Síndrome Klippel-Trenaunay en el embarazo. Reporte de un caso y revisión de la literatura / Klippel-Trenaunay syndrome in pregnancy. Case report and literature review

Resumen Antecedes: El síndrome de Klippel-Trenaunay (KTS) es un síndrome de malformación vascular que comprende una afectación variable de capilares cutáneos, venas y linfáticos con hipertrofia de tejidos blandos y huesos de la extremidad afectada. Durante el embarazo estas malformaciones se incrementan, con afectación pélvica e intraabdominal. Caso clínico: Paciente de 15 años, primigesta, con síndrome Klippel-Trenaunay diagnosticado al nacimiento, con embarazo a término, referida por iniciar con trabajo de parto para finalización del embarazo en un hospital de tercer nivel. No cuenta con control obstétrico, estudios prenatales ni ultrasonidos obstétricos. Se realiza una ecografía pélvica en donde se descartóla presencia de varices pélvicas y un doppler que evidenció un sistema venoso conservado. Se realiza terminación del embarazo vía abdominal obteniendo un recién nacido vivo masculino, peso de 3045 gramos, APGAR 9 y 9 al minuto y a los 5 minutos. Resultados: El embarazo en pacientes con síndrome de Klippel-Trenaunay tiene alto riesgo de tromboembolismo y complicaciones hemorrágicas. La valoración debe ser por un equipo multidisciplinario capacitado para anticiparse a las potenciales complicaciones. Limitaciones del estudio o implicaciones: La principal limitación es la baja incidencia de esta patología. Se puede concluir que el diagnostico de SKT no es indicación de interrupción del embarazo. El éxito del manejo de estas pacientes requiere la participación de un equipo multidisciplinario. Originalidad o valor: Este caso clínico es de primordial relevancia ya que en la bibliografía internacional están reportados menos de 100 casos de embarazos complicados con este síndrome.

Abstract: Background: Klippel-Trenaunay syndrome (KTS) Klippel- Trenaunay syndrome (KTS) is a vascular malformation síndrome that includes variable involvement of skin capillaries, veins and lymphatics with hypertrophy of soft tissues and bones of the affected limb. During pregnancy, these malformations increase, with pelvic and intra-abdominal involvement. Clinical case: 15-year-old patient, gravida 0, with Klippel- Trenaunay syndrome diagnosed at birth, with full-term pregnancy, referred for initiating labor for resolution of pregnancy in a third level hospital. Without obstetric control, without prenatal studies or obstetric ultrasounds. A pelvic ultrasound was performed, which ruled out the presence of pelvic varices and a Doppler that showed a preserved venous system. The pregnancy was terminated by abdominal route, obtaining a male newborn, weighting 3045 grams, APGAR 9 and 9 after 1 and 5 minutes. Results: Pregnancy in patients with Klippel-Trenaunay síndrome has a high risk of thromboembolism and bleeding complications. They should be evaluated by a trained multidisciplinary team to anticipate possible complications. Study limitations or implications: The main limitation is the low incidence of this pathology. It can be concluded thatthe diagnosis of SKT is not an indication for termination of pregnancy. Successful management of these patients requires the participation of a multidisciplinary team. Originality or value: This clinical case is of primary relevance since fewer than 100 cases of complicated pregnancies with this syndrome are reported in the international literature.

Multimodal imaging features of perifoveal exudative vascular anomalous complex / 中华眼底病杂志

