Table 1.
Human phenotypes associated with variants of genes encoding dyneins and dynein-associated proteins
PhenotypeAssociated dynein or dynein-related gene variantsaSelected associated publications (PubMed ID)OMIM MIM number (phenotype subtype)
Primary ciliary dyskinesia (PCD): abnormal ciliary motility, respiratory distress, sinusitis, otitis media, bronchiectasis, laterality defects, infertility DNAH1 11371505 20301301 24360805 617577 (CILD37) 
DNAH5 11062149 11788826 608644 (CILD3) 
DNAH9 30471717 30471718 618300 (CILD40) 
DNAH11 12142464 611884 (CILD7) 
DNAI1 10577904 604366 (CILD1) 
DNAI2 18950741 612444 (CILD9) 
DNAL1 21496787 614017 (CILD16) 
NME8 (alias DNAI8 and TXNDC3) 17360648 610852 (CILD6) 
ODAD1 23261302 23261303 23506398 30291279 32855706 615067 (CILD20) 
ODAD2 23849778 24203976 25186273 615451 (CILD23) 
ODAD3 24067530 25192045 25224326 30504913 31383820 616037 (CILD30) 
ODAD4 27486780 617092 (CILD35) 
DNAAF1 19944400 19944405 27261005 613193 (CILD13) 
DNAAF2 31107948 32638265 34785929 612518 (CILD10) 
DNAAF3 22387996 31186518 606763 (CILD2) 
DNAAF4 23872636 615482 (CILD25) 
DNAAF5 29358401 25232951 23040496 614874 (CILD18) 
DNAAF6 32170493 300991 (CILD36) 
ZMYND10 23604077 23891469 23891471 615444 (CILD22) 
LRRC6 23122589 614935 (CILD19) 
LRRC56 30388400 618254 (CILD39) 
SPAG1 24055112 26228299 615505 (CILD28) 
CFAP298 24094744 615500 (CILD26) 
CFAP300 29727692 29727693 618063 (CILD38) 
Spinal muscular atrophy (SMALED type 1): lower limb atrophy and weakness, mild to moderate cognitive impairment DYNC1H1 24307404 25609763 32788638 158600 (SMALED) 
BICD2 26998597 29353221 32709491 615290 (SMALED2A) 618291 (SMALED2B) 
Charcot-Marie-Tooth type 2: distal lower limb weakness, abnormal gait DYNC1H1 24307404 20697106 22459677 22847149 33242470 614228 (CMT2O) 
DNAH10 26517670 Not listed in OMIM 
Asphyxiating thoracic dystrophies (including Jeune syndrome): skeletal abnormalities that may include short ribs and a chest wall deformity, shortened arm and leg bones, an unusually shaped pelvis, polydactyly, renal and hepatic disease (more rarely, retinal disease) DYNC2H1  19442771 26874042 27925158 31935347 613091 (SRTD3) 
DYNC2I1 23910462 26874042 29271569 615503 (SRTD8) 
DYNC2I2 24183449 24183451 615633 (SRTD11) 
DYNC2LI1 26130459 617088 (SRTD15) 
DYNLT2B 25830415 26044572 28475963 617405 (SRTD17) 
Retinal degeneration DYNC2H1 32753734 Not listed in OMIM 
Nonsyndromic rod-cone dystrophy DYNC2I2 33124039 Not listed in OMIM 
Neurodevelopmental disorder with microcephaly and structural brain anomalies DYNC1I2 31079899 618492 (NEDMIBA) 
Mirror movements type 3: movements on one side of the body are involuntarily mirrored on the other side of the body DNAL4 25098561 616059 (MRMV3) 
Mental retardation autosomal dominant 13 DYNC1H1 23603762 22368300 614563 (MRD13) 
Spermatogenic failure DNAH1 24360805 33989052 617576 (SPGF18) 
DNAH2 30811583 619094 (SPGF45) 
DNAH8 32619401 619095 (SPGF46) 
DNAH17 31178125 31658987 31841227 618643 (SPGF39) 
Lissencephaly: developmental delay, myoclonic jerks and spasms, seizures, hypotonia, microcephaly, dysmorphic facies PAFAH1B1 32692650 20301752 32341547 28886386 601545 (LIS) 
Seckel syndrome: growth retardation, microcephaly, developmental delay NIN 27053665 22933543 614851 (SCKL7) 
