Chronic recurrent multifocal osteomyelitis (CRMO) is an extremely rare and most severe form of chronic nonbacterial osteomyelitis of unknown etiology. Here we present the first case of a six-year-old girl in which was observed that the stress fracture mimic osteomyelitic foci in the course of CRMO

Gordan Gulan

Gordana Zamolo

Iva Bilić

Vojko Rožmanić

Zdravko Jotanović

English

Rožmanić, Vojko

Zamolo, Gordana

Gulan, Gordan

Bilić, Iva

Jotanović, Zdravko

Repository of the University of Rijeka

Coll. Antropol. 37 (2013) 4: 1347–1351Case reportTibial Stress Fracture Simulate OsteomyeliticFoci in the Course of Chronic RecurrentMultifocal OsteomyelitisSandor Roth1, Vojko Ro`mani}2, Gordana Zamolo3, Gordan Gulan4, Iva Bili}2 and Zdravko Jotanovi}41 University of Rijeka, School of Medicine, University Hospital Center Rijeka, Department for Children Orthopaedics, Rijeka,Croatia2 University of Rijeka, School of Medicine, University Hospital Center Rijeka, Clinic for Pediatrics, Rijeka, Croatia3 University of Rijeka, School of Medicine, University Hospital Center Rijeka, Department of Pathology, Rijeka, Croatia4 University of Rijeka, School of Medicine, Clinic for Orthopaedic Surgery Lovran, Lovran, CroatiaA B S T R A C TChronic recurrent multifocal osteomyelitis (CRMO) is an extremely rare and most severe form of chronic nonbacterialosteomyelitis of unknown etiology. Here we present the first case of a six-year-old girl in which was observed that thestress fracture mimic osteomyelitic foci in the course of CRMO.Key words: chronic recurrent multifocal osteomyelitis, bone lesions, negative microbiological cultures, stress fractureIntroductionChronic recurrent multifocal osteomyelitis (CRMO) isan autoinflammatory bone disease which was first de-scribed by Giedion et al. in 19721. It is characterized byan insidious onset of bone pain, soft tissue swelling, feverand osteolysis with an unpredictable course of exacerba-tions and spontaneous remissions2,3. CRMO predomi-nantly affects children and adolescents4–6, primarily girlswith a mean age at onset of 8.5 years and a mean time todiagnosis of 8.5 months7, accounting for 2–5% of allosteomyelitis cases8. It is one of more severe types ofchronic nonbacterial osteomyelitis9. The etiology of CRMOis still not elucidated but it is probably due to infection,immune deficiency or autoimmunity2. Several recentstudies indicate contribution of genetic factors to the eti-ology of CRMO10–12. Localization of inflammatory bonelesions is similar to those of hematogenous osteomyelitisin childhood, i.e. metaphyses of long bones13. However,other skeletal localizations have been described as well,such as the vertebral body, the clavicle, the ribs, the ster-num, the sphenoid bones, the pelvis, and the mandi-ble14–16. The diagnosis of this rare entity is difficult be-cause of its vague clinical presentation and course8, andis made after exclusion14. Given the fact that there is nospecific treatment for it, the goal of its management is ef-fective pain control4,5.Case ReportA 6-year-old female patient was admitted to our de-partment due to the pain in her left leg after weight--bearing. Physical evaluation revealed swelling, rednessand tenderness of the skin above the left ankle joint, andlimping. She was afebrile and in good general condition.Her family and personal history was insignificant. Labo-ratory findings showed an elevated white blood cell (WBC)count, which was 20.2 × 109/L, and an elevated erythro-cyte sedimentation rate (ESR), which was 25 mm/h.Other laboratory values were normal. Radiographs of theleft ankle showed two osteolytic foci above the distal tib-ial growth plate. Computerized tomography (CT) of theleft ankle showed two osteolytic areas with sharp scle-rotic edges. Magnetic resonance