Leiomyomas of the Rectum and Anal Canal: Report
o f Six Cases a n d
R e v i e w of the L i t e r a t u r e *
ROBERTO .E. KUSMINSKY, M.D., WALLACE BAILEY, M.D. Dallas, Texas
A RECENTPATIENTwho had leiomyoma of the anal canal and the unusual characteristics often shown by these tumors stimulated our interest in these infrequently seen lesions. T h e records of Baylor University Medical Center were reviewed for a period starting January 1936, and ending December 1975. I n those 40 years, six cases of leiomyoma of the rectum were.encountered. T o complete this research, an extensive review of the literature was done. This task was greatly simplified by our consultation of the monogram published in 1962 by Skandalakis and Gray.SS This excellent world literature review of smooth-muscle tumors of the alimentary canal was thorough, and collected 2,525 cases through 1959. Of these, 144 were rectal tumors, 89 being leiomyomas and the rest leiomyosarcomas. I n turn, we have tried to record in careful detail the cases reported from 1959 until 1975. I n 1975, however, we are aware that some publications have been omitted. This has been mainly due to our inability to translate certain articles, and; probably we have overlooked a few. We have been able to account for 79 leiomyomas of the rectum reported in the world literature from 1959 to the end of 1975, including the six cases here presented. This amounts to a total of 168 since the first one reported in 1872. Table 1 shows an abbreviated review of the * Received for publication November 29, 1976. Address reprint requests to Dr. Kusminsky: 6064 Village Glen Drive, Apt. 4153, Dallas, Texas 75206.
literature to 1959. Here we have included cases that had unusual or interesting features, those that recurred a n d / o r metastasized, those in which the lesions were located in the anal canal, and those for which we have follow-up data. For a more detailed description, the reader is referred to the work of Skandalakis and Gray. s8 T o emphasize the problems of 9 and metastasis associated with these tumors, two cases 70, 90 included in Table 1 have been described with slight changes in chronology as compared with the original reports. T a b l e 2 lists all cases reported from 1959 to 1975, summarizing symptoms, findings, treatment and results. Historic Review T h e term "myoma" was first employed by Virchow in 1854, in reference to the gravid uterus (myoma telangiectodes). In 1864, F. A. yon Zemker proposed the terms "leiomyoma" for smooth-muscle tumors and "rhabdomyoma" for tumors of striated muscle. An admirable review of the history of these tumors was written by Grigg, 43 who included very interesting quotations of what may have been cases of smooth-muscle tumors of the rectum as early as 1728 (Boerheave), 1768 (Bloch), and 1843 (Lamballe). Records of the resection of an apparent smooth-muscle tumor of the rectal ~submucosa by Cambrai in 1847 also exist. 88 T h e first histologically confirmed leiomyoma of the rectum was reported by Malassez in 1872, 63 although most writers assign the 580
Dis. Col. & Rect. October 1977
Volume 20 Number 7
Volume 20 Number 7
ANORECTAL
credit for such a report to Carlier and Van der Espt (1881), 2~ probably because it was a well-described case. Since then several reviews have been written: H u n t 4a collected 20 cases in 1921, and in 1933 H a r t m a n n et al. 45 gathered 33 cases from the literature. An almost classic article was written by Golden and Stout 40 in 1941. Since then, increasing numbers of reports on this subject have been published. Incidence A few reports mention the incidence of smooth-muscle tumors of the rectum including cases found at autopsy31; however, the n u m b e r of leiomyomas of the rectum producing clinical manifestations is so limited that very few surgeons gain wide experience treating these lesions. Therefore, including tumors found at autopsy is probably not realistic, even though it might be more accurate. In fact, O b e r h e l m a n et al. 72 noted that of 115 cases of gastrointestinal leiomyomas collected from the literature, approximately 30 per cent were incidental findings at autopsy or operation. Golden and Stout 4~ reported the incidence of smooth-muscle tumors of the rectum to be 7 per cent, with the bulk of these lesions occurring in the stomach (61 per cent). T h e y arrived at these figures after analyzing 1,018 cases of smooth-muscle tumors reported in the literature to 1940, with 69 of these tumors located in the rectum. However, by 1959, with increasing numbers of reports and better means of diagnosis, the incidence cited by Skandalakis and Gray from their analysis of 2,525 cases was slightly different: 5.7 per cent for smooth-muscle tumors of the rectum and 45 per cent for gastric lesions. Anderson et al.3 also mentioned a 7 per cent incidence of rectal smooth-muscle tumors, adding that one leiomyoma is seen for every 2,000 to 3,000 rectal tumors. These figures coincide with those of Somervel and Myer, 9~ who, after a computer search of
.~ 1
LEIOMYOMAS
a 17-year period, found an incidence of one leiomyosarcoma for every 1,300 rectal tumors. Finally, it would a p p e a r that the incidences of rectal leiomyomas and leiomyosarcomas are a p p r o x i m a t e l y equal, although slight variations are found in different reports.
