Splenic cord capillary hemangioma and anemia: Resolution after splenectomy

Kato, Meredith; Lubitz, Carrie C.; Finley, David J.; Chadburn, Amy; Fahey, Thomas J.

doi:10.1002/ajh.20595

Cited by 7 publications

(6 citation statements)

References 11 publications

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“…[31][32][33] Cord capillary hemangioma is a relative rare entity first described in 1993 by Krishnan et al 18 in an abstract, whereas few other cases have also been subsequently reported. 34,35 It was hypothesized by Krishnan and Frizzera 17 that cord capillary hemangioma may represent a 'capillary-vessels-rich' variant of splenic hamartoma. Owing to the presence of both lobular architecture and significant sclerosis (as seen in two of our cord capillary hemangiomacases 4 and 6), a proportion of cord capillary hemangiomas show overlapping features with yet another putatively pseudotumoral splenic lesion, which has been termed 'sclerosing angiomatoid nodular transformation' of the spleen.…”

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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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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“…Os relatos de estudos em necropsias, apontam para incidências do hemangioma esplênico que variam entre 0,03 % e 14 % 11,12,13 . Na maior parte dos casos, ele é assintomático e seu diagnóstico se faz por acaso em um exame de imagem do abdome, por outros motivos 13,14 . Suas dimensões são geralmente menores de quatro centímetros, porém o hemangioma pode crescer e, nesse caso, acompanha-se de desconforto no hipocôndrio esquerdo 11,12 .…”

Section: Introductionunclassified

Esplenectomia parcial para tratar hemangioma esplênico

Petroianu

Brandt

Alencar

2008

Rev. Col. Bras. Cir.

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Apesar de a esplenectomia ser eficaz na abordagem terapêutica de pacientes com hemangioma esplênico, esse procedimento é acompanhado de elevada morbidade e até mortalidade, principalmente devido à sepse, quando realizado em crianças e adolescentes com sistema imunitário ainda imaturo. Para prevenir os efeitos adversos da asplenia, propõe-se neste artigo a esplenectomia parcial, com a retirada apenas da região do hemangioma, mantendo o restante do baço e preservando suas importantes funções.

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“…Before Chiu et al . presented seven cases of CCH in 2011, there had been three reported cases of CCH . When strictly defined according to the relative proportion of the types of constituent vessels, there have only been 8 documented cases of CCH to date …”

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“…5 Before Chiu et al presented seven cases of CCH in 2011, 6 there had been three reported cases of CCH. 4,7,8 When strictly defined according to the relative proportion of the types of constituent vessels, there have only been 8 documented cases of CCH to date. 6,8 Herein, we present an additional case of CCH of the spleen, which was strictly diagnosed according to the constituent vessels.…”

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confidence: 99%

A case of cord capillary hemangioma of the spleen: A recently proven true neoplasm

Tajima

Koda

2015

Pathology International

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Cord capillary hemangioma (CCH) of the spleen is an extremely rare lesion. Once classified under splenic hamartoma, CCH is now differentiated from hamartoma, as its clonality has recently been proven. Herein, we present the case of an incidentally found CCH, measuring 6 × 5.5 × 5 cm, in a 45-year-old man. He underwent splenectomy and has been recurrence-free for 8 years. Macroscopically, the cut surface of the mass showed a central stellate scar with peripheral interspersed reddish areas of variable sizes and intervening fibrous bands. Microscopically, the mass was well demarcated from the splenic tissue. The macroscopic reddish areas were found to correspond to nodules of various sizes, which were predominantly composed of CD34-expressing capillaries without the presence of CD8-expressing sinuses. The relative proportion of these two types of vessels distinguishes CCH from sclerosing angiomatoid nodular transformation (SANT), as SANT shows the apparent presence of CD8-expressing sinuses. In longstanding cases of SANT, sinuses might become ambiguous and the differences between CCH and SANT might become subtle. Nonetheless, immunohistochemistry should be performed when these conditions are suspected, since accurate distinction is usually achieved as a result.

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Splenic cord capillary hemangioma and anemia: Resolution after splenectomy

Cited by 7 publications

References 11 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

Esplenectomia parcial para tratar hemangioma esplênico

A case of cord capillary hemangioma of the spleen: A recently proven true neoplasm

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