Diaphragmmatic eventration: long-term follow-up and results of open-chest plicature

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1 Europen Journl of Crdio-thorcic Surgery 36 (2009) Diphrgmmtic eventrtion: long-term follow-up nd results of open-chest plicture Pulo Clvinho, Cláudi Bstos, João E. Bernrdo, Luís Eugénio, Mnuel J. Antunes * Crdiothorcic Surgery, University Hospitl, Coimbr, Portugl Received 23 October 2008; received in revised form 11 My 2009; ccepted 19 My 2009; Avilble online 24 July 2009 Abstrct Objective: Diphrgmmtic eventrtion is reltively uncommon entity with simple surgicl correction technique pliction of the diphrgm. This study ims to ssess the clinicl nd ventiltory impct of this technique. Mterils: From April 1988 to Februry 2007, we operted on 20 ptients (12 men) with diphrgmmtic eventrtion using the ero-lterl pproch nd correction by rdil pliction. The men ge of the ptients studied ws yers (rnge: yers). A trumtic cuse ws identified in 13 ptients; one ptient hd congenitl cuse nd the reminder were of idiopthic origin. Chronic obstructive pulmonry disese nd rteril hypertension were sent in one-hlf of the study group, while dibetes mellitus ws sent in three ptients. Dyspnoe ws the most common complint in 75% of the ptients, nd thorcic pin ws sent in 25%. The men forced expirtory volume in 1 s (FEV 1 ) nd vitl cpcity (VC) were % nd % of the dicted vlues, respectively. Results: There ws no opertive mortlity. Aprt from ptient with moderte/severe pin nd nother who hd pneumoni, there were no other importnt periopertive complictions. Averge dringe time ws dys (rnge: 2 7 dys). Hospitlistion time ws dys (5 10 dys). Follow-up ws complete, for men of months (4 206 months). There were three lte deths (one sudden, one stroke nd one trum). Eight of the 17 survivors (47%) re symptomtic. According to the MRC/ATS grding system, the dyspnoe score ws opertively nd opertively ( p = 0.007). At follow-up, the FEV 1 ws % nd the VC ws % ( p > 0.1). Two ptients hd chronic pin. Conclusion: Pliction of the diphrgm is sfe nd efficient procedure. Most ptients experienced significnt clinicl improvement with enhncement of the FEV 1 nd VC. Chronic surgicl pin still remins potentil problem with the clssicl pproch. # 2009 Europen Assocition for Crdio-Thorcic Surgery. Published by Elsevier B.V. All rights reserved. Keywords: Diphrgm; Eventrtion; Herni; Plicture; Surgery 1. Introduction Diphrgmmtic eventrtion is defined s permnent elevtion of hemidiphrgm without defects of continuity. The musculr insertions re norml, the norml orifices re seled nd there is no interruption of the pleurl or peritonel lyers [1]. The eventrtion my be of congenitl origin, with lrge unilterl elevtion of the diphrgm becuse of secondry hypoplsi of the homolterl lung, usully senting with severe crdiorespirtory symptoms in the newborn. Eventrtion tht occurs in older children nd in dults is thought to be cused by prlysis of diphrgmmtic lef, normlly ccompnied by dyspnoe. Gstrointestinl symptoms my be sent due to disloction of bdominl viscer [2]. Although it is conceivble tht long-lsting diphrgm prlysis leds to cor pulmonle, there is no evidence Presented t the 22nd Annul Meeting of the Europen Assocition for Crdio-thorcic Surgery, Lisbon, Portugl, September 14 17, * Corresponding uthor. Tel.: ; fx: E-mil ddress: ntunes.cct.huc@spo.pt (M.J. Antunes). substntiting this ssumption [3]. Mlignncy, trum, infection, cervicl spondylosis nd itrogeny (surgery on the medistinum/crdic) nd neuromusculr disorders re the most common cuses of this condition. There re number of ptients in whom cuse cnnot be explined (idiopthic eventrtion). The exct incidence of this pthology is not known, nd cn most probbly be ttributed to under-recognition. The first report of surgicl repir ws published by Morrison in 1923 [4]. Since then, severl series were published describing different techniques nd results of diphrgmmtic pliction. With this technique, the muscle is fixed in the position of mximum inspirtion, decresing the ventiltory work. This is prticulrly importnt in ptients with chronic obstructive lung disese, for it cretes lrger spce to these hyper-expnded lungs [2]. However, surgicl tretment is infrequently performed for unilterl diphrgmmtic prlysis in dults. Brriers to wider use of pliction of the diphrgm include lck of ttribution of symptoms to unilterl diphrgmmtic prlysis, uncertinty bout the potentil benefits of pliction nd the perceived need for thorcotomy [5]. In /$ see front mtter # 2009 Europen Assocition for Crdio-Thorcic Surgery. Published by Elsevier B.V. All rights reserved. doi: /j.ejcts Downloded from

