Successful Treatment of Inguinal Lymphocele After Angiomyomatous Hamartoma Resection During Inguinal Hernia Repair

Each year, more than 20 million inguinal hernia repairs are performed worldwide.¹ The most common complications following inguinal hernia repair include hematoma, seroma, wound/superficial infection, mesh/deep infection, vascular injury, visceral injury, chronic pain, chronic paresthesia, and recurrence.² These complications can be reduced with use of an open repair technique, such as the Lichtenstein repair.³ 

Lymphocele is defined as a cystic collection of lymphatic fluid without any inflammatory or granulomatous changes at the lymph leakage site, which occurs following trauma to the lymphatic channels.⁴ There have been few reports of lymphocele after open inguinal hernia repair; however, this complication occurs in up to 8% of inguinal lymph node dissections and lymphadenectomies for oncologic procedures.^5,6 Treatment of symptomatic lymphoceles frequently involves needle aspiration, drain placement, and sclerosis.⁴ Even with use of these techniques, surgical intervention is often required to treat lymphoceles and prevent recurrence.⁷ Although negative pressure wound therapy (NPWT) has been documented as a treatment for many types of wounds, to the authors’ knowledge, there is only one reported case in which NPWT was used to manage postoperative lymphocele.^8,9 

In general, NPWT promotes formation of granulation tissue, removes fluid from the wound, and provides an approximation of tissues in open wounds.⁹ It is important to note that wounds must be cleaned and debrided prior to therapy to avoid making the condition worse with the use of NPWT. Conversely, NPWT can cause injury to healthy skin in the form of blister formation or bleeding, and in some studies it has been associated with protein loss, in which case nutritional status and support are important.^10,11 

An even rarer finding than a lymphocele is of an angiomyomatous hamartoma. This pathology was first described in 1992 by Chan et al¹² and has been reported fewer than 50 times since its discovery.^13-16 These malformations are usually diagnosed in an excisional manner. The most common pathologic finding is the replacement of parenchymal lymph tissue by blood vessels, smooth muscle, and fibrous tissue without cellular fascicle formation.¹² The exact cause of this pathology is unknown; however, it has been proposed that it is due to disruption of lymphatic flow¹⁷ or inflammation near the lymph nodes themselves.¹² Neither recurrence nor metastasis has been reported after excision of angiomyomatous hamartoma. There seems not to be any malignant potential.¹³

The case of an inguinal hernia repair in which an angiomyomatous hamartoma was found incidentally, then complicated by inguinal lymphocele, is presented herein. Treatment included wound exploration and ligation of lymphatic channels, followed by NPWT. To the authors’ knowledge, this technique for repair has been documented only once previously⁸; it was used following a lymphoma excision.⁸ 

A 59-year-old African American male with a previous history of smoking presented for evaluation of left groin discomfort that was found to be a reducible inguinal hernia with no obstructive symptoms. The patient had no prior medical history and had not undergone surgery previously. After smoking cessation, the patient was deemed an adequate surgical candidate and underwent open inguinal hernia repair with mesh.

During the procedure, a curvilinear incision was made between the left pubic tubercle and the left anterior superior iliac spine. On exploration of the subcutaneous tissues and on top of the external oblique aponeurosis at the level of the pubic tubercle, a large confluence of lymphoid tissue was encountered. There were no signs of other lymphoid tissue. To achieve adequate exposure of the external inguinal ring, this lymphoid tissue was resected sharply, suture ligated, and sent for pathologic evaluation. The rest of the procedure was completed as per usual, and repair was performed with a polypropylene mesh in a Lichtenstein fashion. 

The patient tolerated the procedure well and was discharged the same day. Five days later, the pathology results were obtained and indicated 5 benign lymph nodes spanning an area of 5 cm × 4 cm with features consistent with angiomyomatous hamartoma. Follow-up was done by telephone, and the patient reported feeling well; thus, a clinic follow-up was scheduled for 3 weeks postoperatively. On postoperative day 20, the patient called to report increasing swelling and pain at the surgical site for several days. On physical examination at the clinic the following day, the patient was found to have mild scrotal swelling with a small fluid collection beneath the surgical incision, with no local signs of infection. A postoperative seroma was suspected, and the patient was counseled and opted for watchful waiting and close evaluation.

The follow-up appointment 10 days later (postoperative week 4) was significant for increased swelling and discomfort. Due to worsening of the collection the patient was taken to the operating suite, where he underwent wound exploration (Figure 1). The previous surgical incision was opened, and a large amount of red/clear fluid was obtained from the subcutaneous space. Approximately 700 mL of this fluid was drained, and a sample was sent to the pathology department for analysis and culture. The fluid collection was anterior to the external oblique fascia and the prior hernia repair was intact, with no signs of mesh exposure or hydrocele. There were, however, small, medial portions of actively draining lymphatic vessels; these were secured with metallic clips and fibrin sealant was also applied (Figure 2). Further exploration revealed a large connecting hydrocele, which was suture ligated. The wound was loosely closed with interrupted nylon sutures and packed with sterile gauze (Figure 3). Final fluid analysis was negative for any organisms, and triglyceride content could not be determined owing to the presence of old blood products. 

