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Rheumatic syndromes secondary to guinea worm infestation.

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BRIEF REPORT
RHEUMATIC SYNDROMES SECONDARY TO GUINEA WORM
INFESTATION
GEOKGE E. McLAUGHLIN. PETER D. UTSINGER. WILLIAM F. TRAKAT, DONALD RESNICK,
and ROBERT A. MOIDEL
Dracunculosis, also known as dracontiasis or
dracunculiasis, is an endemic parasitic infection
caused by the nematode (round worm) Dracunculus
medinensis or guinea worm. An obligate parasite of
man, the organism may have been recognized in
biblical times as the “fiery serpent” which afflicted the
Israelites and was described in the Old Testament,
Numbers 21. Dmcunculus mediriensis has recently
been identified radiographically in a mummy almost
3,000 years old (1). Although dracunculosis is considered to be geographically restricted to southeastern
Asia, northern Africa, and the eastern Mediterranean
countries, reports indicate that up to 50 million people
may be affected with this disorder (2). We recently
studied a patient with rheumatic complaints and
From the Departments of Rheumatic Disease, Medicine.
‘and Immunology, Temple University. Philadelphia, Pennsylvania;
the Department of Medicine, North Penn Hospital, Lansdale.
Pennsylvania; and the Department of Radiology, University of
California, San Diego.
Support for this publication was provided by the Arthritis
Research Fund. Germantown Hospital and Medical Center, Philadelphia, Pennsylvania.
George E. McLaughlin, MD, FACP: Chief. Department of
Rheumatic Disease, Germantown Hospital and Medical Center.
Clinical Associate Professor of Medicine, Temple University; Peter
D. Utsinger, MD: Chief of Research and Director of the Department
of Immunology, Germantown Hospital and Medical Center, Professor of Clinical Medicine, Temple University; William F. Trakat,
DO: North Penn Hospital: Donald Resnick, MD: Professor of
Radiology. University of California, San Diego: Robert A. Moldel,
MD: Department of Rheumatic Disease, Germantown Hospital and
Medical Center.
Address reprint requests to Peter D. Utsinger, MI>, Irnrnunology Laboratory, Germantown Hospital and Medical Center, Two
Penn Boulevard, Philadelphia, PA 19144.
Submitted for publication March 29. 1983; accepted in
revised form January 13, 1984.
Arthritis and Rheumatism, Vol. 27, So. 6 (June 1984)
unique radiographic findings secondary to dracunculosis. This report, emphasizing these findings and the
rheumatic syndromes associated with dracunculosis,
serves to remind the clinician that in this era of modern
travel, diseases endemic to one geographic area may
be seen far afield (3,4).
Case report. A 33-year-old Indian woman came
to one of us (PDU) with the chief complaint of
intermittent mild pain in the dorsal aspect of both feet
and ankles of approximately 6 months duration and
difficulty with hand grasp because of pain and stiffness
in the fingers. She was born and raised in the Madras
area of southeastern India but has lived in the United
States for 20 years. She denied symptoms suggestive
of dracunculosis in the past, and her medical history
was completely unremarkable.
On physical examination, small (2-3 mm), firm
subcutaneous nodules were palpated overlying the
dorsal aspect of the proximal interphalangeal joints. A
larger mass, measuring 0.8 x 0.5 cm was noted
overlying the dorsolateral aspect of the left fifth metacarpophalangeal joint. A discrete, linear firm mass
overlying the dorsal aspect of each ankle was evident
on palpation. These lesions were nontender. There
was no evidence of any synovitis in the involved
joints, nor evidence of joint effusion.
Laboratory results, including a complete blood
count, sedimentation rate (Westergren), multichannel
chemistry, antinuclear factor, and rheumatoid factor
were all within normal limits or negative.
Radiographs demonstrated a linear stippled calcific density in the subcutaneous tissues overlying
both ankles (Figure 1). Hand radiographs (Figure 2)
revealed fluffy periarticular densities overlying both
the metacarpophalangeal and proximal interphalangeal
BRIEF REPORTS
695
Figure 1. Linear stippled calcific dehsity in the subcutaneous tissues overlying the ankle.
joints, but sparing the distal interphalangeal joints. A
larger, dense calcific deposit was noted overlying the
left fifth metacarpophalangeal joint (Figure 3). Articu-
Figure 2. Fluffy penarticular densities overlying the metacarpophalangeal and proximal interphalangeal joints.
lar surfaces were normal except for a few unexplained
cystic lesions in the distal aspect of the left third
proximal phalanx. Nematode infection was suspected.
