MAEDICA – a Journal of Clinical Medicine

Mædica -a
2018; 13(4): 336-xxx Journal of Clinical Medicine 2018; 13(4): 336-339
https://doi.org/10.26574/maedica.2018.13.4.336
C ase reports

Coexistence of Psoriasis, Vitiligo and

Oral Lichen Planus – a New Variant of
Multiple Autoimmune Syndrome?
Cristina VAJAITUa, Vlad Mihai VOICULESCUa, b, Carmen DRAGHICIa, Iulia SOLOMONa,
Mihai LUPUb, c
Department of Dermatology and Allergology, “Elias” Emergency University Hospital,
a

Bucharest, Romania
b
”Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania
MEDAS Medical Center, Bucharest, Romania
c

ABSTRACT
Multiple autoimmune syndrome is a rare condition, defined by the association of at least three autoimmune
disorders in the same patient, and first described by Humbert and Dupond in 1988. Psoriasis, vitiligo, and
oral lichen planus are all common dermatological disorders in the general population, and the coexistence of
these three diseases in these exact clinical variants has never been reported in the literature before. Herein, we
describe an unusual case of co-occurrence of psoriasis vulgaris, vitiligo, and oral lichen planus. A 67-year-
old Romanian female patient with a history of both vitiligo and psoriasis presented with white, shiny striae,
organized in a reticular pattern on the mucosa of the cheek and whitish homogeneous irregularities similar
to leukoplakia involving the dorsal side of the tongue. Histology examination confirmed oral lichen planus.
The rarity of this case is highlighted by the coexistence of psoriasis, vitiligo, and oral lichen planus in
the absence of cutaneous lichen planus lesions, an association which may be a newly described variant of
multiple autoimmune syndrome.
Keywords: vitiligo, lichen planus, oral, psoriasis, hypopigmentation, female, autoimmunity.

INTRODUCTION ported in the literature, although an immuno-

P
logical link between them has been previously
soriasis, oral lichen planus, and vitiligo suggested (1-3).
are all common dermatological disor- Psoriasis is a common, chronic inflammatory
ders in the general population. Coexis- skin disease characterized by T-cell mediated hy-
tence of these three diseases, in these perproliferation of keratinocytes. Despite the ad-
clinical variants, has never been re- vances in the last decades, the pathogenesis of
Address for correspondence:
Mihai Lupu, MD, PhD student
Targu Neamt Street No 9, Bucharest, Postal code 062052, Romania
Tel: 00-40-740237450
Email: lupu.g.mihai@gmail.com

Article received on the 11th of October 2018 and accepted for publication on 11th of December 2018.

336 Maedica A Journal of Clinical Medicine, Volume 13, No.4, 2018

Coexistence of Psoriasis, Vitiligo and Oral Lichen Planus - a New Variant of Multiple Autoimmune Syndrome?

psoriasis is still not fully understood but current

data indicates that human leukocyte antigen
Cw0602 is associated with an increased suscep-
tibility for psoriasis (4). Two cytokines, interleu-
kin (IL)-23 and IL-22, have been found to play a
crucial role in psoriasis pathogenesis (5).
Vitiligo is an acquired disorder of pigmenta-
tion due to a substantial loss of functioning epi-
dermal and/or hair follicle melanocytes. Recent
research has shown that IL-17 secreting cells in-
duced by IL-1β are present in increased numbers
in the serum of vitiligo patients, thus inducing a
proinflammatory state (6). In the meantime, the
number of regulatory T cells is reduced and their
ability to suppress CD8+ cells is diminished (7).
Oral lichen planus (OLP) is a chronic, T cell-
mediated autoimmune disease, in which auto-
cytotoxic CD8+ T cells attack the basal layer. FIGURE 1. Clinical presentation of colocalized vitiliginous and
Cytokines such as IFN-gamma, TNF-alpha, psoriatic lesions (1A) and OLP located on the tongue (1B) and cheek
IL-1-alpha, IL-6 and IL-22 associated with T hel­ mucosa (C-D)
per cell response may play a role in the pathoge­
nesis of lichen planus (8, 9). between psoriatic plaques and vitiliginous
It has been suggested that a combination of patches.
genetic susceptibility and environmental factors, One year after the onset of psoriasis, she de-
which underlie most autoimmune conditions, veloped white, shiny striae, disposed in a reticu-
explains the higher possibility of multiple auto- lar pattern, localised on the mucosa of the cheek
immune diseases appearing in one patient. (Figure 1C-D), and whitish homogeneously di­
Lopez-Jornet et al. have shown a higher inci- stributed irregularities, similar to leukoplakia, in-
dence of other autoimmune diseases in patients volving the dorsal side of the tongue (Figure 1B).
with OLP when compared with patients without Oral cavity examination revealed that she had
OLP (7% vs 4%), thus suggesting a higher chance dental fillings. A biopsy was taken from the le-
for these patients of associating vitiligo or psoria- sions on the oral mucosa, which histologically
sis compared to the rest of the population (10). q showed hyperkeratosis with irregular acanthosis
and focal thickening in the granular layer, Civatte
CASE REPORT bodies in the lower epidermis, a band-like in-
flammatory infiltrate composed of histiocytes
A 67-year-old woman presented with an
18 year history of hypopigmented skin
patches on the extremities, trunk, and face
and lymphocytes, mainly T cells, localised in the
superficial dermis (Figure 2B), thus confirming
(Figure 1A), which had already been diagnosed the diagnosis of oral lichen planus. There was no
as vitiligo after clinical examination, Wood lamp
examination, and skin biopsy. About two years
prior to presentation and sixteen years after the
onset of vitiligo, she developed scaly erythema-
tous plaques on the elbows, dorsum of the
hands, and scalp (Figure 1A). A skin biopsy from
one of these plaques revealed epidermal acan-
thosis with hyperkeratosis, parakeratosis, and in-
filtrations of neutrophils into the stratum corne-
um and subcorneal zone, compatible with the FIGURE 1. Histopathological aspect of the biopsy taken from a
clinical diagnosis of psoriasis vulgaris (PV) psoriatic lesion (2A) and from an OLP lesion located on the cheek
(Figure 2A). Note that there was co-localization mucosa (2B) (Hematoxylin and eosin stain).