imaging (MRI) of the leftankle was recommended for better visualization of thecancellous bone and the surrounding bone marrow, par-ticularly due to the vicinity of the physis. It revealed two1347Received for publication May 1, 2012osteolytic foci above the physis involved in the tibialmetaphyses with cortical erosion and signs of swelling ofthe surrounding spongiosis, indicating osteomyelitis (Fi-gure 1). Because of the above-mentioned CT and MRIfindings, we performed open biopsy, trepanation and cu-rettage of the osteolytic foci. The patient was treated byceftriaxone for seven days, when pustules on both fore-arms and shins appeared. Because of suspicion that thepustules were a consequence of an allergic reaction to theantibiotics applied, the treatment was changed to amoxi-cillin in combination with clavulanic acid perorally twotimes daily in doses of 475 mg through seven days.Osteolytic foci smears were negative, both to bacteriaand acid resistant bacteria. Pathohistological findingsshowed acute osteomyelitis (Figure 2). On the 11th post-operative day the wound started to moist. Wound smearwas taken, stitches removal postponed, and the woundwas rebandaged every second day. Cast immobilization ofthe shin without weight-bearing was ordered and shewas discharged from the hospital.The first smear of the wound was sterile. The secondwound smear showed MRSE (methicillin-resistant Sta-phylococcus epidermidis). The patient was readmitted tohospital and was given trimetoprim-sulfometoxasol per-orally two times daily in doses of 480 mg through four-teen days. The wound dried but did not heal completely.Mild, temporary toothache appeared and panoramic ra-diographs of the jaw showed granulomas on the roots oftwo teeth. After extraction of the teeth, the wound on theshin healed completely. The smear of the root of the teethshowed Streptococcus viridans. The smear previouslytaken from the shin revealed MRSE. Evidently, therewas no correlation between the osteomyelitic foci andteeth granulomas. We believed that the MRSE was skinsuperinfection and that the patient had been cured.Four months later, she complained of pain, swellingand redness in the right shin. She was subfebrile with el-evated ESR (27 mm/h) and WBC (11 × 109/L). Radio-graphs of the right shin were negative. Due to suspectedosteomyelits, bed rest and ceftriaxone through 15 dayswere ordered. After that we continued with a 3rd genera-tion cephalosporin perorally through 9 days. Control ra-diographs, bone scan and CT showed stress fracture atthe proximal part of the right tibial diaphysis (Figures3a, 3b and 3c), so we immediately stopped the antibiotictherapy. Two months after, the stress fracture healed af-ter a resting period. Once again we thought that the pa-tient had been cured.However, the patient returned two years later, limp-ing on the right leg and complaining of a pain and mildswelling in the area of the right heel. She was subfebrilewith elevated ESR (26 mm/h). Radiographs, bone scanand MRI showed osteolytic lesions in the talus and cal-caneus (Figures 4a, 4b and 4c). A needle biopsy was con-ducted and the pathohistological findings indicatedosteomyelitis. Microbiological findings were negative.Ibuprofen and clindamycin were administrated perorallythrough 15 days and resulted in resolution of the symp-toms. The recurrence of symptoms, the chronic course,multifocality and lack of evidence of a specific cause ofosteomyelitis led us to suspect CRMO. We also isolatedDNA (deoxyribonucleic acid) for molecular analysis ofthe LPIN2 (lipin 2) gene (exon 4 and 17) because of sus-pected Majeed syndrome. Molecular analysis of theLPIN2 genes showed none of the mutations searched for.A year later, the patient returned complaining of painand mild swelling in the area of the