CASE REPORTS

NOVEMBER 2019 • VOLUME 104 • NUMBER 11

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CASE REPORTS

5421 Biochemically Silent Sympathetic Paraganglioma, Pheochromocytoma, or Metastatic Disease in SDHDMutation CarriersKoen M. A. Dreijerink, Johannes A. Rijken, C. J. Compaijen, Henri J. L. M. Timmers,Anouk N. A. van der Horst-Schrivers, Rachel S. van Leeuwaarde, P. Sytze van Dam, C. Rene Leemans,Eveline W. C. M. van Dam, Chris Dickhoff, Charlotte J. Dommering, Pim de Graaf, G. J. C. Zwezerijnen,Paul van der Valk, C. Willemien Menke-Van der Houven van Oordt, Erik F. Hensen, Eleonora P. M. Corssmit,and E. Marelise W. EekhoffPrecis: Ten SDHD patients developed biochemically silent sympathetic paraganglioma, pheochromocytoma, ormetastases. Intensified imaging may be warranted irrespective of hormonal activity in SDHD patients.

5427 Hypernatremic Hydrophobic Transient Adipsia Without Organic or Severe Psychiatric DisorderAleida Rodriguez, Leon Fogelfeld, and Gary RobertsonPrecis: Psychogenic adipsia can occur in those with mild depression.

5462 Adrenalectomy Completely Cured Hypertension in Patients With Familial Hyperaldosteronism Type I WhoHad Somatic KCNJ5 MutationYu-Fang Lin, Kang-Yung Peng, Chia-Hui Chang, Ya-Hui Hu, Vin-Cent Wu, Jeff S. Chueh, and Kwan-Dun WuPrecis: Two siblings of a Taiwanese family with GRA were found to have adrenal adenomas and somatic KCNJ5mutation. Complete clinical cure was achieved after unilateral adrenalectomy.

5478 Pasireotide and Pegvisomant Combination Treatment in Acromegaly Resistant to Second-Line Therapies:A Longitudinal StudySabrina Chiloiro, Chiara Bima, Tommaso Tartaglione, Antonella Giampietro, Marco Gessi, Liverana Lauretti,Carmelo Anile, Cesare Colosimo, Guido Rindi, Alfredo Pontecorvi, Laura De Marinis, and Antonio BianchiPrecis: Acromegaly treated with pasireotide/pegvisomant combination therapy after resistance to first- andsecond-line therapies is described and compared with acromegaly responsive to second-line therapies.

5499 Checkpoint Inhibitor–Associated Autoimmune Diabetes Is Distinct From Type 1 DiabetesVenessa H. M. Tsang, Rachel T. McGrath, Roderick J. Clifton-Bligh, Richard A. Scolyer, Valerie Jakrot,Alexander D. Guminski, Georgina V. Long, and Alexander M. MenziesPrecis: CIADM represents a new class and increasingly common cause of autoimmune diabetes that is distinct fromT1D in onset and cause; it is characterized by sudden complete b-cell failure after immune checkpointinhibitor therapy.

P5 Pediatrics

THE JOURNALOF CLINICAL ENDOCRINOLOGY&METABOLISM (Print ISSN 0021-972X; Online ISSN 1945-7197; USPS 278-920) is an official journal of theEndocrine Society, published monthly by the Endocrine Society, 2055 L Street NW, Suite 600, Washington, DC 20036. Periodicals postage paid atWashington, DC, and at additional mailing offices. Printed in the USA. POSTMASTER: Send address changes to The Journal of Clinical Endocrinology &Metabolism, 2055 L Street NW, Suite 600, Washington, DC 20036. Printed on acid-free paper. Table of Contents and abstracts for each issue availableon the issuemail date at https://academic.oup.com/jcem. 2019 Subscription information is located on the Resources page. Copyright© 2019 Endocrine Society.

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META-ANALYSIS

5325 Improvement but No Normalization of Quality of Life and Cognitive Functioning After Treatment ofCushing SyndromeLeonie H. A. Broersen, Cornelie D. Andela, Olaf M. Dekkers, Alberto M. Pereira, and Nienke R. BiermaszPrecis: This systematic review shows that treatment of Cushing syndrome improves quality of life and cognitivefunctioning, without achieving normalization compared with healthy controls.

5349 Caffeine, Type of Coffee, and Risk of Ovarian Cancer: A Dose–Response Meta-Analysis ofProspective StudiesAsma Salari-Moghaddam, Alireza Milajerdi, Pamela J. Surkan, Bagher Larijani, and Ahmad EsmaillzadehPrecis: Coffee and caffeine consumption have been examined in relation to the risk of ovarian cancer. We found nosignificant association among caffeine intake or coffee type and the risk of ovarian cancer.

MINI-REVIEWS

4949 Disturbances in Insulin–Glucose Metabolism in Patients With Advanced Renal Disease With andWithout DiabetesMarie-Noel Rahhal, Naser Eddin Gharaibeh, Leili Rahimi, and Faramarz Ismail-BeigiPrecis: In patients with advanced CKD, changes in several factors underlying insulin–glucose dynamics, in additionto decreased renal insulin clearance, necessitate detailed attention to insulin dosing.

5274 Therapeutic Effects of Endogenous Incretin Hormones and Exogenous Incretin-Based Medications in SepsisFaraaz Ali Shah, Hussain Mahmud, Teresa Gallego-Martin, Michael J. Jurczak, Christopher P. O’Donnell,and Bryan J. McVerryPrecis: This manuscript provides a bench-to-bedside review of the evidence supporting a potential therapeutic rolefor endogenous incretin hormones and exogenous incretin-derived medications in sepsis.

