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CASE REPORT
J Res Med Sci 2021,  26:32

Chronic recurrent multifocal osteomyelitis in a 3.5-year-old boy


1 Department of Orthopedics, Isfahan University of Medical Sciences, Isfahan, Iran
2 Bone Joint and Related Tissues Research Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran

Date of Submission03-Apr-2020
Date of Decision11-Jul-2020
Date of Acceptance31-Dec-2020
Date of Web Publication27-May-2021

Correspondence Address:
Dr. Seyed Mohamad Hossein Tabatabaei Nodushan
Department of Orthopedics, Isfahan University of Medical Sciences, Isfahan
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jrms.JRMS_338_20

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  Abstract 


We report an extremely rare case of multifocal bone disorder in a 3.5-year-old boy who appeared for left forearm and arm pain and multiple periods of fever with an unusual presentation of lymphoma/leukemia and highlight diagnostic challenges leading to a misdiagnosis, which was then diagnosed and treated for chronic recurrent multifocal osteomyelitis (CRMO). Based on a left arm biopsy and whole-body scans, he was eventually diagnosed with CRMO. Taken together, in this case, we noticed a notable amelioration after a 5-month treatment with nonsteroidal anti-inflammatory drugs on multiple bone pains.

Keywords: Bone, chronic recurrent multifocal osteomyelitis, eosinophilic granuloma, lymphoma


How to cite this article:
Tahririan MA, Nodushan SM, Farrokhi M. Chronic recurrent multifocal osteomyelitis in a 3.5-year-old boy. J Res Med Sci 2021;26:32

How to cite this URL:
Tahririan MA, Nodushan SM, Farrokhi M. Chronic recurrent multifocal osteomyelitis in a 3.5-year-old boy. J Res Med Sci [serial online] 2021 [cited 2021 Dec 1];26:32. Available from: https://www.jmsjournal.net/text.asp?2021/26/1/32/316989




  Introduction Top


Chronic recurrent multifocal osteomyelitis (CRMO), or chronic nonbacterial osteomyelitis, is an extremely scarce condition in children with low-incidence, noninfectious inflammatory bone disease.[1] CRMO which occurs fundamentally in children and adolescents may be misdiagnosed as osteomyelitis, lymphoma/leukemia, arthritis, or cancer.[2],[3],[4] Furthermore, CRMO has unknown etiology, but agents or factors containing the disorders of inflammatory, genetic, autoimmunity, juvenile seronegative spondyloarthropathy, and infectious organism have been shown to play a major role as well as CRMO epidemiological data are sparse and contain little case series and zonal cohorts plus, clinical symptoms presented are various, such as bone pain, local swelling, scarcely skin redness, and heat.[1],[2],[3],[4],[5] In addition, in the early stages of diagnosis, complete body imaging using magnetic resonance imaging (MRI) (turbo inversion recovery magnitude) techniques should be performed to diagnose silent clinical lesions, exclusively in the vertebral column, and also to assess CRMO activity during follow up, the exploration and monitoring the consequences of CRMO, which may include fractures, inflammatory involvement, and tissue damage to surrounding structures and histopathology findings show the chronic inflammation together with granulocytes, lymphocytes, plasma cells, and monocytes in later stages.[1],[2],[3],[4],[5],[6],[7] We hereby describe the clinical and radiological data on a rare case of CRMO treated with nonsteroidal anti-inflammatory drugs (NSAIDs).


  Case Report Top


A 3.5-year-old male patient with left forearm pain and fever was admitted to the pediatrician. After initial examinations, including hemoglobin: 8, hematocrit: 32.3, white cell count (white blood cell): 8100, lymph: 51.7%, platelet count: 462,000, erythrocyte sedimentation rate: 72 and X-ray [Figure 1]a, lymphoma/leukemia was suggested. The patient was also examined by MRI, biopsy of left radius, flow cytometry, and bone marrow aspirate. The pediatrician, due to the significance of the issue of initiating treatment, suggested the patient's referral to the orthopedic department. With the suspicion of eosinophilic granuloma, the patient underwent a biopsy of the forearm, in which many necroses were reported without a decisive diagnosis, but was not referred due to his recovery. The probability of eosinophilic granuloma was considered and the patient did not receive treatment for lymphoma/leukemia and only took the medication and becomes a follow-up. Five months later, the patient returns with the right forearm pain and left arm (and the previous location of the lesion in the left forearm X-ray showed that the lesion was removed) [Figure 1]b. The patient was re-examined for supplementary lymphoma/leukemia, and a whole-body scan was taken from the patient (metabolically active bony lesion of the left humerus and right forearm); as a result, diagnosis of eosinophilic granuloma was rejected. Finally, as a result of the left arm biopsy, CRMO was diagnosed. Taken together, the patient was treated with NSAIDs and recovered quickly and his graphs ultimately became normal.
Figure 1: (a) Increased density in medulla of both ulna and radius bones associated with permeative lytic lesions in radius is seen. Cortical irregularity is also evident in some areas. No obvious soft tissue. The wrist seems to be intact. (b) There is a lytic lesion with a wide zone of transition in midshaft of the left humerus bone. Adjacent cortical irregularity and disruption is also noted associated with lamellated periosteal reaction. No obvious soft tissue is evident

