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Expanding the Phenotype of a Neurofibromatosis Type 1–Like Syndrome: A Patient With a SPRED1 Mutation and Orbital Manifestations

2009· article· en· 3 citations· W4233296897 on OpenAlex· 10.1097/iop.0b013e3181b59eea

Why is this work in the frame?

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

Canadian affiliationAn author listed a Canadian institution. This is the only route the usual frame has.

Post-publication record

Nature
Retraction
Reason
Error in Data;Error in Results and/or Conclusions;Miscommunication with/by Author;Unreliable Data;Unreliable Results and/or Conclusions;
Date
3/1/2010 0:00
Flagged by OpenAlex?
Yes

Source: Retraction Watch, joined by DOI. OpenAlex records retraction as is_retracted, a boolean over a state space with at least four values, so it cannot express an expression of concern, a correction or a reinstatement — it reports them as false, which reads as “fine”.

Abstract

A 4-year-old child with no medical history presented for evaluation of a small, palpable nodule near the left inferolateral rim. The lesion had a bluish hue and had been slowly enlarging over the course of several months. MRI of the orbits revealed a heterogenous and infiltrative preseptal and extraconal mass which enhanced with gadolinium, and sphenoid wing dysplasia on the left. A complete ophthalmic and physical examination failed to reveal any other stigmata of neurofibromatosis type 1 (NF1) or neurofibromatosis type 2 (NF2). An incisional biopsy of the palpable mass revealed a plexiform neurofibroma. Molecular sequencing of the NF1 and NF2 genes did not reveal causative mutations. Further investigation revealed a loss of function mutation in SPRED1 on chromosome 15. Although loss of function mutations in the SPRED1 gene are known to cause several dermatologic changes associated with the NF1-like phenotype, to our knowledge, this is the first description of a SPRED1 gene mutation resulting in ophthalmic abnormalities.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

The record

Venue
Ophthalmic Plastic and Reconstructive Surgery
Topic
Neurofibromatosis and Schwannoma Cases
Field
Medicine
Canadian institutions
Object Research Systems (Canada)
Funders
Massachusetts General Hospital
Keywords
MedicineNeurofibromatosisDermatologyPathologyMutationBiopsyNeurofibromatosis type 2NeurofibromatosesGeneticsGeneBiology
Has abstract in OpenAlex
yes