1113
RAGBEER AND STONE
23.
24. 25. 26. 27.
ing hypopharyngeal carcinoma. J Laryngol Otol 99: 1261, 1985 Benninger MS, Lavertu P, Linden MD, et al: Multiple parotid gland primary neoplasms after radiation therapy. Otolaryngol Head Neck Surg 98:250. 1988 Blanck C, Eneroth CM, Jakobsson PA: Bilateral tumours of the parotid gland. Opuscula Medica 19:30, 1974 lbi A, Yokobayashi T, Kawasaki T, et al: Bilateral Warthin’s tumor: Report of case and review of Japanese literature. J Oral Surg 39:362, 1981 Shugar JMA, Som PM, Biller HF: Warthin’s tumor, a multiple disease. Ann Otol Rhino1 Laryngol 9:246. 1982 Tveteris K, Kristensen S: Warthin’s tumour with bilateral
J Oral Maxlllofac
28. 29.
30.
3 1.
synchronous presentation. Survey of the literature and a new case. J Laryngol Otol 100:487, 1986 Baugh RF, McClatchey KD: Papillary cystadenoma lymphomatosum syndrome. J Otolaryngol 15: 166, 1986 Stokes JD, Park YW: Pathologic quiz case 1: Papillary cystadenoma lymphomatosum (Warthin’s tumor), bilateral. Arch Otolaryngol Head Neck Surg 113:1000. 1987 Bundaaard N. Eriksen HE. Greisen 0: Inflammed adenol;mphoma with cholest&ol granuloma. J Laryngol Otol 101:967, 1987 Dietert SE: Papillary cystadenoma lymphomatosum (Warthin’s tumor) in patients in a general hospital over a 24year period. Am J Clin Path01 63:866, 1975
Surg
48.1113-1117.1990
Vascular Leiomyoma of the Nasal Cavity: Report of a Case and Review of Literature MOHAN S. RAGBEER, MB, BS, DCP (LoND), FRCPATH, FRCS(C),* AND JAMES STONE, DDS, MSC, MD, DIP OMFSt
Leiomyomas are benign myogenic tumors found mainly in the uterus, skin (pilar muscle and vessels), the alimentary tract, and, rarely, in deep soft tissues, peritoneum, and other sites. In one study of 7,748 cases by Enzinger and Weiss,’ 95% of the tumors were from the female genitalia, 3% from skin, and the remainder from various sites, about half of them from the gastrointestinal tract. They are quite unusual in the nasal cavity, and a search of the English literature revealed only 13 prior reports of 19 patients.2-8 We report the clinical and histological features of a case, including its immunohistochemistry and ultrastructure. Report of a Case A 49-year-old with
an
white woman of Middle Eastern 18-month history of pain, suppuration,
origin and
bloody discharge from the right anterior nasal floor was seen. The symptoms and signs were intermittent, with no clear temporal or causal associations. Both nares were clear. The patient’s health was otherwise normal. Clinical examination demonstrated a tenderness in the right anterior nasal floor approximately 1 cm from the Received from McMaster University, Hamilton, Ontario, Canada. *Associate Professor, Department of Pathology. tPrivate Practice and Consultant in Chronic Pain. Address correspondence and reprint requests to Dr Ragbeer: Department of Pathology, McMaster University. Hamilton, Ontario, Canada L8N 325. 0 1990 American geons 0278-2193/90/481
Association O-001 9$3.00/O
of Oral and Maxillofacial
Sur-
anterior nasal border. with crusting over the discharge site. Intraorally, the tissues were normal; no clinical evidence of dental disease was present. Radiographic examination was within normal limits. The lesion was excised through an incision in the anterior maxillary mucobuccal fold. A 1.5 x 1.1 X l-cm smooth, oval mass located entirely within the soft tissues of the nasal floor was immediately evident. The lesion was easily dissected from its bed. The postoperative course was uneventful. The patient remains well, without symptoms or recurrence 12 months postoperatively. HISTOLOGICAL
FINDINGS