5299 A Narrative Review of Placental Contribution to Adverse Pregnancy Outcomes in Women With PolycysticOvary SyndromeAngela S. Kelley, Yolanda R. Smith, and Vasantha PadmanabhanPrecis: In this review, we summarize current knowledge of placental dysfunction in human and animal models ofPCOS and propose future directions for placental research in the population with PCOS.

5406 Antidiuretic Hormone and Serum Osmolarity Physiology and Related Outcomes: What Is Old, What Is New,and What Is Unknown?Mehmet Kanbay, Sezen Yilmaz, Neris Dincer, Alberto Ortiz, Alan A. Sag, Adrian Covic,Laura G. Sanchez-Lozada, Miguel A. Lanaspa, David Z. I. Cherney, Richard J. Johnson, and Baris AfsarPrecis: Recent studies show that increased serum osmolarity and high serum vasopressin levels might be novelindependent risk factors for the development of hypertension and kidney and cardiovascular disease.

5431 Angiogenic Abnormalities in Diabetes Mellitus: Mechanistic and Clinical AspectsGian Paolo Fadini, Mattia Albiero, Benedetta Maria Bonora, and Angelo AvogaroPrecis: Angiogenesis is a fundamental biological process in health and disease. We herein review alterations ofangio/vasculogenesis in diabetes and how they contribute to disease onset and complications.

PERSPECTIVE

5372 Molecular Mechanisms of Insulin Resistance in Polycystic Ovary Syndrome: Unraveling the Conundrum inSkeletal Muscle?Nigel K. Stepto, Alba Moreno-Asso, Luke C. McIlvenna, Kirsty A. Walters, and Raymond J. RodgersPrecis: This perspective explores current understanding of the molecular mechanisms of PCOS-specific insulinresistance in skeletal muscle. Here, we highlight major gaps in knowledge and new opportunities.

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CLINICAL RESEARCH ARTICLES

4941 Stage II Differentiated Thyroid Cancer Is a High-Risk Disease in Patients <45/55 Years OldZeming Liu, Xiaopei Shen, Rengyun Liu, Guangwu Zhu, Tao Huang, and Mingzhao XingPrecis: This study showed a sharply higher mortality risk of stage II thyroid cancer in patients,45/55 years old thanin older patients, emphasizing the importance of age in differentiating stage II DTC.

4967 Illicit Upregulation of Serotonin Signaling Pathway in Adrenals of Patients With High Plasma or Intra-AdrenalACTH LevelsJulie Le Mestre, Celine Duparc, Yves Reznik, Fideline Bonnet-Serrano, Philippe Touraine, Olivier Chabre,Jacques Young, Mari Suzuki, Mathilde Sibony, Françoise Gobet, Constantine A. Stratakis, Gerald Raverot,Jerome Bertherat, Herve Lefebvre, and Estelle LouisetPrecis: The serotonin signaling pathway is upregulated in adrenal tissues exposed to high levels of ACTH orharboring constitutive activation of the cAMP protein kinase A pathway.

4981 Thyroid Cancer Patients With No Evidence of Disease: The Need for Repeat Neck UltrasoundGiorgio Grani, Valeria Ramundo, Rosa Falcone, Livia Lamartina, Teresa Montesano, Marco Biffoni,Laura Giacomelli, Marialuisa Sponziello, Antonella Verrienti, Martin Schlumberger, Sebastiano Filetti,and Cosimo DurantePrecis: Neck lymph node recurrences are rare in low/intermediate-risk PTCs with no evidence of disease aftersurgery. Subsequent sonography can be deferred if the serum Tg level is ,1 ng/mL.

4990 Hypothyroidism and Its Association With Sleep Apnea Among Adults in the United States:NHANES 2007–2008Subhanudh Thavaraputta, Jeff A. Dennis, Passisd Laoveeravat, Kenneth Nugent, and Ana M. RivasPrecis: This is a population study conducted to investigate the association between hypothyroidism and sleep apneain the United States. A significant association between these two conditions was found.

4998 Clinical Importance of Free Thyroxine Concentration Decline After Radiotherapy for Pediatric and AdolescentBrain TumorsLaura van Iersel, Jiahui Xu, Brian S. Potter, Heather M. Conklin, Hui Zhang, Wassim Chemaitilly,Hanneke M. van Santen, and Thomas E. MerchantPrecis: In a longitudinal study, declining FT4 concentrations, suggestive for central hypothyroidism, were associatedwith adverse physical health outcomes in children with brain tumor after radiotherapy.

5008 Genetic Association Study of Eight Steroid Hormones and Implications for Sexual Dimorphism of CoronaryArtery DiseaseJanne Pott, Yoon Ju Bae, Katrin Horn, Andrej Teren, Andreas Kuhnapfel, Holger Kirsten, Uta Ceglarek,Markus Loeffler, Joachim Thiery, Jurgen Kratzsch, and Markus ScholzPrecis: Genome-wide analysis of eight steroid hormones revealed 15 novel loci involving several steroid-convertingenzymes. Mendelian randomization found four steroids causally related to coronary artery disease.

5024 Can Menopause Prediction Be Improved With Multiple AMH Measurements? Results From the ProspectiveDoetinchem Cohort StudyAnnelien C. de Kat, Yvonne T. van der Schouw, Marinus J. C. Eijkemans, Simone L. Broer,W. M. Monique Verschuren, and Frank J. M. BroekmansPrecis: Compared with a single AMH measurement, the use of serial AMH measurements does not improve theprediction of menopause.