Etiology T h e etiologic factors involved in the origin of leiomyomas are, of course, merely speculative. T h e m a i n problem once centered around the fact that smooth-muscle cells were not frequently seen dividing, and in the early part of the century investigators were faced with the existence of tumors (leiomyomas) made up of cells not often seen to multiply. T h i s helped to originate the theory that these tumors could arise from residual embryonic material, hypothetically supported by reported cases of congenital leiomyomas of the gastrointestinal tract. 24, 100. ~06 Bensaude et al3 o found aberrant hair follicles and sweat glands in two patients with rectal leiomyomas examined by them; they felt that the presence of these elements in an unexpected area strongly supported the theory of congenital origin. T h e y postulated that this residual heterotopic material within the wall of the rectum could at some point develop into these tumors. However, the multiplication of smooth-muscle cells has now been demonstrated frequently enough to suggest that the mitotic division of smooth muscle is responsible for most, if not all, smoothmuscle cell tumors in the gastrointestinal tract.as It has been frequently suggested that a relationship exists between the [ H of the contents of the gastrointestinal tract and the appearance of smooth-muscle tumors, pointing out that the areas with the highest acidic content correlate with the segments that have the most frequent occurrence of these tumors (lower esophagus, stomach).
582
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o f Six Cases a n d
R e v i e w of the L i t e r a t u r e *
ROBERTO .E. KUSMINSKY, M.D., WALLACE BAILEY, M.D. Dallas, Texas
A RECENTPATIENTwho had leiomyoma of the anal canal and the unusual characteristics often shown by these tumors stimulated our interest in these infrequently seen lesions. T h e records of Baylor University Medical Center were reviewed for a period starting January 1936, and ending December 1975. I n those 40 years, six cases of leiomyoma of the rectum were.encountered. T o complete this research, an extensive review of the literature was done. This task was greatly simplified by our consultation of the monogram published in 1962 by Skandalakis and Gray.SS This excellent world literature review of smooth-muscle tumors of the alimentary canal was thorough, and collected 2,525 cases through 1959. Of these, 144 were rectal tumors, 89 being leiomyomas and the rest leiomyosarcomas. I n turn, we have tried to record in careful detail the cases reported from 1959 until 1975. I n 1975, however, we are aware that some publications have been omitted. This has been mainly due to our inability to translate certain articles, and; probably we have overlooked a few. We have been able to account for 79 leiomyomas of the rectum reported in the world literature from 1959 to the end of 1975, including the six cases here presented. This amounts to a total of 168 since the first one reported in 1872. Table 1 shows an abbreviated review of the * Received for publication November 29, 1976. Address reprint requests to Dr. Kusminsky: 6064 Village Glen Drive, Apt. 4153, Dallas, Texas 75206.