The fixed specimen was an intact, ovoid, smooth, pale grey, solid rubbery mass measuring 1.3 x 1 x 0.8 cm. The cut surface showed homogeneous, pale tissue with no hemorrhage or necrosis. Sections were processed for light and electron microscopy and histochemistry using standard methods. Slices were investigated by peroxidase-antiperoxidase methods (PAP), using commercial kits, for keratin, vimentin, alpha-l antichymotrypsin (AACT), factor VIII, desmin, myoglobin, SlOO (Dako Corp, Santa Barbara, CA), and smooth muscle-specific actin-HHF35 (Enzo Diagnostics Inc, New York, NY). All methods followed manufacturers’ instructions, which conformed to the principles and practices detailed by Stemberger.’ Histologically, the lesion was thinly encapsulated and made up of sinuous and arborizing groups of plump spindle or round cells with ovoid blunt-ended nuclei and abundant clear or pale pink cytoplasm, often with vacuoles, in a collagenous matrix. Mitoses were not seen. Spindle cells were prominent peripherally, and clear cells, many with perinuclear halos, dominated the center of the lesion. Most of the groups enclosed narrow, slitlike vascular lumina with strands of elastic fibers and circumferentially arranged smooth muscle cells in their
1114
VASCULAR
LEIOMYOMA
OF THE NASAL CAVLTY
FIGURE 1. A thin capsule (thick arrow, left) encloses sinuous strands of spindle and clear cells around thin-walled vessels. Note clear cells in vessel walls merging with peripheral ones (thin arrows) (PAS after diastase digestion, original magnification x 1851. Inset shows cells at higher magnification. They are plump with abundant cytoplasm and ovoid nuclei in a fibrillar matrix (Masson’s trichrome. original magnification x460).
walls suggesting veins. The outer layers merged with the surrounding cells. Between the bundles were strands of collagen and a few scattered adipocytes (Figs 1 and 2). The periodic acid-Schiff (PAS) reaction before and after diastase digestion showed abundant positive granules, especially in the clear cells (Fig 3), confirmed as glycogen in smooth muscle cells by electron microscopy (Fig 4 inset). Immunohistologically, the cells showed strong cytoplasmic positivity for HHF35 antigen (smooth musclespecific) and desmin (Fig 4), faint to moderate staining for vimentin, and focal positivity for myoglobin. The other reactions in the tumor cells were negative. The linings of the slits and groups of flattened cells in the solid regions. however, stained brightly for factor VIII antigen, confirming their endothelial nature (Fig 2 inset).
Discussion The first report of an intranasal leiomyoma, that of Maesaka et al concerning an angioleiomyoma,’ appeared in 1966. Since then, 19 cases have been reported, the present included, indicating the extreme rarity of this condition even with underreporting taken into account. This rarity is partly attributable to the fact that smooth muscle is sparsely present in the nasal cavity apart from the walls of blood vessels. It is possible that the lesion may represent a hamartoma rather than a true neoplasm, taking into account the occasional occur-
FIGURE 2. A field from the center of the lesion showing arborizing bundles of spindle cells, thick-walled vessels, and adipocytes (hematoxylineosin, original magnification x 150). Znser shows endothelial slits in solid areas, confirming their vascular nature (PAP method for factor VIII antigen, original magnification x460).
RAGBEER AND STONE
FIGURE 3. Center of lesion shows clear cells, many with central nuclei (PAS after diastase digestion, original magnification x750). Inset shows diffuse dark cytoplasmic staining and discrete granules due to glycogen (PAS, original magnification x750).
FIGURE 4. A, Most of the cells contain dark granules that represent sites of smooth muscle-specific actin (SMSA) (PAP method for HHF35 antigen, original magnification x 185). Insets show higher power views of positive cells. B, Positive intracytoplasmic granules of SMSA (HHF35, original magnification x750). C, Positive intracytoplasmic and perinuclear desmin granules (PAP method for desmin, original magnification x460). D, Electron micrograph of part of a clear cell showing typical smooth muscle features: electron-dense external lamina (basement membranelike material) (arrowhead): caveolae at cytoplasmic border (arrow); and microfilaments (x). Glycogen granules (*) are unusually plentiful in this cell.
