Ophthalmic Manifestation in Neurofibromatosis Type 2
Source abstract
Abstract
Neurofibromatosis type 2 (NF2) is a genetically determined tumor-predisposing syndrome. Ocular manifestations include cataracts, epiretinal membranes, retinal hamartomas, optic disk gliomas, and optic nerve sheath meningiomas. Moreover, optic disk edema, optical atrophy, motility disorders, pupil and lid dysfunction, and neurotrophic keratitis can be observed as indirect signs. An observational study was conducted with the aim to collect clinical data and describe the most frequent NF2 ocular manifestations. Fourteen patients affected by NF2, according to the Manchester criteria, were enrolled. All patients underwent complete ophthalmologic and orthoptic evaluation and a spectral domain optical coherence tomography. Ocular manifestations were present in all patients. The slit lamp evaluation of the anterior segment highlighted cataracts in five patients, keratitis in two patients, corneal leukoma in two patients, and corneal pannus in one patient. Fundus oculi and OCT evaluation identified epiretinal membranes in four patients, vitreoretinal tufts in three patients, optic nerve edema in one patient, and retinal hamartoma in one patient. Moreover, the orthoptic evaluation identified different types of ocular motility disorders in seven patients. This is a descriptive study of a rare disease with poor previous literature. Clinical data are shown, emphasizing the role of NF2-specific ophthalmological and orthoptic findings to help establish an early diagnosis.
Source: crossref · Quality: baseline
Research context
OpenAlex topics: Neurofibromatosis and Schwannoma Cases · Vascular Malformations Diagnosis and Treatment · Meningioma and schwannoma management
SciVX discovery tags are terminology-based. OpenAlex topics are source-provided classifications.
Affiliations
Department of Sense Organs, Sapienza University of Rome, Policlinico Umberto I, Viale del Policlinico 155, 00161 Rome, Italy
Department of Dermatology and Venereology, Sapienza University of Rome, Policlinico Umberto I, 00161 Rome, Italy
Ophthalmology Unit, Head and Neck Department, Policlinico Umberto I University Hospital, Sapienza University of Rome, 00161 Rome, Italy
References (34)
Show source-provided references
1. von Recklinghausen, F. (1882). Ueber die Multiplen Fibrome der Haut und Ihre Beziehung zu den Multiplen Neuromen: Festschrift zur Feier des Fünfundzwanzigjährigen Bestehens des Pathologischen Instituts zu Berlin Herrn Rudolf Virchow, A Hirschwald.
2. Bilateral acoustic neurofibromas: A clinical study and field survey of a family of five generations with bilateral deafness in thirty-eight members10.1001/archneurpsyc.1930.02220080050005 ↗
3. Birth incidence and prevalence of tumor-prone syndromes: Estimates from a UK family genetic register service10.1002/ajmg.a.33139 ↗
4. A novel moesin-, ezrin-, radixin-like gene is a candidate for the neurofibromatosis 2 tumor suppressor10.1016/0092-8674(93)90406-G ↗
5. Neurofibromatosis 2 (NF2): Clinical characteristics of 63 affected individuals and clinical evidence for heterogeneity10.1002/ajmg.1320520411 ↗
6. An update on the CNS manifestations of neurofibromatosis type 210.1007/s00401-019-02029-5 ↗
7. Updated diagnostic criteria and nomenclature for neurofibromatosis type 2 and schwannomatosis: An international consensus recommendation
8. Genetic Severity Score predicts clinical phenotype in NF210.1136/jmedgenet-2017-104519 ↗
9. Ophthalmologic findings and long-term course in patients with neurofibromatosis type 210.1016/j.ajo.2005.12.042 ↗
10. Neurofibromatosis Type 2-Related Eye Disease Correlated With Genetic Severity Type
11. Revisiting neurofibromatosis type 2 diagnostic criteria to exclude LZTR1-related schwannomatosis10.1212/WNL.0000000000003418 ↗
12. A genetic study of type 2 neurofibromatosis in the United Kingdom. II. Guidelines for genetic counselling10.1136/jmg.29.12.847 ↗
13. Predictors of the risk of mortality in neurofibromatosis 210.1086/342716 ↗
14. Genotype-phenotype correlations for nervous system tumors in neurofibromatosis 2: A population-based study10.1086/422700 ↗
15. Lens opacities in neurofibromatosis 2: Further significant correlations10.1136/bjo.77.6.354 ↗
16. Epiretinal membranes indicate a severe phenotype of neurofibromatosis type 210.1097/IAE.0b013e3181dc58bf ↗
17. Ocular alterations, molecular findings, and three novel pathological mutations in a series of NF2 patients10.1007/s00417-019-04348-5 ↗
18. Optical coherence tomography characteristics of epiretinal membranes in neurofibromatosis 210.3928/15428877-20080101-13 ↗
19. Ocular pathologic findings of neurofibromatosis type 210.1001/archopht.125.3.389 ↗
20. Ocular fundus in neurofibromatosis type 210.1136/bjo.77.10.646 ↗
21. Structural Abnormalities of the Central Retina in Neurofibromatosis Type 210.1159/000519143 ↗
22. Spectral-Domain Optical Coherence Tomography Findings in Neurofibromatosis Type 210.1167/iovs.15-18919 ↗
23. Revised diagnostic criteria for neurofibromatosis type 1 and Legius syndrome: An international consensus recommendation
24. An update on choroidal abnormalities and retinal microvascular changes in neurofibromatosis type 110.1186/s13023-022-02369-8 ↗
25. Monocular elevator paresis in neurofibromatosis type 210.1212/WNL.56.9.1222 ↗
26. Severe optic disc edema without hydrocephalus in neurofibromatosis 210.1016/S0021-5155(98)00027-6 ↗
27. Neurofibromatosis type 210.1016/S0140-6736(09)60259-2 ↗
28. Combined retinal hamartomas leading to the diagnosis of neurofibromatosis type 210.1080/13816810802206507 ↗
29. Familial occurrence of combined pigment epithelial and retinal hamartomas associated with neurofibromatosis 210.1097/00006982-199212020-00005 ↗
30. Neurofibromatosis 2 leads to higher incidence of strabismological and neuro-ophthalmological disorders10.1111/j.1600-0420.2007.01088.x ↗
31. Ocular abnormalities in neurofibromatosis 210.1016/S0002-9394(14)72210-X ↗
32. Recurrent third nerve palsy as the presenting feature of neurofibromatosis 2
33. Neurofibromatosis type II presenting as vertical diplopia10.1080/09273970490491178 ↗
34. Ocular findings associated with neurofibromatosis type II10.1016/S0161-6420(92)31789-0 ↗