Generic selectors
Exact matches only
Search in title
Search in content
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 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 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
Revision Corner
Self Assessment Programme
SEMINAR
Seminar: Chronic Arsenicosis in India
Seminar: HIV Infection
Short Communication
Short Communications
Short Report
Special Article
Specialty Interface
Studies
Study Letter
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
Tables
Technology
Therapeutic Guidelines
Therapeutic Guidelines - IADVL
Therapeutics
Therapy
Therapy Letter
View Point
Viewpoint
What’s new in Dermatology
View/Download PDF
CROSSMARK LOGO Buy Reprints
PDF

Translate this page into:

Case Report
ARTICLE IN PRESS
doi:
10.25259/IJDVL_909_20

Unusual cutaneous manifestations of dracunculiasis: Two rare case reports

Department of Dermatology, KEM Hospital, Mumbai, Maharashtra, India
MGM Medical College, Aurangabad, Mumbai, Maharashtra, India
UBM Institute and Dr. Bhatt Sonography Centre, Mumbai, Maharashtra, India
Corresponding author: Dr. Bhushan Amol Darkase, KEM Hospital, Mumbai - 400 012, Maharashtra, India. bhushandarkase@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: Darkase BA, Ratnaprkhi T, Bhatt K, Khopkar U. Unusual cutaneous manifestations of dracunculiasis: Two rare case reports. Indian J Dermatol Venereol Leprol, doi: 10.25259/IJDVL_909_20

Abstract

A nematode parasite, Dracunculus medinensis, causes dracunculiasis. Despite being non-fatal, this condition causes significant morbidity. Dracunculiasis is considered an eradicated disease in India since 1999. We report two cases that document the unusual linear morphea-like morphology of the calcified D. medinensis and the rare periorbital location of the worm. The cases presented here are rare and a diagnostic challenge, considering the eradicated status of dracunculiasis.

Keywords

Dracunculiasis
morphea
periorbital

Introduction

Dracunculiasis, also called guinea worm disease, is caused by Dracunculus medinensis which belongs to the nematode superfamily Dracunculoidea of the order Spirurida which are tissue parasites.1

In 1986, 20 Asian and African countries recorded 3.5 million cases. In 2002, 13 African countries reported >55,000 cases.2

The global incidence of new cases of dracunculiasis has decreased to 25 in 2016, mostly confined to three countries such as Chad, Sudan and Ethiopia.3

In India, the last reported case was in July 1996, though subsequently, three more cases were reported from parts of Rajasthan.4 The goal is global eradication of this disease by 2020.5

The disease is not fatal but its complications may cause considerable disability in acute and chronic stages.

The cases presented here show that though considered an eradicated disease, occasional guinea worm cases with unusual cutaneous manifestations may occur and require a high index of clinical suspicion.

Case Report

The first case was a 41-year-old female housewife, from Punjab complaining of of mild swelling, itching and discomfort around the right ankle for five years. There was no history of trauma or associated pain.

On examination, we noted a serpiginous swelling with intermittent break-up over the medial aspect of her right ankle and Achilles tendon. Overlying skin showed brownish-black hyperpigmentation with the pulled up appearance and puckering at a few places [Figure 1].

Figure 1:: A serpiginous swelling with intermittent break-up with brownish-black hyperpigmentation and the pulled-up appearance and puckering at few places

On palpation, along the lesional length, non-tender, thick, indurated cord-like swelling was appreciated. The patient denied biopsy but consented to a local ultrasound examination. Differentials considered were morphea, resolved thrombophlebitis and fibromatosis.

On ultrasound biomicroscopy [Figure 2] with a 50 MHz frequency probe, the dermis was hypoechoic in echo-texture and increased in thickness [red line]. A few small calcifications (purple arrow) were present in the superficial region of the subcutaneous tissue.

Figure 2:: The dermis is hypoechoic and is increased in thickness (red line). A few small calcifications (purple arrow) are noted in the superficial region of the subcutaneous tissue.

On high-frequency ultrasonography with 7–15 MHz multifrequency probes with color and spectral Doppler [Figure 3], we identified a worm having static linear elongated to curvilinear cord-like structure measuring 1.5–2 mm in diameter [red arrow]. No color flow/vascularity was present within this cord-like structure on color and spectral Doppler study. A thin linear anechoic region within the worm was observed possibly representing fluid in the worm’s gastrointestinal tract [Figure 4, blue arrow]. The above findings suggest a dead worm with inflammation of the skin and subcutaneous tissue around the ankle joint.

Figure 3:: A static linear cord-like structure, which is the worm, measures 1.5–2 mm in diameter (red arrow)
Figure 4:: A thin linear anechoic region within the worm is most probably fluid in the worm's gastrointestinal tract (blue arrow)

The clinical presentation, along with the findings on ultrasonography, pointed to the diagnosis of dracunculiasis. Unfortunately, this patient refused to undergo any surgical intervention and was lost to follow up for further management.

The second case was a 40-year-old female housewife, from Maharashtra, who presented with periorbital swelling and redness associated with itching and crawling like sensation in the skin for 15 days. There was no history of traveling, trauma or associated pain.

Examination showed an erythematous, non-indurated periorbital swelling with a worm-like serpiginous outline along the lateral canthus of the right eye [Figure 5, black arrow].

Figure 5:: Erythematous, edematous, inflamed non-indurated periorbital swelling with rope-like structure at (black arrow) lateral canthus

Ultrasound biomicroscopy of the skin and subcutaneous tissue in the lateral canthus and supraorbital region of the right eye was performed [Figure 6].