5032 Maternal and Offspring Genetic Risk of Type 2 Diabetes and Offspring Birthweight Among AfricanAncestry PopulationsMohammad L. Rahman, Deepika Shrestha, Tsegaselassie Workalemahu, Jing Wu, Chunming Zhu,Cuilin Zhang, and Fasil Tekola-AyelePrecis:Maternal and offspring genetic risks of type 2 diabetes were independently and differentially associated withoffspring birthweight among African ancestry populations.

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P 5043 Maternal Heavy Metal Exposure, Thyroid Hormones, and Birth Outcomes: A Prospective Cohort StudyXiaojie Sun, Wenyu Liu, Bin Zhang, Xiantao Shen, Chen Hu, Xiaomei Chen, Shuna Jin, Yangqian Jiang,Hongxiu Liu, Zhongqiang Cao, Wei Xia, Shunqing Xu, and Yuanyuan LiPrecis: Changes in maternal thyroid hormone were postulated to mediate the associations between metal exposureand birth outcomes. We found maternal FT3 or FT3/FT4 ratio were mediators in these associations.

5056 Patients With Aldolase B Deficiency Are Characterized by Increased Intrahepatic Triglyceride ContentNynke Simons, François-Guillaume Debray, Nicolaas C. Schaper, M. Eline Kooi, Edith J. M. Feskens,Carla E. M. Hollak, Lucas Lindeboom, Ger H. Koek, Judith A. P. Bons, Dirk J. Lefeber, Leanne Hodson,Casper G. Schalkwijk, Coen D. A. Stehouwer, David Cassiman, and Martijn C. G. J. BrouwersPrecis: Patients with aldolase B deficiency, a defect in fructose metabolism, are characterized by increased hepaticfat content and glucose intolerance compared with age-, sex-, and BMI-matched controls.

5065 Glucocorticoid Activity of Adrenal Steroid Precursors in Untreated Patients With CongenitalAdrenal HyperplasiaManon Engels, Karijn J. Pijnenburg-Kleizen, Agustini Utari, Sultana M. H. Faradz, Sandra Oude-Alink,Antonius E. van Herwaarden, Paul N. Span, Fred C. Sweep, and Hedi L. Claahsen-van der GrintenPrecis: Increased adrenal steroid precursors that accumulate in untreated patients with congenital adrenalhyperplasia have glucocorticoid activity in vitro.

5073 Health-Related Quality of Life in Turner Syndrome and the Influence of Growth Hormone Therapy: A20-Year Follow-UpEmily Krantz, Kerstin Landin-Wilhelmsen, Penelope Trimpou, Inger Bryman, and Ulla WidePrecis: In this 20-year follow-up study of HRQoL of women with Turner syndrome, no association between HRQoLand previous GH treatment was found, calling the relevance of GH treatment into question.

5084 Relationship Between Number of Multiple Risk Factors and Coronary Artery Disease Risk With and WithoutDiabetes MellitusMayuko Yamada-Harada, Kazuya Fujihara, Taeko Osawa, Masahiko Yamamoto, Masanori Kaneko,Masaru Kitazawa, Yasuhiro Matsubayashi, Takaho Yamada, Nauta Yamanaka, Hiroyasu Seida, Wataru Ogawa,and Hirohito SonePrecis: Composite control of modifiable risk factors, such as glucose, BP, or current smoking, was sufficient to bringCAD risk in patients with DM below that for the non-DM group who had two targets achieved.

5091 The Role of 68Ga-DOTA-Octreotate PET/CT in Follow-Up of SDH-Associated Pheochromocytomaand ParagangliomaGrace Kong, Tess Schenberg, Christopher J. Yates, Alison Trainer, Nirupa Sachithanandan, Amir Iravani,Aravind Ravi Kumar, Michael S. Hofman, Tim Akhurst, Michael Michael, and Rodney J. HicksPrecis: We report the incremental diagnostic value with consequent management impact of GaTate PET/CTcompared with MRI or CT in follow-up of patients with SDHx pheochromocytoma/paraganglioma.

5120 Comorbidities and Survival in Patients With Lipodystrophy: An International Chart Review StudyBaris Akinci, Elif A. Oral, Adam Neidert, Diana Rus, Wendy Y. Cheng, Philippe Thompson-Leduc,Hoi Ching Cheung, Pamela Bradt, Maria Cristina Foss de Freitas, Renan Magalh~aes Montenegro, Jr.,Virgınia Oliveira Fernandes, Elaine Cochran, and Rebecca J. BrownPrecis: This international chart review of patients with lipodystrophy syndromes describes the disease natural historyand highlights differences between generalized and partial forms.

5136 Safety and Efficacy of 5 Years of Treatment With Recombinant Human Parathyroid Hormone in AdultsWith HypoparathyroidismMichael Mannstadt, Bart L. Clarke, John P. Bilezikian, Henry Bone, Douglas Denham, Michael A. Levine,Munro Peacock, Jeffrey Rothman, Dolores M. Shoback, Mark L. Warren, Nelson B. Watts, Hak-Myung Lee,Nicole Sherry, and Tamara J. VokesPrecis: Treatment of hypoparathyroidism rhPTH(1-84) for 5 years had a safety profile consistent with other studiesand improved key biochemical parameters, including normalization of urinary calcium level.


