An Overlooked Potentially Treatable Disorder: Idiopathic Mesenteric Panniculitis

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1 Originl Pper Med Princ Prct 27;26: DOI:.59/48465 Received: Jnury 3, 27 Accepted: October 26, 27 Published online: October 26, 27 An Overlooked Potentilly Tretble Disorder: Idiopthic Mesenteric Pnniculitis Abdurrhmn Shin Hkn Arts b Yesim Eroglu b Nurettin Tunc Ulvi Demirel Ibrhim Hlil Bhcecioglu Mehmet Ylniz Division of Gstroenterology nd Heptology nd b Deprtment of Rdiology, Firt University School of Medicine, Elzig, Turkey Significnce of the Study In this study, ptients with idiopthic mesenteric pnniculitis (MP) presented with severe pin nd were successfully treted with nonsteroidl nti-inflmmtory drugs (NSAID) nd ntibiotics. This finding indictes tht the inflmmtion in this ptient subgroup might be of n infectious origin. Hence, ntibiotics nd NSAID could be considered in the tretment of symptomtic MP cses. Keywords Idiopthic mesenteric pnniculitis Tretment Antibiotics Nonsteroidl nti-inflmmtory drugs Abstrct Objective: The im of this study ws to determine the prevlence of mesenteric pnniculitis (MP) nd to describe its clinicl chrcteristics, therpy, nd outcome. Subjects nd Methods: This retrospective study ws crried out mong ptients with MP bsed on computed tomogrphy (CT) scns from Jnury 22 to December 25. The CT imges were renlyzed by study rdiologists to confirm the previous MP dignosis. Ptients were divided into 2 groups, i.e., idiopthic nd secondry, bsed on the presence or bsence of ssocited predisposing fctors such s trum, mlignncy, utoimmune disorders, ischemi, or previous b- dominl surgery. The clinicl chrcteristics of the 2 groups, s well s tretments, were ssessed. Results: Among the 9,869 CT scns, 36 ptients (.8%) with MP were identified (i.e., 9 [53%] femles nd 7 [47%] mles). The medin ge ws 54 yers (rnge 26 76). Twenty-four ptients (67%) were ctegorized into the idiopthic group. Mlignncy ws the predisposing fctor in 8 (22%) of those ptients. Furthermore, bdominl pin ws the crdinl symptom observed in 22 ptients (92%) in the idiopthic group. In the idiopthic group, 5 ptients (63%) were treted with ntibiotics nd 6 (67%) were treted with nonsteroidl nti-inflmmtory drugs (NSAID). One unresponsive ptient ws treted with colchicine. Symptomtic relief ws chieved in ll of the treted ptients. Conclusion: In this study, symptomtic idiopthic subgroup of ptients with MP did not hve ny ssocited disorder. The response to tretment with ntibiotics nd NSAID ws effective in most of the ptients. Bsed E-Mil krger@krger.com 27 The Author(s) Published by S. Krger AG, Bsel This is n Open Access rticle licensed under the Cretive Commons Attribution-NonCommercil-4. Interntionl License (CC BY-NC) ( pplicble to the online version of the rticle only. Usge nd distribution for commercil purposes requires written permission. Abdurrhmn Shin Division of Gstroenterology nd Heptology Firt University School of Medicine Yunus Emre Avenue 2, TR 239 Elzig (Turkey) E-Mil firt.edu.tr

2 on these findings, nti-inflmmtory tretments beyond NSAID nd surgery should be reserved for ptients who re unresponsive to ntibiotics nd NSAID. Introduction 27 The Author(s) Published by S. Krger AG, Bsel Mesenteric pnniculitis (MP) is rre, nonspecific fibroinflmmtory disorder of unknown etiology tht ffects the dipose tissue of the root of the mesentery []. It consists of 2 pthologicl subgroups: inflmmtory nd fibrotic lesions. When inflmmtion nd ft necrosis predominte over fibrosis, the condition is known s MP. Conversely, when fibrosis nd retrction predominte, it is clled sclerosing mesenteritis [ 3]. The gold stndrd for the dignosis of MP is to