Overview
Definition:
Alport syndrome is a rare inherited disorder characterized by progressive hearing loss, kidney disease, and eye abnormalities, caused by mutations in genes encoding type IV collagen chains (COL4A3, COL4A4, COL4A5).
Epidemiology:
It affects approximately 1 in 5,000 to 1 in 50,000 live births
The X-linked form (COL4A5 mutations) is the most common, accounting for about 80% of cases
autosomal recessive (COL4A3/COL4A4 mutations) and autosomal dominant forms are less frequent.
Clinical Significance:
Early recognition and meticulous long-term follow-up are crucial to prevent irreversible kidney damage, manage hearing loss effectively, and provide genetic counseling to affected families, impacting long-term patient outcomes and quality of life.
Clinical Presentation
Symptoms:
Sensorineural hearing loss, typically high-frequency
Hematuria, often microscopic initially, progressing to proteinuria and kidney failure
Recurrent urinary tract infections
Ocular abnormalities like lenticonus, retinopathy.
Signs:
Normal initial blood pressure
Edema may appear in advanced stages
Funduscopic examination may reveal anterior lenticonus
Audiometry shows bilateral sensorineural hearing loss
Urinalysis reveals hematuria and proteinuria.
Diagnostic Criteria:
Diagnosis is suspected in individuals with a family history of Alport syndrome or isolated hematuria/proteinuria with sensorineural hearing loss and/or ocular abnormalities
Definitive diagnosis requires genetic testing or renal biopsy showing characteristic ultrastructural abnormalities of the glomerular basement membrane.
Diagnostic Approach
History Taking:
Detailed family history including kidney disease, hearing loss, and eye problems across generations
Age of onset of symptoms
Progression of renal symptoms
Previous audiometry and ophthalmology reports
History of recurrent infections or unexplained hematuria.
Physical Examination:
Complete physical examination focusing on blood pressure, signs of edema
Ocular examination for lenticonus
Detailed neurological assessment for hearing deficits
Skin examination for leiomyomatosis in some rare cases (in connection with Laugier-Hunziker syndrome).
Investigations:
Urinalysis: persistent hematuria (red blood cell casts may be present), proteinuria
Serum creatinine and estimated glomerular filtration rate (eGFR) to assess renal function
Complete blood count to assess for anemia
Audiometry: bilateral sensorineural hearing loss, often high-frequency
Ocular examination: slit-lamp examination for anterior lenticonus
Renal biopsy (if genetic testing is inconclusive or for definitive diagnosis): electron microscopy shows thinning and splitting of the glomerular basement membrane
Genetic testing: sequencing of COL4A3, COL4A4, and COL4A5 genes to identify causative mutations.
Differential Diagnosis:
Other causes of hereditary nephropathies (e.g., thin basement membrane nephropathy, focal segmental glomerulosclerosis)
IgA nephropathy
Systemic lupus erythematosus
Hereditary nephritis without hearing loss or ocular findings.
Management
Initial Management:
Early diagnosis and initiation of renal protective measures
Regular monitoring of renal function, blood pressure, and proteinuria
Referral to audiology and ophthalmology for baseline assessment and ongoing management.
Medical Management:
Renin-angiotensin-aldosterone system (RAAS) inhibitors (ACE inhibitors or ARBs) are the cornerstone of renal management to reduce proteinuria and slow disease progression
Target blood pressure typically <130/80 mmHg
Strict glycemic control in diabetic patients
Management of anemia and electrolyte imbalances
Consider immunosuppressants only in select cases of rapidly progressive glomerulonephritis.
Supportive Care:
Regular audiology follow-up and provision of hearing aids or cochlear implants as needed
Genetic counseling for patients and families regarding inheritance patterns and reproductive options
Lifestyle modifications including salt restriction and avoiding nephrotoxic agents
Nutritional support and management of comorbidities.
Renal Transplantation:
Kidney transplantation is the definitive treatment for end-stage renal disease (ESRD)
Living related donors should be carefully screened to avoid donor-specific antibodies or pre-existing kidney disease
Gene therapy is an investigational approach.
Hearing And Ocular Follow Up
Audiological Monitoring:
Baseline audiometry in all diagnosed children and regular follow-up every 6-12 months, or more frequently if hearing loss is detected
Audiological rehabilitation with hearing aids, assistive listening devices, or cochlear implants when indicated
Counseling on speech and language development support.
Ophthalmological Monitoring:
Annual eye examinations starting in childhood to detect anterior lenticonus, retinal flecks, and other ocular manifestations
Prompt referral to ophthalmologist for evaluation and management of refractive errors, cataracts, or glaucoma
Regular monitoring for visual acuity and visual field defects.
Timing Of Manifestations:
Hearing loss typically begins in late childhood or adolescence, often starting with high-frequency deficits
Ocular abnormalities like lenticonus may be present from birth or develop in early childhood
These manifestations can precede significant renal dysfunction.
Renal Follow Up
Monitoring Frequency:
Regular monitoring of urinalysis (hematuria, proteinuria) and renal function (serum creatinine, eGFR) at least every 6-12 months, or more frequently based on disease severity and progression
Blood pressure monitoring at each visit.
Proteinuria Management:
Aggressive management of proteinuria with RAAS inhibitors is critical
Quantify proteinuria (e.g., spot urine protein-to-creatinine ratio) regularly
Aim for significant reduction in proteinuria to slow CKD progression.
Progression To Esrd:
Patients require close monitoring for progression to end-stage renal disease (ESRD)
Factors like severity of proteinuria, degree of hematuria, genetic mutation type, and age at diagnosis influence the rate of progression
Dialysis and transplantation are options for ESRD management.
Genetic Counseling:
Essential for affected families to understand the inheritance patterns, recurrence risks, and implications for future generations
Carrier screening and prenatal diagnosis may be offered.
Key Points
Exam Focus:
Alport syndrome is a type IV collagenopathy
Key triad: hearing loss, renal disease, ocular abnormalities
X-linked is most common
RAAS inhibitors are crucial for renal protection
Early audiological and ophthalmological assessment is vital.
Clinical Pearls:
Suspect Alport syndrome in any child with unexplained hematuria or persistent proteinuria
Always perform audiometry and fundoscopy in such cases
Long-term multidisciplinary follow-up is key to optimal outcomes.
Common Mistakes:
Delaying diagnosis by attributing hematuria solely to benign causes
Underestimating the progressive nature of hearing loss and kidney disease
Inadequate monitoring of renal function and proteinuria
Failure to offer genetic counseling to families.