Human parvovirus B19 is the cause of several distinct clinical syndromes.1-3 The most common is erythema infectiosum, but there are also hematologic (transient aplastic crisis), rheumatologic (arthritis) and even fetal (hydrops/death) manifestations.3, 4 Routine diagnosis is performed with high sensitivity and specificity by immunoglobulin M detection in sera by enzyme immunoassays.5, 6 No specific therapy is known.
We present the case report of a child admitted to our hospital with a mononucleosis-like syndrome complicated by peripheral facial palsy and parotitis with intraparotid lymphadenitis, associated with parvovirus B19 infection.
Case report. A 16-month-old boy presented to our hospital with left peripheral facial palsy and parotitis. Family and personal histories were unremarkable except for the present illness. He had a 1-week history of nonproductive cough and fever (below 39°C). There was no clinical improvement with a 4-day course of amoxicillin/clavulanate therapy. His parent noticed in the hours before admission the onset of "masses" in both sides of the neck and facial asymmetry with crying. When seen for the first time at our hospital, the patient had fever, left peripheral facial palsy, multiple bilateral cervical, inguinal and axillary adenopathy (larger on left side), left parotid gland enlargement, hepatomegaly (6 cm below the costal margin), splenomegaly (3 cm below the costal margin), purulent tonsillopharyngeal exudate and bilateral acute otitis media. The clinical picture was interpreted as a mononucleosis-like syndrome complicated with parotitis and peripheral facial palsy, and therapy with ceftriaxone together with prednisone, ibuprofen and vitamin B12 was started.
The white blood cell count was 10.3 × 109/l with 40% neutrophils and 40% lymphocytes, and the rest of the blood count findings were normal. Monospot tests were negative on admission and 2 weeks later. Peripheral blood smear examination revealed no abnormalities. The other laboratory studies, including cerebrospinal fluid analysis; serum chemistry; coagulation; erythrocyte sedimentation rate; serum concentrations of IgG, IgM, IgA, IgE, C3 and C4 complement factors; rheumatic tests; and protein analysis were all negative or normal. Serial blood, cerebrospinal fluid, urine, tonsillopharyngeal exudate and stool cultures were negative.
Multiple bilateral enlarged lymph nodes of different sizes were detected by ultrasound examination not only in the cervical region but also in the intraparotid. Abdominal ultrasonography showed homogeneous liver and spleen enlargement. These findings were confirmed by cervical and abdominal computerized tomography. Electromyographic study of facial nerves showed lack of spontaneous activity and a 40% decrease in evoked motor response in the left facial nerve; electroneuronography confirmed these findings. Bilateral acute otitis media was corroborated otomicroscopically; the tympanocentesis samples were negative culture by. Evoked otoacoustic emissions were normal.
Acute parvovirus B19 infection was diagnosed in our patient by means of enzyme immunoassay; specific IgM antibody was positive whereas Ig G was negative (13 days later anti-B19 IgG was positive). Other serologic tests including Epstein-Barr virus, mumps, rubella, cytomegalovirus, Toxoplasma, herpesvirus, coxsackievirus (A9 to A21 and B1 to B6 serotypes), poliovirus, echovirus and HIV were negative. A mycobacterial origin was also excluded by sputum culture and polymerase chain reaction.
The lymph nodes progressively enlarged and the peripheral facial palsy became worse, showing a curious postural behavior; it worsened with standing and improved in the supine position. On the sixth day an itching, mainly centripetal erythematous maculopapular rash appeared. Initially the face, hands and feet were not involved, but 24 h later the eruption became generalized and confluent, encompassing face, hands, feet and even the palms and soles with mild concomitant acral edema. On palpation the affected skin was firm and had a rubbery feel, leaving after pressure a pale red to brownish residual color. Several petechial and purpuric lesions appeared, mainly on the hands and feet. The initial lesions disappeared in 6 to 7 days, leaving a transient residual hyperpigmentation, but new lesions continued to erupt for 2 weeks. Afterward the rash began to resolve distally, the face being the last location to be free of lesions on the 26th day. No mucous membrane changes were seen. The peripheral facial palsy resolved slowly by 40 days. One year later the physical examination is normal.
Discussion. Our case was initially interpreted as a heterophil-antibody-negative mononucleosis syndrome complicated by parotid and facial nerve involvement. We interpret the positive serologic testing for parvovirus B19 antibodies and the negative results for the remaining serologic studies and all the cultures performed as enough to establish parvovirus B19 as the cause, although quantitative enzyme-linked immunosorbent assay or serum polymerase chain reaction testing for parvovirus B19 were not performed in our patient.
Intraparotid lymphadenitis is an uncommon manifestation of infectious or neoplastic diseases.7, 8 In our case there was not only parotid enlargement but also intraparotid lymphadenitis. Tuberculosis and acquired immunodeficiency syndrome and the possibility of an underlying neoplasm were excluded in our patient.
The peripheral facial palsy in our patient was likely secondary to intraparotid facial nerve involvement, according to the clinical, electromyographic and electroneuronographic findings. We cannot ascertain whether it was caused by mechanical involvement of the nerve or to direct viral effect.
The rash in our patient involved hands and feet and even palms and soles with papular-purpuric lesions and mild acral edema which could resemble the "glove and stocking" distribution that has been related to parvovirus B199; nevertheless in our case the rash was generalized and no enanthem appeared throughout its evolution.
Parvovirus B19 is a widespread virus responsible for different clinical syndromes, most of them benign and self-limited. Neurologic complications are uncommon, although isolated cases of encephalitis, meningitis, plexitis, amyotrophy and motor paresis associated with parvovirus B19 infection have been reported.1-4
Federico Martinón-Torres, M.D.
María José Fernández Seara, M.D.
Ines Del Río Pastoriza, M.D.
Manuel Bravo Mata, M.D.
Manuel Castro-Gago, M.D.
Department of Pediatrics; Complejo Hospitalario Universitario de Santiago de Compostela (FMT, MJFS, IDRP, MBM)
Faculty of Medicine; University of Santiago de Compostela (MCG)
Santiago de Compostela, Spain
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