituitary adenoma. In addition to overt hypoglycemia, the patient's laboratory examination was compatible with primary hyperparathyroidism, hyperprolactinemia and acromegaly. Prolonged fasting test revealed hypoglycemia due to endogenous hyperinsulinism. Multiple hypodense mass lesions in the tail and body of pancreas were detected on the computed tomography scan. After surgical excision of these lesions, histopathologic examination of the specimens showed a grade 1 NET, which was positive for glucagon and chromogranin, negative for insulin. Histopathologic and immunostaining evaluation suggested that there were two pancreatic NETs with different characteristics. The patient was diagnosed as multiple endocrine neoplasia type-1 (MEN-1) syndrome due to clinical coexistence of the all 3 components, family history and genetic confirmation (frameshift mutation in MEN1 gene due to pathogenic deletion). After the operation, hypoglycemic symptoms were ameliorated. After 9 years of lost to follow-up period, the patient admitted to the emergency Department with complaints of nausea, vomiting and abdominal pain. In the evaluation of patient, recurrences of hyperparathyroidism, pituitary adenoma and PNET were observed. Coexistence of insulinoma and glucagonoma is a rare condition and the underlying physiopathological mechanisms have not been fully understood. However, it is important to note that two simultaneous functional tumors may coexist in PNETs associated with MEN-1 syndrome.

胰腺神经内分泌肿瘤(PNETs)是一种罕见的胰腺恶性肿瘤,根据功能进行分类。两个功能性PNETs共存是极其罕见的,但在文献中遇到的病例报告。我们报告一位23岁的男性病人,他在检查垂体腺瘤时因反复出现低血糖症状而被转诊。除了明显的低血糖,患者的实验室检查符合原发性甲状旁腺功能亢进、高催乳素血症和肢端肥大症。延长空腹试验显示内源性高胰岛素血症引起低血糖。计算机断层扫描发现胰腺尾部及体部多发低密度肿块。手术切除这些病变后,标本的组织病理学检查显示为1级NET,胰高血糖素和嗜铬粒蛋白阳性,胰岛素阴性。组织病理学和免疫染色评价提示存在两种不同特征的胰腺NETs。经3种成分临床共存、家族史及基因证实(致病性缺失导致MEN1基因移码突变),诊断为多发性内分泌肿瘤1型(MEN1)综合征。术后低血糖症状得到改善。失访期9年后,患者以恶心、呕吐、腹痛等主诉入住急诊科。观察患者甲状旁腺功能亢进、垂体腺瘤、PNET的复发情况。胰岛素瘤和胰高血糖素瘤共存是一种罕见的情况,其潜在的生理病理机制尚未完全了解。然而,值得注意的是,与men1综合征相关的PNETs中可能同时存在两种功能性肿瘤。
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引用次数: 1
Primary hyperparathyroidism presenting with acute pancreatitis, complicated by raised IgG-4 and positive Covid-19 IgG antibody 原发性甲状旁腺功能亢进伴急性胰腺炎,并发IgG-4升高和新冠肺炎IgG抗体阳性
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/j.jecr.2021.100086
Taimoor Hussain , Farukhzad Hafizyar , Abdul Habib Eimal Latif , Muzhda Shafi Mujadidi , Khalida Walizada , Zahra Mushtaq , Jasvindar Kumar

Background

Gall stone and alcohol are the two most common causes of pancreatitis. Other etiologies include trauma, post endoscopic retrograde cholangiopancreatography (ERCP), bacterial, viral, fungal, and parasitic infections, hypertriglyceridemia, medications, and scorpion bite to name a few. Hypercalcemia is a rare cause of acute pancreatitis. Hypercalcemia can be due to primary hyperparathyroidism (PHPT) manifested by raised parathyroid hormone and serum calcium level. In addition acute pancreatitis is a rare presenting complain of PHPT. PHPT can be asymptomatic or present with symptoms of bone pain, kidney stones, nephrocalcinosis, renal failure, peptic ulcer disease, accelerated atherosclerotic heart disease, hypertension and proximal muscle weakness. Osteitis fibrosa cystica is the term for radiologically visible characteristic feature of skeletal involvement.

