Endocrinology, diabetes & metabolism case reports最新文献

{"title":"A silent myocardial infarction in the diabetes outpatient clinic: case report and review of the literature.","authors":"M S Draman,&nbsp;H Thabit,&nbsp;T J Kiernan,&nbsp;J O'Neill,&nbsp;S Sreenan,&nbsp;J H McDermott","doi":"10.1530/EDM-13-0058","DOIUrl":"https://doi.org/10.1530/EDM-13-0058","url":null,"abstract":"<p><strong>Unlabelled: </strong>Silent myocardial ischaemia (SMI), defined as objective evidence of myocardial ischaemia in the absence of symptoms, has important clinical implications for the patient with coronary artery disease. We present a dramatic case of SMI in a diabetes patient who attended annual review clinic with ST elevation myocardial infarction. His troponin was normal on admission but raised to 10.7 ng/ml (normal <0.5) when repeated the next day. His angiogram showed diffused coronary artery disease. We here discuss the implications of silent ischaemia for the patient and for the physician caring for patients with diabetes.</p><p><strong>Learning points: </strong>Silent myocardial ischaemia (SMI) is an important clinical entity.SMI is common and occurs with increased frequency in patients with diabetes.SMI is an independent predictor of mortality.Recognition may lead to early intervention.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130058"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1530/EDM-13-0058","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40298897","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}

{"title":"Methimazole-induced myositis: a case report and review of the literature.","authors":"R Bou Khalil,&nbsp;M Abou Salbi,&nbsp;S Sissi,&nbsp;N El Kara,&nbsp;E Azar,&nbsp;M Khoury,&nbsp;G Abdallah,&nbsp;J Hreiki,&nbsp;S Farhat","doi":"10.1530/EDM-13-0008","DOIUrl":"https://doi.org/10.1530/EDM-13-0008","url":null,"abstract":"<p><strong>Unlabelled: </strong>Methimazole is an anti-thyroid drug commonly used to treat hyperthyroidism and is a relatively safe medication. Several side effects have been reported and usually develop within 3 months of therapy. Well-known adverse reactions include agranulocytosis, hepatitis, skin eruptions, and musculoskeletal complaints such as myalgia, arthralgia, and arthritis. So far, myositis secondary to carbimazole was described in the context of a lupus-like syndrome or other rare cases of anti-neutrophil cytoplasmic antibodies-associated vasculitis. Methimazole-induced myositis occurring independently of such reactions was rarely stated. We report a patient with hyperthyroidism who, early after therapy with methimazole, developed hepatitis, eosinophilia, and fever that resolved completely after stopping the medication as well as a delayed onset of biopsy-proven eosinophilic myositis and fasciitis of gluteal muscles that resolved eventually without any additional therapy. Therefore, we raise the awareness regarding a rare side effect of methimazole: myositis.</p><p><strong>Learning points: </strong>Several differential diagnoses arise when managing a hyperthyroid patient with muscle complaints.Both hyperthyroidism and methimazole are associated with myositis.Methimazole-induced myositis is a rare clinical entity.Resolution of symptoms may occur after stopping methimazole.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130008"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1530/EDM-13-0008","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40300545","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}

