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strongyloides stercoralis hyperinfection mimicking systemic lupus erythematosus flare.

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Strongyloides stercoralis hyperinfection mimicking
systemic lupus erythematosus flare
To the Editor:
Strongyloides stercoralis is an intestinal nematode
that usually causes a mild or asymptomatic gastrointestinal
illness. In an immunocompromised host, such as a patient
with corticosteroid-treated systemic lupus erythematosus
(SLE), dissemination of the parasite may occur and may
present as an acute fulminant systemic disease called hyperinfection (1,2). The case report presented here demonstrates how coexistence and similar manifestations of the
diseases may lead to a delay in detection and treatment of
strongyloidiasis, resulting in death.
The patient was a 43-year old Hispanic woman. Four
months prior to admission, she developed pain in her shoulders and hands and a malar rash. Fluorescent antinuclear
antibodies and anti-DNA antibodies were elevated, and the
hemolytic complement level was low. Her urine contained
red blood cell casts and protein. The patient was diagnosed
as having SLE, and treatment with prednisone was initiated.
Two weeks prior to admission, the patient developed
fever, myalgias, arthralgias, malar and generalized rashes,
and peripheral edema. Findings on admission were compatible with an SLE flare and included, in addition to the fever,
arthritis, skin rash, and diffuse proliferative glomerulonephritis (by biopsy) with nephrotic syndrome and pericarditis
(on echocardiogram). The complement level was 0, but the
anti-DNA antibody level was normal. Blood and urine
cultures gave negative results. The hospital course was
complicated by the development of anemia, thrombocytopenia, Enterobacter cloacae sepsis, and an episode of acute
pulmonary edema. The patient was treated with corticosteroids, antibiotics, and diuretics. She improved markedly and
was discharged 1 month after admission.
The patient was readmitted to the hospital 2 days
later because of severe abdominal pain, temperature of
38.9"C, and a pruritic macular rash. Her abdomen was
tender and distended, with normal bowel sounds and without
signs of peritoneal irritation. The white blood cell (WBC)
count was 13,00O/pI with left shift. The urine showed 4 +
protein and blood. The CH50 level was 75 units (normal
150-250), and anti-DNA antibodies were negative. Urine
cultures grew Klebsiella. Results of blood cultures, amylase
studies, sigmoidoscopy, computed tomography scan of the
abdomen and pelvis, peritoneal lavage, and angiogram of the
superior mesenteric axis were negative or normal. Ampicillin and gentamicin were given and the steroid dosage was
increased, without clinical improvement.
A week after the second admission, the patient
developed acute shortness of breath, necessitating mechanical ventilation. A chest radiograph showed bilateral alveolar
infiltrates. An abdominal radiograph revealed ileus. Catheterization of the pulmonary artery showed a normal capillary
wedge pressure. The clinical picture and study results were
compatible with adult respiratory distress syndrome. An
open lung biopsy showed marked hyaline membrane formation, lymphoplasmocytic infiltration in the interstitium, and
Arthritis and Rheumatism, Vol. 31, No. 7 (July 1988)
focal edema. Stains for organisms gave negative findings.
The patient's condition deteriorated further and she became
comatose, with signs of meningeal irritation. The spinal fluid
showed 10,985 WBC/pI with 83% polymorphonuclear cells
and 16% lymphocytes. Results of stains for organisms and
cultures were negative.
At that time the patient developed diarrhea. Stool
examinations were performed, revealing S stercorulis. The
parasite was found also in the sputum and, on reexamination, in the lung biopsy specimen (Figure 1). Thiabendazole
was added to the treatment regimen and given until stool and
sputum cultures became negative. Despite this treatment,
the patient remained febrile, obtunded, and respiratordependent. She died a month after her second admission.
Autopsy revealed acute confluent bronchopneumonia, nonbacterial verrucous endocarditis, infected thrombi in the
ventricles, and diffuse proliferative glomerulonephritis. S
stercoralis was not found in the lung or other organs.
Early in the course of this patient's disease, a diagnosis of SLE had been made based on symptoms and
findings that met the American Rheumatism Association
revised criteria for diagnosis of the disease (3). Despite
treatment with steroids, multiorgan involvement developed
sequentially, in a manner consistent with a lupus flare. The
Figure 1. Biopsy sample of the patient's lung. An adult Strongyloides stercoralis worm is seen (hematoxylin and eosin stained,
original magnification X 630).
93 1
manifestations were therefore attributed to SLE activity that
was inadequately controlled by steroid treatment. Retrospectively, even though the patient had active SLE, the
strongyloidiasis superinfection could have accounted for
most of the clinical picture. Strongyloides is not endemic in
the northern US. It is conceivable that the patient acquired
the parasite in Puerto Rico more than 30 years prior to the
hospital admission, when she immigrated. Prolonged survival of the parasite in the bowel of a carrier has been
reported (4) and is partly due to autoinfection cycles. The
dissemination of the parasite was probably secondary to the
steroid treatment, as has been described (43).