medial condyle of theright tibia. Radiographs, bone scan (Figure 5) and MRIshowed identical changes in the area next to the proxi-mal physis of the right tibia, the left sacrum and ilium tothose found on previous locations. Biopsy of the right tib-ial foci showed the same pathohistological findings andthe smear did not show any bacteria. We treated the pa-tient with symptomatic therapy (effective pain control)and the patient was without symptoms.S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511348Fig. 1. MRI of the left ankle show two osteolytic foci (black ar-rows) above epiphysal cartilage plate involved in the tibial meta-physes with denivelation of cortical bone and signs of swelling ofthe surrounding spongiosis.Fig. 2. Photomicrograph of bone fragment from osteolytic focishow edematous connective tissue stroma (green arrow) amongbone trabecula (blue arrow) with lymphocytes infiltration (redarrow) (hematoxylin and eosin, original magnification x 200).S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511349Fig. 3 a) The control radiographs, b) bone scan, and c) CT showing stress fracture (black arrows) at the proximal part of diaphysis of theright tibia.Fig. 4 a) Lateral radiographs, b) bone scan, and c) MRI of the right ankle showing osteolytic lesions in talus and calcaneus (black arrows).DiscussionCRMO is a well-described but poorly understood clini-cal entity with diagnostic and therapeutic dilemmas. Oneof such poorly understood parts of the puzzle calledCRMO are its long-term sequelae (complications). Wecan tentatively divide them in two main groups. The firstgroup are skeletal complications, such as sclerosis, pre-mature epiphyseal fusion, long bone deformity, growtharrest, progressive kyphosis17,18, »migratory« or »salta-tory« spondylitis which represents spinal involvementfrom the mid-cervical spine to the sacrum, and ulti-mately vertebra plana – vertebral deformity due to com-pression and total collapse19. The second groups are theso-called non-skeletal complications, such as school diffi-culties, psoriasis and the inflammatory bowel disease20.According to our knowledge, no short-term skeletal com-plications of the CRMO, such as stress fracture of thetibia in our case, have been described.A stress fracture is caused by repetitive bone over-loading which exceeds its mechanical capacity21. Thereare two types of stress fractures: fatigue fractures, cau-sed by excessive loads in normal bones, and insufficiencyfractures, caused by normal loads in bones with reducedmechanical properties22,23. When the overload persistsduring the phase of bone weakening, microdamage accu-mulation may also cause a stress fracture24. Pediatricstress fractures are rare25. Approximately one half of allstress fractures in children and adults are tibial frac-tures26,27. Tibial stress fractures are the most commonlower extremity stress fracture26,28. They are especiallycommon in sports involving running and jumping28,29.Pediatric tibial stress fractures usually occur in the ante-rior proximal one third of the bone, whereas in adults,they occur at the junction of the middle and the distalthirds of the tibia27.We believe that in our case the stress fracture of theproximal part of the tibial diaphysis was a consequenceof long-lasting overloading of the right leg and unloadingof the left leg because of a threatening fracture of the dis-tal part of the left tibia after abundant curettage. Also,this stress fracture of the proximal part of the right tibialdiaphysis, being an unusual and so far not describedcomplication in the course of CRMO, was concealed withthe common clinical picture of osteolytic foci.In conclusion, when dealing with CRMO, possibleshort-term skeletal complications, such as stress fractureof the healthy leg after