Xanthogranulomatous inflammation involving latissimus dorsi donor site and implant breast reconstruction: case report and literature review
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Xanthogranulomatous inflammation is a rare clinico-pathological condition involving many organ systems. Breast involvement with this rare condition reported from a few cases of mastitis has been limited to only microscopic involvement on histology. We would like to report an unusual presentation of this inflammatory process presenting as a solid lump mimicking malignancy in latissimus dorsi donor site scar and implant-based breast reconstruction as a result of a ruptured silicone gel implant. To our knowledge there have been no previous reports on similar presentation published in the literature. This case highlights a rare complication of a leaked silicone gel implant triggering a xanthomatous response in the absence of the usual infective or obstructing etiologies. This condition is of benign nature with complete clearance on surgical excision and excellent clinical prognosis reported from other organ involvement.
KeywordsGall Bladder Mastitis Breast Reconstruction Prussian Blue Latissimus Dorsi
magnetic resonance imaging
Xanthogranulomatous inflammation involves various organ systems such as the gall bladder, kidney, pancreas, appendix, eyes, female genital tracts, colon, and urachus [1, 2, 3, 4, 5, 6, 7, 8]; breast involvement seen with this inflammation has been limited to only few rare cases of mastitis. We would like to report an unusual involvement of this inflammatory process presenting as a solid lump resembling a malignancy in latissimus dorsi donor site scar and breast implant in a previously treated breast cancer patient. Occurrence of this inflammation from a ruptured implant and silicone gel leak in not known; to our knowledge, there have been no previous reports on similar presentations.
Xanthogranulomatous inflammation is a benign pathological condition considered to be a primary disease process of unknown etiology. Various organ system can be involved to varying degrees; most commonly affected are the kidneys and gall bladder with a few rare cases of colonic involvement. Evidence of breast involvement comes from cases of inflammatory mastitis  showing features of xanthomatous change on microscopy. There is no associated age- or sex-related preponderance with the condition, with a few cases from an early pediatric age group also noted .
The exact etiology of the disease is unknown; organ obstruction, suppurative infections and hemorrhage trigger tissue damage within the involved organs, usually eliciting a microscopic response of the disease process. The case study by Luc and colleagues  following long-standing infections with mycoplasma hominis supports the infective etiology theory of this condition. These triggers, either on their own or synchronously (as postulated in the obstructive hypothesis of gall bladder and kidney pathologies), cause extensive chronic inflammation of the involved organ resulting in a xanthogranulomatous transformation. A similar obstructive trigger would also explain the changes in xanthogranulomatous appendicitis following chronic inflammation of the obstructed lumen with faecolith.
Xanthogranulomatous inflammation disease presentation is variable, with both localized and diffuse responses. A diffuse inflammation is the more severe form of the disease, resulting in a complete obliteration of the native parenchymal tissue and marked fibrosis which clinically presents as solid lumps. Xanthogranulomatous inflammationcan also have a cutaneous involvement which can be primary or secondary . Secondary disease represents a severe form of an internal disease process and, clinically, patients present with a discharging skin lesion that communicates through an underlying fistula with the involved organ.
Radiological diagnosis of xanthogranulomatous inflammation using US and CT is challenging. Diagnostic accuracy can be increased using contrast-based imaging, as demonstrated using sonazoid contrast enhancing agent with US scan in the diagnosis of chronic inflamed and thickened gall bladders, and CT urography studies for renal xanthogranulomatous inflammatory disease [13, 14]. The characteristic findings on imaging are the areas of low attenuation nodules corresponding to the xanthomatous change on histology.
The microscopic appearances of xanthogranulomatous inflammation on histology show the characteristic multinucleate giant cells interspersed with lipid-laden macrophages which impart the characteristic yellow macroscopic appearance. This appearance, however, should be differentiated from a pseudo xanthomatous inflammation or malakoplakia characterized by Michaelis-Gutman bodies which stain positive with Von Kossa calcium and Prussian blue stains .
In our case, an implant leak triggered the inflammatory process. Presentation of a well localized mass in the LD back scar was a result of an extensive tissue inflammation and fibrosis from posterior tracking of the leaked silicone. However, what surprisingly was absent in our case was the postulated triggers of obstruction or infection at surgery; gram staining of both implant and back tissue was negative too. The pre-operative imaging was mostly helpful in delineating the anatomy of the mass and excluding any systemic spread from previously treated breast primary; failure to accurately diagnose the condition confirms the limitations of both CT and MRI in these cases.
The recommended treatment of solid lumps with diffuse involvement is usually surgical as this condition is not pre-malignant . A complete surgical excision negates the need for any further treatment. There is no evidence in the literature to support any recurrence in the native or other organs following the primary surgical treatment.
The silicone gel-filled breast implants are safe with a prolonged life span; however, implant-related problems increases with time (usual complications are capsular contractures, asymmetry and ruptures) and, therefore, surgeons should be wary of these unusual presentations when dealing with the complications of long-standing implants.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
- 1.Kansakar PB, Rodrigues G, Khan SA: Xanthogranulomatous cholecystitis: a clinicopathological study from a tertiary care health institution. Kathmandu Univ Med J (KUMJ). 2008, 6: 472-475.Google Scholar
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