Further evidence supporting this diagnosis came from
radiographic interpretation by one of the authors. It
was believed the lesions represented calcified Dracunculus medinensis.
Therapy with nonsteroidal antiinflammatory drugs provided adequate relief of the patient’s
symptoms.
Discussion. Dracunculus mrdinensis is ingested
by drinking water containing the microcrustacean Cyclops, which serves as the intermediate host for dracunculosis. Larvae, freed from Cyclops in the stomach
of humans, migrate through the intestinal wall and
incubate in the abdominal wall for up to 12 months.
The adult female, which may reach a length of 1 meter,
then migrates, usually to gravitationally dependent
areas, where she seeks the skin surface to extrude her
larvae and restart the cycle (5). Little is known about
the fate of the adult male (which measures 30-40 m m
in length). Cohen’s report of a “calcified male worm”
probably represented loaisis ( 6 ) . Should the female
worm die before reaching the surface, her calcified
remains may provide the only evidence of dracunculosis, as was probably the case in our patient. Radiographs showing a linear subcutaneous stippled calcific
density marking the remnant of the body of the dead
female are probably pathognomonic of dracunculosis
(798)*
Symptoms secondary to dracunculosis are pro-
BRIEF REPORTS
696
tean, predicatcd upon the migratory pattern of the
female. Pleurisy (9), pancreatitis (lo), spinal cord
compression ( 1 1,12), and inguinal adenopathy (13)
have been described. Dracunculosis is believed to be a
cause of placental bleeding in pregnancy and subsequent abortion (14). Khajavi noted over I5 malignant
neoplasms, most of which were bladder tumors proximate to the calcified worm in a study of 83 drdcunculosis patients (15).
The rheumatic manifestations of dracunculosis
are outlined in Table 1 . Periarticular deposits in subcu-
Figure 3. Large, dense calcific deposit overlying the fifth metacarpophalangeal joint.
Table 1.
Rheumatic manifestations of Dracunculus infestation
Acute monarthritis secondary to direct invasion by female
worm
a. Secondary mixed bacterial infection
2. Arthralgic syndromes secondary to penarticular calcifications
a. “Sympathetic effusions”
b. Localized periarthralgic syndromes
1.
taneous tissues (as in our patient) may result in joint
pain or even “sympathetic” effusions (16,17). The
fluffy periarticular radiologic densities (Figure 2) may
represent remains of the elusive male worm, but this is
conjectural. Direct invasion of large joints (particularly the knees) may result in an intense destructive
arthropathy (4,18-20), and the joint may become secondarily infected. Cartilage destruction and flexion
contractures are frequent sequelae of this aspect of
dracunculosis-induced arthritis. Synovial fluid has
been described as turbid, yellow, and highly inflammatory (18-20), but precise details are lacking.
An immunologic mechanism seems unlikely as
an etiologic factor in our patient, in view of the lack of
evidence of acute inflammation clinically and serologically, and the long latent period (20 years) before the
onset of symptoms.
The incidence of rheumatologic manifestations
or neoplasms in Dracunculus infestation is not known
at present; these case reports (4,15-20) are anecdotal,
and epidemiologic studies have not evaluated these
facets of Dracunculus infection.
The effect of dracunculosis on the economy of
endemic areas is difficult to assess accurately, but up
to fifty million people may be affected annually (21,
particularly at the time of planting and harvesting
(21,22), which suggests a major economic impact. The
World Health Organization has designated 1981-1990
as the “International Drinking Water and Sanitation
Decade” in an attempt to eliminatc water-transmitted
diseases.
In spite of the international significance of
dracunculosis, little recognition of rheumatic manifestations of parasitic infection is evident in the American
literature (23), and neithcr of 2 leading rheumatology
textbooks makes note of the joint manifestations
(24,25). Hopefully this report will serve to emphasize
the incidence and significance of this form of parasitic
infestation, and alert physicians to the consideration of
dracunculosis in the differential diagnosis of polyarthralgia and arthritis.
BRIEF REPORTS
Acknowledgments. The authors are grateful to Ms
Kathleen Leigh and Ms Christine McDermond who assisted
in the preparation of this manuscript.
REFERENCES
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Parasitology. Ninth edition. New York, Academic
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Exot Filiales 71:85-89, 1978
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