abundant curettage of the oppo-site leg, should always be considered. The above-men-tioned unusual and hitherto not described complicationscan be disguised and easily misdiagnosed with the usualclinical picture of osteolytic foci in the course of CRMO.S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511350Fig. 5. Bone scan show increased radionuclide uptake in the area next to the proximal growth plate of the right tibia and left sacroiliacaljoint (black arrows).R E F E R E N C E S1. GIEDION A, HOLTHUSEN W, MASEL LF, VISCHER D, Ann Ra-diol, 15 (1972) 329. — 2. EL-SHANTI HI, FERGUSON PJ, Clin OrthopRelat Res, 462 (2007) 11. — 3. MIETTUNEN PMH, WEI X, KAURA D,RESLAN WA, AGUIRRE AN, KELLNER JD, Pediatr Rheumatol OnlineJ, 7 (2009) 2. — 4. PIDDO C, REED MH, BLACK GB, Skeletal Radiol, 29(2000) 94. — 5. HUBER AM, LAM PY, DUFFY CM, YEUNG RS, DIT-CHFIELD M, LAXER D, COLE WG, KERR GRAHAM H, ALLEN RC,LAXER RM, J Pediatr, 141 (2002) 198. — 6. JANSSON A, RENNER ED,RAMSER J, MAYER A, HABAN M, MEINDL A, GROTE V, DIEBOLD J,JANSSON V, SCHNEIDER K, BELOHRADSKY BH, Rheumatology (Ox-ford), 46 (2007) 154. — 7. INSALACO A, BOZZOLA E, CAMPANA A, PA-GNOTTA G, TONIOLO RM, CIOFETTA GC, BARBUTI D, CORTIS E(2008) Pediatr Rheumatol, 6 (2008) 188. — 8. BRILAKIS E, AGGOURISK, POTAMITIS N, GKIOKAS A, E.E.X.O.T., 60 (2009) 41. — 9. GIR-SCHICK HJ, RAAB P, SURBAUM S, TRUSEN A, KIRSCHNER S,SCHNEIDER P, PAPADOPOULOS T,MÜLLER-HERMELINK HK, LIP-SKY PE, Ann Rheum Dis, 64 (2005) 279. — 10. GOLLA A, JANSSON A,RAMSER J, HELLEBRAND H, ZAHN R, MEITINGER T, BELOHRAD-SKY BH, MEINDL A, Eur J Hum Genet, 10 (2002) 217. — 11. FERGU-SON PJ, CHEN S, TAYEH MK, OCHOA L, LEAL SM, PELET A, MUN-NICH A, LYONNET S, MAJEED HA, EL-SHANTI H, J Med Genet, 42(2005) 551. — 12. FERGUSON PJ, BING X, VASEF MA, OCHOA LA,MAHGOUB A, WALDSCHMIDT TJ, TYGRETT LT, SCHLUETER AJ,EL-SHANTI H, Bone, 38 (2006) 41.— 13. BJÖRKSTÉN B, BOQUIST L,J Bone Joint Surg Br, 62 (1980) 376.— 14. DABOV G, Osteomyelitis. In:CANALE T. Cambell’s operative orthopaedics (Mosby Inc, St. Louis,USA, 10th ed., 2003). — 15. CHUNCSY, Pediatrics, 113 (2004) 380. — 16.CARR AJ, COLE WG, ROBERTON DM, CHOW CW, J Bone Joint SurgBr, 75 (1993) 582. — 17. BROWN T, WILKINSON RH, Radiology, 166(1988) 493. — 18. KING SM, LAXER RM, MANSON D, GOLD R, PediatrInfect Dis J, 6 (1987) 907. — 19. SCHILLING F, FEDLMEIER M, EC-KARDT A, KESSLER S, Rofo, 174 (2002) 1236. — 20. HUBER AM, LAMPY, DUFFY CM, YEUNG RS, DITCHFIELD M, LAXER D, COLE WG,KERR GRAHAM H, ALLEN RC, LAXER RM, J Pediatr, 141 (2002) 198.— 21. FOTTNER A, BAUR-MELNYK A, BIRKENMAIER C, JANSSONV, DÜRR HR, Int Orthop, 33 (2009) 489. — 22. DAFFNER RH, PAVLOVH, Am J Roentgenol, 159 (1992) 245. — 23. PENTECOST RL, MURRAYRA, BRINDLEY HH, JAMA, 28 (1964) 1001. — 24. STERLING JC,EDELSTEIN DW, CALVO RD, WEBB R, Sports Med, 14 (1992) 336. —25. DE LA CUADRA P, ALBINANA J, Int Orthop, 24 (2000) 47. — 26.BENNELL KL, BRUKNER PD, Clin Sports Med, 16 (1997) 179. — 27.COADY CM, MICHELI LJ, Clin Sports Med, 16 (1997) 225. — 28. MON-TELEONE GP, Orthop Clin North Am, 26 (1995) 423. — 29. MAITRARS, JOHNSON DL, Clin Sports Med, 16 (1997) 259.S. RothUniversity of Rijeka, School of Medicine, University Hospital Center Rijeka, Department for Children Orthopaedics,Istarska 43, 51000 Rijeka, Croatiae-mail: rothmed@ri.htnet.hrPRIJELOM ZAMORA GOLJENICE OPONA[A OSTEOMIJELITI^NO @ARI[TE U TIJEKUKRONI^NOG REKURENTNOG MULTIFOKALNOG OSTEOMIJELITISAS A @ E T A KKroni~ni rekurentni multifokalni osteomijelitis (CRMO) je iznimno rijedak, a ujedno i najte`i oblik kroni~nog ne-bakterijskog osteomijelitisa nepoznate etiologije. U ovom prikazu slu~aja opisujemo prvi slu~aj da prijelom zamoraopona{a osteomijeliti~no `ari{te u tijeku CRMO kod {estogodi{nje djevoj~ice.S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511351