{"title":"Paternally inherited ABCC8 mutation causing diffuse congenital hyperinsulinism.","authors":"Suresh Chandran,&nbsp;Fabian Yap Kok Peng,&nbsp;Victor Samuel Rajadurai,&nbsp;Yap Te Lu,&nbsp;Kenneth T E Chang,&nbsp;S E Flanagan,&nbsp;S Ellard,&nbsp;Khalid Hussain","doi":"10.1530/EDM-13-0041","DOIUrl":"https://doi.org/10.1530/EDM-13-0041","url":null,"abstract":"<p><strong>Background: </strong>Congenital hyperinsulinism (CHI) is a rare genetic disorder characterised by inappropriate insulin secretion in the face of severe hypoglycaemia. There are two histological subtypes of CHI namely diffuse and focal. Diffuse CHI is most common due to recessive mutations in ABCC8/KCNJ11 (which encode the SUR/KIR6.2 components of the pancreatic β-cell KATP channel) whereas focal CHI is due to a paternally inherited ABCC8/KCNJ11 mutation and somatic loss of heterozygosity for the 11p allele inside the focal lesion. Fluorine-18-l-dihydroxyphenylalanine positron emission tomography/computed tomography ((18)F-DOPA-PET/CT) is used in the pre-operative localisation of focal lesions prior to surgery. Diffuse CHI if medically unresponsive will require a near total pancreatectomy whereas focal CHI will only require a limited lesionectomy, thus curing the patient from the hypoglycaemia.</p><p><strong>Aims: </strong>To report the first case of genetically confirmed CHI in Singapore from a heterozygous paternally inherited ABCC8 mutation.</p><p><strong>Methods/results: </strong>A term male infant presented with severe hyperinsulinaemic hypoglycaemia (HH) after birth and failed medical treatment with diazoxide and octreotide. Genetic testing (paternally inherited mutation in ABCC8/p.D1472N) suggested focal disease, but due to the unavailability of (18)F-DOPA-PET/CT to confirm focal disease, a partial pancreatectomy was performed. Interestingly, histology of the resected pancreatic tissue showed changes typical of diffuse disease.</p><p><strong>Conclusion: </strong>Heterozygous paternally inherited ABCC8/KCNJ11 mutations can lead to diffuse or focal CHI.</p><p><strong>Learning points: </strong>HH is a cause of severe hypoglycaemia in the newborn period.Paternal mutations in ABCC8/KCNJ11 can lead to diffuse or focal disease.(18)F-DOPA-PET/CT scan is the current imaging of choice for localising focal lesions.Gallium-68 tetra-aza-cyclododecane-N N'N″N-‴-tetra-acetate octreotate PET scan is not a useful imaging tool for localising focal lesions.The molecular mechanism by which a heterozygous ABCC8 mutation leads to diffuse disease is currently unclear.Focal lesions are curable by lesionectomy and so genetic studies in patients with HH must be followed by imaging using (18)F-DOPA-PET/CT scan.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130041"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1530/EDM-13-0041","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40298890","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}

{"title":"Cushing's disease presenting with gastrointestinal perforation: a case report.","authors":"Takuma Hara,&nbsp;Hiroyoshi Akutsu,&nbsp;Tetsuya Yamamoto,&nbsp;Eiichi Ishikawa,&nbsp;Masahide Matsuda,&nbsp;Akira Matsumura","doi":"10.1530/EDM-13-0064","DOIUrl":"https://doi.org/10.1530/EDM-13-0064","url":null,"abstract":"<p><strong>Unlabelled: </strong>Gastrointestinal perforation is a complication associated with steroid therapy or hypercortisolism, but it is rarely observed in patients with Cushing's disease in clinical practice, and only one case has been reported as a presenting symptom. Herein, we report a rare case of Cushing's disease in which a patient presented with gastrointestinal perforation as a symptom. A 79-year-old man complained of discomfort in the lower abdomen for 6 months. Based on the endocrinological and gastroenterological examinations, he was diagnosed with Cushing's disease with a perforation of the descending colon. After consultation with a gastroenterological surgeon, it was decided that colonic perforation could be conservatively observed without any oral intake and treated with parenteral administration of antibiotics because of the mild systemic inflammation and lack of abdominal guarding. Despite the marked elevated levels of serum cortisol, oral medication was not an option because of colonic perforation. Therefore, the patient was submitted to endonasal adenomectomy to normalize the levels of serum cortisol. Subsequently, a colostomy was successfully performed. Despite its rarity, physicians should be aware that gastrointestinal perforation may be associated with hypercortisolism, especially in elderly patients, and immediate diagnosis and treatment of this life-threatening condition are essential. If a perforation can be conservatively observed, endonasal adenomectomy prior to laparotomy is an alternative treatment option for hypercortisolism.</p><p><strong>Learning points: </strong>Thus far, only one case of gastrointestinal perforation as a presenting clinical symptom of Cushing's disease has been reported.Physicians should be aware that gastrointestinal perforation might be associated with hypercortisolism in elderly patients because elevated levels of serum cortisol may mask the clinical signs of perforation. Because of this masking effect, the diagnosis of the perforation also tends to be delayed.Although parenteral administration of etomidate is a standard treatment option for decreasing the elevated levels of serum cortisol, endonasal adenomectomy prior to laparotomy is an alternative treatment option if etomidate therapy is unavailable.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130064"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3922247/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40299812","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}