PhenotypeAssociated dynein or dynein-related gene variantsaSelected associated publications (PubMed ID)OMIM MIM number (phenotype subtype)
Primary ciliary dyskinesia (PCD): abnormal ciliary motility, respiratory distress, sinusitis, otitis media, bronchiectasis, laterality defects, infertility DNAH1 11371505 20301301 24360805 617577 (CILD37) 
DNAH5 11062149 11788826 608644 (CILD3) 
DNAH9 30471717 30471718 618300 (CILD40) 
DNAH11 12142464 611884 (CILD7) 
DNAI1 10577904 604366 (CILD1) 
DNAI2 18950741 612444 (CILD9) 
DNAL1 21496787 614017 (CILD16) 
NME8 (alias DNAI8 and TXNDC3) 17360648 610852 (CILD6) 
ODAD1 23261302 23261303 23506398 30291279 32855706 615067 (CILD20) 
ODAD2 23849778 24203976 25186273 615451 (CILD23) 
ODAD3 24067530 25192045 25224326 30504913 31383820 616037 (CILD30) 
ODAD4 27486780 617092 (CILD35) 
DNAAF1 19944400 19944405 27261005 613193 (CILD13) 
DNAAF2 31107948 32638265 34785929 612518 (CILD10) 
DNAAF3 22387996 31186518 606763 (CILD2) 
DNAAF4 23872636 615482 (CILD25) 
DNAAF5 29358401 25232951 23040496 614874 (CILD18) 
DNAAF6 32170493 300991 (CILD36) 
ZMYND10 23604077 23891469 23891471 615444 (CILD22) 
LRRC6 23122589 614935 (CILD19) 
LRRC56 30388400 618254 (CILD39) 
SPAG1 24055112 26228299 615505 (CILD28) 
CFAP298 24094744 615500 (CILD26) 
CFAP300 29727692 29727693 618063 (CILD38) 
Spinal muscular atrophy (SMALED type 1): lower limb atrophy and weakness, mild to moderate cognitive impairment DYNC1H1 24307404 25609763 32788638 158600 (SMALED) 
BICD2 26998597 29353221 32709491 615290 (SMALED2A) 618291 (SMALED2B) 
Charcot-Marie-Tooth type 2: distal lower limb weakness, abnormal gait DYNC1H1 24307404 20697106 22459677 22847149 33242470 614228 (CMT2O) 
DNAH10 26517670 Not listed in OMIM 
Asphyxiating thoracic dystrophies (including Jeune syndrome): skeletal abnormalities that may include short ribs and a chest wall deformity, shortened arm and leg bones, an unusually shaped pelvis, polydactyly, renal and hepatic disease (more rarely, retinal disease) DYNC2H1  19442771 26874042 27925158 31935347 613091 (SRTD3) 
DYNC2I1 23910462 26874042 29271569 615503 (SRTD8) 
DYNC2I2 24183449 24183451 615633 (SRTD11) 
DYNC2LI1 26130459 617088 (SRTD15) 
DYNLT2B 25830415 26044572 28475963 617405 (SRTD17) 
Retinal degeneration DYNC2H1 32753734 Not listed in OMIM 
Nonsyndromic rod-cone dystrophy DYNC2I2 33124039 Not listed in OMIM 
Neurodevelopmental disorder with microcephaly and structural brain anomalies DYNC1I2 31079899 618492 (NEDMIBA) 
Mirror movements type 3: movements on one side of the body are involuntarily mirrored on the other side of the body DNAL4 25098561 616059 (MRMV3) 
Mental retardation autosomal dominant 13 DYNC1H1 23603762 22368300 614563 (MRD13) 
Spermatogenic failure DNAH1 24360805 33989052 617576 (SPGF18) 
DNAH2 30811583 619094 (SPGF45) 
DNAH8 32619401 619095 (SPGF46) 
DNAH17 31178125 31658987 31841227 618643 (SPGF39) 
Lissencephaly: developmental delay, myoclonic jerks and spasms, seizures, hypotonia, microcephaly, dysmorphic facies PAFAH1B1 32692650 20301752 32341547 28886386 601545 (LIS) 
Seckel syndrome: growth retardation, microcephaly, developmental delay NIN 27053665 22933543 614851 (SCKL7) 
a

Note that for some of these phenotypes, there are several variants with varying degrees of severity, and different genes may be associated with different types of these genetic conditions.

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