SHORT COMMUNICATION
Chronic Recurrent Multifocal Osteomyelitis and Inflammatory Bowel Disease
Grace K. Audu, yKornilia Nikaki, zDaniel Crespi, yChristine Spray, and Jenny Epstein
ABSTRACT Chronic recurrent multifocal osteomyelitis (CRMO) has been reported in association with inflammatory bowel disease (IBD), mostly in children. We describe the UK paediatric experience of CRMO and IBD and review the global literature. Three cases of CRMO and IBD were identified in UK children during the last 10 years. This adds to the previously published 24 cases worldwide (15 children). We provide further evidence for the true association of CRMO and IBD, and a greater understanding of disease course. CRMO may be considered a rare extraintestinal complication of IBD. Key Words: chronic recurrent multifocal osteomyelitis, Crohn disease, inflammatory bowel disease, SAPHO syndrome, ulcerative colitis
(JPGN 2015;60: 586–591)
What Is Known
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare, sterile bone inflammatory disorder that occurs at multiple skeletal sites. CRMO has been reported in association with IBD in children and (mostly) young adults.
What Is New
The UK experience of CRMO in association with IBD in children is described. We provide evidence for a strong greater-thanchance association of CRMO and IBD. We evaluate the disease course and natural history of the 2 conditions. CRMO can be recognised as a rare extraintestinal complication of IBD.
Received September 30, 2014; accepted December 3, 2014. From the Department of Paediatric Gastroenterology, Chelsea and Westminster Hospital, London, the yDepartment of Paediatric Gastroenterology, Bristol Royal Hospital for Children, Bristol, and the zDepartment of Paediatric Gastroenterology, Royal Free Hospital, London, UK. Address correspondence and reprint requests to Dr Jenny Epstein, MBChB, PhD, Chelsea and Westminster Hospital, 369 Fulham Rd, London SW10 9NH, UK (e-mail: [email protected]). The authors report no conflicts of interest. Copyright # 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition DOI: 10.1097/MPG.0000000000000663
C
hronic recurrent multifocal osteomyelitis (CRMO) is a rare disease of children and adolescents characterised by aseptic inflammation of long bones. Metaphyses are most commonly affected, although lesions can occur anywhere in the skeleton (1). It was first described by Giedion et al in 1972 (2). Patients typically present with gradual onset of bone pain, local tenderness, and swelling. Symptoms are usually self-limiting with episodes of exacerbation and remission. Some authors describe presentation with nonspecific constitutional symptoms, and investigations in these patients reveal elevated erythrocyte sedimentation rate (ESR), leucocytosis and radiologically lytic bone lesions. Culture of the lesions is sterile and symptoms do not respond to classical antimicrobial treatments. The aetiology of CRMO is unknown but an autoimmune process is implicated, with predisposing genetic factors and some rare syndromic forms (1). First-line treatment of CRMO is with nonsteroidal antiinflammatory drugs (NSAIDs), which induce response in the majority of patients (3). (Where CRMO occurs with inflammatory bowel disease (IBD), clearly NSAIDs are relatively contraindicated in view of their recognised association with colitis flare.) Other treatments used include corticosteroids, methotrexate, sulfasalazine, colchicine, and azithromycin (4). There are no comparative or randomised trials to inform therapeutic choice, and few prospective studies. More recently, several groups have reported success with antitumour necrosis factor (TNF) agents and bisphosphonates (reviewed by Ferguson and Sandu (1)), in children who fail first-line management. The response to pamidronate at approximately 80% appears particularly promising, and may be considered as second-line therapy in children with refractory CRMO. As a caveat, bisphosphonate therapy carries the risk of osteonecrosis and atypical fracture, and its effect on growing bones is unknown. Associations of CRMO with other diseases have been reported. These include IBD, both ulcerative colitis (UC) and Crohn disease (CD), parenchymal lung disease, pyoderma gangrenosum, Wegener granulomatosis, psoriasis, and Sweet syndrome (febrile neutrophilic dermatosis) (5). The majority of CRMO cases are reported in children and adolescents, with only a small number of adult cases described (6–9). Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome (SAPHO) appears to be a condition related to CRMO and may represent its adult form (1,9,10). Bony lesions in SAPHO preferentially involve the anterior chest wall and spine (11). A modest number of case reports of CRMO in association with IBD have previously been published. Here we report our experience of a UK case series of 3 children with CRMO and IBD (Table 1), and we review the world literature on the subject.