1116
VASCULAR
rence, as in this case, of other mesenchymal elements, such as adipose tissue and collagen. Table 1 presents a summary of 14 cases, including the 10 tabulated by Hanna et al,* the original reports of which also were reviewed. Another five were found,* but were excluded from the Table for lack of data. Ten tumors were found in women. The main symptoms were epistaxis, pain, and obstruction, seven cases exhibiting the first two symptoms and five the third, either alone or in combination with the others. Two patients were asymptomatic and one showed excessive lacrimation in addition to other symptoms4 The age range was 5 to 76 years, with 13 of the 14 cases in adults aged 30 and over, and with a mean of 54. The single case in a 5year-old girl was labeled “leiomyoblastoma” because of its component of clear cells reminiscent of some gastric leiomyomas subgrouped as lieomyoblastoma.3 This term may actually be a misnomer and should be abandoned because no significant clinical behavioral difference seems to exist between it and the clear cell leiomyoma. However, the presence of abundant glycogen in that case and Table 1. Report No.
OF THE NASAL CAVITY
in ours, a feature not referred to in clear cell leiomyomas, may indicate a functional or metabolic difference between the cells of this variant and the ordinary smooth muscle cell. All the tumors were classified as benign, none showing mitoses or the presence of atypia to suggest possible malignant behavior. Complete surgical excision is the treatment of choice and was used in all the cases reported. Recurrence following complete excision is not known to have taken place in any of these cases; recurrence in the case of “leiomyoblastoma” is thought to have been due to incomplete initial excision. In the series of Hachisuga et al of 562 cases of vascular leiomyomas from all sites, only two recurrences were noteds; the sites were not given. Leiomyomas of the skin and subcutis are usually subdivided into vascular or nonvascular types,” the latter presumably including all those cases arising in association with the arrector pili muscle or in the dartos, vulva, or nipple. These usually are solid lesions made up of uniform smooth muscle cells in bundles. In the nasal cavity, hair and associated
Intranasal Leiomyoma Authors
Sex/Age (yr)
Clinical Problem
Location of Lesion
Histology
Maesaka et al (1966)’ Ram (1971)* Wolfwitz and Schmamann (1973)* Schwartzman and Schwartzmann (1973)*
F/49 M/40 F/42
Facial pain Nasal obstruction Epistaxis
Vestibule Inferior turbinate Inferior turbinate
Vascular Fibromyoma Vascular
Ml57
Nasal obstruction and headache
Vascular
Fu and Perzin (1975)
F/60* M/46* F/l6
None None Epistaxis
Maxillary, ethmoidal, and sphenoidal sinuses, and posterior nasal fossa Nasal polyp Nasal polyp Inferior turbinate
F/5
Epistaxis and nasal obstruction Facial pain
Middle turbinate
Headache, right nasal swelling and obstruction, lacrimation Epistaxis
Middle trubinate
3.5 x 2.5 x 1 cm Erectile tissue Vascular
Posterior end of left inferior turbinate Inferior turbinate
Nonvascular 4X3x3cm Vascular
Nasal vestibule
Not stated
Right anterior nasal floor
Vascular
McCaffrey et al (1978)* Papavasiliou and Michaels (1981)3 Lijovetsky et al (1985)* Daisley ( 1987)4
F/73 F/32
M/56
10
Tang and Tse (1988)5
11
Hanna et al (1988)6
F/64
f2
Nam, Kaufman, and Wolff (1989)’ Present study*
F/59
13
LEIOMYOMA
F/49
Nasal obstruction, epistaxis, and facial pain Recurrent epistaxis and intermittent pain Pain, bloody intermittent discharge, and suppuration
Table excludes five cases mentioned in series by Hachisuga et al8 * Report reviewed. See Hanna et al6