literature to 1959. Here we have included cases that had unusual or interesting features, those that recurred a n d / o r metastasized, those in which the lesions were located in the anal canal, and those for which we have follow-up data. For a more detailed description, the reader is referred to the work of Skandalakis and Gray. s8 T o emphasize the problems of 9 and metastasis associated with these tumors, two cases 70, 90 included in Table 1 have been described with slight changes in chronology as compared with the original reports. T a b l e 2 lists all cases reported from 1959 to 1975, summarizing symptoms, findings, treatment and results. Historic Review T h e term "myoma" was first employed by Virchow in 1854, in reference to the gravid uterus (myoma telangiectodes). In 1864, F. A. yon Zemker proposed the terms "leiomyoma" for smooth-muscle tumors and "rhabdomyoma" for tumors of striated muscle. An admirable review of the history of these tumors was written by Grigg, 43 who included very interesting quotations of what may have been cases of smooth-muscle tumors of the rectum as early as 1728 (Boerheave), 1768 (Bloch), and 1843 (Lamballe). Records of the resection of an apparent smooth-muscle tumor of the rectal ~submucosa by Cambrai in 1847 also exist. 88 T h e first histologically confirmed leiomyoma of the rectum was reported by Malassez in 1872, 63 although most writers assign the 580
Dis. Col. & Rect. October 1977
Volume 20 Number 7
Volume 20 Number 7
ANORECTAL
credit for such a report to Carlier and Van der Espt (1881), 2~ probably because it was a well-described case. Since then several reviews have been written: H u n t 4a collected 20 cases in 1921, and in 1933 H a r t m a n n et al. 45 gathered 33 cases from the literature. An almost classic article was written by Golden and Stout 40 in 1941. Since then, increasing numbers of reports on this subject have been published. Incidence A few reports mention the incidence of smooth-muscle tumors of the rectum including cases found at autopsy31; however, the n u m b e r of leiomyomas of the rectum producing clinical manifestations is so limited that very few surgeons gain wide experience treating these lesions. Therefore, including tumors found at autopsy is probably not realistic, even though it might be more accurate. In fact, O b e r h e l m a n et al. 72 noted that of 115 cases of gastrointestinal leiomyomas collected from the literature, approximately 30 per cent were incidental findings at autopsy or operation. Golden and Stout 4~ reported the incidence of smooth-muscle tumors of the rectum to be 7 per cent, with the bulk of these lesions occurring in the stomach (61 per cent). T h e y arrived at these figures after analyzing 1,018 cases of smooth-muscle tumors reported in the literature to 1940, with 69 of these tumors located in the rectum. However, by 1959, with increasing numbers of reports and better means of diagnosis, the incidence cited by Skandalakis and Gray from their analysis of 2,525 cases was slightly different: 5.7 per cent for smooth-muscle tumors of the rectum and 45 per cent for gastric lesions. Anderson et al.3 also mentioned a 7 per cent incidence of rectal smooth-muscle tumors, adding that one leiomyoma is seen for every 2,000 to 3,000 rectal tumors. These figures coincide with those of Somervel and Myer, 9~ who, after a computer search of
.~ 1
LEIOMYOMAS
a 17-year period, found an incidence of one leiomyosarcoma for every 1,300 rectal tumors. Finally, it would a p p e a r that the incidences of rectal leiomyomas and leiomyosarcomas are a p p r o x i m a t e l y equal, although slight variations are found in different reports.
Etiology T h e etiologic factors involved in the origin of leiomyomas are, of course, merely speculative. T h e m a i n problem once centered around the fact that smooth-muscle cells were not frequently seen dividing, and in the early part of the century investigators were faced with the existence of tumors (leiomyomas) made up of cells not often seen to multiply. T h i s helped to originate the theory that these tumors could arise from residual embryonic material, hypothetically supported by reported cases of congenital leiomyomas of the gastrointestinal tract. 24, 100. ~06 Bensaude et al3 o found aberrant hair follicles and sweat glands in two patients with rectal leiomyomas examined by them; they felt that the presence of these elements in an unexpected area strongly supported the theory of congenital origin. T h e y postulated that this residual heterotopic material within the wall of the rectum could at some point develop into these tumors. However, the multiplication of smooth-muscle cells has now been demonstrated frequently enough to suggest that the mitotic division of smooth muscle is responsible for most, if not all, smoothmuscle cell tumors in the gastrointestinal tract.as It has been frequently suggested that a relationship exists between the [ H of the contents of the gastrointestinal tract and the appearance of smooth-muscle tumors, pointing out that the areas with the highest acidic content correlate with the segments that have the most frequent occurrence of these tumors (lower esophagus, stomach).
582
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