*Purpose: Data to guide the evaluation of living related donor (LRD) candidates for kidney transplant recipients with Alport syndrome (AS) spectrum are limited. We examined a cohort of LRD to recipients with AS to improve understanding of the clinical course and outcomes of living donation in this context.

*Methods: The cohort of recipients and their LRD was retrospectively identified from data at 5 transplant centers (1987-2021). LRD were followed for postdonation kidney function measured by estimated glomerular filtration rate (eGFR), proteinuria, major cardiac events (MACE) and death.

*Results: The cohort comprised 33 LD, where relationship to recipient included mother (14, 42%), father (10, 30%), sibling (5, 32%), grandparent (1, 3%), uncle (1, 3%) and unrelated (2, 6%). Long term outcomes were evaluated in 27 LRD during a follow up of 12 years (IQR, 5-16). None of the donors developed kidney failure (eGFR<15 ml/min/1.73m² or dialysis) during follow up. Last follow up serum mean (SD) creatinine, eGFR and proteinuria levels were 1.1 (0.2) mg/dL, 68.3 (16.0) ml/min/1.73m², and 0.19 (0.36) g/g, respectively (Table 1). During follow up, 13 (48%) and 6 (22%) donors developed hypertension and diabetes mellitus, respectively. Five donors (19%) developed MACE [acute coronary ischemia, n=4 (15%), severe congestive heart failure, n=1, (4%)] at a median 5.5 (IQR, 4.5-10.3) years after donation. MACE rate was significantly higher in patients who developed hypertension compared to normotensives after donation (0% vs 35.7%, p=0.017) (Fig. 1). Three donors died during follow-up at median 14 (5-15) years after donation.

*Conclusions: Although the kidney failure risk might be minimized by careful donor evaluation, our findings suggest that the cardiovascular risk after kidney donation was higher than expected in LRD of recipients with AS.

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