{"title":"Pituitary metastases from papillary carcinoma of thyroid: a case report and literature review.","authors":"Viral Chikani,&nbsp;Duncan Lambie,&nbsp;Anthony Russell","doi":"10.1530/EDM-13-0024","DOIUrl":"https://doi.org/10.1530/EDM-13-0024","url":null,"abstract":"<p><strong>Unlabelled: </strong>Metastases to the pituitary gland are an uncommon complication of thyroid cancer. They resemble pituitary neoplasms posing a diagnostic challenge. We present a case of an aggressive non-radioiodine avid papillary thyroid cancer with recurrent pituitary metastases and a review of the literature. A 70-year-old woman with a history of papillary thyroid cancer and bony metastases presented with symptoms of hypoadrenalism and peripheral vision loss. Magnetic resonance imaging showed a large pituitary mass impinging on the optic chiasm. She underwent transsphenoidal resection followed by (131)I ablation. Post-therapy scintigraphy showed no iodine uptake in the sellar region or bony metastases. Histology of the pituitary mass confirmed metastatic papillary thyroid cancer. Fifteen months later, she had a recurrence of pituitary metastases affecting her vision. This was resected and followed with external beam radiotherapy. Over 2 years, the pituitary metastases increased in size and required two further operations. Radioactive iodine was not considered due to poor response in the past. Progressively, she developed a left-sided III and IV cranial nerve palsy and permanent bitemporal hemianopia. There was a rapid decline in the patient's health with further imaging revealing new lung and bony metastases, and she eventually died 8 months later. To our knowledge, this is the first case of pituitary metastases from a radioiodine-resistant papillary thyroid cancer. Radioiodine-resistant metastatic thyroid cancer may exhibit rapid aggressive growth and remain poorly responsive to the currently available treatment.</p><p><strong>Learning points: </strong>Differentiated thyroid cancer (DTC) has an excellent prognosis with <5% of the cases presenting with distant metastases, usually to lung and bone.Metastasis to the pituitary is a rare complication of DTC.The diagnosis of pituitary insufficiency secondary to pituitary metastases from DTC may be delayed due to the non-specific systemic symptoms of underlying malignancy and TSH suppression therapy for thyroid cancer.The imaging characteristics of metastases to the pituitary may be similar to non-functioning pituitary adenoma.Radioiodine refractory metastatic thyroid cancer has significantly lower survival rates compared with radioactive iodine-avid metastases due to limited therapeutic options.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130024"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3922192/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40300436","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}

{"title":"Pasireotide therapy in a rare and unusual case of plurihormonal pituitary macroadenoma.","authors":"Rajesh Rajendran,&nbsp;Sarita Naik,&nbsp;Derek D Sandeman,&nbsp;Azraai B Nasruddin","doi":"10.1530/EDM-13-0026","DOIUrl":"https://doi.org/10.1530/EDM-13-0026","url":null,"abstract":"<p><strong>Unlabelled: </strong>We report the use of pasireotide in a rare and unusual case of pituitary macroadenoma co-secreting GH, prolactin and ACTH. A 62-year-old Caucasian man presented with impotence. Clinically, he appeared acromegalic and subsequent investigations confirmed GH excess and hyperprolactinaemia. Magnetic resonance imaging (MRI) of pituitary revealed a large pituitary macroadenoma. He underwent trans-sphenoidal surgery and histology confirmed an adenoma with immunohistochemistry positive for ACTH, GH and prolactin. Acromegaly was not cured following surgery and inadequately controlled despite subsequent octreotide therapy. He underwent further debulking pituitary surgery, following which IGF1 levels improved but still high. This time adenoma cells showed immunohistochemistry positivity for ACTH only, following which subsequent investigations confirmed intermittent hypercortisolaemia compatible with pituitary Cushing's disease. We recommended radiotherapy, but in view of the pluripotential nature of the tumour, we proceeded with a trial of s.c. pasireotide therapy on the basis that it may control both his acromegaly and Cushing's disease. After 3 months of pasireotide therapy, his mean GH and IGF1 levels improved significantly, with improvement in his symptoms but intermittent hypercortisolaemia persists. His glycaemic control deteriorated requiring addition of new anti-diabetic medication. MRI imaging showed loss of contrast uptake within the tumour following pasireotide therapy but no change in size. We conclude that our patient has had a partial response to pasireotide therapy. Long-term follow-up studies are needed to establish its safety and efficacy in patients with acromegaly and/or Cushing's disease.</p><p><strong>Learning points: </strong>Plurihormonal pituitary adenomas are rare and unusual.Patients with pituitary adenomas co-secreting ACTH and GH are more likely to present with acromegaly because GH excess can mask hypercortisolaemia.Pasireotide holds potential where conventional somatostatin analogues are not effective in acromegaly due to higher affinity for somatostatin receptor subtypes 1, 2, 3 and 5.Significant deterioration in glycaemic control remains a concern in the use of pasireotide.Currently, long-term safety and efficacy of pasireotide in patients with acromegaly and/or Cushing's disease are not fully clear.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130026"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3922038/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40300437","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}

