• Am J Med Surg October 2023; 13 (3) 1-6

Atypical eyelid hidradenocarcinoma. A case report

Nayeli Tatiana Medina España M.D.
Daniel Atl López Fábila M.D. Background
Cutaneous hidradenocarcinoma appears as a de novo head and neck
cutaneous neoplasia from sweet glands. It represents 6 % of all cutaneous
eccrine tumors and 0.01 % of all the malignant cutaneous tumors. The risk
factors are age (≥65 years), ultraviolet radiation, tumor size ≥ 2cm and
smoking. It arises as a cutaneous nodular hyperpigmented lesion with
ulceration, propensity to dissemination and poor survival. A 78-years-old
female arrived with a 0.5 cm diameter ulcerated fibrotic nodular lesion in the
medial cantal right eyelid of 1 year of evolution. A failed excisional biopsy
was performed reporting a moderate non-differentiated hidradenocarcinoma
(0.5x0.7cm) with positive margins. Clinical diagnosis was made with
negative imaging to dissemination in an IA stage eyelid tumor (T1bN0M0-
G2). A new resection was done with a peripheral 2-3 cm widening in the
medial canthal, upper and inferior right eyelid. The right half of the eyelid
was reconstructed with compound ipsilateral glabellar and a cervicofacial
(Mustarde) flaps with a favorable evolution after 2 years of surveillance.
Hidradenocarcinoma is a rare eccrine neoplasia. Radiotherapy and
chemotherapy are applied in severely selected cases. Contrary to literature in
initial eyelid stages tumors, hidradenocarcinoma can be managed with
surgical treatment and clinical surveillance only. The cutaneous
reconstruction with multiple flaps must be considered in complex eyelid
defects. The glabellar flap is a useful tool combined with another flaps
(nasolabial rotation, rhombic/Limberg, cervicofacial/Mustarde),
Jalisco, Mexico
nasal/Rintala) in complex defects of the eyelids. The description and
Case Report management of this rare neoplasia is paramount to clarify an adequate
consensus in dermatology.
Dermatology
Keywords: Eyelid cutaneous neoplasia, adnexal carcinoma,
hidradenocarcinoma, primary eccrine adenocarcinoma, eyelid reconstruction.

C
utaneous hidradenocarcinoma is also known as
primary eccrine adenocarcinoma, clear cells
hidradenocarcinoma, malignant acrospiroma
and hidroadenoma of malignant clear cells, among
many other names. It is an extremely rare neoplasia
with de novo presentation originated in intradermal
duct of eccrine sweet glands. (1) Adnexal carcinomas
are rare malignant cutaneous tumors representing less
than 1 % of all the malignant cutaneous tumors. They
derived according to their histogenesis from eccrine or
apocrine sweet glands, follicular structures and
pilosebaceous units. (Figure 1) From these ones the
tumors derived from sweet glands represent 62%-82%
of all the adnexal carcinomas, being the most frequent
the porocarcinoma. (2) Cutaneous hidradenocarcinoma
represents 6 % of all cutaneous eccrine tumors, 0.01 %
of all the cutaneous malignant tumors and 0.001 % of
all the malignant tumors. (3,4) Any eyelid cancer has a
prevalence of 145.1 per 100 000 population. By the
type of cancer, basal cell carcinoma is the most
frequently found in 60 % of all cases. The eccrine or
sebaceous tumors are found in 3 % of all the eyelid
cutaneous tumors. (5)
The associated risk factors to develop an
eyelid cancer from adnexal malignant tumors are older
age (≥ 65 years), ultraviolet radiation, male, (male:
female ratio 1.4:1), I and II skin phototypes, tumoral
size (≥ 2 cm), radiation, immunosuppression, Muir-
Torre syndrome, and smoking. (5) The etiopathogenesis
is unknown. Occasionally tumors have arisen within a
pre-existing benign clear cell hidradenoma. (6)
Hidradenocarcinoma presents more frequently in the
head and neck (especially face) and rarely in
extremities or trunk. It appears as a solitary
subcutaneous firm nodule or erythematous plaque with
telangiectasia and/or ulceration, pruritus, and slow
circumferential expansion. It may appear fleshy red,
gray, or violet with normal overlying skin. (7) In the
hidradenocarcinoma the recurrence rate is described in
10 %-30 %, with a survival rate of 30 % in 5 years. (8)
The standard treatment is surgical resection with free
margins considering functional and aesthetic concerns
in the location of the residual defect from resection. In