demonstrte inflmmtory nd fibrotic chnges in the mesentery histopthologiclly [3]. However, MP is usully suspected with MP-like findings on computed tomogrphy (CT) scns incidentlly during investigtions into other disorders [4, 5]. In lrge series, imging findings consistent with MP were reported in.6 2.5% of ptients undergoing bdominl CT for vrious indictions [6, 7]. In ddition to symptomtic cses, MP could be ssocited with severl symptoms, including bdominl pin, nuse, vomiting, dirrhe, constiption, fever, weight loss, nd chylous scites [8]. Although it is frequently mnged conservtively, tretment is wrrnted in dvnced or progressive cses [9]. Vrious drugs hve been used to tret MP, including corticosteroids, colchicine, cyclophosphmide, zthioprine, tmoxifen, nd thlidomide, s well s hormonl therpies [, ]. However, the previous therpeutic interventions hve shown vrying degrees of success. Surgery is nother option in refrctory cses [2, 3]. MP is thought to be ssocited with trum, bdominl mlignncy, utoimmune disorders, ischemi, or previous bdominl surgery [8]. Mlignncy is the most commonly cited disorder in reltion to MP [6]. The im of this retrospective nlysis ws to describe the clinicl chrcteristics of ptients with MP nd to investigte its mngement nd outcome. Subjects nd Methods This retrospective study ws crried out t the Firt University Hospitl, Elzig, Turkey. The rdiologicl reports of ptients who hd undergone bdominl CT nd were ged 8 yers of were obtined from the hospitl s dtbse using the keywords mesenteric pnniculitis nd sclerosing mesenteritis from Jnury 22 to December 25. The CT imges were subsequently ressessed by rdiologists (H.A. nd Y.E.) to confirm the presence of rdiologic chrcteristic fetures of MP. The rdiologic dignosis of MP ws defined ccording to the study of Coulier [5], which defined 5 typicl CT signs: (i) the presence of well-defined mss effect on neighboring structures, (ii) constituted by mesenteric ft tissue of n inhomogeneous higher ttenution thn djcent retroperitonel or mesocolonic ft, (iii) contining smll soft tissue nodes, (iv) ftty hlo sign indicting the preservtion of hlo of ft round the involved vessels; nd (v) hyper-ttenuting pseudocpsule. The dignosis of MP ws estblished if 3 of these signs were present (Fig., b). Demogrphic fetures, complints, clinicl fetures, nd tretments were lso retrieved from the hospitl dtbse. Lbortory mesurements, including white blood cell counts, hemoglobin, pltelet counts, the men pltelet volume nd red cell distribution width, fsting glucose, liver function tests, blood ure nitrogen, cretinine, nd totl protein nd lbumin vlues were lso obtined from the hospitl dtbse. The study popultion ws divided into 2 subgroups: ptients with MP secondry to mlignncy, such s rheumtologic disese or history of previous bdominl surgery, nd ptients with no relted disorder. The ltter group ws clled idiopthic MP. Ethicl pprovl for this study ws obtined from the Institutionl Review Bord of the Firt University Fculty of Medicine. Sttisticl Anlysis Descriptive sttistics were used to nlyze the dt. Ctegoricl vribles re expressed s numbers (%). Continuous vribles re expressed s medins (rnge). Ctegoricl vribles were compred using the χ 2 test or Fischer s exct test. Differences between continuous vribles were nlyzed using the Mnn-Whitney U test. p <.5 ws considered sttisticlly significnt. Sttisticl nlyses were performed using the Sttisticl Pckge for the Socil Sciences (SPSS ) for Windows (v. 2.; SPSS, USA). Results Among totl of 9,869 bdominl CT exmintions, 36 ptients (.8%) were dignosed with MP. Of those 36 ptients, 9 (53%) were femle nd 7 (47%) were mle. The medin ge of the women ws 54 yers (rnge 26 76), nd