AACE Clinical Case Reports最新文献

Insulin-Induced Severe Lipohypertrophy 胰岛素诱发的严重脂肪肥厚症

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.03.005

引用次数: 0

Noonan Syndrome and Celiac Disease in an Adolescent With Short Stature and Delayed Puberty 一名身材矮小、青春期延迟的青少年患有努南综合征和乳糜泻

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.05.002

Justin Lee BA , Sabitha Sasidharan Pillai MD , Avani Ganta MD , Chanika Phornphutkul MD , Jose Bernardo Quintos MD

Background/Objective

We present an adolescent male with Noonan syndrome (NS) and celiac disease (CD) who attained normal adult height with growth hormone (GH) treatment and gluten-free diet (GFD).

Case Report

A 15 ½ year old healthy male presented with short stature and delayed puberty. His mother and maternal grandmother were short with heights 142.2 cm and 147.3 cm, respectively. Examination showed bilateral epicanthal folds and down slanting eyes like his mother, fifth finger clinodactyly, height 147.5 cm (<1%; standard deviation score, −2.96), growth velocity 2.5 cm/y, weight 48.2 kg (11%; standard deviation score, −1.24), Tanner 2 pubic hair and Tanner 1 genitalia. Midparental target height was 169.1 cm. He had normal screening studies for GH deficiency and thyroid disorders, prepubertal gonadotropins and testosterone levels, and normal total immunoglobulin A, and elevated antitissue transglutaminase immunoglobulin A 134.7units/mL (0-20). Bone age was 13 years. Genetic evaluation revealed heterozygous missense variant of BRAF gene in him and his mother confirming a diagnosis of NS. He was diagnosed with CD by intestinal biopsy. Patient was started on GH therapy and a GFD with subsequent improvement in growth velocit (6.8-12.3 cm/y) and advancement of puberty. The patient stopped GH therapy at 17 ½ years with a height 165.9 cm.

Discussion

Coexistence of NS caused by BRAF missense variant and CD has not been previously reported. Our patient attained normal adult height with GH therapy and GFD.

Conclusion

NS and CD can co-occur and addressing both these disorders can help patients attain normal height potential.

背景/目的我们介绍了一名患有努南综合征（NS）和乳糜泻（CD）的青少年男性患者，通过生长激素（GH）治疗和无麸质饮食（GFD），他的成年身高达到了正常水平。他的母亲和外祖母身高分别为 142.2 厘米和 147.3 厘米。检查结果显示，他和母亲一样有双侧上睑皱褶和下斜眼，五指挛缩，身高 147.5 厘米（<1%；标准偏差分值，-2.96），生长速度 2.5 厘米/年，体重 48.2 千克（11%；标准偏差分值，-1.24），阴毛为 Tanner 2 型，生殖器为 Tanner 1 型。父母的目标身高为 169.1 厘米。他的 GH 缺乏症和甲状腺疾病筛查结果正常，青春期前促性腺激素和睾酮水平正常，总免疫球蛋白 A 正常，抗组织转谷氨酰胺酶免疫球蛋白 A 升高，为 134.7 单位/毫升（0-20）。骨龄为 13 岁。遗传学评估显示，他和他母亲的 BRAF 基因存在杂合错义变异，确诊为 NS。通过肠道活检，他被诊断为 CD。患者开始接受 GH 治疗和 GFD，随后生长速度得到改善（6.8-12.3 厘米/年），青春期提前。患者在 17 岁半时停止了 GH 治疗，身高达到 165.9 厘米。结论NS和CD可同时存在，解决这两种疾病可帮助患者获得正常的身高潜能。

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引用次数: 0

Non–Islet-Cell Tumor Hypoglycemia Secondary to Malignant Phyllodes Tumor of the Breast 继发于乳腺恶性植物瘤的非胰岛细胞肿瘤性低血糖症（NICTH）

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.002

Si Min Lee MRCP, MMed, MBBS (Hons) , Timothy Peng Lim Quek MRCP, MMed, MRCPSG, Dip, MSc, DLSHTM, DipRCPath, MBBS (Hons) , Cherng Jye Seow MBBS, DFD, GDFM, MRCP, MRCPS, MCI , Pei Shan Yeo MBBS, MRCP, MMed

Background/Objective

Non–islet cell tumor hypoglycemia (NICTH) is an uncommon condition, of which only a few cases caused by malignant phyllodes tumor of the breast have been reported. We describe a case of NICTH secondary to malignant phyllodes tumor with good response to glucocorticoid therapy.