From the Plastic Surgery Department at Jalisco Institute of Cancerology (IJC). Jalisco, México. Received on August 30, 2023. Accepted on September 5, 2023.
Published on September 6, 2023.

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Medina España NT et al. • Am J Med Surg October 2023; 13 (3). 1-6

Figure 1. Origin of adnexal tumors. Skin specimen section. The arrows point to the tissues and structures that can be the origin of the
appendageal tumors. The cancers described are listed in brackets.

eyelid there are multiple choices of reconstruction
with cutaneous grafts, compound grafts (cartilage,
mucosa, periosteal flaps, fat) and compound flaps
depending on the anatomical structures involved. The
aim of this study is to describe an unusual pathology
due to a lack of consensus in the approach and
treatment of this rare cutaneous disease.
Case report
A 78-years-old female arrived with a clinical
presentation with a nodular lesion of 0.5 X 0.5 cm
diameter with telangiectasia and progressive
enlargement with ulceration in the medial canthal right
eyelid of 1 year of evolution. She mentioned pruritus
and occasional bleeding of the tumor. In her medical
background she had an excisional biopsy of a non-
specified tumor in her right forehead with covering of
the defect with a skin graft without actual recurrence.
She mentioned exposure to sunlight and no other
associated risk factors. She denied any other medical
background of importance for current pathology. A
failed excisional biopsy was performed in an outside
hospital environment which reported a histological

Figure 2. A. A right internal eyelid 0.5 cm diameter violaceous ulcerated nodular lesion is seen in the right eye. B. Erythematosus
fibrotic plaque in medial cantal right eyelid after a failed excisional biopsy with residual positive tumoral margins. Notice the skin
photodamage and the marking of the new tentative ampliation of margins resection.
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Medina España NT et al. • Am J Med Surg October 2023; 13 (3). 1-6

undifferentiated moderate hidradenocarcinoma (0.5x

Figure 3. Second excisional biopsy description which reports an
infiltrative and diffuse growth pattern in dermis with prevalence of
neoplastic clear cells with atypia and marked nuclear
pleomorphism with clear and pale eosinophilic cytoplasm with
polygonal polyhedric cells arranged in anastomosing cords and
chromatin in poorly distributed lumps suggestive of moderate
undifferentiated hidradenocarcinoma (G2) of 0.4 x 0.3 x 0.2 cm
dimensions with negative margins (respecting posterior eyelid
lamella) and low mitotic rate (≤1 mitoses/mm) without
lymphovascular and perineural invasion.
0.7x 0.7 cm) (G2 / Moderately differentiated) with
positive margins to profound and inferior tissues. On
clinical examination after 3 months of the initial
biopsy we observed a nodular scar with telangiectasia
in the medial canthal right eyelid accompanied with
skin photodamage. (Figure 2)
The management of eyelid malignancies is
dependent on histopathologic type and disease stage
according to the American Joint Committee on Cancer
(AJCC) staging system. A regional ultrasound and
computed axial tomography screenings were
performed to detect regional or distant oncologic
dissemination which were negative. A clinical staging
was performed scoring a clinical stage IA (T1b (tumor
≤10 mm that invades the eyelid margin) N0M0 (No
evidence of lymph node involvement and distant
metastasis). (9) A new surgical approach was decided
with peripheral 2 to 3 cm margins depending on the
functional eyelid margins. (10) The anterior right eyelid
lamella in the medial canthus, superior and inferior
sites were resected and a transoperative excisional
biopsy was performed to rule out posterior eyelid
lamella involvement and positive margins from

