Predominance of IgG4+ plasma cells and CD68 positivity in sclerosing angiomatoid nodular transformation (SANT)

Nagai, Yurie; Hayama, N; Kishimoto, Takashi; Furuya, Mitsuko; Takahashi, Yoshi; Otsuka, Masayuki; Miyazaki, Masaru; Nakatani, Yukio

doi:10.1111/j.1365-2559.2008.03118.x

Cited by 48 publications

(29 citation statements)

References 9 publications

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“…Of interest is that both pediatric cases mimicked chronic inflammatory states as evidenced by the patient's growth retardation and elevated erythrocyte sedimentation rate in the report by Kuybulu et al [10] and the initial concern for inflammatory bowel disease in ours. These findings, coupled with reports of dense lymphocytic and IgG4 + plasma cell infiltrates within SANT lesions [3,11], suggest that there may be an immune component to the disease pathogenesis. Whether early age onset of SANT represents a more aggressive form of the disease seen in adults remains to be determined.…”

Section: Discussionmentioning

confidence: 52%

Sclerosing angiomatoid nodular transformation of the spleen in an adolescent with chronic abdominal pain

Bamboat

Masiakos

2010

Journal of Pediatric Surgery

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Section: Discussionmentioning

confidence: 52%

Sclerosing angiomatoid nodular transformation of the spleen in an adolescent with chronic abdominal pain

Bamboat

Masiakos

2010

Journal of Pediatric Surgery

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“…In addition, similarly to inflammatory pseudotumor, EBV participation has been documented in a few cases. 41,42 In their 2003 review article, 17 Krishnan and Frizzera stressed the concept that splenic sinuses and cord capillaries are both characteristic structures of the red pulp and that their respective relative predominance could result in classical hamartoma or cord capillary hemangioma, respectively. However, they also acknowledged that the striking histologic and immunohistochemical differences between classical hamartoma and cord capillary hemangioma may support the contention that they represent two distinct entities with an unclear biologic relationship.…”

Section: Discussionmentioning

confidence: 99%

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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“…SANT has previously been reported to be associated with EBV infection [11], [12]. On the other hand, SANT is considered to be related to IgG4 associated disease [13], [14], [15]. The presence of a significantly higher number of IgG4+ plasma cells and an increased IgG4/IgG ratio has been reported in some studies [13], [14], [15].…”

Section: Discussionmentioning

confidence: 93%

Sclerosing angiomatoid nodular transformation of the spleen presenting rapid growth after adrenalectomy: Report of a case

Nagai

Satoh

Matsukawa

et al. 2017

International Journal of Surgery Case Reports

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Predominance of IgG4+ plasma cells and CD68 positivity in sclerosing angiomatoid nodular transformation (SANT)

Cited by 48 publications

References 9 publications

Sclerosing angiomatoid nodular transformation of the spleen in an adolescent with chronic abdominal pain

Sclerosing angiomatoid nodular transformation of the spleen in an adolescent with chronic abdominal pain

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 presenting rapid growth after adrenalectomy: Report of a case

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