Inguinal Spermatic Cord Lymphangioma a new case and literature review

Abdelhedi Chérif | Hedfi Mohamed | Bayoudh Wafa | Sridi Azza | Boulma Rami | Khouni Hassen | Sassi Karim | Chouchène Adnène |

La tunisie chirurgicale - 2017 ; Vol 2017

Resumé

Inguinal location of cystic lymphangioma is exceptional. We report the case of a 54-year- old man who presented a spermatic cord lymphangioma simulating a strangled inguinal hernia. The tumor was surgically excised.

Mots Clés

Inguinal, Spermatic, Cord Lymphangioma

Introduction :

Cystic lymphangioma is a benign tumor. Inguinal location is exceptional. In this work we report the first case simulating a strangled inguinal hernia.

Article

We report a case of a 54-year- old man who presented with progressively   increasing swelling of the right inguinal region which rapidly increased in size following a physical effort , clinical exam revealed no fever, a painful right inguinal tumefaction which is irreducible and non impulsive on cough. Furthermore, the patient didn’t have transit disorders. Testicular exam reported no anomalies. The diagnosis of strangulated hernia was retained.

Surgical exploration via a left inguinal incision found a cystic tumor developed at the expense of funiculars structures; of about 6 cm size containing clear serous fluid (fig 1). There was no inguinal or femoral hernia.  Complete resection was performed respecting the noble elements of the spermatic cord (fig2).

The anatomo-pathological exam revealed a large irregular shaped cyst spaces which are lined by a single layer of endothelium situated in a loose connective tissue stroma. Sections revealed congested fibrofatty tissue infiltrated with multiples dilated spaces lined bye endothelial cells and filled with lymph and scattred lymphocytes; concordant with the  diagnosis of lymphangioma.

The postoperative period was uneventful, and the patient was followed-up for a period of 1 year without complaints.

Fig 1: Image intraoperative: cystic structure that comes out of the spermatic cord

White Star: Inguinal cystic lymphangioma

C: cremaster muscle

 

Fig 2:  specimen:  Complete resection of the spermatic cord cystic lymphangioma

 

Discussion:

Lymphangiomas are thought to be congenital anomalies due to the abnormal development of the lymphatic vessels, arising from sequestrations of lymphatic tissues that fail to communicate normally with the lymphatic system[1].

Lymphangiomas are usually seen in the cervical region. Spermatic cord localization is, however, exceedingly rare[1-3].

To our knowledge, 18 cases of spermatic cord lyphangioma have been reported in the literature (Table I).

 

Table I: Literature Review of spermatic cord lymphangioma

Auteur

Age (-year-old)

Site of lymphangioma

Year of publisched

number of cases

El-Badawi AA[9]

unknown

Spermatic cord

1965

01

Ratan SK[6]

7

Spermatic cord

2003

01

Vázquez[10]

2

Spermatic cord

2000

01

Huang PF[11]

unknown

Spermatic cord

1987

01

ORMOND JK[12]

unknown

Spermatic cord

1951

02

Jung JL[3]

29

Spermatic cord

1996

01

Li DJ[5]

71

Spermatic cord

2007

01

GUEUDJIAN [13]

unknown

Spermatic cord

1955

09

YASIR S[8]

3

Spermatic cord

2009

01

 

 

In the case reported, the patient presented with an atypical manifestation of a strangled hernia and was directly operated. The diagnosis was re-established per operatively and then confirmed by the anatomo-pathological exam.

We discuss here our attitude of directly operating the patient even if the clinical picture was atypical we maybe should have explored the patient with further complementary exams.

Trauma and physical effort are known to be incriminated in accelerating the evolution of lymphangioma[4,5] .Therefore; we should not hesitate to push the explorations in case of doubt.

Cystic lymphangioma is more common in children[1,4–6]. This is a rare case where the lesion is discovered in an adult. The spermatic cord has a deep anatomical position explaining the fact that small lesions may remain asymptomatic for a long time.

 The importance of this case lies in the differential diagnosis of inguinal tumefaction. Its cystic nature, location and relationship to neighboring structures can be determined by ultrasound and better shown on CT scan and MRI even if these characteristics are not pathognomonic of spermatic cord lymphangioma[1,4,6–8].

 

Conclusion:

Although cystic lyphangioma is a rare entity, its diagnosis should be suspected in an inguinal tumefaction. Surgical excision must be complete to prevent recurrence.

Références

1.         Hema Kini1, Jessica Minal1, Laxman G. G Prabhu2, Pooja K Suresh1, Sridevi H Basavaiah1. Epididymal lymphangioma: An unusual cause for retrotesticular mass in an adult Kini H, Minal J, Prabhu LG, Suresh PK, Basavaiah SH - Med J DY Patil Univ [Internet].

2.         Epididymal lymphangioma: An unusual cause for retrotesticular mass in an adult Kini H, Minal J, Prabhu LG, Suresh PK, Basavaiah SH - Med J DY Patil Univ [Internet].

 3.        Jung JL, Abouelfadel Z, Straub P. Cystic lymphangioma of the spermatic cord. Report of a new case. Prog En Urol J Assoc Fr Urol Société Fr Urol. 1996 Dec;6(6):948–9.

4.          Diakité M. L, Berthé H JG, Tembely A, Cissé M C, Maiga M, Diallo M, Diakité A, Ouattara K. LE LYMPHANGIOME KYSTIQUE DU SCROTUM A PROPOS D’UN CAS. MALI MEDICAL 2011 TOME XXVI N°1;23-5.

5.         Dj L, Yt X, Wh G, Rq H, W G, Xw X, et al. Cystic lymphangioma of the spermatic cord in old man: a case report and review of the literature. Zhonghua Nan Ke Xue Natl J Androl. 2007 Sep;13(9):815–7.

6.         Ratan SK, Ratan KN, Sehgal T, Maggu S. Spermatic cord lymphangioma in a 7-year-old child masquerading as a hydrocele. BJU Int. 2003 Dec;92 Suppl 3:e33.

7.         Grossgold ET, Kusuda L. Scrotal Lymphangioma in an Adult. Urology. 2007 Sep 1;70(3):590.e1–590.e2.

8.         Jamal Yasir S, Abdel-Rahman R. E, Moshref SS, Mazen O. Kurdi. Cystic Lymphangioma of Spermatic Cord: A Case Report and Literature Review.

9.         el-Badawi AA, al-Ghorab MM. Tumors of the spermatic cord: a review of the literature and a report of a case of lymphangioma. J Urol. 1965 Oct;94(4):445–50.

10.       Vázquez Rueda F, Moñiz Mora MV, Salas Molina J, Pizarro de Celis FJ, Ocaña Losa JM. Lymphangioma of the spermatic cord. Actas Urol Esp. 2000 Mar;24(3):260–1.

11.       Huang PF, Farnum JB. Lymphangioma of the spermatic cord. J Tenn Med Assoc. 1987 May;80(5):270–2.

12.       Ormond JK, Culp OS. Lymphangioma of spermatic cord: report of two cases. J Urol. 1951 May;65(5):906–10.

13.       GUEUDJIAN SA. Lymphangioma of the groin and scrotum. 24th ed. J. Int. Coll. Surg.; 1955; 159–70.