Generic selectors
Exact matches only
Search in title
Search in content
Post Type Selectors
Search in posts
Search in pages
Filter by Categories
15th National Conference of the IAOMFP, Chennai, 2006
Abstract
Abstracts from current literature
Acne in India: Guidelines for management - IAA Consensus Document
Addendum
Announcement
Art & Psychiatry
Article
Articles
Association Activities
Association Notes
Award Article
Book Review
Brief Report
Case Analysis
Case Letter
Case Letters
Case Notes
Case Report
Case Reports
Clinical and Laboratory Investigations
Clinical Article
Clinical Studies
Clinical Study
Commentary
Conference Oration
Conference Summary
Continuing Medical Education
Correspondence
Corrigendum
Cosmetic Dermatology
Cosmetology
Current Best Evidence
Current Issue
Current View
Derma Quest
Dermato Surgery
Dermatopathology
Dermatosurgery Specials
Dispensing Pearl
Do you know?
Drug Dialogues
e-IJDVL
Editor Speaks
Editorial
Editorial Remarks
Editorial Report
Editorial Report - 2007
Editorial report for 2004-2005
Errata
Erratum
Focus
Fourth All India Conference Programme
From Our Book Shelf
From the Desk of Chief Editor
General
Get Set for Net
Get set for the net
Guest Article
Guest Editorial
History
How I Manage?
IADVL Announcement
IADVL Announcements
IJDVL Awards
IJDVL AWARDS 2015
IJDVL Awards 2018
IJDVL Awards 2019
IJDVL Awards 2020
IJDVL International Awards 2018
Images in Clinical Practice
Images in Dermatology
In Memorium
Inaugural Address
Index
Knowledge From World Contemporaries
Leprosy Section
Letter in Response to Previous Publication
Letter to Editor
Letter to the Editor
Letter to the Editor - Case Letter
Letter to the Editor - Letter in Response to Published Article
LETTER TO THE EDITOR - LETTERS IN RESPONSE TO PUBLISHED ARTICLES
Letter to the Editor - Observation Letter
Letter to the Editor - Study Letter
Letter to the Editor - Therapy Letter
Letter to the Editor: Articles in Response to Previously Published Articles
Letters in Response to Previous Publication
Letters to the Editor
Letters to the Editor - Letter in Response to Previously Published Articles
Letters to the Editor: Case Letters
Letters to the Editor: Letters in Response to Previously Published Articles
Media and news
Medicolegal Window
Messages
Miscellaneous Letter
Musings
Net Case
Net case report
Net Image
Net Images
Net Letter
Net Quiz
Net Study
New Preparations
News
News & Views
Obituary
Observation Letter
Observation Letters
Oration
Original Article
ORIGINAL CONTRIBUTION
Original Contributions
Pattern of Skin Diseases
Pearls
Pediatric Dermatology
Pediatric Rounds
Perspective
Presedential Address
Presidential Address
Presidents Remarks
Quiz
Recommendations
Regret
Report
Report of chief editor
Report of Hon : Treasurer IADVL
Report of Hon. General Secretary IADVL
Research Methdology
Research Methodology
Resident page
Resident's Page
Resident’s Page
Residents' Corner
Residents' Corner
Residents' Page
Retraction
Review
Review Article
Review Articles
Reviewers 2022
Revision Corner
Self Assessment Programme
SEMINAR
Seminar: Chronic Arsenicosis in India
Seminar: HIV Infection
Short Communication
Short Communications
Short Report
Snippets
Special Article
Specialty Interface
Studies
Study Letter
Study Letters
Supplement-Photoprotection
Supplement-Psoriasis
Symposium - Contact Dermatitis
Symposium - Lasers
Symposium - Pediatric Dermatoses
Symposium - Psoriasis
Symposium - Vesicobullous Disorders
SYMPOSIUM - VITILIGO
Symposium Aesthetic Surgery
Symposium Dermatopathology
Symposium-Hair Disorders
Symposium-Nails Part I
Symposium-Nails-Part II
Systematic Review and Meta-Analysis
Systematic Reviews and Meta-analyses
Systematic Reviews and Meta-analysis
Tables
Technology
Therapeutic Guideline-IADVL
Therapeutic Guidelines
Therapeutic Guidelines - IADVL
Therapeutics
Therapy
Therapy Letter
Therapy Letters
View Point
Viewpoint
What’s new in Dermatology
Generic selectors
