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:

Letter to the Editor - Case Letter
2016:82:4;442-444
doi: 10.4103/0378-6323.181461
PMID: 27279314

Multiple glomeruloid hemangiomas without POEMS syndrome

Vishal Gupta1 , Ajay Rai1 , Asit Ranjan Mridha2 , Vinod Kumar Sharma1
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:
Vinod Kumar Sharma
Department of Dermatology and Venereology, Teaching Block, All India Institute of Medical Sciences, Ansari Nagar, New Delhi - 110 029
India
How to cite this article:
Gupta V, Rai A, Mridha AR, Sharma VK. Multiple glomeruloid hemangiomas without POEMS syndrome. Indian J Dermatol Venereol Leprol 2016;82:442-444
Copyright: (C)2016 Indian Journal of Dermatology, Venereology, and Leprology

Sir,

Glomeruloid hemangioma is considered to be a specific cutaneous marker of POEMS (polyneuropathy, organomegaly, endocrinopathy, M-band, skin changes) syndrome. We report a case of multiple glomeruloid hemangiomas in a young man who had no features of POEMS syndrome. A brief review of literature of similar cases is also presented.

A 24-year-old man presented to us with a 5-year history of multiple, asymptomatic, erythematous papules on his neck. The lesions began as a small number of papules on the left side of the neck, gradually increased in size and number and progressed to involve the other side as well. There was no history of any spontaneous resolution, fluctuation in size or bleeding from the lesions. He had no systemic complaints. On examination, there were multiple soft, non-tender, partially compressible, erythematous papules ranging in size from 0.5 to 1.5 cm, clustered on the left side of the neck extending up to the submandibular region with fewer lesions on the right side. Two similar papules were also present on the right upper eyelid [Figure - 1]. There was no loco-regional lymphadenopathy. Skin biopsy from a neck papule showed several closely aggregated capillaries lined by plump endothelial cells intertwined with each other in a plexiform architecture. Variable sized, homogenous periodic acid–Schiff (PAS)-positive, diastase-resistant globules were seen in the cytoplasm of endothelial cells. The endothelial cells stained positively for CD34. The histological features were diagnostic of glomeruloid hemangioma [Figure - 2]. Results of complete blood count, serum biochemistry, fasting blood sugar and thyroid profile were within normal limits. There was no Bence-Jones proteinuria and the serum and protein electrophoresis did not show any M-band. No osteosclerotic lesions were noted on skeletal survey. A computerized tomography scan of chest and abdomen was normal. Nerve conduction studies were also normal.

Figure 1: (a) Multiple erythematous papules on the left side of neck extending to the mandible (b) few similar papules grouped on the right submandibular region (c) two papules on the lateral aspect of right upper eyelid
Figure 2: (a) Dilated capillaries in the dermis with aggregates of capillaries in ectatic vascular spaces (black arrow) (H and E, ×40). (b) The capillary tufts resemble renal glomeruli (black arrow) (H and E, ×200). (c) Immunohistochemistry with CD34 highlights the endothelial lining of capillaries (H and E, ×200)

Glomeruloid hemangioma is a rare benign cutaneous vascular neoplasm first described by Chan et al.[1] These may present as dermal erythematous to red-purple papules and nodules, subcutaneous bluish compressible nodules, wine-red sessile or pedunculated papules or lesions with cerebriform morphology.[2] The pathogenesis of glomeruloid hemangiomas is still a matter of speculation. Theories include deposition of immunoglobulins within endothelial cells, raised estrogen levels, role of human herpesvirus-8 and increased release of cytokines with angiogenic action such as interleukin-6, interleukin-1β, tumor necrosis factor-α and vascular endothelial growth factor.[3] Unlike their clinical presentation that is varied and non-descript, the histopathological appearance that resembles renal glomeruli is quite characteristic. The periodic acid–Schiff-positive, diastase-resistant eosinophilic globules in the cytoplasm of endothelial cells were initially thought to represent circulating immunoglobulins.[1] However, Lee et al. found that the interior of these eosinophilic globules did not stain positive for either kappa or lambda chains and hemoglobin A.[4] Electron microscopic examination suggested that these structures represent secondary lysosomes (thanatosomes) expanded by phagocytosed proteinaceous material.

