We describe a case of Lyme carditis with intra-hisian 2:1 atrioventricular (AV) block documented by electrophysiological study. To our knowledge, only two cases of AV block at the level of the His bundle has been described in the literature. Sinus rhythm was restored after 4 days of i.v. ceftriaxon

Adamec, R.

de Freudenreich, J.

Fleisch, M.

Rey, M.-J

Viquerat, C.

Zimmermann, M.

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European Heart Journal (1991) 12, 1048-1051Intra-hisian 2:1 atrioventricular block secondary to LymediseaseM.-J. REY j M. ZIMMERMANN, R. ADAMEC, M. FLEISCH, C. VIQUERAT AND J. DE FREUDENREICHCardiology Center, University Hospital, Geneva, SwitzerlandKEY WORDS: Intra-hisian atrioventricular block, Lyme disease, ceftriaxone.We describe a case ofLyme carditis with intra-hisian 2:1 atrioventricular (A V) block documented by electrophysiologicalstudy. To our knowledge, only two cases ofA V block at the level ofthe His bundle has been described in the literature. Sinusrhythm was restored after 4 days ofi.v. ceftriaxone.IntroductionLyme disease is a tick-borne spirochetal infection, charac-terized by erythema chronicum migrans and an acute sys-temic illness. Neurological, cardiac and joint involvementmay develop weeks to months later. Some manifestationsmay last for years or even decadesl'I Cardiac disturbancesmay rarely become permanent'", Lyme carditis is charac-terized by various degrees of atrioventricular (AV) blockwith or without syncope, which usually resolve within afew weeks. The level ofblock has been described as supra-hisian in most cases in whom an electrophysiologicalstudy was performed during the acute phase of the illness.In this report, we describe a patient who presented withintra-hisian 2:1 AV block related to Lyme disease.Case reportA 49-year-old man complained of sudden discomfortwith sweating and unusual shortness of breath occurringduring exercise. No syncope or chest pain was reported.An ECG showed 2:1 second degree AV block j and thepatient was admitted to hospital. The patient rememberedthat 7 weeks earlier he had had arthritis in all the toes ofhisleft foot 2 weeks after a bite by an unidentified insect. Healso developed a flu-like syndrome with diffuse myalgia,arthralgia, tiredness, and an erythematous itching rash ofthe lower left thigh. There was nothing remarkable in hispast medical history. On examination, the patient waswell with bilateral inguinal lymphadenopathies. His tem-perature was 37°C, the pulse was regular at 60 beats perminute and blood pressure was 140/80 mmHg. A proto-systolic murmur was heard at the left sternal border.Pulmonary auscultation was normal. Liver and spleenwere of normal size. There was oedema of the left ankle;the left toes were warm and pain was elicited by pressureover the foot. Neurological. examination was normal.Erythrocyte sedimentation rate was 22 mm per hour, hae-moglobin 13,7%, white blood cells count 6100. mm",Submitted for publication on 13August 1990, and in revised form 29 October1990,Correspondence: Marc Zimmermann, MD, Cardiology Center, UniversityHospital, 24 rue Micheli-du-Crest, 1211 Geneva 4, Switzerland.0195-668X/91j091048+04 $03.00/0An electrocardiogram showed sinus rhythm at a rate of80 min -1 and 2:1 second degree AV block (Fig. 1).An exercise stress test showed persistent 2:1 AV blockwith a normal increase in sinus rate to 140 beats. min-I at125 W (Fig. 2). Echocardiography