COLLINS AND JONES
Acknowledgment The authors thank Dr Jean Pringle of Stanmore Institute of Orthopaedics (London) for her expert opinion on this case.
References 1. Tang JSH, Gold RH, Mirra JM, et al: Hemangiopericytoma of bone. Cancer 62:848, 1988 2. Gt~dran R: Hemangiopericytoma in otolaryngology. J Laryngol Otol 93:477, 1993 3. Birzgalis AR, Ramsden RT, Lye RH, et al: Haemangiopericytoma of the temporal bone. J Laryngol Otol 104:998, 1990 4. O'Brien P, Brasfield RD: Hemangiopericytoma. Cancer 18:249, 1965 5. Sage HH., Salman I: Malignant hemangiopericytoma in the area of a previous ameloblastoma of the mandible. Oral Surg Oral Med Oral Pathol 26:275, 1968 6. Rewell RE: Hemangiopericytoma of the gum. J Laryngol Otol 82:261, 1968
7. Shukla GK, Dayal D, Gupta KR: Hemangiopericytoma of maxilla. J Laryngol Otol 86:399, 1972 8. Caldarelli DD, Sperling RL: Hemangiopericytoma of the maxilia. Arch Otolaryngol 102:49, 1976 9. Wold LE, Unni KK, Cooper KL, et al: Hemangiopericytoma of bone. Am J Surg Pathol 6:53, 1982 10. Dahlin DC: Bone Tumors: General Aspects and Data on 8452 Cases (ed 4). Springfield, IL, Charles C Thomas, 1986, pp 394-405 11. Stout AP: Hemangiopericytoma: A study of twenty-five new cases. Cancer 2:1027, 1949 12. Yaghmai I: Angiographic manifestations of soft tissue and osseous hemangiopericytoma. Radiology 126:653, 1978 13. Anthony PP, RamaniP: Endothelial markersin malignant vascular tumours of the liver: Superiority of QB-END/10 over Von Willebrand factor and Ulex europaeus agglutinin 1. J Clin Pathol 44:29, 1991 14. Sellke FW, Laszewski MJ~ Robinson RA, etal: Hemangiopericytoma of the sternum. Arch Pathol Lab Med 115:242, 1991 15. Enzinger FM, Smith BH: Hemangiopericytoma: An analysis of 106 cases. Hum Pathol 7:61, 1976 16. Smullens SN, Scotti D J, Oterholm JL, et al: Preoperative embolization of retroperitoneal hemangiopericytomas as an aid in their removal. Cancer 50:1875, 1982
J Oral MaxillofacSurg 53:707-711, 1995
Multiple Granular Cell Tumors of the Oral Cavity: Report of a Case and Review of the Literature BOBBY M. COLLINS, DDS,* AND ANNE CALE JONES, DDS1Granular cell tumors are uncommon soft tissue lesions that most often arise in the tongue, skin, and subcutaneous tissues. ~ However, these lesions have also been described in viscera and submucosal locations. The granular cell tumor was originally described by Abrikossoff 2 in 1926. Because of the apparent association between granular cells and skeletal muscle, Abrikossoff coined the term "granular cell myoblastoma." Subsequent tumors have been associated with or.thought to arise from histiocytes, fibroblasts, myoFrom the Division of Oral and Maxillofacial Pathology, Department of Oral Diagnostic Sciences, University of Florida College of Dentistry, Gainesville, FL. * Formerly, Resident; currently Fellow, Head and Neck Surgery, UPMC--Montefiore University Hospital, Pittsburgh, PA. ~-Assistant Professor. Address correspondence and reprint requests to Dr Collins: UPMC--Montefiore University Hospital, NW 623 Department of Pathology, 200 Lothrup St, Pittsburgh, PA 15213-2582. © 1995 American Association of Oral and Maxillofacial Surgeons 0278-2391/95/5306-001553.00/0
epithelium, and nerve cell connective tissue. 35 However, the granular cells are now believed to be of primitive Schwann cell origin, and immunohistochemical stains (s-100 protein and neuron-specific enolase) favor neural differentiation. Most granular cell tumors are solitary, but in 7% to 25% of cases multiple lesions o c c u r . 1"6'7 Multiple granular cell tumors are most often located in the oral mucosa, skin, gastrointestinal tract, respiratory tract, and biliary systemJ '6'8-H These lesions occur most often in black women. ~'6 Rifkin et al ~2 state that multiple tumors may be found in up to 20% of black patients who present with an isolated granular cell tumor. Granular cell tumors have been reported in patients ranging in age from 4 months to 89 years. The mean age occurs between the fourth and sixth decades of life. 6'9A1 Most studies demonstrate a female predilectionl'6'8"9; however, the apparent differences in the sex ratio may depend on the tumor site. ~3 The difference varies from slight in some studies to a 5:1 female predilection for granular cell tumors of the larynx.
