Nevus Hori Treated with Laser Combination: A Case Report

Chesia Christiani Liuwan, M. Yulianto Listiawan

Department of Dermatology and Venerology, School of Medicine, Universitas Airlangga, Dr. Soetomo General Hospital,

Surabaya, Indonesia

Keywords: Nevus Hori, Facial Dermal Melanocytosis, CO

Fractional Laser, 1064 nm Nd:YAG laser

Abstract: Introduction: Dermal melanocytosis include the mongolian spot, blue nevus, nevus of Ota, nevus of Ito, and

nevus hori. Nevus hori is also known as acquired bilateral nevus of Ota-like macules (ABNOM). Nevus

Hori is characterized by its bilateral hyperpigmentation on the forehead, eyelids, cheeks, and/or nose and

appears at third decade of life. Objective: To evaluate the clinical manifestation and management of Nevus

Hori. Case: A 16-year-old Javanese female patient, complaint about dark patches in left her cheek since 2

years ago. Histopathology has not been done because patient refused to do the biopsy. Diagnosis of Nevus

Hori is made based on anamnesis and clinical manifestations. The patient is treated with combination CO

fractional laser

and 1064 nm Nd:YAG laser for 3 sessions and shows improvement. Conclusion: Among

facial dermal melanocytosis (FDM), nevus of Ota and Nevus Hori are clinically similar and both diseases

cause aesthetic problems as they develop on the face and are not self-limited. This laser combination aimed

to increase the ability to remove pigment. Nevus Hori is diagnosed based on anamnesis and clinical finding.

Laser therapy is the therapy of choice because of excellent results but cost and availability are the limiting

factors.

1 INTRODUCTION

Disorders of melanin pigmentation can be divided

on morphological grounds into two types. The first

is hypermelanosis, where there is an increased

amount of melanin in the skin. The second type is

hypomelanosis, where there is a lack of pigment in

the skin. Furthermore, hypermelanosis can be

divided on histological grounds into epidermal

hypermelanosis, dermal hypermelanosis, and mixed

epidermal and dermal hypermelanosis (Lee et al.,

2004). The dermal hypermelanois due to the

presence of melanin–producing dendritic

melanocytes that lie in the dermis is named dermal

melanocytosis, which includes nevus of Ota, nevus

of Ito, Mongolian spots (Watanabe, 2014).

Dermal melanocytosis is usually localized,

especially common among Asians. Clinically, it

shares bluish–grey coloration. When a sufficient

number of melanin–containing cells are present in

the dermis, various clinical forms are reported,

depending on their onset and distribution (Lee et al.,

2004; Watanabe, 2014). While most dermal

melanocytosis are congenital or have an onset in

early childhood, there is a group that is clearly

acquired, with an onset in adult life. Park et al.,

2014). In this report, dermal melanocytosis

appearing on the face, named facial dermal

melanocytosis (FDM) has been reviewed.

Acquired bilateral nevus of Ota-like macules

(ABNOM), also named Hori nevus, was first

described by Hori et al in 1984. Clinically, ABNOM

is characterized by multiple speckled blue-brown

and/or slate-gray macules occurring bilaterally on

the malar regions or less commonly forehead, upper

eyelids, and cheeks and nose. It most commonly

presents in Asian women after the third decade of

life with 89% described as having Fitzpatrick skin

phototype IV (Lee et al., 2004; Watanabe, 2014).

We report a case of an Indonesian female, aged

16 years old, who suffered Nevus Hori. She

complained about the dark patches which appeared

in her left cheek since 2 years ago. She has been

treated with combination of CO

fractional laser and

Nd:YAG laser. This report discusses about the

clinical presentation, diagnosis, and treatment.

The

aim of this study is to evaluate the clinical

manifestation and management of Nevus Hori.

456

Liuwan, C. and Listiawan, M.

Nevus Hori Treated with Laser Combination: A Case Report.

