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New-onset childhood extensive cutaneous lichen planus following asymptomatic COVID-19 infection: report of a case

Líquen plano cutâneo extenso de início recente na infância após infecção assintomática por COVID-19: relato de caso

ABSTRACT

Objective:

The objective of this study was to describe a case of cutaneous lichen planus (LP) that appeared following COVID-19 infection.

Case description:

We report a case of extensive cutaneous classic familial LP in a 4-year-old male child after an asymptomatic serologically confirmed COVID-19 infection. The patient developed intensely itchy, purple, flat-topped papules and plaques, mainly on the dorsal surface of the hands, feet, forearms, and shins. Histopathological examination of the skin biopsy showed vacuolar and apoptotic degeneration of the basal cell layer with a band-like lymphocyte infiltrate at the dermo-epidermal junction and confirmed the diagnosis of LP.

Comments:

LP could be considered among the differential diagnoses of pediatric post-COVID inflammatory skin lesions, either in the patients recovering from COVID-19 infection or in the suspicious asymptomatic cases in close contact with COVID-19-infected patients.

Keywords:
Lichen planus; COVID-19; Child, preschool; Skin diseases; Therapy

RESUMO

Objetivo:

Descrever um caso de líquen plano cutâneo (LP) após infecção por COVID-19.

Descrição do caso:

Relatamos um caso de LP familiar clássico extenso cutâneo em uma criança de quatro anos de idade após uma infecção por COVID-19 assintomática e sorologicamente confirmada. O paciente desenvolveu pápulas e placas intensamente pruriginosas, roxas e achatadas, principalmente na superfície dorsal das mãos, pés, antebraços e canelas. O exame histopatológico da biópsia de pele mostrou degeneração vacuolar e apoptótica da camada basal com infiltrado de linfócitos em faixa na junção dermoepidérmica e confirmou o diagnóstico de líquen plano.

Comentários:

O líquen plano pode ser considerado entre os diagnósticos diferenciais de lesões cutâneas inflamatórias pós-COVID pediátricas, tanto em pacientes em recuperação de infecção por COVID-19 quanto em casos assintomáticos suspeitos em contato próximo com pacientes infectados por COVID-19.

Palavras-chave:
Líquen plano; COVID-19; Criança pré-escolar; Dermatopatias; Terapia

INTRODUCTION

Skin manifestations are among the frequently described signs of COVID-19 infection in adults and children.11 Huynh T, Sanchez-Flores X, Yau J, Huang JT. Cutaneous manifestations of SARS-CoV-2 infection. Am J Clin Dermatol. 2022;23:277-86. https://doi.org/10.1007/s40257-022-00675-2
https://doi.org/10.1007/s40257-022-00675...
55 Andina D, Belloni-Fortina A, Bodemer C, Bonifazi E, Chiriac A, Colmenero I, et al. Skin manifestations of COVID-19 in children: Part 3. Clin Exp Dermatol. 2021;46:462-72. https://doi.org/10.1111/ced.14483
https://doi.org/10.1111/ced.14483...
These cutaneous features include chilblain-like lesions, acute urticaria, erythema multiforme-like eruption, papulovesicular eruption, and non-specific skin lesions.22 Panda M, Agarwal A, Hassanandani T. Dermatological manifestations of COVID-19 in children. Indian Pediatr. 2022;59:393-9. https://doi.org/10.1007/s13312-022-2521-6
https://doi.org/10.1007/s13312-022-2521-...
Besides the skin lesions that develop during active disease, COVID-19 infection can rise or exacerbate some skin diseases.66 Chularojanamontri L, Tuchinda P, Rujitharanawong C, Hunnangkul S, Pochanapan O, Panjapakkul W, et al. New-onset and exacerbated skin diseases after COVID-19 infection: a systematic review. J Dermatol. 2022;49:e419-21. https://doi.org/10.1111/1346-8138.16501
https://doi.org/10.1111/1346-8138.16501...
88 Olson N, Eckhardt D, Delano A. New-onset bullous pemphigoid in a COVID-19 patient. Case Rep Dermatol Med. 2021;2021:5575111. https://doi.org/10.1155/2021/5575111
https://doi.org/10.1155/2021/5575111...
There are few reports of skin lesions during or after asymptomatic COVID-19 infection.99 Ingravallo G, Mazzotta F, Resta L, Sablone S, Cazzato G, Cimmino A, et al. Inflammatory skin lesions in three SARS-CoV-2 swab-negative adolescents: a possible COVID-19 sneaky manifestation? Pediatr Rep. 2021;13:181-8. https://doi.org/10.3390/pediatric13020025
https://doi.org/10.3390/pediatric1302002...