Objective:To observe the multimodal imaging features and explore the treatment of parafoveal exudative vascular anomaly complex (PEVAC).Methods:A retrospective study. Six patients (6 eyes) with PEVAC diagnosed in Tianjin Eye Hospital were included in this study from July 2018 to December 2021. All patients were female with monocular disease. The age was (61.1±9.3) years. All patients showed a sudden painless decline in monocular vision with metamorphopsia. All patients underwent best corrected visual acuity (BCVA), color fundus photography, fundus fluorescein angiography (FFA), optical coherence tomography (OCT) and OCT angiography (OCTA). Indocyanine green angiography (ICGA) was performed in 4 eyes. In 6 eyes, 3 eyes were treated with intravitreal injection of anti-vascular endothelial growth factor drug; 5 eyes were treated with micropulse laser photocoagulation and/or local thermal laser photocoagulation; 1 eye was treated with photodynamic therapy. Five patients were followed up for (9.2±7.4) months, and 1 patient was lost. At follow-up, the same equipment and methods were used as at the initial diagnosis. The clinical manifestations, multimodal image features and treatment response were observed.Results:Baseline BCVA of affected eyes were ranged from 0.1 to 0.5. PEVAC was isolated in 6 eyes, and the fundus showed isolated hemangioma-like leision, accompanied by small bleeding and hard exudation. There were 2 isolated hemangiomatous lesions adjacent to each other in 2 eyes. In the early stage of FFA, punctate high fluorescence lesions near the macular fovea were seen, and the leakage was enhanced in the late stage. There was no leakage in the early stage of ICGA, or slight leakage with late scouring. OCT showed an oval lesion with high reflection wall and uneven low reflection. The central macular thickness (CMT) was (431±76) μm. OCTA showed blood flow signals in PEVAC, 2 eyes in the superficial capillary plexus (SCP), and it was also observed in the deep capillary plexus (DCP), but the intensity of blood flow signal was slightly weaker than that in the SCP. The blood flow signal was visible only in DCP in 2 eyes. SCP and DCP showed similar intensity of blood flow signals in 2 eyes. After treatment, the bleeding was absorbed basically in 4 eyes, the hard exudation partially subsided, the CMT decreased, the intercortical cystic cavity of the fovea nerve decreased, the hemangiomatous lesions narrowed, and BCVA increased. In 1 eye, the macular sac was reduced and partially absorbed by hard exudation, which was later relapsed due to blood pressure fluctuation.Conclusions:The majority of PEVAC patients had monocular onset. The fundus is characterized by solitary or structure with strong reflex walls, with or without retinal cysts, hard exudates, and subretinal fluid, and visible blood flow signals inside.

Application of phantom for dose measurement in interventional therapy for pediatric vascular anomalies / 中国辐射卫生

@#<b>Objective</b> To apply a phantom for dose measurement in interventional therapy for pediatric vascular diseases, and calculate the effective dose (<i>E</i>) and conversion coefficient of dose area product (DAP) to <i>E</i>, and to provide a dose reference for studying radiation dose and radiation protection in children. <b>Methods</b> Thermoluminescent dosimeters were placed in the organs of the phantom. Low-, medium-, and high-dose groups were set for three types of vascular anomalies based on the duration of fluoroscopy. Digital subtraction angiography was used to simulate exposure conditions at different dose levels. The organ dose was measured, and the effective dose was calculated. <b>Results</b> For the three groups of vascular anomalies in the head and face, the red bone marrow doses were 8.15, 30.34, and 43.53 mGy, respectively, the effective doses were 12.88, 47.84, and 73.12 mSv, respectively; and the average conversion coefficient of DAP to <i>E</i> was 2.16. For the three groups of vascular anomalies in the trunk, the red bone marrow doses were 2.11, 15.62, and 31.21 mGy, respectively; the effective doses were 12.39, 70.56, and 134.60 mSv, respectively, and the average conversion coefficient of DAP to <i>E</i> was 3.03. For the three groups of vascular anomalies in the lower extremities, the red bone marrow doses were 3.58, 6.50, and 12.28 mGy, respectively, the effective doses were 3.64, 7.04, and 14.85 mSv, respectively, and the average conversion coefficient of DAP to <i>E</i> was 0.73. <b>Conclusion</b> Patient dose and DAP-to-<i>E</i> conversion coefficient are in the following order: vascular anomalies in the trunk > vascular anomalies in the head and face > vascular anomalies in the lower extremities. The dose data obtained can be used to estimate children’s radiation exposure.