Vestibule
Nonvascular Nonvascular Vascular 0.4 cm Leiomyoblastoma 1.5 X 1 X lcm Not stated
1117
RAGBEER AND STONE
tissue are present only in the anterior part of the vestibule. Elsewhere in the nasal cavity, smooth muscle is found in vein walls as circular or longitudinal strands. It seems more likely, therefore, that any leiomyoma that develops in the nasal cavity should be of vascular origin, though a hamartomatous basis cannot be excluded. Seven of the reported cases were classified as vascular, live were nonvascular (including the case of “leiomyoblastoma”), and no classification was given for the remaining two (reports 8 and 12). In the series of Hachisuga et al8 375 of 562 cases were found in the lower limb, 125 in the upper limb, 48 in the head (with 5 in the nasal cavity), and 14 in the trunk. They comprised 5% of all benign softtissue tumors and 25% to 50% of all superficial leiomyomas. Overall, they were more frequent in women than in men, with a ratio of 1.7: 1. In the head, however, the male:female ratio was reversed. Hachisuga et al did not provide details of their 5 nasal cavity cases but excepting them, the cumulative ratio of the 14 remaining cases shows a 2.5:1 female preponderance, which may not be signilicant in view of the small number of cases, but prima facie is at variance with the overall data of Hachisuga et al on “head” cases. Whether the differential incidence is associated in any way with the presence of estrogen and/or progesterone receptors on smooth muscle cells is as yet speculative, although some experimental evidence exists to show a relationship between the two.” Also, lesions of disseminated peritoneal leiomyomatosis are known to regress spontaneously after conclusion of pregnancy.’ It is possible that nonvascular nasal lesions could represent a late evolutionary stage in the progression of lesions from pre-
dominantly vascular, neoplastic, or hamartomatous, to solid.12 The demonstration of factor VIII positive cells and elastic tissue in an arrangement suggesting residual vessels in solid lesions may support this view. Acknowledgments We thank Dr B. Jackson for referral of the case and Dr G. Simon for the electron micrograph.
References 1. Enzinger FM, Weiss SW: Soft Tissue Tumours (2nd ed). St Louis, Mosby, 1988, pp 383-401 2. Maesaka A, Keyaki Y, Nakahashi T: Nasal angioleiomyoma and leiomyosarcoma-Report of 2 cases. Otologia (Fukuoka) 12:42, 1966 3. Papavasiliou A, Michaels L: Unusual leiomyoma of the nose (leiomyoblastoma): Report of a case. J Laryngol Otol 951281, 1981 4. Daisley H: Leiomyoma of the nasal cavity. West Indian Med J 36:181, 1987 5. Tang SO, Tse CH: Leiomyoma of the nasal cavity. J Laryngo1 Otol 102:831, 1988 6. Hanna GS, Akosa AB, Ali MH: Vascular leiomyoma of the inferior turbinate-Report of a case and review of the literature. J Larvnaol Otol 102:1159. 1988 7. Nam HK, Kaufman MW, Woolf AP: Pathology quiz, case 2. Arch Otolaryngol Head Neck Surg 115:244, 1989 8. Hachisuga T, Hashimoto H, Enjoji M: Angioleiomyoma: A clinicopathological reappraisal of 562 cases. Cancer 54: 126, 1984 9. Stemberger LA: Immunocytochemistry. Englewood Cliffs, NJ, Prentice-Hall, 1974 10. Stout AP, Lattes R: Tumours of the soft tissues (revised). Washington, DC, Armed Forces Institute of Pathology, 1981, pp 60-65 11. Fujii S, Makashima N, Okamura H, et al: Progesteroneinduced smooth muscle like cells in subperitoneal nodules produced by estrogen: Experimental approach to leiomyomatosis peritonealis disseminate. Am J Obstet Gynecol 139:164, 1981 12. Duhig JT, Ayer JP: Vascular leiomyoma-A study of 61 cases. Arch Pathol 68:424, 1959
RAGBEER AND STONE
23.