2 884 P. Clvinho et l. / Europen Journl of Crdio-thorcic Surgery 36 (2009) this work, we describe our experience with 20 ptients who hd unilterl non-mlignnt diphrgmmtic eventrtion, focussing the nlysis on the symptomtic sttus, especilly with regrds to the degree of dyspnoe, nd on the pulmonry function during long-term follow-up. 2. Ptients nd methods 2.1. Popultion Between April 1988 nd Februry 2007, 20 consecutive ptients, 12 (60%) mles, with men ge of yers (rnge: yers), underwent surgicl tretment for diphrgmmtic eventrtion. Since most ptients hd the eventrtion for t lest 1 yer, the chnce of spontneous recovery ws smll, which, in combintion with the symptoms, ws the indiction for surgicl tretment. For inclusion in this study, the ptients were identified retrospectively by consulting the surgicl reports. The following dt hd been registered prospectively in the clinicl records: ge, sex, clinicl history, etiology, symptoms, side (loction) of disese, vlues of spirometry (vitl cpcity nd 1-s forced expirtory volume) in the sitting position, opertive findings nd procedures, durtion of chest dringe, peri- nd opertive complictions nd durtion of hospitl sty. Previous roentgenogrms were reviewed, if vilble. Thorcic nd upper bdominl computed tomogrphic (CT) scns were lwys done. Severl ptients lso hd mgnetic resonnce imging (MRI) ordered by the referring physicin prior to hospitlistion Aetiology nd ssocited pthology All ptients with mlignnt diphrgmmtic prlyses/ eventrtions were excluded from the study. After creful nlysis of the nmnesis nd physicl exmintion of ll ptients, trum ws identified s the only objective cuse of diphrgmmtic eventrtion in 13 ptients (65%). A 13-yerold ptient hd n eventrtion tht ws clerly of congenitl origin. The remining six were clssified s idiopthic (Tble 1). The etiologicl ssessment ws performed by the ssistnt clinicin nd sented for decision by the pneumologist nd the thorcic surgeon. Obstructive chronic pulmonry disese nd rteril hypertension were identified in hlf of the study group (10 ptients). Dibetes mellitus ws dignosed in three ptients nd one ptient hd coronry disese. Five ptients (25%) hd no ssocited pthology Pulmonry function tests Vitl cpcity (VC) nd 1-s forced expirtory volume (FEV 1 ) were mesured in ll ptients in the sitting position, opertively nd t follow-up, ccording to the guidelines of the Europen Respirtory Society (ERS), nd re exssed s percentge of the dicted vlues Dyspnoe index Assessment of dyspnoe ws done using the Medicl Reserch Council/Americn Thorcic Society (MRC/ATS) grding system [6], which correltes the ptient s functionl Tble 1 Ptient dt. Ptient Sex Age Aetiology Side of eventrtion impirment with the mgnitude of tsk needed to evoke dyspnoe (Tble 2) Surgicl procedure Yer of opertion 1 M 40 Trumtic Left M 74 Unknown Left M 52 Trumtic Left M 68 Unknown Left F 65 Trumtic Right F 67 Trumtic Right M 60 Unknown Right F 62 Unknown Left M 50 Trumtic Right M 51 Trumtic Right F 28 Trumtic Right F 56 Trumtic Left F 73 Trumtic Left F 68 Unknown Left M 66 Trumtic Right F 65 Unknown Right M 56 Trumtic Right M 43 Trumtic Left M 69 Trumtic Right M 13 Congenitl Right 1999 Tble 2 MRC/ATS system of grding dyspnoe. Grde 0 NONE No trouble with brething except with strenuous exercise Grde 1 MILD Shortness of breth when hurrying on level or wlking up slight hill Grde 2 MODERATE Wlk slower thn people of sme ge on the level or hs to stop for breth wlking t own pce on the level Grde 3 SEVERE Stop for breth when wlking up to 100 m or fter few minutes on the level Grde 4 VERY SEVERE Too brethless to leve the house or brethless when dressing or