was normal. An elec-trophysiological study was performed 4 days after thebeginning of symptoms: it showed two hisian potentials,HI and H2j but these were never recorded in sequence.However, on the tracing showing 2:1 block, the HI poten-tial appeared every time after a blocked P wave; the A-HIinterval was 130 ms. The H2 potential was recorded onlyin the case of AV conduction. The A-Hz interval was160 ms and the H-V interval 50 ms on conducted beat(Fig. 3). Although the His potential was difficult to vis-ualize, the sequence suggesting intra-hisian block washighly reproducible with constant A-HI and A-Hz inter-vals (Fig. 4). Atrial pacing rapidly aggravated the degreeofAV block (3 to 1, 4 to 1)and atropine did not restore I: IAV conduction. No other electrophysiological abnor-malities were observed and a temporary pacemaker wasinserted. Ceftriaxone (2 g per day i.v.) was administeredfor 15days together with only one oral dose ofprednisone50 mg. Lyme disease was confirmed by serology, showingpositive borrelia burgdorferi IgG (titre: 1/64) and positiveborrelia burgdorferi IgM antibodies. The clinical coursewas uneventful: normal AV conduction resumed after 4days of treatment and the pacemaker was removed. Atfollow-up, five months later, the patient was asympto-matic; the physical examination, the 12 lead ECG and aHolter recording were all normal.DiscussionCarditis occurs in 4 to 12% of cases of Lyme disease,usually at a mean of 4·8 weeks (range 4 days to 7 months)after the initial disease'!', Varying degrees of AV block arethe commonest manifestations of Lyme carditis(Z,3J: from56 reported cases, 49 (87'5%) had documented AV block;in 32 cases complete or high-grade AV block wasobserved, usually with syncope. The escape rhythm rarelyhad a wide QRS pattern (11 cases) or a rate below 40beats. min-I (IOcases). Fluctuating or intermittent left orright bundle branch block has also been observed(2,4-6l.© 1991The European Society of CardiologyIntra-hisian atrioventricular block 1049nJ.IIIIIrt.VR.VFV2V6Figure 1 Resting l2-lead electrocardiogram showing 2:1 AV block (atrial rate 80beats. min-I, ventricular rate 40 beats. min-I).V5Figure 2 Exercise electrocardiogram showing persistant 2:I AV block with anatrial rate of 140beats. min-I (arrows) and a ventricular rate of70 beats. min-I.Several reports have mentioned transient lack of anyescape rhythm, with asystole of 4 to 9 seconds[2,4,5,7-9]. Anelectrophysiological study was performed in only 12patients with Lyme carditis during the acute or subacutephase of the disease (Table 1); in the majority of cases(lOjl2), the level of AV block was considered to be abovethe bundle of His. Intra or infra-hisian block has beenonly rarely suspected or documented'P'", Van der Lindeet al.[2] reported a case where the electrophysiologicalstudy showed a small positive His bundle deflection with-out a negative deflection at all sites mapped in the Hisbundle region. The AH interval was normal, but there wasno relation between His bundle activity and QRS com-plexes. In the case reported by McAlister et alP], a pro-longed HV interval (70 ms) suggested an infra-hisianconduction delay. In the case published by Cornuauet al)?], the electrophysiological study showed a normalAH interval (80 ms), major intra-hisian conduction delay(H 1-H2 120ms) and infra-hisian block. The recordingpublished by Dunica et al.