Many of the manifestations that occurred in our
patient have been described in reports of the hyperinfection
syndrome. Skin manifestations include a pruritic erythematous rash and migratory urticarial lesions. Severe abdominal
pain is accompanied by nausea, vomiting, diarrhea or constipation, and features of ileus (5). Lung involvement may
present as asthma, lung opacities, and diffuse or focal
infiltrates in a patient experiencing pulmonary failure. Central nervous system disease presents as mental status
changes, with confusion, agitation, obtundation, coma, meningeal irritation, and pleocytosis and elevated protein in the
spinal fluid (6). Secondary septicemia and individual organ
infestation (4) may each account for the clinical picture.
In our patient, the diagnosis was based on isolation
of the parasite from the sputum and stool. Usually, isolation
of the organism in stool concentrates, jejunal aspirates, and
a duodenal biopsy is required for accurate diagnosis (5). A
diagnosis of Strongyloides hyperinfection based on sputum
examination has been described (7). Eosinophilia, a marker
of parasitic infestation, was absent in our patient and is
absent frequently in Strongyloides hyperinfection in patients
receiving corticosteroids (43). A delay in the diagnosis
frequently results in mortality, despite intensive treatment
(1,2). in this patient, the delay was due to the similarity of
the manifestations of hyperinfection and SLE. Another case
of Strongytoides hyperinfection masquerading as SLE cerebral vasculitis has been described (6). Otherwise, Strongyloides hyperinfection complicating SLE has been reported
only rarely (1,6,7). We could not find any other published
case in the English-language rheumatologic literature.
Thus, strongyloidiasis, like hypernephroma, porphyria, and leprosy, is an entity that, under certain circumstances, may mimic SLE. When initiation of corticosteroid
therapy in an SLE patient is followed sequentially by abdominal pain, respiratory failure, septicemia, and cerebral
disease, the physician should consider the possibility of
disseminated strongyloidiasis, in addition to evaluating the
need for more aggressive therapy for lupus.
Avi Livneh, MD
Eugene A. Coman, MD
Sangho Cho, MD
Esther Lipstein-Kresch, MD
Albert Einstein College of Medicine
Bronx. N Y
1. Rivera E, Maldonado N, Velez-Garcia E, Grillo AJ, Malaret G:
Hyperinfection syndrome with Strongyloides stercoralis. Ann
Intern Med 72:199-204, 1970
2. Purtile DT, Meyers WM, Connor DH: Fatal strongyloidiasis in
immunosuppressed patients. Am J Med 56:488-493, 1974
3. Tan EM, Cohen AS, Fries JF, Masi AT, McShane DJ, Rothfield
NF, Schaller JG, Tala1 N, Winchester RJ: The 1982 revised
criteria for the classification of systemic lupus erythematosus.
Arthritis Rheum 25:1271-1277, 1982
4. Igra-Siegman Y, Kapila R, Sen P, Kaminski ZC, Louria DB:
Syndrome of hyperinfection with Strongyloides stercoralis. Rev
Infect Dis 3:397-407, 1981
5. Scowden EB, Schafher W , Stone WJ: Overwhelming strongyloidiasis and unappreciated opportunistic infection. Medicine
(Baltimore) 57527-544, 1978
6. Wachter RM, Burke AM, MacGregor RR: Strongyloidiasis stercoralis hyperinfection masquerading as cerebral vasculitis. Arch
Neurol41:1213-1216, 1984
7. Beger R, Kraman S, Paciotti M: Pulmonary strongyloidiasis
complicating therapy with corticosteroids: report of a case with
secondary bacterial infection. Am J Trop Med Hyg 29:31-34,
Acute periarthritis in a patient with infectious
To the Editor:
Acute arthritis is a very unusual complication of
infectious mononucleosis. In 1983, Sigal et al (1) reported a
case and reviewed the 3 previously reported cases. I am
aware of one subsequent report of a patient with an acute
polyarthritis and infectious mononucleosis (2), as well as a
report of 4 additional patients with acute polyarthritis and
evidence of acute or continuing infection by Epstein-Barr
virus (EBV) (3). Interestingly, in only 2 of these 9 previously
reported cases were heterophil antibodies detected. I describe here a patient with acute periarthritis of the ankle in
whom heterophil-positive infectious mononucleosis was
subsequently diagnosed.
The patient, an 18-year-old woman, developed a
fever and pain in her left ankle in February 1987. She had
been completely well prior to the onset of the ankle pain and
fever, and she denied having any symptoms suggestive of a
specific rheumatic disease. She was sexually active, with
one steady partner, and denied having any symptoms suggestive of a diagnosis of gonorrhea. She stated that she had
experienced mild anorexia during the preceding 2 weeks, but
she had not had a sore throat.
At physical examination, her oral temperature was
40°C. There was no icterus, lymphadenopathy, hepatomegaly, or splenomegaly. Marked pitting edema of the left foot
and ankle, ascending the left leg to just below the knee, was
noted. There was erythema and tenderness over the medial
malleolus, which extended over the entire ankle area, although to a milder degree. The ankle was fixed in 15" of
plantar flexion, and she was unable to move the ankle
because of pain. A drop of blood was aspirated from the
ankle joint and sent to the laboratory for culture. Because a
diagnosis of gonorrhea was suspected and there was a
history of a possible allergy to penicillin, treatment was
begun with cefotaxime sodium, given intravenously.
Laboratory findings at that time included a white
blood cell (WBC) count of 7.8 x 109/liter. Automated
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lupus, mimicking, flare, strongyloides, hyperinfection, systemic, erythematosus, stercoralis
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