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      Insulinoma in an adolescent female with weight loss: a case report and literature review on pediatric insulinomas

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          Abstract

          Summary

          Insulinomas are a rare cause of persistent hypoglycemia in a previously healthy child. In addition to symptoms of hypoglycemia, individuals with insulinomas usually present with a history of incessant caloric intake and weight gain due to a constant need to counter hypoglycemia. In addition to an extensive review of the literature, we report the first case of an insulinoma coexisting with reduced appetite secondary to anorexia nervosa in an adolescent female.

          Learning points

          • Eliciting a detailed family history is important in hypoglycemia cases.

          • Obtaining a thorough dietary intake, weight history, and menstrual cycles (in females) and considering a psychiatric consultation for an eating disorder when indicated.

          • Although rare in the pediatric population, multiple endocrine neoplasia type 1 syndrome should be considered in the evaluation of children and adolescents with hypoglycemia who also have a family history of pituitary, pancreatic, and/or parathyroid endocrinopathies.

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          Most cited references10

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          Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1).

          Rajesh V Thakker, Paul J Newey, Gerard V Walls … (2012)
          The aim was to provide guidelines for evaluation, treatment, and genetic testing for multiple endocrine neoplasia type 1 (MEN1). The group, which comprised 10 experts, including physicians, surgeons, and geneticists from international centers, received no corporate funding or remuneration. Guidelines were developed by reviews of peer-reviewed publications; a draft was prepared, reviewed, and rigorously revised at several stages; and agreed-upon revisions were incorporated. MEN1 is an autosomal dominant disorder that is due to mutations in the tumor suppressor gene MEN1, which encodes a 610-amino acid protein, menin. Thus, the finding of MEN1 in a patient has important implications for family members because first-degree relatives have a 50% risk of developing the disease and can often be identified by MEN1 mutational analysis. MEN1 is characterized by the occurrence of parathyroid, pancreatic islet, and anterior pituitary tumors. Some patients may also develop carcinoid tumors, adrenocortical tumors, meningiomas, facial angiofibromas, collagenomas, and lipomas. Patients with MEN1 have a decreased life expectancy, and the outcomes of current treatments, which are generally similar to those for the respective tumors occurring in non-MEN1 patients, are not as successful because of multiple tumors, which may be larger, more aggressive, and resistant to treatment, and the concurrence of metastases. The prognosis for MEN1 patients might be improved by presymptomatic tumor detection and undertaking treatment specific for MEN1 tumors. Thus, it is recommended that MEN1 patients and their families should be cared for by multidisciplinary teams comprising relevant specialists with experience in the diagnosis and treatment of patients with endocrine tumors.
          Article 0 comments Cited 236 times – based on 0 reviews     Review now
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            Functioning insulinoma--incidence, recurrence, and long-term survival of patients: a 60-year study.

            F John Service, M Molly McMahon, PETER C. O'BRIEN … (1991)
            For the 60-year period from 1927 through 1986, we assessed the incidence, recurrence, and long-term survival among all Mayo Clinic patients with histologically confirmed functioning insulinoma. With use of the complete medical record system at Mayo and the comprehensive epidemiologic data base of residents of Olmsted County, Minnesota, we found 224 patients in whom an initial pancreatic exploration at Mayo had confirmed the presence of insulinoma. The median age (and range) of these patients at surgical diagnosis was 47 (8 to 82) years, and 59% were female patients. During the study period, eight cases of insulinoma occurred among residents of Olmsted County; their age and gender distributions were similar to those of the total cohort. The incidence of insulinoma among residents of Olmsted County increased during the study period to a stable level during the last 2 decades of 4 cases per 1 million person-years. For the total cohort, 7.6% had multiple endocrine neoplasia type I (MEN I), and 5.8% had malignant insulinoma. The risk of recurrence was greater among patients with MEN I (21% at 10 and 20 years) than in those without MEN I (5% at 10 years and 7% at 20 years). Although survival of the total cohort was not significantly impaired, it was significantly worse than expected for patients with malignant insulinoma (29% versus 88% expected at 10 years postoperatively). We conclude that insulinoma is less rare than previously suspected. After successful surgical removal, the long-term risk of recurrent insulinoma is relatively high in patients with MEN I; for patients with benign disease, the long-term survival is normal.
            Article 0 comments Cited 195 times – based on 0 reviews     Review now
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              Mechanism of awareness of hypoglycemia. Perception of neurogenic (predominantly cholinergic) rather than neuroglycopenic symptoms.