MULTIPLE GRANULAR CELL TUMORS
FIGURE 1. Multiple granular cell tumors of the tongue. Three lesions are present on the left side and two lesions are present on the right side of the tongue. Most granular cell tumors are benign, but in approximately 10% of cases clinically m a l i g n a n t behavior or m a l i g n a n t tumors have been described. 7'14 This report describes a case of multiple granular cell tumors i n v o l v i n g the oral mucosa, vaginal mucosa, and facial skin, and we review reported cases of multiple granular cell tumors with intraoral i n v o l v e m e n t in the English language literature.
FIGURE 3. Photomicrographof a granular cell tumor demonstrating pseudoepitheliomatoushyperplasia and granular cells (hematoxylin-eosin stain, original magnification x 100).
University of Florida College of Dentistry for evaluation of nodules on the tongue and floor of the mouth. The patient stated that she had experienced pain in the left posterior tongue when eating spicy foods or when brushing her teeth. The patient's medical history was remarkable for hyperten-
On November 14, 1993, a 39-year-old black woman presented to the Oral and Maxillofacial Surgery Clinic at the
FIGURE 2. Two granular cell tumors of the floor of the mouth.
FIGURE 4. Photomicrographof a granular cell tumor demonstrating characteristic granular cells (hematoxylin-eosin stain, original magnification x400).
COLLINS AND JONES
709 dies. In four of the eight specimens, the stratified squamous epithelium was surfaced by a thickened layer of orthokeratin. The spinous cell layer was thickened, and focal areas of pseudoepitheliomatous hyperplasia were apparent (Fig 3). The subjacent connective tissue was composed of loosely and densely arranged collagen fibers interspersed with fibroblasts. The most conspicuous feature of the fibrous stroma was the presence of a diffuse proliferation of relatively large oval-shaped cells with eosinophilic, granular cytoplasm. The nuclei were uniform, basophilic, and centrally located (Fig 4). The granular cells were interspersed between the regional striated muscle bundles. Eight of the lesions were diagnosed as granular cell tumors (5, tongue; 2, floor of the mouth; 1, skin). The remaining lesion showed normal skeletal muscle.
sion, hypothyroidism, gonorrhea, and multiple surgeries. A vulvar biopsy performed in 1993 demonstrated carcinoma in situ. Nodules removed from the tongue in 1985 and 1986 and a vulvar lesion removed in 1987 were diagnosed as granular cell tumors. There was no family history of isolated or multiple granular cell tumors. Clinical examination showed multiple, firm, smooth-surfaced lesions involving the right and left lateral tongue and the floor of the mouth (Figs 1, 2). A single nodule was noted on the facial skin at the angle of the left mandible. In total, nine lesions were excised from the head and neck region, eight of which were diagnosed as granular cell tumors. One specimen represented normal skeletal muscle. MICROSCOPIC FEATURES
Discussion Light microscopic examination demonstrated multiple specimens composed of stratified squamous epithelium, subjacent connective tissue, and regional striated muscle bun-
A r e v i e w o f the E n g l i s h l a n g u a g e literature identified 16 p r e v i o u s l y r e p o r t e d e x a m p l e s o f m u l t i p l e gran-
Multiple Granular Cell Tumors With Oral and Extraoral Involvement Intraoral Site
Bangle5 Arnn and Gordon 2j
Colberg and Hubay ~5
T T (2) BM T
Strong et al9 Garancis et al ~6
Bernstein et al j7 Noonan et al4
T T (2)
White et al ~8
Jones et al ~3
T P Larynx
Head and Neck Site
Baden et al j9 Pressoir and Chung 6
T (2) T
13 14, 15
Manara et al2° Apisarnthanarax ~
T T (2)
L Nose Scalp
T (6) FOM (2)
Rifkin et a112
SP T B
Collins and Jones
Toe Abdomen Foot Forearm Abdomen Toe Bronchus Wrist Back Leg Abdominal wall Back Clavicle Elbow Forearm Chest
Bronchus 8 Subcutaneous LL Labia majora Breast Thigh LL Scalp Shoulder Vocal cord Arm Back Chest 3
Extremities Breast Vulva Trunk Back Vulva
Total No. of Tumors 2 6 4 2 3 NS 5 NS
4 NS 2 7
Abbreviations: B, black; BM, buccal mucosa; F, female; FOM, floor of mouth; L, lip; LL, lower lip; M, male; NS, not stated; P, palate; SP, soft palate; T, tongue; UL, upper lip; W, white.