Artéria subclávia direita aberrante: relato de caso e revisão de literatura / Aberrant right subclavian artery: case report and literature review

Resumo A artéria subclávia direita aberrante, também conhecida como artéria lusória, é a anomalia do arco aórtico mais comum, ocorrendo entre 0,5 e 1% da população. Possui prevalência em mulheres e normalmente está associada a outras variações anatômicas, como o nervo laríngeo não recorrente, presente em 86,7% dos casos. Em sua maioria, a artéria subclávia direita aberrante não apresenta sintomas. Descrevemos essa alteração em uma paciente de 82 anos, hipertensa e assintomática, que havia sido submetida a uma angiotomografia toracoabdominal para a avaliação de uma dissecção crônica tipo III (DeBakey) associada à dilatação de aorta descendente. No achado, a artéria subclávia direita aberrante apresentava percurso retroesofágico associado a um divertículo de Kommerell. Devido à raridade, realizamos revisão bibliográfica integrativa das bases de dados MEDLINE, UpToDate, LILACS, SciELO e Portal CAPES dos últimos 6 anos e discutimos as alterações anatômicas mais frequentes, a sintomatologia e as condutas terapêuticas adotadas.

Abstract The aberrant right subclavian artery, also known as the arteria lusoria, is the most common aortic arch anomaly, occurring in 0.5 to 1% of the population. There is a higher prevalence in women and it is usually associated with other anatomical variations, such as the non-recurrent laryngeal nerve, present in 86.7% of cases. In the majority of cases, the aberrant right subclavian artery causes no symptoms. We describe this anomaly in an 82-year-old, hypertensive, and asymptomatic patient who had undergone a thoracoabdominal angiography to investigate a chronic DeBakey type III aortic dissection with dilation of the descending aorta. The aberrant right subclavian artery followed a retroesophageal course and was associated with a Kommerell diverticulum. In view of its rarity, we conducted an integrative bibliographic review of literature from the last 6 years indexed on the Medline, UpToDate, Lilacs, Scielo, and Portal Capes databases and discuss the most frequent anatomical changes, symptomatology, and therapeutic management adopted.

Doble cuarto arco aórtico persistente en un perro (Canis familiaris): reporte de caso

El doble arco aórtico persistente es una patología caracterizada por anomalías embrionarias en la vascularización, que pueden afectar de manera indirecta a otros sistemas, como el digestivo y el respiratorio. El objetivo de este documento es reportar un caso de doble arco aórtico, persistente en un cachorro Bull terrier, de seis meses de edad. El paciente ingresó a consulta por motivo de regurgitaciones frecuentes y pérdida ponderal. En el estudio radiográfico, se evidenció dilatación esofágica craneal a la base del corazón y en la toracotomía, se confirmó un doble arco aórtico persistente. Se realizó manejo nutricional y posteriormente, corrección quirúrgica de la anomalía vascular. Este es el primer caso de una anomalía de este tipo en Colombia. Se concluye, que un manejo quirúrgico enfocado a liberar el anillo estenosante y a recuperar la función esofágica, son la base terapéutica de este tipo de alteraciones.

Persistent double aortic arch is a pathology characterized by embryonic vascularization anomalies, which can indirectly affect other systems such as the digestive and respiratory systems. The objective of this document is to report a case of persistent double aortic arch in a six-month-old Bull Terrier puppy. The patient was admitted for consultation due to frequent regurgitation and weight loss. The radiographic study revealed cranial esophageal dilation at the base of the heart, and a thoracotomy confirmed a persistent double aortic arch. Nutritional management was performed and subsequently, surgical correction of the vascular anomaly. This is the first case of an anomaly of this type in Colombia. It is concluded that surgical management focused on releasing the stenosing ring and recovering esophageal function are the therapeutic basis for this type of alteration.