Case presentation

We report a case of 20 years old male with chief complaint of epigastric and left hypochondrium pain associated with vomiting for two months. Abdominal pain was gradual in onset, radiating to back, and progressive. He also had complains of constipation, blood tinged feces and pain during defecation. Initial lab tests were significant for increased amylase, lipase and a diagnosis of acute pancreatitis was made which was confirmed by CT scan. After ruling out the common etiologies of acute pancreatitis, further investigations for uncommon etiologies revealed increased IgG-4 values suggesting autoimmune pancreatitis. However further investigations were done and it did not meet the diagnostic criteria for autoimmune pancreatitis. Covid-19 IgG antibodies were positive, however patient did not have any other symptoms of Covid-19 and PCR Covid-19 test was negative. Although Covid-19 can cause pancreatitis, given the ongoing pandemic, he might have been asymptomatically infected with Covid-19 and developed immunity. Therefore we did not believe Covid-19 to be the cause of his pancreatitis. Thus a search for other rare etiologies was initiated which revealed serum calcium of 15 mg/dL. Serum parathyroid hormone and urinary calcium were increased several times suggesting primary hyperparathyroidism. DEXA scan revealed osteoporosis.

Conclusion

Hypercalcemia is one of the rare etiologies of pancreatitis. Thus it should be considered as one of the etiologies of acute pancreatitis after ruling out the more common ones. Secondly, this Case report highlights acute pancreatitis as one of the rare presenting complain of primary hyperparathyroidism which may be associated with several times increased IgG-4 values. Covid-19 has been associated with a number of presenting complains and can cause acute pancreatitis, amid the ongoing pandemic physicians should exercise caution to avoid spurious association of Covid-19 with acute pancreatitis or other presenting complains.

背景胆结石和酒精是胰腺炎最常见的两个原因。其他病因包括创伤、内镜下逆行胰胆管造影术(ERCP)、细菌、病毒、真菌和寄生虫感染、高甘油三酯血症、药物和蝎子咬伤等。高钙血症是急性胰腺炎的罕见病因。高钙血症可能是由于原发性甲状旁腺功能亢进(PHPT),表现为甲状旁腺激素和血清钙水平升高。此外,急性胰腺炎是一种罕见的PHPT症状。PHPT可能无症状或表现为骨痛、肾结石、肾钙沉着症、肾衰竭、消化性溃疡、加速动脉粥样硬化性心脏病、高血压和近端肌肉无力。纤维性膀胱炎是指骨骼受累的放射学可见特征。病例介绍:我们报告一例20岁男性,主要主诉上腹部和左疑病症疼痛伴呕吐两个月。腹痛开始时是渐进的,向背部放射,并进行性疼痛。他还抱怨便秘、粪便带血和排便时疼痛。最初的实验室测试对淀粉酶和脂肪酶的增加具有重要意义,并通过CT扫描确诊为急性胰腺炎。在排除了急性胰腺炎的常见病因后,对不常见病因的进一步研究显示IgG-4值增加,提示自身免疫性胰腺炎。然而,进行了进一步的研究,它不符合自身免疫性胰腺炎的诊断标准。新冠肺炎IgG抗体呈阳性,但患者没有任何其他新冠肺炎症状,PCR新冠肺炎检测呈阴性。尽管新冠肺炎可能会导致胰腺炎,但鉴于疫情持续,他可能已无症状感染新冠肺炎并产生免疫力。因此,我们不认为新冠肺炎是他胰腺炎的原因。因此,开始寻找其他罕见的病因,其显示血清钙为15mg/dL。血清甲状旁腺激素和尿钙增加数倍,提示原发性甲状旁腺功能亢进。DEXA扫描显示骨质疏松。结论高钙血症是胰腺炎的罕见病因之一。因此,在排除更常见的病因后,应将其视为急性胰腺炎的病因之一。其次,本病例报告强调急性胰腺炎是罕见的原发性甲状旁腺功能亢进的主诉之一,可能与IgG-4值增加数倍有关。新冠肺炎与许多出现的投诉有关,并可能导致急性胰腺炎,在持续的大流行中,医生应谨慎行事,避免将新冠肺炎与急性胰腺炎或其他出现的投诉虚假关联。
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引用次数: 2
Primary hyperparathyroidism presenting with acute pancreatitis, complicated by raised IgG-4 and positive Covid-19 IgG antibody 原发性甲状旁腺功能亢进,表现为急性胰腺炎,并伴有IgG-4升高和Covid-19 IgG抗体阳性
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/j.jecr.2021.100086
Taimoor Hussain, Farukhzad Hafizyar, Abdul Habib Eimal Latif, Muzhda Shafi Mujadidi, Khalida Walizada, Zahra Mushtaq, J. Kumar
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引用次数: 2
Pituitary macroadenoma: A case of balanced deficiencies 垂体大腺瘤:一例平衡缺陷
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/j.jecr.2021.100091
Robert D. Leimbach , Mohamed K.M. Shakir , Thanh D. Hoang

Introduction

We present a patient with a non-functional pituitary macroadenoma who developed diabetes insipidus after initiation of thyroid hormone and cortisol replacement for central hypothyroidism and adrenal insufficiency.

