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Cellulitis Case Tx737

Cellulitis Case Tx737

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Published by: Sarah Beth Breck on Aug 25, 2011
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Human Facial Cowpox Obtained from a Cat
CID 2006:43 (15 September)
737
R E V I E W A R T I C L E
A Case of Facial Cellulitis and NecrotizingLymphadenitis due to Cowpox Virus Infection
Roland Pahlitzsch,
1
Anna-Lena Hammarin,
2
and Anders Widell
3
1
Department of Otolaryngology, Blekinge County Hospital, Karlskrona,
2
Department of Virology, Swedish Institute of Infectious Disease Control,Stockholm, and
3
Department of Medical Microbiology, Malmo¨ University Hospital, Lund University, Malmo¨, Sweden
We describe a patient with facial cellulitis/erysipelas due to cowpox virus inoculation in the respiratory epithelium of the nose. A cytopathic agent was isolated in cell culture, and the diagnosis of cowpox wasconfirmed by electron microscopy and polymerase chain reaction. The most likely source of infection wasexposure to the family cats. In addition to the severe edematous cellulitis of the face, the clinical course wasdominated by several areas of subcutaneous, necrotizing lymphadenitis, from one of which a huge abscessformed that had to be incised. Hyperbaric oxygen treatment was provided to prevent development of dermalnecrosis. The healing process in the numerous areas of lymphadenitis was markedly protracted,and1persisting node (which yielded positive results on polymerase chain reaction) had to be excised 2 years after onset of disease. This is the first reported case of inoculation of cowpox virus in the respiratory mucosa of the nose.It resulted in a clinical course totally different than that for inoculation in the skin. We also present a shortreview of findings on orthopoxvirus infection that focuses on the chain of transmission.
Facial infection of the skin and soft tissues can be dueto a number of bacterial agents, but it can also be dueto viruses such as herpes simplex virus type 1, varicellazoster virus, and a range of poxviruses. Formerly, va-riola virus had humans as its exclusive host, but withits eradication through mass vaccination, the totalnumber of poxvirus infections has been dramatically reduced. However, cases of zoonotic poxvirus infectionhave occasionally involved humans. Such cases may present diagnostic difficulties and may have devastatingoutcomes [1]. We report a case of cowpox infectionwith a prolonged disease course, and we discuss theviral, diagnostic, epidemiological, and therapeuticaspects.
CASE REPORT
In early November 2000, a 7-year-old girl living in thesoutheast of Sweden sought medical attention from a
Received 28 February 2006; accepted 23 May 2006; electronically published 10August 2006.Reprints or correspondence: Dr. Roland Pahlitzsch, Dept. of Otolaryngology,Blekinge County Hospital, S-37185, Karlskrona, Sweden (roland.pahlitzsch@ltblekinge.se).
Clinical Infectious Diseases 2006;43:737–42
2006 by the Infectious Diseases Society of America. All rights reserved.1058-4838/2006/4306-0012$15.00
general practitioner. She presented with an inflamma-tory, erythematous swelling of the skin beneath theright ear and painless lymphadenopathy in the regionalparotid and upper jugular lymph nodes. She had nohistory of skin injury in the affected region of the headand neck. Two weeks before the onset of symptoms,the patient had visited Spain. She had received child-hood vaccinations against mumps, rubella,measles,po-liomyelitis,
Haemophilus influenzae,
and diphtheria-tet-anus-pertussis, but she had not received vaccinationagainst tuberculosis or smallpox. There was no history of immunodeficiency or atopia.The patient had a low-grade fever (temperature,
38
C) and general malaise, and because a bacterialinfection was suspected, she was treated orally withpenicillin. Despite the administration of treatment, thesymptoms progressed, and the patient was referred tothe Department of Otolaryngology at Blekinge County Hospital (Karlskrona, Sweden) on 26 November 2000.Three weeks after the onset of the first symptoms, hercondition worsened. Extensive edema developed in theupper part of the right side of the neck and on theright side of the face, extending into the contralateralside and involving the eyelids; it also developed on themental region and the upper part of the right side of the neck (figure 1
A
and 1
).
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738
CID 2006:43 (15 September)
Pahlitzsch et al.
Figure 1.
