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Received: 15 July 2022    Revised: 6 September 2022    Accepted: 21 October 2022

DOI: 10.1111/jocd.15489

REVIEW ARTICLE

Eruptive syringomas: Summary of ninety cases and a brief


literature review

Hao Lei MM1  | Ziyang Wang MD1  | Xinyu Ma MM1 | Zhaohan Zhang MM1 |


Yiguo Feng MD, PhD2 | Yan Zheng MD, PhD1

1
Department of Dermatology, The First
Affiliated Hospital of Xi'an Jiaotong Abstract
University, Xi'an, China
Background: Eruptive syringomas is a rare variant of syringoma, which is a benign
2
Department of Dermatology, The Second
Affiliated Hospital of Xi'an Jiaotong
adenoma differentiated from the terminal ducts of the eccrine glands. Nowadays, it's
University, Xi'an, China widely valued because of obvious skin lesions, large scope of influence, and high mis-

Correspondence
diagnosis rate.
Yan Zheng, Department of Dermatology, Objectives: We aim to explore the clinical features of eruptive syringomas and the
The First Affiliated Hospital of Xi'an
Jiaotong University, Xi'an, 710061, China.
current research progress.
Email: zenyan66@126.com Materials and Methods: We firstly summarized the clinical features of 90 cases of
eruptive syringomas. Then, the chi-­square test was used to analyze the relationship
between the onset site of eruptive syringomas and age, as well as gender. Finally, we
briefly reviewed the previous literature.
Results: During 12 years, 90 cases of eruptive syringomas were diagnosed in our hos-
pital, including 28 males (31.1%) and 62 females (68.9%). The average diagnosed age
was 28.8. Patients from 20 to 40 years old is 63 (70%), which is the most. 60 (66.7%)
patients had the course for more than 1 year. Among onset sites, the neck, chest,
and abdomen were in the top three. The chi-­square test showed that there were no
significant differences in the onset sites of patients aged ≤ 20 and >20 years old (p-­
value = 0.181), as well as male and female (p-­value  = 0.363).
Conclusion: We found that more female than male was affected, and the most com-
mon onset sites were the neck, chest, and abdomen. Neither age nor gender was
significantly associated with onset site distribution. Our study provides some data
support for the research of eruptive syringomas.

KEYWORDS
eruptive syringomas, etiology, onset site, review, treatment

1  |  I NTRO D U C TI O N There are four types of syringomas: local type, eruptive type, Down
syndrome associated type, and familial type. Among them, the local
Syringoma, affecting roughly 1% people worldwide, is a benign ad- type is the most common, which is identified by symmetrical pap-
enoma differentiated from the terminal ducts of the eccrine glands. ules around the eyes. 2 However, it's often ignored by patients due
It is characterized by hemispherical or flat lesions of pale yellow or to the small scope of influence, inconspicuous skin lesions, and no
1
brownish-­yellow color, 1–­4 mm in diameter, dense and not confluent. symptoms.3

This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium,
provided the original work is properly cited.
© 2022 The Authors. Journal of Cosmetic Dermatology published by Wiley Periodicals LLC.

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LEI et al.       1129

Eruptive syringomas is a rare variant compared with the other three diagnosed in our hospital, including 28 males (31.1%) and 62 females
types, but it brings great stress to patients due to obvious skin lesions, (68.9%). The average diagnosed age was 28.8 years old, the youngest
large scope of influence, and high misdiagnosis rate.2 Eruptive syringo- was 8 years old, and the oldest was 65 years old. Patients from 20 to
mas generally occurs in adolescence or early adulthood and has also been 40 years old is 63 (70%), which is the most. 60 (66.7%) patients had
reported in children and the elderly. It manifests as a sudden, massive the disease course for more than 1 year.
appearance that affects two or more anatomical parts of the body, such
as the anterior chest, abdomen and so on.4 The pathological features
of eruptive syringomas are similar to the local type, including multiple 3.2  |  Characters of onset site
cystic lumens and cords of solid cells in the dermis, and a comma-­shaped
appearance in few ducts.5 It's also shown in (Figure 1). What's more, the Firstly, we divided the onset site into four parts, face, neck, trunk and
etiology of the disease is not clear, and the treatment effect is not satis- limbs, and then subdivided it according to the specific site. Here, we
factory.6 Therefore, we summarized the clinical features of eruptive sy- present some characteristic photographs of the patient (Figure  2).
ringomas in our hospital and conducted a literature review, focusing on Also, more clinical photographs of the patients were shown in the
the etiology and possible treatment options of the disease. supporting information (Figure S1). There were more than half of the
patients with at least two affected sites. According to Table  2, 86
patients had a rash on the trunk site, the most common site. Next, 37
2  |  M E TH O D S patients had a rash on the neck. The limbs were the least affected,
with 21 people. Among single lesions, neck, chest, and abdomen
2.1  |  Study population are the most frequent, while peripheral, lower jaw, shoulder, breast,
back, and vulva are sporadic. A summary of the sites of disease
We searched the pathology database of Our hospital for eruptive sy-
ringomas and collected clinical photos of patients. After reviewed by
TA B L E 1  Baseline information
three pathology professors, patients whose clinical and pathological
characteristics matched the criteria were included in the analysis. N (%) Patients

