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Inicio Enfermedades Infecciosas y Microbiología Clínica Pediatric tubular acute lymphangitis caused by Rickettsia sibirica mongolitimona...
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Vol. 40. Núm. 4.
Páginas 218-219 (Abril 2022)
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Vol. 40. Núm. 4.
Páginas 218-219 (Abril 2022)
Letter to the Editor
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Pediatric tubular acute lymphangitis caused by Rickettsia sibirica mongolitimonae: Case report and literature review
Linfangitis tubular aguda pediátrica por Rickettsia sibirica mongolitimonae: reporte de caso y revisión bibliográfica
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Álvaro Vázquez-Péreza,
Autor para correspondencia
avzqpz@gmail.com

Corresponding author.
, Javier Rodríguez-Grangerb, Elizabeth Calatrava-Hernándezb, Juan Luis Santos-Péreza
a Department of Pediatrics, Virgen de las Nieves Children's Hospital, Granada, Spain
b Department of Microbiology, Virgen de las Nieves University Hospital, Granada, Spain
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Table 1. Infections by Rickettsia sibirica mongolitimonaea in children.
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Dear Editor,

In a recent report, Salazar Alarcón et al.1 described a case of lymphangitis-associated rickettsiosis (LAR) caused by Rickettsia sibirica mongolitimonae (R. sibirica mongolotimonae), with no cases in the pediatric population previously reported in the literature. In this letter, we report another pediatric case of LAR by R. sibirica mongolitimonae in southern Spain, which confirms an expanded distribution of this agent in our country. Moreover, we aim to highlight the potential risks associated with its main vector, the species of the genus Hyalomma.2 Human transmission by Hyalomma ticks of other life-threatening zoonotic agents such as Crimean-Congo hemorrhagic fever virus (CCHFV) has already been reported.3

We report the case of a 4-year-old boy with fever of up to 39.7°C for 5 days. In the 24h before the onset of fever, he presented a lesion compatible with a bite on the inner side of the right thigh. Physical examination revealed a circular lesion (5mm) with a necrotic eschar, surrounded by a slightly raised inflammatory halo (Fig. 1). He associated rope-like lymphangitis running from the eschar up to the inguinal region, where a painful adenopathy (2cm×1cm) was found. The rest of the physical examination was normal. A shave biopsy was performed to determine a Polymerase chain reaction (PCR) for Rickettsia. Analytical control was performed with blood count: 10,180 leukocytes (70.8% N, 16.4% L, 12.6% M); Red and platelet series, biochemical profile, and basic coagulation were normal. CRP 24.9mg/L. He began treatment with oral azithromycin and intravenous amoxicillin-clavulanate. Serological studies of Rickettsia conorii, Borrelia Burgdoferi, and Bartonella henselae were negative. He showed a favorable evolution and was discharged on the fifth day. Follow-up confirmed good evolution and no seroconversion. The diagnosis was confirmed with the results of PCR in the eschar sample performed at the National Center for Microbiology: PCR Borrelia sp. negative and positive for Rickettsia sibirica mongolotimonae. Bands compatible with Rickettsia (SFG) infection were detected, using as targets fragments of the ompA and ompB genes (conventional PCR), and 23S rRNA (real-time PCR). Definitive identification was obtained by molecular sequencing of ompA and ompB genes (491 and 464bp, respectively) that revealed 100% identity with Rickettsia sibirica subsp. mongolitimonae.

Fig. 1.

Necrotic eschar on the inner side of the right thigh, surrounded by a slightly raised inflammatory halo.

(0,18MB).

Different clinical manifestations of the infection have been described, such as Dermacentor-borne necrosis erythema and lymphadenopathy (DEBONEL) and mainly, lymphangitis-associated rickettsiosis (LAR).2 The few reported cases of R. sibirica mongolotimonae infection in children1,4–6 are presented in Table 1. The median age of LAR cases was 5 y.o. The three of them were detected in late spring and early summer. In all cases fever was present and just one case presented a generalized skin rash. In the DEBONEL pictures, the presence of eschar and adenopathy also stands out, generally in the upper half of the body and scalp; fever may not always be present and usually occur in cold months.2 In adults, severe manifestations have been described,7 such as septic shock, acute kidney injury, myopericarditis, retinal vasculitis, and neurological alterations, including encephalitis, among others.

Table 1.

Infections by Rickettsia sibirica mongolitimonaea in children.