MRI findings and pathological analysis of intramuscular fibro-adipose vascular anomaly / 中华放射学杂志

Objective:To investigate the MRI and pathological features of intramuscular fibro-adipose vascular anomaly (FAVA).Methods:The clinical and imaging data of 44 patients with intramuscular FAVA confirmed by pathology from December 2012 to March 2021 in Henan Province People′s Hospital were retrospectively analyzed. Twenty-five females and 19 males were included, with the age of (15±6), from 5 to 29 years old. The clinical and MRI features including the type, location, boundary, signal intensity, enhancement mode and degree, and the vascular flow voids in the lesion were summarized and compared with pathological results.Results:The thigh and calf muscles were involved in 1 patient simultaneously, and 1 site was involved in 43 patients, including 20 calf muscles, 15 thigh muscles, 5 forearm muscles, 1 upper arm muscle, 1 gluteal muscle, and 1 shoulder muscle. The gastrocnemius muscle of lower leg was most commonly involved (13/44), followed by soleus muscle (10/44) and quadriceps femoris muscle (9/44). All the lesions were solid on MRI, including 24 cases of focal mass type, 15 cases of diffuse infiltration type and 5 cases of local infiltration type. The long axis of all the lesions were consistent with the long axis of the muscles. All lesions showed inhomogeneously moderate hyperintensity on T 1WI and T 2WI, and significantly hyperintensity on fat suppression T 2WI. All lesions showed tortuous and dilated abnormal vessels, of which 18 cases showed vascular flow voids. Thrombosis was found in 10 cases. On contrast-enhanced imaging, the lesions showed moderate to obvious inhomogeneous enhancement. Pathologically, the diseased skeletal muscle was infiltrated by fibrous tissue, fat components, irregular abnormal veins and vessels, which led to inhomogeneous MRI signals. Among the 7 patients who underwent human PIK3CA gene mutation detection, and 6 were mutant. Conclusions:Intramuscular FAVA has certain characteristics in clinic, MRI imaging and histopathology, and its MRI signal characteristics can reflect its complex pathological components.

A rare case of idiopathic vascular renal tumor renal hemangioma: A case report

@#Renal hemangioma is a rare benign vascular tumor which can present with painful or painless gross hematuria. Its preoperative diagnosis is extremely difficult due to non-specific imaging findings and can also mimic other disease entity.This is a case of a 31-year-old female who presented with recurrent gross hematuria with no antecedent cause. KUB ultrasound and CT stonogram showed insignificant findings of the cause of hematuria. Renal Angiogram revealed multiple vascular channels with arterial and venous connections at the upper pole of the right kidney with the impression of gross hematuria secondary to AV malformation. Simple Nephrectomy was done to address the hematuria wherein histopathologic findings were indicative of renal hemangioma. Patient did not report any episode of gross hematuria since the operation.

Aberrant right subclavian artery in a dog - case report / [Ectopia de artéria subclávia direita em cão - relato de caso]

The brachiocephalic trunk and the left subclavian artery originate from the aortic arch, and both supply blood to the head, neck, and thoracic limbs. Anatomical variations, such as an aberrant right subclavian artery, are congenital conditions rarely observed in dogs, Thus, the objective of the present report was to describe a case of aberrant right subclavian artery in a 9-year-old Dalmatian. However, this anomaly was a finding in which the patient was asymptomatic during its 9 years of life and only at this age did he exhibit signs including sialorrhea, vomiting, hyporexia, and noisy deglutition. Blood count, biochemical profile, and thoracic radiography led to a diagnosis of megaesophagus and aspiration pneumonia. Despite the recommended treatment, the patient did not respond well; as such, the owner elected to euthanize the animal. On necropsy, the right subclavian artery originated directly from the aortic arch, followed a route from left to right dorsally to the esophagus, and then formed an impression of the vascular path over the muscular wall of the esophagus. The esophagus, in turn, exhibited a flaccid wall and dilation in the caudal portion to the vascular path made by the ectopic position of the right subclavian artery.(AU)