Case Report

A 62-year-old woman with a rapidly enlarging left breast mass presented with drowsiness and a capillary blood glucose level of 32.4 mg/dL. Her plasma glucose and insulin levels were 36.0 mg/dL (reference range, 72-144 mg/dL) and 0.6 mIU/L (reference range, 0.0-25.0 mIU/L), respectively. Her beta-hydroxybutyrate and c-peptide levels were undetectable. The insulin-like growth factor (IGF)-I and IGF-II levels were 37 μg/L (reference range, 43-220 μg/L) and 1062 ng/mL (reference range, 333-967 ng/mL), respectively, with an IGF-II:IGF-I molar ratio of 29.4. Prednisolone 30 mg per day was initiated with improvement in hypoglycemia. Outpatient flash glucose monitoring profile was stable with mild hypoglycemia (glucose level, 54-68.5 mg/dL) detected 5% of the time. The patient underwent left mastectomy with axillary clearance 4 weeks later. Histology was reported as malignant phyllodes tumor with extensive ductal carcinoma in situ. Prednisolone was stopped after surgery. The patient was treated with letrozole and adjuvant radiotherapy. There was no recurrence of hypoglycemia during the subsequent 24-month follow-up.

Discussion

The mainstay of treatment for NICTH is surgical resection of the culprit tumor. Although glucocorticoid treatment has also been widely used for NICTH, few reports have demonstrated efficacy for NICTH secondary to phyllodes tumor.

Conclusion

We report a rare case of malignant phyllodes tumor of the breast resulting in NICTH and demonstrated good response to glucocorticoids as a bridge to definitive surgery.

背景/目的非胰岛细胞瘤性低血糖症（NICTH）是一种不常见的疾病，其中由乳腺恶性梭形细胞瘤引起的病例仅有少数报道。我们描述了一例继发于恶性梭形细胞瘤的 NICTH 病例，该病例对糖皮质激素治疗反应良好。病例报告：一名 62 岁女性，左侧乳房肿块迅速增大，出现嗜睡和毛细血管血糖水平 32.4 mg/dL。她的血浆葡萄糖和胰岛素水平分别为 36.0 mg/dL（参考范围 72-144 mg/dL）和 0.6 mIU/L（参考范围 0.0-25.0 mIU/L）。她的β-羟丁酸和c-肽水平检测不到。胰岛素样生长因子（IGF）-I和IGF-II水平分别为37微克/升（参考范围：43-220微克/升）和1062纳克/毫升（参考范围：333-967纳克/毫升），IGF-II与IGF-I的摩尔比为29.4。开始使用泼尼松龙，每天 30 毫克，低血糖症状有所改善。门诊闪光灯血糖监测结果稳定，5%的时间检测到轻度低血糖（血糖水平，54-68.5 mg/dL）。4 周后，患者接受了左侧乳房切除术和腋窝清扫术。组织学报告为恶性植物瘤，伴有广泛的导管原位癌。术后停用了泼尼松龙。患者接受了来曲唑治疗和辅助放疗。讨论 NICTH 的主要治疗方法是手术切除原发肿瘤。尽管糖皮质激素治疗也被广泛用于 NICTH，但很少有报道证实其对继发于蝶骨瘤的 NICTH 有疗效。

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引用次数: 0

A Case of Nonislet Cell Tumor Hypoglycemia Due to Metastatic Salivary Myoepithelial Carcinoma 唾液腺肌上皮癌转移导致非小细胞肿瘤性低血糖症一例