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5148 Medroxyprogesterone Acetate in Gender-Affirming Therapy for Transwomen: Results Froma Retrospective StudyJaison Jain, Daniel Kwan, and Michelle ForcierPrecis: A retrospective analysis of transwomen using MPA found minimal side effects, unchanged E levels, anda decline in testosterone.

P 5157 miRNA Profiles in Extracellular Vesicles From Serum Early in Pregnancies Complicated by GestationalDiabetes MellitusVirginie Gillet, Annie Ouellet, Yulia Stepanov, Rodosthenis S. Rodosthenous, Erika Kathe Croft, Kasey Brennan,Nadia Abdelouahab, Andrea Baccarelli, and Larissa Takser

5170 Blood Pressure Profile, Catecholamine Phenotype, and Target Organ Damage inPheochromocytoma/ParagangliomaOndrej Petrak, Jan Rosa, Robert Holaj, Branislav Strauch, Zuzana Kratka, Jan Kvasnicka, Judita Klımova,Petr Waldauf, Barbora Hamplova, Alice Markvartova, Kvetoslav Novak, David Michalsky, Jirı Widimsky, Jr.,and Tomas ZelinkaPrecis: Predominantly pure noradrenaline, without concomitant adrenaline overproduction, has a long-term effecton the cardiovascular system in pheochromocytoma/functional paraganglioma.

P 5183 Endocrine and Metabolic Disorders in Survivors of Childhood Cancers and Health-Related Quality of Life andPhysical ActivityKamnesh R. Pradhan, Yan Chen, Sogol Moustoufi-Moab, Kevin Krull, Kevin C. Oeffinger, Charles Sklar,Gregory T. Armstrong, Kirsten K. Ness, Leslie Robison, Yutaka Yasui, and Paul C. NathanPrecis: Chronic endocrine and metabolic conditions adversely affected health-related quality of life and physicalactivity among adult survivors of childhood cancers followed in a large North American cohort.

5195 Body Mass Index and Mortality in Individuals With Type 1 DiabetesEmma H. Dahlstrom, Niina Sandholm, Carol M. Forsblom, Lena M. Thorn, Fanny J. Jansson, Valma Harjutsalo,and Per-Henrik Groop, on behalf of the FinnDiane Study GroupPrecis: In this prospective study on BMI and mortality in type 1 diabetes, we found a nonlinear relationship betweenBMI and mortality that was modified by diabetes-onset age, sex, and diabetic nephropathy.

5205 Impaired 11b-Hydroxysteroid Dehydrogenase Type 2 in Glucocorticoid-Resistant PatientsGeraldine Vitellius, Brigitte Delemer, Philippe Caron, Olivier Chabre, Jerome Bouligand, Eric Pussard,Severine Trabado, and Marc LombesPrecis: Glucocorticoid receptor (GR) regulates HSD11B2 expression and activity, accounting forpseudohypermineralocorticism in patients harboring heterozygous GR loss-of-function mutations.

5217 Morning (Fasting) vs Afternoon Resistance Exercise in Individuals With Type 1 Diabetes: A RandomizedCrossover StudySaeed Reza Toghi-Eshghi and Jane E. YardleyPrecis: Morning RE performed while fasting increases blood glucose, whereas RE performed later in the daydecreases blood glucose in individuals with type 1 diabetes.

5225 Transcriptome Profiling of Adipose Tissue Reveals Depot-Specific Metabolic Alterations Among Patientswith Colorectal CancerMariam Haffa, Andreana N. Holowatyj, Mario Kratz, Reka Toth, Axel Benner, Biljana Gigic, Nina Habermann,Petra Schrotz-King, Jurgen Bohm, Hermann Brenner, Martin Schneider, Alexis Ulrich, Esther Herpel,Peter Schirmacher, Beate K. Straub, Johanna Nattenmuller, Hans-Ulrich Kauczor, Tengda Lin, Claudia R. Ball,Cornelia M. Ulrich, Hanno Glimm, and Dominique SchererPrecis: Transcriptome profiling of adipose tissue reveals depot-specific alterations in patients with colorectal cancerand shows that BMI poorly correlates with fat mass–related changes in visceral adipose tissue.


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5238 Influences of Hypoxia Exercise on Whole-Body Insulin Sensitivity and Oxidative Metabolism inOlder IndividualsKristine Chobanyan-Jurgens, Renate J. Scheibe, Arne B. Potthast, Markus Hein, Andrea Smith, Robert Freund,Uwe Tegtbur, Anibh M. Das, Stefan Engeli, Jens Jordan, and Sven HaufePrecis: We observed that 8 weeks of hypoxia training did not further enhance insulin sensitivity and markers ofoxidative phosphorylation compared with endurance training under normoxia in older women and men.

5253 Efficacy and Safety of Alirocumab 300 mg Every 4 Weeks in Individuals With Type 2 Diabetes on MaximallyTolerated StatinDirk Muller-Wieland, Daniel J. Rader, Patrick M. Moriarty, Jean Bergeron, Gisle Langslet, Kausik K. Ray,Garen Manvelian, Desmond Thompson, Maja Bujas-Bobanovic, and Eli M. RothPrecis: Alirocumab 300 mg Q4W was generally well tolerated and efficacious for improving lipid levels (e.g., LDL-Cand non–HDL-C) in individuals with T2DM receiving maximally tolerated statin therapy.