{"title":"Benign adrenal adenomas secreting excess mineralocorticoids and glucocorticoids.","authors":"Vivienne Yoon,&nbsp;Aliya Heyliger,&nbsp;Takashi Maekawa,&nbsp;Hironobu Sasano,&nbsp;Kelley Carrick,&nbsp;Stacey Woodruff,&nbsp;Jennifer Rabaglia,&nbsp;Richard J Auchus,&nbsp;Hans K Ghayee","doi":"10.1530/EDM-13-0042","DOIUrl":"https://doi.org/10.1530/EDM-13-0042","url":null,"abstract":"<p><strong>Objective: </strong>To recognize that benign adrenal adenomas can co-secrete excess aldosterone and cortisol, which can change clinical management.</p><p><strong>Methods: </strong>We reviewed the clinical and histological features of an adrenal tumor co-secreting aldosterone and cortisol in a patient. Biochemical testing as well as postoperative immunohistochemistry was carried out on tissue samples for assessing enzymes involved in steroidogenesis.</p><p><strong>Results: </strong>A patient presented with hypertension, hypokalemia, and symptoms related to hypercortisolism. The case demonstrated suppressed renin concentrations with an elevated aldosterone:renin ratio, abnormal dexamethasone suppression test results, and elevated midnight salivary cortisol concentrations. The patient had a right adrenal nodule with autonomous cortisol production and interval growth. Right adrenalectomy was carried out. Postoperatively, the patient tolerated the surgery, but he was placed on a short course of steroid replacement given a subnormal postoperative serum cortisol concentration. Long-term follow-up of the patient showed that his blood pressure and glucose levels had improved. Histopathology slides showed positive staining for 3β-hydroxysteroid dehydrogenase, 11β-hydroxylase, and 21 hydroxylase.</p><p><strong>Conclusion: </strong>In addition to the clinical manifestations and laboratory values, the presence of these enzymes in this type of tumor provides support that the tumor in this patient was able to produce mineralocorticoids and glucocorticoids. The recognition of patients with a tumor that is co-secreting aldosterone and cortisol can affect decisions to treat with glucocorticoids perioperatively to avoid adrenal crisis.</p><p><strong>Learning points: </strong>Recognition of the presence of adrenal adenomas co-secreting mineralocorticoids and glucocorticoids.Consideration for perioperative and postoperative glucocorticoid use in the treatment of co-secreting adrenal adenomas.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130042"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3922398/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40298891","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}

{"title":"Ectopic prolactin secretion secondary to an ovarian tumour.","authors":"Sowmya Gururaj,&nbsp;K Nisal,&nbsp;Q Davies,&nbsp;S Deen,&nbsp;P G McNally","doi":"10.1530/EDM-13-0016","DOIUrl":"https://doi.org/10.1530/EDM-13-0016","url":null,"abstract":"<p><strong>Unlabelled: </strong>Ectopic hormone secretion is a well-recognised phenomenon; however, ectopic prolactin secretion is exceptionally rare. Hoffman and colleagues reported the first ever well-documented case of ectopic prolactin secretion secondary to a gonadoblastoma. We report a lady who presented with galactorrhoea and a large ovarian tumour that was found to secrete high levels of prolactin.</p><p><strong>Learning points: </strong>Aim of this case report is to highlight the occurrence of this condition.Lack of awareness can often lead to a diagnostic conundrum.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130016"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3922040/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40300433","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}

