Bootsma, H.

Dagal, E. F.

Delli, K.

Spijkervet, F. K. L.

Vissink, A.

English

University of Groningen

   University of GroningenPatient-reported change of sensibility and pain after parotid and labial gland biopsy appliedfor primary Sjogren's syndrome diagnosticsDelli, K.; Dagal, E. F.; Bootsma, H.; Vissink, A.; Spijkervet, F. K. L.Published in:Clinical and Experimental RheumatologyIMPORTANT NOTE: You are advised to consult the publisher's version (publisher's PDF) if you wish to cite fromit. Please check the document version below.Document VersionPublisher's PDF, also known as Version of recordPublication date:2018Link to publication in University of Groningen/UMCG research databaseCitation for published version (APA):Delli, K., Dagal, E. F., Bootsma, H., Vissink, A., & Spijkervet, F. K. L. (2018). Patient-reported change ofsensibility and pain after parotid and labial gland biopsy applied for primary Sjogren's syndromediagnostics: One-year follow-up study. Clinical and Experimental Rheumatology, 36(Suppl. 112), S173-S176. http://www.clinexprheumatol.org/abstract.asp?a=12935CopyrightOther than for strictly personal use, it is not permitted to download or to forward/distribute the text or part of it without the consent of theauthor(s) and/or copyright holder(s), unless the work is under an open content license (like Creative Commons).The publication may also be distributed here under the terms of Article 25fa of the Dutch Copyright Act, indicated by the “Taverne” license.More information can be found on the University of Groningen website: https://www.rug.nl/library/open-access/self-archiving-pure/taverne-amendment.Take-down policyIf you believe that this document breaches copyright please contact us providing details, and we will remove access to the work immediatelyand investigate your claim.Downloaded from the University of Groningen/UMCG research database (Pure): http://www.rug.nl/research/portal. For technical reasons thenumber of authors shown on this cover page is limited to 10 maximum.S-173Clinical and Experimental Rheumatology 20181Oral and Maxillofacial Surgery; 2Rheumatology and Clinical Immunology, University of Groningen, University Medical Centre Groningen, The Netherlands.Konstantina Delli, DDS, Dr Med Dent, PhDEssam F. Dagal, MDHendrika Bootsma, MD, PhDArjan Vissink, DDS, MD, PhDFred K.L. Spijkervet, DDS, PhDPlease address correspondence to:Dr Fred K.L. Spijkervet, Department of Oral and Maxillofacial Surgery, University of Groningen, University Medical Centre Groningen, HPC BB70, P.O. Box 30.001, 9700 RB Groningen, The Netherlands.E-mail: f.k.l.spijkervet@umcg.nlReceived on April 25, 2018; accepted in revised form on June 4, 2018.Clin Exp Rheumatol 2018; 36 (Suppl. 112): S173-S176.© Copyright CliniCal and ExpErimEntal rhEumatology 2018.Key words: Sjögren’s syndrome, biopsy, pain, patient satisfactionCompeting interests: none declared.ABSTRACTObjective. To assess how patients per-ceived pain and change of sensibility of the biopsied area after having under-gone parotid and labial gland biopsy as part of the diagnostic work-up of primary Sjögren’s syndrome (pSS).Methods. Simultaneously, parotid and labial salivary gland biopsies were tak-en under local anesthesia. One week, 6 months and 12 months post-opera-tively, each patient was sent a postal questionnaire to quantify the severity of pain and change of sensibility in the biopsied areas with a visual analogue scale (VAS; range 0-100). Results. 110 patients were included. The median age of patients was 54 years (IQR=47-65) and 92% were fe-male. Changes in sensibility and pain in the biopsied area were significantly higher after a parotid gland biopsy than after a labial gland biopsy at one week and 6 months post-operatively, but rather minor in both areas. At 12 months post-operatively, the change in sensibility and pain level was negligi-ble in most patients and comparable for both biopsied areas. The duration of the technique, outcome of the biopsy, exposure of nerve branches during the biopsy and bleeding during the biopsy did not affect the reported change of sensibility or pain in the biopsied area. ESSPRI was not related to pain level or change of sensibility at any time point (r<0.3 and p>0.05).Conclusion. Patient-reported post-op-erative change of sensibility and pain in the area of the parotid and labial gland biopsy are minor and compara-ble. Parotid and labial gland biopsies are diagnostic techniques well toler-ated by patients suspected with pSS.IntroductionPrimary Sjögren’s