Lichen planus (LP) is a rare complication of COVID-19 infection1010 Diaz-Guimaraens B, Dominguez-Santas M, Suarez-Valle A, Fernandez-Nieto D, Jimenez-Cauhe J, Ballester A. Annular lichen planus associated with coronavirus SARS-CoV-2 disease (COVID-19). Int J Dermatol. 2021;60:246-47. https://doi.org/10.1111/ijd.15338
https://doi.org/10.1111/ijd.15338...
and is mainly reported after COVID-19 vaccination.1111 Merhy R, Sarkis AS, Kaikati J, El Khoury L, Ghosn S, Stephan F. New-onset cutaneous lichen planus triggered by COVID-19 vaccination. J Eur Acad Dermatol Venereol. 2021;35:e729-e730. https://doi.org/10.1111/jdv.17504
https://doi.org/10.1111/jdv.17504...
Some post-COVID LP lesions involved only oral mucosa,1212 Burgos-Blasco P, Fernandez-Nieto D, Selda-Enriquez G, Melian-Olivera A, De Perosanz-Lobo D, Dominguez-Santas M, et al. COVID-19: a possible trigger for oral lichen planus? Int J Dermatol. 2021;60:882-3. https://doi.org/10.1111/ijd.15529
https://doi.org/10.1111/ijd.15529...
,1313 Alabdulaaly L, Sroussi H, Epstein JB. New onset and exacerbation of oral lichenoid mucositis following SARS-CoV-2 infection or vaccination. Oral Dis. 2022;28 Suppl 2:2563-7. https://doi.org/10.1111/odi.14257
https://doi.org/10.1111/odi.14257...
and the other cases were cutaneous and/or mucocutaneous LP.1010 Diaz-Guimaraens B, Dominguez-Santas M, Suarez-Valle A, Fernandez-Nieto D, Jimenez-Cauhe J, Ballester A. Annular lichen planus associated with coronavirus SARS-CoV-2 disease (COVID-19). Int J Dermatol. 2021;60:246-47. https://doi.org/10.1111/ijd.15338
https://doi.org/10.1111/ijd.15338...
,1414 Castillo JG, de la Torre Gomar FJ, Aguirre AS. Lichen planus after SARS-CoV-2 infection treated with lopinavir/ritonavir. Med Clin (Barc). 2021;156:468-9. https://doi.org/10.1016/j.medcli.2020.12.014
https://doi.org/10.1016/j.medcli.2020.12...
,1515 Saleh W, Shawky E, Halim GA, Ata F. Oral lichen planus after COVID-19, a case report. Ann Med Surg (Lond). 2021;72:103051. https://doi.org/10.1016/j.amsu.2021.103051
https://doi.org/10.1016/j.amsu.2021.1030...

LP may occur in children, although it is not a common skin disorder in this age group.1616 Payette MJ, Weston G, Humphrey S, Yu J, Holland KE. Lichen planus and other lichenoid dermatoses: kids are not just little people. Clin Dermatol. 2015;33:631-43. https://doi.org/10.1016/j.clindermatol.2015.09.006
https://doi.org/10.1016/j.clindermatol.2...
Herein, we report the appearance of extensive cutaneous LP following an asymptomatic COVID-19 infection in a child.

