Limits...
Caseous granuloma: tuberculosis or chronic recurrent multifocal osteomyelitis?

Assari R, Ziaee V, Ahmadinejad Z, Vasei M, Moradinejad MH - Iran J Pediatr (2014)

Bottom Line: About five months after initiation of the anti-mycobacterial treatment, he was referred to the rheumatology clinic with left elbow pain, effusion and decreased range of motion, and bilateral erythematous palmar pustulosis.The diagnosis of CREMO should be considered in the patients with lytic bone lesions.In addition, the clinicians should be aware of the possibility of caseating granuloma in the cases with possible diagnosis of CREMO.

View Article: PubMed Central - PubMed

Affiliation: Children's Medical Center, Pediatrics Center of Excellence.

ABSTRACT

Background: Chronic recurrent multifocal osteomyelitis (CREMO) is one of the autoinflammatory bone disorders due to disturbance in innate immune system. Up to now, there is no reported case of caseous granulomas in the CREMO. We report a boy with sterile granolumatous osteomyelitis.

Case presentation: A four-year-old boy presented with swelling and pain in the left wrist, malaise and bilateral erythematous pustulosis on the palmar region which had resolved spontaneously after about 7 days. The histopathology of the lesions showed severe acute and chronic inflammatory process and chronic granulomatous reaction with caseating necrosis (granulomatous osteomyelitis). The direct smear, culture and PCR for the mycobacterium tuberculosis and atypical mycobacteria were negative. About five months after initiation of the anti-mycobacterial treatment, he was referred to the rheumatology clinic with left elbow pain, effusion and decreased range of motion, and bilateral erythematous palmar pustulosis. He was diagnosed as CREMO based on two exacerbations, repeatedly negative cultures, and concomitant acute and chronic lesions in the histopathology and X-ray. Naproxen and pamidronate every 3 months were started and all other medications were stopped. Two months after the first dose of pamidronate, he became symptom-free and forearm X-ray showed disappearance of the osteolytic lesions and periosteal reactions.

Conclusion: The diagnosis of CREMO should be considered in the patients with lytic bone lesions. In addition, the clinicians should be aware of the possibility of caseating granuloma in the cases with possible diagnosis of CREMO.

No MeSH data available.


Related in: MedlinePlus

Multiple lytic lesions in ulnar, two chronic osteomyelitis lesions on the distal ulnar, and periosteal reaction over the radius bone.
© Copyright Policy
Related In: Results  -  Collection


getmorefigures.php?uid=PMC4442841&req=5

Figure 2: Multiple lytic lesions in ulnar, two chronic osteomyelitis lesions on the distal ulnar, and periosteal reaction over the radius bone.

Mentions: In spite of negative investigations for the cause of granulomatous osteomyelitis, four drugs anti-mycobacterial regimen was prescribed: Isoniazid, Rifampin, Pyrazinamide, and Ethambutol, plus B6. About five months after initiation of the anti-mycobacterial treatment, he was referred to the rheumatology clinic of the Children’s Medical Center with left elbow pain, effusion and decreased range of motion, and bilateral erythematous palmar pustulosis. The X-ray of the left forearm revealed multiple lytic lesions in ulnar, two chronic osteomyelitis lesions on the distal ulnar, and periosteal reaction over the radius bone (Fig. 2). The bone biopsy showed granulomatous osteomyelitis with some area of necrosis. The direct smear and acid-fast stained smear were negative. The cultures for bacteria, acid-fast bacilli and fungi were negative similar to the previous evaluations.


Caseous granuloma: tuberculosis or chronic recurrent multifocal osteomyelitis?

Assari R, Ziaee V, Ahmadinejad Z, Vasei M, Moradinejad MH - Iran J Pediatr (2014)

Multiple lytic lesions in ulnar, two chronic osteomyelitis lesions on the distal ulnar, and periosteal reaction over the radius bone.
© Copyright Policy
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC4442841&req=5

Figure 2: Multiple lytic lesions in ulnar, two chronic osteomyelitis lesions on the distal ulnar, and periosteal reaction over the radius bone.
Mentions: In spite of negative investigations for the cause of granulomatous osteomyelitis, four drugs anti-mycobacterial regimen was prescribed: Isoniazid, Rifampin, Pyrazinamide, and Ethambutol, plus B6. About five months after initiation of the anti-mycobacterial treatment, he was referred to the rheumatology clinic of the Children’s Medical Center with left elbow pain, effusion and decreased range of motion, and bilateral erythematous palmar pustulosis. The X-ray of the left forearm revealed multiple lytic lesions in ulnar, two chronic osteomyelitis lesions on the distal ulnar, and periosteal reaction over the radius bone (Fig. 2). The bone biopsy showed granulomatous osteomyelitis with some area of necrosis. The direct smear and acid-fast stained smear were negative. The cultures for bacteria, acid-fast bacilli and fungi were negative similar to the previous evaluations.

Bottom Line: About five months after initiation of the anti-mycobacterial treatment, he was referred to the rheumatology clinic with left elbow pain, effusion and decreased range of motion, and bilateral erythematous palmar pustulosis.The diagnosis of CREMO should be considered in the patients with lytic bone lesions.In addition, the clinicians should be aware of the possibility of caseating granuloma in the cases with possible diagnosis of CREMO.

View Article: PubMed Central - PubMed

Affiliation: Children's Medical Center, Pediatrics Center of Excellence.

ABSTRACT

Background: Chronic recurrent multifocal osteomyelitis (CREMO) is one of the autoinflammatory bone disorders due to disturbance in innate immune system. Up to now, there is no reported case of caseous granulomas in the CREMO. We report a boy with sterile granolumatous osteomyelitis.

Case presentation: A four-year-old boy presented with swelling and pain in the left wrist, malaise and bilateral erythematous pustulosis on the palmar region which had resolved spontaneously after about 7 days. The histopathology of the lesions showed severe acute and chronic inflammatory process and chronic granulomatous reaction with caseating necrosis (granulomatous osteomyelitis). The direct smear, culture and PCR for the mycobacterium tuberculosis and atypical mycobacteria were negative. About five months after initiation of the anti-mycobacterial treatment, he was referred to the rheumatology clinic with left elbow pain, effusion and decreased range of motion, and bilateral erythematous palmar pustulosis. He was diagnosed as CREMO based on two exacerbations, repeatedly negative cultures, and concomitant acute and chronic lesions in the histopathology and X-ray. Naproxen and pamidronate every 3 months were started and all other medications were stopped. Two months after the first dose of pamidronate, he became symptom-free and forearm X-ray showed disappearance of the osteolytic lesions and periosteal reactions.

Conclusion: The diagnosis of CREMO should be considered in the patients with lytic bone lesions. In addition, the clinicians should be aware of the possibility of caseating granuloma in the cases with possible diagnosis of CREMO.

No MeSH data available.


Related in: MedlinePlus