undressing Adpted from Mhler et l. [6]. Surgery ws performed under generl nesthesi, with single-lumen intubtion nd limited ero-lterl thorcotomy through the seventh or eighth intercostl spce (Fig. 1). The thorcic cvity nd its structures (lung, medistinum nd phrenic nerve) were crefully exmined to rule out ny unsuspected pthology. The diphrgm ws then plicted in severl rows (normlly four to five), until it becme tught nd firm, using polypropylene U-stitches buttressed with Teflon pledgets to vent cutting through the tissue. Usully, the first row strts t the erior-most portion of the phrenic centre nd runs in rdil fshion. Then, other suture lyers re used to complete the shortening s required (Fig. 2). Redundnt tissue ws flttened both nterior nd erior to the plicture by using running sutures. Extreme cre ws exerted to void injuring bdominl viscer with the sutures. A single intercostl drin ws left in plce nd the Downloded from

3 P. Clvinho et l. / Europen Journl of Crdio-thorcic Surgery 36 (2009) Results 3.1. In-hospitl mortlity nd morbidity Fig. 1. The 6 7 cm muscle-spring thorcotomy. There were no deths in-hospitl or within 30 dys following the procedure. The men hospitl sty ws of dys (rnge: 5 10 dys), with medin of 6 dys, nd dringe time ws dys (rnge: 2 7 dys). The ptient with longer dringe time hd n emphysemtous lung nd prolonged ir-lek. Most of the ptients hd the drin for 2 or 3 dys. One ptient hd nosocomil lobr pneumoni requiring prolonged oxygen therpy. He ws successfully treted with ntibiotics nd ws free from respirtory symptoms t the time of dischrge. With the exception of some nuse relted to mediction, there were no gstrointestinl complictions. Almost ll ptients experienced feeling of tightness/slight pin in the lower chest/upper bdominl re following the procedure, which subsided with nlgesics Follow-up thorcotomy ws closed in lyers. Pin control ws chieved with dorsl/lombr epidurl nlgesi for h or by intercostl blockde using 7.5% ropivcine. All ptients were extubted in the operting thetre immeditely following completion of the procedure Follow-up Ptients were contcted telephoniclly nd invited to come for consulttion t our institution where creful evlution of the dyspnoe score ws done, chronic pin or its equivlent ssessed nd spirometry ws performed. As some ptients were unble to ttend this consulttion, the interview ws conducted telephoniclly nd the spirometry solicited from the ptient s ssistnt physicin. All ptients were ccounted for Sttisticl nlysis Fig. 2. First rows of sutures. Continuous dt re exssed s men stndrd devition. Vlues of VC nd FEV 1 re exssed s percentge dicted for ge nd gender nd body surfce. Follow-up dt were comd with opertive vlues using the Wilcoxon signed-rnk test. The follow-up ws complete for n verge length of months (rnge: months). Nineteen ptients hd follow-up period greter thn 12 months. There were three lte deths (one sudden, one due to stroke nd one trumtic); eight of the 17 survivors (47%) re symptomtic. There re two ptients with chronic pin nd nother one with sthesi in the scr re. The reminder hve different degrees of dyspnoe, s discussed below. Two ptients sented, 75 nd 117 months fter surgery, with recurrence of the eventrtion on the follow-up chest X- ry. The first ptient requires nightly oxygen therpy, is on tretment for chronic pin nd hs dyspnoe grde 3. He refused to undergo surgery gin. The second ptient hd the sme dyspnoe score s before surgery plus the feeling of fullness fter mels. He ws identified t follow-up nd hs since been re-operted, nd since then he is symptomtic Dyspnoe score According to the MRC/ATS grding system (Tble 2), one ptient hd no dyspnoe before surgery. Seven ptients hd dyspnoe grde 3, nd seven grde 2. The remining five ptients hd shortness of breth when hurrying on level or wlking up slight hill. At long-term follow-up, grde of dyspnoe improved 1 point in 10 ptients, 2 points in two, 3 points in two nd remined unchnged in nother two. It worsened 3 points in one ptient. The men dyspnoe score ws opertively nd opertively ( p = 0.007; Tble 3) Spirometry The results of spirometry re summrised in Tbles 3 nd 4. Although the VC nd FEV 1 men vlues were higher fter surgery, the non-prmetric Wilcoxon signed-rnk test showed non-significnt result ( p > 0.05), probbly becuse of the smll size of the smple. Downloded from