tht of the men ws 54 yers (rnge 32 76). Of the 36 ptients, 24 (67%) were in the idiopthic group nd 2 (33%) were in the secondry group. The mjor complint ws bdominl pin, which ws present in 26 (72%) ptients with MP. Other MP-relted symptoms were nuse nd vomiting in (3%) ptient nd dirrhe in nother (3%) ptient. Bsed on the dignostic criteri of Coulier [5], sign 2 nd sign 3 were present in the whole study popultion. Twenty-four ptients (66%) hd mss effect (sign ). A hyperttenuting pseudocpsule (sign 5) ws evident in 29 (8%) ptients. The lest common rdiologic feture 568 Med Princ Prct 27;26: DOI:.59/48465 Shin/Arts/Eroglu/Tunc/Demirel/ Bhcecioglu/Ylniz

3 Tble. Comprison of idiopthic mesenteric pnniculitis nd secondry mesenteric pnniculitis Idiopthic group p Ptients Age, yers Femle/mle rtio (26 76) 55.5 (34 72).65 /3 8/4.3 Crdinl symptoms Asymptomtic Abdominl pin Fever nd dysuri Dyspne Nuse-vomiting Dirrhe 2 (8) 22 (92) 6 (5) 4 (33) (8) (8) 2 (8) (8) Abdominopelvic surgery Secondry group Associted disorders Mlignncy Colorectl crcinom Pncres crcinom Renl cell crcinom Bldder crcinom Brest crcinom Over crcinom Rheumtologic disorders Ankylosing spondylitis Scleroderm Miscellneous Portl vein thrombosis Nephrolithisis Acute cholecystitis Dibetes mellitus Coronry rteril disese COPD Hert filure Vlues re presented s numbers, medins (rnge), or numbers (%) unless otherwise stted. COPD, chronic obstructive pulmonry disese. b Fig.. Seventy-six-yer-old mn with chronic hert filure nd cute cholecystitis. Contrst-enhnced xil () nd coronl (b) bdominl computed tomogrphy imges showing mesenteric pnniculitis s well-circumscribed, inhomogeneous ftty mss displying higher ttenution thn norml retroperitonel ft with hyperttenuting pseudocpsule (rrowheds). ws ftty hlo sign (sign 4), which ws detected in only 2 (5.6%) ptients. The secondry group ws composed of 2 ptients in whom MP ws found in conjunction with selected disorders or mjor surgery. The men ge ws 55.5 yers (rnge Idiopthic MP 34 72) nd 4 of them were men. Six of them were symptomtic t the time of CT exmintion. The complint of ptient who hd been dignosed with brest cncer ws dyspne. Fever nd dysuri relted to urinry trct infection were the dmission symptoms in nother ptient with bldder crcinom. Four ptients in this group hd bdominl pin (2 ptients hd pncres crcinom). One ptient hd previous bdominl surgery due to intestinl perfortion nd ptient hd portl vein thrombosis. MP ws relted to mlignncy in 8 (22%) ptients, i.e., 2 with colorectl crcinom, nother 2 with pncretic crcinoms, nd 4 ech with renl cell crcinom, Med Princ Prct 27;26: DOI:.59/

4 Tble 2. Demogrphic nd clinicl chrcteristics nd tretments of ptients with idiopthic mesenteric pnniculitis Ptient No. Age, Sex Symptoms yers Associted disorder Tretment Other informtion 34 F Asymptomtic None None 2 63 M Asymptomtic DM None duodenl diverticul, nephrolithisis 3 5 M Right lower qudrnt pin DM Etodolc 6 mg po bid for 8 dys 4 76 F Epigstric pin None Dexketoprofen 25 mg po bid for dys 5 54 M Periumbilicl pin None Dexketoprofen 5 mg i.v. bid for dys nd IBS-relted symptoms moxifloxcin, then colchicine 6 45 F Suprpubic pin None Dexketoprofen 25 mg po bid for 3 dys 7 48 M Left flnk pin CAD Nimesulid mg po bid for 7 dys 8 43 F Periumbilicl pin None Diclofenc 5 mg po bid for 7 dys nd ciprofloxcin 9 6 M Left upper qudrnt pin, dirrhe CAD Ciprofloxcin 63 F Epigstric pin CAD, HT Nproxen 75 mg po qd for dys 58 F Left flnk pin None Cefpodoxime 2 52 M Left flnk pin None Flurbiprofen mg po bid for 8 dys 3 59 M Right flnk pin DM Ceftrixone 4 56 F Epigstric pin None Diclofenc 75 mg po bid for 5 dys 5 45 M Left flnk pin None Dexketoprofen 5 mg i.v. bid for 3 dys nd then