DOI: 10.5220/0008159604560460

In Proceedings of the 23rd Regional Conference of Dermatology (RCD 2018), pages 456-460

ISBN: 978-989-758-494-7

2 CASE

16-years-old female, came to Dermato-Venereology

Outpatient Clinic of Dr. Soetomo General Hospital

Surabaya on December 22

2016 with main

complain dark patches at her left cheek since 2 years

ago. The dark patches ware getting wider. She never

complain about itchy, numbness or pain sensation on

her left cheek. She had no complained about visual

disturbances or dizzy.

She already gone to general doctor at Jombang

and got 3 type of cream for morning and night, but

there was no improvement. She usually uses facial

foam that she get from supermarket. She never has

same complaint before. There are no family

members who have the same complaint as her. There

were no history of food and drug allergy in the

patient and her family, no history of atopi in the

patient and her family.

She is a high school student and usually goes to

school by bicycle or on foot. She prefer to stay at

home and helping her mother doing the housework

than playing outside. She only has direct sun

exposure during going to school and going home.

General physical examination was within normal

limit, with no sign of anemic, icterus, cyanotic or

respiratory distress. The blood pressure was 110/70,

pulse rate was 96 times per minute, respiratory rate

20 times per minute and body temperature was 36,3

°C. No abnormalities found on thorax and

abdominal examination. No swelling on his

extremity.

From dermatological examination on left cheek

region, there were hyperpigmented macule, vary in

size, bluish in color, sharply marginated, and from

oculi region there was no episcleral pigmentation.

Laboratory result revealed the complete blood

count all within normal limit. The histology

examination has not been done yet because patient

still refuse to do the biopsy. Patient has been

consulted to ophtalmologist and the result is all

within normal limit. The patient’s visus is normal

(6/6) and there are no pigmentation in her eyes. The

tonometry and funduscopy examination also

revealed normal result and no sign of glaukoma in

this patient.

The patient was treated with CO

fractional laser

and Nd:YAG laser 1064 nm for 3 sessions and the

lesion became lighter. The progression and

improvement of the patient can be seen in the figures

3 DISCUSSION

Pigmentation disorders of the skin can either be

hypomelanotic, hypermelanotic, or may present with

a pattern of mixed hypo- and hypermelanosis. The

diagnosis of these disorders can be quite challenging

(Lee et al., 2004). Dermal melanocytosis define a

broad group of congenital and acquired melanocytic

lesions characterized by the presence of intradermal

dendritic, variably pigmented, spindle shaped

melanocytes with or without presence of dermal

melanophages. This group includes Mongolian spot,

nevus of Ota, nevus of Ito and acquired bilateral

nevus of Ota-like macules (ABNOM), and other

unusual cases of dermal melanocytosis that have

been introduced to the literature as part of this

category on the basis of similar histopathological

findings (Watanabe, 2014).

From the standpoint of age of onset, there is

overlap between classical nevus of Ota and Hori’s.

The distribution of pigmentation is identical between

nevus of Ota and Hori’s nevi, although mostly

unilateral in nevus of Ota. The histology of Hori’s is

identical to nevus of Ota.

Figure 1: Comparison of 1

Laser session (left), 2

Laser session (middle), and 3

Laser session (right).

Nevus Hori Treated with Laser Combination: A Case Report

457

3.1 Nevus Hori

Acquired bilateral nevus of Ota-like macules

(ABNOM), also named Hori nevus, was first

described by Hori et al in 1984. Clinically, ABNOM

is characterized by multiple speckled blue-brown

and/or slate-gray macules occurring bilaterally on

the malar regions or less commonly forehead, upper

eyelids, and cheeks and nose. It most commonly

presents in Asian women after the third decade of

life (Cho et al., 2009; Park et al., 2014; Watanabe,

2014).

Hori et al hypothesized that the pathogenesis of

ABNOM may be attributed to later reactivation of

preexisting misplaced dermal melanocytes that may

result from faulty migration during embryological

development, dropping off from the basal layer of

epidermis or migration from follicular bulb

melanocytes. Mizoguchi and Mizushima concluded

that there are ‘‘two hits’’ are needed for the

development of ABNOM: the first representing the

ectopic placement of inactive, poorly melanized

dermal melanocytes at birth or soon thereafter and

the second, the activation of these melanocytes in

response to ultraviolet exposure, excessive sex

hormone, chronic inflammation such as atopic

dermatitis, or other unknown triggers (Murakami,

2000; Park et al., 2014).