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.06.002

Margaret C. Slack MD , Samantha Sovich MD , Chana R. Sachs MD , Dorothy Martinez MD , Run Yu MD

Background/Objective

Nonislet cell tumor hypoglycemia (NICTH) is an uncommon cause of hypoglycemia due to a relative surplus of insulin-like growth factor 2 (IGF-2) or its precursor molecule. The diagnosis is confirmed by an elevated ratio of IGF-2 to insulin-like growth factor 1 (IGF-1). Myoepithelial carcinoma (MECA) is a rare and aggressive salivary gland cancer that has not been previously associated with NICTH.

Case Report

A 63-year-old female with a past medical history of metastatic salivary MECA, type 2 diabetes mellitus previously on metformin, hypertension, and hypothyroidism presented to her oncologist for chemotherapy and was found to have a serum glucose of 30 mg/dL (reference: 65-99). She was admitted for further diagnostic work-up which revealed an insulin level of <1 μU/mL (reference: 3-25), C-peptide <0.5 ng/mL (reference: 1.1-4.3), IGF-1 of 15 ng/mL (reference: 41-279), and IGF-2 of 147 ng/mL (reference: 180-580) with an IGF-2:IGF-1 molar ratio of 10, consistent with NICTH. The patient’s hypoglycemia unfortunately was quite resistant to treatment, requiring a combination of corticosteroids, continuous dextrose infusion, and somatostatin injections. The patient died 3 weeks after presenting with hypoglycemia.

Discussion

Salivary MRCAs commonly contain pleomorphic adenoma gene 1 oncogene rearrangements which are associated with increased IGF-2 production and may predispose patients to hypoglycemia.

Conclusion

This case demonstrates that NICTH can be associated with metastatic salivary MECA. The hypoglycemia in this scenario is challenging to manage and is associated with poor prognosis.

背景/目的非小细胞肿瘤性低血糖症（NICTH）是由于胰岛素样生长因子2（IGF-2）或其前体分子相对过剩而引起的一种不常见的低血糖症。IGF-2与胰岛素样生长因子1（IGF-1）的比值升高即可确诊。病例报告一名 63 岁女性患者，既往病史为转移性唾液腺上皮癌（MECA）、2 型糖尿病，曾服用二甲双胍、高血压和甲状腺功能减退症，因化疗到肿瘤科就诊，发现血糖为 30 mg/dL（参考值：65-99）。她入院接受进一步诊断检查，结果显示胰岛素水平为 1 μU/mL（参考值：3-25），C 肽为 0.5 ng/mL（参考值：1.1-4.3），IGF-1 为 15 ng/mL（参考值：41-279），IGF-2 为 147 ng/mL（参考值：180-580），IGF-2:IGF-1 摩尔比为 10，符合 NICTH。不幸的是，患者的低血糖症非常难治，需要联合使用皮质类固醇、持续输注葡萄糖和注射体生长抑素。讨论唾液腺 MRCA 通常含有多形性腺瘤基因 1 的癌基因重排，这与 IGF-2 生成增加有关，可能导致患者出现低血糖。本病例表明，NICTH 可与转移性唾液腺肿瘤伴发，这种情况下的低血糖症极难处理，且与预后不良有关。

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引用次数: 0

Treatment of Bisphosphonate-Associated Atypical Femur Fracture With a Combination of Teriparatide and a Novel Surgical Technique 特立帕肽与新型外科技术相结合治疗双膦酸盐相关非典型股骨骨折

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.05.001

Background/Objective

Atypical femur fractures (AFFs) caused by long-term bisphosphonate use are associated with high rates of delayed healing and nonunion.

Case Report

A 64-year-old woman with osteopenia on alendronate for 15 years sustained a displaced left AFF following a fall from standing height. Imaging showed an acute displaced transverse diaphyseal left femur fracture with lateral cortical thickening and beaking. She underwent an open reduction and internal fixation with insertion of a cephalomedullary nail placed in compression mode, utilizing a novel technique involving intraoperative removal of the endosteal hypertrophied cortical bone at the fracture site. Alendronate was stopped and teriparatide was initiated postoperatively. Radiographs at 3.5 months postsurgery showed evidence of normal fracture union with mature callus formation.