5263 Identification of Haptoglobin as a Readout of rhGH Therapy in GH DeficiencyMarilisa De Feudis, Gillian Elisabeth Walker, Giulia Genoni, Marcello Manfredi, Emanuela Agosti,Mara Giordano, Marina Caputo, Luisa Di Trapani, Emilio Marengo, Gianluca Aimaretti, Nicoletta Filigheddu,Simonetta Bellone, Gianni Bona, and Flavia ProdamPrecis: Proteomics and experiments in HepG2 cells showed that haptoglobin is dysregulated in GHD. GH treatmentinhibits Hp secretion through mechanisms involving both GH and potentially IGF-1.

P 5285 Umbilical Cord miRNAs in Small-for-Gestational-Age Children and Association With Catch-Up Growth:A Pilot StudyBerta Mas-Pares, Sılvia Xargay-Torrent, Alexandra Bonmatı, Esther Lizarraga-Mollinedo,Jose Maria Martınez-Calcerrada, Gemma Carreras-Badosa, Anna Prats-Puig, Francis de Zegher,Lourdes Iba~nez, Abel Lopez-Bermejo, and Judit BassolsPrecis: We describe several umbilical cord miRNAs nominally associated with catch-up growth and identifymiR-576-5p as a novel potential predictor of increased waist circumference and renal fat at 6 years.

5316 Recovery of the Hypothalamo-Pituitary-Adrenal Axis After Transsphenoidal Adenomectomy forNon–ACTH-Secreting MacroadenomasRiccardo Pofi, Sonali Gunatilake, Victoria Macgregor, Brian Shine, Robin Joseph, Ashley B. Grossman,Andrea M. Isidori, Simon Cudlip, Bahram Jafar-Mohammadi, Jeremy W. Tomlinson, and Aparna PalPrecis: After TSA, pre- and postoperative SST cortisol levels can predict 6-week HPA axis recovery. Recoveryoccurs even 12 months after TSA, demonstrating the need for periodic retesting.

5338 Early Life Exposures to Perfluoroalkyl Substances in Relation to Adipokine Hormone Levels at Birth andDuring ChildhoodColleen Shelly, Philippe Grandjean, Youssef Oulhote, Peter Plomgaard, Ruth Frikke-Schmidt,Flemming Nielsen, Denis Zmirou-Navier, Pal Weihe, and Damaskini ValviPrecis: Prenatal and early postnatal exposures to perfluoroalkyl substances were associated with decreases inadipokine hormone levels over the childhood period in a sex-specific manner.

5360 Oxidative Stress in Response to Saturated Fat Ingestion Is Linked to Insulin Resistance andHyperandrogenism in Polycystic Ovary SyndromeFrank Gonzalez, Robert V. Considine, Ola A. Abdelhadi, and Anthony J. ActonPrecis: We studied the effect of lipid on oxidative stress in PCOS. We found increased obesity-independent lipid-stimulated oxidative stress in PCOS that is linked to insulin resistance and hyperandrogenism.

5382 Androgens During the Reproductive Years: What Is Normal for Women?Marina A. Skiba, Robin J. Bell, Rakibul M. Islam, David J. Handelsman, Reena Desai, and Susan R. DavisPrecis: We have shown that the serum levels of classic and nonclassic C19-steroids decline with age and providedreference ranges for estrogens and androgens stratified by menstrual cycle stage, BMI, and age.

5393 Lavender Products Associated With Premature Thelarche and Prepubertal Gynecomastia: Case Reports andEndocrine-Disrupting Chemical ActivitiesJ. Tyler Ramsey, Yin Li, Yukitomo Arao, Ajanta Naidu, Laurel A. Coons, Alejandro Diaz, and Kenneth S. KorachPrecis: Lavender-containing fragrances may cause premature thelarche and prepubertal gynecomastia.


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5445 Association of Inpatient Glucose Measurements With Amputations in Patients Hospitalized With AcuteDiabetic FootShahar Peled, Rena Pollack, Ofer Elishoov, Amir Haze, and Avivit CahnPrecis:We studied the association of inpatient glucose data with amputations in patients admitted for acute diabeticfoot. Hypoglycemia emerged as an independent risk factor for any and major amputations.

5453 Plasma Agouti-Related Protein Levels in Acromegaly and Effects of Surgical or Pegvisomant TherapyPamela U. Freda, Carlos Reyes-Vidal, Zhezhen Jin, Mya Pugh, Sunil K. Panigrahi, Jeffrey N. Bruce,and Sharon L. WardlawPrecis: Plasma AgRP levels are elevated in active acromegaly and decrease with treatment with surgery orpegvisomant, providing evidence of a stimulatory effect of GH on plasma AgRP in humans.

5467 Metabolomic Urine Profile: Searching for New Biomarkers of SDHx-Associated Pheochromocytomasand ParagangliomasRaquel G. Martins, Luıs G. Gonçalves, Nuno Cunha, and Maria Jo~ao BugalhoPrecis: The metabolomic urine profile of SDHx mutation carriers with pheochromocytomas and paragangliomasis distinct from that of asymptomatic carriers and from that of their sporadic tumor counterparts.

5483 Genetic Variation of the Vitamin D Binding Protein Affects Vitamin D Status and Response toSupplementation in InfantsMaria Enlund-Cerullo, Laura Koljonen, Elisa Holmlund-Suila, Helena Hauta-alus, Jenni Rosendahl,Saara Valkama, Otto Helve, Timo Hytinantti, Heli Viljakainen, Sture Andersson, Outi Makitie,and Minna PekkinenPrecis: In this vitamin D intervention study on 913 infants, GC genotype, diplotype, and haplotype affectedvitamin D status and response to supplementation in participants randomized to receive 30 mg/d.