dexketoprofen 25 mg i.v. bid 5 dys nd cefuroxime 6 32 M Periumbilicl pin None Ceftrixone 7 65 M Right lower qudrnt pin None Dexketoprofen 5 mg i.v. bid for 4 dys nd then Ureterolithisis dexketoprofen 25 mg i.v. bid for 5 dys nd cefuroxime 8 35 F Right flnk pin None Nproxen 75 mg po qd for 8 dys nd cefuroxime Nephrolithisis 9 7 F Periumbilicl pin, nuse-vomiting CAD, COPD Dexketoprofen 5 mg i.v. bid for 3 dys nd ceftrixone 2 49 M Left lower qudrnt pin None Cefuroxime Nephrolithisis 2 6 F Left flnk pin DM Ciprofloxcin Nephrolithisis nd UTI (Pseudomons eruginos) M Right upper qudrnt pin, Hert filure Dexketoprofen 25 mg i.v. bid for 5 dys nd ceftrixone Acute cholecystitis nuse-vomiting M Periumbilicl pin None Nproxen 75 mg po qd for 5 dys nd moxicillin/ clvulnic cid F Left lower qudrnt pin None Dexketoprofen 5 mg i.v. qd for 2 dys nd ceftrixone bid, twice dily; CAD, coronry rteril disese; COPD, chronic obstructive pulmonry disese; DM, dibetes mellitus; F, femle; HT, hypertension; M, mle; qd, once dily; UTI, urinry trct infection; po, orlly; IBS, irritble bowel syndrome; UTI, urinry trct infection. bldder crcinom, ovrin crcinom, nd brest crcinom. Cncer dignosis nd surgicl interventions hd preceded the dignosis of MP in ll the ptients with mlignncy. Abdominl surgery for intestinl perfortion ws considered the leding cuse of MP in ptient. Only 2 ptients in the idiopthic group hd cholecystectomy fter the dignosis of MP. Demogrphic nd clinicl dt re presented in Tble. Of the 24 ptients in the idiopthic group, 3 (54.2%) were men, nd the medin ge ws 53 yers (rnge, yers). The medin ges of idiopthic group nd secondry group did not differ (p =.65). Despite femle dominnce (n = 8; 67%) in the secondry MP group, femle ptients ccounted for bout hlf of the idiopthic MP group (n = ; 46%). However, it did not rech sttisticl significnce (p =.3). Five (2%) ptients in the idiopthic group hd nephrolithisis t presenttion, while ptient ws lso dignosed with cute cholecystitis. As opposed to the symptomtic presenttions of ptients in the secondry group, the mjority (n = 22; 92%) of ptients in the idiopthic group presented with bdominl pin. The demogrphic nd clinicl dt of the idiopthic MP ptients re presented in Tble 2. Most of those ptients were dmitted to the hospitl with severe loclized bdominl pin. In ddition to bdominl pin, ptient suffered from nuse nd severe vomiting nd nother hd dirrhe in conjunction with pin. Weight loss, fever of unknown origin, constiption, nd scites were not noted in the idiopthic MP popultion. The tretment of these ptients minly consisted of NSAID for pin relief nd ntibiotics. Antibiotics were prescribed in 5 ptients, nd NSAID were used in 6 ptients. The pre- 57 Med Princ Prct 27;26: DOI:.59/48465 Shin/Arts/Eroglu/Tunc/Demirel/ Bhcecioglu/Ylniz

5 scribed ntibiotics were cephlosporins in ptients, quinolones in 4 ptients, nd moxicillin/clvulnic cid in ptient. All of the ptients becme symptom free, except for ptient in whom colchicine tretment ws strted fter lck of response to tretment with NSAID nd moxifloxcin. Tht ptient s severe bdominl pin decresed grdully fter 6-month tretment with colchicine. Becuse ptients in the secondry group were symptomtic or hd symptoms relted to primry disorders, dditionl tretments were not given to them. There ws no difference between the 2 groups regrding white blood cell counts, hemoglobin, pltelet counts, liver function tests, or cretinine. The medin protein nd lbumin levels were lso lower in the secondry MP group thn in the idiopthic group (7.3 vs. 6.9; p =.5, nd 4.3 vs. 4.; p =.4, respectively). The erythrocyte sedimenttion rte nd C-rective protein vlues were not vilble for the entire study popultion. Histopthologicl