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Dermpath & Clinic: Exogenous ochronosis

Article  in  European Journal of Dermatology · April 2019

DOI: 10.1684/ejd.2019.3563

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Cecile Lesort Clemmie Martin

CHU de Lyon - Groupement Hospitalier Edouard Herriot CHU de Lyon - Hôpital Femme-Mère-Enfant
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Axel Patrice Villani Jean Kanitakis

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 failed to respond to long-term conventional antifungal ther-
apy with itraconazole. For the in vitro study, five sporothrix
strains were assigned to six different groups: LED
irradiation with photosensitizer (L+M+), LED irradiation
without photosensitizer (L+M-), photosensitizer alone (L-
M+), without exposure to LED light or photosensitizer
(L-M-), and finally two groups of LED irradiation plus
photosensitizer with and without itraconazole (L+M+I+
and L+M+I-). Although L-M-, L+M- and L-M+ groups
revealed similar residual antimicrobial activities, in the
L+M+ group, residual antimicrobial activities were signif-
icantly lower than in other groups. These results support
the idea that photosensitizer or LED light alone have no
fungicidal effect, and that PDT plus itraconazole has a syn-
ergistic effect against sporothrix globosa. For the in vivo
study, mice were infected with sporothrix and treated with
sodium chloride (control group), antifungal itraconazole
(intraconazole group), a combination of itraconazole and
PDT (itraconazole + PDT group), and PDT (PDT group).
Two days after the injection of the fungus, mice developed
Copyright © 2019 John Libbey Eurotext. Téléchargé par M. JEAN KANITAKIS le 22/05/2019.
soft subcutaneous nodules. The nodules reached a peak in
size on Day 20 (control group) and Day 8 (all other groups).
The decrease in nodule size in the itraconazole + PDT group
was significantly greater than in the PDT and itraconazole
groups. However, no significant difference was observed
between the PDT and itraconazole groups. It seems that
PDT and itraconazole have a synergistic effect and are
effective as treatment for sporotrichosis in mouse mod-
els. Similar outcomes have been described for candida spp.
in other studies. This study presents valuable information
about PDT effectiveness in the treatment of sporotrichosis.
Combination therapy with PDT plus itraconazole could be
an effective treatment for those patients who fail to respond
to itraconazole alone. In the future, PDT may be a valuable
adjuvant therapy for a wide range of infectious disorders.
References
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in Japanese dermatitis herpetiformis. Br J Dermatol 2016; 174:
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Dermpath & Clinic: Exogenous ochronosis
Joann LE BORGNE DE LAVILLANDRE, Cécile LESORT,
Clemmie MARTIN, Axel Patrice VILLANI,
Jean KANITAKIS
Hospices Civils de Lyon, Dermatology Department, Edouard Herriot
Hospital, Claude Bernard Lyon University, Lyon, France
e-mail: cecile.lesort@laposte.net
A 47-year-old woman from Cameroon with skin phototype
V consulted for dyschromic macules of the face. Physi-
cal examination revealed bluish-black pigmentation on the
temporal, perioral region, frontal areas, and nose, which had
appeared in merely two weeks after sun exposure in France
(figure 1A). The patient had been applying a skin-lightening
cream containing hydroquinone on her face for the past two
months. We initially suspected lupus erythematosus or der-
matomyositis for this facial eruption after sun exposure and
performed a skin biopsy from a temporal lesion. Histologi-
cal examination showed clusters of irregularly shaped thick
fibres with round or indented/angular contours in the pap-
illary dermis. These fibres were ochre-coloured and had a
basophilic/violaceous peripheral rim (figures 1B, C); they
were weakly stained by orcein but were negative with alcian
blue, toluidine blue, Perls, and Fontana-Masson stains, and
were not birefringent under polarized light. The patient’s
urinary levels of homogentisic acid were normal. The diag-
nosis of exogenous ochronosis was established.
Ochronosis is a rare disorder characterized by blue-black or
grey-blue pigmentation of the skin, which reflects the his-
tological finding of abnormal yellow-brown deposits in the
dermis. Exogenous ochronosis (EO) is clinically and histo-
logically similar to endogenous ochronosis (alkaptonuria),
an autosomal recessive condition due to deficiency in
homogentisate 1,2-dioxygenase, but lacks systemic symp-
toms and is not inherited. Predisposing factors of EO
include sun exposure, application of skin-lightening agents,
such as hydroquinone, phenol or resorcinol, or follow-
ing the use of systemic antimalarials. The pathogenesis is
poorly understood but the most likely hypothesis stipulates