MULTIPLE GRANULAR CELL TUMORS
Table 2. Clinical Characteristics of Multiple Granular Cell Tumors With Oral and Extraoral involvement
Location of intraoral tumors (n = 35) T FOM BM P SP Age (yrs; n = 17) Mean Range Sex (n = 17) Female Male Race (n = 17) Black White NS Total no. of tumors (n = 113) Mean Range
3O (85.7%) 2 (5.7%) 1 (2.8%) 1 (2.8%) 1 (2.8%)
38.0 23-54 11 (64.7%) 6 (35.3%) 12 (70.6%) 1 (5.9%) 4 (23.5%) 6.6 2-57
Abbreviations: BM, buccal mucosa; FOM, floor of mouth; NS, not stated; P, palate; SP, soft palate; T, tongue.
ular cell tumors, all of which included intraoral lesions. These cases, along with the current case, are listed in Table 1. Table 2 summarizes the clinical features of the 17 examples of multiple granular cell tumors with intraoral involvement. Seventeen patients presented with a total of 113 tumors; the range was 2 to 57, and the mean number of lesions was 6.6. There were 35 intraoral tumors, and the tongue was the most prominent site (85.7%). Subcutaneous involvement accounted for the bulk of the remaining tumors. The ages ranged from 23 to 54 years, with a mean age of 38.0 years. A female (64.7%) and black (70.6%) predilection was noted in those cases where sex and race were stated. This predilection parallels that of keloids, leiomyomas, and fibroadenomas, v In seven cases (cases 5, 8, 10, 11, 13, 16, and 17), the multicentric tumors were synchronous, and in four cases (cases 2, 4, 7, and 9) they were nonsynchronous. In five cases (cases 1, 3, 12, 14, and 15), there was no indication of whether the tumors coexisted or arose over time. The remaining example (case 6) demonstrated features of concurrent multiple tumors and sequential tumor development over time. Our patient reported a single tumor on the tongue on initial presentation in December 1985, which she stated had been present for approximately 3 years. An incisional biopsy indicated a granular cell tumor. The patient returned for excision of the tumor in February 1986. A vulvar granular cell tumor was excised in October 1987, and tumors of the tongue, floor of the mouth, and facial skin were excised in November
1993. Thus, our case illustrates multiple tumors arising after surgical resection of an initial tumor, as has been reported in the literature. Often the subsequent granular cell tumors are remote from the initial surgical site. 7'2~ The multicentricity of some granular cell tumors has been likened to neurofibromas or viral w a r t s , 7 but Arnn and Gordon 2~ state that granular cell tumor multicentricity is more characteristic of a hamartomatous process rather than of true neoplastic growth. Granular cells are histologically distinctive and have been described in other well-recognized lesions. The congenital epulis of the newborn, a2'23 granular cell ameloblastoma, 24 granular cell odontogenic tumor, 25 peripheral odontogenic fibroma, 26 postsurgical granular cell lesion, z7 and some schwannomas 5 demonstrate granular cells in either the stroma or the epithelial component of the lesion. Although the resemblance is striking, the granular ceils of the aforementioned lesions show differences in immunohistochemical staining and ultrastructural features. The various locations of the lesions, collectively and individually, may account for the divergent opinions concerning the histogenesis of granular cells. An interesting observation is the female predilection in lesions containing granular cells except for the granular cell schwannoma and postsurgical granular cell lesion. Histiocytes, smooth and skeletal muscle cells, fibroblasts, neural connective tissue, and undifferentiated mesenchymal cells have been proposed as progenitor cells. 