Exact matches only
Search in title
Search in content
Post Type Selectors
Search in posts
Search in pages
Filter by Categories
15th National Conference of the IAOMFP, Chennai, 2006
Abstract
Abstracts from current literature
Acne in India: Guidelines for management - IAA Consensus Document
Addendum
Announcement
Art & Psychiatry
Article
Articles
Association Activities
Association Notes
Award Article
Book Review
Brief Report
Case Analysis
Case Letter
Case Letters
Case Notes
Case Report
Case Reports
Clinical and Laboratory Investigations
Clinical Article
Clinical Studies
Clinical Study
Commentary
Conference Oration
Conference Summary
Continuing Medical Education
Correspondence
Corrigendum
Cosmetic Dermatology
Cosmetology
Current Best Evidence
Current Issue
Current View
Derma Quest
Dermato Surgery
Dermatopathology
Dermatosurgery Specials
Dispensing Pearl
Do you know?
Drug Dialogues
e-IJDVL
Editor Speaks
Editorial
Editorial Remarks
Editorial Report
Editorial Report - 2007
Editorial report for 2004-2005
Errata
Erratum
Focus
Fourth All India Conference Programme
From Our Book Shelf
From the Desk of Chief Editor
General
Get Set for Net
Get set for the net
Guest Article
Guest Editorial
History
How I Manage?
IADVL Announcement
IADVL Announcements
IJDVL Awards
IJDVL AWARDS 2015
IJDVL Awards 2018
IJDVL Awards 2019
IJDVL Awards 2020
IJDVL International Awards 2018
Images in Clinical Practice
Images in Dermatology
In Memorium
Inaugural Address
Index
Knowledge From World Contemporaries
Leprosy Section
Letter in Response to Previous Publication
Letter to Editor
Letter to the Editor
Letter to the Editor - Case Letter
Letter to the Editor - Letter in Response to Published Article
LETTER TO THE EDITOR - LETTERS IN RESPONSE TO PUBLISHED ARTICLES
Letter to the Editor - Observation Letter
Letter to the Editor - Study Letter
Letter to the Editor - Therapy Letter
Letter to the Editor: Articles in Response to Previously Published Articles
Letters in Response to Previous Publication
Letters to the Editor
Letters to the Editor - Letter in Response to Previously Published Articles
Letters to the Editor: Case Letters
Letters to the Editor: Letters in Response to Previously Published Articles
Media and news
Medicolegal Window
Messages
Miscellaneous Letter
Musings
Net Case
Net case report
Net Image
Net Images
Net Letter
Net Quiz
Net Study
New Preparations
News
News & Views
Obituary
Observation Letter
Observation Letters
Oration
Original Article
ORIGINAL CONTRIBUTION
Original Contributions
Pattern of Skin Diseases
Pearls
Pediatric Dermatology
Pediatric Rounds
Perspective
Presedential Address
Presidential Address
Presidents Remarks
Quiz
Recommendations
Regret
Report
Report of chief editor
Report of Hon : Treasurer IADVL
Report of Hon. General Secretary IADVL
Research Methdology
Research Methodology
Resident page
Resident's Page
Resident’s Page
Residents' Corner
Residents' Corner
Residents' Page
Retraction
Review
Review Article
Review Articles
Reviewers 2022
Revision Corner
Self Assessment Programme
SEMINAR
Seminar: Chronic Arsenicosis in India
Seminar: HIV Infection
Short Communication
Short Communications
Short Report
Snippets
Special Article
Specialty Interface
Studies
Study Letter
Study Letters
Supplement-Photoprotection
Supplement-Psoriasis
Symposium - Contact Dermatitis
Symposium - Lasers
Symposium - Pediatric Dermatoses
Symposium - Psoriasis
Symposium - Vesicobullous Disorders
SYMPOSIUM - VITILIGO
Symposium Aesthetic Surgery
Symposium Dermatopathology
Symposium-Hair Disorders
Symposium-Nails Part I
Symposium-Nails-Part II
Systematic Review and Meta-Analysis
Systematic Reviews and Meta-analyses
Systematic Reviews and Meta-analysis
Tables
Technology
Therapeutic Guideline-IADVL
Therapeutic Guidelines
Therapeutic Guidelines - IADVL
Therapeutics
Therapy
Therapy Letter
Therapy Letters
View Point
Viewpoint
What’s new in Dermatology
View/Download PDF