METHODS UK cases were identified via a nationwide email survey of members of the British Society of Paediatric Gastroenterology,
586 JPGN Volume 60, Number 5, May 2015 Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 60, Number 5, May 2015
Chronic Recurrent Multifocal Osteomyelitis and IBD
TABLE 1. UK case series of CRMO in association with IBD
Case no.
Age, sex of patient
Which condition was diagnosed first? (time)
IBD type
Autoantibodies
ESR, mm/h
Treatment used for IBD
1 2 3
9 y, M 10 y, F 2 y, M
CRMO (1 mo) CRMO (2 y) CRMO (3 y)
UC CD CD
pANCA positive ANA positive pANCA negative
66 72 45
Steroid, 5-ASA Steroid (for CRMO), no other treatment for CD Steroid, 5-ASA, diet
ANA ¼ anti-nuclear antibodies; ASA ¼ 5-aminosalicylic acid; CD ¼ Crohn disease; CRMO ¼ chronic recurrent multifocal osteomyelitis; ESR ¼ erythrocyte sedimentation rate; IBD ¼ inflammatory bowel disease; pANCA ¼ perinuclear anti-neutrophil cytoplasmic antibodies; UC ¼ ulcerative colitis.
Hepatology, and Nutrition (BSPGHAN). Each of the 3 cases identified were subject to detailed case note review. MEDLINE and Embase were interrogated for ‘‘chronic recurrent multifocal osteomyelitis’’ and ‘‘inflammatory bowel disease.’’ All of the articles from 1972 (first publication) to 2014 were retrieved, with no age limits or language restriction applied. After excluding duplicates, the resulting 184 articles were examined by 2 authors for their relevance. Twelve articles were selected for inclusion in our systematic review (11 English language and 1 German), which reported 24 cases of CRMO in association with IBD. One case (12) was excluded because the case had been reported in another article by the same authors (13). A
UK CASE REPORTS Case 1 A 9-year-old boy of Bangladeshi origin presented with right ankle pain and swelling. He had been diagnosed as having right ankle sprain 6 weeks earlier. There was a 2- to 3-month history of intermittent abdominal pain. C-reactive protein (CRP) was 9 mg/L, ESR 66 mm/h, white cell count (WCC) 13.9 109/L, and platelets 410 109/L. X-ray of the right ankle showed soft tissue swelling over the lateral malleolus. Magnetic resonance imaging (MRI) of the right leg showed features of osteomyelitis in the right tibia, fibula, and calcaneus (Fig. 1A). A 6-week course of intravenous ceftriaxone was commenced. He subsequently developed left ankle
B
MRI right ankle
C
MRI right ankle
D
MRI right elbow
Bone scan E
F
Colonoscopy active disease
Colonoscopy remission
FIGURE 1. A–C, MRI showing osteomyelitis of right ankle and right elbow. D, Bone scan showing increased uptake at distal right humerus, distal right tibia and fibula, and left distal radial epiphysis. E, Colonoscopy during active disease. F, Colonoscopy in remission. MRI ¼ magnetic resonance imaging.
www.jpgn.org
587
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
Epstein et al
JPGN
pain. Repeat MRI and full-body bone scan showed features in keeping with active CRMO involving the distal right humerus, distal right tibia and fibula, and left distal radial epiphysis (Fig. 1B–D). ELISPOT and Mantoux were negative, bone biopsy showed active chronic inflammation only, and all of the cultures were negative. Abdominal pain persisted, and a month later, he developed bloody diarrhoea. He underwent upper and lower gastrointestinal (GI) endoscopy, which showed mild focal active colitis (Fig. 1E). He was given hydrocortisone enemas, oral mesalazine, and oral steroids, which were gradually weaned. His GI symptoms settled, and a repeat endoscopy 3 months later showed mild microscopic focal inflammation in the caecum only (Fig. 1F). Presently, 17 months after the first presentation, he continues on maintenance mesalazine only. His bone pain, abdominal pain, and diarrhoea have resolved, and his inflammatory markers are within normal limits (ESR 10 mm/h).