COMMENTARY

5053 Turning Up the Heat: Endoscopic Ablation of Pancreatic Neuroendocrine NeoplasmsJohannes Hofland, Wouter W. de Herder, and Peter H. Kann

LETTERS TO THE EDITOR

5100 Response to Letter to the Editor: “Gestational Gigantomastia Complicated byPTHrP-Mediated Hypercalcemia”Taher Modarressi, Michael A. Levine, and Amna N. Khan

5102 Response to Letter to the Editor: “Endocrine Treatment of Gender-Dysphoric/Gender-Incongruent Persons:An Endocrine Society Clinical Practice Guideline”Stephen M. Rosenthal, Wylie C. Hembree, Peggy T. Cohen-Kettenis, Louis Gooren, Sabine E. Hannema,Walter J. Meyer, M. Hassan Murad, Joshua D. Safer, Vin Tangpricha, and Guy G. T’Sjoen

5104 Letter to the Editor: “The Inflammation-Based Index Can Predict Response and Improve Patient Selection inNETs Treated With PRRT: A Pilot Study”Johannes Hofland, Tessa Brabander, and Wouter W. de Herder

5106 Letter to the Editor: “Defects in GLP-1 Response to an Oral Challenge Do Not Play a Significant Role in thePathogenesis of Prediabetes”Galina Smushkin, Airani Sathananthan, Chiara Dalla Man, Michael Camilleri, Claudio Cobelli, Robert A. Rizza,and Adrian Vella

5108 Letter to the Editor: “Time to Separate Persistent From Recurrent Differentiated Thyroid Cancer: DifferentConditions With Different Outcomes”Lynn A. Burmeister and Tyler Drake


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5110 Response to Letter to the Editor: “Time to Separate Persistent From Recurrent Differentiated ThyroidCancer: Different Conditions With Different Outcomes”Giulia Sapuppo, Martina Tavarelli, Antonino Belfiore, Riccardo Vigneri, and Gabriella Pellegriti

5112 Letter to the Editor: "CT Characteristics of Pheochromocytoma: Relevance for the Evaluation ofAdrenal Incidentaloma"Zbynek Tudos and Filip Ctvrtlık

5114 Response to Letter to the Editor “CT Characteristics of Pheochromocytoma: Relevance for the Evaluation ofAdrenal Incidentaloma”Letizia Canu and Henri J. L. M. Timmers

5116 Letter to the Editor: “IHH Gene Mutations Causing Short Stature With Nonspecific Skeletal Abnormalities andResponse to Growth Hormone Therapy”Gianluca Tornese

5118 Response to Letter to the Editor: “IHH Gene Mutations Causing Short Stature With Nonspecific SkeletalAbnormalities and Response to Growth Hormone Therapy”Gabriela A. Vasques and Alexander A. L. Jorge

5181 Letter to the Editor: “Pharmacological Management of Osteoporosis in Postmenopausal Women:An Endocrine Society Clinical Practice Guideline”Toshihiro Sugiyama

5249 Letter to the Editor: “Hepatic Insulin Extraction in NAFLD Is Related to Insulin Resistance Rather Than LiverFat Content“Fernando Bril and Kenneth Cusi

5251 Response to Letter to the Editor: “Hepatic Insulin Extraction in NAFLD Is Related to Insulin Resistance RatherThan Liver Fat Content”Kristina M. Utzschneider, Steven E. Kahn, and David C. Polidori

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CLINICAL RESEARCH ARTICLES

5507 The GH Axis in Relation to Accepting an Early Macronutrient Deficit and Outcome of Critically Ill PatientsLisa Van Dyck, Inge Derese, Sarah Vander Perre, Pieter J. Wouters, Michael P. Casaer, Greet Hermans,Greet Van den Berghe, and Ilse VanhorebeekPrecis: Withholding early parenteral nutrition to supplement insufficient enteral nutrition improves clinicaloutcome of critically ill patients despite further suppressing the GH axis.

5519 The Steroid Profile of Adrenal Incidentalomas: Subtyping Subjects With High Cardiovascular RiskGuido Di Dalmazi, Flaminia Fanelli, Guido Zavatta, Silvia Ricci Bitti, Marco Mezzullo, Andrea Repaci,Carla Pelusi, Alessandra Gambineri, Paola Altieri, Cristina Mosconi, Caterina Balacchi, Rita Golfieri,Eugenio Roberto Cosentino, Claudio Borghi, Valentina Vicennati, Renato Pasquali, and Uberto PagottoPrecis: Patients with incidentally discovered adrenal tumors showed different steroid profiles, depending onadrenal gland morphology, hormone secretion, and sex, with implications for blood pressure control.


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5529 Trimethylamine N-Oxide Metabolites in Early Pregnancy and Risk of Gestational Diabetes: A NestedCase-Control StudyXiaoxu Huo, Jing Li, Yun-Feng Cao, Sai-Nan Li, Ping Shao, Junhong Leng, Weiqin Li, Jinnan Liu, Kai Yang,Ronald C. W. Ma, Gang Hu, Zhong-Ze Fang, and Xilin YangPrecis:We found that TMAO, TMA, and its precursors were all associated with GDM. A high conversion rate to TMAand a low rate to TMAO had a synergistic effect on GDM.