4 886 P. Clvinho et l. / Europen Journl of Crdio-thorcic Surgery 36 (2009) Tble 3 Mesured respirtory vlues nd grde of dyspnoe. Ptient VC Tble 4 VC nd FEV 1 vlues, exssed s men stndrd devition of dicted. 4. Discussion VC FEV 1 FEV 1 Dyspnoe VC p vlue FEV 1 p vlue Preopertive Postopertive VC: vitl cpcity; nd FEV 1 : forced expirtory volume in 1 s. Dyspnoe VC: vitl cpcity; FEV 1 : forced expirtory volume in 1 s; : opertive; nd : opertive. Lte deth. Atrophy, thinned wll nd progressive distension of the diphrgm resent the min chrcteristics of diphrgmmtic eventrtion. Newborns usully sent with cute respirtory filure, but in dulthood the most common symptoms re dyspnoe nd thorcic pin. Simnsky et l., in 2002, reported tht the indiction for surgery for ll children ws filure to wen from ventiltory support, while the indictions for surgery in the dult group were ventiltor dependency (n = 4) nd symptomtic dyspnoe (n = 7) [7]. Erlier, Grhm et l. lso indicted shortness of breth s the min symptomtic feture of diphrgmmtic prlysis [8]. Although diphrgmmtic eventrtion nd prlysis re two different entities, the symptomtic consequences nd the therpeutic procedures re very similr, nd the two groups of ptients re quite nlogous. DeVries et l. clerly stte tht phrenic nerve injury inducing chronic diphrgmmtic prlysis leds to eventrtion [9]. A prlysis of the hemidiphrgm cn produce significnt ltertions in the respirtory physiology. These include telectsis nd ventiltion perfusion mismtch, worsening of pulmonry spirometry, ferentil redistribution of pulmonry blood flow to the contrlterl lung, shift of the mobile medistinum to the contrlterl side nd prdoxicl movement of the ffected hemidiphrgm [5]. For prcticl purposes, in most situtions, the differentition of these entities is of limited vlue. The pthophysiology of these entities is equivlent, hence comprison between both groups of ptients with regrds to symptomtology, therpy nd outcomes is possible. Diphrgmmtic pliction is stndrd, well-described technique to tret diphrgmmtic eventrtion [2] but there hve been only limited number of reports published, most with smll series nd short follow-ups. Higgs et l. described group of 19 ptients with long-term follow-up in 15 [10] nd Versteegh et l. reported on 22 ptients with follow-up in 17 [3]. Both clerly proved tht the min nd probbly most importnt result is the significnt nd long-lsting improvement in the subjective symptomtology. To our knowledge, these re the lrgest series published with long follow-ups. In this work, we describe the long-term follow-up of 20 ptients, with mximum of up to 17 yers. Most of our ptients hd n importnt impirment of their dily ctivities opertively, nd ll except three improved their physicl sttus. One ptient referred worsening of the COPD, one hd recurrence of the eventrtion (probble technicl filure) nd the third complined of persistent disbling pin. The reports by Higgs et l. [10] nd Versteegh et l. [3] demonstrted sttisticlly significnt differences between opertive nd opertive vlues of VC nd FEV 1. Our ptients chieved similr improvements, lthough we could not demonstrte sttisticl significnce, probbly ttributble to the smll number of ptients nlysed. Also, with such severe symptomtic disese, one should expect lower vlues thn dicted, opertively, hence with lrger difference to opertive vlues. In ny cse, our vlues re within the rnge reported by most other surgicl groups (65 70% of dicted, opertively, nd 70 79% of dicted, opertively). Recently, diphrgmmtic pliction hs lso been performed through minimlly invsive techniques, either lproscopic [11] or thorcoscopic [5,12 14]. Most of the published ppers on the use of this technique concern cse reports or smll series, except one tht ws published by Freemn et l. [5] which reports on 22 ptients. Our series dtes from 20 yers go when thorcoscopic techniques were not yet widely used, hence we describe the clssicl pproch in this