Case 2 A 10-year-old white girl presented with a 3-week history of lower back pain and difficulty walking. Investigations showed ESR 72 mm/h and CRP 23 mg/L. Spinal MRI showed 2 areas of signal abnormality in the sacral spine (S1 and S2) with a pathological fracture of the superior end plate of S1 (Fig. 2A–C). Bone biopsy confirmed CRMO with negative bacterial cultures. Whole-body MRI showed distal tibial changes bilaterally, and changes in the right tibial metaphysis, cervicothoracic junction, and right sacroiliac joint were consistent with the diagnosis of CRMO. Her bone pains were managed with NSAIDs. Two years later, her weight had dropped from 9th to 0.4th percentile. There was a history of occasional upper abdominal burning sensation. Investigations showed albumin 44 g/L, faecal calprotectin 492 mg/g, and a positive antineutrophil antibody. Upper and lower GI endoscopy showed eosinophilic oesophagitis and moderately active terminal ileitis in keeping with CD. Small bowel MRI showed no more widespread small bowel disease. She became more symptomatic from her bone pains. Repeat MRI
A
Volume 60, Number 5, May 2015
showed progression of her CRMO, and she was treated with oral steroids and pamidronate infusion. Her IBD remains asymptomatic several years later, and she is presently receiving no specific treatment for this; however, anti-TNF treatment is under consideration to manage her CRMO.
Case 3 A 2-year-old white boy presented with pyrexia and a limp. He had a red swollen right ankle and left first toe. Haemoglobin (Hb) was 8.8 g/dL, WCC 20.9 109/L, platelets 579 109/L, CRP 169 mg/L, and albumin 30 g/L. He subsequently developed multiple bone lesions in >10 separate locations, involving upper and lower limbs bilaterally (Fig. 3A and B), as well as nonspecific rashes and recurrent splenic abscesses (Fig. 3C). His bone and splenic lesions were aspirated and intravenous antibiotics administered, although all of the aspirate cultures were negative. He received intravenous immunoglobulins (IVIGs) and his symptoms improved. During the following year, he continued to have inflammatory repeat activations of his bony lesions and recurrent splenic abscesses. At the age of 3 years, he received a second dose of IVIG to which he had an allergic reaction. He was treated with oral prednisolone and partial splenectomy. His bone symptoms and radiological lesions subsided, although he experienced further episodes of sterile splenic abscesses, treated with intermittent oral steroids for the ensuing 2 years. At 5 years, he developed perianal skin tags and fissures, abdominal pain, vomiting, and faecal urgency. There was a seconddegree family history of UC. Inflammatory markers, blood counts, and albumin were normal. He underwent proctoscopic examination. Histology showed inflamed anal epithelium with underlying nonnecrotising granulomatous proctitis, whereas rectal mucosal biopsies showed no inflammation. He was treated with metronidazole. Chronic granulomatous disease was excluded by neutrophil oxidative burst test, and detailed immunological function tests were normal. Small bowel contrast imaging was normal. Upper and lower GI endoscopy was macroscopically and histologically normal. His perianal disease was treated with topical tacrolimus,
B
MRI pelvis coronal
C
MRI pelvis axial
MRI spine sagittal
FIGURE 2. A–C, MRI of spine showing 2 areas of signal abnormality in the sacral spine (S1 and S2) with a pathological fracture of the superior end plate of S1. MRI ¼ magnetic resonance imaging.
588
www.jpgn.org
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 60, Number 5, May 2015 A
Chronic Recurrent Multifocal Osteomyelitis and IBD B
X-ray left ankle: lucency and periosteal reaction
C
Bone scan lower limbs: anterior view
D
White cell scan: photopaenic splenic lesions
Colonoscopy
FIGURE 3. A, X-rays of left ankle showing lucent bone lesions and periosteal reaction. B, Bone scan of lower limbs showing multiple areas of increased uptake. C, Abdominal white cell scan showing photopaenic lesions in the spleen. D, Colonoscopy showing scattered aphthous ulcers in the transverse colon.
