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Cystic lymphangioma: Trauma may be a significant cause?
International Journal of Pediatric Otorhinolaryngology (2007) 71, 1921—1923
www.elsevier.com/locate/ijporl
CASE REPORT
Cystic lymphangioma: Trauma may be a significant cause? Han Su Kim *, Eun A Kim, Sung Min Chung, You Ree Shin Department of Otolaryngology, Ewha Womans University School of Medicine, 911-1 MokDong, YangCheon-Gu, Seoul, Republic of Korea Received 15 December 2006; received in revised form 16 August 2007; accepted 16 August 2007
KEYWORDS Lymphangioma; Etiology; Trauma
Summary Cystic lymphangioma is a rare congenital anomaly of the lymphatic system. Its embryopathogenesis is still controversial. Most lesions present in infancy or early childhood with a swelling found in the head and neck region. We report the case of a woman who presented with a cystic swelling in the left supraclavicular region that appeared after left side thyroidectomy. Surgical excision achieved complete removal. Histology showed the lesion to be a cystic lymphangioma. It can be hypothesized that an embryogenetic origin of the adult lymphangioma is associated with some precipitating trauma such as surgery. # 2007 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Cystic lymphangiomas are almost diagnosed by 3 years of age and 90% of lymphangiomas are found in 1-year-old [1]. An occurrence of cystic lymphangioma in an adult is very rare and it is thought that the cause of this condition results from a congenital origin [2]. We report a case of cystic lymphangioma in an adult, which suggests that trauma is probably a cause of occurrence.
2. Case report A 62-year-old woman visited the otolaryngology department for the evaluation of a non-painful * Corresponding author. Tel.: +82 2 2650 2686; fax: +82 2 2653 5135. E-mail address: [email protected] (H.S. Kim).
swelling on the left supraclavicular area, which had been found incidentally 7 days prior. She underwent a left thyroid lobectomy due to papillary thyroid papillary carcinoma 5 years ago. Examination showed a 4 cm 5.5 cm sized cyst-like mass on the left supraclavicular region (Fig. 1). There was no erythema, warmth or tenderness on palpation. Aspiration of the mass showed the presence of a bloody-colored serous fluid. Computed tomograpy imaging of the neck showed a cystic mass deep to the sternocleidomastoid muscle (Fig. 2). Although there was no evidence of malignancy following an analysis of the aspirate, we could not completely rule out the possibility of recurrence of thyroid malignancy with cystic change; therefore, the patient underwent a surgical excision of the lesion (Fig. 3). The lesion was well encapsulated and contained straw-colored serous fluid. A histological examination of the lesion revealed a network of spaces varying in size with some large cystic spaces
0165-5876/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2007.08.018
1922
Fig. 1 Preoperative findings. A 4 cm x5.5 cm sized soft cyst-like mass was palpated on the left supraclavicular region.
Fig. 2 Contrast-enhanced Neck CT scan. A: axial scan and B: coronal scan; there is a 31 mm 43 mm 36 mm sized, well-defined homogenous low density mass in the left supraclavicular fossa (arrow). The left thyroid is not present due to thyroid lobectomy. This mass compresses the internal jugular vein. The left sternocleidomastoid muscle is displaced anteriorly.
H.S. Kim et al.
Fig. 3 Intra-operative findings. A dark-yellow cystic mass was excised.
and smaller channels. The spaces were lined by bland and flattened cells (Fig. 4A and B). These features are indicated in the diagnosis of a cystic lymphangioma.
Fig. 4 Histopathologic findings. (A) In the fibrovascular stroma, lymphatic vessels covered with single layer endothelial cells seemed to become a cyst (H&E stain, 40); (B) the lymphocytes are located under the endothelial layer with a small amount of erythrocytes in the lumen (H&E stain, 200).
Cystic lymphangioma in an adult
1923 it was a single lymphatic sac with a good plane of dissection. Recently picibanil injection has been used for the treatment of cystic lymphangioma and seems to provide good results [8]. We feel if the diagnosis is certain and the lesion is cystic, injection of picibanil is an alternative treatment modality that can be utilized instead of surgical excision in adult cases.
4. Conclusion Fig. 5 Pre-thyroid lobectomy ultrasonogram. A 0.51 cm sized intra-parenchymal nodule was noted at the left thyroid gland. Other abnormal findings such as cyst were not found.
