54 Arch Dis Child 2001;85:54–55

SHORT REPORT Arch Dis Child: first published as 10.1136/adc.85.1.54 on 1 July 2001. Downloaded from

Facial nerve palsy associated with conorii infection

M Bitsori, E Galanakis, C E Papadakis, S Sbyrakis

Abstract Chest x ray was normal. Serology for Epstein– Facial nerve palsy has been occasionally Barr virus, cytomegalovirus, herpesviruses 1 attributed to infectious agents, but Rick- and 2, adenovirus, coxsackie viruses, Myco- ettsiae species have not been documented plasma pneumoniae, Chlamydia pneumoniae, and as causative agents. We report two adoles- Borrelia burgdorferi was negative for acute cent girls with facial nerve palsy and sero- infection. IgM and IgG antibodies against R logical evidence of R conorii infection. conorii at titres of 1/960 and 1/3840 respectively These cases indicate that rickettsioses were detected by indirect immunofluorescence should be included among the causes of test. IgM antibodies against R typhi and facial nerve palsy, particularly in endemic at a titre of 1/100 and IgG areas. antibodies at titres of 1/960 and 1/400 respec- (Arch Dis Child 2001;85:54–55) tively were also detected. Oral clarithromycin and prednisolone 2 Keywords: Bell’s palsy; facial nerve palsy; Rickettsia mg/kg were administered. The fever subsided, conorii; rickettsial infections but the facial weakness progressed to paralysis over the next three days. Prednisolone was Acute unilateral facial nerve palsy has occa- continued at the same dose for two more days sionally been attributed to infectious agents, and subsequently tapered within five days. Two including the herpes, mumps, rubella, influ- weeks later IgM and IgG antibodies against R enza, respiratory syncytial, human immuno- conorii were 1/3840 and 1/15 360 respectively,

deficiency, and coxsackie viruses, and Borrelia thus confirming acute R conorii infection. http://adc.bmj.com/ burgdorferi, Mycoplasma pneumoniae, Chlamydia Titres for R typhi and Coxiella burnetii remained pneumoniae, and group B streptococci.1 A non- low. Facial palsy subsided three months later. specific immunological response to infection, leading to facial nerve compression and degen- CASE 2 eration has been suggested as the pathogenic A 12 year old girl was admitted with a five day mechanism. Rickettsiae species have not been history of progressive right facial side weak- implicated in the aetiology of facial nerve palsy. ness, following a flu like febrile illness two We report two cases of facial nerve palsy in weeks previously. Physical examination re- on September 30, 2021 by guest. Protected copyright. adolescents with serological evidence of vealed a drooped mouth corner, impaired facial Rickettsia conorii infection. muscle movement, and eye closure on the right side. Liver and spleen were palpable at 4 cm Case reports and 3 cm respectively. Blood pressure was CASE 1 125/70 mm Hg. Examination of the other cra- A 14 year old girl was admitted with a six day nial nerves and tympanoscopy was normal. Department of history of fever, cough, and conjunctivitis. Tympanometry revealed a normal tympano- Paediatrics, University Physical examination revealed bilateral non- gram of type A in both ears. The acoustic Hospital of Heraklion, Crete, Greece purulent conjunctivitis, palpable liver and reflexes, measured by ipsilateral and contralat- M Bitsori spleen 3 cm subcostally, cervical lymphaden- eral stimulation, were absent in the right and E Galanakis opathy, and right facial weakness. Blood normal in the left ear. White blood cell count S Sbyrakis pressure was 110/60 mm Hg. Chest ausculta- was 5 × 109/l; neutrophil count, 1.85 × 109/l; tion, examination of the remaining cranial haemoglobin, 1.89 mmol/l; and platelet count, Department of nerves, and tympanoscopy were normal. Tym- 390 × 109/l. Erythrocyte sedimentation rate was Otorhinolaryngology, University Hospital of panometry revealed a normal tympanogram of 31 mm/h; alanine aminotransferase, 48 U/ml; Heraklion type A in both ears. The acoustic reflexes, and aspartate aminotransferase, 60 U/ml. C E Papadakis measured by ipsilateral and contralateral Serology for Epstein–Barr virus, cytomegalovi- stimulation, were absent in the right and rus, herpesviruses 1 and 2, adenovirus, cox- Correspondence to: normal in the left ear. White blood cell count sackie viruses, Mycoplasma pneumoniae, Dr E Galanakis, Department 9 9 of Paediatrics, University of was 13.7 × 10 /l; neutrophil count, 6.98 × 10 /l; Chlamydia pneumoniae, and Borrelia burgdoferi Crete, PO Box 1393, 715 00 haemoglobin, 1.72 mmol/l; and platelet count, was negative for acute infection. Indirect Heraklion, Greece 292 × 109/l. Erythrocyte sedimentation rate was immunofluorescence test was positive for R [email protected] 11 mm/h; alanine aminotransferase, 45 U/ml; conorii, with an IgM titre of 1/400 and IgG titre Accepted 28 March 2001 and aspartate aminotransferase, 68 U/ml. of 1/960. IgG antibodies against R typhi and