Serial photographs and a CT scan for a patient with facial cowpoxvirus infection.
A,
Lesion and facial edema 2 weeks after onset ofinfection.
B,
CT obtained 2 weeks after onset.
C,
Abscess prior to hyperbaric oxygen treatment and surgical incision 6 weeks after onset.
D,
Status4 months after hyperbaric oxygen treatment and incision.
E,
Close-up of long-lasting lesion in medial canthus 24 month after onset.
F,
Late results,36 months after onset, showing a minimum of scarring.
G,
Electron microscopic image of orthopoxvirus obtained from our patient. Consent to appearin the photographs was obtained from the patient’s guardian.
Physical examination was performed at the time of hospitaladmission and revealed a whitish, necrotizing mass in the rightanterior nasal cavity that was surrounded by granulations atthe anterior border of the lower nasal concha. Numerous solidlymph nodes (diameter, 3–25 mm) were palpable in the rightcheek, near the right nostril, near the right eye (including theupper eyelid), and more laterally, anterior to the border of theright parotid gland.The WBC count ( cells/L) was nearly normal (nor-
9
11.3
10mal WBC count,
!
cells/L). Other laboratory values
9
9.6
10were slightly elevated: the C-reactive protein level was 14 mg/mL (normal level,
!
5 mg/mL), the serum alkaline phosphataselevel was 7.9
m
kat/L (normal range, 1.0–4.5
m
kat/L), and thelactate dehydrogenase level was 9.3
m
kat/L (normal range, 3.0–7.0
m
kat/L). Plasma electrophoresis revealed a slightly raisedlevel of IgG3 (1.4 g/L; reference values, 0.13–1.05 g/L), but thefindings were otherwise normal.
Bacteriologic study findings.
A nasal swab culture yielded
Streptococcus mitis 
(low numbers),
Haemophilus parainfluenzae 
(low numbers), and
Neisseria 
species, including 1 oxidase pos-itive rod-like bacterium. Samples were obtained for
Mycobac-terium tuberculosis 
culture, and the results were negative.The overall interpretation was that the local skin infectionwas a phlegmon caused by 
H. influenzae 
(although culture re-sults were negative). Treatment with intravenous cefuroximewas started on a tentative basis.
Histologic study findings.
Biopsy from the nose mass re-vealed necrotic material with granulocytes.
Radiographic study findings.
CT revealed deep edema of the right cheek, the right parotid gland, and the right side of the neck. Edema of the respiratory epithelium of the anteriorethmoidal cells and the nasal concha on the right side couldalso be visualized (figure 1
B
).
Subsequent clinical course.
One week after the acute ex-acerbation a slight improvement of the cellulitis under the eyeoccurred. Around the areas of facial lymphadenitis the skin stillshowed a bluish discoloration. However, since the patient’sgeneral condition improved, she was discharged from the hos-pital and followed up on weekly visits to our outpatient clinic.The cellulitis completely resolved within 3 weeks after onset,
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Human Facial Cowpox Obtained from a Cat
CID 2006:43 (15 September)
739
but the lymphadenitis in the buccal and periorbital areas de-veloped into subcutaneous abscess cavities, which ranged insize from 3 to 40 mm. The largest of these abscess cavitiesmeasured mm and extended from the paranasal region40
25into the right cheek (figure 1
). The subcutaneous fat layer atthe site of the abscess cavity underwent a complete necrosis.Repeated needle punctures produced a whitish, smeary dis-charge, a specimen of which was sent for general and specificbacterial culture and, subsequently, for virus isolation on 19December 2000.Approximately 6 weeks after the onset of symptoms, thedermis in the region of the paranasal abscess became extremely thin and was at risk of necrosis and perforation. Three punc-tuations, which were made under general anesthesia during aninterval of 3–5 days, had no effect on the condition, and in-cision became inevitable. Because of the risk of dermalnecrosis,hyperbaric oxygen (HBO) treatment was chosen as therapy, toimprove the chances that the infection would resolve and tostabilize the atrophied skin before incision. The patient un-derwent 5 courses of HBO treatments, in 6 hourly intervals,between the 21st and the 23rd of December in the pressurechamber at the Karlskrona Naval Base.After the first course of HBO treatment, the huge paranasalabscess was evacuated by incision, and a drain was inserted.The discharge diminished