O tronco braquiocefálico e a artéria subclávia esquerda emergem do arco aórtico e são responsáveis por fazerem o suprimento sanguíneo para cabeça, pescoço e membros torácicos. Variações anatômicas, como a ectopia da artéria subclávia direita, são alterações congênitas raramente encontradas em cães, cujas alterações do sistema digestivo acontecem em pacientes recém-desmamados e não em adultos. Assim, o objetivo deste relato é descrever um caso de ectopia da artéria subclávia direita em uma cadela, Dálmata, de nove anos de idade. No entanto, essa anomalia foi um achado do qual o paciente foi assintomático durante os nove anos de vida e somente com essa idade apresentou sinais como sialorreia, vômito, hiporexia e deglutição ruidosa. O hemograma e os perfis bioquímicos, associados à radiografia torácica, levaram a um diagnóstico de megaesôfago e pneumonia aspirativa. Mesmo seguindo o tratamento recomendado, houve piora clínica do quadro e o animal foi submetido à eutanásia. À macroscopia, a artéria subclávia direita originava-se direto do arco aórtico, fazia um percurso da esquerda para a direita dorsalmente ao esôfago e, então, formava uma impressão do trajeto vascular sobre a parede muscular do esôfago. O esôfago, por sua vez, apresentava parede flácida e dilatação na porção caudal ao trajeto vascular feito pela posição ectópica da artéria subclávia direita.(AU)

Aberrant right subclavian artery (arteria lusoria) aneurysm with a Kommerell's diverticulum / Aneurisma de artéria subclávia direita aberrante (artéria lusória) com divertículo de Kommerell

The treatment options for aberrant right subclavian artery vary depending on the presence of Kommerell's diverticulum. Because there is a tendency not to report mortalities of these rare cases in the literature, it is hard to reach a conclusion on treatments from the limited data on post-interventional results in these patients. We report our experience with a 67-year old patient with an aberrant right subclavian aneurysm with Kommerell's diverticulum, diagnosed by chance

As opções de tratamento para artéria subclávia direita aberrante variam dependendo da presença de divertículo de Kommerell. Como há uma tendência a não relatar mortalidade nos raros casos descritos na literatura, é difícil chegar a uma conclusão sobre tratamentos tendo em vista os dados limitados sobre resultados pós-intervenção nesses pacientes. Relatamos aqui nossa experiência com um paciente de 67 anos de idade com aneurisma de artéria subclávia aberrante direita com divertículo de Kommerell diagnosticado ao acaso

Congenital Lymphatic Malformation

Lymphatic malformation is one of common benign disease in head and neck lesion. It frequently arises from oral cavity, pharynx, skin, and mediastinum. Huge lymphatic malformation may result in dysfunction of vital structure. Lymphatic malformation, which has commonly ill-defined margin, would affect surrounding structures which include upper aero-digestive tract with unresectable margin. Lymphatic malformation is mainly treated with conservative methods. Recently, various treatment modalities have been introduced. So, we intended to describe this disease entity with the review of recent literature.

Retrospective clinical analysis of 34 cases with fibro-adipose vascular anomaly on the lower extremities / 中华整形外科杂志

Objective@#To define a new subtype of vascular anomaly, named fibro-adipose vascular anomaly(FAVA)and to discuss the methods of diagnosis and treatment in order to decrease the misdiagnosis rate and improve the recovery rate.@*Methods@#From Jan. 2011 to Dec. 2015, 34 cases with FAVA on the lower extremities received surgical treatment in our center. The clinical data were collected to summarize the imaging and pathological characteristics for diagnosis.@*Results@#The misdiagnosis rate was 76.5%(26/34) in all these 34 patients. The cure rate was 100% after operation. The patients were followed-up for 1-3 years(average, 19 months) with normal function and no recurrence.@*Conclusions@#FAVA is usually misdiagnosed as hemangioma or vascular malformation. The disease should be well defined to help the diagnosis and treatment. Surgical excision is one of the optional treatments.

