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
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
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
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
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
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
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:

Case Letter
ARTICLE IN PRESS
doi:
10.25259/IJDVL_932_2023

Unusual presentation of digital papillary adenocarcinoma in a sporotrichoid pattern

Department of Pathology, ICMR National Institute of Pathology, Safdarjung Hospital Campus, New Delhi, India
Department of Dermatology and Venereal diseases, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India

Corresponding author: Dr. Shruti Sharma, Department of Pathology, ICMR National Institute of Pathology, Safdarjung Hospital Campus, New Delhi, India. shrutigautam@rediffmail.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, transform, 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: Gupta A, Khullar G, Divyashree R, Sharma S. Unusual presentation of digital papillary adenocarcinoma in a sporotrichoid pattern. Indian J Dermatol Venereol Leprol. doi: 10.25259/IJDVL_932_2023

Dear Editor,

Digital papillary adenocarcinoma (DPAC) is a rare, slow-growing, malignant tumour of eccrine glands with a predilection for acral sites.1 It is known for its high recurrence rate and metastatic potential. Here, we report a unique presentation of DPAC as multiple cutaneous nodules and a verrucous plaque in a sporotrichoid distribution on the left upper limb, masquerading as an infectious condition.

A 62-year-old woman presented with a 6-year history of a gradually progressive asymptomatic plaque on the left forefinger and nodules on the left forearm. The lesions had increased in size in the last 6 months. Cutaneous examination revealed an ill-defined verrucous plaque, focally ulcerated, measuring 4 × 3 cm on the distal aspect of the left forefinger, causing nail dystrophy and flexion deformity, and two well-defined, firm, skin-coloured nodules measuring 1 × 1 cm, proximally on the extensor surface of the left forearm and elbow in a linear distribution [Figures 1a and 1b]. Lymph nodes were not palpable and systemic examination was unremarkable. Clinically, differential diagnoses of lymphocutaneous sporotrichosis, cutaneous leishmaniasis, atypical mycobacterial infection and tuberculosis verrucosa cutis were considered. Two punch biopsies were performed, one from the verrucous plaque and the other from the nodule on the forearm. Histopathological examination from both the lesions showed unremarkable epidermis with an underlying grenz zone separating it from a poorly circumscribed tumour filling the dermis. The tumour was composed of multiple, variably dilated and interconnected duct-like structures lined by two cell layers: inner lined by cuboidal to columnar cells and outer lined by myoepithelial cells. Focal areas of multi-layering and papillary infoldings, mild to moderate atypia and frequent mitotic figures were seen [Figures 2a and 2b]. Solid areas with back-to-back glands were also noted. Presence of solid areas ruled out the possibility of tubular papillary adenoma. Immunohistochemistry (IHC) revealed diffuse positivity for pan cytokeratin, CD15 and S-100 in the tumour cells and smooth muscle actin in myoepithelial cells. There was a high proliferative index of approximately 40% with Ki-67 [Figures 2c, 2d, 2e and 2f]. Histopathology images from the verrucous plaque also show similar morphology [Figure 3]. Tissue cultures for fungus, leishmaniasis and atypical and typical mycobacterial infection were all negative. Based on the clinical and histological findings, a final diagnosis of sporotrichoid DPAC was made. Routine haematological and biochemical investigations were unremarkable. Positron emission tomography scan did not reveal any metastasis. The patient was referred to the Department of Surgical Oncology, where her forefinger was amputated along with excision of both the nodules followed by radiotherapy. There was no evidence of local recurrence or distant metastasis after 1 year of follow-up and the patient is doing well.