The photos shown in this paper fully protect the privacy of patients, Gender
and pathological photos were collected by NDP.view2 software. Male 28 (31.1%)
Female 62 (68.9%)
Age at diagnosis(years)
2.2  |  Statistics
Mean (range) 28.8 (8 ~ 65)
<20 15 (16.7%)
During the analysis, data missing values were marked as blank and
20 ~ 40 63 (70.0%)
involved in the ratio calculation. All statistical analyses were finished
>40 12 (13.3%)
by SPSS 22.0 software.
Course of disease
<1 m 1 (1.1%)

3  |  R E S U LT S 1 m ~ 1 year 4 (4.4%)
>1 year 60 (66.7%)
3.1  |  Baseline information Blank 20 (22.2%)

Note: It shows the clinical data of 90 cases of eruptive syringomas in


The results (Table 1) showed that during the 12 years from August our hospital, including gender, age, and course of disease. Blank value
2010 to March 2022, a total of 90 cases of eruptive syringomas were represents some patients with no history of disease.

F I G U R E 1  Photomicrograph of
eruptive syringomas. The magnification on
the left is 10X, and the right is 20X. Arrow
shows the characteristic comma-­shaped
appearance.
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1130      LEI et al.

(A) (B) (C)

(D) (E)

(H)

(F) (G)

F I G U R E 2  The typical onset sites of eruptive syringomas. It shows some characteristic sites of the eruptive syringomas: (A) forehead; (B)
neck; (C) neck and chest; (D) armpit; (E) chest and abdomen; (F) abdomen; (G) back; (H) chest.

onset, based on age and gender, has also been provided. We further old. The results of the chi-­square test showed that there was no sta-
analyzed the relationship between the onset site of the patients and tistically significant difference in their onset sites. Meanwhile, we
age, gender. The results showed that there were no significant dif- explored whether gender affects the onset site, and the statistical
ferences in the onset sites of the patients aged ≤ 20 and >20 years results showed no statistical difference between males and females.
old (p value = 0.181), as well as male and female (p value = 0.363) Maybe it because that almost all skin glands on the human body sur-
(Table 3). face are eccrine glands, which total about 2 million, regardless of
age and gender.9 Also, maybe our insufficient sample size masks its
inherent differences.
4  |   D I S C U S S I O N Usually, eruptive syringomas are misdiagnosed clinically be-
cause of the low specificity of the rash, the common location and
Eruptive syringomas often occurs in adolescence or early adulthood, low incidence. It is important to distinguish it from many other dis-
7
especially in female. In our study, female patients were twice more eases, including folliculitis, flat wart, cutaneous mastocytosis, lichen
than males, and the average age of patients was 28.8 years old. These planus, disseminated granuloma annulare, Fox–­
Fordyce disease,
data support the view above. Eruptive syringomas usually involve exanthematous velvet syndrome, pilaris epithelioma, Sebaceous
the anterior chest, abdomen, axillary, and periumbilical regions.8 We hyperplasia, sweat sac tumor, etc.10–­12 Thus, it is important to com-
found that the most common site of disease was the neck. Chest, bine the various histopathological features in order to make an
abdomen, trunk scattered, and upper extremity are also more com- accurate diagnosis. Despite this, the willingness of clinically newly
mon affected sites. We further divided the patients into adolescence diagnosed patients to undergo biopsy is relatively low. Therefore,
and post-­adolescence according to whether they were over 20 years researchers chose dermoscopy, a non-­invasive method, to verify the
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LEI et al.       1131