Translate this page into:

Net Letter
2019:85:1;130-130
doi: 10.4103/ijdvl.IJDVL_1025_16
PMID: 29176249

Partial unilateral lentiginosis with ipsilateral ocular involvement and seizures

Vishal Gupta1 , Neha Taneja1 , Binod K Khaitan1 , Manish Singh2
1 Department of Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi, India
2 Department of Pathology, All India Institute of Medical Sciences, New Delhi, India

Correspondence Address:
Binod K Khaitan
Department of Dermatology and Venereology, All India Institute of Medical Sciences, Ansari Nagar, New Delhi - 110 029
India
Published: 24-Nov-2017
How to cite this article:
Gupta V, Taneja N, Khaitan BK, Singh M. Partial unilateral lentiginosis with ipsilateral ocular involvement and seizures. Indian J Dermatol Venereol Leprol 2019;85:130
Copyright: (C)2019 Indian Journal of Dermatology, Venereology, and Leprology

Sir,

A 21-year-old, otherwise healthy female consulted us for asymptomatic, brown-colored “spotty” pigmentation on the right side of her face noticed 5 years back. Cutaneous examination showed multiple round-to-oval discrete, 0.1–0.5-cm sized, dark brown-colored macules distributed in the ophthalmic and maxillary divisions of the trigeminal nerve on the right-side of the face with a sharp midline demarcation [Figure - 1]a. There was no background pigmentation suggestive of a café-au-lait- macule. Eye examination revealed brown-colored macules on the bulbar conjunctiva of the right eye [Figure - 1]b. Conjunctiva on the left side, oral, and nasal mucosa were not affected. Dermoscopic examination of the pigmented macule under nonpolarized as well as polarized light (x10 magnification) showed an accentuated, regular, brown-colored pigment network [Figure - 2]. With the provisional clinical diagnosis of nevus of Ota and segmental lentiginosis, a skin biopsy from one of the pigmented macules was planned. During the biopsy procedure, the patient developed an episode of generalized tonic seizure lasting for less than a minute. The mother informed that she had been experiencing such transient episodes intermittently since the age of 10 years, but these were considered trivial and no medical consultation was ever sought. Histopathological examination showed accentuated epidermal basilar pigmentation with an increase in the number of melanocytes, consistent with a lentigo [Figure - 3]. No features of dermal melanocytosis were seen. A computerized tomographic scan of the brain, electroencephalogram, and ophthalmological examination were within normal limits. A final diagnosis of partial unilateral lentiginosis with ipsilateral ocular involvement was made. The patient was started on oxcarbazepine for seizures after neurological evaluation, and is being planned for Q-switched Nd: YAG laser for lentigines.

Figure 1
Figure 2: Dermoscopic examination under non-polarized light showing an accentuated brown-coloured regular pseudoreticular pigment network
Figure 3: Photomicrograph showing increased basilar pigmentation with an increase in the number of melanocytes (H and E, ×100)

Partial unilateral lentiginosis is an unusual pigmentary disorder characterized by multiple lentigines clustered over a localized area with a sharp midline demarcation, usually confined to one or more dermatomes. The onset is usually in childhood, and any body site can be affected. When on the face, partial unilateral lentiginosis can be mistaken for nevus of Ota, especially if ocular pigmentation is present, like in our patient. Interestingly, ocular pigmentation has been reported in a few patients with partial unilateral lentiginosis as well.[1],[2],[3] However, unlike the mottled blue-gray pigmentation in nevus of Ota, the pigmentation in partial unilateral lentiginosis is brown-colored, both in the skin as well as the eye. Histopathology can distinguish between the two, showing features of lentigo in partial unilateral lentiginosis and dermal melanocytosis in nevus of Ota.

Central nervous system manifestations have been rarely reported in nevus of Ota and partial unilateral lentiginosis, though the association with nevus of Ota as meningeal melanocytomas and intracranial melanomas is more well-documented in the literature.[4] Our patient had brown-colored macules on one side of her face and ipsilateral bulbar conjunctiva and skin biopsy showed features of a lentigo. She also had a seizure disorder which had been so far unrecognized. There are only a few earlier cases of partial unilateral lentiginosis associated with central nervous system abnormalities. Schaffer et al.[1] reviewed previously reported cases of partial unilateral lentiginosis and their associations [Table - 1].[1],[3],[5] Though the majority of the reported “associations” were regarded as coincidental, central nervous system abnormalities were considered to have a possible association explained by a common developmental defect in the neural crest. We cannot be certain whether the seizures in our patient represent a true association or a mere coincidence. Nonetheless, we report this case to increase the awareness among dermatologists regarding the ocular involvement in partial unilateral lentiginosis, and its possible association with central nervous system abnormalities.

Table 1: Reported associations of partial unilateral lentiginosis

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References
1.
Schaffer JV, Lazova R, Bolognia JL. Partial unilateral lentiginosis with ocular involvement. J Am Acad Dermatol 2001;44:387-90.
[Google Scholar]
2.
Kim EH, Kang HY. Partial unilateral lentiginosis with ocular involvement. Eur J Dermatol 2006;16:582-3.
[Google Scholar]
3.
Serarslan G. Partial unilateral lentiginosis with ipsilateral ocular nevus. J Eur Acad Dermatol Venereol 2007;21:281-3.
[Google Scholar]
4.
Franceschini D, Dinulos JG. Dermal melanocytosis and associated disorders. Curr Opin Pediatr 2015;27:480-5.
[Google Scholar]
5.
González-Sixto B, De la Torre C, Posada C, Sevillano C. Partial unilateral lentiginosis associated with nevus of Ota. J Am Acad Dermatol 2010;63:353-5.
[Google Scholar]

Fulltext Views
3,058

PDF downloads
4,696
Show Sections