that local competitive inhibition of the enzyme homogen-
tisic oxidase by hydroquinone leads to accumulation of
homogentisic acid in the skin and ochronotic pigment in
the papillary dermis [1].
Histological examination is mandatory to confirm the diag-
nosis; it shows ochre-coloured, sometimes banana-shaped
fibres in the papillary dermis and swollen collagen bun-
dles. Dermoscopy and reflectance confocal microscopy are
non-invasive tools which may help to establish the diag-
nosis of EO [2, 3]. Typical dermoscopy findings include
irregular brown-grey globular-like and arciform-to-annular
structures around the follicular openings. Reflectance con-
focal microscopy reveals dark, well-defined, round-to-oval
and banana-shaped structures in the papillary dermis, which
correlate with the histological findings. However, these
239
 A B
Figure 1. A) Dark brown pigmented macules of the face. B, C) Histological examination after haematoxylin-eosin-saffron
Copyright © 2019 John Libbey Eurotext. Téléchargé par M. JEAN KANITAKIS le 22/05/2019.
staining shows characteristic ochre-coloured fibres with a basophilic rim in the papillary dermis ([B] ×100, [C] ×250).
structures do not correspond to any previously described
structures of the skin.
The treatment of EO is complex. Skin-lightening products
should be discontinued. Several topical treatments have
been tried with inconsistent results [3], including 0.05%
retinoic acid, 20% azelaic acid, and 1% hydrocortisone.
More recent alternatives include the use of Q-switched
lasers of longer wavelengths, alone or in combination with
CO2 laser, and a combination of laser and dermabrasion. 
Disclosure. Conflicts of interest: none.
References
1. Bhattar P, Zawar V, Godse K, Patil S, Nadkarni N, Gautam M.
Exogenous Ochronosis. Indian J Dermatol 2015; 60: 537-43.
2. Gil I, Segura S, Martínez-Escala E, et al. Dermoscopic and
reflectance confocal microscopic features of exogenous ochronosis.
Arch Dermatol 2010; 146: 1021-5.
3. Simmons BJ, Griffith RD, Bray FN, Falto-Aizpurua LA, Nouri
K. Exogenous ochronosis: a comprehensive review of the diag-
nosis, epidemiology, causes, and treatments. Am J Clin Dermatol
2015; 16: 205-12.
doi:10.1684/ejd.2019.3563
DermSurgery: Z-plasty transposition flap
for reconstruction of the medial eyebrow
Álvaro MACHADO1 , Mónica CAETANO1,2,3 ,
Manuela SELORES1,2,3
1 Department of Dermatology, Centro Hospitalar,
e-mail: alvmac@gmail.com
2 Instituto de Ciências Biomédicas Abel Salazar,
3 Dermatology Research Unit, Centro Hospitalar Universitário do Porto,
Porto, Portugal
240
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An 81-year-old female patient presented with an 18-mm
nodular basal cell carcinoma of the left medial eyebrow
(figure 1A). The tumour was surgically removed with a
4-mm margin, resulting in a 26-mm wide defect. For
reconstruction, a Z-plasty transposition flap was performed
(figures 1B, C). Six months after surgery, no recurrence
was noted and a good functional and aesthetic outcome
was obtained.
The eyebrows are a prominent feature of the upper third
of the face and play an important role in facial aesthetics,
emotional expression, and non-verbal communication [1].
In addition, the skin of the eyebrows is thick and contains
abundant sweat and sebaceous glands [2], therefore, in cases
of inevitable skin transplantation, donor sites should have
similar properties. Hence, the reconstruction of eyebrow
defects may represent a challenge for the dermatological
surgeon. If possible, direct closure should be performed,
especially for small defects.
In our case, direct closure was not feasible given the defect
size and the fact that this would have had a significant func-
tional and aesthetic impact. Several techniques have been
described for larger defects, including grafts and numer-
ous flaps, such as Limberg, Dufourmentel, O-Z or A-T.
Transposition flaps take advantage of regional laxity by
mobilizing tissue from an adjacent area of excess laxity
into the area without slack. Also, the primary advantages
of transposition flaps include less undermining, compared
to large sliding flaps, and the superior ability to displace
tension away from the defect and from free margins [3].
In particular, the Z-plasty transposition flap is an excellent
choice for scar camouflage [4]. Thus, a Z-plasty transposi-
tion flap offered an excellent reconstructive option in our
case.
Z-plasty is a procedure which involves the transposition of
two interdigitating triangular flaps [5]. Traditional Z-plasty
is designed to ensure that the length of both the lateral and
central limbs and the angles created by their junction are
equal (figure 1B). In our case, due to the size and location of
the defect, a modified approach was performed (figure 1B).
In this technique, full-thickness skin flaps are undermined
at the level of the subcutaneous fat. Adequate undermining
EJD, vol. 29, n◦ 2, March-April 2019