              P Cryer, C Havlin, Tara Craft … (1993)
              We sought 1) to determine which symptoms of hypoglycemia are reproducible, 2) to pharmacologically distinguish neurogenic (autonomic) from neuroglycopenic symptoms, and 3) to test the hypothesis that awareness of hypoglycemia is the result of perception of neurogenic rather than neuroglycopenic symptoms. Awareness of hypoglycemia and 19 symptoms were quantitated in 10 normal, young adults, each studied on four occasions in random sequence, during 1) clamped euglycemia (approximately 5 mM), 2) clamped hypoglycemia (approximately 2.5 mM), 3) clamped hypoglycemia with combined alpha- and beta-adrenergic blockade (phentolamine and propranolol), and 4) clamped hypoglycemia with pan-autonomic blockade (phentolamine, propranolol and atropine). Significant (ANOVA, P < 0.001) treatment effects on the awareness of hypoglycemia ("blood sugar low") were noted. No change occurred in the score for this during euglycemia, but the mean +/- SE increase was 2.1 +/- 0.4 during hypoglycemia. This increase was not reduced significantly by adrenergic blockade (1.6 +/- 0.5), but was reduced significantly and substantially (approximately 70%) by pan-autonomic blockade (0.6 +/- 0.3). Significant neurogenic symptoms included shaky/tremulous (P < 0.001), heart pounding (P < 0.001), and nervous/anxious (P = 0.002), all adrenergic; and sweaty (P < 0.001), hungry (P < 0.001), and tingling (P = 0.009), all cholinergic. Significant neuroglycopenic symptoms, those produced by hypoglycemia but not reduced by pan-autonomic blockade, included warm (P < 0.001), weak (P = 0.011), difficulty thinking/confused (P = 0.004), and tired/drowsy (P = 0.003). We conclude that muscarinic cholinergic mechanisms mediate an important and previously uncharacterized component of the neurogenic symptoms of hypoglycemia and awareness of hypoglycemia.(ABSTRACT TRUNCATED AT 250 WORDS)
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                Author and article information

                Journal
                Journal ID (nlm-ta): Endocrinol Diabetes Metab Case Rep
                Journal ID (iso-abbrev): Endocrinol Diabetes Metab Case Rep
                Journal ID (hwp): EDM
                Title: Endocrinology, Diabetes & Metabolism Case Reports
                Publisher: Bioscientifica Ltd (Bristol )
                ISSN (Electronic): 2052-0573
                Publication date (Electronic): 11 February 2022
                Publication date Collection: 2022
                Volume: 2022
                Electronic Location Identifier: 21-0206
                Affiliations
                [1 ]Division of Endocrinology, Department of Pediatrics , Children’s Healthcare of Atlanta, Emory University School of Medicine, Atlanta, Georgia, USA
                [2 ]Division of Endocrinology , Department of Pediatrics, Connecticut Childrens Medical Center, Farmington, Connecticut, USA
                [3 ]Aflac Cancer and Blood Disorders Center of Children’s Healthcare of Atlanta , Atlanta, Georgia, USA
                Author notes
                Correspondence should be addressed to P Gupta; Email: pranav.gupta@ 123456emory.edu
                Author information
                http://orcid.org/0000-0003-1205-4391
                Article
                Publisher ID: EDM210206
                DOI: 10.1530/EDM-21-0206
                PMC ID: 8942319
                PubMed ID: 35264462
                SO-VID: 743a2fd4-906d-497c-b197-6bf2deece1e3
                Copyright © © The authors
                License:

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                Date received : 07 February 2022
                Date accepted : 11 February 2022
                Categories
                Subject: Adolescent/young adult
                Subject: Female
                Subject: Asian - Indian
                Subject: United States
                Subject: Pancreas
                Subject: Parathyroid
                Subject: Pituitary
                Subject: Endocrine-Related Cancer
                Subject: Genetics
                Subject: Paediatrics
                Subject: Psychology/Psychiatry
                Subject: Surgery
                Subject: Unique/Unexpected Symptoms or Presentations of a Disease
                Subject: Unique/Unexpected Symptoms or Presentations of a Disease

                Keywords: adolescent/young adult,female,asian - indian,united states,pancreas,parathyroid,pituitary,endocrine-related cancer,genetics,paediatrics,psychology/psychiatry,surgery,unique/unexpected symptoms or presentations of a disease,march,2022
                Data availability:
                Keywords: adolescent/young adult, female, asian - indian, united states, pancreas, parathyroid, pituitary, endocrine-related cancer, genetics, paediatrics, psychology/psychiatry, surgery, unique/unexpected symptoms or presentations of a disease, march, 2022

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