CASE REPORT

A 4-year-old male child was admitted to the dermatology ward of our referral hospital in Shiraz, South of Iran, due to generalized, intensely itchy lesions that started about 3 months before admission. The lesions were violaceous, flat-topped papules and plaques on the trunk, extremities, and face. The skin lesions gathered especially on the dorsal surface of hands, feet, forearms, and shins (Figure 1A). There was no sign of mucosal, hair, or nail involvement.

Figure 1
Clinical features. (A) Pruritic violaceous erythematous flat-topped papules and plaques on the upper extremities after asymptomatic COVID-19 infection. (B) Evolution of the lesions 3 months after treatment. (C) Post-inflammatory hyperpigmentation in 10 months follow-up.

The patient was a known case of single kidney disease and hypoglycemia since birth without recent significant problems. His mother had a history of localized cutaneous LP controlled by topical corticosteroid. He had not started any new medications recently and was not taking any drugs chronically. He had close household contact with a confirmed case of COVID-19 infection (his mother) about 2 weeks before the development of the lesions, without any symptoms of upper or lower respiratory tract infection, gastrointestinal infection, skin rash, or fever.

The clinical differential diagnoses included LP and psoriasis. Histopathological examination of the skin punch biopsy revealed hyperkeratosis without parakeratosis, focal increase in the granular cell layer, irregular acanthosis, and vacuolar degeneration of the basal cell layer with a band-like lymphocyte infiltrate at the dermo-epidermal junction. Colloid bodies were present at the lower level of the epidermis (Figure 2). Hence, the diagnosis of LP was confirmed.

Figure 2
Histopathologic features. (a) Hyperorthokeratosis, focal hypergranulosis, irregular acanthosis, vacuolar degeneration of the basal cell layer, and a band-like lymphocyte infiltrate at the dermo-epidermal junction (H&E stain, 100X). (b) Colloid bodies (green arrow) in the basal layer and pigment incontinence (blue arrows) in the papillary dermis (H&E stain, 400X).

Laboratory work-up revealed normal results of complete blood count, liver function tests, blood urea nitrogen, creatinine, fasting blood sugar, and C-reactive protein. Serological examination showed high titers of IgM for COVID-19.

The patient was treated with systemic corticosteroid (prednisolone 7.5 mg daily, increased to 12.5 mg daily), clobetasol propionate ointment at night, and topical tacrolimus 0.03% ointment twice daily (for face and flexural areas). Oral sedative antihistamines were prescribed to improve pruritus and disturbed sleep. After 1 month of follow-up, the skin lesion improved significantly (Figure 1B). Hence, we tapered prednisolone for 3 months and then discontinued it (Figure 1B). No relapse occurred in 10 months of follow-up (Figure 1C).

The patient's parent signed written informed consent and permitted the publication of the case and photography without identifying data. Maintaining the patient's confidentiality is guaranteed by researchers. The institutional ethics committee approved the case report (ethics code: IR.SUMS.MED.REC.1401.403).

