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Laryngeal leiomyoma
International Elsevier
Journal of Pediatric
Otorhinolatyngology,
285
19 (1990) 285-288
PEDOT 00650
Laryngeal leiomyoma Sefa Kaya ‘, Levent Saydam I, and Sevket Ruacan Departments
of ’ Otorhinolaryngology
and 2 Pathology, Hacettepe Ankara (Turkey)
*
University, Medical Faculty,
(Received 21 December 1989) (Accepted 21 February 1990)
Key word: Subglottic laryngeal leiomyoma
Abstract
A case of leiomyoma arising from the subglottic area of a child is reported. This neoplasm is extremely rare in the larynx as only 14 cases have been described in the literature so far. Only 4 of these could be defined as subglottic. The clinical and histopathological features of laryngeal leiomyomas are briefly discussed.
Introduction
Leiomyomas of the larynx are extremely rare tumors. We observed such a case in a child. In our search of the literature we could find no more than 15 cases of laryngeal leiomyomas. Only 4 of these were localized in the subglottic area [3].
Case report
An 11-year-old female patient was admitted to the E.N.T. clinic of Hacettepe University Hospitals, with the complaint of suddenly developing respiratory distress. On examination all signs of upper respiratory airway obstruction were present. Chest X-ray showed no abnormality, According to the history given by parents a foreign body in the upper respiratory airway was suspected. Indirect laryngoscopy could not be performed, because of the patient’s state of panic. A direct laryngoscopic and bronchoscopic examination under general anaesthesia was performed. Correspondence: Ankara, Turkey. 0165-5876/90/$03.50
S. Kaya, Hacettepe aniversitesi
Tip Fakiiltesi, Kulak-Burun-BO&Z
0 1990 Elsevier Science Publishers B.V. (Biomedical Division)
Anabilim Dali,
286
Fig. 1. Section of biopsy specimen showing crossing bundles of smooth muscle tissue underneath the mt :taplasic su rface epithelium. Scattered inflammatory cells and dilated vessels are also noted. (H + E X 103.5)
Fig.
2. Higher
magnification cytoplasm.
of tumor Numerous
tissue. Tumor cells are fusiform with eosinophilic mitoses are found in each field. (H + E x 652)
elonga ted
281
On examination a spherical mass of approximately 1 cm in diameter was present 2 cm superior to the tracheal bifurcation. It was removed with forceps. The patient did well after the operation and respiratory distress disappeared. The histopathological examination of the specimen was reported as ‘smooth muscle tumor leiomyoma’ (Figs. 1 and 2). Seven months after the first operation the patient was again admitted to the clinic with respiratory distress. The patient’s clinical condition was similar to what was previously described. Direct laryngoscopy under general anaesthesia revealed a pinkish purple coloured mass attached to the subglottic lateral wall of the larynx. The mass was once again excised and the patient had an uneventful postoperative period. A tracheotomy was not needed. Histopathological results were consistent with earlier findings of leiomyoma.
Discussion The occurrence of a leiomyoma outside the uterus and gastrointestinal tract is considerably rare because of the paucity of smooth muscle elsewhere [3]. To account for the presence of smooth muscle tumors in areas normally deficient in smooth muscle 3 explanations have been given [l]: (1) Originating from aberrant undifferentiated mesenchyme; (2) Originating from smooth muscle elements in the walls of blood vessels; (3) Originating from both sources. Smooth muscle tumors are rarely seen in children and consequently very little is known about this particular type of neoplasia. But, there are constant features differentiating the adult type of tumor from those seen in the pediatric age group. For example, benign and malignant smooth muscle tumors of the uterus are most common in female adults but no comparable cases in children have been reported. Another feature according to Yannopoulos and Stout [8] is the extreme rarity of smooth muscle tumors in the soft tissues of children. In the larynx the tumor may be located both in supraglottic or subglottic spaces. In this study we presented a subglottic leiomyoma. In upper aerodigestive tract different localizations of tumors have been reported such as the nose [5], the pharynx [6] and the trachea [4]. In the subglottic space the tumor may originate from the posterior wall and the walls of small blood vessels. Histopathologically, the tumor consists of interlacing bundles of smooth muscle fibers. Duhing and Ayer [2] have presented an interesting theory of histogenesis. They postulated a progressive development of smooth muscle tumors, beginning with a hemangioma progressing to an angioma with much non-striated muscle, then gradually to a highly vascular mass, and finally a leiomyoma is formed. According to this developmental chain, vascular leiomyoma (angiomyoma) is a state rather than a distinct type of leiomyoma. We could find two cases of laryngeal angiomyomas reported in the literature [3,7]. It is important to distinguish benign leiomyoma from leiomyosarcoma. Elevated mitotic activity, anaplasia and bizarre cell forms are criterias used to differentiate
288
the malignant from the non-malignant forms. But, Yannopoulos and Stout [8] concluded that none of these are reliable and definitive features for malignancy. In laryngeal leiomyoma the aim of treatment is to maintain the air passage. Surgical removal of tumor may be performed either by endoscopic or external approach. Benign supraglottic and glottic tumors may be removed through the endoscopic route, but for sizeable subglottic tumors thyrotomy is the choice of surgical treatment [3].