24. 25. 26. 27.
ing hypopharyngeal carcinoma. J Laryngol Otol 99: 1261, 1985 Benninger MS, Lavertu P, Linden MD, et al: Multiple parotid gland primary neoplasms after radiation therapy. Otolaryngol Head Neck Surg 98:250. 1988 Blanck C, Eneroth CM, Jakobsson PA: Bilateral tumours of the parotid gland. Opuscula Medica 19:30, 1974 lbi A, Yokobayashi T, Kawasaki T, et al: Bilateral Warthin’s tumor: Report of case and review of Japanese literature. J Oral Surg 39:362, 1981 Shugar JMA, Som PM, Biller HF: Warthin’s tumor, a multiple disease. Ann Otol Rhino1 Laryngol 9:246. 1982 Tveteris K, Kristensen S: Warthin’s tumour with bilateral
J Oral Maxlllofac
28. 29.
30.
3 1.
synchronous presentation. Survey of the literature and a new case. J Laryngol Otol 100:487, 1986 Baugh RF, McClatchey KD: Papillary cystadenoma lymphomatosum syndrome. J Otolaryngol 15: 166, 1986 Stokes JD, Park YW: Pathologic quiz case 1: Papillary cystadenoma lymphomatosum (Warthin’s tumor), bilateral. Arch Otolaryngol Head Neck Surg 113:1000. 1987 Bundaaard N. Eriksen HE. Greisen 0: Inflammed adenol;mphoma with cholest&ol granuloma. J Laryngol Otol 101:967, 1987 Dietert SE: Papillary cystadenoma lymphomatosum (Warthin’s tumor) in patients in a general hospital over a 24year period. Am J Clin Path01 63:866, 1975
Surg
48.1113-1117.1990
Vascular Leiomyoma of the Nasal Cavity: Report of a Case and Review of Literature MOHAN S. RAGBEER, MB, BS, DCP (LoND), FRCPATH, FRCS(C),* AND JAMES STONE, DDS, MSC, MD, DIP OMFSt
Leiomyomas are benign myogenic tumors found mainly in the uterus, skin (pilar muscle and vessels), the alimentary tract, and, rarely, in deep soft tissues, peritoneum, and other sites. In one study of 7,748 cases by Enzinger and Weiss,’ 95% of the tumors were from the female genitalia, 3% from skin, and the remainder from various sites, about half of them from the gastrointestinal tract. They are quite unusual in the nasal cavity, and a search of the English literature revealed only 13 prior reports of 19 patients.2-8 We report the clinical and histological features of a case, including its immunohistochemistry and ultrastructure. Report of a Case A 49-year-old with
an
white woman of Middle Eastern 18-month history of pain, suppuration,
origin and
bloody discharge from the right anterior nasal floor was seen. The symptoms and signs were intermittent, with no clear temporal or causal associations. Both nares were clear. The patient’s health was otherwise normal. Clinical examination demonstrated a tenderness in the right anterior nasal floor approximately 1 cm from the Received from McMaster University, Hamilton, Ontario, Canada. *Associate Professor, Department of Pathology. tPrivate Practice and Consultant in Chronic Pain. Address correspondence and reprint requests to Dr Ragbeer: Department of Pathology, McMaster University. Hamilton, Ontario, Canada L8N 325. 0 1990 American geons 0278-2193/90/481
Association O-001 9$3.00/O
of Oral and Maxillofacial
Sur-
anterior nasal border. with crusting over the discharge site. Intraorally, the tissues were normal; no clinical evidence of dental disease was present. Radiographic examination was within normal limits. The lesion was excised through an incision in the anterior maxillary mucobuccal fold. A 1.5 x 1.1 X l-cm smooth, oval mass located entirely within the soft tissues of the nasal floor was immediately evident. The lesion was easily dissected from its bed. The postoperative course was uneventful. The patient remains well, without symptoms or recurrence 12 months postoperatively. HISTOLOGICAL
FINDINGS
