Abstract
Xantho-granulomatous inflammation is a rare type of chronic inflammation, when seen in the bones it is known as xanthogranulomatous osteomyelitis (XO). Here we present a rare case XO of the manubrium sterni, to best of our knowledge this is the first diagnosed. XO of the manubrium sterni mimics radiologically as tuberculosis and as eosinophillic granuloma. We have a 23 year old lady who presented with pain and swelling in the anterior chest, radiology was done and finally the lesion was biopsied. We accepted the patient as Xanthomatous Osteomyelitis and planned for into excision of manubrium sterni, with a flap comprising of ribs and muscle advancements to cover the midline gap in the chest wall, that would have been created. Meanwhile she was put on a short course of antibiotics. It had been observed that the first open biopsy scar had exhibited a tendency for keloid formation. Hence, the risk of final surgery incision producing a disfiguring keloid was informed to the patient, which unfortunately led the patient deter surgery indefinitely, since she was to be married soon. We proposed a local injection of steroid into the lesion to minimize the inflammation. Under image intensifier we injected 40 mg of methyl prednisolone into the tumor. There was some tolerable discomfort the next day of injection that eventually subsided in 3 days. However, the biopsy came inconclusive second core needle biopsy came as XO. Since 1984 with the first case by Cozzutto et al, only 17 cases have been reported in the worldwide literature including us.
Highlights
Xantho-granulomatous inflammation (XI) is a very rare variety of chronic inflammation, though usually witnessed in bladder, kidney, pancreas, fallopian tube, ovary, epididymis, testis, prostate and salivary glands [1] but when seen in bones, it is k/a Xanthogranulomatous osteomyelitis (XO)
Cysticercosis disease is caused by the larval form of the Taenia solium parasite, known to induce inflammatory sequence, in tissues surrounding the parasite, and quite distant[4] Cysticercosis casts it’s oncogenic immunosuppressive effect through various pathways viz. differential expression of antigens, molecular mimicry of human leukocyte antigens, and alterations to the human immune system [5] Cysticercosis causes synthesis of proteins that decrease the production of cytokines IL-2, IL-4, and IFN-γ and recruitment of macrophages by TNF-α, thereby rendering Th1 and Th2 immune responses and pro-inflammatory cytokines ineffective, which act as the defense mechanism against neoplasia[6]
Xanthomatous Inflammation needs to be differentiated from Tubercular osteomyelitis (Gene Expert / CBNAAT is required to rule out ), Eosinophilic Granuloma (S100 and CD1a both are positive), Hand Schuller Christian syndrome, PseudoXanthomatous inflammation or malakoplakia, Erdheim-Chester disease
Summary
Xantho-granulomatous inflammation (XI) is a very rare variety of chronic inflammation, though usually witnessed in bladder, kidney, pancreas, fallopian tube, ovary, epididymis, testis, prostate and salivary glands [1] but when seen in bones, it is k/a Xanthogranulomatous osteomyelitis (XO). Case presentation: This 23year old lady, presented to us with pain and swelling in front of upper chest, in and around manubrium sterni, with local signs of inflammation Her blood parameters showed slight increase in ESR, CRP, alkaline phosphatase with normal serum cacium levels. We accepted the patient as Xanthomatous Osteomyelitis and planned for intoto excision of manubrium sterni, with a flap comprising of ribs and muscle advancements to cover the midline gap in the chest wall, that would have been created. She was put on a short course of antibiotics. Though the tumor tissue was beyond posterior bony confines of manubrium, yet it was not invading adjacent mediastinal sensitive structures
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