The diagnosis of ABNOM was made by clinical

appearances, according to the description by Hori et

al.and skin biopsies were not performed. The color

of ABNOM was categorized into one of four groups,

namely brown, slate-gray, brown–blue, and blue

(Cho et al., 2009).

3.2 Nevus Ota

Nevus of Ota or nevus fuscocaeruleus

ophthalmomaxillaris was first described by the

Japanese dermatologist Ota in 1939 as a dermal

melanocytic hamartoma that presents as bluish

hyperpigmentation along the ophthalmic, maxillary

and mandibular branches of the trigeminal nerve

(Metha & Balachandran, 2007; Lapreere et al.,

2012). It is most frequently seen in the Asian

population, has a female predominance, and is

usually congenital, although appearance in early

childhood or at puberty has been described (Kumari

& Thappa, 2006; Lapeere et al., 2012).

The pigmentation of Ota’s nevus is composed of

flat blue black or slate grey macules intermingled

with small brown specks. The intensity of

pigmentation may be influenced by fatigue,

menstruation, insomnia and weather.

Mucosal

pigmentation may occur involving conjunctiva,

sclera, and tympanic membrane (oculodermal

melanocytosis), or other sites.

Ocular melanosis in

22-77% cases is almost always ipsilateral and deep

in the conjunctiva (Metha & Balachandran, 2007;

Lapeere et al., 2012). Pigmentation may also affect

the sclera, cornea, iris, choroid and less commonly

the optic nerve, retrobulbar fat, orbit, periosteum and

extraocular muscles (Metha & Balachandran, 2007).

The pigmentation of mucous membranes of the head

and neck is variable; tympanic membrane being

most frequently affected although nasal, buccal,

pharyngeal and rarely palatine mucosa may be

involved (Sharan et al., 2005). At present, it is

believed that nevus of Ota is caused by heteroplasia

that occurs in melanocyte migration during

embryonic development (Huang et al., 2013).

Nevus of Ota involves innervated areas of the

first branch (V1) and second branch (V2) of the

trigeminal nerve mainly affects the eye region and

pars zygomatica, and the color of the skin lesion is

brown or blue, the diameter of the area is 1–10 cm

or larger.

Tanino classified nevus of Ota into 4

types according to the skin lesion involvement area:

Type I was mild, Type II was moderate, Type III

was severe, Type IV was bilateral type (Huang et al.,

2013).

In our report, the patient is female, age 16 years

old with the symptom dark patches at her left cheek

since 2 years ago. There are no patches since she

birth. The dark patches were getting wider, and she

also has ultraviolet exposure since she usually going

to school by bicycle and on foot. According to this

situation is suitable with the clinical manifestation of

Nevus Hori or ABNOM. From the theory, ABNOM

is an acquired dermal melanocytosis which induced

by ultraviolet exposure, sex hormone, and chronic

inflammation.

She never complain about itchy, numbness or

pain sensation on her cheek. She had no complained

about visual disturbances or dizzy. The patient’s

visus is normal (6/6) and there are no pigmentation

in her eyes. The tonometry and funduscopy

examination also revealed normal result and no sign

of glaukoma in this patient. Nevus Hori is said to

have lack mucosal involvement.

From physical examination at left cheek region

there were hyperpigmented macule, vary in size,

bluish in color, sharply marginated. In this case, the

histopathology examination has not been done yet

because the patient still refuse to do the biopsy.

Nevus Hori tends to appear symmetrically at both

cheek (malar area). In the other hands, Nevus Ota

can appear unilaterally in one side of face. In this

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458

case because the clinical manifestations appear on

the left side only, so based on Tanino classification

included in type I.

Pigmentary disorders appearing on the face, even

if they are benign, frequently cause cosmetic and

psychological problems to many people, especially

women. As with most dermal pigmentary disorders,

single treatment with topical bleaching agents or

superficial-to medium-depth chemical peels is

generally not effective for long-term pigmentary

reduction or elimination.

