Berard, Roberta A.

Hunter, Julia A.

Levy, Deborah M.

English

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Western University Scholarship@Western Paediatrics Publications Paediatrics Department 3-1-2021 Raynaud phenomenon with lingual involvement in a 6-year-old female Julia A. Hunter Western University Deborah M. Levy Western University Roberta A. Berard Western University, roberta.berard@lhsc.on.ca Follow this and additional works at: https://ir.lib.uwo.ca/paedpub Citation of this paper: Hunter, Julia A.; Levy, Deborah M.; and Berard, Roberta A., "Raynaud phenomenon with lingual involvement in a 6-year-old female" (2021). Paediatrics Publications. 791. https://ir.lib.uwo.ca/paedpub/791 Berard, et al: Pediatric lingual RP 465Images in RheumatologyRaynaud Phenomenon With Lingual Involvement in a  6-year-old FemaleJulia A. Hunter, MB BCh BAO; Deborah M. Levy, MD, MS, FRCPC; Roberta A. Berard, MD, MSc, FRCPC, Division of Pediatric Rheumatology, Western  University, London, Ontario, Canada. Address correspondence to Dr. R. Berard, Children’s Hospital, London Health Sciences Centre, London, ON N6A5W9, Canada. Email: Roberta.berard@lhsc.on.ca. Written and verbal consent were provided by the patient’s mother to publish this case. Ethical board approval was not required according to the authors’ institution, as this was an image for which we have received consent from the patient’s family.Raynaud phenomenon (RP) may affect the tongue as has pre-viously been reported in cases of secondary RP1,2,3. We are not aware of lingual involvement being previously described in indi-viduals with primary RP, particularly in children. A 6-year-old prepubertal, previously healthy female was referred for rheumatological assessment. Following cold exposure, she described a typical triphasic RP of the fingers. After initial episodes involving the digits, she subsequently experienced epi-sodes of nonpainful blanching of the anterior left one-third of the tongue associated with tingling. These episodes were precipitated by exposure to cold weather with an open mouth, but not inges-tion of cold liquids or foods. Exact episode duration is unknown, though there was rapid resolution after mouth closure. There was no history of lingual ulceration or other associated symp-toms. Lingual and digital episodes did not occur concurrently (Figures 1A,B). History and examination, including nailfold capillaros-copy were normal. Laboratory workup revealed positive antinuclear antibodies (ANA; titer of 1:2560 in a mitotic pat-tern). Extractable nuclear antigen (ENA) panel was negative. The patient was evaluated 9 months later, with repeat ANA revealing a titer of 1:160 (mitotic). ENA remained negative. There was no evolution in symptoms to suggest secondary RP. Retrospective data have shown that ANA positivity in chil-The Journal of Rheumatology 2021;48:465–6doi:10.3899/jrheum.200258Figure 1. A 6-year-old female with a history of blanching and tingling of fingers developed anterior predominantly left-sided tongue discoloration consistent with lingual RP. (A) Superior surface of the tongue. (B) Typical blanching of the fingers as seen in RP. RP: Raynaud phenomenon.Personal non-commercial use only. The Journal of Rheumatology Copyright © 2021. All rights reserved. www.jrheum.orgDownloaded on August 8, 2022 from 466 The Journal of Rheumatology 2021;48:3; doi:10.3899/jrheum.200258Personal non-commercial use only. The Journal of Rheumatology Copyright © 2021. All rights reserved. Personal non-commercial use only. The Journal of Rheumatology Copyright © 2021. All rights reserved.dren with RP is associated with evolution to involve other fea-tures of a systemic autoimmune disease4. Despite not having secondary RP at the time of assessment, given this patient’s elevated ANA and unusual lingual presentation, she will be followed closely for development of an associated rheumatic disease.REFERENCES 1. Lioger B, Diot E. Raynaud’s phenomenon of the tongue: uncommon presentation of a classical sign. QJM 2012;106:583-4. 2. Katada Y, Tanaka T. Lingual Raynaud’s phenomenon. N Engl J Med 2012;366:e12. 3. Cohen JC, Palomba ML, Morris LG. Raynaud’s phenomenon of the tongue. J Rheumatol 2013;40:336. 4. Falcini F, Rigante D, Candelli M, Martini G, Corona F, Petaccia A, et al. Anti-nuclear antibodies as predictor of outcome in a  multi-center cohort of italian children and adolescents with Raynaud’s phenomenon. Clin Rheumatol 2015;34:167-9. www.jrheum.orgDownloaded on August 8, 2022 from 

Raynaud phenomenon with lingual involvement in a 6-year-old female

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Raynaud phenomenon with lingual involvement in a 6-year-old female

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