Traumatic Neuroma of the Anus After Milligan-Morgan Hemorrhoidectomy

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324 Annals of Clinical & Laboratory Science, vol. 44, no. 3, 2014 Traumatic Neuroma of the Anus after Milligan-Morgan Hemorrhoidectomy

Catherine Takawira1,2*, Suzan Shenouda2*, Gregor Mikuz3, and Consolato Sergi2

1Department of Oncology and Developmental Biology, Faculty of Health, Medicine & Life Sciences, Maastricht University, Maastricht, Netherlands, 2Department of Laboratory Medicine and Pathology, University of Alberta, Edmonton, AB, Canada, and 3Institute of Pathology, Medical University, Innsbruck, Austria

Abstract. There are several clinical settings of traumatic neuroma and a few may occur following surgical procedures. A 42-year-old man presented with anal pain five years after a Milligan Morgan hemorrhoidectomy for prolapsing hemorrhoids. A 4x4x3 mm sized anal polyp was seen during a rectal examination at a follow-up five years after surgery. The patient complained of point tenderness, pruritus, and anal discomfort as well as fecal retention. An endoscopy revealed a rectal polyp. Remarkably, histopathological examination and immunohistochemistry of the excised polyp showed a polypoid traumatic neuroma of the rectal plexus. After the excision of the polyp, the patient’s complaint resolved completely. Traumatic neuromas may be a cause of significant pain and tenderness in patients with anal surgery or repair of anal lacerations. Interest- ingly, this is the second case of anal traumatic neuroma since Dr. Marks’ first case in 1956 and is a possible complication of Milligan Morgan hemorrhoidectomy for prolapsing hemorrhoids. Similar complications of rectal surgery are reviewed.

Keywords: hemorrhoids; surgery; constipation; polyp and traumatic neuroma

Introduction Other complications that arise because of both hemorrhoidectomy and hemorrhoidopexy or other Traumatic neuroma is an exuberant but actually similar procedures are similarly reviewed. Stapled non-neoplastic proliferation of a nerve occurring in hemorrhoidopexy for varicosities of the hemor- response to injury or surgery [1]. After sharp trau- rhoidal plexus and Milligan Morgan hemorrhoid- ma to a peripheral nerve, the nerve ends try to re- ectomy are two surgical options. In both procedures establish continuity by an orderly growth of axons complications have been indeed reported, includ- from proximal to distal stumps through the prolif- ing postoperative bleeding, urinary retention, sep- eration of Schwann cells. However, if close apposi- sis, recto-vaginal fistula, fecal incontinence, fecal tion of the ends of a nerve is not maintained or if urgency, anal stricture, persistent anal pain, and the distal stump is destroyed, a disorganized prolif- incidence of residual disease [4,5,6]. eration of Schwann cells at the proximal end occurs, giving rise to a neuroma. Aberrant repair oc- Case Report curs when the regenerative tissues encounter a scar or otherwise cannot re-establish correct innervation A 42-year-old man presented with anal pain, pruritus, [2]. Painful neuromas can even be associated with and general paraesthesias in the perianal region. Five blunt trauma or retraction of a nerve without the years earlier, the patient underwent anal surgery because actual nerve division [3]. of varicosities of the hemorrhoidal plexus. During rec- toscopy, the patient showed a 4x4x3-mm sized polyp localized in the anus. The polyp was excised and the his- Here we describe a traumatic neuroma in the anus topathological examination of the specimen showed a following a Milligan-Morgan hemorrhoidectomy. traumatic neuroma (Figure 1a). Immunohistochemistry confirmed the proliferation of the nerve bundles using * Those two authors contributed equally to the manuscript. Address an antibody against S-100 protein (Figure 1b). One year correspondence to Dr. Consolato Sergi, MSc, MD, PhD (habil.), FRCPC; Department of Lab Medicine and Pathology, University of after the neuroma excision, the patient did not complain Alberta, Edmonton, Alberta, T6G 2B7, Canada; phone: +1 780 407 of any perianal discomfort although mild varices were 7951; e mail: [email protected] eventually observed.

Figure 1. a. Haphazard proliferation of nerve fascicles, including axons with myelin, Schwann cells and fibroblasts (Hematoxylin and eosin staining, 40X). b. Immunohistochemical detection of S-100 expressing cells in the neuroma (anti- S-100 staining, Avidin-Biotin-Complex detection method, 100X).

Discussion Since the procedure for the prolapsed hemorrhoids was first introduced in Italy in 1997, it has become Traumatic anal neuroma is a rarely documented a common practice around the world for the surgi- finding in the setting of anal pain. In both reported cal treatment of hemorrhoids. The surgical proce- cases, there was a history of trauma. The tangled dure for varicosities of the hemorrhoidal plexus is a mass of crushed nerve fibers and theox pr imal end routine one. Two surgical options are the Milligan- regenerates, but fails to meet adequately and cor- Morgan hemorrhoidectomy and the staple hemor- rectly the distal end. The traumatic neuroma is con- rhoidopexy [4,5,6,7,8]. Possible surgical complica- stituted by all the elements of a nerve, including tions of the Milligan-Morgan procedure include axons, Schwann cells, and perineural fibroblasts. In early or delayed postoperative hemorrhage, recur- some cases, scar tissue may also be observed. rence of hemorrhoids, and passive or urge incontinence. The Milligan-Morgan technique can also To reach a diagnosis of traumatic neuroma [7], the cause secondary stenosis of the anal canal through following diagnostic criteria should be applied: adhesions of the preserved skin areas. 1. Haphazard arrangement of tangles of variably sized regenerating nerve twigs; We searched PubMed in English for relevant arti- 2. Axonic terminations ensheathed by Schwann cles on complications of Milligan-Morgan hemor- cells surrounded by perineum; rhoidectomy and stapled hemorrhoidopexy to in- 3. No evidence of a capsule; clude articles published from 1965 up to May 4. Limited or no extension into adipose tissue or 2013. We used the key words “hemorrhoids”, “sur- skeletal muscle; gery” and “complication” as well as appropriate 5. Myxoid (young) or fibrous (old) stroma ac- medical subject headings (MeSH). Articles were cording to the age of the lesion; also identified from the reference lists of retrieved 6. Regenerating nerve twigs containing neurofila- literature. Only English language studies were re- ments or Bielschowsky positive terminal axons (op- viewed. To the best of our knowledge, this is the tional special stain or immunohistochemistry); second case of traumatic neuroma subsequent to 7. S100 positive surrounding Schwann cells (op- rectal surgery. tional immunohistochemistry); 8. Nerve twigs optionally stained with epithelium In fact, the first case of traumatic neuroma of the membrane antigen (EMA) and Glucose transporter anus was described in 1956 [9]. In the original re- 1 (GLUT1) monoclonal antibodies to the perineu- port by Dr. Marks, a 58-year-old man presented rium, and with pain on defecation. Similarly, to our patient, 9. Curative excision. symptoms have been slowly increasing in severity 326 Annals of Clinical & Laboratory Science, vol. 44, no. 3, 2014 since his rectal operation seven years prior to exami- The effectiveness of the hemorrhoidopexy asade- nation. In addition, in this case of last century, the finitive cure in patients with fourth-degree hemor- anal outlet was well healed, with minimal scarring, rhoids remains controversial. Long-term results in- and was adequate in size and insignificant apart of dicate that persistence of pain occurs in stapled the nodule and had some some rebuilding of anal hemorrhoidopexy on a scale ranging from 1.6 to 31 varices. percent, while it is very rare after the Milligan- Morgan procedure [15,16]. The persistence of se- Clinically, traumatic neuromas may also arise as a vere pain after rectal operation is intriguing. The result of surgery and are often mistaken for scar tis- importance of avoiding the inclusion of smooth sue or adenomas; they are rare events in the gastro- muscle fibers in the fixation ofthe vascular pedicles intestinal tract. In the oral cavity, the most com- in a correct Milligan-Morgan procedure to reduce monly affected sites are the lip, tongue, and chin pain is well known and this theory has been sug- region. Pressure on the suspected area usually pro- gested for the pain arising after the hemorrhoido- vokes pain [10]. In our case, the patient’s presenta- pexy procedure. However, we cannot exclude that tion is particular because of pruritus and general some of the causes of some persisting discomfort paresthesias in the perianal region, which dramati- after hemorrhoidopexy could also be due to minute cally ceased after excision of the lesion. Traumatic anal neuromas. Traumatic neuromas following anal neuroma of the cystic duct in the absence of previ- surgery should be taken in the differential diagnosis ous surgery has been found in a 60-year-old man with hyperplastic, adenomatous, mesenchymal, or admitted to investigate obstructive jaundice [11]. serrated and tumor-like lesions. In this patient, traumatic neuroma was observed 17 years after a cholecystectomy and common bile Acknowledgements duct exploration. Another case of traumatic neuro- We are very grateful to Dr. Friedrich Wieser, Internal Medicine, ma of the cystic duct was found in a markedly fi- Andrea Krouz Gasse 5, 9900, Lienz, Austria for referral of the tissue specimen for pathological examination and external brotic gallbladder of an 88-year-old man [12]. consultation. However, this patient did not undergo surgery and the authors postulated that the stimulus for the References neural and fibrous proliferation was leakage of bile 1. Enzinger FM, Weiss SW. 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