Discussion

AFFs caused by prolonged bisphosphonate use have a high rate of delayed healing and nonunion due to abnormal bone remodeling. Use of teriparatide postoperatively has been shown to reduce healing time in small observational studies in surgically treated patients. Our case demonstrates an expedited healing time of 3.5 months using teriparatide combined with a novel surgical technique involving removal of a portion of the abnormally remodeled bone and placement of an intramedullary nail in compression mode.

Conclusion

Our case demonstrates an expedited healing time of 3.5 months compared to the average reported healing time for AFF of 10.7 months, supporting the use of the combination of teriparatide and a novel surgical technique.

背景/目的：长期使用双膦酸盐引起的非典型股骨骨折（AFF）与高延迟愈合率和非愈合率有关。病例报告：一名 64 岁的女性患者患有骨质疏松症，服用阿仑膦酸盐 15 年，一次从站立高度摔倒后，左侧股骨骨折移位。影像学检查显示，左股骨横骺急性移位骨折，外侧皮质增厚并呈蜂窝状。她接受了切开复位和内固定术，在加压模式下植入了头髓内钉，采用了一种新技术，即术中去除骨折部位骨膜内肥厚的皮质骨。术后停用阿仑膦酸钠，并开始使用特立帕肽。术后3.5个月的X光片显示骨折愈合正常，并有成熟的胼胝形成。讨论长期使用双膦酸盐引起的骨折由于骨质重塑异常，延迟愈合和不愈合的发生率很高。小型观察性研究显示，术后使用特立帕肽可缩短手术治疗患者的愈合时间。我们的病例显示，使用特立帕肽并结合一种新的手术技术（切除部分异常重塑的骨骼并在加压模式下放置髓内钉），患者的愈合时间缩短了 3.5 个月。结论我们的病例显示，与所报道的 AFF 平均愈合时间 10.7 个月相比，患者的愈合时间缩短了 3.5 个月，这支持了特立帕肽和新手术技术的结合使用。

{"title":"Treatment of Bisphosphonate-Associated Atypical Femur Fracture With a Combination of Teriparatide and a Novel Surgical Technique","authors":"","doi":"10.1016/j.aace.2024.05.001","DOIUrl":"10.1016/j.aace.2024.05.001","url":null,"abstract":"<div><h3>Background/Objective</h3><p>Atypical femur fractures (AFFs) caused by long-term bisphosphonate use are associated with high rates of delayed healing and nonunion.</p></div><div><h3>Case Report</h3><p>A 64-year-old woman with osteopenia on alendronate for 15 years sustained a displaced left AFF following a fall from standing height. Imaging showed an acute displaced transverse diaphyseal left femur fracture with lateral cortical thickening and beaking. She underwent an open reduction and internal fixation with insertion of a cephalomedullary nail placed in compression mode, utilizing a novel technique involving intraoperative removal of the endosteal hypertrophied cortical bone at the fracture site. Alendronate was stopped and teriparatide was initiated postoperatively. Radiographs at 3.5 months postsurgery showed evidence of normal fracture union with mature callus formation.</p></div><div><h3>Discussion</h3><p>AFFs caused by prolonged bisphosphonate use have a high rate of delayed healing and nonunion due to abnormal bone remodeling. Use of teriparatide postoperatively has been shown to reduce healing time in small observational studies in surgically treated patients. Our case demonstrates an expedited healing time of 3.5 months using teriparatide combined with a novel surgical technique involving removal of a portion of the abnormally remodeled bone and placement of an intramedullary nail in compression mode.</p></div><div><h3>Conclusion</h3><p>Our case demonstrates an expedited healing time of 3.5 months compared to the average reported healing time for AFF of 10.7 months, supporting the use of the combination of teriparatide and a novel surgical technique.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 170-173"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000464/pdfft?md5=ae0dffd2b476cdfe83bacb296492c509&pid=1-s2.0-S2376060524000464-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141034914","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