Verrucous plaque on the forefinger showing focal ulceration and causing nail dystrophy distally.
Figure 1a:
Verrucous plaque on the forefinger showing focal ulceration and causing nail dystrophy distally.
Verrucous plaque on the forefinger and skin-coloured nodule on the forearm in a linear arrangement.
Figure 1b:
Verrucous plaque on the forefinger and skin-coloured nodule on the forearm in a linear arrangement.
Tumour is composed of multiple variably dialated and anastomosing duct like structures in the dermis (Haematoxylin and eosin, x40).
Figure 2a:
Tumour is composed of multiple variably dialated and anastomosing duct like structures in the dermis (Haematoxylin and eosin, x40).
Tumour composed of multiple variably dialated and anastomosing duct like structures in the dermis (Haematoxylin and eosin, x400).
Figure 2b:
Tumour composed of multiple variably dialated and anastomosing duct like structures in the dermis (Haematoxylin and eosin, x400).
Immunohistochemistry (IHC) reveals diffuse positivity for pancytokeratin in tumour cells IHC CK; ×100).
Figure 2c:
Immunohistochemistry (IHC) reveals diffuse positivity for pancytokeratin in tumour cells IHC CK; ×100).
Diffuse positivity for S-100 in tumour cells (IHCS100; ×100).
Figure 2d:
Diffuse positivity for S-100 in tumour cells (IHCS100; ×100).
Smooth muscle actin positivity in myoepithelial cells (IHC-SMA; ×200).
Figure 2e:
Smooth muscle actin positivity in myoepithelial cells (IHC-SMA; ×200).
Tumour cells showing positivity for Ki 67 (IHC-Ki67; ×100).
Figure 2f:
Tumour cells showing positivity for Ki 67 (IHC-Ki67; ×100).
Histopathology image from verrucous plaque showing marked hyperkeratosis and parakeratosis. Entire dermis is infiltrated by a tumour composed of ducts and solid areas. (H&E x40). Inset show tumour in high magnification (x400).
Figure 3:
Histopathology image from verrucous plaque showing marked hyperkeratosis and parakeratosis. Entire dermis is infiltrated by a tumour composed of ducts and solid areas. (H&E x40). Inset show tumour in high magnification (x400).

In the past, digital papillary adenoma and adenocarcinoma were considered as separate entities, but neither the clinical features nor the histomorphology predicted the incidence of recurrences or metastasis. Hence, in 2006, WHO classified both the terminologies under a single entity of DPAC, in the absence of any benign counterpart.2 DPAC presents as a solitary mass that frequently involves the distal aspect of the finger/thumb or palm and sole. Our case is unique in terms of multiple lesions of the upper limb. The clinical differential diagnoses include digital mucous cyst, foreign body granuloma, paronychia, benign adnexal tumour and vascular tumour-like pyogenic granuloma and glomus tumour. Histologically, it may resemble sweat gland tumours like spiradenocarcinoma or metastatic adenocarcinoma from colon, thyroid or breast.3

Surgical management options for DPAC include wide excision, digital amputation, partial digital amputation or Mohs micrographic surgery.4 Long-term follow-up is important for the early detection of local recurrence and metastasis to lymph nodes and lungs.

The present case was unique since sporotrichoid pattern of involvement in DPAC mimicking an infectious aetiology has not been described in literature. Besides infections, rarely cutaneous tumours such as metastatic cutaneous squamous cell carcinoma, lymphomas, melanoma, epithelioid sarcoma, peripheral nerve sheath tumour and keratoacanthomas have been reported to present in a sporotrichoid fashion.5 Digital papillary adenocarcinoma is known for local recurrence and distant metastasis, mostly to the lungs, owing to the haematogenous spread. However, lymph node involvement is also described in literature explaining the lymphatic spread. Hence, it appears to be spread via both haematogenous and lymphatic routes. Lymphatic spread appears to be the most probable explanation in our case.

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.

Use of artificial intelligence (AI)-assisted technology for manuscript preparation

The authors confirm that there was no use of artificial intelligence (AI)-assisted technology for assisting in the writing or editing of the manuscript and no images were manipulated using AI.

References

  1. , , . Aggressive digital papillary adenocarcinoma (aggressive digital papillary adenoma and adenocarcinoma revisited) Am J Surg Pathol. 2000;24:775-84.
    [CrossRef] [PubMed] [Google Scholar]
  2. , , , . World Health Organization classification of tumours: Pathology and genetics of skin tumours. Lyon: IARC Press; . p. :133-4.
  3. , , , , , . Aggressive digital papillary adenocarcinoma: A report of two diseases and review of the literature. J Am Acad Dermatol. 2009;60:331-9.
    [CrossRef] [PubMed] [Google Scholar]
  4. , , , , , . Aggressive digital papillary adenocarcinoma with multiple organ metastases: A case report and review of the literature. Am J Dermatopathol. 2016;38:910-14.
    [CrossRef] [PubMed] [Google Scholar]
  5. , . Sporotrichoid keratoacanthomas: Case report and review of neoplasms presenting in a sporotrichoid pattern. Cureus. 2018;10:3196.
    [Google Scholar]

Fulltext Views
2,356

PDF downloads
1,950
View/Download PDF
Download Citations
BibTeX
RIS
Show Sections