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Recurrent symptomatic vertebral hemangioma during two consecutive pregnancies: Case report and review of the literature.

ABSTRACT: Pregnancy-related changes can exacerbate the symptoms/signs of vertebral hemangiomas. Here, we report a patient who experienced symptomatic vertebral hemangiomas resulting in cord compression during two consecutive pregnancies.A 28-year-old female 34 weeks pregnant, presented with a progressive spastic paraparesis. Magnetic Resonance Imaging (MRI) demonstrated an T5 vertebral body signal change attributed to a hemangioma resulting in cord compression. Following a cesarean section, she had a trans thoracic T5 corpectomy with spinal fusion. Indeed, the histopathology was consistent with a vertebral hemangioma. She fully recovered after this first surgery. However, six years later, she again presented with a spastic paraparesis and sphincter deficit now 29 weeks pregnant. The MR demonstrated cord compression one more at the T5 level attributed to the hemangioma; following a T5 and T6 laminectomy, the left paracentral epidural vascular mass totally resected. Her child was successfully delivered 2 months later at which point she exhibited only mild residual lower limb spasticity.Patients with known vertebral hemangiomas should be closely monitored during pregnancy as increased growth during these pregnancies may result in progressive spinal cord compression.

SUBMITTER: Fereydonyan N

PROVIDER: S-EPMC5473073 | biostudies-other | 2017

REPOSITORIES: biostudies-other

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Project description:RationaleGeller et al reported a rare mutation in the mineralocorticoid receptor (MR) resulting in constitutive MR activity. Progesterone, normally an MR antagonist, acts as a potent agonist with this mutation. Progesterone levels can increase 100-fold during pregnancy and thus lead to increased MR activity in this setting, resulting in hypertension (HTN) and hypokalemia during pregnancy and resolution of hypokalemia after delivery.Presenting concernsOur patient was a 33-year-old African American female with a history of pregnancy-induced HTN associated with hypokalemia during her last pregnancy. She presented with muscle weakness from profound hypokalemia complicated by nephrogenic diabetes insipidus (DI) and rhabdomyolysis.DiagnosisHer admission potassium was 1.9 mmol/L (3.5-5.1 mmol/L) with a 24-hour urine potassium of 35 mmol per day and an unmeasurable serum aldosterone level. Her potassium normalized 1 day after delivery off potassium supplementation and amiloride, which were last given 1 day prior to her delivery. Recurrent hypokalemia from nonaldosterone-mediated renal potassium wasting during pregnancy (with normal potassium in a nongestational state) is consistent with the cases of gain-of-function mutation in MR that Geller et al report. A definite diagnosis requires genetic analysis.InterventionsHer hypokalemia was refractory to potassium replacement but quickly responded to an inhibitor of the epithelial sodium channel (ENaC), amiloride.OutcomesHer potassium normalized on amiloride 10 mg per day and KCL 40 mEq daily during the remainder of her pregnancy, and her nephrogenic DI resolved after this correction of hypokalemia. After her delivery, her potassium remained normal off the potassium supplements and amiloride.Novel findingsPregnancy-induced hypokalemia from an activating MR mutation has rarely been reported. Pregnancy-induced HTN is often the first differential diagnosis in a patient who develops worsening in her HTN during pregnancy. We should also consider the possibility of a gain-of-function mutation in MR in these patients who also have associated hypokalemia.

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Recurrent symptomatic vertebral hemangioma during two consecutive pregnancies: Case report and review of the literature.

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