3. Discussion Cystic lymphangioma is a rare congenital anomaly usually observed in children [1,3] and seen very rarely in adults [2]. The origin of the lymphatic system and its anomalies are still not clearly defined. It has been proposed that lymphangiomas occur when there is failure of the lymphatic system to connect with the venous system [4]. The etiology of lymphangioma in adults is likely due to delayed proliferation of resting cells. This delayed proliferation can occur in adults because of an instigating stimulus such as infection or trauma [5,6]. In the case of our patient, the triggering event might have been the left thyroid lobectomy as there were no cystic anomalies seen on an ultrasonogram which was performed prior to the thyroid lobectomy (Fig. 5). The common treatment of cystic lymphangioma has been surgical excision; however, as the lymphatic channels invade the adjacent tissues and destroy the normal plane of dissection, a complete removal of lesion is very difficult. Nevertheless some lesions are well encapsulated [7]. In this case,
Although this is a single clinical case report in an adult, our case supports a clinically important assumption that a cystic lymphangioma can occur by not only congenital causes but also as a result of a physical injury, such as surgery or trauma. Therefore it is important to take into account the diagnosis of cystic swelling that may appear following trauma in pediatric patients.
References [1] D.R. James, Craniofacial anomalies, in: A.G. Kerr, J.B. Booth (Eds.), Scott Brown’s Otolaryngology, 5th ed., ButterworthHeinemann, London, 1987, p. 267. [2] S. Baer, J. Davis, Cyst hygroma presenting in adulthood, J. Laryngol. Otol. 103 (1989) 976—977. [3] B.J. Hancock, D. St-Vil, F.I. Luks, M. Di Lorenzo, H. Blanchard, Complications of lymphangiomas in children, J. Pediatr. Surg. 27 (1992) 220—226. [4] J.L. Guarisco, Congenital head and neck masses in infants and children, Ear Nose Throat J. 70 (1991) 75—82. [5] S. Suk, M. Sheridan, J.S. Saenger, Adult lymphangioma: a case report, Ear Nose Throat J. 76 (1997) 881—883. [6] W.J. Wiggs Jr., A. Sismanis, Cystic hygroma in the adult: two case reports, Otolaryngol. Head Neck Surg. 110 (1994) 239—241. [7] M.K. Aneeshkumar, S. Kale, M. Kabbani, V.C. David, Cystic lymphangioma in adults: can trauma be the trigger? Eur. Arch. Otorhinolaryngol. 262 (4) (2005) 335—337. [8] C. Brewis, J.P. Pray, D.M. Albert, Treatment of lymphangiomas of the head and neck in children by intralesional injection of OK-432, Clin. Otolaryngol. 25 (2) (2000) 130—134.
www.elsevier.com/locate/ijporl
CASE REPORT
Cystic lymphangioma: Trauma may be a significant cause? Han Su Kim *, Eun A Kim, Sung Min Chung, You Ree Shin Department of Otolaryngology, Ewha Womans University School of Medicine, 911-1 MokDong, YangCheon-Gu, Seoul, Republic of Korea Received 15 December 2006; received in revised form 16 August 2007; accepted 16 August 2007
KEYWORDS Lymphangioma; Etiology; Trauma
Summary Cystic lymphangioma is a rare congenital anomaly of the lymphatic system. Its embryopathogenesis is still controversial. Most lesions present in infancy or early childhood with a swelling found in the head and neck region. We report the case of a woman who presented with a cystic swelling in the left supraclavicular region that appeared after left side thyroidectomy. Surgical excision achieved complete removal. Histology showed the lesion to be a cystic lymphangioma. It can be hypothesized that an embryogenetic origin of the adult lymphangioma is associated with some precipitating trauma such as surgery. # 2007 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Cystic lymphangiomas are almost diagnosed by 3 years of age and 90% of lymphangiomas are found in 1-year-old [1]. An occurrence of cystic lymphangioma in an adult is very rare and it is thought that the cause of this condition results from a congenital origin [2]. We report a case of cystic lymphangioma in an adult, which suggests that trauma is probably a cause of occurrence.
2. Case report A 62-year-old woman visited the otolaryngology department for the evaluation of a non-painful * Corresponding author. Tel.: +82 2 2650 2686; fax: +82 2 2653 5135. E-mail address: [email protected] (H.S. Kim).
swelling on the left supraclavicular area, which had been found incidentally 7 days prior. She underwent a left thyroid lobectomy due to papillary thyroid papillary carcinoma 5 years ago. Examination showed a 4 cm 5.5 cm sized cyst-like mass on the left supraclavicular region (Fig. 1). There was no erythema, warmth or tenderness on palpation. Aspiration of the mass showed the presence of a bloody-colored serous fluid. Computed tomograpy imaging of the neck showed a cystic mass deep to the sternocleidomastoid muscle (Fig. 2). Although there was no evidence of malignancy following an analysis of the aspirate, we could not completely rule out the possibility of recurrence of thyroid malignancy with cystic change; therefore, the patient underwent a surgical excision of the lesion (Fig. 3). The lesion was well encapsulated and contained straw-colored serous fluid. A histological examination of the lesion revealed a network of spaces varying in size with some large cystic spaces
0165-5876/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2007.08.018
1922
Fig. 1 Preoperative findings. A 4 cm x5.5 cm sized soft cyst-like mass was palpated on the left supraclavicular region.