5540 The Effects of Amiodarone on Thyroid Function in Pediatric and Young Adult PatientsBrett Barrett, Colin P. Hawkes, Amber Isaza, and Andrew J. BauerPrecis: This is a retrospective review of thyroid function in children and young adults treated with amiodarone. Wefound approximately one-third of patients developed amiodarone-induced thyroid dysfunction.

5547 Endometriosis Triggers Excessive Activation of Primordial Follicles via PI3K-PTEN-Akt-Foxo3 PathwayArisa Takeuchi, Kaori Koga, Erina Satake, Tomoko Makabe, Ayumi Taguchi, Mariko Miyashita,Masashi Takamura, Miyuki Harada, Tetsuya Hirata, Yasushi Hirota, Osamu Yoshino, Osamu Wada-Hiraike,Tomoyuki Fujii, and Yutaka OsugaPrecis: The endometriosis-related reduction of ovarian reserve is at least partially caused by the excessive activationof primordial follicles, which is mediated by the PI3K-PTEN-Akt-Foxo3 pathway.

5555 Lipid Profiling of Peri-implantation Endometrium in Patients With Premature Progesterone Rise in the LateFollicular PhaseJingjie Li, Yue Gao, Lihuan Guan, Huizhen Zhang, Pan Chen, Xiao Gong, Dongshun Li, Xiaoyan Liang,Min Huang, and Huichang BiPrecis: Lipid profiling alterations at the window of implantation in patients with premature P rise on the day of humanchorionic gonadotrophin administration.

5566 Resting Energy Expenditure and Substrate Oxidation in Malnourished Patients With Type 1 GlycogenosisSilvio Buscemi, Davide Noto, Carola Buscemi, Anna Maria Barile, Giuseppe Rosafio, Valentina Settipani,Antonina Giammanco, and Maurizio AvernaPrecis: Type 1 glycogenosis is a rare disease associated with malnutrition. The measured REE was elevated in fivepatients and contributed to explaining the occurrence of malnutrition.

5573 IL13RA2 Is Differentially Regulated in Papillary Thyroid Carcinoma vs Follicular Thyroid CarcinomaSiao Ting Chong, Khee Ming Tan, Catherine Y. L. Kok, Shou Ping Guan, Siang Hui Lai, Cindy Lim,Jiancheng Hu, Charles Sturgis, Charis Eng, Paula Y. P. Lam, and Joanne NgeowPrecis: Our study revealed that IL13RA2 is differentially regulated in thyroid cancer and is correlated with advancedtumor stage.

5585 Characteristics of Slow Progression to Type 1 Diabetes in Children With Increased HLA-ConferredDisease RiskPetra M. Pollanen, Johanna Lempainen, Antti-Pekka Laine, Jorma Toppari, Riitta Veijola, Jorma Ilonen,Heli Siljander, and Mikael KnipPrecis: We set out to characterize slow progression from seroconversion to overt type 1 diabetes. We founddifferences in autoantibodies and the season of birth, but no features specific for slow progression.

5595 Serum Parathyroid Hormone and Risk of Coronary Artery Disease: Exploring Causality UsingMendelian RandomizationHakan Melhus, Karl Michaelsson, and Susanna C. LarssonPrecis: This Mendelian randomization study found no association of genetic predisposition to higher serumparathyroid hormone concentrations with coronary artery disease.

5601 Therapy of Hypoparathyroidism With rhPTH(1-84): A Prospective, 8-Year Investigation of Efficacy and SafetyYu-Kwang Donovan Tay, Gaia Tabacco, Natalie E. Cusano, John Williams, Beatriz Omeragic,Rukshana Majeed, Maximo Gomez Almonte, John P. Bilezikian, and Mishaela R. RubinPrecis: rhPTH(1-84) therapy is safe and effective in hypoparathyroidism. Supplemental calcium and active vitamin Drequirements decrease and serum calcium is maintained; renal function is stable.


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5611 Comparison of Teriparatide and Denosumab in Patients Switching From Long-Term Bisphosphonate UseHouchen Lyu, Sizheng S. Zhao, Kazuki Yoshida, Sara K. Tedeschi, Chang Xu, Sagar U. Nigwekar,Benjamin Z. Leder, and Daniel H. SolomonPrecis: This is a head-to-head comparison study using 14 years of electronic medical records. We comparedteriparatide with denosumab on BMD in patients switching from long-term bisphosphonate.

5621 Circulating Oxytocin Is Genetically Determined and Associated With Obesity and Impaired Glucose ToleranceMark Florian Joachim Weingarten, Markus Scholz, Tobias Wohland, Katrin Horn, Michael Stumvoll,Peter Kovacs, and Anke TonjesPrecis: By measuring circulating oxytocin (OXT), we show that OXT is genetically determined and associated withhuman obesity and impaired glucose tolerance.

5633 HDL-Cholesterol, Its Variability, and the Risk of Diabetes: A Nationwide Population-Based StudySeung-Hwan Lee, Hun-Sung Kim, Yong-Moon Park, Hyuk-Sang Kwon, Kun-Ho Yoon, Kyungdo Han,and Mee Kyoung KimPrecis: Low mean and high variability in HDL-C were independent predictors of diabetes, and an additive effect ofthese two parameters was noted.

