Discussion
Three main factors of keloids are
known: infection, deep injury and mechanical trauma [5]. So
far, only four cases of keloid
scars following pemphigus have been reported [1–4]. Gupta et al.
reported a case of uninfected PV erosions that healed with keloids
despite high dose of systemic steroids [2]. Shivaswamy et al.
described keloids in a patient whose pemphigus was treated, inter
alia , with cyclosporin. The onset of keloids in this patient was
attributed to cyclosporin [3]. In the latest two cases, such as in
our patient, extensive keloids appeared in patients whose pemphigus had
been superinfected. Authors suggested that secondary infection induced
keloids via dermal damage. However, the paucity of cases of keloids
following PV flares contrasts with the frequency of superinfections
which are common in pemphigus, suggesting the presence of other unknown
factors [1,4].
Our case is notable due to the severity of the PV flare which was
refractory to conventional treatment and then complicated by ecthyma
gangrenosum. Re-epithelialization took more than one month and ended
with extensive keloids after three months. It is reasonable that
superinfection led to deep intradermal inflammation and keloid
formation. In fact, keloids occurred in previously superinfected PV
areas. In this context, there are reports of keloid onset following
herpetic infections, with the keloids involving previously infected
areas [6]. An alternative hypothesis is that erosions in our patient
took exceptionally long to heal, which maintained a long inflammation
and scarring process with a chronic fibroblast activation. This does not
exclude the potential role of apoptotic cells as well as increased
levels of tumor necrosis factor-alpha, found in PV, that may contribute
to the dysregulation of fibroblasts, and lead to keloids in susceptible
patients [7]. Intralesional steroids are generally proposed in
patients with keloids. Yet, our patient is under high systemic doses of
steroids, making this alternative debatable as we can opt for
compression clothing and active monitoring.