Case description

A 61-year-old man with a history of a 1.7cm non-functional pituitary macroadenoma that was stable over a five-year period was found to have an interval growth of 2.1cm on annual follow-up. He was in a good state of health, but laboratory findings revealed central hypothyroidism and central adrenal insufficiency. He was started on glucocorticoid and thyroid hormone replacement with subsequent development of diabetes insipidus. A follow-up MRI demonstrated apoplexy with decrease in tumor size. Pituitary surgery was cancelled.

Conclusion

Subclinical apoplexy may occur in a pituitary macroadenoma with pituitary hormone deficiency as the only indication of an occult process. Treating underlying glucocorticoid or thyroid deficiency may unmask diabetes insipidus. Alternative diagnosis should be investigated in a patient with a pituitary macroadenoma who suddenly develops diabetes insipidus.

我们报告一例无功能垂体大腺瘤患者,因中枢性甲状腺功能减退和肾上腺功能不全而开始甲状腺激素和皮质醇替代治疗后发生尿囊症。病例描述1例61岁男性,既往有1.7cm无功能垂体大腺瘤病史,5年稳定,每年随访发现间断性生长2.1cm。他的健康状况良好,但实验室结果显示中枢性甲状腺功能减退和中枢性肾上腺功能不全。他开始使用糖皮质激素和甲状腺激素替代,随后发展为尿崩症。后续MRI显示中风伴肿瘤缩小。垂体手术取消。结论垂体大腺瘤有可能发生亚临床卒中,且垂体激素缺乏是其隐匿过程的唯一指征。治疗潜在的糖皮质激素或甲状腺缺乏可能揭开尿崩症的面纱。对于突然发生尿崩症的垂体大腺瘤患者,应考虑其他诊断。
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引用次数: 0
Precocious puberty in a healthy 2 years old girl: Ovarian Sertoli-Leydig cells cancer 健康2岁女孩性早熟:卵巢Sertoli-Leydig细胞癌症
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/j.jecr.2021.100090
Jennifer Monroy , Carolina Jaramillo , Sasha González

Precocious puberty refers to appearance of secondary sexual characteristics, increased growth rate and reproductive capacity before 8 years in girls, and 9 years in boys. The etiology can be diverse, either central activation of hypothalamus-pituitary gonadal axis or by endogenous production or exogenous administration of sex steroids. Peripheral precocious puberty is less common and Leydig-Sertoli cell tumors comprise 1–2% of the pediatric ovarian malignancies. (1) These tumors commonly present in the second or third decade of life but can present as early as 2-years of age.(2).

We describe a case of precocious puberty caused by ovarian Sertoli-Leydig cancer in a young girl with no pre-existing medical issues. The 2-year-old patient was referred from Saint Martin to the Pediatric endocrinology for evaluation of pubic hair and breast development which started 3-month prior to presentation. On exam, the patient was noted to have abdominal prominence which prompted obtaining abdominal imaging which showed an ovarian tumor which was subsequently removed.

早熟是指女孩在8岁前出现第二性征,生长速度和生殖能力增加,男孩在9岁前出现。病因可能多种多样,要么是下丘脑-垂体-性腺轴的中枢激活,要么是内源性产生或外源性给予性类固醇。外周性早熟不太常见,Leydig支持细胞肿瘤占儿童卵巢恶性肿瘤的1-2%。(1) 这些肿瘤通常出现在生命的第二个或第三个十年,但最早可以出现在2岁。(2) 我们描述了一例由卵巢癌症引起的性早熟病例,该病例发生在一名没有预先存在的医学问题的年轻女孩身上。这名2岁的患者从圣马丁转诊到儿科内分泌科,在就诊前3个月开始评估阴毛和乳房发育。在检查中,发现患者腹部突出,这促使获得腹部成像,显示卵巢肿瘤,随后切除。
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引用次数: 0
Neuroglycopaenia presenting as isolated aphasia 神经性糖少症表现为孤立性失语
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/j.jecr.2021.100089
Zhemin Wang , Eng Loon Tng

Aphasia is an uncommon presentation of neuroglycopaenia. We herein report a case of a 55-year-old man with type 2 diabetes mellitus with neuroglycopaenia presenting with speech difficulties that recovered over an unusually prolonged period. The clinical course and laboratory results are presented. The patient's symptoms persisted for 48 hours despite correction of hypoglycaemia but he made significant recovery. This case highlights an unusual presentation of neuroglycopaenia manifesting mainly with extended speech deficits. The delayed recovery despite correction of hypoglycaemia in this patient shows that Whipple's Triad may not be reliably applied in patients treated with long-acting insulin secretagogues or those with neuronal vulnerability to hypoglycaemia.

