Laryngeal leiomyoma: a case report and literature review

DOI: https://doi.org/10.21203/rs.3.rs-1790415/v1

Abstract

Leiomyomas (LMs) are mesenchymal tumors that derive from smooth muscle cells. Here, we report a rare case of laryngeal leiomyoma (LLM) admitted to our center. We also reviewed and summarized the clinical characteristics of reported LLMs to help clinicians gain a better understanding of this uncommon disease and improve the diagnosis, treatment, and patient postoperative recovery.

Introduction

Leiomyomas (LMs) are mesenchymal tumors that derive from smooth muscle cells. Most commonly leiomyomas are seen in the organs with abundance of smooth muscle such as uterus and gastrointestinal tract. However, LMs are rarely seen in the head and neck region, and the incidence of oral involvement is only 0.065%, accounting for 0.42% of all soft tissue tumors in the oral cavity. To the best of our knowledge, there were no more than 50 cases reported till now. Here, we report a rare case of laryngeal leiomyoma (LLM) admitted to our center. We also reviewed and summarized the clinical characteristics of reported LLMs to help clinicians gain a better understanding of this uncommon disease and improve the diagnosis, treatment, and patient postoperative recovery.

Case Presentation

On xxx, a 49-year-old male patient admitted to our E.N.T outpatient clinic with a chief complaint of pharynx discomfort for two months. During the course of the disease, the patient had no fever, cough, sore throat, and difficulties in swallowing or breathing. He was a non-smoker without remarkable medical history and family history of head and neck tumors. Electronic laryngoscopy performed under topical anesthesia showed a solitary, pink mass at the tubercle of epiglottis. Biopsy was obtained via laryngoscopy (Fig. 1), and the pathological examination reported spindle cell proliferation, prone to smooth muscle-derived tumor lesions. Operation through laryngeal endoscopy was carried out under general anesthesia and the lesion was striped easily (Fig. 2). The tissues submitted for pathological examination showed polypoid tumor tissue at the base. Hematoxylin and eosin (HE) staining confirmed that the mass was composed of myogenic, indistinct shape tumor cells surrounding the submucosal small salivary glands without sign of necrosis or mitotic. The positive immunohistochemical (IHC) staining of desmin and smooth-muscle actin indicated a smooth muscle origin (Fig. 3). After operation, the patient was in stable condition, and was discharged 2 days after surgery. During the one year postoperative following-up, the patient was in stable condition without evidence of recurrence. The informed consent for publication was also obtained from the patient.

Discussion

LMs are benign connective tissue tumors involving the smooth muscle components of the organs. They occur mostly in the uterus, small intestine and esophagus. In contrast, LMs occur rarely in the head and neck region due to the paucity of smooth muscle [1]. Three different types of leiomyoma are recognized: the common leiomyoma, the vascular leiomyoma (angiomyoma) and the epithelioid leiomyoma. Although the smooth muscle were rarely seen in laryngeal tissues, all three types have been found in the larynx [2]. Histopathologically, angiomyoma is made up of many variant blood vessels. Whirlpools of smooth muscle fibers can be observed around the vascular endothelium, but lacking mitotic and well-differentiated smooth muscle bundles, and sometimes accompanied by mucoid lesions[3]. Epithelioid leiomyomas manifest as tumor cell masses with round or oval nucleus, with or without vacuoles, and clear cell changes in the rich eosinophilic cytoplasm [4]. The common leiomyoma are characterized by intertwined spindle cells, cigar-shaped nuclei, eosinophilic cytoplasm, and a positive reaction to smooth-muscle actin or desmin[1]. The case we reported here is consistent with the pathological diagnosis of common leiomyomas. LMs that occur in the larynx are extremely rare. We searched the PubMed and Geenmedical databases using laryngeal leiomyoma as a keyword and analyzed the search results (Table 1). To our knowledge, including this case, there are only 12 literatures reported relevant patients. Among them, there were 12 men and 5 women between 7 and 75 years old, half of which were between 40 and 70 years old and the median was 48 years old. Apparently, a male dominance (11/16) was observed as shown in Table 1. LMs that occur in the upper aerodigestive tract can be distributed in the nose, pharynx, and trachea. The most commonly reported sites of laryngeal LMs were the supraglottic area (7/16), the glottal area (5/16) and the subglottic area (4/16) in order. The common symptoms of LMs in the glottic region were hoarseness. Most subglottic LMs may originate from the posterior wall and the walls of small blood vessels[5], and most of patients were admitted to the emergency department due to dyspnea. Dysphagia and foreign body sensation in the throat were also usual symptoms. The case we presented here is a supraglottic LMs. In addition, it is believed that although the malignant transformation of laryngeal LMs has not been reported, benign LMs must be distinguished from malignant leiomyosarcoma, and the mitotic rate is the most reliable standard indicating the malignancy of LMs [2].