Figure 4. A. Markings with an approximately 3 cm resection margin respecting the eye limited in the medial right eyelid canthus.
B. Ampliation of resection margin and marking of compound eyelid reconstruction with cheek (Mustarde) and glabellar flaps. C.
Immediate postoperative result with drain placement in the lower region of the right eye. D. Late postoperative result after a 2
years follow up without clinical recurrence and adequate functional and aesthetic result.
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Medina España NT et al.
Figure 5. Summary of some regional multiple flap choices in
complex eyelid reconstruction. A. Temporal forehead flap
(Fricke) for upper eyelid. B. Semicircular myocutaneous flap
(Tenzel) for inferior eyelid. C. Cervicofacial flap (Mustarde) for
inferior eyelid. D. Dorso nasal flap (Rintala) for upper and medial
canthal eyelid. E. Glabellar nasal flap for upper, medial canthal
and medial inferior eyelid. F. Nasolabial flap for medial canthal
and inferior eyelid. G. Frontonasal flap for upper, medial canthal,
lateral canthal and inferior eyelid. H. Upper eyelid flap (Tripier)
for medial canthus and inferior eyelid. I. Rhomboidal flap
(Limberg) for medial canthal, lateral canthal and inferior eyelid.
histopathology. The result of the second excisional
biopsy revealed the same histological diagnosis with
an undifferentiated moderate hidradenocarcinoma
(0.4x0.3x0.2cm) without lymphovascular and
perineural invasion with negative margins. (Figure 3)
The markings of an ipsilateral compound flaps for the
right eyelid reconstruction (medial canthus, superior
and inferior eyelid) were marked and performed with a
glabellar and a cervicofacial (Mustarde) flaps due to
the complexity of the defect. A drain was set in the
inferior eyelid at the cervicofacial flap, and the stitches
were removed with the drain after 7 days of
postoperative recovery. A 2-year follow-up was done
with monthly revisions without clinical recurrences
and complications. (Figure 4)
Discussion
Hidradenocarcinoma is an extremely rare
cutaneous eccrine neoplasia. There is a lack of
adequate consensus to lead a therapeutic approach,
because of the limited and ambiguous literature. In the
eyelid the differential diagnosis of
hidradenocarcinoma must be distinguished from
tumors showing conspicuous cytoplasmic vacuolations
such as clear cell squamous carcinoma, clear cell
melanoma, trichilemmal carcinoma, and metastatic
clear cell carcinoma from kidney, bronchus, and
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• Am J Med Surg October 2023; 13 (3). 1-6
liver.(10) However, the clinical and pathologic staging
TNM is paramount in eyelid carcinoma regardless of
the subtype to direct the treatment approach. (11) Eyelid
malignancies require different considerations from
other cutaneous malignancies of the same cell type due
to the unique anatomic considerations in the periocular
region and the functional impact of surgical resection
and reconstruction on ocular protection and visual
function. Because of this Mohs micrographic surgery
can have an important role in surgical treatment. Mohs
micrographic surgery has been advocated for eyelid
malignancies to ensure maximal preservation of
normal tissue and to obtain margins free of disease.
The standard treatment is surgical excision with
negative margins. In patients at high risk for local
recurrence, adjuvant radiotherapy may be needed in
selected cases of severity. In patients at high risk for
lymph node metastasis, sentinel lymph node (SLN)
biopsy may be considered. There is no clear benefit
from chemotherapy in early stages. In some
instances, topical chemotherapy may be considered.
Therapy needs to be tailored in each case.
A wide local excision or complete surgical
excision and block dissection of involved nodes
followed by radiotherapy and chemotherapy must be
considered if there is distant metastasis. Nash et al.