References 1 Batsakis, J.G., Tumors of the Head and Neck, Williams and Wilkins, Baltimore, MD, 1974, pp. 270. 2 Duhing, J.T. and Ayer, J.P., Vascular leiomyoma: a study of 71 cases, Arch. Pathol., 68 (1956) 424. 3 Karma, P., Hyrynkangas, K. and Rasanen, O., Laryngeal leiomyoma, J. Laryngol. Otol., 92 (1978) 411-415. 4 Paludetti, G. and Rossignobi, M., Leiomyoma of the trachea: report of case and review of the literature, J. Laryngol. Otol., 98 (1984) 947-951. 5 Papavasiliou, A. and Micheals, L., Unusual leiomyoma of the nose (leiomyomablastoma): Report of a case, J. Otol., 95 (1981) 1281-1286. 6 Saymour-Jones, A., Leiomyoma of the pharynx with case report, J. Laryngol. Otol., 73 (1959) 396-398. 7 Shibata, K. and Kamure, S., Laryngeal angiomyoma (vascular leiomyoma); clinicopathological findings, Laryngoscope, 90 (1980) 1880-1886. 8 Yannopoulos, K. and Stout, A.P., Smooth muscle tumors in children, Cancer, 15 (1962) 958-971.
Journal of Pediatric
Otorhinolatyngology,
285
19 (1990) 285-288
PEDOT 00650
Laryngeal leiomyoma Sefa Kaya ‘, Levent Saydam I, and Sevket Ruacan Departments
of ’ Otorhinolaryngology
and 2 Pathology, Hacettepe Ankara (Turkey)
*
University, Medical Faculty,
(Received 21 December 1989) (Accepted 21 February 1990)
Key word: Subglottic laryngeal leiomyoma
Abstract
A case of leiomyoma arising from the subglottic area of a child is reported. This neoplasm is extremely rare in the larynx as only 14 cases have been described in the literature so far. Only 4 of these could be defined as subglottic. The clinical and histopathological features of laryngeal leiomyomas are briefly discussed.
Introduction
Leiomyomas of the larynx are extremely rare tumors. We observed such a case in a child. In our search of the literature we could find no more than 15 cases of laryngeal leiomyomas. Only 4 of these were localized in the subglottic area [3].
Case report
An 11-year-old female patient was admitted to the E.N.T. clinic of Hacettepe University Hospitals, with the complaint of suddenly developing respiratory distress. On examination all signs of upper respiratory airway obstruction were present. Chest X-ray showed no abnormality, According to the history given by parents a foreign body in the upper respiratory airway was suspected. Indirect laryngoscopy could not be performed, because of the patient’s state of panic. A direct laryngoscopic and bronchoscopic examination under general anaesthesia was performed. Correspondence: Ankara, Turkey. 0165-5876/90/$03.50
S. Kaya, Hacettepe aniversitesi
Tip Fakiiltesi, Kulak-Burun-BO&Z
0 1990 Elsevier Science Publishers B.V. (Biomedical Division)
Anabilim Dali,
286
Fig. 1. Section of biopsy specimen showing crossing bundles of smooth muscle tissue underneath the mt :taplasic su rface epithelium. Scattered inflammatory cells and dilated vessels are also noted. (H + E X 103.5)
Fig.