syndrome (pSS) is a chronic, systemic autoimmune disease, which is characterised by inflammation of the exocrine glands and prevalence of 0.05% (1). Most patients with pSS suffer from xerostomia, keratocon-junctivitis sicca and extreme fatigue (2). Currently, several criteria sets are available for the classification of pSS, with the American College of Rheu-matology-European League Against Rheumatism (ACR-EULAR) criteria being endorsed by both ACR and EU-LAR (3). Biopsy of the labial salivary glands is generally considered as one of the most important items of the ACR-EULAR criteria, carries a weight of 3 out of 4 and is considered the cor-nerstone of the diagnosis of pSS.The diagnostic accuracy of the parotid gland biopsy is comparable to the la-bial gland biopsy when applied for pSS diagnostics (4). Parotid biopsy demon-strates next to the labial salivary gland biopsy a number of additional advan-tages, i.e. repeated biopsies from the same parotid gland are possible, parot-id biopsy can predict patients’ respon-siveness to rituximab treatment (5) and since MALT lymphomas occur more frequently in parotid glands, they can be easily diagnosed by parotid gland biopsy. Additionally parotid gland ul-trasound can be directly compared to parotid gland histopathology (6). Not-withstanding these aforementioned ad-vantages, biopsies of the parotid gland have not become a common place because of concerns for damaging fa-cial nerve branches, development of sialoceles or salivary fistulae and high-er morbidity than labial gland biopsies, concerns which so far have never been confirmed or proven (7). Therefore, the aim of this study was to assess the perceived pain and change of sensibility in the biopsied area after a parotid and labial gland biopsy per-formed as part of a routine diagnostic work-up of pSS. Patient-reported change of sensibility and pain after parotid and labial gland biopsy applied for primary Sjögren’s syndrome diagnostics: one-year follow-up studyK. Delli1, E.F. Dagal1, H. Bootsma2, A. Vissink1, F.K.L. Spijkervet1S-174 Clinical and Experimental Rheumatology 2018Biopsy in Sjögren’s syndrome / K. Delli et al.Patients and methodsPatientsThis was a prospective, observa-tional, single-centre study. Patients were recruited at the multidisciplinary Sjögren’s expertise centre, University Medical Centre Groningen (UMCG), the Netherlands. All patients were ≥18 years and were clinically suspected with pSS. Patients were included if they were willing to be subjected to a complete diagnostic work-up according to the ACR-EULAR criteria including a labial and parotid biopsy. Previous radiation to head and neck area, confirmed hepatitis C infection, human immunodeficiency virus (HIV) infection, sarcoidosis, graft versus host disease (GvHD), oral can-cer and pregnancy based on self-report were considered as exclusion criteria. The study protocol was approved by the ethics committee of the University Medical Center Groningen (METC ap-proval: 2013.066). All patients provided their written informed consent.MethodsThe parotid gland biopsy and labial gland biopsies were simultaneously tak-en under local anesthesia and with the use of loupe glass magnification (3.5x) (7, 8) as part of a routine diagnostic work up for pSS. All biopsies were per-formed by a single surgeon (F.K.L.S.).Pre-operatively, on the day of the biop-sies, patients were asked if the sensibil-ity of the pre-auricular and labial area was normal. Possible complications during the biopsy, e.g. bleeding, unin-tended exposure of nerves branches and duration of each biopsy were recorded. Additionally, the patients filled in the European League Against Rheumatism (EULAR) Sjögren’s syndrome Patient-reported Index (ESSDAI) prior to the biopsies. One week, 6 months and 12 months post-operatively, each patient was sent a postal questionnaire. The question-naire comprised 4 questions quanti-fying the patient-reported severity of pain in biopsied areas and the change of sensibility (Supplementary Figure 1). Patients were asked to state their experience using a visual analogue scale (VAS; range: 0–100).  