ARTICLE
Auteur(s) : Işıl KILINÇ, Glsm GENÇOGLAN, Işıl INANIR*
Tugrul DERELI
Ege University Medical Faculty, Department of Dermatology,
35100 Bornova, Izmir, Turkey. *Celal Bayar University Medical
Faculty, Department of Dermatology, Manisa, Turkey.
Reprints: T. Dereli 108\32 sok. No: 32\6
35350 Esenyali‐Izmir Turkey. Fax: (+90)
232 339 97 02 E‐mail: tddereliyahoo.com
Article accepted on 29\06\03 Key words: Solid facial
edema of acne is an uncommon skin condition which occurs as a
complication of acne vulgaris. Though this persistent, nontender
solid swelling can involve the whole face, it mainly locates on the
forehead, upper eyelids, nasal saddle, nasolabial folds, and
cheeks. Clinical appearance of the patient is typical with a
persistent and prominent swelling of the upper eyelid and widening
of the nasal saddle [1‐8]. Besides the unclear etiopathogenesis,
treatment is also unsatisfactory. A wide range of treatment
modalities, including antibiotics, systemic steroids, X‐rays, and
compression garments have been tried, resulting in various
responses [1, 3, 6]. Isotretinoin is reported to be the most
beneficial modality either alone [7] or in combination with
ketotifen [6] or clofazimine [5] which results in partial or
complete remission. We report a case of solid facial edema of acne,
which was unresponsive to isotretinoin. The pathogenesis and
possible causes leading to the failure of treatment are also
discussed.
Case report
A 25‐year‐old man attended to our clinic in February 2002
with a persistent swelling on his upper face existing for
four years. Initially, the edema on the upper eyelids was more
prominent in the morning and resolved during the day. Intramuscular
triamcinolone acetonide 40 mg per month was administrated for
three months by the physician he had consulted, and a complete
resolution was obtained. But the edema recurred after a short time,
had gradually spread over the nasal saddle, forehead, and upper
part of the cheeks. It has been persistent and unchanged for the
last three years. The anamnesis revealed that he had suffered from
acne vulgaris since the age of 14, but his acne recovered one year
before the onset of facial edema. His personal and familial medical
history was otherwise normal.
On dermatological examination, solid swelling over the forehead,
nose, cheeks, periorbital and glabellar areas was observed (Fig. 1). There were
also multiple pitting scars on the whole face but no inflammatory
acne lesions. Punch biopsy taken from the forehead revealed a dense
perivascular, periadnexial infiltrate consisting of lymphocytes,
histiocytes, plasma cells and numerous mast cells as well as
fibrosis in deep dermis.
.
Laboratory investigations were all within normal limits.
After a course of oral tetracycline therapy, 500 mg b.i.d.
for 2 months, there was no recovery. Isotretinoin, 40 mg
daily (0.5 mg\kg) administrated for 4 months also did not
reveal any improvement. The therapy was stopped owing to severe
headaches and high serum triglyceride level up to 360 mg\dl in
the last month.
The administration of colchicum 1.5 mg daily in three doses
for the regulation of collagen synthesis was unsatisfactory. For
the last five months, he has been treated with intralesional
injections of 5 mg\ml triamcinolone once monthly. The maximum
amount in one application for all of the face was 20 mg. A
temporary improvement for 2‐3 weeks was obtained and the
patient reported a better quality of life.
Discussion
The pathogenesis of solid facial edema associated with acne is
unknown. Conelly and Winkelmann who reported the first cases in
1985, assumed that chronic cutaneous inflammation resulting from
acne may cause a limited cellulitis and a progressive edema
resembling the edema of the legs after recurrent cellulitis
[1].
Friedman et al. pointed out that there was no apparent
correlation between the clinical course of acne vulgaris and edema
[7]. It is not necessary for all patients to have very severe acne
lesions. In almost all reported cases, the edema had appeared at
least one year after the onset of acne. The edema in our case was
interesting since it had begun after the remission of acne
vulgaris. The long preceding acne period supports the hypothesis
that persistent edema is a consequence of the chronic
inflammation.
In our case, histopathological examination revealed remarkable
mast cell accumulation in the fibrotic dermis. Jungfer et
al., who also showed numerous mast cells in the infiltrate and
fibrosis in the dermis, claimed that these mast cells might be
responsible for fibrosis and play a role in the pathogenesis [6].
Numerous mast cells in reticular dermis have also been demonstrated
similarly in patients with chronic venous insufficiency [9].
Perhaps inherited factors are also involved in the pathogenesis as
shown in the twin cases of Tosti et al. [4].
Solid facial edema of acne is difficult to treat. The two months
course of oral tetracycline therapy was ineffective in our patient.
After treatment with isotretinoin, 0.5 mg\kg per day, for
4 months, there was also no remarkable change in the edema. In
contrast to our case, isotretinoin was reported to be successful by
several authors with complete or partial improvement [5‐7, 10].
As seen in Table I, the duration of
swelling was less than two years in all the cases treated with
isotretinoin (mean 14 months). The beneficial effect of
isotretinoin was explained by its anti‐inflammatory effect similar
to corticosteroids [7]. Our case had a good response to systemic
corticosteroid therapy at onset. In fact, all therapy alternatives
are essentially effective in the early stages of the disease.
Therefore, Mendez suggested a surgical modality in the later stages
[11].
Table I. Data from the patients of solid facial
edema of acne treated with isotretinoin.
| Patient number |
Author\year |
Sex\age (years) |
Duration of acne
(years) |
Duration of swelling (years) |
Dose and period of isotretinoin treatment |
Additional drug |
Isotretinoin response |
| 1 |
Friedman et al.
1986 |
M\17 |
3 |
1 |
1.0 mg\kg
5 months |
‐‐ |
Partial |
| 2 |
Helander & Aho
1987 |
F\20 |
4 |
4\12 |
0.6 mg\kg
6 months |
Clofazimine +
Lymph massage |
Partial |
| 3 |
|
M\18 |
4 |
9\12 |
0.8 mg\kg
6 months |
Lymph massage |
Partial |
| 4 |
Humbert et al.
1990 |
F\15 |
¿ |
1 |
1.0 mg\kg
6 months |
‐‐ |
Nearly complete |
| 5 |
|
M\20 |
¿ |
2 |
1.0 mg\kg
8 months |
‐‐ |
Partial |
| 6 |
Jungfer et al.
1993 |
M\20 |
8 |
2 |
0.5 mg\kg
4 months |
Ketotifen |
Nearly complete |
| 7 |
Present case |
M\25 |
11 |
4 |
0.5 mg\kg
4 months |
‐‐ |
None |
.
In conclusion, based on our unresponsive case, we suggest that the
anti‐inflammatory drugs, isotretinoin or corticosteroids, are only
worth trying in the earlier stages of swelling before the
development of fibrosis and permanent edema.
References
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complication of acne vulgaris. Arch Dermatol 1985; 121:
87‐90.
2 . Mahajan PM. Solid facial edema as a complication of
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129‐30
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