rticle. We strongly believe tht the diphrgm should be rendered s tut s possible, which my not be possible with video-ssisted technique becuse of the lck of full tctile feedbck. The risk of dmge to the bdominl viscer through such thin diphrgm is sent nd is probbly greter with less invsive pproch. To inspect the entire rised diphrgmmtic cupul my be quite difficult. In ny cse, the incidence nd intensity of -thorcotomy pin does not seem to be very different [15]. Obviously, there re no hed-to-hed comprisons to dte [3], nd the nswer to this dilemm will only come with further study. In conclusion, this work demonstrtes tht dult ptients with chronic dyspnoe due to unilterl, non-mlignnt eventrtion significntly benefit from diphrgmmtic plicture. We showed tht there is significnt nd long-lsting clinicl improvement in this group of ptients, with return to norml life fter the surgicl correction. Subjective ssess- Downloded from

5 P. Clvinho et l. / Europen Journl of Crdio-thorcic Surgery 36 (2009) ment in order to estblish the surgicl indiction nd to scertin the clinicl results of the procedure hs been of much more vlue thn the objective dt from the spirometric studies. References [1] Mouroux J, Venissc N, Leo F, Alifno M, Guillot F. Surgicl tretment of diphrgmtic eventrtion using video-ssisted thorcic surgery: prospective study. Ann Thorc Surg 2005;79: [2] Shields TW. Diphrgmtic function, diphrgmtic prlysis, nd eventrtion of the diphrgm. In: Shields TW, Locicero III J, Ponn RB, Rusch VW, editors. Generl Thorcic Surgery. Lippincot Willims & Wilkins; p [3] Versteegh MIM, Brun J, Voigt PG, Bosmn DB, Stolk J, Rbe KF, Dion RAE. Diphrgm pliction in dult ptients with diphrgm prlysis leds to long-term improvement of pulmonry function nd level of dyspne. Eur J Crdiothorc Surg 2007;32: [4] Morrison JMW. Eventrtion of diphrgm due to unilterl phrenic nerve prlysis. Arch Rdiol Electrotherp 1923;28:72 5. [5] Freemn RK, Woznik TC, Fitzgerld EB. Functionl nd physiologic results of video-ssisted thorcoscopic diphrgm pliction in dult ptients with unilterl diphrgm prlysis. Ann Thorc Surg 2006;81: [6] Mhler DA, Weinberg DH, Wells CK, Feinstein AR. The mesurement of dyspne. Chest 1984;85: [7] Simnsky DA, Pley M, Refely Y, Yellin A. Diphrgm pliction following phrenic nerve injury: comprison of peditric nd dult ptients. Thorx 2002;57: [8] Grhm DR, Kpln D, Evns CC, Hind CRK, Donnelly RJ. Diphrgmtic pliction for unilterl diphrgmtic prlysis: 10-yer experience. Ann Thorc Surg 1990;49: [9] DeVries TS, Koens BL, Vos A. Surgicl eventrtion cused by phrenic nerve injury in the newborn. J Peditr Surg 1998;33: [10] Higgs SM, Hussin A, Jckson M, Donnelly RJ, Berrisford RG. Longterm results of diphrgmtic pliction for unilterl diphrgm prlysis. Eur J Crdiothorc Surg 2002;21: [11] Hüttl TP, Wichmnn MW, Reichrt B, Geiger TK, Schildberg FW, Meyer G. Lproscopic diphrgmtic pliction, long-term results of novel technique for opertive phrenic nerve plsy. Surg Endosc 2004;18: [12] Ghrgozloo F, McReynolds SD, Snyder L. Thorcoscopic pliction of the diphrgm. Surg Endosc 1995;9: [13] Hwng Z, Shin JS, Cho YH, Sun K, Lee IS. A simple technique for the thorcoscopic pliction of the diphrgm. Chest 2003;124: [14] Hines MH. Video-ssisted diphrgm pliction in children. Ann Thorc Surg 2003;76: [15] Rogers ML, Duffy JP. Surgicl spects of chronic -thorcotomy pin. A review rticle. Eur J Crdiothorcic Surg 2000;18: Appendix A. Conference discussion Dr S. Eli (Rome, Itly): You sid you hd one sudden deth. Wht ws tht due to? Dr Clvinho: No, I cn t sy it ws sudden deth. It ws during the follow-up, lmost 6 yers fter the surgery. He ws t home nd he just didn t wke up. Dr Eli: So it ws long fter the procedure. Dr Clvinho: Yes, 6 yers fter the procedure. Dr Eli: How did you tret the three ptients who didn t do well? Did you reoperte on them? Dr Clvinho: No. One hd worsening COPD, so he ws not surgicl ptient. In the other one, we hd to replce the diphrgm with Mrlex net becuse the diphrgm just tore off by the knots. It ws probbly technicl filure. The lst ptient is being followed in pin clinic t our institution nd is doing better. Downloded from