失语是神经性糖少症的罕见表现。我们在此报告一例55岁男性2型糖尿病伴神经性糖少症,表现为言语困难,恢复异常长时间。介绍了临床过程和实验室结果。患者的症状持续了48小时,尽管纠正了低血糖,但他明显恢复。本病例表现为神经性糖减少症,主要表现为扩展性语言缺陷。尽管该患者的低血糖得到了纠正,但恢复缓慢,这表明惠普尔三联征可能并不可靠地适用于长效胰岛素分泌剂治疗的患者或低血糖神经易感性患者。
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引用次数: 0
Precocious puberty in a healthy 2 years old girl: Ovarian Sertoli-Leydig cells cancer 2岁健康女童性早熟:卵巢上皮间质细胞癌
Q4 Medicine Pub Date : 2021-09-01 DOI: 10.1016/J.JECR.2021.100090
J. Monroy, C. Jaramillo, Sasha González
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引用次数: 0
Management of the patient with incidental bilateral adrenal nodules 偶发双侧肾上腺结节患者的处理
Q4 Medicine Pub Date : 2021-06-01 DOI: 10.1016/j.jecr.2021.100082
Ann T. Sweeney , Padmavathi Srivoleti , Michael A. Blake

Bilateral incidental adrenal nodules represent 10–23% of all incidental adrenal nodules. The general approach to these nodules follows the same premise as for unilateral incidental adrenal nodules however there are features unique to bilateral nodules including the differential diagnosis, the diagnostic approach as well as the management. The majority (75%) of bilateral truly incidental adrenal nodules are benign nonfunctioning adenomas however bilateral lesions are more likely to display hormone excess than unilateral lesions with subclinical Cushing's being the most prevalent abnormality followed by Cushing's syndrome, Hyperaldosteronism as well as Congenital Adrenal Hyperplasia (CAH). Though bilateral pheochromocytomas are less common, a patient presenting with bilateral pheochromocytomas will usually have a germline pathogenic variant and be syndromic. The overall diagnostic approach to bilateral nodules involves determining based on the patients' clinical history and examination as well as the imaging phenotype of each lesion whether the lesions could represent a malignancy, exhibit hormonal excess and whether they could represent a familial syndrome. In patients with bilateral infiltrative lesions, adrenal insufficiency needs to be excluded. In patients with bilateral myelolipomas, screening for CAH should be considered. We present an illustrative case of a patient with incidental bilateral nodules and review the systematic approach to the patient including the key radiographic diagnostic features of adrenal nodules (unenhanced CT attenuation, adrenal washout and chemical shift MRI) to best determine the etiology of these lesions. An overview of the management of patients with such bilateral adrenal nodules is provided with a particular focus on pheochromocytomas.

双侧偶发肾上腺结节占所有偶发肾上腺结节的10-23%。这些结节的一般方法遵循与单侧偶发肾上腺结节相同的前提,但双侧结节有其独特的特征,包括鉴别诊断、诊断方法和治疗。大多数(75%)双侧偶发肾上腺结节是良性无功能腺瘤,但双侧病变比单侧病变更容易表现为激素过量,亚临床库欣综合征是最常见的异常,其次是库欣综合征、醛固酮增多症和先天性肾上腺增生症(CAH)。虽然双侧嗜铬细胞瘤不太常见,但双侧嗜铬细胞瘤患者通常有种系致病变异和综合征。双侧结节的整体诊断方法包括根据患者的临床病史和检查以及每个病变的影像学表型来确定病变是否代表恶性肿瘤,是否表现为激素过量,是否代表家族综合征。对于双侧浸润性病变的患者,应排除肾上腺功能不全。对于双侧骨髓瘤患者,应考虑筛查CAH。我们提出一例偶发双侧结节的病例,并回顾了该患者的系统方法,包括肾上腺结节的关键影像学诊断特征(未增强CT衰减,肾上腺冲掉和MRI化学移位),以最好地确定这些病变的病因。这类双侧肾上腺结节患者的管理概述提供了特别关注嗜铬细胞瘤。
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引用次数: 3
Papillary thyroid cancer with unusual late onset pituitary and orbital choroidal metastases. Case report and literature review 甲状腺乳头状癌伴不寻常的晚发性垂体及眼眶脉络膜转移。病例报告及文献复习
Q4 Medicine Pub Date : 2021-06-01 DOI: 10.1016/j.jecr.2021.100081
Nada Bin Hareez , Mohammad Jay , Heather Lochnan