Tibial Stress Fracture Simulate Osteomyelitic Foci in the Course of Chronic Recurrent Multifocal Osteomyelitis

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Coll. Antropol. 37 (2013) 4: 1347–1351Case reportTibial Stress Fracture Simulate OsteomyeliticFoci in the Course of Chronic RecurrentMultifocal OsteomyelitisSandor Roth1, Vojko Ro`mani}2, Gordana Zamolo3, Gordan Gulan4, Iva Bili}2 and Zdravko Jotanovi}41 University of Rijeka, School of Medicine, University Hospital Center Rijeka, Department for Children Orthopaedics, Rijeka,Croatia2 University of Rijeka, School of Medicine, University Hospital Center Rijeka, Clinic for Pediatrics, Rijeka, Croatia3 University of Rijeka, School of Medicine, University Hospital Center Rijeka, Department of Pathology, Rijeka, Croatia4 University of Rijeka, School of Medicine, Clinic for Orthopaedic Surgery Lovran, Lovran, CroatiaA B S T R A C TChronic recurrent multifocal osteomyelitis (CRMO) is an extremely rare and most severe form of chronic nonbacterialosteomyelitis of unknown etiology. Here we present the first case of a six-year-old girl in which was observed that thestress fracture mimic osteomyelitic foci in the course of CRMO.Key words: chronic recurrent multifocal osteomyelitis, bone lesions, negative microbiological cultures, stress fractureIntroductionChronic recurrent multifocal osteomyelitis (CRMO) isan autoinflammatory bone disease which was first de-scribed by Giedion et al. in 19721. It is characterized byan insidious onset of bone pain, soft tissue swelling, feverand osteolysis with an unpredictable course of exacerba-tions and spontaneous remissions2,3. CRMO predomi-nantly affects children and adolescents4–6, primarily girlswith a mean age at onset of 8.5 years and a mean time todiagnosis of 8.5 months7, accounting for 2–5% of allosteomyelitis cases8. It is one of more severe types ofchronic nonbacterial osteomyelitis9. The etiology of CRMOis still not elucidated but it is probably due to infection,immune deficiency or autoimmunity2. Several recentstudies indicate contribution of genetic factors to the eti-ology of CRMO10–12. Localization of inflammatory bonelesions is similar to those of hematogenous osteomyelitisin childhood, i.e. metaphyses of long bones13. However,other skeletal localizations have been described as well,such as the vertebral body, the clavicle, the ribs, the ster-num, the sphenoid bones, the pelvis, and the mandi-ble14–16. The diagnosis of this rare entity is difficult be-cause of its vague clinical presentation and course8, andis made after exclusion14. Given the fact that there is nospecific treatment for it, the goal of its management is ef-fective pain control4,5.Case ReportA 6-year-old female patient was admitted to our de-partment due to the pain in her left leg after weight--bearing. Physical evaluation revealed swelling, rednessand tenderness of the skin above the left ankle joint, andlimping. She was afebrile and in good general condition.Her family and personal history was insignificant. Labo-ratory findings showed an elevated white blood cell (WBC)count, which was 20.2 × 109/L, and an elevated erythro-cyte sedimentation rate (ESR), which was 25 mm/h.Other laboratory values were normal. Radiographs of theleft ankle showed two osteolytic foci above the distal tib-ial growth plate. Computerized tomography (CT) of theleft ankle showed two osteolytic areas with sharp scle-rotic edges. Magnetic resonance imaging (MRI) of the leftankle was recommended for better visualization of thecancellous bone and the surrounding bone marrow, par-ticularly due to the vicinity of the physis. It revealed two1347Received for publication May 1, 2012osteolytic foci above the physis involved in the tibialmetaphyses with cortical erosion and signs of swelling ofthe surrounding spongiosis, indicating osteomyelitis (Fi-gure 1). Because of the above-mentioned CT and MRIfindings, we performed open biopsy, trepanation and cu-rettage of the osteolytic foci. The patient was treated byceftriaxone for seven days, when pustules on both fore-arms and shins appeared. Because of suspicion that thepustules were a consequence of an allergic reaction to theantibiotics applied, the treatment was changed to amoxi-cillin in combination with clavulanic acid perorally twotimes daily in doses of 475 mg through seven days.Osteolytic foci smears were negative, both to bacteriaand acid resistant bacteria. Pathohistological findingsshowed acute osteomyelitis (Figure 2). On the 11th post-operative day the wound started to moist. Wound smearwas taken, stitches removal postponed, and the woundwas rebandaged every second day. Cast immobilization ofthe shin without weight-bearing was ordered and shewas discharged from the hospital.The first smear of the wound was sterile. The secondwound smear showed MRSE (methicillin-resistant Sta-phylococcus epidermidis). The patient was readmitted tohospital and was given trimetoprim-sulfometoxasol per-orally two times daily in doses of 480 mg through four-teen days. The wound dried but did not heal completely.Mild, temporary toothache appeared and panoramic ra-diographs of the jaw showed granulomas on the roots oftwo teeth. After extraction of the teeth, the wound on theshin healed completely. The smear of the root of the teethshowed Streptococcus viridans. The smear previouslytaken from the shin revealed MRSE. Evidently, therewas no correlation between the osteomyelitic foci andteeth granulomas. We believed that the MRSE was skinsuperinfection and that the patient had been cured.Four months later, she complained of pain, swellingand redness in the right shin. She was subfebrile with el-evated ESR (27 mm/h) and WBC (11 × 109/L). Radio-graphs of the right shin were negative. Due to suspectedosteomyelits, bed rest and ceftriaxone through 15 dayswere ordered. After that we continued with a 3rd genera-tion cephalosporin perorally through 9 days. Control ra-diographs, bone scan and CT showed stress fracture atthe proximal part of the right tibial diaphysis (Figures3a, 3b and 3c), so we immediately stopped the antibiotictherapy. Two months after, the stress fracture healed af-ter a resting period. Once again we thought that the pa-tient had been cured.However, the patient returned two years later, limp-ing on the right leg and complaining of a pain and mildswelling in the area of the right heel. She was subfebrilewith elevated ESR (26 mm/h). Radiographs, bone scanand MRI showed osteolytic lesions in the talus and cal-caneus (Figures 4a, 4b and 4c). A needle biopsy was con-ducted and the pathohistological findings indicatedosteomyelitis. Microbiological findings were negative.Ibuprofen and clindamycin were administrated perorallythrough 15 days and resulted in resolution of the symp-toms. The recurrence of symptoms, the chronic course,multifocality and lack of evidence of a specific cause ofosteomyelitis led us to suspect CRMO. We also isolatedDNA (deoxyribonucleic acid) for molecular analysis ofthe LPIN2 (lipin 2) gene (exon 4 and 17) because of sus-pected Majeed syndrome. Molecular analysis of theLPIN2 genes showed none of the mutations searched for.A year later, the patient returned complaining of painand mild swelling in the area of the medial condyle of theright tibia. Radiographs, bone scan (Figure 5) and MRIshowed identical changes in the area next to the proxi-mal physis of the right tibia, the left sacrum and ilium tothose found on previous locations. Biopsy of the right tib-ial foci showed the same pathohistological findings andthe smear did not show any bacteria. We treated the pa-tient with symptomatic therapy (effective pain control)and the patient was without symptoms.S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511348Fig. 1. MRI of the left ankle show two osteolytic foci (black ar-rows) above epiphysal cartilage plate involved in the tibial meta-physes with denivelation of cortical bone and signs of swelling ofthe surrounding spongiosis.Fig. 2. Photomicrograph of bone fragment from osteolytic focishow edematous connective tissue stroma (green arrow) amongbone trabecula (blue arrow) with lymphocytes infiltration (redarrow) (hematoxylin and eosin, original magnification x 200).S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511349Fig. 3 a) The control radiographs, b) bone scan, and c) CT showing stress fracture (black arrows) at the proximal part of diaphysis of theright tibia.Fig. 4 a) Lateral radiographs, b) bone scan, and c) MRI of the right ankle showing osteolytic lesions in talus and calcaneus (black arrows).DiscussionCRMO is a well-described but poorly understood clini-cal entity with diagnostic and therapeutic dilemmas. Oneof such poorly understood parts of the puzzle calledCRMO are its long-term sequelae (complications). Wecan tentatively divide them in two main groups. The firstgroup are skeletal complications, such as sclerosis, pre-mature epiphyseal fusion, long bone deformity, growtharrest, progressive kyphosis17,18, »migratory« or »salta-tory« spondylitis which represents spinal involvementfrom the mid-cervical spine to the sacrum, and ulti-mately vertebra plana – vertebral deformity due to com-pression and total collapse19. The second groups are theso-called non-skeletal complications, such as school diffi-culties, psoriasis and the inflammatory bowel disease20.According to our knowledge, no short-term skeletal com-plications of the CRMO, such as stress fracture of thetibia in our case, have been described.A stress fracture is caused by repetitive bone over-loading which exceeds its mechanical capacity21. Thereare two types of stress fractures: fatigue fractures, cau-sed by excessive loads in normal bones, and insufficiencyfractures, caused by normal loads in bones with reducedmechanical properties22,23. When the overload persistsduring the phase of bone weakening, microdamage accu-mulation may also cause a stress fracture24. Pediatricstress fractures are rare25. Approximately one half of allstress fractures in children and adults are tibial frac-tures26,27. Tibial stress fractures are the most commonlower extremity stress fracture26,28. They are especiallycommon in sports involving running and jumping28,29.Pediatric tibial stress fractures usually occur in the ante-rior proximal one third of the bone, whereas in adults,they occur at the junction of the middle and the distalthirds of the tibia27.We believe that in our case the stress fracture of theproximal part of the tibial diaphysis was a consequenceof long-lasting overloading of the right leg and unloadingof the left leg because of a threatening fracture of the dis-tal part of the left tibia after abundant curettage. Also,this