Figure 6:: There is a well-defined tubular hypoechoic lesion measuring 1.5 mm in diameter and 4 mm in length with a thin, linear, hyperechoic area within it, noted in the dermis and the subcutaneous tissue. This is the worm (green arrow). There is a well-defined, oval, hypoechoic lesion measuring 4 × 3.7 mm (yellow arrow) noted on the right side of the worm and surround the worm in the subcutaneous tissue.

We observed a well-defined, tubular, hypoechoic lesion measuring 1.5 mm in diameter and 4 mm in length with a thin, linear, hyperechoic area within it (the worm, green arrow) in the dermis and the subcutaneous tissue. The thin, hyperechoic line indicates the gastrointestinal system of the worm. We also noted a well-defined, oval, hypoechoic lesion measuring 4 × 3.7 mm (yellow arrow) noted on the right side of the worm and surrounding it in the subcutaneous tissue. The hypoechoic, tubular lesion with branching pattern in the subcutaneous tissue below the worm possibly occurred due to local tissue inflammation.

The patient consented to undergo surgical intervention for further confirmation. During surgery, a moving worm was identified and extracted by forceps [Figure 7].

Figure 7:: Extracted worm winding around the forceps

The extracted worm was identified as Dracunculiasis medinensis by the microbiologist.

Discussion

Dracunculiasis, also known as guinea worm disease, is a preventable water-borne parasitic disease. It is transmitted when people drink stagnant water contaminated with copepods that carry guinea worm larvae. Humans are the principal definitive host, and Cyclops is the intermediate host. A significant transmission route is drinking unsafe water containing small Cyclops infected with the larvae of D. medinensis.6 Transmission is limited among remote rural settings without a safe portable water supply.

Dracunculiasis was previously considered an exclusively a water-borne anthroponosis. Recent reports of infection by ingestion of paratenic (frogs) or transport (fish) hosts support that dracunculiasis could also be a food-borne zoonosis.5

The clinical features of dracunculiasis include mild fever, itchy rash, nausea, vomiting, diarrhea and dizziness. Nearly one year after infection, the female worm induces a blister on the skin, generally on the distal lower extremity, which ruptures. Acute stage complications include cellulitis, abscesses, septic shock and septic arthritis, while late-stage calcification of worm and joint deformities can occur.7

If the worm fails to reach the skin, it disintegrates or becomes calcified which becomes readily appreciable on the x-ray.8

In our first case, the calcified worm was present in subcutaneous tissue, whereas the second case presented with unusual periorbital location and mild cellulitis like features. The calcified worm may remain indolent or rarely causes intermittent mild discomfort and itching. Both of our cases presented with discomfort and itching.

Guinea worm calcification may take several forms, ranging from linear elongated to curvilinear to oval shapes. Muscle movement may break up the worm in several places, leading to elongated, nodular, beaded and fragmented appearance.8

The characteristic appearance is long linear, serpiginous or coiled, whorled “chain mail” type of calcification in the soft tissues, mostly in the lower extremity.7

In our first case, linear elongated to curvilinear worm was present in the dermis and subcutaneous tissue.

Multiple “rice grain” calcifications oriented along the direction of the muscle fibers are seen in cysticercosis.8 Loa loa and Onchocerca volvulus may calcify but show small, coiled masses of calcification and occasionally may be linear, but never as large or extensive as the guinea worm.7

The localization and the characteristic linear elongated and curvilinear appearance of worm with calcification on ultrasound in our patient were diagnostic of calcified guinea worm presenting as linear morphea-like lesion. In the second case, a live worm was extracted after surgical intervention.

Although dracunculiasis has been eradicated from several countries, it might present in an unusual location (periorbital) with acute skin manifestations. The remnant of disease can still be detected in some patients as a calcified worm. A high index of clinical suspicion is required to diagnose these cases of guinea worm disease.

Conclusion

Although dracunculiasis has been considered as an eradicated worm from several countries, unusual acute skin manifestations and the ghost of the disease in the form of a calcified worm can still be seen in some patients. A high index of clinical suspicion and collaboration with radiologists is required to diagnose these cases of guinea worm disease.

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. , , . Dracunculiasis (guinea worm disease) and the eradication initiative. Clin Microbiol Rev. 2002;15:223-4.
    [CrossRef] [PubMed] [Google Scholar]
  2. . Dracunculiasis (guinea worm disease) CMAJ. 2004;170:495-500.
    [Google Scholar]
  3. , , , , . Progress toward global eradication of dracunculiasis. MMWR Morb Mortal Wkly Rep. 2017;66:1327-31.
    [CrossRef] [PubMed] [Google Scholar]
  4. , , . Dracunculiasis in tribal region of southern Rajasthan, India: A case report. J Parasit Dis. 2010;34:94-6.
    [CrossRef] [PubMed] [Google Scholar]
  5. . Dracunculiasis: Water-borne anthroponosis vs. food-borne zoonosis. J Helminthol. 2019;94:e76.
    [CrossRef] [PubMed] [Google Scholar]
  6. , , . Dracunculiasis-the saddle is virtually ended. Parasitol Res. 2008;102:343-7.
    [CrossRef] [PubMed] [Google Scholar]
  7. . Dracunculiasis: Two cases with rare presentations. J Cutan Aesthet Surg. 2012;5:281-3.
    [CrossRef] [PubMed] [Google Scholar]
  8. . Serpentine calcification: A radiological stigma. J Neurosci Rural Pract. 2011;2:203-4.
    [CrossRef] [PubMed] [Google Scholar]
Show Sections