Although methods such

as dermabrasion, cryotherapy, surgical excision, and

cosmetic camouflage had been attempted for FDM

in the past, these have been largely replaced by

pigment-selective lasers given the lower risk of scar

formation and permanent hypopigmentation or

depigmentation with these devices (Kunachak et al.,

1996; Kar et al., 2011).

Three types of Q-switched lasers have been used

widely to treat FDM. These include the Q-switched

694 nm Ruby laser, Q-switched 755 nm Alexandrite

laser and the Q-switched 1064 nm Nd:YAG laser.

Previous studies have shown that all of them were

able to provide excellent results in treating FDM.

Because Q-switched (QS) laser devices have

been widely accepted as the treatment of choice for

nevus of Ota on the principle of selective

photothermolysis and because ABNOM is

histopathologically similar to nevus of Ota, QS

lasers such as the QS ruby laser (QSRL), QS

neodymium:yttrium-aluminum-garnet laser

(QSNYL), and QS alexandrite laser (QSAL) have all

been used for the treatment of ABNOM since the

first report of Hori et al (Lee et al., 2009; Watanabe,

2014).

The laser fluence used was 7 to 10 J/cm2, at a

repetition rate of 1 Hz, and with a spot size of 2 to 4

mm. The number of treatment sessions ranged from

1 to 6 (mean 2.3 sessions) with short treatment

intervals (mean 2.2 weeks) (Watanabe, 2004).

In addition to local thermal destruction and

stimulation, fractionated devices may also play an

important role for drug delivery into the tissue and

for extruding material out of the skin, as in the

studies by Haedersdal et al. This has also been

recently reported by Brian Wei Cheng using a

combination between non ablative fractionated

erbium : YAG laser and Q-switches Nd:YAG laser

ias an effective and safe treatment to treat Nevus

Hori (Sakamoto et al., 2013; Tian, 2015).

After laser treatment, ABNOM showed a higher

degree of erythema as well as a higher incidence and

degree of Post Inflammatory Hyperpigmentation

(PIH) compared to that of nevus of Ota. Several

causes for the increased prevalence of PIH in

ABNOM after laser treatment are considered. First,

the lesion in ABNOM was located in the superficial

dermal layer and there were few epidermal

melanocytes and melanin pigment. In the treatment

of Q-switches laser, melanin acts as a chromophore,

therefore melanin in the epidermis allows laser to be

selectively absorbed so that the epidermal tissue

becomes vacuolized due to the heat. The

melanocytes and melanin pigment of the vacuolated

epidermis are dropped into the dermis, and are

presumed to induce PIH.

Next, melanocytes were especially clustered in

the perivascular area in ABNOM, whereas in nevus

of Ota melanocytes were evenly distributed

throughout the dermal layer in between collagen

fibers. The presence of many melanocytes in the

perivascular area may lead to indirect vascular

damage, increase melanogenesis, induce many types

of inflammatory responses, and produce chemical

substances. These process can induce PIH

(Watanabe, 2014).

Patient already had Q-switched 1064 Nd:YAG

laser combined with fractional CO

for three times

and the lesion is having improvement although she

has not satisfied yet. The patient is advised to

continue the laser treatment and observed the

occurrence of PIH. The prognosis of this case is

good.

4 CONCLUSION

Pigmentary disorders appearing on the face

frequently cause cosmetic and psychological

problems to many people, especially women. The

diagnosis of FDM can be quite challenging because

of the similarity manifestation of Nevus of Ota and

ABNOM. Q-switched laser give a promising result

to treat this pigmentary problems. Combination with

fractional laser aimed to increase the ability to

remove pigment.

REFERENCES

Cho, S.B., Park, S.J., Kim, M.J., Bu, T.S., 2009.