5642 Correlation Between Differentiation of Adrenocortical Zones and Telomere Lengths Measured by Q-FISHKeisuke Nonaka, Junko Aida, Kaiyo Takubo, Yuto Yamazaki, Shoichiro Takakuma, Mototsune Kakizaki,Yoko Matsuda, Naoshi Ishikawa, Toshiyuki Ishiwata, Ja-Mun Chong, Tomio Arai, and Hironobu SasanoPrecis: Telomere lengths in the cells from three adrenocortical zones were examined, and distinctive differenceswere detected among these zones in adult but not in pediatric adrenal glands.

5651 Somatic PRKACA Mutations: Association With Transition From Pituitary-Dependent to Adrenal-DependentCushing SyndromeGuido Di Dalmazi, Henri J. L. M. Timmers, Giorgio Arnaldi, Benno Kusters, Marina Scarpelli, Kerstin Bathon,Davide Calebiro, Felix Beuschlein, Ad Hermus, and Martin Reincke

5658 Adrenal Insufficiency After Unilateral Adrenalectomy in Primary Aldosteronism: Long-Term Outcome andClinical ImpactDaniel A. Heinrich, Christian Adolf, Finn Holler, Benjamin Lechner, Holger Schneider, Anna Riester,Nina Nirschl, Lisa Sturm, Xiao Wang, Roland Ladurner, Max Seidensticker, Martin Bidlingmaier,Felix Beuschlein, and Martin ReinckePrecis: We identified many cases of adrenal insufficiency after unilateral adrenalectomy for PA. We recommendperforming postoperative ACTH stimulation tests in all patients undergoing this procedure.

5665 A Cohort Analysis of Clinical and Ultrasound Variables Predicting Cancer Risk in 20,001 ConsecutiveThyroid NodulesTrevor E. Angell, Rie Maurer, Zhihong Wang, Matthew I. Kim, Caroline A. Alexander, Justine A. Barletta,Carol B. Benson, Edmund S. Cibas, Nancy L. Cho, Gerard M. Doherty, Peter M. Doubilet, Mary C. Frates,Atul A. Gawande, Jeff F. Krane, Ellen Marqusee, Francis D. Moore, Jr., Matthew A. Nehs, P. Reed Larsen,and Erik K. AlexanderPrecis: Association between cancer risk and clinical variable analyzed in 20,001 consecutive thyroid nodules: usingpatient and nodule data may provide more precise and individualized nodule risk assessment.

5673 Epigenetic Deregulation of Protocadherin PCDHGC3 in Pheochromocytomas/Paragangliomas AssociatedWith SDHB MutationsCristobal Bernardo-Casti~neira, Nuria Valdes, Lucıa Celada, Andres San Jose Martinez, I. Saenz-de-Santa-Marıa,Gustavo F. Bayon, Agustın F. Fernandez, Marta I. Sierra, Mario F. Fraga, Aurora Astudillo, Paula Jimenez-Fonseca,Juan Carlos Rial, Miguel Angel Hevia, Estrella Turienzo, Carmen Bernardo, Lluis Forga, Isabel Tena,Marıa-Jose Molina-Garrido, Laura Cacho, Carles Villabona, Teresa Serrano, Bartolome Scola, Isabel Chirivella,Maribel del Olmo, Carmen Luz Menendez, Elena Navarro, Marıa Tous, Ana Vallejo, Shobana Athimulam,Irina Bancos, Carlos Suarez, and Marıa-Dolores ChiaraPrecis: We discovered epigenetic silencing of PCDHGC3 in metastatic SDHB-PPGLs associated with short overalldisease survival and the acquisition of metastatic capabilities in tumor cells.


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5693 Low-Normal Thyroid Function Predicts Incident Anemia in the General Population With Euthyroid StatusYeqing Gu, Vu Thi Quynh Chi, Qing Zhang, Li Liu, Ge Meng, Hongmei Wu, Xue Bao, Shunming Zhang,Shaomei Sun, Xing Wang, Ming Zhou, Qiyu Jia, Kun Song, and Kaijun NiuPrecis: The current study demonstrates that thyroid hormones significantly predict future anemia and annualchanges in Hb, even in the euthyroid population.

5703 Effects of Nicotinamide Riboside on Endocrine Pancreatic Function and Incretin Hormones in NondiabeticMen With ObesityOle L. Dollerup, Samuel A. J. Trammell, Bolette Hartmann, Jens J. Holst, Britt Christensen, Niels Møller,Matthew P. Gillum, Jonas T. Treebak, and Niels JessenPrecis: Nicotinamide riboside supplementation for 12 weeks in men with obesity did not affect b-cell function orfasting or OGTT-induced concentrations of glucose, insulin, C-peptide, glucagon, GLP-1, or GIP.

5715 Atg7 Knockdown Reduces Chemerin Secretion in Murine AdipocytesSascha Heinitz, Claudia Gebhardt, Paolo Piaggi, Jacqueline Kruger, Henrike Heyne, Juliane Weiner,John T. Heiker, Michael Stumvoll, Matthias Bluher, Leslie Baier, Assaf Rudich, Peter Kovacs, and Anke TonjesPrecis: Human, mouse, and in vitro studies suggest that ATG7 regulates chemerin secretion in adipose tissue/adipocytes. Elevated adipose autophagy in obesity may therefore manifest by elevated chemerin levels.

5729 Diabetes-Related Fracture Risk Is Different in African Americans Compared With Hispanics and CaucasiansRajesh K. Jain, Mark G. Weiner, Huaqing Zhao, Kevin Jon Williams, and Tamara VokesPrecis: In a retrospective study, DM was significantly associated with the incidence of MOFs in Caucasians andHispanics but not in African Americans.

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CASE REPORTS

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