This case contributes to the limited literature on severe PTH-independent hypercalcaemia during pregnancy and lactation. A 38-year-old primiparous woman presented in early pregnancy with a 6-year history of intermittent mild PTH-independent hypercalcaemia and elevated 1,25-hydroxyvitamin D (1,25VitD) level without identified cause (Figure 1a) or suspicion of calcitriol use.
In early second trimester she developed severe PTH-independent hypercalcaemia (corrected calcium 3.38mmol/L (range 2.1-2.6)) that persisted during her pregnancy (Figure 1b). Her third trimester PTHrp was elevated 42.27pmol/L (non-pregnant range <1.0) with a 1,25VitD (835pmol/L) twice the expected pregnancy range. She had minimal response to intravenous fluids and did not respond to 10-day high-dose glucocorticoid. A modest response to strict low calcium diet (<150g per day) was observed. There was no evidence of end-organ damage until development of pre-eclampsia at 36-weeks. A healthy infant was delivered via emergency caesarean-section at 37-weeks gestation. Maternal hypercalcaemia persisted post-partum and worsened with establishment of lactation. Fluconazole (1-alpha reductase inhibitor) was commenced in attempt to reduce 1,25VitD but ultimately severe hypercalcaemia (3.53mmol/L) necessitated cessation of lactation with subsequent rapid improvement of calcium levels. Initial neonatal hypercalcaemia (day 8 corrected calcium 3.08mmol/L (range 1.85-2.8)) resolved by day 14 post-partum.
Pregnancy and lactation-induced significant elevation of 1,25VitD and PTHrp postulated to be driving the PTH-independent hypercalcaemia although underlying aetiology remained under investigation; a post-partum FDG-PET was non-diagnostic. Similar biochemical and clinical pattern are well-reported in rare cases of 24-hydroxylase loss-of-function mutations. Genetic testing for CYP24A1 mutations are pending.
Key points: