Retiform Hemangioendothelioma Developed on the Site of an Earlier Cystic Lymphangioma in a Six-Year-Old Girl

Albertini, Anne-Fore; Brousse, Nicole; Bodemer, Christine; Calonje, Eduardo; Fraïtag, Sylvie

doi:10.1097/dad.0b013e31821b0a9f

Cited by 26 publications

(24 citation statements)

References 16 publications

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“…The patient had rapid tumor local recurrence 3 months after excision and died of disease 6 months after excision [14]. Also, Albertini et al reported a lethal RH case occurring in a 6-year-old girl, in which the patient died of hypovolemic shock 8 months after surgery because of pre-existed diffuse lymphangiomatosis [18]. Those lethal cases indicate that RH may exhibit more aggressive behavior than originally described by Calonje [2].…”

Section: Discussionmentioning

confidence: 79%

Unusual multiple cutaneous retiform hemangioendothelioma on forearm and neck misdiagnosed as angiosarcoma with metastasis

Pan¹,

Wang²,

Huang³

et al. 2017

Clin Diagn Pathol

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Retiform hemangioendothelioma (RH) is extremely rare, and often involves the skin and subcutaneous tissues of distal extremities in young adults or children. Since its first description by Calonje in 1994, only a few primary multiple cases have been described in the literature. We present a case of unusual primary multiple RH on forearm and neck occurring in a 56 years old female patient. The patient presented with a slow-growing cutaneous plaque-like lesion on her left forearm, followed by another lesion at the site of neck for several years. In the skin biopsy examination, a diagnosis of angiosarcoma with cutaneous metastasis was made based on multiple lesions at different anatomic sites and vasoformative growth pattern with anastomosing channels under the microscopy. However, postoperative histological diagnosis of the lesion was primary multiple RH by thoroughly microscopical inspection and the presence of thin-walled interconnecting vascular channels arranged in a retiform pattern and absence of lymph node metastasis. Despite wide surgical excision with tumor-free margin, the tumor recurred at the neck 3 months after surgery. The second local excision was performed. The patient was on regular follow-up for 18 months without additional treatments. There was no sign of recurrence or lymph node metastasis. Due to its rarity, multiple RH might be erroneously interpreted as angiosarcoma with metastasis by those who were not familiar with this condition, or conduce to aggressive postoperative treatment. Therefore, thoroughly microscopical inspection and a long-term follow-up are helpful for surgeons and pathologists to obtain an accurate diagnosis and avoid clinical over-treatment.

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Section: Discussionmentioning

confidence: 79%

Unusual multiple cutaneous retiform hemangioendothelioma on forearm and neck misdiagnosed as angiosarcoma with metastasis

Pan¹,

Wang²,

Huang³

et al. 2017

Clin Diagn Pathol

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“…Therefore, the clinical recognition of RH is extremely difficult because of its non-specific appearance with differential diagnosis There is a wide variability in the age of patients diagnosed with RH; usually it occurs in the second to fourth decade of life with a mean age of 36 years (1,2). RH has also been reported in patients with less than 10 years of age (6). The tumor shows a predilection for females.…”

Section: Discussionmentioning

confidence: 99%

Retiform hemangioendothelioma of the lower limbs: a case-based review

Zarian¹

2015

cderm

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SummaryRetiform hemangioendothelioma is a rare entity included in the spectrum of vascular tumors. It is considered an uncommon vascular neoplasm of intermediate or borderline malignancy (between entirely benign hemangiomas and highly malignant angiosarcomas). Retiform hemangioendothelioma is clinically characterized by exophytic nodules and plaques in the dermal or subcutaneous compartments, which grows slowly involving more often lower limbs, upper limbs and trunk. Histologically, it shows a typical vascular pattern characterized by microscopic feature of arborizing, elongated, thin-walled cavernous blood vessels dissecting the dermal collagen. In this article, we report a case of retiform hemangioendothelioma in a 86-year-old male and review the literature concerning this rare entity.

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“…There are generally three isoforms of HE; an intermediate-locally aggressive isoform (kaposiform), an intermediate-rarely metastasizing isoform (retiform/composite), and a malignant isoform (epithelioid) [1,2]. Pathologic isoforms exhibit differences in malignancy, recurrence, metastasis, and prognosis.…”

Section: Discussionmentioning

confidence: 99%

“…Retiform/composite HE shows a recurrence rate and metastasis rate of less than 2% [1,2]. Epithelioid HE has recurrence rate of 10% to 15%, a metastasis rate of 20% to 30%, and a mortality rate of 10% to 20% [1,3,10].…”

Section: Discussionmentioning

confidence: 99%

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Soft Tissue Hemangioendothelioma in Children

Youn

Jung

Kim

et al. 2017

J Korean Assoc Pediatr Surg

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Purpose: Soft tissue hemangioendothelioma (STHE) is a rare vascular tumor, which has a similar prognosis to borderline malignancy. The disease is poorly understood in pediatric cases because of its low incidence; therefore, we investigated treatment strategies for STHE in children. Methods: We retrospectively analyzed 8 patients with STHE, who were pathologically confirmed between January 1995 and June 2015. The median duration of follow-up was 72 months. Results: Five were male and the median age at the time of surgery was 1.2 years. Six patients presented with a palpable mass, and 2 patients experienced facial paralysis. The median tumor size was 4.0 cm. The following tumor locations were observed head (2 patients), neck (2 patients), chest wall (1 patient), sacrococcyx (1 patient), upper limb (1 patient), and lower limb (1 patient). The patients underwent either microscopic complete resection (R0) (3 patients), macroscopic complete resection (R1) (2 patients), or macroscopic incomplete resection (R2) (3 patients). After histopathological examination, 6 patients were diagnosed with kaposiform hemangioendothelioma (HE), one with retiform HE, and one with epithelioid HE. Postoperative sequelae occurred as gait disturbance, hearing impairment, and vocal cord palsy. Tumor recurrence or regrowth occurred in 4 patients. These patients underwent reoperation and IFN therapy; however, in the patient with epithelioid HE, metastasis to the scalp occurred after these therapies. The patient with the head tumor who underwent R2 resection, underwent resection three more times, but died 11 months after the first surgery. Conclusion: When treating STHE in children, R0 resection should be first considered, but recurrence and metastasis should be monitored depending on the size, pathology, and location of the lesion. When major sequelae are expected, function-preserving surgery could be considered, depending on tumor location, size, and nearby organs.

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Retiform Hemangioendothelioma Developed on the Site of an Earlier Cystic Lymphangioma in a Six-Year-Old Girl

Cited by 26 publications

References 16 publications

Unusual multiple cutaneous retiform hemangioendothelioma on forearm and neck misdiagnosed as angiosarcoma with metastasis

Unusual multiple cutaneous retiform hemangioendothelioma on forearm and neck misdiagnosed as angiosarcoma with metastasis

Retiform hemangioendothelioma of the lower limbs: a case-based review

Soft Tissue Hemangioendothelioma in Children

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