The fixed specimen was an intact, ovoid, smooth, pale grey, solid rubbery mass measuring 1.3 x 1 x 0.8 cm. The cut surface showed homogeneous, pale tissue with no hemorrhage or necrosis. Sections were processed for light and electron microscopy and histochemistry using standard methods. Slices were investigated by peroxidase-antiperoxidase methods (PAP), using commercial kits, for keratin, vimentin, alpha-l antichymotrypsin (AACT), factor VIII, desmin, myoglobin, SlOO (Dako Corp, Santa Barbara, CA), and smooth muscle-specific actin-HHF35 (Enzo Diagnostics Inc, New York, NY). All methods followed manufacturers’ instructions, which conformed to the principles and practices detailed by Stemberger.’ Histologically, the lesion was thinly encapsulated and made up of sinuous and arborizing groups of plump spindle or round cells with ovoid blunt-ended nuclei and abundant clear or pale pink cytoplasm, often with vacuoles, in a collagenous matrix. Mitoses were not seen. Spindle cells were prominent peripherally, and clear cells, many with perinuclear halos, dominated the center of the lesion. Most of the groups enclosed narrow, slitlike vascular lumina with strands of elastic fibers and circumferentially arranged smooth muscle cells in their
1114
VASCULAR
LEIOMYOMA
OF THE NASAL CAVLTY
FIGURE 1. A thin capsule (thick arrow, left) encloses sinuous strands of spindle and clear cells around thin-walled vessels. Note clear cells in vessel walls merging with peripheral ones (thin arrows) (PAS after diastase digestion, original magnification x 1851. Inset shows cells at higher magnification. They are plump with abundant cytoplasm and ovoid nuclei in a fibrillar matrix (Masson’s trichrome. original magnification x460).
walls suggesting veins. The outer layers merged with the surrounding cells. Between the bundles were strands of collagen and a few scattered adipocytes (Figs 1 and 2). The periodic acid-Schiff (PAS) reaction before and after diastase digestion showed abundant positive granules, especially in the clear cells (Fig 3), confirmed as glycogen in smooth muscle cells by electron microscopy (Fig 4 inset). Immunohistologically, the cells showed strong cytoplasmic positivity for HHF35 antigen (smooth musclespecific) and desmin (Fig 4), faint to moderate staining for vimentin, and focal positivity for myoglobin. The other reactions in the tumor cells were negative. The linings of the slits and groups of flattened cells in the solid regions. however, stained brightly for factor VIII antigen, confirming their endothelial nature (Fig 2 inset).
Discussion The first report of an intranasal leiomyoma, that of Maesaka et al concerning an angioleiomyoma,’ appeared in 1966. Since then, 19 cases have been reported, the present included, indicating the extreme rarity of this condition even with underreporting taken into account. This rarity is partly attributable to the fact that smooth muscle is sparsely present in the nasal cavity apart from the walls of blood vessels. It is possible that the lesion may represent a hamartoma rather than a true neoplasm, taking into account the occasional occur-
FIGURE 2. A field from the center of the lesion showing arborizing bundles of spindle cells, thick-walled vessels, and adipocytes (hematoxylineosin, original magnification x 150). Znser shows endothelial slits in solid areas, confirming their vascular nature (PAP method for factor VIII antigen, original magnification x460).
RAGBEER AND STONE
FIGURE 3. Center of lesion shows clear cells, many with central nuclei (PAS after diastase digestion, original magnification x750). Inset shows diffuse dark cytoplasmic staining and discrete granules due to glycogen (PAS, original magnification x750).
FIGURE 4. A, Most of the cells contain dark granules that represent sites of smooth muscle-specific actin (SMSA) (PAP method for HHF35 antigen, original magnification x 185). Insets show higher power views of positive cells. B, Positive intracytoplasmic granules of SMSA (HHF35, original magnification x750). C, Positive intracytoplasmic and perinuclear desmin granules (PAP method for desmin, original magnification x460). D, Electron micrograph of part of a clear cell showing typical smooth muscle features: electron-dense external lamina (basement membranelike material) (arrowhead): caveolae at cytoplasmic border (arrow); and microfilaments (x). Glycogen granules (*) are unusually plentiful in this cell.