To date, the treatment of LLMs has not been standardized due to its rarity, so treatment decisions are often made empirically from case to case. In these 16 case reports, the most recommended treatment is complete surgical resection through endoscopy (13 cases) or external surgery (3 cases). The method of surgery depends on the location of the tumor. Due to the large mass, 1 out of 16 patients with laryngeal LMs requires emergency tracheal intubation at the time of diagnosis. If the tumor seriously affects breathing, a tracheotomy should be performed first, and then the tumor can be removed through a neck approach. In addition, if the video laryngoscope shows a smooth surface and no new organisms with a pedicle, no biopsy should be taken to avoid bleeding. Recurrence after complete resection is rare, and only 3 literature reports recurrence in patients diagnosed with laryngeal LMs after surgery. Even though it is clear that no case of external incisions has reported perioperative complications or subsequent local recurrence, endoscopic surgery is still favored due to its less damage to tissues. With the development of transoral instruments, laser technology and low-temperature plasma in the past 20 years, it is not difficult to foresee that the treatment of laryngeal LMs will be more diverse.

Conclusion

As a very rare tumor, surgical resection is still the preferred treatment for LLMs. Although the recurrence of leiomyomas after resection is rare, it is still possible. Therefore, its early diagnosis and differential diagnosis with malignant leiomyosarcoma still have important clinical significance.

Declarations

Ethics approval and consent to participate: Ethics approval had been granted by the institutions for all of the manuscripts involved, along with written informed consent and corresponding ethics codes and approval.

Consent for publication: Not applicable.

Availability of data and materials: All data generated or analysed during this study are included in this published article

Competing interests: The authors declare that they have no competing interests.

Funding: Not applicable

Authors' contributions - provide individual author contribution: .Yu Wu.Tingting Yu and Xin Wang worte the main manuscript text,and Shengrui Gao prepared figure1.All authors reviewed the manuscript.

Acknowledgements: Not applicable

Authors' information (optional): 

Yu Wu, Tingting Yu,Shengrui Gao and Xin Wang all in the Department of Otolaryngology, Head and Neck Surgery, the First Hospital of Jilin University, Changchun, China.

Academic degrees for all authors:

Yu Wu: Master degree; Tingting Yu:Doctor degree; Shengrui Gao: Master degree;Xin Wang: Doctor degree.

E-mail addresses for all authors:

Yu Wu:[email protected];Tingting Yu: [email protected]; Shengrui Gao:[email protected]; Xin Wang:[email protected]

References

  1. Nagaraju, G., et al., Recurrent laryngeal leiomyoma. Journal of Laryngology and Voice, 2012. 2(2): p. 95.
  2. McKiernan, D.C. and G.W. Watters, Smooth muscle tumours of the larynx. J Laryngol Otol, 1995. 109(1): p. 77-9.
  3. Xu, Y., S. Zhou, and S. Wang, Vascular leiomyoma of the larynx: a rare entity. Three case reports and literature review. ORL J Otorhinolaryngol Relat Spec, 2008. 70(4): p. 264-7.
  4. Hellquist, H.B., et al., Epithelioid leiomyoma of the larynx. Histopathology, 1994. 24(2): p. 155-9.
  5. Kaya, S., L. Saydam, and S.J.I.j.o.p.o. Ruacan, Laryngeal leiomyoma. 1990. 19(3): p. 285-8.
  6. KARATAY, S. and B.J.A.M.A.a.o.o. PARS, Leiomyoma laryngis. 1959. 69(2): p. 224-6.
  7. Karma, P., et al., Laryngeal leiomyoma. 1978. 92(5): p. 411-5.
  8. Gallagher, T., J.J.E. Sinacori, nose,, and t. journal, Laryngeal leiomyoma. 2010. 89(8): p. 346-7.
  9. NEIVERT, H. and L.J.A.o.o. ROYER, Leiomyoma of the larynx. 1946. 44: p. 214-8.
  10. Kendall, C.J.H., Leiomyoma with atypical cells (atypical leiomyoma) in the larynx. 2000. 37(5): p. 475-6.
  11. Chang, Y., et al., Subglottic leiomyoma: report of a case. 2002. 101(11): p. 795-7.
  12. Burzej-Uchto, M., et al., [A rare case of laryngeal leiomyoma]. Otolaryngol Pol, 2006. 60(2): p. 207-9.
  13. Ebert, W. and H.J.Z.f.a.P.u.p.A. Scholz, Leiomyomas of the larynx. 1979. 123(6): p. 580-3.

Tables

Table 1 is available in the Supplementary Files section.