Laryngeal leiomyoma: A case report and review of literature

Abstract

BACKGROUND

Leiomyomas (LMs) are mesenchymal tumors that arise from smooth muscle cells. LMs most commonly arise in organs with an abundance of smooth muscle such as the uterus and gastrointestinal tract. Conversely, LMs are rarely detected in the head and neck region. In this study, we report a rare case of laryngeal LM (LLM) and summarized the clinical characteristics of reported LLMs to help clinicians better understand this rare disease and improve its diagnosis, treatment, and postoperative course.

CASE SUMMARY

A 49-year-old man was admitted to our ENT outpatient clinic with a chief complaint of pharynx discomfort for 2 months. Laryngoscopy performed under topical anesthesia revealed a solitary, pink mass at the tubercle of epiglottis. Surgery via laryngeal endoscopy was performed under general anesthesia, and the lesion was excised easily. Positive immunohistochemical staining for desmin and smooth-muscle actin indicated a smooth muscle origin and the diagnosis was laryngeal leiomyoma. After surgery, the patient’s condition was stable, and he was discharged 2 d after surgery. During the 1-year postoperative period, the patient’s condition remained stable without evidence of recurrence.

CONCLUSION

Surgical resection is the preferred treatment for LLMs, its early diagnosis and differential diagnosis have important clinical significance.

Key Words: Leiomyoma, Larynx, Smooth tumor, Endoscopic surgery, Case report

Core Tip: We present a rare case of laryngeal leiomyoma (LLM) admitted to our institution, while also providing a comprehensive review and synthesis of the clinical characteristics associated with reported LLMs. Our aim is to enhance clinicians' comprehension of this uncommon disease, thereby facilitating improved diagnosis, treatment strategies, and postoperative patient recovery.

INTRODUCTION

Leiomyomas (LMs) are mesenchymal tumors that originate from smooth muscle cells. LMs most commonly arise in organs with an abundance of smooth muscle such as the uterus and gastrointestinal tract. Conversely, LMs are rarely detected in the head and neck region, and the incidence of oral LM is only 0.065%, accounting for 0.42% of all soft tissue tumors in the oral cavity. To the best of our knowledge, no more than 50 cases have been reported to date. In this study, we report a rare case of laryngeal LM (LLM) in a patient 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 postoperative course.

CASE PRESENTATION

Chief complaints

A 49-year-old man was admitted to our ENT outpatient clinic with a chief complaint of pharynx discomfort persisting for 2 months.

History of present illness

During his clinical course, he had no fever, cough, sore throat, or difficulties in swallowing or breathing.

History of past illness

He had no previous medical history.

Personal and family history

He was a non-smoker without a remarkable medical history or family history of head and neck tumors.

Physical examination

Through indirect laryngoscopy, revealed a solitary, pink mass at the tubercle of epiglottis.

Laboratory examinations

Biopsy was performed via laryngoscopy, and the pathological examination revealed spindle cell proliferation, which is typical of smooth muscle-derived tumors. The tissues were submitted for pathological examination, revealing polypoid tumor tissue at the base. Hematoxylin and eosin staining demonstrated that the mass was composed of myogenic, indistinct shape tumor cells surrounding the submucosal small salivary glands without sign of necrosis or mitoses. Positive immunohistochemical staining for desmin and smooth-muscle actin indicated a smooth muscle origin (Figure 1).

Figure 1 Pathological section. A: Microphotograph of laryngeal leiomyoma showing spindle cells arranged in fascicles and storiform pattern (Hematoxylin and eosin, × 10), microphotograph showing; B: Smooth-muscle actin; C: Desmin positivity.

Imaging examinations

Laryngoscopy performed under topical anesthesia revealed a solitary, pink mass at the tubercle of epiglottis.

FINAL DIAGNOSIS

LLM.

TREATMENT

Surgery via laryngeal endoscopy was performed under general anesthesia, and the lesion was excised easily (Figure 2). No other interventions were given during treatment.

Figure 2 Laryngoscopy images. A: Laryngoscopy imaging showing the solitary mass on the epiglottis; B: Intraoperatively, the mass located on the epiglottis was completely removed.

OUTCOME AND FOLLOW-UP

After surgery, the patient condition was stable, and he was discharged 2 d after surgery (Figure 3). During the 1-year postoperative period, the patient condition remained stable without evidence of recurrence. The patient provided informed consent for publication of his case.

Figure 3 Timeline.