1116
VASCULAR
rence, as in this case, of other mesenchymal elements, such as adipose tissue and collagen. Table 1 presents a summary of 14 cases, including the 10 tabulated by Hanna et al,* the original reports of which also were reviewed. Another five were found,* but were excluded from the Table for lack of data. Ten tumors were found in women. The main symptoms were epistaxis, pain, and obstruction, seven cases exhibiting the first two symptoms and five the third, either alone or in combination with the others. Two patients were asymptomatic and one showed excessive lacrimation in addition to other symptoms4 The age range was 5 to 76 years, with 13 of the 14 cases in adults aged 30 and over, and with a mean of 54. The single case in a 5year-old girl was labeled “leiomyoblastoma” because of its component of clear cells reminiscent of some gastric leiomyomas subgrouped as lieomyoblastoma.3 This term may actually be a misnomer and should be abandoned because no significant clinical behavioral difference seems to exist between it and the clear cell leiomyoma. However, the presence of abundant glycogen in that case and Table 1. Report No.
OF THE NASAL CAVITY
in ours, a feature not referred to in clear cell leiomyomas, may indicate a functional or metabolic difference between the cells of this variant and the ordinary smooth muscle cell. All the tumors were classified as benign, none showing mitoses or the presence of atypia to suggest possible malignant behavior. Complete surgical excision is the treatment of choice and was used in all the cases reported. Recurrence following complete excision is not known to have taken place in any of these cases; recurrence in the case of “leiomyoblastoma” is thought to have been due to incomplete initial excision. In the series of Hachisuga et al of 562 cases of vascular leiomyomas from all sites, only two recurrences were noteds; the sites were not given. Leiomyomas of the skin and subcutis are usually subdivided into vascular or nonvascular types,” the latter presumably including all those cases arising in association with the arrector pili muscle or in the dartos, vulva, or nipple. These usually are solid lesions made up of uniform smooth muscle cells in bundles. In the nasal cavity, hair and associated
Intranasal Leiomyoma Authors
Sex/Age (yr)
Clinical Problem
Location of Lesion
Histology
Maesaka et al (1966)’ Ram (1971)* Wolfwitz and Schmamann (1973)* Schwartzman and Schwartzmann (1973)*
F/49 M/40 F/42
Facial pain Nasal obstruction Epistaxis
Vestibule Inferior turbinate Inferior turbinate
Vascular Fibromyoma Vascular
Ml57
Nasal obstruction and headache
Vascular
Fu and Perzin (1975)
F/60* M/46* F/l6
None None Epistaxis
Maxillary, ethmoidal, and sphenoidal sinuses, and posterior nasal fossa Nasal polyp Nasal polyp Inferior turbinate
F/5
Epistaxis and nasal obstruction Facial pain
Middle turbinate
Headache, right nasal swelling and obstruction, lacrimation Epistaxis
Middle trubinate
3.5 x 2.5 x 1 cm Erectile tissue Vascular
Posterior end of left inferior turbinate Inferior turbinate
Nonvascular 4X3x3cm Vascular
Nasal vestibule
Not stated
Right anterior nasal floor
Vascular
McCaffrey et al (1978)* Papavasiliou and Michaels (1981)3 Lijovetsky et al (1985)* Daisley ( 1987)4
F/73 F/32
M/56
10
Tang and Tse (1988)5
11
Hanna et al (1988)6
F/64
f2
Nam, Kaufman, and Wolff (1989)’ Present study*
F/59
13
LEIOMYOMA
F/49
Nasal obstruction, epistaxis, and facial pain Recurrent epistaxis and intermittent pain Pain, bloody intermittent discharge, and suppuration
Table excludes five cases mentioned in series by Hachisuga et al8 * Report reviewed. See Hanna et al6
