Case Report: A Neuro-Ophthalmological Assessment of Vision Loss in a Pediatric Case of McCune-Albright Syndrome

被引:4
|
作者
Lemme, Jordan D. [1 ,2 ]
Tucker-Bartley, Anthony [1 ,2 ,3 ]
Drubach, Laura A. [4 ,5 ]
Shah, Nehal [5 ,6 ]
Romo, Laura [2 ,7 ]
Cay, Mariesa [1 ,2 ]
Voss, Stephan [4 ,5 ]
Kwatra, Neha [4 ,5 ]
Kaban, Leonard B. [8 ]
Hassan, Adam S. [9 ]
Boyce, Alison M. [10 ]
Upadhyay, Jaymin [1 ,2 ,11 ,12 ]
机构
[1] Boston Childrens Hosp, Dept Anesthesiol Crit Care & Pain Med, Boston, MA 02115 USA
[2] Harvard Med Sch, Boston, MA 02115 USA
[3] Massachusetts Gen Hosp, Dept Anesthesiol Crit Care & Pain Med, Boston, MA USA
[4] Boston Childrens Hosp, Dept Radiol, Boston, MA USA
[5] Harvard Sch, Boston, MA USA
[6] Brigham & Womens Hosp, Dept Radiol, Boston, MA USA
[7] Massachusetts Eye & Ear, Dept Radiol, Head & Neck Imaging, Boston, MA USA
[8] Harvard Sch Dent Med, Massachusetts Gen Hosp, Dept Oral & Maxillofacial Surg, Boston, MA USA
[9] Eye Plast & Facial Cosmet Surg, Grand Rapids, MI USA
[10] Natl Inst Dent & Craniofacial Res, Metab Bone Disorders Unit, NIH, Bethesda, MD USA
[11] McLean Hosp, Dept Psychiat, Belmont, MA USA
[12] Harvard Med Sch, Belmont, MA USA
关键词
McCune-Albright Syndrome; craniofacial lesions; vision loss; headache; central nervous system; OPTIC-NERVE DECOMPRESSION; FIBROUS DYSPLASIA INDICATIONS; BLINDNESS; EFFICACY; SKULL; BONE;
D O I
10.3389/fmed.2022.857079
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Patients diagnosed with McCune-Albright Syndrome (MAS) frequently manifest craniofacial fibrous dysplasia (FD). Craniofacial FD can impinge nerve fibers causing visual loss as well as craniofacial pain. Surgical decompression of affected nerves is performed, with variable efficacy, in an attempt to restore function or alleviate symptoms. Here, we present a case of a 12-year-old MAS patient with visual deficits, particularly in the left eye (confirmed by enlarged blind spots on Goldmann visual field testing), and craniofacial pain. Decompression surgery of the left optic nerve mildly improved vision, while persistent visual deficits were noted at a 3-month follow-up assessment. An in-depth, imaging-based evaluation of the visual system, including the retinal nerve fiber layer, optic nerves, and central nervous system (CNS) visual pathways, revealed multiple abnormalities throughout the visual processing stream. In the current FD/MAS patient, a loss of white matter fiber density within the left optic radiation and functional changes involving the left primary visual cortex were observed. Aberrant structural and functional abnormalities embedded within central visual pathways may play a role in facilitating deficits in vision in FD/MAS and contribute to the variable outcome following peripheral nerve decompression surgery.
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页数:5
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