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:

Therapy Letter
87 (
4
); 581-584
doi:
10.25259/IJDVL_822_20

Tufted angioma successfully treated with topical timolol gel-forming solution

Department of Dermatology, and Venereology, AIIMS, Bhubaneswar, Odisha, India
Department of Dermatology, Venereology and Leprology, Pondicherry Institute of Medical Sciences, Puducherry, India
Department of Dermatology,Venereology and Leprology, JIPMER, Puducherry, India
Department of Dermatology, North Eastern Indira Gandhi Regional Institute of Health and Medical Sciences, Shillong, Meghalaya, India
Department of Pathology,JIPMER, Puducherry, India

Corresponding author: Dr. Laxmisha Chandrashekar, Department of Dermatology, Venereology and Leprology, JIPMER, Puducherry - 605 006, India. laxmishac@gmail.com

Licence
This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: Behera B, Remya R, Chandrashekar L, Thappa DM, Gochhait D. Tufted angioma successfully treated with topical timolol gel-forming solution. Indian J Dermatol Venereol Leprol 2021;87:581-4.

Sir,

A four-month-old male child born out of nonconsanguineous marriage was brought by his parents with the complaint of a slowly growing pigmented lesion on the right leg since birth. The lesion was not associated with any history of bleeding or ulceration. Cutaneous examination revealed a solitary, well-defined, reddish-brown, firm plaque of size 6 cm × 3cm circumferentially encircling two-thirds of the right lower leg in a horseshoe manner, sparing the posterior-lateral area [Figure 1]. There was no thrill or bruit. Other mucocutaneous and systemic examinations were within normal limits. Differential diagnoses of infantile hemangioma, tufted angioma, vascular malformation and congenital dermatofibrosarcoma protuberans were considered. A skin biopsy showed tufts of proliferating vessels in the dermis and superficial subcutis in a cannonball distribution surrounded by empty crescent-shaped vessel and fibrous tissue. The vascular tufts were composed of closely packed epithelioid and spindle-shaped endothelial cells and were surrounded by pericytes. On immunohistochemistry, the endothelial cells were highlighted by CD31 and the pericytes by smooth muscle antigen [Figure 2]. A diagnosis of tufted angioma was made. The coagulation profile was within the normal limits. The child was treated with timolol 0.5% gel-forming solution thrice daily. Four months after starting treatment, there was almost complete resolution of the lesion [Figure 3], following which the dose was made twice daily, then once daily, and stopped over the next two months. There was no recurrence of the lesion one year after stopping treatment. During the therapy, the parents did not complain about any adverse reactions. Before the initiation of timolol therapy, the child’s blood pressure, heart rate and glucose level were normal. On day-one, the child’s blood pressure and heart rate were measured at a 6-h interval. The parents were advised to measure the blood pressure and heart rate every week at a nearby hospital. The mother was educated to breastfeed the child frequently and notice for any hypoglycemic symptoms. At each monthly follow-up visit, the child’s blood sugar was within normal limits.

Solitary well-defined reddish-brown plaque on the right lower leg
Figure 1a:
Solitary well-defined reddish-brown plaque on the right lower leg
Closer view of the plaque
Figure 1b:
Closer view of the plaque
Tufts of proliferating vessels in a cannonball distribution surrounded by empty crescent-shaped vessels and fibrous tissue (H and E, ×50)
Figure 2a:
Tufts of proliferating vessels in a cannonball distribution surrounded by empty crescent-shaped vessels and fibrous tissue (H and E, ×50)
A tuft of proliferating vessels (H and E, ×100)
Figure 2b:
A tuft of proliferating vessels (H and E, ×100)
Immunohistochemistry highlighting CD31 positive endothelial cells (IHC, ×400)
Figure 2c:
Immunohistochemistry highlighting CD31 positive endothelial cells (IHC, ×400)
Immunohistochemistry showing smooth muscle antigen-positive pericytes (IHC, ×400)
Figure 2d:
Immunohistochemistry showing smooth muscle antigen-positive pericytes (IHC, ×400)
Complete resolution of the tufted angioma
Figure 3a:
Complete resolution of the tufted angioma
Complete resolution of the tufted angioma
Figure 3b:
Complete resolution of the tufted angioma

Tufted angioma is a rare benign vascular tumor that commonly occurs in infancy and childhood. It can be both congenital and acquired and rarely affects adults. It usually presents as a dusky red or violaceous solitary tumor or infiltrating plaque. Associated hyperhidrosis or hypertrichosis can rarely be seen.1 Tufted angioma can have a variable clinical outcome. It can spontaneously regress partially or completely or can enlarge rapidly, leading to life-threatening coagulopathy called the Kasabach-Merritt phenomenon.