28 The granularity may be caused by lysosomes, phagocytized or stored material, angulate bodies, or cellular degeneration products. 13'28 The granular cell lesion reported in sites of prior surgery, particularly surgery involving smooth muscle, has a putative histiocytic origin. 27 A lesion from an unrelated case observed by one of the authors (BC) resembled a granular cell tumor except that it exhibited pleomorphism and occasional binucleate granular cells. The lesion was from a previous orthopedic surgery site. Surgical excision is the treatment of choice for granular cell tumors regardless of whether the lesions occur as isolated tumors or demonstrate multifocal involvement. In most cases this procedure is curative, even if the tumor is incompletely excised. However, a small percentage have recurred when tumor remained in the margins of excision. ~2 The phenomenon of multiple granular cell tumors developing after surgical procedures has been reported in a number of cases, 7'21 and subcutaneous tumors have arisen subsequent to surgical excision of deep tumors. Spontaneous regression also has been reported. 28 The prognosis following treatment is excellent. References
1. Apisarnthanarax P: Granular cell tumor. J Am Acad Derrnatol 5:171, 1981
STRULL AND DYM 2. Abrikossoff A: Uber myome ausgehend vonder quergestreiften willkurlichen muskulatur. Virchows Arch [A] 260:215, 1926 3. Rubenstein D, Shanker DB, Finlayson L, et al: Multiple cutaneous granular cell tumors in children. Pediatr Dennatol 4:94, 1987 4. Noonan JD, Horton CE, Old WL, et al: Granular cell myoblastoma of the head and neck: Review of the literature and 10 year experience. Am J Surg 138:611, 1979 5. Bangle RB Jr: A morphological and histochemical study of the granular cell myoblastoma. Cancer 5:950, 1952 6. Pressoir R, Chung EB: Granular cell tumor in black patients. J Natl Med Assoc 72:1171, 1980 7. Moscovic EA, Azar HA: Multiple granular cell tumors ("myoblastomas"): Case report with electron microscopic observations and review of the literature. Cancer 20:2032, 1967 8. Eisen RN, Kirby WM, O'Quinn JL: Granular cell tumor of the biliary tree. Am J Surg Pathol 15:460, 1991 9. Strong EW, McDivitt RW, Brasfield RD: Granular cell myoblastoma. Cancer 25:415, 1970 10. Powell EB: Granular cell myoblastoma. Arch Pathol 42:517, 1946 11. Radin DR, Zelner R, Ray MJ, et al: Multiple granular cell tumors of the skin and gastrointestinal tract. Am J Radiol 147:1305, 1986 12. Rifkin RH, Blocker SH, Palmer JO, et al: Multiple granular cell tumors: A familial occurrence in children. Arch Surg 121:945, 1986 13. Jones JK, Kuo T, Griffiths CM, et al: Multiple granular cell tumor. Laryngoscope 90:1646, 1980 14. LackEE, Worsham GF, Callihan MD, et al: Granular cell tumor: A clinicopathologic study of 110 patients. J Surg Oncol 13:301, 1980 15. Colberg JE, Hubay CA: Granular cell myoblastoma: A problem in diagnosis. Surgery 53:226, 1963
