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Juvenile xanthogranuloma-related central diabetes insipidus and pregnancy: Case report and literature review

A 27-year-old patient, G1P0A0, presented with diabetes insipidus caused by juvenile xanthogranuloma, diagnosed at 11 years of age due to the appearance of xanthomas in the skin and central nervous system (CNS). Skin biopsy evidenced the presence of numerous foamy cytoplasm cells and Touton giant cells, expressing CD68 and S100 protein (Figures 1 and 2). She was followed up at the High-Risk Prenatal Care Unit of the University of Brasília Hospital, together with the Endocrinology Team. On the eighth month of gestation, it was necessary to increase the number of nasal desmopressin puffs from three to seven per day (0.1–2.5 mg/ml); no other alterations were observed in the pre-natal period. A C-section delivery was performed at 39 weeks and 3 days, due to fetal tachysystole observed at cardiotocography. The child was born at term, with Apgar score of 8/8 and weighing 4,015 g. The anatomopathological assessment of the placenta did not evidence any alterations. After delivery, the desmopressin dose was reduced back to three puffs per day. In the post-delivery consultation, the patient was clinically stable.