Addition-ally, patients were asked to indicate Fig. 2. Duration of salivary gland biopsies. Horizon-tal lines indicate median values.Fig. 1. Flowchart of patients’ response to questionnaires. S-175Clinical and Experimental Rheumatology 2018Biopsy in Sjögren’s syndrome / K. Delli et al.which biopsied area caused more post-operative complaints. Detailed written instructions were given to patients on how complete the questionnaire. Statistical analysisStatistical analyses were performed using IBM SPSS Statistics 23 (SPSS, Chicago, IL, USA). Descriptive pa-rameters were expressed as number of patients (%) and median (Interquartile range (IQR)=Q1-Q3) when appropri-ate. Mann-Whitney U-test was used to evaluate differences in VAS scores between the parotid and labial gland biopsy. Wilcoxon signed-rank test was used to compare differences over time. Spearman’s  correlation coefficient (r) was used to analyse the relationship between ESSPRI and the VAS scores. P-values<0.05 were considered statis-tically significant. ResultsOne hundred and fifty-six patients suspected with pSS were invited to participate, of which 110 consented to undergo both parotid and labial gland biopsy as part of a routine diagnostic pSS work-up. Four patients had to be excluded from further evaluation for the purposes of this study because they presented at the day of the biopsy with an already altered sensibility of the biopsied area. The median age of patients was 54 years (IQR=47-65) and 92% were female. A flowchart of patients’ response to questionnaires is shown in Figure 1. Analysis of missing data did not detect a pattern on patients response, indicating a completely ran-dom type of missing data. The median duration of the surgical procedure (from incision to complete closure of the wound) of parotid gland biopsy was significantly longer than of the lip (9.5 min (IQR=8.6-10.5) vs. 5.4 min (IQR=4.7-7.0), p<0.001; Fig. 2). Bipolar electrocautery was needed to control bleeding during 4 (4%) parotid and 2 (2%) labial gland biopsy proce-dures, while exposure of sensory nerve branches was detected in 5 (5%) parot-id (branches of the auricular temporal nerve, no branches of the facial nerve) and 68 (64%) labial (branches of the mental nerve) gland biopsies. None of Fig. 3A. Change of sensibility VAS score of patients after biopsies. Horizontal lines indicate medians.Fig. 3B. Pain VAS score of patients after biopsies. Horizontal lines indicate medians.Fig. 4. Type of biopsy perceived by patients (%) as most bothersome.S-176 Clinical and Experimental Rheumatology 2018Biopsy in Sjögren’s syndrome / K. Delli et al.the patients experienced post-operative bleeding, paresis of the facial nerve, fistula or sialoceles formation.  Patient-reported changes in the sensi-bility of the biopsied area were more common after a parotid biopsy than after a labial salivary gland biopsy at one week and 6 months post-opera-tively. Although significantly different, the change of sensibility in the area of the labial and parotid gland was minor for both types of biopsy. At 12 months post-operatively, the change in sensi-bility of the biopsied areas was minor and comparable in both biopsied areas (Fig. 3A; Supplementary Table I).Patients experienced significantly more pain in the area of the parotid gland biopsy than in the area of the labial gland biopsy at one week and 6 months post-operative visits, but again pain in both areas was at the low levels. At 12 months postoperatively, pain level was very low in most patients and compa-rable for both biopsied areas (Fig. 3B; Supplementary Table II). Figure 4 shows which biopsied area was perceived as the most bothersome at each time-point. Labial biopsies were experienced as less bothersome than parotid biopsies by the majority of patients one 1 week and 6 months post-operatively (12.7% and 19.5%, respec-tively). Twelve months post-operative-ly, half of the patients did not report any of the biopsied areas as annoying. The duration of the technique, out-come of the biopsy, exposure of nerve branches during the biopsy and bleed-ing during the biopsy did not affect the reported change of sensibility or pain in the biopsied area. ESSPRI and ESS-PRI-pain domain did not correlate with the pain level or change of sensibility at any time point (r<0.3 and p>0.05).DiscussionThis study showed that at 12 months post-operatively pain was very low and comparable in both biopsied ar-eas. These results are in agreement with the study of Pijpe et al. (4), who assessed the post-operative pain in a study including 15 patients with pSS and 20 healthy controls. Pijpe et al. (4) reported, however, a similar pattern also at one week and six months post-operatively, while in the present study patients reported significantly less pain in the area of the labial gland biopsy compared to the parotid at these time points. Number of patients participat-ing in the studies and the fact that we included patients suspected with pSS, while Pijpe et al. (4) included patients already diagnosed with pSS and com-pared them to healthy controls, might explain to some extend the discrepancy between both studies.  