Spontaneous subepithelial hemorrhage of renal pelvis and ureter (Antopol-Goldman lesion) in hemophilia A patient with inhibitor

Abstract

Introduction: The Antopol-Goldman lesion (AGL), which expresses subepithelial hemorrhage in the renal pelvis, was first defined by Antopol and Goldman in 1948. The objective of this study is to report the first case of AGL in patients with congenital hemophilia and review the relevant literature. Patient concerns: A 32-year-old male patient diagnosed with congenital hemophilia A (FVIII = %4) with high responding inhibitors (7.4 BU) was admitted to our emergency department with gross hematuria and sudden onset flank pain. Diagnosis: Abdominal computed tomography (CT-scan) presented a hyperdense lesion in the left ureteropelvic junction with Hounsfield Units of 56 compatibles with hemorrhage. Interventions: The patient was given 4500 IU of factor eight inhibitor bypass activity (FEIBA) intravenously twice daily for 5 days. Subsequently, 4500 IU of FEIBA was administrated once a day for 2 days. Outcomes: The patient's complaints disappeared on the fourth day of treatment. Macroscopic and microscopic hematuria was not seen in the following days. Follow-up CT was done 3 months after discharge and showed normal left renal pelvis without hyperdenosis. Follow-up CT was performed 3 months after discharge and presented normal left renal pelvis with no hyperdense lesion. Conclusion: Although very rare, AGL should be kept in mind in the differential diagnosis of renal pelvic hemorrhage. In the patient who has an underlying history of coagulopathy nephrectomy can be avoided when there is awareness of AGL.

Recombinant FVIIIFc Versus BAY 94-9027 for Treatment of Patients with Haemophilia A: Comparative Efficacy Using a Matching Adjusted Indirect Comparison

“…Similarly, adjustment for differences in the assessment periods of the two studies could not be included in the analysis, as these data were not available for PROTECT VIII. Furthermore, detailed clinical data for the patients in these studies are limited, and information on, for example, spontaneous bleeds such as the recently reported case of a spontaneous subepithelial haemorrhage (Antopol-Goldman lesion) in the renal pelvis and ureter of a patient with haemophilia A and inhibitors [ 25 ]. The absence of a placebo group in these clinical studies means that there can be no adjustment for residual confounding; other confounding factors not reported in the trials may also have an influence on the results reported here.…”

Successful Chemical Synovectomy in a Patient with Acquired von Willebrand Syndrome with Chronic Synovitis Due to Recurrent Knee Hemarthrosis: A Case Report

“…On the other hand, inhibitors observed in around one-third of patients with congenital hemophilia A have different kinetic features compared to anti-FVIII antibodies found in acquired hemophilia A. However, in both cases, the interference of these antibodies with FVIII replacement therapy leads to the need of bypassing agents [19]. Indeed, in the case of inherited bleeding disorders such as hemophilia A and B and VWD, when plasma levels of FVIII are markedly reduced, joint bleeding is a typical presentation [20][21][22].…”

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“…UTUCs are another kind of common renal pelvis malignant tumor. Patients with UTUCs typically have a history of hematuria[ 11 ]. In contrast, symptoms of renal pelvis SFTs are mostly due to pressure effect of the lesion, and these patients rarely develop urinary symptoms.…”