Postoperatively, the dressing was changed twice a day for the first 2 days, after which the patient was transitioned to NPWT. A polyvinyl alcohol sponge followed by a polyurethane sponge were placed and -125 mm Hg continuous NPWT applied to allow complete drainage of exudate. The patient was discharged on postoperative day 4 with home NPWT with dressing changes to be performed twice a week. Three weeks after surgery, the incision closed to 1 cm. At that time NPWT was stopped and the patient was transitioned to AQUACEL Ag Hydrofiber Dressing (ConvaTec). At the 2-month follow-up, the wound completely healed, and all wound care was stopped.

The authors presented a case of an open inguinal hernia repair with an incidentally discovered and excised angiomyomatous hamartoma. Postoperative recovery was complicated by inguinal lymphocele, which was explored and closed secondarily with NPWT with good results. Another consideration for the development of this postoperative fluid collection would have been an untreated communicating hydrocele; however, the authors did not find evidence of this during the second exploration. Although closer follow-up may have helped identify the problem early, initially the authors would have opted for conservative treatment based on the small size; thus, the outcome likely would have been the same. 

Complications following inguinal hernia repair occur in up to 19% of cases; the most common complications are persisting pain and hematoma.² Lymphocele is a common complication following groin surgery (eg, inguinal lymphadenectomy, femoral artery exposure during vascular interventions).^5,6 Lymphocele is estimated to occur in approximately 1% to 4% of femoral artery dissections.¹⁸ The major consequence of these lymph leaks or lymphocele is the development of wound infection, which has been reported in up to 57% of cases.¹⁹ Attempted nonoperative repair of lymphoceles, such as aspiration, drainage, and sclerosis, is often unsuccessful. Long-term repair often includes surgical exploration with ligation of lymphatic vessels.⁷

With the advent of NPWT for effluent control and to aid in wound healing, this therapy has been used in the management of lymph leaks, although only case reports have been published to date.^8,20,21 In most of these cases, a persistent leak developed by the second or third postoperative week; in some instances, such leaking was refractory to other therapies such as exploration and ligation of visible ducts. Resolution of the leak after therapy initiation has been reported at about 2 to 3 weeks after start of therapy, which is compatible with the present case.^8,20 It is the authors’ belief that in the absence of infection and with an adequate nutritional status, NPWT can be a safe and effective treatment option.^8,20,22 Historically, one of the main disadvantages of NPWT was the large device needed; however, portable devices are now available that permit patients to be more mobile and undergo treatment at home. 

Inguinal masses can originate as primary masses from tissues native to the inguinal region or as metastatic masses from distant locations. Primary masses can form from connective tissue, nerve sheaths, muscle, fat, blood vessels, and lymphoid tissue.²³ Angiomyomatous hamartoma is a very rare primary mass of the lymph nodes, which has a predisposition for the inguinal region. Since the first description of angiomyomatous hamartoma in 1992,¹² there have been less than 50 documented cases.^13-16 Chan et al¹² described the condition as replacement of lymphoid tissue with vascular, smooth muscle, and connective tissues, starting at the hilum of the lymph node and spreading to the capsule. Due to these histological characteristics, diagnosis of angiomyomatous hamartoma is definitively made only after excision and pathologic examination, as was done in the case presented herein. In terms of lymphocele, to the authors’ knowledge, there are no reports of it after angiomyomatous hamartoma excision; however, a case of lymphocele after a groin lymphoma excision treated successfully with NPWT has been reported.⁸

In general, extreme care should be taken during any inguinal dissection, whether for hernia repair or other purposes to prevent postoperative lymph leak when damaging lymphatic structures and potential lymphatic masses such as the angiomyomatous hamartoma. Injury to the structures risks postoperative lymphocele, which can require reoperation and advanced wound care. 

The main limitation of this study is that it is a case report on a rare disease. Further studies are needed to provide specific recommendations. The authors recommend that adequate control of lymphatics (either with metallic clips or suture) always be ensured when operating on the groin area to prevent lymphocele. 

This case report details a complication following excision of an angiomyomatous hamartoma in the inguinal region. After disruption of the lymphatic vessels by excision of the hamartoma, lymphocele formation can occur. Nonoperative intervention frequently fails, requiring reoperation to drain the lymphocele and ligate the leaking lymphatic vessels. Following this procedure, NPWT is a plausible and convenient option to manage the lymphocele wound and accelerate wound closure. To the authors’ knowledge, this is the first documented use of NPWT for lymphocele following angiomyomatous hamartoma excision during inguinal hernia repair.

Authors: Raphael H. Parrado, MD; Christopher S. Thomas, MD, MS; and David Countryman, MD

Affiliation: Division of General Surgery, Department of Surgery, Medical University of South Carolina, Charleston, SC

Correspondence: Raphael Parrado, MD, 96 Jonathon Lucas Street, CSB 423-A, MSC 616, Charleston, SC 29425; Parrado@musc.edu

Disclosure: The authors disclose no financial or other conflicts of interest.