Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis
Background. Langerhans Cell Histiocytosis (LCH) is a rare inflammatory neoplasm characterized by an infiltration of organs by Langerin + (CD207+) and CD1a+ histiocytes. Diabetes insipidus is a frequent manifestation of the disease, while diabetes mellitus is very rare. We report the first case of a...
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| Format: | Article |
| Language: | English |
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Wiley
2019-01-01
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| Series: | Case Reports in Endocrinology |
| Online Access: | http://dx.doi.org/10.1155/2019/2719364 |
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| author | Mathilde Sollier Marine Halbron Jean Donadieu Ahmed Idbaih Fleur Cohen Aubart Corinne Vigouroux Martine Auclair Olivier Bourron Marie Bastin Géraldine Béra Philippe Touraine Jacques Young Héléna Mosbah Agnès Hartemann Fabrizio Andreelli Chloé Amouyal |
| author_facet | Mathilde Sollier Marine Halbron Jean Donadieu Ahmed Idbaih Fleur Cohen Aubart Corinne Vigouroux Martine Auclair Olivier Bourron Marie Bastin Géraldine Béra Philippe Touraine Jacques Young Héléna Mosbah Agnès Hartemann Fabrizio Andreelli Chloé Amouyal |
| author_sort | Mathilde Sollier |
| collection | DOAJ |
| description | Background. Langerhans Cell Histiocytosis (LCH) is a rare inflammatory neoplasm characterized by an infiltration of organs by Langerin + (CD207+) and CD1a+ histiocytes. Diabetes insipidus is a frequent manifestation of the disease, while diabetes mellitus is very rare. We report the first case of a 20-year-old man suffering from hypothalamopituitary histiocytosis and diabetes mellitus with serum anti-insulin receptor antibodies. Case Presentation. A 20-year-old patient was admitted for the evaluation of growth delay and hyperphagia. HbA1c level and fasting blood glucose were in the normal range. The diagnosis of hypothalamopituitary histiocytosis was based on histological features after biopsy of a large suprachiasmatic lesion identified on magnetic resonance imaging (MRI). Association of vinblastine and purinethol was started followed by a second-line therapy by cladribine. During the follow-up, the patient was admitted for recurrence of hyperglycemic states and extreme insulin resistance. The screening for serum anti-insulin receptor antibodies was positive. Each episode of hyperglycemia appeared to be correlated with tumoral activity and increase in serum anti-insulin receptor antibodies and appeared to be improved when the disease was controlled by chemotherapy. Conclusion. We report the first description of a hypothalamopituitary histiocytosis associated with serum anti-insulin receptor antibodies, extreme insulin resistance, and diabetes. Parallel evolution of glucose levels and serum anti-insulin receptor antibodies seemed to be the consequence of immune suppressive properties of cladribine. |
| format | Article |
| id | doaj-art-fcd5f624168741899d3022bc411784cd |
| institution | Kabale University |
| issn | 2090-6501 2090-651X |
| language | English |
| publishDate | 2019-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Endocrinology |
| spelling | doaj-art-fcd5f624168741899d3022bc411784cd2025-02-03T05:48:19ZengWileyCase Reports in Endocrinology2090-65012090-651X2019-01-01201910.1155/2019/27193642719364Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells HistiocytosisMathilde Sollier0Marine Halbron1Jean Donadieu2Ahmed Idbaih3Fleur Cohen Aubart4Corinne Vigouroux5Martine Auclair6Olivier Bourron7Marie Bastin8Géraldine Béra9Philippe Touraine10Jacques Young11Héléna Mosbah12Agnès Hartemann13Fabrizio Andreelli14Chloé Amouyal15Diabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceHemato-Oncology Dpt, Trousseau Hospital, APHP, 75012 Paris, FranceSorbonne Université, Inserm, CNRS, UMRS 1127, Institut du Cerveau et de la Moelle épinière, ICM, AP-HP, Hôpitaux Universitaires La Pitié Salpêtrière -Charles Foix, Service de Neurologie 2, Mazarin, 75013 Paris, FranceDepartment of Internal Medicine 2, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceInserm UMRS 938, Centre de Recherche Saint–Antoine, Sorbonne Université, Reference Center for Rare Diseases of Insulin Secretion and Insulin Sensitivity, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Paris, APHP, Saint Antoine Hospital, Paris, FranceInserm UMRS 938, Centre de Recherche Saint–Antoine, Sorbonne Université, Reference Center for Rare Diseases of Insulin Secretion and Insulin Sensitivity, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Paris, APHP, Saint Antoine Hospital, Paris, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceNuclear Medicine Dpt, Sorbonne Université, APHP, Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceDepartment of Endocrinology and Reproductive Medicine, Pitié-Salpêtrière Hospital, APHP, Reference Center for Rare Endocrine Diseases of Growth, Reference Center for Rare Gynecological Pathologies, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Sorbonne University, 75013 Paris, FranceReproductive Endocrinology Department, Paris Sud University, APHP, Le Kremlin-Bicêtre, 94275, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceDiabetology-Metabolism Dpt, Sorbonne Université, APHP, Institut Hospitalo–Universitaire de Cardiometabolisme et Nutrition (ICAN), Pitié-Salpêtrière-Charles Foix Hospital, 75013 Paris, FranceBackground. Langerhans Cell Histiocytosis (LCH) is a rare inflammatory neoplasm characterized by an infiltration of organs by Langerin + (CD207+) and CD1a+ histiocytes. Diabetes insipidus is a frequent manifestation of the disease, while diabetes mellitus is very rare. We report the first case of a 20-year-old man suffering from hypothalamopituitary histiocytosis and diabetes mellitus with serum anti-insulin receptor antibodies. Case Presentation. A 20-year-old patient was admitted for the evaluation of growth delay and hyperphagia. HbA1c level and fasting blood glucose were in the normal range. The diagnosis of hypothalamopituitary histiocytosis was based on histological features after biopsy of a large suprachiasmatic lesion identified on magnetic resonance imaging (MRI). Association of vinblastine and purinethol was started followed by a second-line therapy by cladribine. During the follow-up, the patient was admitted for recurrence of hyperglycemic states and extreme insulin resistance. The screening for serum anti-insulin receptor antibodies was positive. Each episode of hyperglycemia appeared to be correlated with tumoral activity and increase in serum anti-insulin receptor antibodies and appeared to be improved when the disease was controlled by chemotherapy. Conclusion. We report the first description of a hypothalamopituitary histiocytosis associated with serum anti-insulin receptor antibodies, extreme insulin resistance, and diabetes. Parallel evolution of glucose levels and serum anti-insulin receptor antibodies seemed to be the consequence of immune suppressive properties of cladribine.http://dx.doi.org/10.1155/2019/2719364 |
| spellingShingle | Mathilde Sollier Marine Halbron Jean Donadieu Ahmed Idbaih Fleur Cohen Aubart Corinne Vigouroux Martine Auclair Olivier Bourron Marie Bastin Géraldine Béra Philippe Touraine Jacques Young Héléna Mosbah Agnès Hartemann Fabrizio Andreelli Chloé Amouyal Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis Case Reports in Endocrinology |
| title | Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis |
| title_full | Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis |
| title_fullStr | Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis |
| title_full_unstemmed | Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis |
| title_short | Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis |
| title_sort | diabetes mellitus extreme insulin resistance and hypothalamic pituitary langerhans cells histiocytosis |
| url | http://dx.doi.org/10.1155/2019/2719364 |
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