Case Report: IgG4-Related Disease Presenting With Isolated Hypophysitis 病例报告：表现为孤立性肾上腺皮质功能减退症的 IgG4 相关疾病

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.004

Suhaib Radi MD , Michael Tamilia FRCPC

Background/Objective

IgG4-related disease (IgG4-RD) is an immune-mediated condition that affects multiple organs, including the pituitary gland. Here we present a patient with isolated pituitary involvement of IgG4-RD mimicking pituitary apoplexy.

Case Report

A 49-year-old woman presented to the emergency department with abdominal pain, nausea, vomiting, and weight loss. Her blood pressure was low, and she appeared euvolemic with the rest of physical examination being noncontributory. Her electrolytes showed low serum sodium of 118 mmol/L (normal 135-145). Further investigations were significant for low morning cortisol of 20 nmol/L (N:100-500) and low adrenocorticotropic hormone. Magnetic resonant imaging of the pituitary fossa showed a pituitary macroadenoma with hemorrhagic transformation. She was started on glucocorticoids and levothyroxine before undergoing surgical removal of the pituitary tumor. The pathology was positive for IgG-4-related hypophysitis (IgG4-RH) with no evidence of pituitary tumor.

Discussion

IgG4-RD is an immune-mediated condition that can affect many organs including the pituitary gland, in the form of hypophysitis. IgG4-RH can affect anterior, posterior, or both pituitary lobes. In 2011, Leporati et al developed a diagnostic criteria for IgG4-RH which includes the following: imaging, serology, histopathology, and response t glucocorticoids. The mainstay of treatment is glucocorticoids and hormone replacement therapy.

Conclusion

IgG4-RH might be underestimated and should be suspected in those with hypophysitis or unknown cause of hypopituitarism. Moreover, pituitary macroadenoma with hemorrhagic transformation and panhypopituitarism should be considered as rare and unusual presentations of IgG4-RD.

背景/目的IgG4相关疾病（IgG4-RD）是一种免疫介导的疾病，可影响包括垂体在内的多个器官。病例报告一名 49 岁女性因腹痛、恶心、呕吐和体重减轻到急诊科就诊。她的血压很低，看起来没有血容量，其他体格检查也无异常。她的电解质显示血清钠偏低，为 118 毫摩尔/升（正常值为 135-145 毫摩尔/升）。进一步检查显示，她的晨间皮质醇偏低，为 20 nmol/L（正常值：100-500），促肾上腺皮质激素也偏低。垂体窝的磁共振成像显示垂体大腺瘤伴出血性转化。她开始服用糖皮质激素和左甲状腺素，然后接受了垂体瘤手术切除。讨论IgG4-RD是一种免疫介导的疾病，可以影响包括垂体在内的许多器官，表现为垂体功能减退症。IgG4-RH 可影响垂体前叶、后叶或双侧垂体。2011 年，Leporati 等人制定了 IgG4-RH 的诊断标准，其中包括：影像学、血清学、组织病理学和对糖皮质激素的反应。结论IgG4-RH可能被低估，应怀疑患有垂体功能减退症或原因不明的垂体功能减退症。此外，垂体大腺瘤伴出血性转化和泛垂体功能减退症应被视为IgG4-RD罕见的异常表现。

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引用次数: 0

Exaggerated Increases in the Serum Cortisol Level in a Woman Following Oral Contraceptive Treatment 一名妇女口服避孕药后血清皮质醇的过度升高

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.005

Run Yu MD, PhD

Background/Objective

Extreme hypercortisolemia in an otherwise healthy patient can be due to familial dysalbuminemia, generalized glucocorticoid resistance, and estrogen-containing medications. I report a woman who appeared to have an exaggerated increase in the serum cortisol level following oral contraceptive treatment.

