Sclerosing Angiomatoid Nodular Transformation (SANT) of the Spleen: A Report of 3 Cases

Koreishi, Aashiyana F.; Saenz, Adam J.; Fleming, Stephen E.; Teruya‐Feldstein, Julie

doi:10.1177/1066896909342568

Cited by 30 publications

(20 citation statements)

References 22 publications

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“…Similar lesions have been described by others. 36,37 In a more recent series of sclerosing angiomatoid nodular transformation cases, published by Diebold et al, 38 one of the lesions was found adjacent to a splenic hemangioma. This suggests the possibility of a secondary response possibly to vascular obstruction and/or intratumoral thrombosis.…”

Section: Discussionmentioning

confidence: 98%

Clonal X-chromosome inactivation suggests that splenic cord capillary hemangioma is a true neoplasm and not a subtype of splenic hamartoma

et al. 2011

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Splenic hamartoma is a rare tumor-like lesion composed of structurally disorganized red pulp elements. It has been hypothesized that two other splenic lesions, cord capillary hemangioma and myoid angioendothelioma, may fall within the spectrum of splenic hamartoma, simply representing morphological variants. In this study, we compared the vascular and stromal composition of cord capillary hemangioma and myoid angioendothelioma with those of classical hamartoma. In addition, we assessed the clonal vs polyclonal nature of the lesions in nine female cases by performing clonality analysis for X-chromosome inactivation at the human androgen receptor locus (HUMARA) on laser-assisted microdissected samples. In 15 of 17 cases, increased reticulin and/or collagen content was observed. The classical hamartoma cases showed a vasculature predominantly composed of CD8 þ CD31 þ CD34À splenic sinuses, whereas cases of cord capillary hemangioma and myoid angioendothelioma contained many CD8À CD31 þ CD34 þ cord capillaries, but very little CD8 þ vasculature. All cases lacked expression of D2-40 and Epstein Barr virus-encoded RNA. All cases showed a proliferation index of r5% by Ki-67. Cases of classical hamartoma lacked significant perisinusoidal expression of collagen IV and low-affinity nerve growth factor receptor. Both markers were variably expressed in the other lesions. Increased CD163-positive histiocytes were found in four cases (three cord capillary hemangiomas and one myoid angioendothelioma). HUMARA analysis was informative in all nine tested cases, of which three cases showed a non-random X-chromosome inactivation pattern, indicating clonality. All three clonal cases were cord capillary hemangiomas. Our study has shown that in spite of considerable morphologic heterogeneity and overlapping features, classical hamartoma and cord capillary hemangioma and myoid angioendothelioma are different in terms of their vascular and stromal composition. Clonality analysis supports a true neoplastic origin for the cord capillary hemangioma. A larger study using additional immunohistochemical and molecular studies is necessary to further evaluate the biological significance of the current findings.

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Section: Discussionmentioning

confidence: 98%

Clonal X-chromosome inactivation suggests that splenic cord capillary hemangioma is a true neoplasm and not a subtype of splenic hamartoma

et al. 2011

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“…11 This idea is supported further by a recent report of three cases by Koreishi et al , in which all three cases had positive IgG4 plasma cells. 12 A link with Epstein-Barr virus has also been postulated by Weinreb et al ; however, in their set of six cases only one was positive for the virus. 13 Koreishi et al also tested for the Epstein-Barr virus, and in their three cases, all were negative.…”

Section: Discussionmentioning

confidence: 70%

Sclerosing angiomatoid nodular transformation of the spleen masquerading as a sarcoma metastasis

Sitaraman¹,

Linn²,

Wayne³

2010

Rare Tumors

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We report a case of sclerosing angiomatoid nodular transformation (SANT) of the spleen presenting as an incidental splenic mass in a patient with a history of retroperitoneal spindle cell sarcoma. Imaging studies and preoperative fine needle biopsy failed to differentiate this lesion from other vascular splenic lesions or a metastatic focus of a prior sarcoma. The patient was treated with splenectomy, which has proved both diagnostic and therapeutic in this and other cases of SANT. Although histology can lead to the diagnosis of vascular tumor, immunohistochemistry is the only way to confirm the diagnosis of SANT. The etiology of SANT is unknown. SANT of the spleen is a benign lesion that does not recur after splenectomy.

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“…However, due to the few cases in the literature it is not clear whether SANT has imaging characteristics that would allow a defi nitive diagnosis without the requirement of histopathological confi rmation [1]. Koreishi et al [6] reported that PET-Scan revealed positive FDG activity heightening clinical concern for malignancy. Some authors argue that core biopsy of the spleen appears to be a useful and noninvasive procedure in the recognition of SANT.…”

Section: Discussionmentioning

confidence: 98%

“…It is a benign process and only a few case reports are documented in the literature [1][2][3][4][5][6][7][8]. It might have started as infl ammatory pseudotumors due to the appearance of the internodular stroma and the fact that some patients had fever or elevated erythrocyte sedimentation rate (ESR) [2].…”

Section: Introductionmentioning

confidence: 99%

Incidental finding of sclerosing angiomatoid nodular transformation of the spleen

Kornprat

Beham‐Schmid

Parvizi

et al. 2011

Wien Klin Wochenschr

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Sclerosing Angiomatoid Nodular Transformation (SANT) of the Spleen: A Report of 3 Cases

Cited by 30 publications

References 22 publications

Clonal X-chromosome inactivation suggests that splenic cord capillary hemangioma is a true neoplasm and not a subtype of splenic hamartoma

Clonal X-chromosome inactivation suggests that splenic cord capillary hemangioma is a true neoplasm and not a subtype of splenic hamartoma

Sclerosing angiomatoid nodular transformation of the spleen masquerading as a sarcoma metastasis

Incidental finding of sclerosing angiomatoid nodular transformation of the spleen

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