Author(s) : Chun-Hong Zhang, Caiping Zhang, Jianbing Wu, Shi-Jun Shan, Quan-Zhong Liu, Zhiyi Fu, Ying Guo, Huachen Wei, Hong-Duo Chen , Department of Dermatology, The Second Hospital of Shandong University; Jinan, China, Institute of Dermatology, Chinese Academy of Medical Sciences, Peking Union Medical College, Nanjing, China, Department of Dermatology, Tianjin Medical University General Hospital, 154 Anshan Road, Tianjin 300052, China, Ackerman Academy of Dermatopathology, New York, USA, Department of Dermatology, Mount Sinai Medical Center, New York, USA, Department of Dermatology, No.1 Hospital China Medical University, Shenyang, China. |
ARTICLE
Auteur(s) : Chun-Hong Zhang1, Caiping
Zhang2, Jianbing Wu2, Shi-Jun Shan3,
Quan-Zhong Liu3, Zhiyi Fu3, Ying
Guo4, Huachen Wei5, Hong-Duo
Chen6
1Department of Dermatology, The Second Hospital
of Shandong University; Jinan, China
2Institute of Dermatology, Chinese Academy
of Medical Sciences, Peking Union Medical College, Nanjing,
China
3Department of Dermatology, Tianjin Medical
University General Hospital, 154 Anshan Road, Tianjin 300052,
China
4Ackerman Academy of Dermatopathology, New York,
USA
5Department of Dermatology, Mount Sinai Medical
Center, New York, USA
6Department of Dermatology, No.1 Hospital China
Medical University, Shenyang, China
A 19-year-old Chinese female presented with reticular brownish
papules on her neck and trunk for about 2 months. The patient
complained of palpitations, irritability and tremor of the hands
which had started 6 months previously, but the symptoms were
mild. Hyperthyroidism was diagnosed, without medication prescribed.
Several brownish reticular papules developed on her intermammary
and interscapular areas; the lesions gradually increased in number
and the neck, chest and back were subsequently involved. There was
no history of any other disease or drug intake. The family history
was unremarkable.
At presentation, the symptoms of hyperthyroidism were severe;
weight loss (body mass index 18.4; normal value
18.5 ~ 22.9) and ophthalmoptosis were observed.
A heart rate of 107 beats per minute, evagination of
eyeballs, tremor of hands and enlarged thyroid gland with enhanced
Doppler flow were observed. Skin examination revealed numerous
brownish, 1-3 mm, slightly hyperkeratotic papules, coalescing
to form a reticulated pattern and distributed widely on the neck,
chest and abdomen, especially the intermammary and scapular regions
(figure 1A). Mycology
examination of lesion scrapings showed negative results. ECG
examination exhibited sinus tachycardia. Laboratory tests including
blood, urine and stool routine, blood sugar, liver and kidney
function were all within normal limits. The thyroid function test
showed: TSH 0.03 uIU/mL (0.34 ~ 5.60),
FT3 15.16 pg/mL (2.5 ~ 3.9),
FT4 4.71 ng/dL (0.61 ~ 1.22), TPO-Ab
183 IU/mL (< 35), TR-Ab 86.82 U/L (< 14). The
2-hour thyroid iodine uptake rate was 59.5% (4~25%), the 6-hour
88.8% (8~36%) and the 24-hour 94.3% (18 ~ 54%).
A skin biopsy from abdominal papules showed hyperkeratosis,
epidermal papillomatosis and sparse superficial perivascular
lymphocytic infiltrate in the dermis (figure 1C); a
diagnosis of confluent and reticulated papillomatosis (CARP) was
made.
Propylthiouracil (PTU)was prescribed, 300 mg daily, for
hyperthyreosis and propranolol, to lower the heart rate. Neither
local nor systemic treatment was given to skin lesions because they
were asymptomatic. After 2 weeks treatment, skin lesions
cleared completely accompanied with marked improvement of the
hyperthyroidism symptoms. At the 1-month follow-up visit, some
brownish papules recurred (figure 1B) following
arbitrary discontinuation of treatment. Thyroid function tests
changed to TSH 0.28 uIU/mL, FT3 5.6 pg/mL,
FT4 1.28 ng/dL, TPO-Ab 46.2 IU/mL and TR-Ab
22.10 U/L. PTU therapy was reinitiated with 200 mg daily
for one week. The lesions cleared again. After another 4-weeks’
treatment, the hyperthyreosis disappeared and thyroid function
returned to normal. PTU was decreased to maintenance therapy for
another 6 months. No relapse of CARP was observed at 1-year
follow-up visit after PTU withdrawal.
The etiology and pathogenesis of CARP remain unclear. Proposed
causative factors include a keratinization disease, Pityrosporum,
bacterial and Dietzia infection, genetic susceptibility and
endocrinopathy [1, 2]. Familial cases and coincidence with
Greither's disease of CARP have been described; which support the
hypothesis of keratinization alterations and genetic susceptibility
[3, 4]. Hirokawa et al. [5] reported CARP with acanthosis
nigricans in an obese patient with abnormal glucose tolerance and
hyperinsulinemia. To the best of our knowledge, there were no
previous reports about CARP with hyperthyroidism, which may be
associated with pretibial myxedema, hair loss and onycholysis [6].
In our case, with improvement of the hyperthyreosis and thyroid
functions, the CARP lesions cleared completely with PTU
therapy.
Numerous agents have been used to treat CARP [1]. In our case,
with improvement of hyperthyreosis after PTU therapy, the CARP
lesions cleared completely. It is unclear whether the change in
thyroid hormone levels resulting from PTU, or PTU itself was
responsible for the effect. But it suggests that hyperthyroidism
could be an important pathogenetic factor in CARP. Further studies
are needed to elucidate the mechanism of this
phenomenon.Disclosure. Financial support: none. Conflict of
interest: none.
References
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