Treatment of acquired bilateral nevus of Ota-like

macules (Hori’s nevus) using 1064-nm Q-switched

Nd:YAG laser with low fluence. International Journal

of Dermatology 48, 1308–1312. doi:10.1111/j.1365-

4632.2008.04061.x

Huang, W.H., Wang, H.W., Sun, Q.N., Jin, H.Z., Liu,

Y.H., Ma, D.L., Zuo, Y.G., Zheng, H.Y., Wan, K.,

Jing, Q., Zhao, Y.L., 2013. A new classification of

Nevus Hori Treated with Laser Combination: A Case Report

459

nevus of Ota. Chinese Medical Journal 126, 3910–

3914. doi:10.3760/cma.j.issn.0366-6999.20131211

Kar, H.K., Gupta, L., 2014. 1064 nm Q switched Nd:

YAG laser treatment of nevus of Ota: an Indian open

label prospective study of 50 patients. Indian journal

of dermatology, venereology and leprology 77, 565–

70. doi:10.4103/0378-6323.84057

Kumari, R., Thappa, D.M. 2006. Familial nevus of ota.

Indian J Dermatol; 52: 198-9.

Kunachak, S., Kunachakr, S., Sirikulchayanonta, V.,

Leelaudomniti, P., 1996. Dermabrasion is an effective

treatment for acquired bilateral nevus of Ota-like

macules. Dermatologic Surgery 22, 559–562.

doi:10.1111/j.1524-4725.1996.tb00374.x

Lapeere, H. 2012. Hypomelanoses and hypermelanoses In:

Goldsmith, L.A., Katz, S.I., Gilchrest, B.A., Paller.

A.S, Leffell, D.J., Wolf, K., editors. Fitzpatrick’s

dermatology in general medicine. 8

ed. New York:

Mc Graw Hill.p. 823.

Lee, B., You, C.K., Won, H.K., Lee, E.S., 2004.

Comparison of characteristics of acquired bilateral

nevus of Ota-like macules and nevus of Ota according

to therapeutic outcome. Journal of Korean Medical

Science 19, 554–559.

doi:10.3346/jkms.2004.19.4.554.

Lee, W.J., Han, S.S., Chang, S.E., Lee, M.W., Choi, J.H.,

Moon, K.C., Koh, J.K., 2009. Q-switched ND:YAG

laser therapy of acquired bilateral nevus of Ota-like

macules. Annals of Dermatology 21, 255–260.

doi:10.5021/ad.2009.21.3.255

Mehta, V., Balachandran, C. 2007. Case report : bilateral

nevus of ota. J Pakistan Association Dermatol; 17: 59-

61.

Murakami, F., Baba, T., Mizoguchi, M. 2000. Ultraviolet-

induced generalized acquired dermal melanocytosis

with numerous melanophages. Br J

Dermatol;142:184-6

Park, J.M., Tsao, H., Tsao, S., 2009. Acquired bilateral

nevus of Ota-like macules (Hori nevus): Etiologic and

therapeutic considerations. Journal of the American

Academy of Dermatology.

doi:10.1016/j.jaad.2008.10.054.

Sakamoto, F.H., Jalian, H.R., Anderson, R.R. 2013.

Understanding lasers, lights, and tissue interactions In:

Hruza, G., Avram, M., editors. Lasers and lights. 3

Ed. New York: Elsevier.p. 6-8.

Sharan S, Grigg JR, Billson FA. Bilateral nevus of Ota

with choroidal melanoma and diffuse retinal

pigmentation in a dark skinned person. Br J

Ophthalmol 2005; 89: 1529. Sharan, S., Grigg, J.R.,

Billson, F.A., 2005. Bilateral naevus of Ota with

choroidal melanoma and diffuse retinal pigmentation

in a dark skinned person [1]. British Journal of

Ophthalmology. doi:10.1136/bjo.2005.070839

Tian, B.C.A., 2015. Novel treatment of Hori′s nevus: A

combination of fractional nonablative 2,940-nm

Er:YAG and low-fluence 1,064-nm Q-switched

Nd:YAG laser. Journal of Cutaneous and Aesthetic

Surgery 8, 227. doi:10.4103/0974-2077.172198

Watanabe S. 2014. Facial Dermal Melanocytosis. Austin J

Dermatolog; 1(2):1-6

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