A rare variant angioarchitecture of upper abdomen / 대한해부학회지

Vascular anomalies are frequently encountered in abdomen. But they are usually asymptomatic and diagnosed accidently during angiography or surgery leading into severe complications. Thus knowledge of angioarchitecture in abdomen, whether normal or variant, is considered prerequisite for successful, uncomplicated surgeries and interventional radiology. This case report describes one of such varying branching pattern of celiac trunk and superior mesenteric artery. During routine abdominal dissection, gastroduodenal artery was seen arising from celiac trunk along with its usual three branches. Common hepatic artery continued as left hepatic artery after giving rise the right gastric artery and a tortuous replaced right hepatic artery arose from superior mesenteric artery. An unusually long cystic artery arose from left hepatic artery and gave rise to 2-3 small anastomotic branches towards hepatic flexor of colon, in addition to its normal gallbladder supply. Awareness of such variations would certainly be helpful in upper abdominal surgeries.

Variant course and branching of right brachial artery: a case study.

A variant course and branching pattern of the right brachial artery was recorded in a 54-year-old male cadaver during the practical sessions of University College of Medical Sciences, Delhi, India. The right brachial artery divided in the middle third of arm into a medial superficial and lateral deep branch. The superficial medial branch descended anterior to the median nerve and ended by dividing in the cubital fossa into ulnar and radial arteries, whereas the lateral branch descended postero-medial to the median nerve, ending deep to pronator teres as the common interosseous artery. The left brachial artery showed a normal branching pattern by dividing into radial and ulnar arteries in the cubital fossa. The probable origin of such a variation is embryological and familiarity with such variations is imperative as they might affect dynamics of limb function or alter the course of interventional procedures.

Pulsatile Tinnitus Arising from Aberrant Internal Carotid Artery at Nasopharynx

Pulsatile tinnitus arising from an aberrant internal carotid artery at the level of the nasopharynx is rarely found. We present a case of a 74-year-old woman complaining of pulsatile tinnitus in the right ear. The tinnitus was not audible by auscultation. Endoscopic examination revealed pulsating swelling of the nasopharyngeal wall on the right side. Computed tomography angiography of the head and neck region showed medial and superficial transposition of the right internal carotid artery at the level of the nasopharynx. The artery was located right next to the orifice of the Eustachian tube and coursed along the distal portion of the tube. The patient's perception of tinnitus was improved after explaining the cause of the tinnitus and reassuring her about thecondition.

Retroperitoneal vascular anomalies in paraaortic region: Impact on laparoscopic paraaortic lymphadenectomy in patients with gynecologic malignancies / 대한산부인과내시경학회지

OBJECTIVE: To document our experience of the vascular anomalies or variants in paraaortic region and intend to increase vigilance among the gynecological surgeons for presence of variable vascular anomalies or variants. METHODS: We conducted a retrospective chart review of 280 patients with various gynecologic malignancies who had undergone systemic laparotomic or laparoscopic paraaortic lymphadenectomy between November 2003 and July 2011. RESULTS: We discovered total nine patients of vascular anomalies during the surgery. Seven patients had an accessory polar renal artery. One patient had a duplicated inferior vena cava and the other had a right paravertebral vein. There were no vascular complications such as tearing, ligation or transection. CONCLUSION: It is not uncommon to encounter vascular anomalies in paraaortic region during the lymphadenectomy. Hence, the gynecological surgeons must be cognizant of various vascular anomalies occurring within this area to reduce the vascular accidents.

