Taher Modarressi1, Michael A Levine2,3, Julia Tchou4,5, Amna N Khan1,6. 1. Department of Endocrinology, Diabetes and Metabolism, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania. 2. Division of Endocrinology and Diabetes, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania. 3. Department of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania. 4. Abramson Cancer Center, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania. 5. Division of Endocrine and Oncologic Surgery, Department of Surgery, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania. 6. Section of Endocrinology, Philadelphia VA Medical Center, Philadelphia, Pennsylvania.
Abstract
Context: Gestational gigantomastia is an uncommon condition characterized by abnormal and excessive growth of breast tissue during an otherwise uncomplicated pregnancy. Gestational gigantomastia may be accompanied by hypercalcemia, which in some cases has been associated with elevated serum levels of PTHrP. The source of the PTHrP in these cases has been suggested to be the enlarged breasts. Objective: To describe the rapid resolution of hypercalcemia and normalization of serum PTHrP after elective termination of pregnancy, indicating that the placenta was the source of the PTHrP. Design: A retrospective analysis of clinical and biochemical data over a 2-year interval and review of literature. Setting: An academic medical center. Patient: A 33-year-old G8P4 female who presented at week 8 of pregnancy with gestational gigantomastia and subsequently developed marked hypercalcemia at week 13. Serum levels of PTH were suppressed but circulating PTHrP was elevated. There was no history of hypercalcemia or significant breast growth during previous pregnancies. Intervention: Hypercalcemia was poorly responsive to IV saline, prednisone, calcitonin, and cinacalcet. She requested termination of pregnancy at week 20. Results: Serum levels of calcium, PTH, and PTHrP normalized within 48 hours of termination of pregnancy. Conclusion: The rapid resolution of hypercalcemia after termination of pregnancy, despite persistent gigantomastia, provides evidence for a pathologic role of the placenta in the excess production of PTHrP, possibly through an as yet uncharacterized placenta-breast hormonal axis.
Context:Gestational gigantomastia is an uncommon condition characterized by abnormal and excessive growth of breast tissue during an otherwise uncomplicated pregnancy. Gestational gigantomastia may be accompanied by hypercalcemia, which in some cases has been associated with elevated serum levels of PTHrP. The source of the PTHrP in these cases has been suggested to be the enlarged breasts. Objective: To describe the rapid resolution of hypercalcemia and normalization of serum PTHrP after elective termination of pregnancy, indicating that the placenta was the source of the PTHrP. Design: A retrospective analysis of clinical and biochemical data over a 2-year interval and review of literature. Setting: An academic medical center. Patient: A 33-year-old G8P4 female who presented at week 8 of pregnancy with gestational gigantomastia and subsequently developed marked hypercalcemia at week 13. Serum levels of PTH were suppressed but circulating PTHrP was elevated. There was no history of hypercalcemia or significant breast growth during previous pregnancies. Intervention: Hypercalcemia was poorly responsive to IV saline, prednisone, calcitonin, and cinacalcet. She requested termination of pregnancy at week 20. Results: Serum levels of calcium, PTH, and PTHrP normalized within 48 hours of termination of pregnancy. Conclusion: The rapid resolution of hypercalcemia after termination of pregnancy, despite persistent gigantomastia, provides evidence for a pathologic role of the placenta in the excess production of PTHrP, possibly through an as yet uncharacterized placenta-breast hormonal axis.
Authors: Natasha M Appelman-Dijkstra; Diana- Alexandra Ertl; M C Zillikens; Lars Rjenmark; Elizabeth M Winter Journal: Endocrine Date: 2021-02-05 Impact factor: 3.633