{"title":"Steroid cell tumor: a rare cause of hirsutism in a female.","authors":"Jayshree Swain,&nbsp;Shruti Sharma,&nbsp;Ved Prakash,&nbsp;N K Agrawal,&nbsp;S K Singh","doi":"10.1530/EDM-13-0030","DOIUrl":"https://doi.org/10.1530/EDM-13-0030","url":null,"abstract":"<p><strong>Unlabelled: </strong>Ovarian steroid cell tumors are very rare functioning sex-cord stromal tumors. They comprise <0.1% of all ovarian tumors. Previously designated as lipoid cell tumors, one-third of these tumors are considered malignant with the mean age of presentation at around 40 years. We present a case of a 28-year-old female with 2-year history of hirsutism, virilization, and amenorrhea. She was diagnosed with left ovarian tumor, for which she underwent left salpingo-oophorectomy. Histopathology revealed not otherwise specified subtype of steroid cell tumors. The patient resumed menses 2 months after the features of masculinization regressed. Within 1 year of surgery, the patient successfully conceived a full-term baby without any complications. In a young female, the neoplastic etiology of a rapid virilization or menses changing should always be kept in mind. Though commonly observed in adult females, steroid cell tumors have very good surgical outcomes if age at presentation is less and tumor is unilateral, and there are no evidences of bilateral malignancy. Bilateral salpingo-oophorectomy is not required.</p><p><strong>Learning points: </strong>In a case of severe rapid hirsutism and virilization with serum testosterone level more than 200 ng/dl or more than threefold of the normal range, neoplastic conditions should always be suspected.Steroid cell tumor in young women without evidence of malignancy on histopathology has excellent surgical outcomes.Unilateral salpingo-oophorectomy is the surgery of choice.As the frequency of bilateralism is only 6%, prophylactic unaffected side oophorectomy need not be done.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130030"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3922368/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40300438","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}

{"title":"Non-islet cell tumor-induced hypoglycemia: a report of five cases and brief review of the literature.","authors":"Pinaki Dutta,&nbsp;Anuradha Aggarwal,&nbsp;Yashpal Gogate,&nbsp;Uma Nahar,&nbsp;Viral N Shah,&nbsp;Mandeep Singla,&nbsp;N Khandelwal,&nbsp;Anil Bhansali","doi":"10.1530/EDM-13-0046","DOIUrl":"https://doi.org/10.1530/EDM-13-0046","url":null,"abstract":"<p><strong>Unlabelled: </strong>We describe the clinical presentation, diagnostic and management issues in five cases of non-islet cell tumor hypoglycemia (NICTH), diagnosed at a tertiary care institute over a period of 15 years. The clinical, laboratory, and histopathological findings of these patients along with diagnostic utility of IGF2:IGF1 ratio are discussed. The mean age of presentation was 52 years, with a male predominance (3:2). Three patients presented with recurrent episodes of fasting hypoglycemia and it was detected in other two patients during hospitalization. Two patients had acromegaloid features that regressed following treatment. One patient had hypokalemia. Low levels of insulin, C-peptide, GH, and IGF1 were invariably found in all. The IGF2 level was elevated in only one patient; however, IGF2:IGF1 ratio was more than 10 in four of the five patients. The mean tumor size was 16.4 cm and mean weight was 3.6 kg. Four patients had mesenchymal tumors and one had epithelial tumor. NICTH is a rare cause of hypoglycemia. Hypoinsulinemic hypoglycemia with low IGF1 and IGF2:IGF1 ratio more than 10 is suggestive of this entity.</p><p><strong>Learning points: </strong>NICTH should be considered in patients presenting with tumor of mesenchymal origin and hypoglycemia.Hypoinsulinemic hypoglycemia with low IGF1 is a strong biochemical evidence of NICTH.IGF2:IGF1 ratio of more than 10 is a complementary investigation in the absence of an assay facility for IGF2.</p>","PeriodicalId":520608,"journal":{"name":"Endocrinology, diabetes & metabolism case reports","volume":" ","pages":"130046"},"PeriodicalIF":0.9,"publicationDate":"2013-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1530/EDM-13-0046","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40298893","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}