Eccrine angiomatous hamartoma with verrucous hemangioma-like features – an unusual combination

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Eccrine angiomatous hamartoma with verrucous
hemangioma-like features – an unusual combination
Observaon Leer
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How to cite this article: Liu L, Zhou L, Zhao Q, Wei D, Jiang X. Eccrine angiomatous hamartoma with verrucous hemangioma-like features –
an unusual combination. Indian J Dermatol Venereol Leprol doi:10.25259/IJDVL_195_2021
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Sir,
A 25-year-old male presented with a history of asymptomatic
reddish-purple patches on the dorsal right hand since birth.
On examination, there were several clustered, erythematous-
violaceous and hyperkeratotic plaques on the dorsal right
hand [Figure 1a]. Dermoscopy revealed a prominent blue-
white background, with hyperkeratosis in the center of
the lesions, surrounded by purple-brown round lacunae
indicative of underlying dilated vessels [Figure 1b]. Doppler
ultrasonography of the lesions detected dotted blood ow
signals in the dermis and subcutis. Magnetic resonance
imaging demonstrated mixed long T1 and T2 signals of lesions
involving dermal and subcutaneous tissue without involving
tendons and muscles, which was suggestive of a hemangioma.
Skin biopsy showed hyperkeratosis, acanthosis and
papillomatosis in the epidermis. The papillary dermis had
numerous thin-walled, ectatic and irregular vessels [Figures 2a
and 2b]. In the reticular dermis and subcutis, proliferation of
eccrine glands associated with thin walled vessels was observed
[Figure 2c]. On immunohistochemistry, the vascular endothelial
cells were positive for CD31 [Figure 2d] and weakly positive
for GLUT-1 [Figure 2e]. Positive attaining for αSMA was seen
in the yoepithelial layer of secretory coils and vascular smooth
muscle cells [Figure 2f]. We speculated that the diagnosis
could be eccrine angiomatous hamartoma with verrucous
hemangioma-like features. Intralesional Nd: YAG laser (energy
130 mJ, frequency 60 Hz) was utilized to treat the lesions which
improved satisfactorily after two treatments [Figure 3].
In a study of 26 cases of eccrine angiomatous hamartoma,
seven demonstrated mild hyperkeratosis in the epidermis;
another study involving 15 cases reported that one patient
had verrucous changes in the epidermis and abundant
capillaries in the papillary dermis.1,2 Wang et al. analyzed
74 cases of verrucous hemangioma and found hyperplasia
of the eccrine glands around abnormal vessels in four cases.
They opined that these were verrucous hemangiomas with
features of eccrine angiomatous hamartoma.3 However, a
literature search could nd only two reported cases with the
Received: February, 2021 Accepted: May, 2021 EPub Ahead of Print: August, 2021 Published: ***
DOI: 10.25259/IJDVL_195_2021 PMID: ***
Figure 1a: Multiple reddish-purple hyperkeratotic plaques on the dorsal
right hand
Figure 1b: Dermoscopic examination: blue-white background, hyperkeratosis
in the middle of the lesion, surrounded by purple-brown round lacunae

Observation Letter
Indian Journal of Dermatology, Venereology and Leprology | Volume XX | Issue XX | Month 20212
Figure 3: Apparent improvement of the plaques (blue arrow) after treatment
with Nd:YAG laser
Figure 2: Histopathology examinations of lesions from dorsal aspect of the right hand. (a) Low-power view of biopsy (hematoxylin–eosin[HE],original
magnication ×10). (b) High-power view showing hyperkeratosis, acanthosis and papillomatosis in epidermis,thin-walled, ectatic, vessels in papillary dermis
(×100). (c) Proliferation of eccrine glands associated with thin-walled vessels in reticular dermis (×100). (d) Immunohistochemical staining showing that the
vascular endothelial cells (green arrow) were positive for CD31 and eccrine glands (black arrow) were negative (×400). (e) Vessels were weakly positive for
GLUT-1 (×400). (f) Myoepithelial layer of secretory coils (black arrow) and vessels (green arrow) were positive for αSMA (×400)
ab c
f
e
d
diagnosis of eccrine angiomatous hamartoma with verrucous
hemangioma-like features . In Table 1 we have summarized
the characteristics of eccrine angiomatous hamartoma
and verrucous hemangioma so as to highlight their salient
dierences.].
Both eccrine angiomatous hamartoma and verrucous
hemangioma present mostly at birth or childhood with
lesions principally localized to the extremities. Eccrine
angiomatous hamartoma is mostly isolated papules, while
verrucous hemangioma is characterized as multiple and
clustered plaques or nodules. The dermoscopic features of
eccrine angiomatous hamartoma are spitzoid or popcorn
patterns, whereas those of verrucous hemangioma are
reddish-blue or bluish lacunae or dermis with a bluish-
white hue. Clinically and dermoscopically the lesions
seen in our patient were more suggestive of verrucous
hemangioma. The symptoms of eccrine angiomatous
hamartoma include pain and hyperhidrosis, whereas those
of verrucous hemangioma are itch, oozing and bleeding.
However, our patient did not exhibit typical symptoms of
either entity.
Patterson et al. developed the histopathological criteria for
eccrine angiomatous hamartoma including: (a) hyperplasia
of normal or dilated eccrine glands; (b) intermixing of
eccrine glands with abundant capillaries; (c) variable
presence of apocrine, lymphatic or mucinous structures.4
Histology of verrucous hemangioma demonstrates
hyperkeratosis, papillomatosis and acanthosis in the
epidermis with numerous dilated vessels extending into
the subcutaneous tissues. Our case showed histological
characteristics of both eccrine angiomatous hamartoma and
verrucous hemangioma.
GLUT-1 and WT-1 are markers that dierentiate vascular
neoplasms from vascular malformations. Trindade et al.
reported that verrucous hemangioma was positive for
GLUT-1 in 13 cases (100%), suggesting that verrucous
hemangioma may be categorized as a form of vascular