prednisolone, and mesalazine suppositories. Symptoms persisted, and he underwent repeat upper and lower GI endoscopy at 7 years of age, which was again normal. At 9 years, he underwent his third upper and lower GI endoscopy because he developed loose stools, abdominal pain, static weight, and hypoalbuminaemia (31 g/L). ESR was 45 mm/h, CRP 18 years), although 3 of these were first diagnosed as having either CRMO or IBD when they were
Chronic Recurrent Multifocal Osteomyelitis and Inflammatory Bowel Disease
Grace K. Audu, yKornilia Nikaki, zDaniel Crespi, yChristine Spray, and Jenny Epstein
ABSTRACT Chronic recurrent multifocal osteomyelitis (CRMO) has been reported in association with inflammatory bowel disease (IBD), mostly in children. We describe the UK paediatric experience of CRMO and IBD and review the global literature. Three cases of CRMO and IBD were identified in UK children during the last 10 years. This adds to the previously published 24 cases worldwide (15 children). We provide further evidence for the true association of CRMO and IBD, and a greater understanding of disease course. CRMO may be considered a rare extraintestinal complication of IBD. Key Words: chronic recurrent multifocal osteomyelitis, Crohn disease, inflammatory bowel disease, SAPHO syndrome, ulcerative colitis
(JPGN 2015;60: 586–591)
What Is Known
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare, sterile bone inflammatory disorder that occurs at multiple skeletal sites. CRMO has been reported in association with IBD in children and (mostly) young adults.
What Is New
The UK experience of CRMO in association with IBD in children is described. We provide evidence for a strong greater-thanchance association of CRMO and IBD. We evaluate the disease course and natural history of the 2 conditions. CRMO can be recognised as a rare extraintestinal complication of IBD.
Received September 30, 2014; accepted December 3, 2014. From the Department of Paediatric Gastroenterology, Chelsea and Westminster Hospital, London, the yDepartment of Paediatric Gastroenterology, Bristol Royal Hospital for Children, Bristol, and the zDepartment of Paediatric Gastroenterology, Royal Free Hospital, London, UK. Address correspondence and reprint requests to Dr Jenny Epstein, MBChB, PhD, Chelsea and Westminster Hospital, 369 Fulham Rd, London SW10 9NH, UK (e-mail: [email protected]). The authors report no conflicts of interest. Copyright # 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition DOI: 10.1097/MPG.0000000000000663
C
hronic recurrent multifocal osteomyelitis (CRMO) is a rare disease of children and adolescents characterised by aseptic inflammation of long bones. Metaphyses are most commonly affected, although lesions can occur anywhere in the skeleton (1). It was first described by Giedion et al in 1972 (2). Patients typically present with gradual onset of bone pain, local tenderness, and swelling. Symptoms are usually self-limiting with episodes of exacerbation and remission. Some authors describe presentation with nonspecific constitutional symptoms, and investigations in these patients reveal elevated erythrocyte sedimentation rate (ESR), leucocytosis and radiologically lytic bone lesions. Culture of the lesions is sterile and symptoms do not respond to classical antimicrobial treatments. The aetiology of CRMO is unknown but an autoimmune process is implicated, with predisposing genetic factors and some rare syndromic forms (1). First-line treatment of CRMO is with nonsteroidal antiinflammatory drugs (NSAIDs), which induce response in the majority of patients (3). (Where CRMO occurs with inflammatory bowel disease (IBD), clearly NSAIDs are relatively contraindicated in view of their recognised association with colitis flare.) Other treatments used include corticosteroids, methotrexate, sulfasalazine, colchicine, and azithromycin (4). There are no comparative or randomised trials to inform therapeutic choice, and few prospective studies. More recently, several groups have reported success with antitumour necrosis factor (TNF) agents and bisphosphonates (reviewed by Ferguson and Sandu (1)), in children who fail first-line management. The response to pamidronate at approximately 80% appears particularly promising, and may be considered as second-line therapy in children with refractory CRMO. As a caveat, bisphosphonate therapy carries the risk of osteonecrosis and atypical fracture, and its effect on growing bones is unknown. Associations of CRMO with other diseases have been reported. These include IBD, both ulcerative colitis (UC) and Crohn disease (CD), parenchymal lung disease, pyoderma gangrenosum, Wegener granulomatosis, psoriasis, and Sweet syndrome (febrile neutrophilic dermatosis) (5). The majority of CRMO cases are reported in children and adolescents, with only a small number of adult cases described (6–9). Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome (SAPHO) appears to be a condition related to CRMO and may represent its adult form (1,9,10). Bony lesions in SAPHO preferentially involve the anterior chest wall and spine (11). A modest number of case reports of CRMO in association with IBD have previously been published. Here we report our experience of a UK case series of 3 children with CRMO and IBD (Table 1), and we review the world literature on the subject.