DISCUSSION

LMs are benign tumors originating from the smooth muscle components of organs. They mostly arise in the uterus, small intestine, and esophagus. By contrast, LMs rarely occur in the head and neck region because of the paucity of smooth muscle[1]. Three different types of LM are recognized: Common LM, vascular LM (angiomyoma), and epithelioid LM. Although smooth muscle is rarely present in laryngeal tissues, all three types of LM have been found in the larynx[2]. Histopathologically, angiomyoma consists of many variant blood vessels. Whirlpools of smooth muscle fibers can be observed around the vascular endothelium. However, they lack mitotic and well-differentiated smooth muscle bundles, and they are sometimes accompanied by mucoid lesions[3]. Epithelioid LMs manifest as tumor cell masses with round or oval nucleus, with or without vacuoles, and clear cell changes in the rich eosinophilic cytoplasm[4]. Common LMs are characterized by intertwined spindle cells, cigar-shaped nuclei, eosinophilic cytoplasm, and positivity for smooth-muscle actin or desmin[1]. The findings of the current case are consistent with the pathological diagnosis of common LMs. LMs rarely arise in the larynx. We searched the PubMed and Geen Medical databases using LLM as a keyword and analyzed the search results (Table 1). To our knowledge, including this case, only 12 studies reported relevant patients[1-3,5-13]. The patients included 11 men and 5 women aged 7-75 years old, and half of the patients were aged 40–70 years (median age, 48 years). Apparently, a male dominance (11/16) was observed as shown in Table1. 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). The most common symptom of LMs in the glottic region was hoarseness. Most subglottic LMs originate from the posterior wall and the walls of small blood vessels[5], and most patients were admitted to the emergency department because of dyspnea. Dysphagia and foreign body sensation in the throat were also typical symptoms[3,6]. The current case involved a supraglottic LM. In addition, it is believed that although the malignant transformation of laryngeal LMs has not been reported, benign LMs must be distinguished from leiomyosarcoma, and the mitotic rate is the most reliable standard indicating the malignancy of LMs[2].

Table 1 Clinical characteristics of reported laryngeal leiomyomas cases.

Ref.	Age	History	Gender	Symptom	Side and location	Treatment	Follow-up
Nagaraju et al[1]	18	3 wk	Female	Hoarseness	Left vocal cord	Microlaryngoscopy	Recurrence
McKiernan et al[2]	28	6 months	Female	Hoarseness	The right vocal fold	Microlaryngoscopy	No recurrence
Xu et al[3]	53	Not mentioned	Male	Hoarseness	Left vocal cord	Microlaryngoscopy	No recurrence
Xu et al[3]	47	6 yr	Male	Foreign body sensation	Right aryepiglottic fold	Tracheotomy and lateral neck approach	No recurrence
Xu et al[3]	62	Not mentioned	Male	Globus feeling	Lingual surface of the epiglottis	Tracheotomy and lateral neck approach	No recurrence
Kaya et al[5]	11	Suddenly	Female	Respiratory distress	Subglottic lateral wall of the larynx	Microlaryngoscopy	Recurrence
Karatay and Pars et al[6]	40	Not mentioned	Female	Difficulty in swallowing	Right aryepiglottic fold	Indirect laryngoscopy	No recurrence
Karma et al[7]	71	6 months	Female	Respiratory distress	Left lateral subglottic wall	Tracheotomy and thyrotomy route	No recurrence
Gallagher and Sinacori[8]	56	2 yr	Male	Hoarseness	Right false vocal cord	Microlaryngoscopy	No recurrence
Neivert and Royer[9]	10	7 months	Male	Hoarseness	Vestibule of the larynx	Tracheotomy and laryngoscopy	No recurrence
Neivert and Royer[9]	67	Not mentioned	Male	Not mentioned	The left ventricle	Indirect laryngoscopy	No recurrence
Neivert and Royer[9]	75	16 yr	Male	Respiratory distress	Vestibule of the larynx	Tracheotomy and thyrotomy route	No recurrence
Matsumoto et al[10]	53	2 months	Male	Hoarseness	The left vocal cord	Microlaryngoscopy	No recurrence
Chang et al[11]	7	2 wk	Male	Respiratory distress	Below the right vocal cord	Microlaryngoscopy	No recurrence
Lawal et al[12]	7	Suddenly	Male	Respiratory distress	Right false cord and subglottic area	Microlaryngoscopy	No recurrence
Ebert et al[13]	65	Several years	Male	Not mentioned	Left vocal cord	Laryngectomy	No recurrence

To date, the treatment of LLM has not been standardized because of its rarity, and thus treatment decisions are often made empirically from case to case. In these 16 case reports, the most recommended treatment was complete surgical resection via endoscopy (11 cases) or external surgery (5 cases). The method of surgery depends on the location of the tumor. Because of the large mass, one reported patient with laryngeal LM required emergency tracheal intubation at the time of diagnosis. If the tumor seriously affects breathing, tracheotomy should be performed first, and then the tumor can be removed through a neck approach[7]. In addition, if videolaryngoscopy reveals a smooth surface and no new organisms with a pedicle, no biopsy should be taken to avoid bleeding[6,9]. The diagnosis is based on histologic examination and confirmed by immunohistochemistry[8,10]. Recurrence after complete resection is rare[11], and only 2 studies report recurrence after surgery in patients diagnosed with laryngeal LMs[1,5,11]. Although no case of external incisions resulted in perioperative complications or subsequent local recurrence[12,13], endoscopic surgery is favored because it causes less tissue damage. 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 become more diverse.

CONCLUSION

As an extremely rare tumor, surgical resection remains the preferred treatment for LLM. Although the recurrence of LM after resection is rare, it is possible. Therefore, early diagnosis and differentiation of LM from leiomyosarcoma have important clinical significance.