were the first to demonstrate Her-2/neu amplification
in a malignant skin adnexal tumor and suggested
application of trastuzumab for patients with metastatic
adnexal carcinomas. (12) There are some risk factors for
local recurrence eyelid carcinoma and metastasis after
wide excision. Takahashi et al. described some of
them which are simultaneous involvement of both
upper and lower eyelid, multicentric origin, diffuse
pattern, stage T3a, large tumor size ≥ 2 cm, poor
differentiation and Pagetoid spread (presence of
atypical cells at different levels of the skin
epithelium). (13) These risk factors can predict the
necessity of adjuvant and more aggressive treatment. It
is worth noting that in our case hidradenocarcinoma
affects only the medial canthal eyelid and the only
predicting factors were age (78 years) and diffuse
pattern. So, we classified our case as a low-risk eyelid
carcinoma because of its early detection and
management. This was our criteria to treat the
hidradenocarcinoma with wide resection and
surveillance only. In our follow-up contrary at some
literature stands we agree with Gao et al. who states
that the tumor size (≥ 2 cm) is inversely proportional
to patient´s overall survival time affecting the overall
(14)
survival and cancer specific survival. That is how
we explain no relapses in our clinical approach in this
case.
In the eyelid the cutaneous
hidradenocarcinoma of low risk detected in early
stages, can be managed with wide surgical resection or
Mohs surgery depending on the functional periorbital
DOI 10.5281/zenodo.8320824
Medina España NT et al. • Am J Med Surg October 2023; 13 (3). 1-6
tumor eyelid involvement with surveillance with good Conflicts of interests
prognosis. The reconstruction of the eyelid depends on
the depth of affection, the cutaneous anterior lamella, There was no conflict of interest during the
posterior lamella, or both that can affect the study declared by the authors, and it was not funded
reconstruction strategy. In our case the residual defect by any organization.
from the resection was cutaneous (anterior lamella) in
the medial canthal, superior and inferior right eyelid. References
This is the reason why in complex multiple eyelid
cutaneous defects the knowledge of multiple-choice 1.
regional flaps facilitates an adequate planning and
surgical approach for the surgical dermatologist. Some
of the reconstructive options (random or pedicled 2.
periorbital regional flaps) for the eyelid reconstruction
are the glabellar flap, cervicofacial flap (Mustarde), 3.
temporal frontal flap (Fricke) and upper eyelid flap
(Tenzel) among others. (Figure 5) The medial canthal
is a region where multiple sites such as the upper 4.
eyelid, the lower eyelid, the eyebrows, and the nose
5.
converge, and the colour, texture, and thickness of the
skin are different for each site. The skin of the medial
canthal region is slightly thicker than the skin of the
upper and lower eyelids and thinner than the skin of
6.
the nose and the glabella. Taking this into account in
our experience as it has been performed in this case we
agree with Ogino et al. that our preferred selected flaps
are the myocutaneous glabellar flap, upper eyelid flap 7.
(Tripier) and nasal flap (Rintala) for canthal eyelid
(15)
reconstruction. We also recommended the use of
more than one flap in complex eyelid defects like our
case in which we used a glabellar flap with a 8.
cervicofacial flap (Mustarde) without recurrences at 2
years, and excellent functional and aesthetic results.
We also incentivize the use of Mohs surgery in eyelid 9.
(16)
carcinoma for the surgical dermatologist.
Conclusion 10.
In the cutaneous eyelid hidradenocarcinoma
an improving health awareness, initial histological 11.
examination and timely surgery are key points to
improving the prognosis. The detection of
hidradenocarcinoma in early stages (tumor size ≤ 2 cm 12.