Papillary thyroid cancer is the most common type of differentiated thyroid cancer (DTC) and in most cases, disease is localized to the neck and remission of disease is common. Though distant metastasis of DTC is unusual; involved sites are more often the lungs and bones, while less common sites include the liver and kidney. No case of metastasis to both intraocular choroid and the pituitary gland has previously been reported. We describe a case of a 58-year-old man who presented with multifocal papillary thyroid carcinoma (PTC) with cervical node metastasis. According to risk stratifications in DTC, the patient stratified as low risk of recurrence. Five years after initial presentation, he was found to have mediastinal lymph node metastasis. Fifteen years post diagnosis, his PTC metastasized to his liver, bone, lung, pituitary gland, orbital choroid and choroid plexus. Remarkably, the only symptom the patient developed as a result of this metastasis was alteration in his vision, which was treated with bevacizumab. Despite initial improvement, he developed ptosis and third nerve symptoms. His pituitary metastasis was treated with Cyber-Knife Surgery and his systemic metastasis was treated with the tyrosine kinase inhibitor (TKI) lenvantinib, resulting in new improvement of his vision. Lenvantinib treatment led to a very marked decrease in the size of his intraocular and choroid plexus lesions and stabilization of his pituitary mass size. The patient developed central hypoadrenalism that required treatment with glucocorticoid replacement.

A review of literature for the past 5 decades revealed that metastasis of DTC to the orbital choroid and the pituitary gland have been reported in 17 and 16 cases, respectively. This report is unique as it presents the first case of metastatic PTC to both the intraocular choroid and the pituitary gland. Furthermore, no prior study reported simultaneous DTC metastasis to intraocular choroid and the ventricular choroid. This case highlights that despite their rarity, intraocular and pituitary metastasis of DTC should be considered as potential causes of atypical symptoms such as visual changes or hypopituitarism. Finally, use of TKI therapy for DTC with metastasis to the intraocular choroid and pituitary gland can improve the disease course.

乳头状甲状腺癌是分化型甲状腺癌(DTC)最常见的类型,在大多数情况下,疾病局限于颈部,疾病缓解是常见的。虽然DTC的远处转移并不常见;受累部位多见于肺和骨骼,而不太常见的部位包括肝和肾。目前还没有同时转移到眼内脉络膜和脑垂体的病例报道。我们描述了一个58岁的男子谁提出了多灶乳头状甲状腺癌(PTC)与宫颈淋巴结转移。根据DTC的风险分层,将患者分层为低复发风险。初次就诊五年后,他被发现有纵隔淋巴结转移。确诊15年后,他的PTC转移到肝脏、骨骼、肺、脑垂体、眶脉络膜和脉络膜丛。值得注意的是,由于这种转移,患者出现的唯一症状是视力改变,这是用贝伐单抗治疗的。尽管最初有所改善,但他出现了上睑下垂和第三神经症状。他的垂体转移用电子刀手术治疗,他的全身转移用酪氨酸激酶抑制剂(TKI) lenvantinib治疗,使他的视力有了新的改善。Lenvantinib治疗使他的眼内和脉络膜丛病变的大小显著减小,并稳定了垂体肿块的大小。患者出现中枢性肾上腺功能减退,需要糖皮质激素替代治疗。回顾过去50年的文献发现,分别有17例和16例DTC转移到眶脉络膜和垂体。本报告是独一无二的,因为它提出了转移性PTC到眼内脉络膜和脑垂体的第一例。此外,之前没有研究报道DTC同时转移到眼内脉络膜和心室脉络膜。本病例强调,尽管罕见,但DTC的眼内和垂体转移应被视为不典型症状(如视力改变或垂体功能减退)的潜在原因。最后,对于转移到眼内脉络膜和垂体的DTC,使用TKI治疗可以改善病程。
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引用次数: 3
Enterococcus cecorum in diabetic foot infection 盲肠球菌与糖尿病足感染的关系
Q4 Medicine Pub Date : 2021-06-01 DOI: 10.1016/j.jecr.2021.100085
Zsolt Bodnar, Aqeel Alameer
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引用次数: 1
期刊
Journal of Clinical and Translational Endocrinology: Case Reports
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