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Familial congenital palsy of trochlear nerve

MedGen UID:
338185
Concept ID:
C1850996
Disease or Syndrome
Synonyms: Familial congenital fourth cranial nerve palsy; Strabismus from superior oblique palsy; Superior oblique oculomotor palsy, familial congenital; Trochlear nerve palsy, familial congenital
SNOMED CT: Familial congenital palsy of trochlear nerve (782679002)
 
Monarch Initiative: MONDO:0007626
OMIM®: 136480
Orphanet: ORPHA91498

Definition

A rare genetic neuro-ophthalmological disease with characteristics of congenital fourth cranial nerve palsy, manifesting with hypertropia in side gaze, unexplained head tilt, acquired vertical diplopia and progressive increase in vertical fusional vergence amplitudes with prolonged occlusion. Facial asymmetry (for example hemifacial retrusion, upward slanting of mouth on the side of the head tilt, mild enophthalmos of paretic eye) and superior oblique tendon abnormalities (such as absence, redundance, misdirection) are frequently associated. Some asymptomatic cases have been reported. [from SNOMEDCT_US]

Clinical features

From HPO
Fourth cranial nerve palsy
MedGen UID:
75746
Concept ID:
C0271375
Disease or Syndrome
Paralysis of the fourth cranial (trochlear) nerve manifested as weakness of the superior oblique muscle which causes vertical diplopia that is maximal when the affected eye is adducted and directed inferiorly.
Impaired ocular adduction
MedGen UID:
337588
Concept ID:
C1846463
Finding
Reduced ability to move the eye in the direction of the nose.
Abnormality of binocular vision
MedGen UID:
868909
Concept ID:
C4023320
Anatomical Abnormality
An abnormality of binocular vision, that is of the ability to synthesize the visual inputs from both eyes to a single image with perception of depth.
Abnormal visual fixation
MedGen UID:
1390282
Concept ID:
C4477094
Finding
Any anomaly in the process of ocular fixation, which is the maintaining of the visual gaze on a single location.
Superior oblique muscle weakness
MedGen UID:
1640397
Concept ID:
C4703403
Finding
Decreased strength of the superior oblique muscle.

Recent clinical studies

Etiology

Komori M, Suzuki H, Hikoya A, Sawada M, Hotta Y, Sato M
PLoS One 2016;11(12):e0168245. Epub 2016 Dec 16 doi: 10.1371/journal.pone.0168245. PMID: 27992486Free PMC Article
Ellis FJ, Jeffery AR, Seidman DJ, Sprague JB, Coussens T, Schuller J
J AAPOS 2012 Dec;16(6):558-64. doi: 10.1016/j.jaapos.2012.09.005. PMID: 23237754
Caca I, Sahin A, Cingu A, Ari S, Akbas U
J Pediatr Ophthalmol Strabismus 2012 Mar-Apr;49(2):103-8. Epub 2011 Jul 6 doi: 10.3928/01913913-20110628-01. PMID: 21732576
Jiang Y, Matsuo T, Fujiwara H, Hasebe S, Ohtsuki H, Yasuda T
Acta Med Okayama 2005 Apr;59(2):55-62. doi: 10.18926/AMO/31966. PMID: 16049556

Diagnosis

Matalia J, Kasturi N, Brodsky MC
Am Orthopt J 2013;63:80-4. doi: 10.3368/aoj.63.1.80. PMID: 24141756
Ellis FJ, Jeffery AR, Seidman DJ, Sprague JB, Coussens T, Schuller J
J AAPOS 2012 Dec;16(6):558-64. doi: 10.1016/j.jaapos.2012.09.005. PMID: 23237754
Caca I, Sahin A, Cingu A, Ari S, Akbas U
J Pediatr Ophthalmol Strabismus 2012 Mar-Apr;49(2):103-8. Epub 2011 Jul 6 doi: 10.3928/01913913-20110628-01. PMID: 21732576
Kim JH, Hwang JM
Br J Ophthalmol 2010 Mar;94(3):346-50. Epub 2009 Oct 12 doi: 10.1136/bjo.2009.161398. PMID: 19822909
Astle WF, Rosenbaum AL
Arch Ophthalmol 1985 Apr;103(4):532-5. doi: 10.1001/archopht.1985.01050040074021. PMID: 3985833

Prognosis

Caca I, Sahin A, Cingu A, Ari S, Akbas U
J Pediatr Ophthalmol Strabismus 2012 Mar-Apr;49(2):103-8. Epub 2011 Jul 6 doi: 10.3928/01913913-20110628-01. PMID: 21732576

Clinical prediction guides

Wang SM, Zwaan J, Mullaney PB, Jabak MH, Al-Awad A, Beggs AH, Engle EC
Am J Hum Genet 1998 Aug;63(2):517-25. doi: 10.1086/301980. PMID: 9683611Free PMC Article

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