16. Garancis JC, Komorowski RA, Kuzma JF: Granular cell myoblastoma. Cancer 25:542, 1970 17. Bernstein BA, Murnane TW, Maloney PL: Tongue and multiple cutaneous granular-cell myoblastoma. Oral Surg 31:312, 1971 18. White SW, Gallager RL, Rodman OG: Multiple granular-cell tumors. J Dermatol Surg Oncol 6:57, 1980 19. Baden E, Divaris M, Quillard J: A light microscopic and immunohistochemical study of a multiple granular cell tumor and review of the literature. J Oral Maxillofac Surg 48:1093, 1990 20. Manara GC, De Panfilis G, Bacchi AB, et al: Fine structure of granular cell tumor of abrikossoff. J Cutan Pathol 8:277, 1981 21. Arnn ET, Gordon H: Granular-cell myoblastoma: Report of a case with multiple sites of origin. Lab Invest 6:142, 1957 22. Datum DD, Cibull ML, Geissler RH, et al: Investigation into the histogenesis of congenital epulis of the newborn. Oral Surg Oral Med Oral Pathol 76:205, 1993 23. Zuker RM, Buenechea R: Congential epulis: Review of the literature and case report. J Oral Maxillofac Surg 51:1040, 1993 24. Hartman KS: Granular-cell ameloblastoma: A survey of twenty cases from the Armed Forces Institute of Pathology. Oral Surg 38:241, 1974 25. Mirchandani R, Sciubba JJ, Mir R: Granular cell lesions of the jaws and oral cavity: A clinicopathologic, immunohistochemical, and ultrastructural study. J Oral Maxillofac Surg 47:1248, 1989 26. Lownie JF, Altini M, Shear M: Granular cell peripheral odontogenic fibroma. J Oral Pathol 5:295, 1976 27. Sobel HJ, Avrin E, Marquet E, et al: Reactive granular cells in sites of trauma: A cytochemical and ultrastructural study. Am J Clin Pathol 61:223, 1974 28. Regezi JA, Sciubba J: Oral Pathology: Clinical-Pathologic Correlations (ed 2). Philadelphia, PA, Saunders, 1993
J Oral Maxillofac Surg 53:711-713, 1995
Singultus: A Distressing Postsurgical Compfication GREGORY E. STRULL, DMD,* AND HARRY DYM, DDSt S i n g u l t u s , c o m m o n l y k n o w n as h i c c u p s , is a transient and b e n i g n a n n o y a n c e e x p e r i e n c e d o c c a s i o n a l l y b y m o s t p e o p l e . It is d e f i n e d as an i n v o l u n t a r y spasm o d i c c o n t r a c t i o n o f the d i a p h r a g m c a u s i n g a b e g i n n i n g i n s p i r a t i o n that is s u d d e n l y c h e c k e d b y c l o s u r e o f the epiglottis, p r o d u c i n g the c h a r a c t e r i s t i c sound. 1 Received from the Division of Oral and Maxillofacial Surgery, The Brooklyn Hospital Center, Brooklyn, NY. * Senior Resident. t Chief, Division of Oral and Maxillofacial Surgery; Director, Advanced Residency Training Program in Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Dym: Department of Dentistry, The Brooklyn Hospital Center, Box 224, 121 DeKalb Ave, Brooklyn, NY 11201. © 1995 American Association of Oral and MaxillofacialSurgeons 0278-2391/95/5306-001653.00/0
H i c c u p s are also d e s c r i b e d b y their duration. A h i c c u p b o u t is an e p i s o d e c o n s i s t i n g o f m o r e than a f e w hiccups, w h i c h c a n last up to 48 hours; persistent h i c c u p s are t h o s e that last o v e r 48 h o u r s and c o n t i n u e f o r up to a m o n t h ; i n t r a c t a b l e h i c c u p s c a n last l o n g e r than a m o n t h . 2 T h e f o l l o w i n g r e p o r t d e s c r i b e s a patient w h o developed persistent hiccups postoperatively.
Report of Case A 65-year-old African American man was referred to the oral and maxillofacial surgery clinic for evaluation and treatment of a severely atrophic maxilla. Examination of the soft tissues showed mobile fibrous tissue in the anterior maxilla. The remaining anterior mandibular teeth were periodontally compromised but clinically stable. The extraoral examination showed a decreased vertical dimension, with overclosure secondary to inability to wear a maxillary prosthesis.