TA B L E 2  Characters of onset sites


Sites Specific sites Total ≤20 >20 Male Female

Face Forehead 9 1 8 1 8
Around the eye 14 5 9 3 11
Perioral 1 1 0 1 0
Lower jaw 1 0 1 0 1
Face scattered 5 0 5 1 4
Neck and shoulder Neck 37 13 24 10 27
Shoulder 1 1 0 0 1
Trunk Chest scattered 35 8 27 13 22
Breast 1 0 1 0 1
Armpit 9 1 8 4 5
Abdomen 21 6 15 5 16
Torso scattered 17 3 14 7 10
Back 1 0 1 1 0
Vulva 1 0 1 0 1
Limbs Upper extremity 16 1 15 5 11
Lower limbs 5 2 3 0 5

Note: The onset site is divided into four parts, face, neck, trunk, and limbs, then it's subdivided
according to the specific site. Also, onset sites of disease by sex and age is shown here.

TA B L E 3  Analysis of factors affecting the onset site papules after waxing of the pubic region, which may be related to
acute inflammatory irritation.18 A patient with a history of severe ec-
Sites ≤20 >20 Male Female
zema presented with symmetrical papules on both arms, which is the
Face 7 23 6 24
primary site of hyperkeratotic eczema.19 Multiple pathology reports
Neck and shoulder 14 24 10 28
showed abundant lymphocytic inflammatory infiltrates around its
Trunk 18 67 30 55 superficial blood vessels and eccrine ducts.17,20 What's more, one
Limbs 3 18 5 16 patient treated with oral tranilast (300 mg/day) was observed im-
Chi-­square test p = 0.181 p = 0.363 provement both objectively and subjectively after 1 month of treat-
Note: We used the Chi-­square test to analyze whether the disease site ment. Six of the eight cases of syringomas treated with tranilast
was affected by age and gender. p < 0.05 was considered statistically showed improvement. Tranilast may inhibit the proliferation of stro-
significant. mal connective tissue in syringomas by inhibiting the release of IL-­1b
from the eccrine ducts. 21 It provides strong evidence for the inflam-
characteristic changes. Several studies have indicated that skin le- matory response theory. Third, autoimmune disorders also appear
sions appear dermoscopically as brown regular pigmented networks to be associated with eruptive syringomas. Two patients developed
and tiny white spaces between adjacent papules.8,13,14 Glossy white disease following liver transplantation and kidney transplantation,
structures with punctate and linear vessels on a pale pink back- respectively.11,22 Some patients had concomitant autoimmune dis-
ground are seen in the lesions of the vulvar area. In contrast, lichen eases such as alopecia areata and vitiligo. 23 It is noteworthy that
planus, folliculitis, and flat warts can be distinguished from them by patients developed vitiligo first and followed by syringomas in both
their characteristic dermoscopic findings.15 The specificity of this breasts. 23,24 Some researchers performed immunohistochemically
assay remains to be explored. staining on syringomas tissue and found that the autoimmune in-
The etiology of eruptive syringomas is still unclear, and the flammatory infiltrate around the sweat ducts consisted of an equal
current research mainly focuses on the following aspects: endo- mixture of CD4+ and CD8+ T cells, suggesting that reactive hy-
crine factors, inflammatory response around sweat duct, autoim- perplasia of cutaneous eccrine ducts is secondary to autoimmune
mune disorder, and concomitant with other diseases. Firstly, most destruction of their surface components. 25 Finally, some hereditary
eruptive syringomas occur in adolescent or post-­pubertal female. diseases or systemic disease can be combined with eruptive syringo-
The rash also increases in females during pregnancy and before mas, such as systemic disease hyperthyroidism, diabetes, Marfan's
menstruation.16 When immunohisto-­chemistry was performed on syndrome, Ehlers–­Danlos syndrome.4,26 However, it's relatively rare.
patient tissue, overexpression of receptors for estrogen and pro- Current treatments for syringomas include topical and sys-
gesterone receptors was detected,17 reflecting the important role temic medications, chemical peels, CO2 lasers, electrocautery, and
of hormones. Secondly, several studies have linked inflammation to surgery, all of which have shown some relief. 6,27–­31 However, in
eruption of syringomas. A man developed multiple asymptomatic the treatment of eruptive syringomas, the above physical therapy
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1132      LEI et al.

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