smpling of MP hd not been performed in the ptients with MP. Discussion In this study, the frequency of MP ws.8% mong ptients undergoing bdominl CT exmintion. The ptients with MP showed slight femle predominnce nd two thirds of them hd no obvious relted disorder. The incidence of MP hs been reported to be in the rnge of.6 3.4% in previous studies [5, 7]. The frequency of MP in this study (i.e.,.8%) is close to the.6% previously reported [7] but lower thn 3.4% [5]. The differences could be due to the methodology of this study, i.e., retrospective or prospective. Some of the retrospective studies used the keywords mesenteric pnniculitis or pnniculitis or sclerosing mesenteritis for screening of the CT reports [2, 6], while others renlyzed ll CT scns for the presence of MP [5, 7]. Becuse the ptients were selected bsed on rdiologic reports in the current study, we might hve hd lower prevlence of MP in the study popultion. Although the gold stndrd for the dignosis of MP is histopthologicl exmintion of the mesentery, none of our ptients underwent histopthologicl exmintion nd we considered them to hve MP ccording to the MPlike findings on CT exmintions [3]. The hllmrk of this study ws tht the ptients in the idiopthic group were treted with NSAID nd/or severl ntibiotics. Although proof of the bcteril infection ws evident in only 2 ptients, symptomtic relief ws chieved in ll of the ptients except. Responses to brief nti-inflmmtory nd nti-infective tretments could indicte tht the trigger event might be self-limiting inflmmtion or infection tht spred from djcent structures to the mesenteric dipose tissue in most ptients. Although no cler proof exists tht specific microorgnism contributes to the development of MP, predisposition to immunosuppression, which is seen in mlignncy or utoimmune disorders relted to the disese itself or to its tretments, might explin why MP is more common in the immunosuppressive group of ptients. Severl typicl microorgnisms such s Cryptococcus neoformns nd Mycobcterium genvense hve been reported in HIV ptients in reltion to MP [4, 5]. On the other hnd, M. tuberculosis nd Vibrio choler re suspected to be the infectious gents tht cuse MP in immunocompetent ptients [6, 7]. In ddition, vrious cute bdominl inflmmtory disorders in which enteric bcteri ws involved in the etiopthogenesis, such s diverticulitis nd cute ppendicitis, hve been reported in reltion to MP [8]. The response to ntibiotics could indicte tht if the MP is of n infectious origin the microorgnisms might spred from the gut or genitourinry trct. Bcteril trnsloction of microorgnisms from the gut or urinry trct might loclize n occult infection in djcent mesenteric dipose tissue, thereby leding in the development of MP. Another probble explntion for cute cses my be virl enteritis [9]. The importnce of intestinl microbiot nd innte immunity in the pthogenesis of IBD hs been better understood in recent yers [2]. MP shres some fetures of dipose tissue chnges in IBD, such s ft wrpping in Crohn s disese nd ft hlo sign in Crohn s disese nd ulcertive colitis [8, 2]. Mesenteric dipose tissue itself, through dipokines, might be responsible for low-grde inflmmtion nd subsequent fibrosis. The results of the current study could indicte tht this rre entity hs nturl history different from tht in Western countries presumbly due to lower snitry conditions, infections with severl unidentified microorgnisms, nd genetic differences [7, 8]. Therefore, there is need for epidemiologicl studies from Asin nd Africn countries in order to obtin more ccurte epidemiologic dt on MP. This retrospective study hd some limittions; the CT dtbse ws serched using the terms mesenteric pnniculitis nd sclerosing mesenteritis to define the cohort. However, some ptients could hve been overlooked, nd the prevlence of MP therefore could be underestimted. MP ws dignosed rdiologiclly nd none of the ptients hd histopthologic dignosis of MP nd there ws lck of follow-up CT exmintions, especilly in the idiopthic group. Idiopthic MP Med Princ Prct 27;26: DOI:.59/