DISCUSSION

COVID-19 is a multisystem disease, and several skin manifestations are described during and after this infection.11 Huynh T, Sanchez-Flores X, Yau J, Huang JT. Cutaneous manifestations of SARS-CoV-2 infection. Am J Clin Dermatol. 2022;23:277-86. https://doi.org/10.1007/s40257-022-00675-2
https://doi.org/10.1007/s40257-022-00675...
Several cases of newly developed or exacerbated skin disorders are reported after COVID-19 infection, including psoriasis, pityriasis rosea, vasculitis, alopecia areata, vitiligo, bullous pemphigoid, and many other skin diseases.66 Chularojanamontri L, Tuchinda P, Rujitharanawong C, Hunnangkul S, Pochanapan O, Panjapakkul W, et al. New-onset and exacerbated skin diseases after COVID-19 infection: a systematic review. J Dermatol. 2022;49:e419-21. https://doi.org/10.1111/1346-8138.16501
https://doi.org/10.1111/1346-8138.16501...
88 Olson N, Eckhardt D, Delano A. New-onset bullous pemphigoid in a COVID-19 patient. Case Rep Dermatol Med. 2021;2021:5575111. https://doi.org/10.1155/2021/5575111
https://doi.org/10.1155/2021/5575111...
Rarely, inflammatory skin lesions are reported after asymptomatic, serologically-confirmed COVID-19 infection.99 Ingravallo G, Mazzotta F, Resta L, Sablone S, Cazzato G, Cimmino A, et al. Inflammatory skin lesions in three SARS-CoV-2 swab-negative adolescents: a possible COVID-19 sneaky manifestation? Pediatr Rep. 2021;13:181-8. https://doi.org/10.3390/pediatric13020025
https://doi.org/10.3390/pediatric1302002...

LP is a T-cell-mediated autoimmune dermatologic disorder. The development of autoimmunity mostly depends on a combination of factors such as genetic predisposition, inadequate immune response, and environmental triggers such as viral infections.1717 Hussein HM, Rahal EA. The role of viral infections in the development of autoimmune diseases. Crit Rev Microbiol. 2019;45:394-412. https://doi.org/10.1080/1040841X.2019.1614904
https://doi.org/10.1080/1040841X.2019.16...
While the causal effect of microbial agents in the pathogenesis of LP is debated,1818 Baek K, Choi Y. The microbiology of oral lichen planus: is microbial infection the cause of oral lichen planus? Mol Oral Microbiol. 2018;33:22-8. https://doi.org/10.1111/omi.12197
https://doi.org/10.1111/omi.12197...
the association of LP with several viruses (e.g., hepatitis C virus, human herpes virus-7, and Ebstein-Barr virus) is disclosed.1919 Ashraf S, Al-Maweri SA, Alaizari N, Umair A, Ariffin Z, Alhajj MN, et al. The association between Epstein-Barr virus and oral lichen planus: a systematic review and meta-analysis. J Oral Pathol Med. 2020;49:969-76. https://doi.org/10.1111/jop.13093
https://doi.org/10.1111/jop.13093...
2121 Pinelli S, Basile S, Panici PB, D'Erme AM, Romanelli M, Plotti F, et al. Association between HCV infection and cutaneous-mucosal lichen planus: an update. Eur J Dermatol. 2017;27:329-31. https://doi.org/10.1684/ejd.2017.3009
https://doi.org/10.1684/ejd.2017.3009...