A variety of cutaneous vascular tumors are described in the setting of POEMS syndrome: glomeruloid hemangiomas, microvenular hemangiomas, cherry angiomas and multinucleated cell angiohistiocytomas. However, only glomeruloid hemangiomas are considered specific, occurring in 26–44% cases.[1] In 2005, Vélez et al. reported the first case of solitary glomeruloid hemangioma without POEMS syndrome on the chin of an 86-year-old man.[5] Since then, only a handful of cases of glomeruloid hemangioma without this characteristic association have been described. On reviewing the literature, we could find only 11 other cases of cutaneous glomeruloid hemangiomas without an underlying POEMS syndrome.[4],[5],[6],[7],[8],[9],[10] Six of the eight patients, whose clinical information is available, had single-multiple lesions confined to the head and neck area while only two cases had multiple papules on the trunk [Table - 1]. Thus, it appears that the head and neck region, although considered an unusual site for glomeruloid hemangiomas, is more commonly affected in the absence of POEMS syndrome. This is in contrast to what is seen in POEMS syndrome where trunk and proximal extremities are predominantly affected.[3]

Table 1: Previously published cases of glomeruloid hemangiomas without POEMS syndrome

Since glomeruloid hemangiomas have been present in our patient in the absence of any other feature for 7 years (5 years before presentation and a 2 year follow up period), he is probably unlikely to develop POEMS syndrome. However, glomeruloid hemangiomas may rarely precede other manifestations by a long period, so we plan to keep our patient under follow-up.[1]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References
1.
Chan JK, Fletcher CD, Hicklin GA, Rosai J. Glomeruloid hemangioma. A distinctive cutaneous lesion of multicentric Castleman's disease associated with POEMS syndrome. Am J Surg Pathol 1990;14:1036-46.
[Google Scholar]
2.
Chan PT, Lee KC, Chong LY, Lo KK, Cheung YF. Glomeruloid haemangioma with cerebriform morphology in a patient with POEMS syndrome. Clin Exp Dermatol 2006;31:775-7.
[Google Scholar]
3.
Gherardi RK, Bélec L, Soubrier M, Malapert D, Zuber M, Viard JP, et al. Overproduction of proinflammatory cytokines imbalanced by their antagonists in POEMS syndrome. Blood 1996;87:1458-65.
[Google Scholar]
4.
Lee H, Meier FA, Ma CK, Ormsby AH, Lee MW. Eosinophilic globules in 3 case of glomeruloid hemangioma of the head and neck: A characteristic offering more evidence for thanatosomes with or without POEMS. Am J Dermatopathol 2008;30:539-44.
[Google Scholar]
5.
Vélez D, Delgado-Jiménez Y, Fraga J. Solitary glomeruloid haemangioma without POEMS syndrome. J Cutan Pathol 2005;32:449-52.
[Google Scholar]
6.
Piña-Oviedo S, López-Patiño S, Ortiz-Hidalgo C. Glomeruloid hemangiomas localized to the skin of the trunk with no clinical features of POEMS syndrome. Int J Dermatol 2006;45:1449-50.
[Google Scholar]
7.
Forman SB, Tyler WB, Ferringer TC, Elston DM. Glomeruloid hemangiomas without POEMS syndrome: Series of three cases. J Cutan Pathol 2007;34:956-7.
[Google Scholar]
8.
González-Guerra E, Haro MR, Fariña MC, MartÍn L, Manzarbeitia L, Requena L. Glomeruloid haemangioma is not always associated with POEMS syndrome. Clin Exp Dermatol 2009;34:800-3.
[Google Scholar]
9.
Jacobson-Dunlop E, Liu H, Simpson EL, White CR Jr., White KP. Glomeruloid hemangiomas in the absence of POEMS syndrome. J Cutan Pathol 2012;39:402-3.
[Google Scholar]
10.
Gupta J, Kandhari R, Ramesh V, Singh A. Glomeruloid hemangioma in normal individuals. Indian J Dermatol 2013;58:160.
[Google Scholar]

Fulltext Views
3,152

PDF downloads
1,780
Show Sections