Case Report

A 50-year-old woman presented with extreme morning hypercortisolemia—cortisol levels of 61 and 55 mcg/dL (4 and 3 months before presentation, respectively; normal range, 8-25 mcg/dL)—found during workup of mildly increased white cell counts. The morning cortisol level had been 10 mcg/dL after administration of 1-mg dexamethasone. The 24-hour urine free cortisol level had been normal and only slightly increased after correction by creatinine. The patient was anxious about the extremely high cortisol levels but otherwise felt well. She took norgestimate-ethinyl estradiol contraceptive (0.18/0.215/0.25 mg - 35 mcg). Physical examination showed a well-appearing, lean female. The thyroid-stimulating hormone, total thyroxine, free thyroxine, total triiodothyronine, free triiodothyronine, androstenedione, dehydroepiandrosterone sulfate, aldosterone, and renin levels were normal. Morning total cortisol and cortisol-binding globulin (CBG) were tested before and after she held the oral contraceptive for 2 months. The total cortisol and CBG levels decreased from 50 to 26 mcg/dL and from 6.4 to 3.8 mg/dL (normal range, 1.7-3.1 mg/dL), respectively.

Discussion

Increases in the serum cortisol-binding proteins are a well-recognized cause for increases in the serum cortisol levels.

Conclusion

This case suggests that modern oral contraceptives with low to moderate estrogen activity can cause extreme increases in the serum cortisol levels due to marked increases in the CBG levels.

背景/目的原本健康的患者出现极度高皮质醇血症可能是由于家族性白蛋白血症、全身性糖皮质激素抵抗和含有雌激素的药物所致。病例报告一位 50 岁的女性患者在接受口服避孕药治疗后出现血清皮质醇水平的夸张性升高--皮质醇水平分别为 61 和 55 mcg/dL（发病前 4 个月和 3 个月；正常范围为 8-25 mcg/dL）--这是在白细胞计数轻度升高的检查中发现的。在服用1毫克地塞米松后，晨间皮质醇水平为10微克/分升。24 小时尿液游离皮质醇水平正常，经肌酐校正后仅略有升高。患者对皮质醇水平过高感到焦虑，但其他方面感觉良好。她服用了炔诺酮-乙炔雌二醇避孕药（0.18/0.215/0.25 毫克 - 35 微克）。体格检查显示，她是一名面容姣好的瘦弱女性。促甲状腺激素、总甲状腺素、游离甲状腺素、总三碘甲状腺原氨酸、游离三碘甲状腺原氨酸、雄烯二酮、硫酸脱氢表雄酮、醛固酮和肾素水平正常。在她口服避孕药 2 个月之前和之后，检测了晨间总皮质醇和皮质醇结合球蛋白（CBG）。总皮质醇和 CBG 水平分别从 50 毫微克/分升降至 26 毫微克/分升，从 6.4 毫克/分升降至 3.8 毫克/分升（正常范围为 1.7-3.1 毫克/分升）。讨论血清皮质醇结合蛋白的增加是导致血清皮质醇水平升高的公认原因。

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引用次数: 0

Stiff Person Syndrome and Brittle Type 1 Diabetes: Report of 2 Cases 僵人综合征和脆性 1 型糖尿病。两个病例的报告

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.003

Ismael A. Quintal-Medina MD , Francisco J. Gómez-Pérez MD , Paloma Almeda-Valdes MD, PhD

Background/Objective

Stiff person syndrome (SPS) and type 1 diabetes (T1D) are heterogeneous disorders characterized by antibodies (Abs) against glutamic acid decarboxylase (GAD).

Case Report

We describe 2 patients with T1D and autoimmune thyroid disease who presented with muscle rigidity and intermittent spasms that affected gait and with elevated circulating anti-GAD titers. Classic SPS and stiff limb syndrome were diagnosed, respectively. Muscle spasms resolved with immunotherapy and muscle relaxants in both patients, and the ability to ambulate without an assistive device was restored in 1 patient. Patients also had brittle diabetes with high glycemic variability, requiring the use of flash glucose monitoring with an insulin pump and a second-generation basal insulin analog, respectively.