stress fracture of the proximal part of the right tibialdiaphysis, being an unusual and so far not describedcomplication in the course of CRMO, was concealed withthe common clinical picture of osteolytic foci.In conclusion, when dealing with CRMO, possibleshort-term skeletal complications, such as stress fractureof the healthy leg after abundant curettage of the oppo-site leg, should always be considered. The above-men-tioned unusual and hitherto not described complicationscan be disguised and easily misdiagnosed with the usualclinical picture of osteolytic foci in the course of CRMO.S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511350Fig. 5. Bone scan show increased radionuclide uptake in the area next to the proximal growth plate of the right tibia and left sacroiliacaljoint (black arrows).R E F E R E N C E S1. GIEDION A, HOLTHUSEN W, MASEL LF, VISCHER D, Ann Ra-diol, 15 (1972) 329. — 2. EL-SHANTI HI, FERGUSON PJ, Clin OrthopRelat Res, 462 (2007) 11. — 3. MIETTUNEN PMH, WEI X, KAURA D,RESLAN WA, AGUIRRE AN, KELLNER JD, Pediatr Rheumatol OnlineJ, 7 (2009) 2. — 4. PIDDO C, REED MH, BLACK GB, Skeletal Radiol, 29(2000) 94. — 5. HUBER AM, LAM PY, DUFFY CM, YEUNG RS, DIT-CHFIELD M, LAXER D, COLE WG, KERR GRAHAM H, ALLEN RC,LAXER RM, J Pediatr, 141 (2002) 198. — 6. JANSSON A, RENNER ED,RAMSER J, MAYER A, HABAN M, MEINDL A, GROTE V, DIEBOLD J,JANSSON V, SCHNEIDER K, BELOHRADSKY BH, Rheumatology (Ox-ford), 46 (2007) 154. — 7. INSALACO A, BOZZOLA E, CAMPANA A, PA-GNOTTA G, TONIOLO RM, CIOFETTA GC, BARBUTI D, CORTIS E(2008) Pediatr Rheumatol, 6 (2008) 188. — 8. BRILAKIS E, AGGOURISK, POTAMITIS N, GKIOKAS A, E.E.X.O.T., 60 (2009) 41. — 9. GIR-SCHICK HJ, RAAB P, SURBAUM S, TRUSEN A, KIRSCHNER S,SCHNEIDER P, PAPADOPOULOS T, MÜLLER-HERMELINK HK, LIP-SKY PE, Ann Rheum Dis, 64 (2005) 279. — 10. GOLLA A, JANSSON A,RAMSER J, HELLEBRAND H, ZAHN R, MEITINGER T, BELOHRAD-SKY BH, MEINDL A, Eur J Hum Genet, 10 (2002) 217. — 11. FERGU-SON PJ, CHEN S, TAYEH MK, OCHOA L, LEAL SM, PELET A, MUN-NICH A, LYONNET S, MAJEED HA, EL-SHANTI H, J Med Genet, 42(2005) 551. — 12. FERGUSON PJ, BING X, VASEF MA, OCHOA LA,MAHGOUB A, WALDSCHMIDT TJ, TYGRETT LT, SCHLUETER AJ,EL-SHANTI H, Bone, 38 (2006) 41. — 13. BJÖRKSTÉN B, BOQUIST L,J Bone Joint Surg Br, 62 (1980) 376. — 14. 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DE LA CUADRA P, ALBINANA J, Int Orthop, 24 (2000) 47. — 26.BENNELL KL, BRUKNER PD, Clin Sports Med, 16 (1997) 179. — 27.COADY CM, MICHELI LJ, Clin Sports Med, 16 (1997) 225. — 28. MON-TELEONE GP, Orthop Clin North Am, 26 (1995) 423. — 29. MAITRARS, JOHNSON DL, Clin Sports Med, 16 (1997) 259.S. RothUniversity of Rijeka, School of Medicine, University Hospital Center Rijeka, Department for Children Orthopaedics,Istarska 43, 51000 Rijeka, Croatiae-mail: rothmed@ri.htnet.hrPRIJELOM ZAMORA GOLJENICE OPONA[A OSTEOMIJELITI^NO @ARI[TE U TIJEKUKRONI^NOG REKURENTNOG MULTIFOKALNOG OSTEOMIJELITISAS A @ E T A KKroni~ni rekurentni multifokalni osteomijelitis (CRMO) je iznimno rijedak, a ujedno i najte`i oblik kroni~nog ne-bakterijskog osteomijelitisa nepoznate etiologije. U ovom prikazu slu~aja opisujemo prvi slu~aj da prijelom zamoraopona{a osteomijeliti~no `ari{te u tijeku CRMO kod {estogodi{nje djevoj~ice.S. Roth et al.: Tibial Stress Fracture in the Course Of CRMO, Coll. Antropol. 37 (2013) 4: 1347–13511351

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Tibial Stress Fracture Simulate Osteomyelitic Foci in the Course of Chronic Recurrent Multifocal Osteomyelitis

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