On histopathology, the diagnosis is clinched by the presence of proliferating capillaries in cannonball distribution in the dermis and upper subcutis along with lymphatic-like vessels, as in our case.1

Treatment options available for tufted angioma are systemic corticosteroids, vincristine, radiotherapy, interferon, aspirin, ticlopidine, sirolimus, surgical resection and embolization. A meta-analysis by Liu et al. found vincristine to be a better option for the treatment of tufted angioma.2 Sirolimus is increasingly being used in steroid-resistant vascular tumors associated with the Kasabach-Merritt phenomenon.3 The use of topical timolol in vascular proliferation is encouraged by the therapeutic success of oral propranolol in both infantile hemangioma and tufted angioma. A case of tufted angioma without the Kasabach-Merritt phenomenon was successfully managed with oral propranolol.4 We decided to use topical timolol gel-forming solution, instead of oral propranolol, due to the relatively small size of the lesion and no associated complications.

Timolol is a nonselective beta-blocker with activity against both β1 and β2 adrenergic receptors.5 It acts by causing vasoconstriction, blockade of the proangiogenic signaling pathway and apoptosis.5 It has been effectively used in the treatment of both complicated and uncomplicated infantile hemangioma.6 Various formulations like timolol eye drops, gel, gel-forming solution and hydrogel have been used for the treatment of infantile hemangioma with frequencies varying from twice daily to five times daily.7 Timolol gel-forming solution is preferred over other topical solutions because of its less systemic absorption. Systemic side effects like hypotension, bradycardia, hypoglycemia and bronchospasm are rare with topical timolol, but due caution is advised when used in mucosal surfaces or ulcerated lesions, preterm infants, and in occluded areas like diaper region. Dalla Costa et al. have advocated a simplified approach regarding the dose of timolol gel-forming solution to be used, that is, the maximum drops of timolol gel-forming solution to be used per day is equal to the bodyweight of the child in kilograms.8

According to the parents, the plaque was slowly growing until the child presented to us, and the improvement only started after the application of the timolol gel-forming solution. It is difficult to predict the role of spontaneous resolution in the subsidence of the plaque of tufted angioma in the index case. Another advantage of the use of timolol gel-forming solution was the absence of residual skin change following therapy, in contrast to the reports of residual cutaneous changes after spontaneous resolution of tufted angioma.9 A non-regressing painful tufted angioma in a five-year-old girl without any features of the Kasabach-Merritt phenomenon completely resolved by topical timolol maleate 0.5% ophthalmic solution without any recurrence has been reported.10

In conclusion, we are reporting an unusual presentation of tufted angioma as a reddish-brown horseshoe-shaped plaque on the right leg that was successfully treated by a timolol gel-forming solution. Topical timolol gel-forming solution can be a safe and effective therapeutic option for the treatment of uncomplicated tufted angioma.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

  1. , , , , , . Clinical spectrum of tufted angiomas in childhood: A report of 13 cases and a review of the literature. Arch Dermatol. 2010;146:758-63.
    [CrossRef] [PubMed] [Google Scholar]
  2. , , , , , , et al. Treatment of kaposiform hemangioendothelioma and tufted angioma. Int J Cancer. 2016;139:1658-66.
    [CrossRef] [PubMed] [Google Scholar]
  3. , , , , , . Successful management of steroid-resistant vascular tumors associated with the Kasabach-Merritt phenomenon using sirolimus. J Dermatol. 2018;45:580-3.
    [CrossRef] [PubMed] [Google Scholar]
  4. , , , , . Successful treatment of tufted angioma with propranolol. J Dermatol. 2014;41:1120-2.
    [CrossRef] [PubMed] [Google Scholar]
  5. , . Effect of topical timolol on response rate and adverse events in infantile hemangioma: A meta-analysis. Arch Dermatol Res. 2018;310:261-9.
    [CrossRef] [PubMed] [Google Scholar]
  6. , , . Topical timolol: A safer alternative for complicated and un-complicated infantile hemangiomas. Indian J Dermatol. 2013;58:330.
    [CrossRef] [PubMed] [Google Scholar]
  7. , , , , , , et al. Topical application of 0.5% timolol maleate hydrogel for the treatment of superficial infantile hemangioma. Front Oncol. 2017;7:137.
    [CrossRef] [PubMed] [Google Scholar]
  8. , , . Doing the math: A simple approach to topical timolol dosing for infantile hemangiomas. Pediatr Dermatol. 2018;35:276-7.
    [CrossRef] [PubMed] [Google Scholar]
  9. , , , , . Congenital, self-regressing tufted angioma. Arch Dermatol. 2006;142:749-51.
    [CrossRef] [PubMed] [Google Scholar]
  10. , , , , , . Topical timolol maleate for treatment of tufted angioma. J Dermatol. 2019;46:e402-3.
    [CrossRef] [Google Scholar]

Fulltext Views
4,412

PDF downloads
3,641
View/Download PDF
Download Citations
BibTeX
RIS
Show Sections