CaseYear  Geographic location  Month  Risk factors  Age (years)  Sex  Tick bite site  Fever  Rash  Diagnostic  Serology  Reference 
52020  Alcorcón (Madrid – Spain)  July  Rural dwelling.Tick infested dog contact  11  Men  Upper extremity  Yes  No  LARb  Negative for Rickettsia conorii  Echevarría-Zubero et al.4 
42020  Illescas (Madrid – Spain)  June  Rural dwelling.Tick infested dog contact  Men  Genital area  Yes  Yes  LAR  Negative for Rickettsia conorii and Borrelia burgdorferi  Salazar Alarcón et al.1 
32020  Alcalá la Real (Jaén-Spain)  May  Rural dwelling.Tick infested dog contact  Men  Lower extremity  Yes  No  LAR  Negative for Rickettsia conorii, Bartonella Henselae; and Borrelia Burgdoferi  Case presented in this article 
22015  Reus (Tarragona-Spain)  Outside the summer season (N.S.)d  Rural dwelling  N.S.  Head  Yes  Yes  DEBONELc  Negative for Rickettsia conorii  Monterde-Álvarez et al.6 
12009  Arganda del Rey (Madrid-Spain)  October  Rural dwellingFarm animals  Men  Head  Yes  No  DEBONELc  Negative for Rickettsia conorii and Borrelia Burgdoferi  Morales et al.5 
a

In all cases, the diagnosis was confirmed with the results of polymerase chain reaction (PCR) in the biopsy of the eschar sample. DNA samples were tested by PCR targeting fragments of the rickettsial genes ompA and ompB (conventional PCR). Definitive identification was obtained by molecular sequence of ompA and ompB, what showed a 100% identity with Rickettsia sibirica subsp. Mongolitimonae.

b

Lymphangitis-associated rickettsiosis.

c

Dermacentor-borne necrosis erythema and lymphadenopathy.

d

Not specified.

One of the limitations in the study of these cases is the difficulty in identifying the species of tick involved, unknown as in our case. Nevertheless, although it has been isolated in Rhipicephalus pusillus and R. bursa, its main vector is considered to be the species of the genus Hyalomma.2 These species are characterized by their aggressive host-seeking behavior8 (unlike other ticks utilizing a passive ambush strategy as they wait in vegetation), at a surprising speed for its size.8 Perfectly adapted to semi-desert climates, they usually have maximum activity in the hot and dry months.8 They are not very anthropophilic, although, in Spain, a progressive increase in their population is reported3,9 along with a high proportion of CCHFV infected ticks collected from wildlife.3,9 The change in climatic conditions seems to play an important role in this increasement.9,10 The fragmentation of the plant habitat and the abandonment of farmland would also be also decisive in tick populations and their hosts, causing increased contact rates between humans and infected ticks.10 These potential risks emphasize the importance of the development of new prevention strategies and the evaluation of this threat to public health.3

Informed consent

The family of the patient described in this case report gave their informed consent for the inclusion in this publication.

Conflict of interest

The authors declare no conflict of interest in this article.

References
[1]
E. Salazar Alarcón, S. Guillén-Martín, I. Callejas-Caballero, A. Valero-Arenas.
A propósito de un caso: no toda rickettsiosis es fiebre botonosa mediterránea.
Enferm Infecc Microbiol Clin, (2021),
[2]
Á.A. Faccini-Martínez, L. García-Álvarez, M. Hidalgo, J.A. Oteo.
Syndromic classification of rickettsioses: an approach for clinical practice.
Int J Infect Dis, 28 (2014), pp. 126-139
[3]
A. Portillo, A.M. Palomar, P. Santibáñez, J.A. Oteo.
Epidemiological aspects of Crimean-Congo hemorrhagic fever in Western Europe: what about the future?.
Microorganisms, 9 (2021), pp. 649
[4]
R. Echevarría-Zubero, E. Porras-López, C. Campelo-Gutiérrez, J.C. Rivas-Crespo, A.M. Lucas, E. Cobo-Vázquez.
Lymphangitis-associated rickettsiosis by Rickettsia sibirica mongolitimonae.
J Pediatric Infect Dis Soc, 10 (2021), pp. 797-799
[5]
V. Morales, C.R. Garcia Acebes, A.P. Miguelez Hernandez, F. Alfagueme Roldán, A. Rodríguez Albarrán, Y. Alins Sahun, et al.
Infeccion por Rickettsia sibirica subsp. monogolitimonae.
[6]
M.L. Monterde-Álvarez, C. Calbet-Ferré, N. Rius-Gordillo, I. Pujol-Bajador, F. Ballester-Bastardie, J. Escribano-Subías.
Rickettsiosis tras la picadura de una garrapata: una clínica sutil en muchas ocasiones, debemos estar atentos.
Enferm Infecc Microbiol Clin, 35 (2017), pp. 100-103
[7]
M.D.C. Loarte, C. Melenotte, N. Cassir, et al.
Rickettsia mongolitimonae Encephalitis Southern France, 2018.
Emerg Infect Dis, 26 (2020), pp. 362-364
[8]
J.F. Anderson, L.A. Magnarelli.
Biology of ticks.
Infect Dis Clin North Am, 22 (2008),
[9]
A. Moraga-Fernández, F. Ruiz-Fons, M.A. Habela, et al.
Detection of new Crimean-Congo haemorrhagic fever virus genotypes in ticks feeding on deer and wild boar, Spain.
Transbound Emerg Dis, 68 (2021), pp. 993-1000
[10]
A. Estrada-Peña, N. Ayllón, J. de la Fuente.
Impact of climate trends on tick-borne pathogen transmission.
Front Physiol, 3 (2012), pp. 64
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