gradually, and the drainwasremovedafter 5 days. The patient’s condition quickly improved, and 6weeks after she underwent HBO therapy and the incision, theskin had recovered completely, including a reappearance of thesubcutaneous fat layer. However, some lymph nodes continuedto vary in size, and the bluish discoloration of the adjacent skinareas remained. Nodes of 2–3 mm in diameter in generalshowed spontaneous regression, but nodes with a diameter of 5–15 mm required needle aspirations several times, for up to5 months after the onset of the disease. Histologic examinationof the discharge showed degeneratedhyperplasticunidentifiablecells. Examination of fine-needle biopsy specimens from theupper jugular lymph nodes, which never necrotized, revealedreactive lymphadenitis.The aspirate obtained on 19 December was subjected tovirusisolation at the Department of Virology at Malmo¨ University Hospital (Malmo¨, Sweden). Ten days after inoculation ongreen-monkey kidney cells, a cytopathic effect (CPE) appeared,which, on subpassage, yielded syncytial formation of the cells.The CPE was caused by an agent that was filterable through a0.45-
m
m filter. The results of PCR and immunofluorescencetesting for mumps virus were negative.The CPE pattern directed our suspicions to poxvirus infec-tion, and the culture material was sent to the Swedish Institutefor Infectious Disease Control in Stockholmon9February2001for investigation for poxvirus. Orthopoxvirus-like particleswere detected in the culture medium by electron microscopy (figure 1
). The diagnosis was further confirmed by PCR anal- ysis, which noted a 339-bp fragment of the viral thymidinekinase gene, where direct sequencing revealed 100% homology with an earlier published Swedish isolate, H2 [2]; direct se-quencing also revealed, as expected, 99% homology with vac-cinia virus sequences. A serum sample, which had been col-lected 5 weeks after the onset of symptoms, was positive forantibodies to orthopoxvirus (vaccinia virus) by plaque reduc-tion neutralization test.The healing process of the numerous areas of lymphadenitiswas markedly prolonged. Two years after onset of symptoms,a single firm node situated in the medial angulus of the righteye was removed (figure 1
). Electron microscopy evaluationof this node yielded negative results, but PCR of the node waspositive for orthopoxvirus DNA. Sequence analysis showed to-tal homology with the PCR product obtained from the patientin February 2001.
Epidemiological findings.
After cowpox infection was di-agnosed (almost 3 months after the onset of symptoms), thepatient recalled having had close contact with 2 domestic cats.One of the cats used to lick her face and nose. Close contactwith the cats was especially frequent after she came home fromthe holiday in Spain. On this occasion, the cat’s tongue pen-etrated the right nasal cavity;
2 weeks later, the first symptomsappeared. No scratches were found on the girl’s nose. The catswere in good health when examined by a veterinarian in Feb-ruary 2001. None of them had lesions of the paws, mouth, ornose or enlarged lymph nodes of the neck. Serum samplesobtained from both cats were tested for orthopox virus anti-bodies at the Swedish Veterinary Institute (Uppsala), and theresults were negative.
DISCUSSION
Poxviruses are the largest enveloped DNA viruses, with an av-erage dimension of nm. The virus particle contains230
300a linear, double-stranded genome of 140–300 kb in length thatencodes several hundred polypeptides. Vertebrate poxvirusescontain 8 genera, of which 4 (Orthopoxvirus, Parapoxvirus,Yatapoxvirus, and Molluscipoxvirus) contain viruses patho-genic to humans [3], all of which, with the exception of small-pox (variola), vaccinia virus, and molluscipoxvirus, are trans-mitted to humans by animals (zoonosis).Smallpox was caused by the variola virus, a member of theOrthopoxvirus genus. It was globally eradicated in the late1970s by mass vaccination, which was successful, because thevariola virus did not have an animal reservoir. Vaccinia, knownas the “Jenner virus,” was the virus used for successfulsmallpoxvaccination, but the exact origin of vaccinia virus is uncertainand there is no known natural host.Vaccination with vaccinia virus provided cross-protectionagainst several orthopoxviruses, includingmonkeypoxvirus,thethird orthopoxvirus. Normally, vaccinia causes mild and local
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