{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,27]],"date-time":"2026-05-27T18:15:19Z","timestamp":1779905719637,"version":"3.53.1"},"reference-count":26,"publisher":"Wiley","license":[{"start":{"date-parts":[[2019,6,23]],"date-time":"2019-06-23T00:00:00Z","timestamp":1561248000000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Case Reports in Endocrinology"],"published-print":{"date-parts":[[2019,6,23]]},"abstract":"<jats:p><jats:italic>Background<\/jats:italic>. 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.<jats:italic> Case Presentation<\/jats:italic>. 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.<jats:italic> Conclusion<\/jats:italic>. 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.<\/jats:p>","DOI":"10.1155\/2019\/2719364","type":"journal-article","created":{"date-parts":[[2019,6,23]],"date-time":"2019-06-23T19:31:01Z","timestamp":1561318261000},"page":"1-8","source":"Crossref","is-referenced-by-count":1,"title":["Diabetes Mellitus, Extreme Insulin Resistance, and Hypothalamic-Pituitary Langerhans Cells Histiocytosis"],"prefix":"10.1155","volume":"2019","author":[{"given":"Mathilde","family":"Sollier","sequence":"first","affiliation":[{"name":"Diabetology-Metabolism Dpt, Sorbonne Universit\u00e9, APHP, Institut Hospitalo\u2013Universitaire de Cardiometabolisme et Nutrition (ICAN), Piti\u00e9-Salp\u00eatri\u00e8re-Charles Foix Hospital, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marine","family":"Halbron","sequence":"additional","affiliation":[{"name":"Diabetology-Metabolism Dpt, Sorbonne Universit\u00e9, APHP, Institut Hospitalo\u2013Universitaire de Cardiometabolisme et Nutrition (ICAN), Piti\u00e9-Salp\u00eatri\u00e8re-Charles Foix Hospital, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean","family":"Donadieu","sequence":"additional","affiliation":[{"name":"Hemato-Oncology Dpt, Trousseau Hospital, APHP, 75012 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5290-1204","authenticated-orcid":true,"given":"Ahmed","family":"Idbaih","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9, Inserm, CNRS, UMRS 1127, Institut du Cerveau et de la Moelle \u00e9pini\u00e8re, ICM, AP-HP, H\u00f4pitaux Universitaires La Piti\u00e9 Salp\u00eatri\u00e8re -Charles Foix, Service de 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938, Centre de Recherche Saint\u2013Antoine, Sorbonne Universit\u00e9, Reference Center for Rare Diseases of Insulin Secretion and Insulin Sensitivity, Institut Hospitalo\u2013Universitaire de Cardiometabolisme et Nutrition (ICAN), Paris, APHP, Saint Antoine Hospital, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olivier","family":"Bourron","sequence":"additional","affiliation":[{"name":"Diabetology-Metabolism Dpt, Sorbonne Universit\u00e9, APHP, Institut Hospitalo\u2013Universitaire de Cardiometabolisme et Nutrition (ICAN), Piti\u00e9-Salp\u00eatri\u00e8re-Charles Foix Hospital, 75013 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie","family":"Bastin","sequence":"additional","affiliation":[{"name":"Diabetology-Metabolism Dpt, Sorbonne Universit\u00e9, APHP, Institut Hospitalo\u2013Universitaire de Cardiometabolisme et Nutrition (ICAN), Piti\u00e9-Salp\u00eatri\u00e8re-Charles Foix Hospital, 75013 Paris, 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