Lemme Jordan D, Tucker-Bartley Anthony, Drubach Laura A, Shah Nehal, Romo Laura, Cay Mariesa, Voss Stephan, Kwatra Neha, Kaban Leonard B, Hassan Adam S, Boyce Alison M, Upadhyay Jaymin
Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital and Harvard Medical School, Boston, MA, United States.
Department of Anesthesiology, Critical Care and Pain Medicine, Massachusetts General Hospital and Harvard Medical School, Boston, MA, United States.
Front Med (Lausanne). 2022 Mar 15;9:857079. doi: 10.3389/fmed.2022.857079. eCollection 2022.
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.
被诊断为McCune - Albright综合征(MAS)的患者经常表现出颅面骨纤维发育不良(FD)。颅面骨FD可压迫神经纤维,导致视力丧失以及颅面疼痛。为恢复功能或缓解症状,会对受影响的神经进行手术减压,但效果不一。在此,我们报告一例12岁的MAS患者,该患者存在视力缺陷,尤其是左眼(经Goldmann视野测试中盲点扩大证实)以及颅面疼痛。左侧视神经减压手术使视力稍有改善,但在3个月的随访评估中仍存在持续性视力缺陷。对视觉系统进行深入的基于成像的评估,包括视网膜神经纤维层、视神经和中枢神经系统(CNS)视觉通路,发现在整个视觉处理流程中存在多处异常。在当前的FD/MAS患者中,观察到左侧视辐射内白质纤维密度降低以及左侧初级视觉皮层出现功能变化。中枢视觉通路中存在的异常结构和功能异常可能在FD/MAS患者视力缺陷的形成中起作用,并导致外周神经减压手术后效果不一。