[9] suggested supra-hisian blockwith an AH interval of 175ms and an HV interval of45 ms; however the author suspected that the block couldhave also been present at a lower level because there was asmall atrial deflection on the AV lead during the AV blockand because no escape rhythm was present during com-plete AV block. Kapusta et al.[10] also observed a smalldeflection after the first blocked A wave, which could beinterpreted as a partial activity of the His bundle. Ourpatient had an electrophysiological study performed 4days after the onset of cardiovascular symptoms, whichshowed intra-hisian AV block; although the double Hispotential was difficult to visualize, the sequence suggest-ing intra-hisian block was highly reproducible. Allgroups have reported that patients with AV block have an1050 M. -J. Rey et aJ.1 sec1 1TTi TT:J J I' j j- •. I 1.- -1. - :' j' ,' l ~rr"1.-r:tfF T~ ' T t - Hj- ' I J.l t t -j-j j ---r ':: :; J j-IT-I-r.-lTri ! : ~ , ~-'1 - - !-- rt :L -T ii-~ _ ~ 1-' ' 1!~t;L~·j,i : !i -T 1 I! i i . !: --- i' : ' ~' +II p..; , -- , ' ~ -- - " f+ -', ; -,~-. ,J ' - -: - -.~ t " t r I -' .' j-I ' - -" I " Of !-'-:: I t---111 H-- -t- ·_rJ d .__1 h -' r-H+ --;--; -.--i+ ~ . . , ' ~- ;-4. - . -!.-!-----1i' rtCL -+- ~-I- -t-- -Ll. -1..1- '- t1 ' '" IT- , '. I I I, ,', '. I 1- 1 ~.- . I ,-. . , I ' -- I l " ..JJ, ;-'A-- -i "1.2,'/ ,+H- -r' !-.A ' I - ' ~ -- -, ".- ~ +-t ~A--'" -" ~-j : -- ,"BE " J ,. '.1 . T··I +--- - -~. " :-- ",f- " 1' :'-- h ~ T ':+. ~-'~: :-r±.,-1- -- +-LI-- i 1-!- -1-- . i roo, I~_ ' +- ,~~ =l -..j -~- -- ;, --"- 1 1- , " l'_~ ~;"' f---'--i _L. ' j!l8i" ' -- - -- i' I _-t-l -. - -- .~ I I-I -1 ' _.; I ti-' - f-_ Iol""~- -' . I-t- f-" V ,-- .- l- t- - - - - V I--.LRA . _ U : - - _ . - I IU; , . '- _1.. .L: x-FF [tl -r ~-tTI- "--lt.L -- -.. - -~ lI .L, - - - - f - L -j -. r·-+I I - - 1- -- !L!-I_LL _I_ __ _ . __ .L . _i_~L __ L I- _ .I- L L..L -1Figure 3 A 2:I AV block arising with the His bun dle: during sinus rhythm, two Hpotentials (arrows) are identified (H, and H 2) and the block occurs between the two Hdeflections. H BE: His bundle recording; LRA: low right atrium; A: atrium; V:ventricle.A· lf l : 130 m lm.I 1 St'C I 1A· lI l , 160IIBE +::.il:T r "I; ~ ' ! :! I ' ~ ' I : i . : '! : L~ ~ . , ~.~~ :_.; L L~~JJ ~ .; ;_ IFigure 4 A 2:I AV block occurring within the His bundle du ring atrial pacing at acycle length of 700 IDS . Abbreviations same as Fig, 3.Table 1 Electrophysiological data obtainedduring the acute or subacute phase 0/Lym e carditis: reviewofthe literatureFirst authorVan der Linde'"McA lister))Cornuaud!"Reznick'"Dunika'"Kapus ta''"Steere' !'!Lavaud'!"Reyn2III2ECG Level of block Evolutioncomplete AV block intra-hisian pennanent PMcomplete AV block+LBBB,jllnct. escape supra-hisian SRhigh grade AV block supra-hisian SRcomplete AV block intra-hisian SR2:1AVblock supra-hisian SR2:1 AVblockcomplete AV blockincomplete RBBB supra-hisian SR2-3:I A V block, RBBB supra-hisian SRintra-hisian?complete AV block supra-hisian SRcomplete AV bloc supra-hisian SR2:1 AVblock intra-hisian SRAV =atrioventricular; SR = sinus rhythm; LBBD=left bundle branch block; n =number of cases ;RBBB= right bundle branch block; PM = pacemaker.excellent prognosis: most cases resolved within 1 to 2weeks, and three within 6 weeks'". Only Van der Lindeet alP] reported a case who required implantation of apermanent pacemaker after 15 weeks of follow-up.Therefore, the insertion of a permanent pacemaker israrely indicated. Lyme carditis may occur