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Published on: August 9, 2024
Ocular manifestations of oblique facial clefts
Maria Carolina Ortube1, Katrina Dipple, Yoshio Setoguchi
1Department of Ophthalmology, Jules Stein Eye Institute, David Geffen School of Medicine at UCLA, Los Angeles, California, USA.
Insights
This study reports a rare case of bilateral Tessier facial cleft 10 in a patient with ocular abnormalities, including dermoids and morning glory anomaly. The findings highlight potential associations with alopecia and restrictive strabismus.
Area of Science:
- Craniofacial surgery
- Ophthalmology
- Pediatric neurology
Background:
- Tessier clefts are a classification system for craniofacial clefts, numbered 0-14.
- Tessier cleft 10 involves the eyebrows and upper eyelids.
- Bilateral Tessier 10 clefts are exceptionally rare and have not been previously reported.
Observation:
- A male infant presented with bilateral epibulbar dermoids, unilateral morning glory anomaly, and facial dysmorphism following prenatal exposure to oligohydramnios, digoxin, and lisinopril.
- Ocular examination revealed severe restrictive strabismus in the left eye, hypotropia, and esotropia.
- Cranial CT scans identified a left middle cranial fossa arachnoid cyst and calcification of the superior oblique muscle trochlea.
Findings:
- The patient exhibited bilateral Tessier cleft 10, characterized by colobomata of the upper eyelids and eyebrows, extending into the hairline with associated alopecia.
- The ocular findings included bilateral epibulbar dermoids and a unilateral morning glory anomaly of the optic nerve.
- Radiological findings revealed a left middle cranial fossa arachnoid cyst and calcification of ocular structures causing globe displacement and restrictive strabismus.
Implications:
- This case expands the known spectrum of anomalies associated with bilateral Tessier 10 clefts.
- It suggests a potential link between bilateral Tessier 10 clefts and a constellation of findings including alopecia, ocular abnormalities (dermoids, morning glory anomaly), arachnoid cysts, and restrictive strabismus.
- Further research is warranted to understand the embryological basis and clinical management of such complex craniofacial and ocular malformations.
Introduction:
In the Tessier classification, craniofacial clefts are numbered from 0 to 14 and extend along constant axes through the eyebrows, eyelids, maxilla, nostrils, and the lips. We studied a patient with bilateral cleft 10 associated with ocular abnormalities.
Method:
Clinical report with orbital and cranial computed tomography.
Results:
After pregnancy complicated by oligohydramnios, digoxin, and lisinopril exposure, a boy was born with facial and ocular dysmorphism. Examination at age 26 months showed bilateral epibulbar dermoids, covering half the corneal surface, and unilateral morning glory anomaly of the optic nerve. Ductions of the right eye were normal, but the left eye had severely impaired ductions in all directions, left hypotropia, and esotropia. Under anesthesia, the left eye could not be rotated freely in any direction. Bilateral Tessier cleft number 10 was implicated by the presence of colobomata of the middle third of the upper eyelids and eyebrows. As the cleft continued into the hairline, there was marked anterior scalp alopecia. Computed x-ray tomography showed a left middle cranial fossa arachnoid cyst and calcification of the reflected tendon of the superior oblique muscle, trochlea, and underlying sclera, with downward and lateral globe displacement.
Discussion:
Tessier 10 clefts are very rare and usually associated with encephalocele. Bilateral 10 clefts have not been reported previously. In this case, there was coexisting unilateral morning glory anomaly and arachnoid cyst of the left middle cranial fossa but no encephalocele.
Conclusions:
Bilateral Tessier facial cleft 10 may be associated with alopecia, morning glory anomaly, epibulbar dermoids, arachnoid cyst, and restrictive strabismus.
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