METHODS UK cases were identified via a nationwide email survey of members of the British Society of Paediatric Gastroenterology,
586 JPGN Volume 60, Number 5, May 2015 Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 60, Number 5, May 2015
Chronic Recurrent Multifocal Osteomyelitis and IBD
TABLE 1. UK case series of CRMO in association with IBD
Case no.
Age, sex of patient
Which condition was diagnosed first? (time)
IBD type
Autoantibodies
ESR, mm/h
Treatment used for IBD
1 2 3
9 y, M 10 y, F 2 y, M
CRMO (1 mo) CRMO (2 y) CRMO (3 y)
UC CD CD
pANCA positive ANA positive pANCA negative
66 72 45
Steroid, 5-ASA Steroid (for CRMO), no other treatment for CD Steroid, 5-ASA, diet
ANA ¼ anti-nuclear antibodies; ASA ¼ 5-aminosalicylic acid; CD ¼ Crohn disease; CRMO ¼ chronic recurrent multifocal osteomyelitis; ESR ¼ erythrocyte sedimentation rate; IBD ¼ inflammatory bowel disease; pANCA ¼ perinuclear anti-neutrophil cytoplasmic antibodies; UC ¼ ulcerative colitis.
Hepatology, and Nutrition (BSPGHAN). Each of the 3 cases identified were subject to detailed case note review. MEDLINE and Embase were interrogated for ‘‘chronic recurrent multifocal osteomyelitis’’ and ‘‘inflammatory bowel disease.’’ All of the articles from 1972 (first publication) to 2014 were retrieved, with no age limits or language restriction applied. After excluding duplicates, the resulting 184 articles were examined by 2 authors for their relevance. Twelve articles were selected for inclusion in our systematic review (11 English language and 1 German), which reported 24 cases of CRMO in association with IBD. One case (12) was excluded because the case had been reported in another article by the same authors (13). A
UK CASE REPORTS Case 1 A 9-year-old boy of Bangladeshi origin presented with right ankle pain and swelling. He had been diagnosed as having right ankle sprain 6 weeks earlier. There was a 2- to 3-month history of intermittent abdominal pain. C-reactive protein (CRP) was 9 mg/L, ESR 66 mm/h, white cell count (WCC) 13.9 109/L, and platelets 410 109/L. X-ray of the right ankle showed soft tissue swelling over the lateral malleolus. Magnetic resonance imaging (MRI) of the right leg showed features of osteomyelitis in the right tibia, fibula, and calcaneus (Fig. 1A). A 6-week course of intravenous ceftriaxone was commenced. He subsequently developed left ankle
B
MRI right ankle
C
MRI right ankle
D
MRI right elbow
Bone scan E
F
Colonoscopy active disease
Colonoscopy remission
FIGURE 1. A–C, MRI showing osteomyelitis of right ankle and right elbow. D, Bone scan showing increased uptake at distal right humerus, distal right tibia and fibula, and left distal radial epiphysis. E, Colonoscopy during active disease. F, Colonoscopy in remission. MRI ¼ magnetic resonance imaging.
www.jpgn.org
587
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
Epstein et al
JPGN
pain. Repeat MRI and full-body bone scan showed features in keeping with active CRMO involving the distal right humerus, distal right tibia and fibula, and left distal radial epiphysis (Fig. 1B–D). ELISPOT and Mantoux were negative, bone biopsy showed active chronic inflammation only, and all of the cultures were negative. Abdominal pain persisted, and a month later, he developed bloody diarrhoea. He underwent upper and lower gastrointestinal (GI) endoscopy, which showed mild focal active colitis (Fig. 1E). He was given hydrocortisone enemas, oral mesalazine, and oral steroids, which were gradually weaned. His GI symptoms settled, and a repeat endoscopy 3 months later showed mild microscopic focal inflammation in the caecum only (Fig. 1F). Presently, 17 months after the first presentation, he continues on maintenance mesalazine only. His bone pain, abdominal pain, and diarrhoea have resolved, and his inflammatory markers are within normal limits (ESR 10 mm/h).
