Hemolymphangioma of the waist A case report and review of the literature

Hemolymphangioma of the waist - A case report and review of the literature脉管瘤

ONCOLOGY LETTERS 9: 2629-2632, 2015

Hemolymphangioma of the waist: A case report and review of the literature

YONGCHAO LI1,2*, XIAODONG PANG2*, HONG YANG2, CHUNHUA GAO2 and BAOGAN PENG2

1

Postgraduate Training Base, General Hospital of Armed Police Force, Liaoning Medical University, Jinzhou, Liaoning 121000; 2Department of Spinal Surgery, Institute of Spinal Surgery of Armed Police Force,

General Hospital of Armed Police Force, Beijing 100039, P.R. China

Received June 26, 2014; Accepted February 26, 2015

DOI: 10.3892/ol.2015.3071

Abstract. Hemolymphangioma is a malformation of the lymphatic and blood vessels. To the best of our knowledge, only a limited number of hemolymphangioma cases have been reported in the literature thus far, with no cases developed in the waist region. The present study reported the case of a 17-year-old male patient with hemolymphangioma growing on the waist, presented with back pain for four months. Upon physical examination, the lesion was identified to be oval in shape, soft and compressible, with mild tenderness. No abnor-malities were detected in the results of laboratory examinations. However, a magnetic resonance imaging (MRI) scan revealed a tumor with low signal intensity on T1-weighted imaging (WI) and high signal intensity on T2-WI. The mass was successfully removed during surgery. During the seven-month follow-up period, the patient was asymptomatic with no evidence of recurrence. The present study discussed the imaging findings and pathological features of this uncommon case and reviewed the relevant literature.

Hemolymphangioma is a congenital malformation that may be asymptomatic for a long period of time (3). This lesion is typically considered to be a benign and noninvasive disorder, characterized by the presence of dilated lymphatic spaces, extravasation of red blood cells, hemosiderin deposi-tion and fibrosis (4). Hemolymphangioma formation may be due to venolymphatic communication obstruction between the dysembrioplastic vascular tissue and systemic circulation (5).The incidence of hemolymphangioma is 1.2-2.8 per 1,000 newborn infants (6). Hemolymphangioma has been previously detected at the pancreas (5,7-12), spleen (13-16), stomach (1,17), rectum (18), mediastinum (19-21), chest wall (22-25), small intestine (26), extremities (3,27,28), cervix (29,30), pericardium (31), oral region (32), esophagus (33), axilla (34), retroperitoneal space (35,36), adrenal gland (37), abdomen (38), duodenum (4) and hepatica (39), as well as on the tongue (40,41) and orbit (42,43). However, to the best of our knowledge, no studies have reported this type of tumor in the waist region, as determined by a review of the

Introductionmedical literature until June 2014 using the PubMed data-base (http://www.ncbi.nlm.nih.gov/pubmed; accessed on 9th Lymphangioma, also known as angioma lymphaticum, June 2014). Complete excision is considered the optimal treat-is a congenital malformation of the vascular system, ment for hemolymphangioma, which exhibits a low recurrence comprising newly-formed lymph spaces and channels (1). rate. Non-surgical treatments are also used, including aspira-Landing and Farber (2) classified this benign malformation tion and drainage, cryotherapy, injection of sclerotic agents, in four categories, including capillary, cavernous and cystic laser therapy and radiotherapy, however, to date, the outcomes (hygroma) lymphangioma, and hemolymphangioma, which is of such treatments have been unsatisfactory (3,22). In cases a combination of hemangioma and lymphangioma.of tumor recurrence, conservative treatment methods such as

laser therapy, may be applied (3,44). Generally, the prognosis of hemolymphangioma is good (3,4,13,17,18), however, careful follow-up is required.

The present study reported the case of a 17-year-old male patient with hemolymphangioma on the waist and reviewed the

Correspondence to: Professor Baogan Peng, Department of Spinal characteristics of this disease based on the existing literature.

Surgery, Institute of Spinal Surgery of Armed Police Force, General Hospital of Armed Police Force, 69 Yongding Road, Beijing 100039, P.R. China

E-mail: pengbaogan76611@

*

Case report

A 17-year-old male was admitted at the General Hospital of Armed Police Force (Beijing, China) in September 2013, complaining of a mass on the right side of the waist and back pain for approximately four months. The back pain was significantly increased when the patient was sedentary and was slightly alleviated by rest. On admission, the patient was

Contributed equally

Key words: waist, hemolymphangioma, back pain

Hemolymphangioma of the waist - A case report and review of the literature脉管瘤

2630LI et al: HEMOLYMPHANGIOMA OF THE WAIST

A

B

C

Figure 1. Magnetic resonance imaging (MRI) findings. The scans demonstrate the (A) coronal T1 weighted imaging (WI), (B) coronal T2 WI and -(C) axial T2 WI. A mass (arrows) was identified, with low signal intensity on T1-WI and high signal intensity on T2-WI in the right side of the waist

subcutaneous tissue with the fifth lumbar level parallel.