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  Discussion Top


Similar to our diagnosis approach, Girschick et al., 2007, reported the major role of pathologic biopsy in ruling out other diagnoses such as cancer and infectious diseases, so biopsy must be considered, particularly since it is often hard to create a definite detection among oncological bone lesions and those related to CRMO.[8] Gicchino et al., 2018, concluded that the most common differential diagnosis of CRMO is shown by infections, malignant bone tumors, and Langerhans cell histocytosis.[9] In addition, a study by Fritz et al., 2015, revealed that the use of the whole-body MRI was beneficial in reaching the diagnosis,[10] which is consistent with our case. Furthermore, our study revealed that a whole-body MRI scan seems to have even better diagnostic potential and sensitivity. A number of studies have been reported that that whole-body MRI is very valuable, although expensive and this method may become a diagnostic standard in the work-up of doubtful CRMO in future.[11] Moreover, a review evaluation by Schultz et al. of case reports of CRMO showed that 79% of patients had a good response to NSAIDs.[12] In addition, Walsh et al. recommend that CRMO signs are generally well controlled by NSAID therapy; therefore, in our experience, NSAIDs are the first choice for the initial treatment in our case.[13] Furthermore, a report by Pham et al. examined a 38-year-old man who presented with lower extremity musculoskeletal pain with the presentation of Hodgkin lymphoma as a misdiagnosis instead of CRMO.[14]


  Conclusion Top


This case report demonstrates the diagnostic difficulties of a musculoskeletal presentation of lymphoma, challenges of nondiagnostic bone pain, and yields challenges of diagnostic delays in providing a potential therapeutic treatment for CRMO. We consider that whole-body scans and bone biopsy can be reliably performed in children for the diagnosis of CRMO. Therefore, we propose that children with bone pain due to CRMO involvement be considered for NSAIDs therapy primary after diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Hofmann SR, Kapplusch F, Girschick HJ, Morbach H, Pablik J, Ferguson PJ, et al. Chronic recurrent multifocal osteomyelitis (CRMO): Presentation, pathogenesis, and treatment. Curr Osteoporos Rep 2017;15:542-54.  Back to cited text no. 1
    
2.
Borzutzky A, Stern S, Reiff A, Zurakowski D, Steinberg EA, Dedeoglu F, et al. Pediatric chronic nonbacterial osteomyelitis. Pediatrics 2012;130:e1190-7.  Back to cited text no. 2
    
3.
Schnabel A, Range U, Hahn G, Siepmann T, Berner R, Hedrich CM. Unexpectedly high incidences of chronic non-bacterial as compared to bacterial osteomyelitis in children. Rheumatol Int 2016;36:1737-45.  Back to cited text no. 3
    
4.
Hofmann C, Wurm M, Schwarz T, Neubauer H, Beer M, Girschick H, et al. A standardized clinical and radiological follow-up of patients with chronic non-bacterial osteomyelitis treated with pamidronate. Clin Exp Rheumatol 2014;32:604-9.  Back to cited text no. 4
    
5.
Ferguson B, Gryfe D, Hsu W. Chronic recurrent multifocal osteomyelitis in a 13 year old female athlete: A case report. J Can Chiropr Assoc 2013;57:334-40.  Back to cited text no. 5
    
6.
Schilling F, Kessler S. Chronic recurrent multifocal osteomyelitis – I. Review. Klin Padiatr 2001;213:271-6.  Back to cited text no. 6
    
7.
Alshammari A, Usmani S, Elgazzar AH, Ashkanani RA. Chronic recurrent multifocal osteomyelitis in children: A multidisciplinary approach is needed to establish a diagnosis. World J Nucl Med 2013;12:120-3.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Girschick HJ, Zimmer C, Klaus G, Darge K, Dick A, Morbach H. Chronic recurrent multifocal osteomyelitis: What is it and how should it be treated? Nat Clin Pract Rheumatol 2007;3:733-8.  Back to cited text no. 8
    
9.
Gicchino MF, Diplomatico M, Granato C, Capalbo D, Marzuillo P, Olivieri AN, et al. Chronic recurrent multifocal osteomyelitis: A case report. Ital J Pediatr 2018;44:26.  Back to cited text no. 9
    
10.
Fritz J, Lurie B, Potter HG. MR imaging of knee arthroplasty implants. Radiographics 2015;35:1483-501.  Back to cited text no. 10
    
11.
von Kalle T, Heim N, Hospach T, Langendörfer M, Winkler P, Stuber T. Typical patterns of bone involvement in whole-body MRI of patients with chronic recurrent multifocal osteomyelitis (CRMO). Rofo 2013;185:655-61.  Back to cited text no. 11
    
12.
Schultz C, Holterhus PM, Seidel A, Jonas S, Barthel M, Kruse K, et al. Chronic recurrent multifocal osteomyelitis in children. Pediatr Infect Dis J 1999;18:1008-13.  Back to cited text no. 12
    
13.
Walsh P, Manners PJ, Vercoe J, Burgner D, Murray KJ. Chronic recurrent multifocal osteomyelitis in children: Nine years' experience at a statewide tertiary paediatric rheumatology referral centre. Rheumatology (Oxford) 2015;54:1688-91.  Back to cited text no. 13
    
14.
Pham M, Ressler S, Rosenthal A, Kelemen K. Classical Hodgkin lymphoma masquerading as chronic recurrent multifocal osteomyelitis: A case report. J Med Case Rep 2017;11:45.  Back to cited text no. 14
    


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