without locoregional invasion) can be treated with
wide resection or Mohs surgery resection. An
understanding of eyelid anatomy is the first step
toward the best surgical outcome, and there are
various methods for reconstructing eyelid defects. 13.
Defect size, location and analysis of the anterior
lamella, posterior lamella, and the canthal regions help
to create an organized operative plan. Also, the
presence of simultaneous multiples defects forces the
surgical dermatologist to use simultaneous compounds 14.
flaps to give adequate skin coverage in wide eyelid
defects. This rare pathology must be continue studied
to clarify the pathway to perform a more adequate 15.
consensus in its treatment.
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Soni, N. Bansal, V. Kaushal, A.K. Chauhan. Current
management approach to hidradenocarcinoma: a
comprehensive review of the literature. E cancer medical
science. 2015 Mar 19; 9:517.
Seger EW, Neill BC, Tolkachjov SN. Adnexal and
Sebaceous Carcinomas. Dermatol Clin 2023
Jan;41(1):117-132.
Mehregan AH, Hashimoto K and Rahbari H (1983)
Eccrine adenocarcinoma: a clinicopathologic study of 35
cases. Arch Dermatol 1983; 119(2) 104–14
Cooper PH. Carcinoma of sweat glands. Pathol Annu.
1987;22(pt 1):83–124.
Bas Z, Sharpe J, Yaghy A, Zhang Q et al Prevalence of
and Associated Factors for Eyelid Cancer in the
American Academy of Ophthalmology Intelligent
Research in Sight Registry Ophthalmol Sci. 2022 Sep
27;3(1):100227.
Brenn Thomas, Phillip McKee H (2005) Tumors of the
sweat glands. In: McKee Phillip H, Calonje Eduardo,
Granter Scott R (eds) Pathology of the skin with clinical
correlations, 3rd edn. Elsevier, Mosby, p 388.
Yavel R et al (2009) Hidradenoma and
hidradenocarcinoma of the scalp. Managed using Mohs
micrographic surgery and a multidisciplinary surgery
and a multidisciplinary approach: case reports and
review of the literature Dermatol Surg 35(2) 273–81.
Tolkachjov SN, Hocker TL, Hochwalt PC, et al. Mohs
micrographic surgery for the treatment of
hidradenocarcinoma: the mayo clinic experience from
1993 to 2013. Dermatologic Surgery 2015;41:226-231.
Esmaeli B. (2018) Eyelid Carcinoma. En Amin MB.
American Joint of Comittee Cancer 8th Ed., Ch. 64 (787-
794) Springer.
Singh G, Narasimha A, Kumar H, Datti N. Clear cell
hidradenocarcinoma of the eyelid: a case report with
review of the literature. Int Ophthalmol. 2013
Apr;33(2):171-5.
Guillot B (2009) From unusual cutaneous malignancies:
cutaneous adnexal tumors in Management of rare adult
tumors ed Belkacemi Y, Mirimanoff R and Ozsahin M
(Paris: Springer- Verlag France) 471–7.
Nash JW, Barrett TL, Kies M, Ross MI, Sneige N,
Diwan AH, Lazar AJ (2007) Metastatic
hidradenocarcinoma with demonstration of Her-2/neu
gene amplification by fluorescence in situ hybridization:
potential treatment implications. J Cutan Pathol 34:49–
54
Takahashi Y, Takahashi E, Nakakura S, Kitaguchi Y,
Mupas-Uy J, Kakizaki H. Risk Factors for Local
Recurrence or Metastasis of Eyelid Sebaceous
Gland Carcinoma After Wide Excision with Paraffin
Section Control. Am J Ophthalmol. 2016 Nov;171:67-
74.
Gao T, Pan S, Li M, Su
R. Prognostic analysis of hidradenocarcinoma: a SEER-
based observational study. Ann Med. 2022
Dec;54(1):454-463.
Ogino A, Onishi K, Okada E, Nakamichi M. Medial
canthal reconstruction with multiple local flaps. JPRAS
Open. 2017 Sep 29;15:4-9.
DOI 10.5281/zenodo.8320824
Medina España NT et al. • Am J Med Surg October 2023; 13 (3). 1-6

16. Blumenthal SR, Swick M, Bayan CA, Ramanathan D,

Maher I. Complex Eyelid Reconstruction: A Practical
Guide for the Mohs Surgeon. Dermatol Surg. 2022 Sep
1;48(9):916-923.

Nayeli Tatiana Medina España

Internal Medicine Service
Hospital “Dr. Valentin Gomez Farias”
Institute of Security and Social Services for State Workers
(ISSSTE)
Jalisco, México.

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