New-onset LP has been sporadically reported after COVID-19 infection.10,12,14,15 More LP cases are reported after COVID-19 vaccination compared to COVID-19 infection.1111 Merhy R, Sarkis AS, Kaikati J, El Khoury L, Ghosn S, Stephan F. New-onset cutaneous lichen planus triggered by COVID-19 vaccination. J Eur Acad Dermatol Venereol. 2021;35:e729-e730. https://doi.org/10.1111/jdv.17504
https://doi.org/10.1111/jdv.17504...
,1313 Alabdulaaly L, Sroussi H, Epstein JB. New onset and exacerbation of oral lichenoid mucositis following SARS-CoV-2 infection or vaccination. Oral Dis. 2022;28 Suppl 2:2563-7. https://doi.org/10.1111/odi.14257
https://doi.org/10.1111/odi.14257...
The post-COVID LP lesions may involve skin,1111 Merhy R, Sarkis AS, Kaikati J, El Khoury L, Ghosn S, Stephan F. New-onset cutaneous lichen planus triggered by COVID-19 vaccination. J Eur Acad Dermatol Venereol. 2021;35:e729-e730. https://doi.org/10.1111/jdv.17504
https://doi.org/10.1111/jdv.17504...
oral mucosae,99 Ingravallo G, Mazzotta F, Resta L, Sablone S, Cazzato G, Cimmino A, et al. Inflammatory skin lesions in three SARS-CoV-2 swab-negative adolescents: a possible COVID-19 sneaky manifestation? Pediatr Rep. 2021;13:181-8. https://doi.org/10.3390/pediatric13020025
https://doi.org/10.3390/pediatric1302002...
,1010 Diaz-Guimaraens B, Dominguez-Santas M, Suarez-Valle A, Fernandez-Nieto D, Jimenez-Cauhe J, Ballester A. Annular lichen planus associated with coronavirus SARS-CoV-2 disease (COVID-19). Int J Dermatol. 2021;60:246-47. https://doi.org/10.1111/ijd.15338
https://doi.org/10.1111/ijd.15338...
or both.1010 Diaz-Guimaraens B, Dominguez-Santas M, Suarez-Valle A, Fernandez-Nieto D, Jimenez-Cauhe J, Ballester A. Annular lichen planus associated with coronavirus SARS-CoV-2 disease (COVID-19). Int J Dermatol. 2021;60:246-47. https://doi.org/10.1111/ijd.15338
https://doi.org/10.1111/ijd.15338...
,1515 Saleh W, Shawky E, Halim GA, Ata F. Oral lichen planus after COVID-19, a case report. Ann Med Surg (Lond). 2021;72:103051. https://doi.org/10.1016/j.amsu.2021.103051
https://doi.org/10.1016/j.amsu.2021.1030...
To the best of our knowledge, all reported post-COVID LP cases were adults.10,12-15 One of these cases presented with annular LP.1010 Diaz-Guimaraens B, Dominguez-Santas M, Suarez-Valle A, Fernandez-Nieto D, Jimenez-Cauhe J, Ballester A. Annular lichen planus associated with coronavirus SARS-CoV-2 disease (COVID-19). Int J Dermatol. 2021;60:246-47. https://doi.org/10.1111/ijd.15338
https://doi.org/10.1111/ijd.15338...
Sood et al.2222 Sood A, Raghavan S, Batra P, Sharma K, Talwar A. Rise and exacerbation of oral lichen planus in the background of SARS-CoV-2 infection. Med Hypotheses. 2021;156:110681. https://doi.org/10.1016/j.mehy.2021.110681
https://doi.org/10.1016/j.mehy.2021.1106...
proposed the hypothesis of the pathogenesis of post-COVID oral LP. They discussed the related target receptors, abnormal T-cell responses, elevated levels of cytokines, disrupted immune permeability barrier, and vitamin D deficiency as the possible causes of post-COVID oral LP.2222 Sood A, Raghavan S, Batra P, Sharma K, Talwar A. Rise and exacerbation of oral lichen planus in the background of SARS-CoV-2 infection. Med Hypotheses. 2021;156:110681. https://doi.org/10.1016/j.mehy.2021.110681
https://doi.org/10.1016/j.mehy.2021.1106...

LP is not a common dermatologic disorder in the pediatric age group. Most pediatric LP cases occur in school-aged children.1616 Payette MJ, Weston G, Humphrey S, Yu J, Holland KE. Lichen planus and other lichenoid dermatoses: kids are not just little people. Clin Dermatol. 2015;33:631-43. https://doi.org/10.1016/j.clindermatol.2015.09.006
https://doi.org/10.1016/j.clindermatol.2...
The clinical presentation of classic LP is similar in adults and children and includes pruritic, polygonal, flat-topped, purple papules and plaques with fine scale and Wickham's striae. The lesions may involve any body area but have a predilection for flexural parts of extremities, dorsa of the hands, shins, trunk, and sacral area.1616 Payette MJ, Weston G, Humphrey S, Yu J, Holland KE. Lichen planus and other lichenoid dermatoses: kids are not just little people. Clin Dermatol. 2015;33:631-43. https://doi.org/10.1016/j.clindermatol.2015.09.006
https://doi.org/10.1016/j.clindermatol.2...
Oral involvement is less common in pediatric LP compared to adult LP.2323 Merhy R, Sarkis AS, Assaf J, Afiouni R, Zeinaty P, Kechichian E, et al. Pediatric lichen planus: a systematic review of 985 published cases. Int J Dermatol. 2022;61:416-21. https://doi.org/10.1111/ijd.15678
https://doi.org/10.1111/ijd.15678...

