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Primary Neuroleptospirosis
A Case Report and Literature Review
Alfredo Chiappe-Gonzalez, MD,* Csar Ticona-Huaroto, MD,* Valerie Hoerster, MS,
Carlos Coral-Gonzales, MD, and Moiss Sihuincha-Maldonado, MD
Infectious Diseases in Clinical Practice Volume 00, Number 00, Month 2017 www.infectdis.com 1
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Chiappe-Gonzalez et al Infectious Diseases in Clinical Practice Volume 00, Number 00, Month 2017
Hemoglobin 11.2 g/dL Total protein 6.13 g/dL Sodium 138 mEq/L
Hematocrit 32.7% Albumin 2.54 g/dL Potassium 4.14 mEq/L
MCV 74 fl Globulin 3.59 g/dL Chloride 102 mEq/L
MCH 25.3 pg Total bilirubin 0.42 mg% Calcium 8.8 mg/dL
Leucocytes 15,550 Direct bilirubin 0.20 mg% Lumbar puncture
Granulocytes 14,320 Indirect bilirubin 0.22 mg% Glucose 54 mg/dL
Lymphocytes 108 AST 55 U/L Protein 67 mg/dL
Platelet 322,000 ALT 213 U/L G index (csf/ser) 0.6
Glucose 113 mg/dL Arterial gasometry
BUN 24 mg/dL pH 7.47 Chest x-ray Normal
Creatinine 0.8 mg/dL PCO2 29.9 mEq/L Brain CT Normal
CRP 11.2 mg/L HCO3 21.6 mEq/L
LDH 468 U/L PO2 81 mm Hg
Urine test PO2/FiO2 385.7 mm Hg
Leucocytes >100 x/c O2 Sat 96.8%
Erythrocytes 810 x/c G (A-a) 32.4 mm Hg
Epithelial cells Absent FiO2 21%
HAI indicates Hospital Apoyo Iquitos; MCV, mean corpuscular volume; BUN, blood urea nitrogen; CRP, c-reactive protein; LDH, lactate dehydroge-
nase; AST, aspartate transaminase; ALT, alanine transaminase; HCO3, bicarbonate; O2 Sat, oxygen saturation; G (A-a), alveolar-arterial gradient; FiO2, frac-
tion of inspired oxygen; CSF, cerebrospinal fluid; G index (csf/ser), glucose index between CSF and serum glucose; CT, computed tomography.
or biochemical changes; meningeal irritation is unusual. In the 40 years,with high fever, muscular deficits, and renal and/or he-
immune phase, we find meningitis signs, cerebrospinal fluid patic compromise with or without hyperbilirubinemia. Sensory in-
with proteinorrachia, mononuclear pleocytosis, and normal glu- volvement is minimal.911
cose; antibodies are detectable, but bacteria are no longer isolable.6,7 The patient's optic nerve atrophy could correspond to sequalae
The mechanism of neurological compromise has not been of a lesion produced by neuritis or an anterior ischemic stroke, both
elucidated. Postulates include direct effects of the bacteria or an of which have been described in ocular leptospirosis. More fre-
immunologic reaction. In a patient, Lepur et al describe the menin- quently, neuritis, retinal periphlebitis, and panuveitis are reported.12
goencephalitic and PNS compromise after a Leptospira infection It is also interesting to mention the positive Rose Bengal test, which
with detectable anti-GD1a and anti-GM1 antiganglioside antibod- could be explained as a cross-reaction to antibodies against many
ies, which explain the polyneuritis. However, they do not explain other agents; this particular cross-reactivity has also been reported
the diffuse CNS compromise, highlighting the agent's potential in livestock vaccinated against Leptospira.13
ability to simultaneously induce multiple pathogenic mechanisms.8 The patient had a favorable outcome with the established
Our patient was admitted with a febrile neurological syn- therapy. Reports describe a complete clinical recovery 6 to
drome suspicious of GBS with a concurrent UTI, but the neuro- 8 weeks after neurological presentation. Poor prognostic indica-
logical examination detected a CNS and PNS compromise. Such tors described include altered consciousness, proteinorrachia,
compromise has been described as a primary manifestation of and seizure.5,7
neuroleptospirosis by Panicker et al,6 who reported diagnoses The workup focused on looking for infectious and autoim-
of myelopathy, myeloradiculopathy, and GBS representing the mune diseases associated with GBS. Serology for cytomega-
42.5% of all their cases. Other reports similar to ours exist, with lovirus, Epstein-Barr virus, and toxoplasma showed previous
diagnoses of transverse myelitis, paraplegia, axonal motor neurop- exposure. Dengue and chikungunya virus workup ruled out recent
athy, or GBS-like syndromes as the primary presentation. In these infection that would justify the clinical picture. Acute retroviral in-
reports, patients are predominantly male, older than the age of fection was ruled out with a negative human immunodeficiency
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Infectious Diseases in Clinical Practice Volume 00, Number 00, Month 2017 Primary Neuroleptospirosis
virus ELISA test result after the detection window period, as 3. Vargas E. Situacin de la leptospirosis en el Per, aos 20132014
well as neurosyphilis with a negative serum venereal disease re- (a la SE 19). Bol Epidemiol (Lima). 2014;23(19):382385. Available at:
search laboratory. Screening results for autoimmune diseases were http://www.dge.gob.pe/portal/docs/vigilancia/boletines/2014/19.pdf.
also negative. 4. Gancheva GI, Kostadinova MA, Kostadinova PI. Involvement of
Neuroleptospirosis should therefore always be considered in central nervous system in leptospirosis. J Biomed Clin Res.
the differential diagnosis of a patient coming from an endemic 2009;2(2):109114.
area who presents with fever associated with various forms of 5. Berman SJ, Tsai CC, Holmes K, et al. Sporadic anicteric leptospirosis in
acute neurological compromise. South Vietnam. A study in 150 patients. Ann Intern Med. 1973;79(2):
To conclude this case report, it is important to keep in mind 167173.
the following:
6. Panicker JN, Mammachan R, Jayakumar RV. Primary neuroleptospirosis.
Postgrad Med J. 2001;77(911):589590.
Leptospirosis can present as a primary neurological syndrome.
The clinical patterns of the nervous system manifestations 7. Bharucha NE, Bharucha EP, Bhabha SK. Infections of nervous system.
due to Leptospira infection are diverse and include Guillain- In: Bradley WG, Daroft RB, Fenchel GM, et al, eds. Neurology in
Barrlike syndromes. Clinical Practice. Boston, MA: Butterworth-Heinemann; 1996:
The presence of fever with neurological compromise should 12141215.
suggest the diagnosis and justifies the empiric treatment of 8. Lepur D, Himbele J, Klinar I, et al. Anti-ganglioside antibodies-mediated
neuroleptospirosis in highly endemic areas. leptospiral meningomyeloencephalopolyneuritis. Scand J Infect Dis.
It is extremely important to treat neuroleptospirosis with both 2007;39(5):472475.
antibiotics and corticosteroids due to the direct and indirect 9. Mumford C, Dudley N, Terry H. Leptospirosis presenting as a flaccid
(immunologic) damage of the bacteria. paraplegia. Postgrad Med J. 1990;66(773):218220.
10. Kavitha S, Shastry BA. Leptospirosis with transverse myelitis.
J Assoc Physicians India. 2005;53:159160.
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