ARTICLE
Tufted hair folliculitis (THF) is a disease causing alopecia which is
characterized clinically by the presence of a bundle of hair shafts emerging
from single, dilated, follicular openings. It is a rare condition with
only 32 reported cases, mostly from Europe and the United States [1-16].
Various treatments tried in the past have generally been unsuccessful.
We report two characteristic cases of THF, which are the first cases from
Japan to be reported in the international literature and we discuss a
new effective treatment for this condition.
Case reports
Case 1
A 42-year old woman presented with a 20-year history of patchy hair
loss on the scalp. She had been treated with topical steroid ointment
mixed with antibiotics for 10 years. Physical examination showed a well-defined
area of scarring alopecia on the parietal region. Within these areas were
tufts of 7-10 normal-looking hairs arising out of dilated hair follicles
(Fig. 1). A section of
the affected area felt slightly edematous, and pus could be expressed
from some follicular orifices. A sample of the pus was cultured and showed
positive for staphylococcus aureus. Laboratory tests that were
found to be normal or negative included a full blood count and liver and
kidney function tests. We treated this patient with topical nadifloxacin
ointment (NFO), a quinolone carboxylic acid antibacterial agent [10].
This ointment was applied 2 times per day. Although scarring alopecia
persisted, this treatment stopped the expansion of alopecia.
Case 2
A 46-year old woman presented with a three-year history of erythematous
pruritic plaque on the parietal region of the scalp. Physical examination
revealed multiple bundles of hairs emerging from a single dilated follicular
opening.
Histological examination of the tufted areas
of the scalp showed scarring in the papillary to mid-dermis with perifollicular
acute and chronic inflammation. There was a superficial perifollicular
mixed inflammatory infiltrate composed of neutrophilis, lymphocytes and
numerous plasma cells. The foci of fibrosis in the upper dermis and around
grouped follicles were inconsistent findings. Multiple hair shafts, emerging
from a single follicular opening, were observed. The structure of each
hair papilla was normal, and none of the papilla appeared to fuse or subdivide
to form multiple papilla (Fig.
2).
Several fungal cultures from the affected area were negative. However
staphylococcus aureus was found to be present in the bacterial
cultures. Laboratory tests, including full blood count, liver and kidney
function tests, were within normal limits. The patient had been initially
treated with topical steroid ointment mixed with antibiotics. This treatment
produced some improvement with resolution of pruritus, however scarring
alopecia persisted with gradual expansion. We subsequently treated this
patient with NFO which stopped the expansion of alopecia.
Discussion
Smith first described this disorder in 1978 as "Tufted hair folliculitis
of the scalp" [1]. Thereafter, we have been able to find only 32 reported
cases [1-16] including the 2 cases reported here. Twenty-nine of these
cases were reported from the United States and Europe [1-6, 8-16]. Three
cases, including our two, were found in Japan [7]. All 32 patients were
between the ages of 20 and 60 years old (10 patients in the twenties,
8 in the thirties, 6 in the forties, 5 in the fifties, and 3 in the sixties).
There was a male preponderance with 20 out of these 32 patients being
male. The sites of predilection were the occipital area (15 cases) and
the parietal area (10 cases). Frequently reported symptoms were pruritus
(11 cases) and pain (4 cases). Except for two cases, staphylococcus
aureus infection was observed in all patients [9]. Good results
have been reported after surgical excision of the involved areas [16].
However all reported topical treatments have been unsuccessful. Treatment
with oral antibiotics have resulted in temporary cure in a few reported
cases, however, long-term use of systemic antibiotics is best avoided
to prevent possible hepatic or renal side effects. In our 2 patients,
treatment with topical NFO resulted in the disappearance of pruritus and
pustules, and expansion of the alopecia is not observed after 1 year.
Nadifloxacin is a new quinolone antibiotic with a broad-spectrum antibacterial
effect and little resistance. The topical preparation is much preferred
for long-term use over systemic drugs. We believe that nadifloxacin ointment
is an effective treament in patients for the prevention of the spread
of lesions or recurrence.
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