6 Conclusion In this study, symptomtic idiopthic subgroup of ptients with MP did not hve ssocited disorders. Antibiotics nd NSAID tretment ws effective in most of the ptients. Bsed on these findings, nti-inflmmtory tretments beyond NSAID nd surgery should be reserved for ptients who re unresponsive to ntibiotics nd NSAID. Disclosure Sttement The uthors declre no conflicts of interest. References Coulier B: Mesenteric pnniculitis. Prt : MDCT pictoril review. JBR-BTR 2; 94: Bdet N, Silley N, Briquez C, et l: Mesenteric pnniculitis: still n mbiguous condition. Dign Interv Imging 25; 96: Emory TS, Monihn JM, Crr NJ, et l: Sclerosing mesenteritis, mesenteric pnniculitis nd mesenteric lipodystrophy: single entity? Am J Surg Pthol 997; 2: Horton KM, Lwler LP, Fishmn EK: CT findings in sclerosing mesenteritis (pnniculitis): spectrum of disese. Rdiogrphics 23; 23: Coulier B: Mesenteric pnniculitis. Prt 2: prevlence nd nturl course: MDCT prospective study. JBR-BTR 2; 94: Wilkes A, Griffin N, Dixon L, et l: Mesenteric pnniculitis: prneoplstic phenomenon? Dis Colon Rectum 22; 55: vn Putte-Ktier N, vn Bommel EF, Elgersm OE, et l: Mesenteric pnniculitis: prevlence, clinicordiologicl presenttion nd 5-yer follow-up. Br J Rdiol 24; 87: Hussein MR, Abdelwhed SR: Mesenteric pnniculitis: n updte. Expert Rev Gstroenterol Heptol 25; 9: vn Bred Vriesmn AC, Schuttever HM, Coerkmp EG, et l: Mesenteric pnniculitis: US nd CT fetures. Eur Rdiol 24; 4: Bl A, Coderre SP, Johnson DR, et l: Tretment of sclerosing mesenteritis with corticosteroids nd zthioprine. Cn J Gstroenterol 2; 5: Akrm S, Prdi DS, Schffner JA, et l: Sclerosing mesenteritis: clinicl fetures, tretment, nd outcome in ninety-two ptients. Clin Gstroenterol Heptol 27; 5: ; quiz Endo K, Moroi R, Sugimur M, et l: Refrctory sclerosing mesenteritis involving the smll intestinl mesentery: cse report nd literture review. Intern Med 24; 53: Msulovic D, Jovnovic M, Ivnovic A, et l: Sclerosing mesenteritis presenting s pseudotumor of the greter omentum. Med Princ Prct 26; 25: Alonso Socs MM, Vlls RA, Gomez Sirvent JL, et l: Mesenteric pnniculitis by cryptococcl infection in n HIV-infected mn without severe immunosuppression. AIDS 26; 2: Borde JP, Offensperger WB, Kern WV, et l: Mycobcterium genvense specific mesenteritic syndrome in HIV-infected ptients: new entity of retrctile mesenteritis? AIDS 23; 27: Ege G, Akmn H, Ckiroglu G: Mesenteric pnniculitis ssocited with bdominl tuberculous lymphdenitis: cse report nd review of the literture. Br J Rdiol 22; 75: Roginsky G, Mzulis A, Ecnow JS, et l: Mesenteric pnniculitis ssocited with Vibrio cholere infection. ACG Cse Rep J 25; 3: Smith ZL, Sifuentes H, Deepk P, et l: Reltionship between mesenteric bnormlities on computed tomogrphy nd mlignncy: clinicl findings nd outcomes of 359 ptients. J Clin Gstroenterol 23; 47: Ehrenpreis ED, Roginsky G, Gore RM: Clinicl significnce of mesenteric pnniculitislike bnormlities on bdominl computerized tomogrphy in ptients with mlignnt neoplsms. World J Gstroenterol 26; 22: Ohkus T, Koido S: Intestinl microbiot nd ulcertive colitis. J Infect Chemother 25; 2: Amiti MM, Arzi-Kleinmn T, Avidn B, et l: Ft hlo sign in the bowel wll of ptients with Crohn s disese. Clin Rdiol 27; 62: Med Princ Prct 27;26: DOI:.59/48465 Shin/Arts/Eroglu/Tunc/Demirel/ Bhcecioglu/Ylniz