Discussion

GAD Ab–associated syndromes include SPS, T1D, and other endocrinopathies. The clinical heterogeneity implies variable susceptibility of γ-aminobutyric acid-ergic neurons and pancreatic beta cells to anti-GAD or other autoantibodies.

Conclusion

Our case series represent the heterogeneity in natural history, clinical course, and response to therapy in patients with Abs against GAD-spectrum disorders.

背景/目的僵硬综合征（SPS）和1型糖尿病（T1D）是以谷氨酸脱羧酶（GAD）抗体（Abs）为特征的异质性疾病。病例报告我们描述了2例患有T1D和自身免疫性甲状腺疾病的患者，他们表现为肌肉僵硬和间歇性痉挛，影响步态，循环中抗GAD滴度升高。他们分别被诊断为典型的 SPS 和僵硬肢体综合征。通过免疫疗法和肌肉松弛剂，两名患者的肌肉痉挛都得到了缓解，其中一名患者在没有辅助设备的情况下恢复了行走能力。患者还患有血糖变异性高的脆性糖尿病，需要分别使用胰岛素泵和第二代基础胰岛素类似物进行闪光血糖监测。临床异质性意味着γ-氨基丁酸能神经元和胰岛β细胞对抗GAD或其他自身抗体的易感性各不相同。

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引用次数: 0

Type 1 Diabetes Mellitus Caused by COVID-19 mRNA Vaccination: A Case Report and Literature Review of 17 Published Cases 接种 COVID-19 mRNA 疫苗导致的 1 型糖尿病：病例报告和已发表 17 例病例的文献综述

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.06.001

Background/Objective

Multiple cases of postvaccination immune-related adverse events have been reported. We, hereby, present a patient who presented with new-onset type 1 diabetes mellitus (DM) after COVID-19 messenger RNA (mRNA) vaccination.

Case Report

A 38-year-old Caucasian man presented with sudden onset of polyuria, polydipsia, and blurry vision for 1 month. The patient received the second dose of the COVID-19 mRNA vaccine (Pfizer-BioNTech) 4 weeks prior to symptom onset. Initial workup revealed glucosuria and hemoglobin A1c of 9.4%. Antibodies against multiple pancreatic beta cell autoantigens were detected. The patient was then initiated on insulin.

Discussion

Hypothesized mechanisms for development of type 1 DM after COVID-19 mRNA vaccination include molecular mimicry, autoimmune/inflammatory syndrome induced by adjuvants, and possible interaction between the angiotensin-I converting enzyme-2 receptor on beta cells and viral mRNA. An initial high index of suspicion should be accompanied by early autoantibody testing and initiation of insulin, if indicated. Finally, if diagnosed with type 1 diabetes, patients must have long-term follow-up as there may be brief periods where glycemic control is maintained off insulin.

Conclusion

New-onset type 1 DM has been reported after COVID mRNA vaccination. Clinicians should maintain a high index of suspicion and pursue early testing for the same to reduce adverse outcomes and improve long-term prognosis.

背景/目的已有多例疫苗接种后免疫相关不良事件的报道。病例报告：一名 38 岁的白种男子在接种 COVID-19 信使 RNA（mRNA）疫苗后突然出现多尿、多饮和视力模糊，持续 1 个月。患者在发病前 4 周接种了第二剂 COVID-19 mRNA 疫苗（辉瑞生物技术公司）。初步检查显示患者有葡萄糖尿，血红蛋白 A1c 为 9.4%。检测到多种胰腺β细胞自身抗原抗体。讨论COVID-19 mRNA疫苗接种后发生1型糖尿病的假设机制包括分子模拟、佐剂诱导的自身免疫/炎症综合征以及β细胞上的血管紧张素-I转换酶-2受体与病毒mRNA之间可能存在的相互作用。在最初高度怀疑的同时，应尽早进行自身抗体检测，并在必要时开始使用胰岛素。最后，如果确诊为 1 型糖尿病，患者必须接受长期随访，因为可能会出现短暂的血糖控制不依赖胰岛素的情况。临床医生应保持高度怀疑，并尽早进行相关检测，以减少不良后果并改善长期预后。

