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Spontaneous adrenal hemorrhage is an acute hemorrhage during pregnancy, which can be tragic for the mother and the baby. We report a unique spontaneous hemorrhage during pregnancy in a case with horseshoe kidney with separated adrenal, presented for the first time in the world. Computed tomography scan showed a horseshoe kidney fused with left normal kidney. Interestingly the adrenal gland was remained in right flank and separated from the horseshoe kidney, which prepares a probable physical stress for the hemorrhage. Diagnosis and surgery were done successfully and the case was fully recovered after several days.
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