Observation Letter
3Indian Journal of Dermatology, Venereology and Leprology | Volume XX | Issue XX | Month 2021
Table 1: Comparison between eccrine angiomatous
hamartoma and verrucous hemangioma
Features Eccrine angiomatous
hamartoma
Verrucous hemangioma
Onset Congenital or later in
childhood
Congenital or in early
infancy
Location Distal extremities Distal extremities
Distribution Mostly solitary papules Grouped of plaques or
nodules
Symptom Pain, hyperhidrosis Itch, oozing, bleeding
Dermoscopy A spitzoid pattern or a
popcorn pattern
Bluish-white hue
(hyperkeratosis), reddish-blue
or bluish lacunae
Histopathology Eccrine sweat glands
associated with thin-walled,
aggregated vessels in the
middle and lower dermis
Hyperkeratosis, acanthosis and
papillomatosis in epidermis,
vascular component in dermis
and subcutaneous tissue
GLUT-1
expression
Negative Mostly positive
Therapy Surgery, laser, botulinum
toxin
Surgery, laser, topical steroid
with salicylic acid ointment
tumor.5 However, Wang et al. found that vessels in verrucous
hemangioma were positive for GLUT-1 in 49 cases (66%),
focally positive for Prox1 in 69 (93%) cases, while
negative for WT-1 in 60 cases (81%).3 They proposed that
verrucous hemangioma is a vascular malformation and
an incomplete lymphatic immunophenotype. The reason
why for GLUT-1 is positive in verrucous hemangioma is
not clear, but the staining is less intense than in infantile
hemangiomas.
In conclusion, the mixed histological features comprising
both eccrine gland proliferation as well as vascular
proliferation and weak GLUT-1 positivity support the
diagnosis of eccrine angiomatous hamartoma with
verrucous hemangioma-like features in our case. To the best
of our knowledge, this is the rst reported case of eccrine
angiomatous hamartoma with verrucous hemangioma-like
features, to be characterized in such great detail using a
combination of histopathology, immunohistochemistry and
dermoscopy.
Declaration of patient consent
The authors certify that they have obtained all appropriate
patient consent.
Financial support and sponsorship
National Natural Science Foundation of China (Grant
No. 81872535 and 82003373).
Conicts of interest
There are no conicts of interest.
Lian Liu*, Li Zhou1*, Qian Zhao, Danfeng Wei,
Xian Jiang
Departments of Dermatology, 1Core Facility, West China Hospital,
Sichuan University, Sichuan, China
*Lian Liu and Li Zhou are Co-rst authors
Corresponding author:
Prof. Xian Jiang,
Department of Dermatology, West China Hospital,
Sichuan University, Sichuan, China.
jennyxianj@163.com
References
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hamartoma: A clinicopathological study of 26 cases. Dermatology
2015;231:63-9.
2. Lin YT, Chen CM, Yang CH, Chuang YH. Eccrine angiomatous hamartoma:
A retrospective study of 15 cases. Chang Gung Med J 2012;35:167-77.
3. Wang L, Gao T, Wang G. Verrucous hemangioma: A clinicopathological and
immunohistochemical analysis of 74 cases. J Cutan Pathol 2014;41:823-30.
4. Patterson AT, Kumar MG, Bayliss SJ, Witman PM, Dehner LP,
Gru AA. Eccrine angiomatous hamartoma: A clinicopathologic review
of 18 cases. Am J Dermatopathol 2016;38:413-7.
5. Trindade F, Torrelo A, Requena L, Tellechea O, Del Pozo J, Sacristán F,
et al. An immunohistochemical study of verrucous hemangiomas.
J Cutan Pathol 2013;40:472-6.