www.archdischild.com Facial nerve palsy and R conorii infection 55

Coxiella burnetii at titres of 1/400 and 1/100 these had a clinical presentation and serologi-

respectively were also detected. cal confirmation of acute Epstein–Barr virus Arch Dis Child: first published as 10.1136/adc.85.1.54 on 1 July 2001. Downloaded from Oral prednisolone 2 mg/kg was administered infection; serological evidence of R conorii for five days and gradually tapered within five infection was present in two others. In the days. A repeat serology for R conorii two weeks remaining two cases no aetiological agent was later revealed IgM antibody titre of 1/1600 and found. IgG antibody titre of 1/3840, thus confirming a The indirect immunofluorescence test is recent R conorii infection. Four months later, generally considered to be a sensitive and spe- on a follow up evaluation, the girl had normal cific test for confirming the diagnosis of R right facial nerve function; IgM and IgG titres conorii infection. Titres above 1/150 for IgM against R conorii were 1/100 and 1/1600 and above 1/400 for IgG specific antibodies are respectively. suggestive of acute infection. As low titres of specific IgM antibodies may be present in Discussion 34 many systemic diseases, confirmation of the Mediterranean , the typical sys- diagnosis requires a fourfold increase of the temic R conorii infection, usually presents with spiking fever, myalgia, headache, hepato- specific antibody titre in a second sample, three splenomegaly, and a maculopapular or purpu- to four weeks after the onset of symptoms. ric rash. However, atypical cases have been Tetracyclines and chloramphenicol, given reported.23The disease is considered endemic early in the course of the disease, are in southern Europe, and epidemiological stud- considered to be the treatment of choice for all ies in certain parts of Greece have shown con- rickettsial infections. Our patients did not siderable seroprevalence.4 receive any specific treatment, as confirmation Our first patient was admitted with a of the diagnosis was rather delayed. A short diagnosis of lower respiratory tract infection. course of corticosteroids was administered, Facial palsy was noted later in the course of her although their role in the treatment of facial illness. She was investigated for rickettsial nerve palsy remains controversial.7 infection because of history of animal contact In conclusion, rickettsial infection should be with the household dogs of the family. The included among the causes of facial nerve palsy second patient presented with typical Bell’s in endemic areas, particularly in patients with a palsy and a preceding upper respiratory tract history of animal contact. The outcome of such infection. She was investigated for rickettsial cases associated with rickettsioses seems excel- infection because of the presence of mice on lent. the family’s farm. Tick bite signs were not present in either patient. Neurological involvement in rickettsial in- 1 Morgan M, Nathwani D. Facial palsy and infection: the unfolding story. Clin Infect Dis 1992;14:263–71. fections is encountered in a minority of cases 2 Brouqui P, Tissot Dupont H, Drancourt M, et al. Spotless

with severe systemic manifestations. Distur- . Clin Infect Dis 1992;14:114–16. http://adc.bmj.com/ 3 Cascio A, Dones P, Romano A, Titone L. Clinical and labo- bances of consciousness, , ratory findings of boutonneuse fever in Sicilian children. and a case of Guillain–Barré syndrome in an Eur J Pediatr 1998;157:482–6. 4 Antoniou M, Tselentis Y, Gikas A, et al. The seroprevalence adult patient with R conorii infection have been of ten zoonoses in two villages of Crete, Greece. EurJEpi- 56 reported. Rickettsiae are not currently in- demiol 1995;11:415–23. cluded among the aetiological agents of facial 5 Silpapojacul K, Ukkachoke C, Krisanapan S, Silpapojakul K. Rickettsial meningitis and encephalitis. Arch Intern Med nerve palsy. In a series of R conorii infection 1991;151:1753–7. cases in children from Sicily, two cases of facial 6 De Galan BE, van Kasteren BJ, van den Wall Bake AWL,

Vreugdenhil G. A case of Guillain-Barré syndrome due to on September 30, 2021 by guest. Protected copyright. nerve palsy were noted; however, clinical infection with Rickettsia conorii. Eur J Clin Microbiol Infect details of these two patients were not reported.3 Dis 1999;18:79–80. 7 Unuvar E, Oguz F, Sidal M, Kilic A. Corticosteroid Six patients with facial nerve palsy were hospi- treatment of childhood Bell’s palsy. Pediatr Neurol 1999;21: talised in our department during 1999. Two of 814–16.

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