Vestibule
Nonvascular Nonvascular Vascular 0.4 cm Leiomyoblastoma 1.5 X 1 X lcm Not stated
1117
RAGBEER AND STONE
tissue are present only in the anterior part of the vestibule. Elsewhere in the nasal cavity, smooth muscle is found in vein walls as circular or longitudinal strands. It seems more likely, therefore, that any leiomyoma that develops in the nasal cavity should be of vascular origin, though a hamartomatous basis cannot be excluded. Seven of the reported cases were classified as vascular, live were nonvascular (including the case of “leiomyoblastoma”), and no classification was given for the remaining two (reports 8 and 12). In the series of Hachisuga et al8 375 of 562 cases were found in the lower limb, 125 in the upper limb, 48 in the head (with 5 in the nasal cavity), and 14 in the trunk. They comprised 5% of all benign softtissue tumors and 25% to 50% of all superficial leiomyomas. Overall, they were more frequent in women than in men, with a ratio of 1.7: 1. In the head, however, the male:female ratio was reversed. Hachisuga et al did not provide details of their 5 nasal cavity cases but excepting them, the cumulative ratio of the 14 remaining cases shows a 2.5:1 female preponderance, which may not be signilicant in view of the small number of cases, but prima facie is at variance with the overall data of Hachisuga et al on “head” cases. Whether the differential incidence is associated in any way with the presence of estrogen and/or progesterone receptors on smooth muscle cells is as yet speculative, although some experimental evidence exists to show a relationship between the two.” Also, lesions of disseminated peritoneal leiomyomatosis are known to regress spontaneously after conclusion of pregnancy.’ It is possible that nonvascular nasal lesions could represent a late evolutionary stage in the progression of lesions from pre-
dominantly vascular, neoplastic, or hamartomatous, to solid.12 The demonstration of factor VIII positive cells and elastic tissue in an arrangement suggesting residual vessels in solid lesions may support this view. Acknowledgments We thank Dr B. Jackson for referral of the case and Dr G. Simon for the electron micrograph.
References 1. Enzinger FM, Weiss SW: Soft Tissue Tumours (2nd ed). St Louis, Mosby, 1988, pp 383-401 2. Maesaka A, Keyaki Y, Nakahashi T: Nasal angioleiomyoma and leiomyosarcoma-Report of 2 cases. Otologia (Fukuoka) 12:42, 1966 3. Papavasiliou A, Michaels L: Unusual leiomyoma of the nose (leiomyoblastoma): Report of a case. J Laryngol Otol 951281, 1981 4. Daisley H: Leiomyoma of the nasal cavity. West Indian Med J 36:181, 1987 5. Tang SO, Tse CH: Leiomyoma of the nasal cavity. J Laryngo1 Otol 102:831, 1988 6. Hanna GS, Akosa AB, Ali MH: Vascular leiomyoma of the inferior turbinate-Report of a case and review of the literature. J Larvnaol Otol 102:1159. 1988 7. Nam HK, Kaufman MW, Woolf AP: Pathology quiz, case 2. Arch Otolaryngol Head Neck Surg 115:244, 1989 8. Hachisuga T, Hashimoto H, Enjoji M: Angioleiomyoma: A clinicopathological reappraisal of 562 cases. Cancer 54: 126, 1984 9. Stemberger LA: Immunocytochemistry. Englewood Cliffs, NJ, Prentice-Hall, 1974 10. Stout AP, Lattes R: Tumours of the soft tissues (revised). Washington, DC, Armed Forces Institute of Pathology, 1981, pp 60-65 11. Fujii S, Makashima N, Okamura H, et al: Progesteroneinduced smooth muscle like cells in subperitoneal nodules produced by estrogen: Experimental approach to leiomyomatosis peritonealis disseminate. Am J Obstet Gynecol 139:164, 1981 12. Duhig JT, Ayer JP: Vascular leiomyoma-A study of 61 cases. Arch Pathol 68:424, 1959
