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Case Reports

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Erythermalgia Involving the Face Alone: Two Case Reports

  • Juan-Hua Liu1
  • Zhi-Sheng Weng2
  • Di-Qing Luo1,*,
  • Yu-Kun Zhao1
  • Hui-Hui Wu1
  • Ying-Biao Huang1

1Sun Yat Sen Univ, Dept Dermatol, Eastern Hosp, Affiliated Hosp 1, 183 Huangpu Rd E, Guangzhou 510700, Guangdong, Peoples R China

2Guangzhou Med Univ, Dept Dermatol, Affiliated Hosp 3, Guangzhou, Guangdong, Peoples R China

DOI: 10.11607/ofph.2139 Vol.33,Issue 3,September 2019 pp.15-18

Submitted: 06 January 2018 Accepted: 18 July 2018

Published: 30 September 2019

*Corresponding Author(s): Di-Qing Luo E-mail: luodq@mail.sysu.edu.cn

Abstract

Erythermalgia is a rare cutaneous disease characterized by episodic attacks of burning pain, erythema, and increased temperature. It primarily involves the extremities, with possible extensions to the ears, face, neck, and scrotum; in rare instances, it may afflict the ears, face, or the scrotum alone. Although various medications alone or in different combinations have been tried with significant variations in response, no recommended therapeutics have been established until very recently. This report presents two case histories of a 20-year-old and a 46-year-old woman, respectively, who displayed intermittent facial erythema associated with warmth and pain during the episodes and who presented normal between episodes. Both had good response to combinative treatments of systemic medication and topical lidocaine compounds. The younger was disease- free after more than 4 years of follow-up, and the older had recurrence after stopping the treatment. Conclusion: Erythermalgia may involve the face alone, and combinative approaches may be of choice for its treatment. Topical lidocaine compounds are considered to be a good option for palliative treatment.

Keywords

erythermalgia;erythromelalgia;face;pain;treatment

Cite and Share

Juan-Hua Liu,Zhi-Sheng Weng,Di-Qing Luo,Yu-Kun Zhao,Hui-Hui Wu,Ying-Biao Huang. Erythermalgia Involving the Face Alone: Two Case Reports. Journal of Oral & Facial Pain and Headache. 2019. 33(3);15-18.

References

1. Cook-Norris RH, Tollefson MM, Cruz-Inigo AE, Sandroni P, Davis MD, Davis DM. Pediatric erythromelalgia: A retrospec- tive review of 32 cases evaluated at Mayo Clinic over a 37-year period. J Am Acad Dermatol 2012;66:416–423.

2. Davis MD, O’Fallon WM, Rogers RS 3rd, Rooke TW. Natural history of erythromelalgia: Presentation and outcome in 168 patients. Arch Dermatol 2000;136:330–336.

3. Skeik N, Rooke TW, Davis MD, et al. Severe case and liter- ature review of primary erythromelalgia: Novel SCN9A gene mutation. Vasc Med 2012;17:44–49.

4. Wu HH, Wang MJ, Luo DQ, Lin Q. Localized secondary eryth- romelalgia in an 11-year-old boy. Pediatr Dermatol 2012;30:e 244–e245.

5. Johnson E, Iyer P, Eanes A, Zolnoun D. Erythema and burn- ing pain in the vulva: A possible phenotype of erythromelalgia. Case Rep Med 2011;2011:374167.

6. Liu JH, Feng SW, Luo ZY, Luo DQ. Red scrotum syndrome: A form of corticosteroid addiction, a variant of erythermalgia, or a distinct entity? Dermatologica Sinica 2016;34:170–171.

7. Byun JW, Hong WK, Han SH, et al. Red scrotum syndrome: Successful treatment with oral doxycycline. Int J Dermatol 2012; 51:362–363.

8. Wollina U. Red scrotum syndrome. J Dermatol Case Rep 2011; 5:38–41.

9. Luo DQ, Zhao YK, Xu QF, He XQ, Wu LC. Recurrent facial erythema with burning sensation and increased temperature: A variant of erythromelalgia or a new entity? Pain Med 2014; 15:1007–1010.

10. Lambru G, Bakar NA, Matharu M. SUNA and red ear syn- drome: A new association and pathophysiological consider- ations. J Headache Pain 2013;14:32.

11. Ramirez CC, Kirsner RS. A refractory case of erythromelalgia involving the ears. Am J Otolaryngol 2004;25:251–254.

12. Chen MC, Xu QF, Luo DQ, Li X, He DY. Erythema associated with pain and warmth on face and ears: A variant of eryther- malgia or red ear syndrome? J Headache Pain 2014;15:18.

13. Kalgaard OM, Seem E, Kvernebo K. Erythromelalgia: A clinical study of 87 cases. J Intern Med 1997;242:191–197.

14. Yang Y, Wang Y, Li S, et al. Mutations in SCN9A, encoding a sodium channel alpha subunit, in patients with primary eryther- malgia. J Med Genet 2004;41:171–174.

15. Dessinioti C, Katsambas A. Seborrheic dermatitis: Etiology, risk factors, and treatments: Facts and controversies. Clin Dermatol 2013;31:343–351.

16. Dessinioti C, Antoniou C. The “red face”: Not always rosacea. Clin Dermatol 2017;35:201–206.

17. Purnamawati S, Indrastuti N, Danarti R, Saefudin T. The role of moisturizers in addressing various kinds of dermatitis: A review. Clin Med Res 2017;15:75–87.

18. Moore DE. Drug-induced cutaneous photosensitivity: Inci- dence, mechanism, prevention and management. Drug Saf 2002;25:345–372.

19. Ikizog˘ lu G. Red face revisited: Flushing. Clin Dermatol 2014; 32:800–808.

20. Jørum E, Torbergsen T, Dietrichs E. Combined testing of au- tonomic and sensory dysfunction in patients with unilateral fa- cial flushing and sweating during exercise. Neurophysiol Clin 2012;43:1–10.

21. Cerci FB, Kapural L, Yosipovitch G. Intractable erythromelal- gia of the lower extremities successfully treated with lumbar sympathetic block. J Am Acad Dermatol 2013;69:e270–e272.

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