Ameur Wafa Ben, Kallel Lamia, Maghrebi Houcine, Haouet Slim, Filali Azza
Service de Gastroentérologie A, Hôpital la Rabta, Tunis, Tunisie.
Service de Chirurgie Générale et Digestive A, Hôpital la Rabta, Tunis, Tunisie.
Pan Afr Med J. 2018 May 18;30:48. doi: 10.11604/pamj.2018.30.48.14601. eCollection 2018.
Cystic lymphangioma is a rare benign malformative tumor of the lymphatic vessels which may occur in various locations. Intra-abdominal cystic lymphangioma is less frequent than cervicoaxillary cystic lymphangioma. Clinical presentation is polymorphic. Diagnosis is based on imaging data but it requires histological confirmation. Surgery is the gold standard treatment. We here report a rare case of acquired intraperitoneal cystic lymphangioma secondary to subtotal colectomy in a female patient with severe evolutive Crohn's disease treated with anti-TNF alpha. The patient presented with irreducible right painful paramedian mass with no impulse on coughing, suggesting the diagnosis of strangulated eventration within a surgical scar from midline laparotomy. She underwent emergency surgery. Surgical exploration showed multi-cystic intraperitoneal mass protrunding through the right paramedian eventration. Anatomo-pathological examination helped to confirm the diagnosis of cystic lymphangioma. Postoperatively, the mass was punctured twice to evacuate the fluid, because of incomplete surgical resection. This is the first reported case of cystic lymphangioma in a patient under anti-TNF alpha. It could be caused by disruption of the immune system and more specifically of the lymphocyte population. This association has not hitherto been established and experimental studies are necessary to accept or refuse this hypothesis.
囊性淋巴管瘤是一种罕见的淋巴管良性畸形肿瘤,可发生于身体的各个部位。腹腔内囊性淋巴管瘤比颈腋窝囊性淋巴管瘤少见。临床表现多样。诊断基于影像学资料,但需要组织学证实。手术是金标准治疗方法。我们在此报告一例罕见的获得性腹腔内囊性淋巴管瘤病例,该病例发生在一名患有严重进展性克罗恩病并接受抗TNF-α治疗的女性患者行次全结肠切除术后。患者出现右侧疼痛性正中旁不可复性肿块,咳嗽时无冲动感,提示诊断为中线剖腹手术后手术瘢痕内绞窄性腹疝。她接受了急诊手术。手术探查发现一个多囊性腹腔肿块通过右侧正中旁腹疝突出。解剖病理学检查有助于确诊囊性淋巴管瘤。术后,由于手术切除不完全,肿块被穿刺两次以排出液体。这是首例报道的接受抗TNF-α治疗患者发生囊性淋巴管瘤的病例。它可能是由免疫系统尤其是淋巴细胞群的破坏引起的。这种关联迄今尚未确立,需要进行实验研究来接受或拒绝这一假设。