2. Higher
magnification cytoplasm.
of tumor Numerous
tissue. Tumor cells are fusiform with eosinophilic mitoses are found in each field. (H + E x 652)
elonga ted
281
On examination a spherical mass of approximately 1 cm in diameter was present 2 cm superior to the tracheal bifurcation. It was removed with forceps. The patient did well after the operation and respiratory distress disappeared. The histopathological examination of the specimen was reported as ‘smooth muscle tumor leiomyoma’ (Figs. 1 and 2). Seven months after the first operation the patient was again admitted to the clinic with respiratory distress. The patient’s clinical condition was similar to what was previously described. Direct laryngoscopy under general anaesthesia revealed a pinkish purple coloured mass attached to the subglottic lateral wall of the larynx. The mass was once again excised and the patient had an uneventful postoperative period. A tracheotomy was not needed. Histopathological results were consistent with earlier findings of leiomyoma.
Discussion The occurrence of a leiomyoma outside the uterus and gastrointestinal tract is considerably rare because of the paucity of smooth muscle elsewhere [3]. To account for the presence of smooth muscle tumors in areas normally deficient in smooth muscle 3 explanations have been given [l]: (1) Originating from aberrant undifferentiated mesenchyme; (2) Originating from smooth muscle elements in the walls of blood vessels; (3) Originating from both sources. Smooth muscle tumors are rarely seen in children and consequently very little is known about this particular type of neoplasia. But, there are constant features differentiating the adult type of tumor from those seen in the pediatric age group. For example, benign and malignant smooth muscle tumors of the uterus are most common in female adults but no comparable cases in children have been reported. Another feature according to Yannopoulos and Stout [8] is the extreme rarity of smooth muscle tumors in the soft tissues of children. In the larynx the tumor may be located both in supraglottic or subglottic spaces. In this study we presented a subglottic leiomyoma. In upper aerodigestive tract different localizations of tumors have been reported such as the nose [5], the pharynx [6] and the trachea [4]. In the subglottic space the tumor may originate from the posterior wall and the walls of small blood vessels. Histopathologically, the tumor consists of interlacing bundles of smooth muscle fibers. Duhing and Ayer [2] have presented an interesting theory of histogenesis. They postulated a progressive development of smooth muscle tumors, beginning with a hemangioma progressing to an angioma with much non-striated muscle, then gradually to a highly vascular mass, and finally a leiomyoma is formed. According to this developmental chain, vascular leiomyoma (angiomyoma) is a state rather than a distinct type of leiomyoma. We could find two cases of laryngeal angiomyomas reported in the literature [3,7]. It is important to distinguish benign leiomyoma from leiomyosarcoma. Elevated mitotic activity, anaplasia and bizarre cell forms are criterias used to differentiate
288
the malignant from the non-malignant forms. But, Yannopoulos and Stout [8] concluded that none of these are reliable and definitive features for malignancy. In laryngeal leiomyoma the aim of treatment is to maintain the air passage. Surgical removal of tumor may be performed either by endoscopic or external approach. Benign supraglottic and glottic tumors may be removed through the endoscopic route, but for sizeable subglottic tumors thyrotomy is the choice of surgical treatment [3].
References 1 Batsakis, J.G., Tumors of the Head and Neck, Williams and Wilkins, Baltimore, MD, 1974, pp. 270. 2 Duhing, J.T. and Ayer, J.P., Vascular leiomyoma: a study of 71 cases, Arch. Pathol., 68 (1956) 424. 3 Karma, P., Hyrynkangas, K. and Rasanen, O., Laryngeal leiomyoma, J. Laryngol. Otol., 92 (1978) 411-415. 4 Paludetti, G. and Rossignobi, M., Leiomyoma of the trachea: report of case and review of the literature, J. Laryngol. Otol., 98 (1984) 947-951. 5 Papavasiliou, A. and Micheals, L., Unusual leiomyoma of the nose (leiomyomablastoma): Report of a case, J. Otol., 95 (1981) 1281-1286. 6 Saymour-Jones, A., Leiomyoma of the pharynx with case report, J. Laryngol. Otol., 73 (1959) 396-398. 7 Shibata, K. and Kamure, S., Laryngeal angiomyoma (vascular leiomyoma); clinicopathological findings, Laryngoscope, 90 (1980) 1880-1886. 8 Yannopoulos, K. and Stout, A.P., Smooth muscle tumors in children, Cancer, 15 (1962) 958-971.