Maedica A Journal of Clinical Medicine, Volume 13, No. 4, 2018 337

Coexistence of Psoriasis, Vitiligo and Oral Lichen Planus - a New Variant of Multiple Autoimmune Syndrome?
evidence of LP elsewhere, such as the skin, geni-
talia, nails, or scalp.
Routine laboratory tests were all unremarka­
ble. No finding suggestive of any type of connec-
tive tissue or rheumatic disease was evident.
On the basis of the clinical and histological
findings, the diagnoses of psoriasis vulgaris, vitili-
go and oral lichen planus were established and
she was started on systemic antioxidant therapy,
topical steroids for psoriasis lesions and intrabuc-
cal steroids for the OLP lesions, with a follow-up
visit scheduled for a month later. q
DISCUSSION
C oexistence of psoriasis, vitiligo, and OLP and
has never been reported in the literature be-
fore. There is one report in which psoriasis, viti­
ligo, and cutaneous lichen planus occur together
(2). However, there are more frequent reports
about the association between pairs of these
three immune disorders.
The association of vitiligo and lichen planus
has been previously described (11, 12). Most
cases associated vitiligo with the cutaneous form
of lichen planus (LP), with a great majority of pa-
tients having LP lesions superimposed on vitiligi-
nous areas (13). Lesion distribution was particu-
lar in our case due to the association of vitiligo
with OLP, in the absence of cutaneous LP lesions.
The common pathogenic way of LP and vitiligo
could be explained by the autoimmune destruc-
tion of basal keratinocytes leading to a cross-re-
action with antigens located on melanocytes.
Even though the association between vitiligo
and psoriasis was first described in 1890 (14),
the relationship between the two diseases is still
subject to discussions. Some reported cases de-
scribe the two dermatoses occurring together
with strict co-localization (15-17), while others
deny strict co-localization (3). Some authors (18,
19) conclude that psoriasis lesions have the same
occurring rate on both vitiliginous lesions and on
normal skin. In our case there was, although not
strict, a co-localization between psoriasis and vi­
tiligo lesions. Autoimmunity, common neuro-
peptides, and Koebner’s phenomenon have all
been called to explain the pathogenic link
I n conclusion, we report a case of coexistence of
between the two disorders. The concurrence of
vitiligo and psoriasis highlights the link between
the pathogeneses of the two conditions. In vitro
studies have confirmed that patients with vitiligo
338 Maedica A Journal of Clinical Medicine, Volume 13, No. 4, 2018
have enhanced neurotensin, which in turn leads
to a melanocytic production of TNF-alpha (20)
and Jain et al. showed that patients with vitiligo
present elevated levels of TNF-alpha in peri-viti-
liginous areas (21). TNF-alpha is a key cytokine
in innate immune responses and is increased in
psoriatic lesions (22-24), its central role in psoria-
sis being demonstrated by TNF-alpha blockers
therapies (25). Thus, TNF-alpha may represent a
common link in the pathogeneses of vitiligo and
psoriasis, and deserves further evaluation as a
target for treatment.
Coexistence of OLP and psoriasis is rarely re-
ported in the literature (26), the majority of cases
associating psoriasis with cutaneous LP (27, 28).
Cytokine imbalance, such as excess production
of IFN-alpha, and T-cell-mediated autoimmuni-
ty, as proven by leukocyte migration-inhibition
assays, are known to occur in both conditions.
Although our patient had no identifiable trigger
such as a virus or a drug, some antigenic stimulus
may have triggered IFN-alpha production. It is
speculated that activation of both CD4+ and
CD8+ T cells may be responsible in cases of co-
existence between psoriasis and LP (27, 29).
The white striae characteristic for OLP were
mainly located in the posterior part of the buccal
region, near the dental fillings our patient had. A
mechanism of contact hypersensitivity can be in-
ferred, as metal ions are considered a potential
trigger factor for OLP. Replacement of the dental
fillings may be necessary in this case in order to
obtain disease control. A reduced quantity of
melanin could be an underlying cause for the
development of other disorders, because cell
damage in vitiliginous areas incites immunity re-
actions and Koebner’s phenomenon. Ujiie et al.
already inferred that areas with low amounts of
melanin in association with Koebner’s pheno­
menon and sun exposure can be effective in
initiating psoriasis and LP in patients with
viti­ligo (2). q
CONCLUSION
three immune dermatoses in a single patient by
the common etiological background of autoim-
munity and Koebner’s phenomenon. The rarity
of this case is highlighted by the coexistence of
psoriasis, vitiligo, and oral lichen planus in the
absence of cutaneous lichen planus lesions, an
Coexistence of Psoriasis, Vitiligo and Oral Lichen Planus - a New Variant of Multiple Autoimmune Syndrome?

association which, to the best of our knowledge,
may be a newly described variant of multiple au-
toimmune syndrome. A combination of these
diseases in one patient is a unique situation and,
at the same time, a much greater therapeutic
challenge in comparison to the each condition
occurring separately or in pairs. q
Conflicts of interest: none declared.
Financial support: none declared.

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