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引用次数: 0

Multifocal Nonmetastatic Radioactive Iodine Avidity on Whole Body Scan After Thyroidectomy for Thyroid Cancer 甲状腺癌甲状腺切除术后全身扫描显示多灶性非转移性放射性碘惰性

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.001

Jim T.C. Chen MD , Kirun Baweja MbChB , Lurdes Tse-Agha MD , Sara Awad MBBS, MHPE, FRCPC

Background/Objective

Non-metastatic radioactive iodine (RAI) uptake can complicate the interpretation of whole-body scan (WBS) for differentiated thyroid carcinoma (DTC) post-thyroidectomy. We present a patient with DTC whose follow-up WBS showed nonmetastatic multifocal avidity in skeletal tissue, an uncommonly reported site of RAI uptake.

Case report

A 42-year-old woman underwent a right hemithyroidectomy, followed by completion thyroidectomy and RAI remnant ablation therapy, for a 4.8 cm thyroid tumor consistent with stage pT3aNxMx follicular thyroid cancer. Follow-up WBS showed intense activity in the thyroid bed, right breast, left medial subcortical acetabulum, and several vertebral bodies. Her biochemical and clinical findings were not suggestive of cancer recurrence. Further workup with SPECT/CT and MRI showed no focal vertebral lesions and identified the left femoral lesion as a benign peripheral nerve sheath. Diagnostic mammography and ultrasound showed no evidence of suspicious breast lesions. Neck ultrasound was clear with no suspicious masses or pathologic lymphadenopathy. She remained in remission on continued active surveillance.

Discussion

Nonmetastatic RAI uptake on WBS has many causes, including functional sodium-iodide symporter expression in nonthyroidal tissues, radioiodine accumulation in tissues and bodily fluids, and benign tumors. False-positive uptake can decrease the utility of post-treatment WBS in low-risk patients. Careful clinical examination, biochemical and radiologic follow-up, and close active surveillance can help distinguish false-positive uptake from metastatic or recurrent disease.

Conclusion

We describe an uncommon case of RAI uptake in skeletal tissues after thyroidectomy for DTC, and we outline the steps taken to rule out underlying metastases.

背景/目的甲状腺切除术后分化型甲状腺癌（DTC）的全身扫描（WBS）结果显示，非转移性放射性碘（RAI）摄取可能会使判读复杂化。病例报告：一名 42 岁女性因患 4.8 厘米甲状腺肿瘤（符合 pT3aNxMx 滤泡型甲状腺癌分期）接受了右半甲状腺切除术，随后进行了甲状腺全切术和 RAI 残余消融治疗。随访WBS显示，甲状腺床、右乳房、左侧皮质下髋臼内侧和多个椎体有强烈活动。她的生化和临床检查结果均未提示癌症复发。进一步的 SPECT/CT 和 MRI 检查显示没有椎体病灶，并确定左股骨病变为良性周围神经鞘。诊断性乳房 X 线照相术和超声波检查没有发现可疑的乳房病变。颈部超声检查结果清晰，未发现可疑肿块或病理性淋巴结病变。讨论WBS上的非转移性RAI摄取有多种原因，包括非甲状腺组织中钠离子-碘离子交感器的功能表达、组织和体液中的放射性碘蓄积以及良性肿瘤。假阳性摄取会降低低风险患者治疗后 WBS 的效用。仔细的临床检查、生化和放射学随访以及密切的主动监测有助于将假阳性摄取与转移性或复发性疾病区分开来。结论我们描述了一例不常见的因 DTC 而行甲状腺切除术后骨骼组织摄取 RAI 的病例，并概述了为排除潜在转移瘤而采取的措施。

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