ð¥ å
§ç§å°ç§èåç
Mechanistic Deep Dive
CFTR Structure + Function
- Membrane-spanning protein
- Two transmembrane domains
- Two nucleotide-binding domains (NBD1, NBD2)
- Regulatory R domain
- ATP-driven Cl- channel + indirect HCO3- regulation
- Regulates other channels (ENaC inhibition, ORCC)
Mucus Pathology
- Dehydrated airway surface liquid (ASL)
- Impaired ciliary function
- Mucus plug formation
- Bacterial trapping
- Chronic inflammation
Inflammation in CF
- Even before infection â primary inflammatory state
- Neutrophil-dominant
- NF-κB pathway hyperactivation
- IL-8 secretion
- Persistent + dysregulated
Recent Trials & Updates
VX-661-103, VX-440-103 (ETI Trials, 2019)
- Phase 3 trials
- F508del homozygous + heterozygous + multiple genotypes
- Improved FEV1 (10-15%)
- Reduced exacerbations (60%)
- FDA approval 2019
ETI Expanded Indications (2023-2024)
- Younger ages (2-5 yo)
- Broader mutation panel
- Reduced age criteria
CFRD GLP-1 + SGLT2i Trials
- Limited data
- Mostly insulin
- Future role unclear
Gene Therapy CF
- AAV-based, lentivirus
- Multiple trials
- Limited durability
- Continued investigation
CRISPR CFTR Repair
- Preclinical / early phase
- Ex-vivo edits possible
- In-vivo lung delivery challenging
Organoid-Based Therapy Selection
- Intestinal organoids from rectal biopsy
- Test CFTR modulator response
- Personalize therapy
- Especially for rare mutations
High-Yield Specialist Points
Sweat Test (Pilocarpine Iontophoresis)
- ⥠60 mmol/L: positive
- 30-59 mmol/L: borderline (repeat + further workup)
- < 30 mmol/L: CF unlikely (note: < 40 in children, < 30 in adults)
- Adequate sweat volume needed (⥠75 mg)
- Specialized lab
Atypical CF Presentations
- Recurrent pancreatitis (CFTR-related)
- Idiopathic bronchiectasis adult
- Male infertility (CBAVD)
- Chronic rhinosinusitis without lung disease
- Sweat chloride may be intermediate
- Single CFTR mutation + clinical features
Adult CF Care
- Separate adult center
- CFRD screening annual
- Liver disease surveillance
- Bone health
- Cancer surveillance (colon)
CF Pulmonary Exacerbation Treatment
- IV antibiotics 2-3 weeks
- Based on prior cultures
- Pseudomonas: combination β-lactam + aminoglycoside
- Tobramycin once-daily preferred
- Cefepime, pip-tazo, ceftolozane-tazobactam
- Avoid aminoglycoside in severe ototoxicity history
Vest Therapy (HFCWO)
- Mainstay airway clearance
- 2-3x daily
- Insurance-covered in US for CF
Dornase Alfa (Pulmozyme)
- Recombinant DNase
- Reduces sputum viscosity
- Daily nebulized
- Improved FEV1
Hypertonic Saline 7%
- Restores airway hydration
- â Exacerbations
- BID nebulized
Mannitol (Bronchitol)
- Osmotic agent
- Alternative airway clearance
- Less common in US
Lung Transplantation
- BMI > 17, FEV1 < 30%, hypoxia, hypercapnia, hospitalizations
- 5-year survival 60-70%
- B. cepacia complex exclusion at many centers
- Bilateral lung transplant
Future Directions
- ETI universal? Or replacement gene therapy?
- mRNA therapy
- Pulmonary microbiome modulation
- Anti-inflammatory beyond modulators
Pearls
- CF: autosomal recessive, CFTR mutation; F508del most common
- Multi-organ disease: lung + pancreas + GI + liver + sweat + reproductive
- Diagnosis: sweat chloride ⥠60 + clinical + genetic
- CFTR modulators revolutionary: ETI (Trikafta/Kaftrio) covers ~ 90%
- Pediatric pathogens: S. aureus â adolescent Pseudomonas â adult Pseudomonas + NTM
- B. cepacia worst â may exclude from transplant
- Survival: ⥠60 for newborns now (vs 30s pre-modulator era)
- CFRD: combined insulin deficiency + resistance; CGM; insulin