Fig. 2 Contrast-enhanced Neck CT scan. A: axial scan and B: coronal scan; there is a 31 mm 43 mm 36 mm sized, well-defined homogenous low density mass in the left supraclavicular fossa (arrow). The left thyroid is not present due to thyroid lobectomy. This mass compresses the internal jugular vein. The left sternocleidomastoid muscle is displaced anteriorly.
H.S. Kim et al.
Fig. 3 Intra-operative findings. A dark-yellow cystic mass was excised.
and smaller channels. The spaces were lined by bland and flattened cells (Fig. 4A and B). These features are indicated in the diagnosis of a cystic lymphangioma.
Fig. 4 Histopathologic findings. (A) In the fibrovascular stroma, lymphatic vessels covered with single layer endothelial cells seemed to become a cyst (H&E stain, 40); (B) the lymphocytes are located under the endothelial layer with a small amount of erythrocytes in the lumen (H&E stain, 200).
Cystic lymphangioma in an adult
1923 it was a single lymphatic sac with a good plane of dissection. Recently picibanil injection has been used for the treatment of cystic lymphangioma and seems to provide good results [8]. We feel if the diagnosis is certain and the lesion is cystic, injection of picibanil is an alternative treatment modality that can be utilized instead of surgical excision in adult cases.
4. Conclusion Fig. 5 Pre-thyroid lobectomy ultrasonogram. A 0.51 cm sized intra-parenchymal nodule was noted at the left thyroid gland. Other abnormal findings such as cyst were not found.
3. Discussion Cystic lymphangioma is a rare congenital anomaly usually observed in children [1,3] and seen very rarely in adults [2]. The origin of the lymphatic system and its anomalies are still not clearly defined. It has been proposed that lymphangiomas occur when there is failure of the lymphatic system to connect with the venous system [4]. The etiology of lymphangioma in adults is likely due to delayed proliferation of resting cells. This delayed proliferation can occur in adults because of an instigating stimulus such as infection or trauma [5,6]. In the case of our patient, the triggering event might have been the left thyroid lobectomy as there were no cystic anomalies seen on an ultrasonogram which was performed prior to the thyroid lobectomy (Fig. 5). The common treatment of cystic lymphangioma has been surgical excision; however, as the lymphatic channels invade the adjacent tissues and destroy the normal plane of dissection, a complete removal of lesion is very difficult. Nevertheless some lesions are well encapsulated [7]. In this case,
Although this is a single clinical case report in an adult, our case supports a clinically important assumption that a cystic lymphangioma can occur by not only congenital causes but also as a result of a physical injury, such as surgery or trauma. Therefore it is important to take into account the diagnosis of cystic swelling that may appear following trauma in pediatric patients.
References [1] D.R. James, Craniofacial anomalies, in: A.G. Kerr, J.B. Booth (Eds.), Scott Brown’s Otolaryngology, 5th ed., ButterworthHeinemann, London, 1987, p. 267. [2] S. Baer, J. Davis, Cyst hygroma presenting in adulthood, J. Laryngol. Otol. 103 (1989) 976—977. [3] B.J. Hancock, D. St-Vil, F.I. Luks, M. Di Lorenzo, H. Blanchard, Complications of lymphangiomas in children, J. Pediatr. Surg. 27 (1992) 220—226. [4] J.L. Guarisco, Congenital head and neck masses in infants and children, Ear Nose Throat J. 70 (1991) 75—82. [5] S. Suk, M. Sheridan, J.S. Saenger, Adult lymphangioma: a case report, Ear Nose Throat J. 76 (1997) 881—883. [6] W.J. Wiggs Jr., A. Sismanis, Cystic hygroma in the adult: two case reports, Otolaryngol. Head Neck Surg. 110 (1994) 239—241. [7] M.K. Aneeshkumar, S. Kale, M. Kabbani, V.C. David, Cystic lymphangioma in adults: can trauma be the trigger? Eur. Arch. Otorhinolaryngol. 262 (4) (2005) 335—337. [8] C. Brewis, J.P. Pray, D.M. Albert, Treatment of lymphangiomas of the head and neck in children by intralesional injection of OK-432, Clin. Otolaryngol. 25 (2) (2000) 130—134.