with high-degree AV block during the sub-acute phase of infection:this was the case for our patient who had the first symp-toms of erythema in August 1989, 7 weeks before thecardiovascular manifestations of the disease.Several therapeutic principles of Lyme carditis aregenerally accepted: hospitalization and continuous moni-toring are advisable for patients with second-degree orcomplete AV block, and with first-degree AV block if thePR interval exceeds 0·30 s[lll. At the present time eitherintravenous penicillin G 20 millions units per day or oraltetracycline for 10 to 20 days are recommended; in highdegree AV block, ceftriazone 2 g intravenously once a dayfor 14 days can be prescribed instead of penicillin'l-'". Itseems that the persistence of the spirochetes within themyocardium is an intrinsic part of the pathophysiologyof Lyme carditis. The observation of spirochetes withincardiac tissue[9,13] lends further support to this hypothesis.It remains unclear, however, whether continued diseaseactivity requires 'the persistence of live spirochetes orwhether it results primarily from immune-mediatedmechanisms.In our case, the administration of ceftriazone resultedin rapid restoration of normal AV conduction withoutany sequellae.References[1] Steere AC. Lyme disease. N Engl J Med 1989;321: 586-96.Intra-hisian atrioventricular block 1051[2] Vander LindeMR, Crijns HJGM, de KoningJ. Range of atrio-ventricular conduction disturbances in Lyme borreliosis: areport of four cases and review of other published reports. BritHeart J 1990;63: 162-8.[3J McAlister HG, Klementowicz PT, Andrews C, Fischer JD,Feld M, Furman S. Lyme carditis: an important cause of revers-ible block. Ann Intern Med 1989; Ito: 339-45.[4] Viereys C, AlIal J, Coisne D, Thomas Ph, Neau JP, Breux JP.Aspects europeens de la maladie de Lyme. Presse Med 1987; 16:59-62.[5] Allal J, Coisne D, Thomas P, et al. Manifestations cardiaquesde la maladie de Lyme. Ann Med Interne (Paris) 1986; 137:372-4.[6J Alpert LI, Welsch P, Fischer N. Gallium-positive Lyme diseasemyocarditis, Clin Nucl Med 1985; to: 617.[7] Cornuau C, Bernard M, Daumas P, Oblet B, Poirot G, ValoisM. Les manifestations cardiaques de la maladie de Lyme: apropos de 2 observations. Ann Cardiol Angeiol1984; 33: 395-9.[8] Reznick JW, Braunstein DB, Walsch RL et al. Lyme carditis.E1ectrophysiologicand histopathologic study. Am J Med 1986;81: 923-7.[9] Dunica S, Piette JC, Nassar N, Beaufils P. Une nouvelle causede bloc auriculo-ventriculaire aigu transitoire: 1a maladie deLyme. Arch Mal Coeur 1986;79: 1251-5.[to] Kapusta P, Fauchier JP, Cosnay R, Huguet R, Grezard 0,Rouesnel P. Troubles conductifs sino-auriculaires et auriculo-ventriculaires de la maladie de Lyme. Arch Mal Coeur 1986;9:1361-6.[11] Steere AC, Batsford WP, Weinburg M, et al. Lyme carditis:cardiac abnormalities of Lyme disease. Ann Intern Med 1980;93: 8-16.[12] Dattwyler RJ, Halperin Ll, Pass H, Luft BJ. Ceftriaxone aseffective therapy in refractory Lyme disease. J Infect Dis 1987;155: 1322-5.[13] Stanek G, Jutta K, Bittner R, Glogar D. Isolation of BorreliaBurgdorferi from the myocardium of a patient with longstand-ingcardiomyopathy. N EnglJ Med 1990;322: 249-52.[14] Lavaud P, Etienne J, Chamot E et al. Bloc auriculo-ventricu-laire aigu associe aune ma1adiede Lyme. Presse Med 1985; 14:2020.

Intra-hisian 2:1 atrioventricular block secondary to Lyme disease

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