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Hereditary neuropathy with liability to pressure palsies (HNPP)

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A 25-year-old has recurrent episodes of numbness and weakness after minor nerve compression (e.g., crossing legs, leaning on elbow). Symptoms resolve over weeks.

AD; PMP22 deletion (reciprocal of CMT1A duplication)

  • Recurrent focal neuropathies triggered by pressure/trauma
  • Episodes typically resolve
  • May develop mild background neuropathy over time
  • Clinical suspicion from recurrent painless pressure palsies at common compression sites
  • Nerve conduction studies show a background demyelinating sensorimotor neuropathy with focal conduction slowing at entrapment sites; sural biopsy (if done) shows tomaculous ("sausage-like") myelin
  • Confirm with molecular testing for the recurrent 1.5 Mb 17p12 deletion spanning PMP22 (the reciprocal of the CMT1A duplication); if absent, sequence PMP22 for an intragenic loss-of-function variant
  • Preventive counseling: avoid prolonged limb compression, repetitive trauma, and rapid weight loss; use ergonomic padding and positioning
  • Supportive care: physical/occupational therapy, splints or braces for foot drop, and pain management; most episodes recover spontaneously
  • Caution with positioning during surgery and anesthesia; consider the diagnosis before nerve decompression surgery

"HNPP is the reciprocal of CMT1A": CMT1A = PMP22 duplication (too much myelin protein), HNPP = PMP22 deletion (too little myelin protein). Same gene region on 17p12, opposite dosage effects. Notably, this is opposite the typical paradigm where deletions are more severe than duplications: here the deletion (HNPP) is the milder condition and the duplication (CMT1A) is more severe.

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