Case of Surgical Treatment with Free Flap on Large Size Facial Capillary Malformation

Capillary malformation is common vascular malformation. In case of facial capillary malformation, patients' cosmetic and functional deficits are quite significant. The standard treatment which has been applied so far for capillary malformation is pulsed dye laser with 585nm. But in case of advanced capillary malformation, surgical interventions are inevitable. The problem of large size facial capillary malformation is how to cover the remnant defect, which occurs after resection. In this case, authors have experienced surgical treatment of large size facial capillary malformation and covered the large facial defect with free thoracodorsal artery perforator flap. The flap was thick, so facial asymmetry remained after the first surgery. But with the secondary procedure, authors have made more symmetric figures. The patient was satisfied with the result. Using free flap to replace the defect after resection due to capillary malformation is useful for these kinds of cases.

Airway obstruction by extrinsic tracheal compression during spinal surgery under prone position: A case report / 대한마취과학회지

Tracheal compression by vascular anomalies in adults is uncommon and most related reports are of children. A 79-year-old woman without any respiratory history underwent a lumbar spine surgery under general anesthesia. She suddenly developed airway obstruction after a position change from supine to prone. A fiberoptic bronchoscopy showed the obstruction of endotracheal tube. The obstruction was relieved after we changed the depth of endotracheal tube and supported the patient's neck with a cotton roll. The surgery ended without any other event and the patient recovered safely. A computed tomography revealed the rightward tracheal deviation and tortuous innominate artery contact with trachea. The patient didn't manifest any respiratory related symptoms during postoperative period, and she was discharged without any treatment.

Cervical Radiculopathy Caused by Vertebral Artery Loop Formation : A Case Report and Review of the Literature

Vertebral artery loop formation causing encroachment on cervical neural foramen and canal is a rare cause of cervical radiculopathy. We report a case of 61-year-old woman with vertebral artery loop formation who presented with right shoulder pain radiating to her arm for 2 years. Plain radiograph and computed tomography scan revealed widening of the right intervertebral foramen at the C5-6 level. Magnetic resonance imaging and angiogram confirmed the vertebral artery loop formation compressing the right C6 nerve root. We had considered microdecompressive surgery, but the patient's symptoms resolved after conservative management. Clinician should keep in mind that vertebral artery loop formation is one of important causes of cervical radiculopathy. Vertebral artery should be visualized using magnetic resonance angiography in suspected case.

Immediate Breast Reconstruction after Resection of Cavernous Hemangioma

PURPOSE: Hemangioma of the breast is an infrequent finding and usually encountered incidentally when checking for other disease. Most of hemangiomas of the breast are asymptomatic, not palpable perilobular type. Cavernous hemangioma of the breast is rare and only a few reports about this type of lesion are present. No example has been reported about reconstruction of the breast after resection of large cavernous hemangioma. METHODS: We report here a case of immediate breast reconstruction using a Becker implant after subcutaneous mastectomy for a large cavernous hemangioma involving almost entire breast. RESULTS: Symmetry is well maintained after 3 years without deformity or recurrence. CONCLUSION: The clinical prognosis of breast cavernous hemangioma is good after total excision and reconstruction.

Potential Role of Homer-2a on Cutaneous Vascular Anomaly

Homer protein was identified based on its rapid induction in rat hippocampal granule cell neurons following excitatory synaptic activity. Although the presence of the Homer gene in the peripheral tissues has been observed in previous reports, the physiological function of the Homer protein in these tissues has not been noted. In this experiment, a Homer-2a cDNA fragment was successfully amplified by RTPCR in the involuting phase of human hemangioma but not in the human vascular malformation and normal vessel. After isolation of full Homer cDNA in a mouse liver cDNA library, E1-deleted recombinant adenovirus expressing the Homer protein (Adv.CMV.mHomer-2a) was constructed to determine its physiological function in peripheral tissues. Adv.CMV.mHomer2a, but not Adv.CMV.LacZ (recombinant adenovirus expressing beta-galactosidase), strongly inhibited the growth rate of HUVECs (human umbilical vein endothelial cells) probably via inducing apoptosis determined by acridine orange/ethidium bromide (AO/EB) staining methods. This study suggests that the Homer gene is present in human specimens in the involuting phase of hemangioma, and it might be involved in the growth control.