Figure 2. Histological analysis of biopsy specimen (hematoxylin and eosin stain; magnification, x100). Blood vessels (red arrow) and lymphatic vessels (blue arrow) can be observed in the tumor.

well, with no symptoms of lower extremity numbness and pain, or abdominal pain. Upon physical examination, the lesion was identified to be oval in shape, soft and compressible, with mild tenderness. No abnormalities were detected -in the results of laboratory examinations. An ultrasound (Doppler sonography) detected a cystic lesion (15.0x10.0 cm) with blood flow, and revealed multiple echo and irregular cavities in the subcutaneous fat layer of the right waist. The most likely diagnosis was hemolymphangioma.

performed preoperatively, in order to establish the extent of Magnetic resonance imaging (MRI) examination was the tumor and define its association with the surrounding tissues. A waist MRI scan (Fig. 1) revealed a 12.6x9.7-cm mass, with low signal intensity on T1-weighted imaging (WI) and high signal intensity on T2-WI in the right side of the waist subcutaneous tissue with the fifth lumbar leveUpon performing a tumor biopsy with a 5-ml syringe, 10-ml l parallel. yellow, clear liquid was extracted and laboratory examina-tions were performed. Preoperatively, no abnormalities were revealed in the laboratory data, including the levels of tumor markers [hydrate antigen (CA) 19-9, and CA-125] and concentrated α-fetoprotein, carcinoembryonic antigen, carbo-mycobacterium tuberculosis.

increased upon sitting, surgical excision was decided as Due to the patient experiencing back pain that was the treatment strategy. During surgery, the boundary of the mass was unclear and a bloody yellow exudate was observed. Macroscopically, the mass measured approximately 12.0x6.0x6.0 cm, and was oval and soft. Multiloculated cystic masses filled with blood and yellow fluid were extracted. Histologically, the tumor was composed of lymphatic and blood vessels with polycystic spaces (Fig. 2). Considering these observations, the definitive histological diagnosis was hemolymphangioma of the waist. The postoperative course of the patient was uneventful. In the course of a seven-month follow-up period, no recurrence of hemolymphangioma was observed. This study was approved by the ethics committee of General Hospital of Armed Police Force (Beijing, China)

Hemolymphangioma of the waist - A case report and review of the literature脉管瘤

ONCOLOGY LETTERS 9: 2629-2632, 2015

2631

and written informed consent was obtained from the patient's family.

while the recurrence rates were 50-100% in cases where the lesions were partially removed (4,52). The extend of surgical resection depends mainly on the anatomical location and

Discussioncomplexity of the tumor (3,4,12,52).

In conclusion, hemolymphangioma of the waist is an

Hemolymphangiomas, a congenital malformation of uncommon vascular and lymphatic lesion, presenting the vascular system, can be classified into primary mainly with back pain. Preoperative imaging examinations, and secondary lymphatic vascular tumors. Primary including ultrasound and MRI, are important for a full evalu-tumors are congenital malformations of the lymphatic ation of the tumor in order to confirm the diagnosis and plan vascular system, possibly formed due to obstruc-the surgical strategy. Complete surgical resection is the most tion of the venolymphatic communication between effective treatment with good prognosis.the dysembryoplastic vascular tissue and the systemic circu-lation. By contrast, secondary tumors are likely to be caused by poor lymph drainage and lymphatic damage resulting

from surgery or trauma (43). Hemolymphangioma mainly presents as cystic or cavernous lesions. Histologically,

hemolymphangioma is composed of dense fibrous tissue that develops in bands between the numerous vascular spaces, invading the subcutaneous fat and involving the blood or

lymphatic vessels (3).newborns (45), and the two genders are equally affected. In

The incidence of hemolymphangioma is 1.2-2.8 per 1,000 the present study, a review of the literature up to June 2014 performed using the PubMed database. The search strategy to was identify all possible studies involved use of the word ‘hemo-lymphangioma’type of tumor were identified (1,3 42,44,46 50). However, to . In total, 47 previous studies concerning this the best of our knowledge, no studies have reported hemo-lymphangioma of the waist. In the current case, the patient was a 17-year-old male, and the tumor occurred on the waist and appeared as a cystic lesion.

between a slow-growing cyst over a period of years and an Clinically, the onset of hemolymphangioma can vary aggressive enlarging tumor without invasive ability (3). The

size of these tumors varies due to the different anatomical location and association with the neighboring tissues. In clinical examinations, they are usually described as soft and

compressible masses, loculated in pattern. The most common complications are random or traumatic hemorrhage, rupture and infection (3). However, no abnormal laboratory findings

were observed in the current patient, and the only symptom was back pain for four months.with low signal intensity on T1-WI and high signal intensity In the present study, a waist MRI scan revealed a tumor on T2-WI on the right side of the waist subcutaneous tissue in the fifth lumbar levelthe presence of a lower number of tortuous blood vessels and . These observations may indicate

water based substance in the lesion, which was then confirmed during surgery. Imaging examinations, including ultrasound,

computed tomography and MRI scans, are useful in order to confirm the diagnosis, identify the tumor nature, and its extension and association with the surrounding tissues,

observe assisting the selection of the surgical strategy and follow-up treatment (51). However, a definitive diagnosis of the tumor in the present study was based on histological evidence.

ment for hemolymphangioma. In order to prevent recurrence, Surgical resection appears to be the most effective treat-thorough radical resection may be required during surgery.

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