As far as sensibility is concerned, this study showed that one week post-oper-atively patients reported less change in the area of the labial gland biopsy com-pared to the parotid and that 12 months post-operatively change of sensibility was comparable in both biopsied ar-eas. Likewise, Pijpe et al. (4) reported that at one week post-operatively fewer patients had paresthesia in the area of the lip biopsy compared to the parotid, i.e. 11% versus 26%, respectively. At 6 months post-operatively, however, in our study change of sensibility, al-though still at the lower levels, was sig-nificantly higher in the area of the pa-rotid gland biopsy compared to the lip, i.e. VAS for change of sensibility was 25.7 versus 7.0, respectively, while Pi-jpe et al. (4) showed that none of the patients had any complaints of pares-thesia at the area of the parotid gland biopsy. All the reasons mentioned in the aforementioned paragraph could justify the difference again. Additional-ly, it has to be acknowledged that in the present study, change of sensibility was assessed and quantified by the patients themselves, based on the questionnaire sent to their home, while in the study of Pijpe et al. (4) change of sensibil-ity was assessed by physical testing of the sensibility and was not based on a questionnaire. Whether physicians’ as-sessment reflects patients’ perception of loss of sensibility remains question-able (9). On the other hand, patients’ understanding of questionnaires is also questionable, since even the apparently most simple questions are subject to misinterpretation (10).  ConclusionPost-operative patient-reported change of sensibility and pain in the area of the parotid and labial gland biopsy are mi-nor and comparable. Labial and parotid gland biopsies are diagnostic tech-niques both well tolerated by patients suspected with pSS.AcknowledgementsThe authors wish to thank Ms Janita Bulthuis-Kuiper, clinical research co-ordinator, Department of Rheumatol-ogy and Clinical Immunology, for kindly mailing the questionnaires to the patients.References  1. QIN B, WANG J, YANG Z et al.: Epidemiology of primary Sjögren’s syndrome: a systematic review and meta-analysis. Ann Rheum Dis 2015; 74: 1983-9.  2. FOX RI: Sjögren’s syndrome. Lancet 2005; 366: 321-31.  3. SHIBOSKI CH, SHIBOSKI SC, SEROR R et al.: 2016 American College of Rheumatology/European League Against Rheumatism clas-sification criteria for primary Sjögren’s syn-drome. Ann Rheum Dis 2017; 76: 9-16.  4. PIJPE J, KALK WW, van der WAL JE et al.: Parotid gland biopsy compared with labial biopsy in the diagnosis of patients with pri-mary Sjögren’s syndrome. Rheumatology 2007; 46: 335-41.  5. DELLI K, HAACKE EA, KROESE FG et al.: Towards personalized treatment in primary Sjögren’s syndrome: baseline parotid histo-pathology predicts responsiveness to rituxi-mab treatment. Ann Rheum Dis 2016; 75: 1933-8.  6. MOSSEL E, DELLI K, van NIMWEGEN JF et al.: Ultrasonography of major salivary glands compared with parotid and labial gland biopsy and classification criteria in patients with clinically suspected primary Sjögren’s syndrome. Ann Rheum Dis 2017; 76: 1883-9.  7. DELLI K, VISSINK A, SPIJKERVET FK: Sali-vary gland biopsy for Sjögren’s syndrome. Oral Maxillofac Surg Clin North Am 2014; 26: 23-33.   8. SPIJKERVET FK, HAACKE E, KROESE FG, BOOTSMA H, VISSINK A: Parotid Gland Biopsy, the alternative way to diagnose Sjögren’s Syndrome. Rheum Dis Clin North Am 2016; 42: 485-99.  9. CHISHOLM CD, WEAVER CS, WHENMOUTH LF, GILES B, BRIZENDINE EJ: A comparison of observed versus documented physician assessment and treatment of pain: the physi-cian record does not reflect the reality. Ann Emerg Med 2008; 52: 383-9. 10. RAGAB AA: Validity of self-assessment out-come questionnaires: patient-physician dis-crepancy in outcome interpretation. Biomed Sci Instrum 2003; 39: 579-84.

Patient-reported change of sensibility and pain after parotid and labial gland biopsy applied for primary Sjogren's syndrome diagnostics:One-year follow-up study

https://pure.rug.nl/ws/files/67095505/article_3_.pdf

Patient-reported change of sensibility and pain after parotid and labial gland biopsy applied for primary Sjogren's syndrome diagnostics:One-year follow-up study

Abstract

Similar works

Full text

Available Versions

University of Groningen