Case 2 A 10-year-old white girl presented with a 3-week history of lower back pain and difficulty walking. Investigations showed ESR 72 mm/h and CRP 23 mg/L. Spinal MRI showed 2 areas of signal abnormality in the sacral spine (S1 and S2) with a pathological fracture of the superior end plate of S1 (Fig. 2A–C). Bone biopsy confirmed CRMO with negative bacterial cultures. Whole-body MRI showed distal tibial changes bilaterally, and changes in the right tibial metaphysis, cervicothoracic junction, and right sacroiliac joint were consistent with the diagnosis of CRMO. Her bone pains were managed with NSAIDs. Two years later, her weight had dropped from 9th to 0.4th percentile. There was a history of occasional upper abdominal burning sensation. Investigations showed albumin 44 g/L, faecal calprotectin 492 mg/g, and a positive antineutrophil antibody. Upper and lower GI endoscopy showed eosinophilic oesophagitis and moderately active terminal ileitis in keeping with CD. Small bowel MRI showed no more widespread small bowel disease. She became more symptomatic from her bone pains. Repeat MRI
A
Volume 60, Number 5, May 2015
showed progression of her CRMO, and she was treated with oral steroids and pamidronate infusion. Her IBD remains asymptomatic several years later, and she is presently receiving no specific treatment for this; however, anti-TNF treatment is under consideration to manage her CRMO.
Case 3 A 2-year-old white boy presented with pyrexia and a limp. He had a red swollen right ankle and left first toe. Haemoglobin (Hb) was 8.8 g/dL, WCC 20.9 109/L, platelets 579 109/L, CRP 169 mg/L, and albumin 30 g/L. He subsequently developed multiple bone lesions in >10 separate locations, involving upper and lower limbs bilaterally (Fig. 3A and B), as well as nonspecific rashes and recurrent splenic abscesses (Fig. 3C). His bone and splenic lesions were aspirated and intravenous antibiotics administered, although all of the aspirate cultures were negative. He received intravenous immunoglobulins (IVIGs) and his symptoms improved. During the following year, he continued to have inflammatory repeat activations of his bony lesions and recurrent splenic abscesses. At the age of 3 years, he received a second dose of IVIG to which he had an allergic reaction. He was treated with oral prednisolone and partial splenectomy. His bone symptoms and radiological lesions subsided, although he experienced further episodes of sterile splenic abscesses, treated with intermittent oral steroids for the ensuing 2 years. At 5 years, he developed perianal skin tags and fissures, abdominal pain, vomiting, and faecal urgency. There was a seconddegree family history of UC. Inflammatory markers, blood counts, and albumin were normal. He underwent proctoscopic examination. Histology showed inflamed anal epithelium with underlying nonnecrotising granulomatous proctitis, whereas rectal mucosal biopsies showed no inflammation. He was treated with metronidazole. Chronic granulomatous disease was excluded by neutrophil oxidative burst test, and detailed immunological function tests were normal. Small bowel contrast imaging was normal. Upper and lower GI endoscopy was macroscopically and histologically normal. His perianal disease was treated with topical tacrolimus,
B
MRI pelvis coronal
C
MRI pelvis axial
MRI spine sagittal
FIGURE 2. A–C, MRI of spine showing 2 areas of signal abnormality in the sacral spine (S1 and S2) with a pathological fracture of the superior end plate of S1. MRI ¼ magnetic resonance imaging.
588
www.jpgn.org
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 60, Number 5, May 2015 A
Chronic Recurrent Multifocal Osteomyelitis and IBD B
X-ray left ankle: lucency and periosteal reaction
C
Bone scan lower limbs: anterior view
D
White cell scan: photopaenic splenic lesions
Colonoscopy
FIGURE 3. A, X-rays of left ankle showing lucent bone lesions and periosteal reaction. B, Bone scan of lower limbs showing multiple areas of increased uptake. C, Abdominal white cell scan showing photopaenic lesions in the spleen. D, Colonoscopy showing scattered aphthous ulcers in the transverse colon.
prednisolone, and mesalazine suppositories. Symptoms persisted, and he underwent repeat upper and lower GI endoscopy at 7 years of age, which was again normal. At 9 years, he underwent his third upper and lower GI endoscopy because he developed loose stools, abdominal pain, static weight, and hypoalbuminaemia (31 g/L). ESR was 45 mm/h, CRP 18 years), although 3 of these were first diagnosed as having either CRMO or IBD when they were