Pediatric LP is usually treated with topical corticosteroids in the limited disease and systemic corticosteroids in the more extensive disease. Phototherapy or systemic immunomodulators (e.g., dapsone, antimalarial drugs, methotrexate, and cyclosporine) are other therapeutic choices in the extensive pediatric LP.2323 Merhy R, Sarkis AS, Assaf J, Afiouni R, Zeinaty P, Kechichian E, et al. Pediatric lichen planus: a systematic review of 985 published cases. Int J Dermatol. 2022;61:416-21. https://doi.org/10.1111/ijd.15678
https://doi.org/10.1111/ijd.15678...

Our case was a child of pre-school age that developed extensive, severe classic cutaneous familial LP following asymptomatic serologically confirmed COVID-19 infection. He was treated successfully with topical and systemic corticosteroids. This case is unusual in LP's age, severity, and presentation setting.

To the best of our knowledge, this is the first case of post-COVID LP in the pediatric age group and the first case of LP following asymptomatic COVID-19 infection. Thus, it is essential to consider LP among the differential diagnoses of pediatric post-COVID inflammatory skin lesions, either in the patients recovering from COVID-19 infection or in the suspicious asymptomatic cases that have been in close contact with COVID-19-infected patients.

  • Funding
    This study did not receive any funding.

REFERENCES

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    » https://doi.org/10.1111/odi.14257
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    » https://doi.org/10.1016/j.medcli.2020.12.014
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    Saleh W, Shawky E, Halim GA, Ata F. Oral lichen planus after COVID-19, a case report. Ann Med Surg (Lond). 2021;72:103051. https://doi.org/10.1016/j.amsu.2021.103051
    » https://doi.org/10.1016/j.amsu.2021.103051
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    » https://doi.org/10.1016/j.clindermatol.2015.09.006
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    » https://doi.org/10.1111/omi.12197
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    Ashraf S, Al-Maweri SA, Alaizari N, Umair A, Ariffin Z, Alhajj MN, et al. The association between Epstein-Barr virus and oral lichen planus: a systematic review and meta-analysis. J Oral Pathol Med. 2020;49:969-76. https://doi.org/10.1111/jop.13093
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    Pinelli S, Basile S, Panici PB, D'Erme AM, Romanelli M, Plotti F, et al. Association between HCV infection and cutaneous-mucosal lichen planus: an update. Eur J Dermatol. 2017;27:329-31. https://doi.org/10.1684/ejd.2017.3009
    » https://doi.org/10.1684/ejd.2017.3009
  • 22
    Sood A, Raghavan S, Batra P, Sharma K, Talwar A. Rise and exacerbation of oral lichen planus in the background of SARS-CoV-2 infection. Med Hypotheses. 2021;156:110681. https://doi.org/10.1016/j.mehy.2021.110681
    » https://doi.org/10.1016/j.mehy.2021.110681
  • 23
    Merhy R, Sarkis AS, Assaf J, Afiouni R, Zeinaty P, Kechichian E, et al. Pediatric lichen planus: a systematic review of 985 published cases. Int J Dermatol. 2022;61:416-21. https://doi.org/10.1111/ijd.15678
    » https://doi.org/10.1111/ijd.15678

Publication Dates

  • Publication in this collection
    23 Oct 2023
  • Date of issue
    2024

History

  • Received
    01 Feb 2023
  • Accepted
    20 May 2023
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