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Recurrent vascular leiomyoma of the larynx: Clinical and histopathologic characteristics and treatment.

Abstract

Laryngeal vascular leiomyomas are uncommon benign tumors that seldom recur following complete excision. The choice of excision procedure--via direct laryngoscopy or an open approach--is dictated by tumor size, the expected amount of blood loss, and any comorbidities the patient may have. We report an unusual case of a recurrent laryngeal vascular leiomyoma in a 64-year-old woman who also had a concurrent parathyroid adenoma and a history of breast carcinoma. A surgical resection via an external approach along with laser resection of a small glottic component was needed.

Introduction

Vascular leiomyomas, also called angiomyomas, are uncommon, slowly growing, benign tumors that seldom recur following complete excision. The occurrence of a leiomyoma outside the uterus and gastrointestinal tract is very rare. In cases of laryngeal leiomyoma, symptoms of dyspnea, hoarseness, stridor, and dysphagia develop progressively with tumor growth. The treatment is complete surgical removal via direct laryngoscopy or an external neck approach.

We report an unusual case of a recurrent laryngeal vascular leiomyoma in a patient with a concurrent parathyroid adenoma and a history of breast carcinoma.

Case report

A 64-year-old woman presented to the Emergency Department for evaluation of dyspnea and hoarseness. The patient had progressive dyspnea and stridor associated with severe cachexia that had progressed over the previous year. She was a nonsmoker and denied hemoptysis.

Twelve years earlier, the patient had been hospitalized for the treatment of dyspnea and dysphagia secondary to the presence of a 1-cm reddish mass that had involved the left aryepiglottic fold and caused supraglottic narrowing. At that time, C[O.sub.2] laser debulking and a biopsy of the mass were performed. A significant quantity of blood loss during the procedure was reported. Examination of the pathology specimen identified the mass as a vascular leiomyoma. The patient's history included a bilateral mastectomy with radiation and chemotherapy for lobular breast carcinoma 20 years earlier.

At the most recent presentation, the patient exhibited marked respiratory distress, and she was in an acute state of confusion secondary to hypercapnia and hypoxemia. Flexible laryngoscopy revealed the presence of a large, reddish, spherical, 4-cm mass that had emerged from the left side of the larynx. The airway posterior to the mass was only 1 to 2 mm wide. No cervical lymphadenopathy was palpated. An immediate awake tracheotomy was performed. Once the patient was stable, magnetic resonance imaging (MRI) was obtained. MRI demonstrated a 3.5-cm vascular mass that primarily involved the supraglottis with possible extension into the glottic area (figure 1, A). In addition, a large parathyroid mass was detected incidentally (figure 1, B). When laboratory testing revealed elevations of the serum calcium and parathyroid hormone (PTH) levels, the parathyroid mass was presumed to be an adenoma.

The patient was taken to the operating room for removal of both tumors. An external neck approach to access the laryngeal mass via a supraglottic thyrotomy and removal of the parathyroid adenoma were performed. The intraoperative PTH level dropped from 20.03 to 6.91 pmol/L, and quick section identified parathyroid tissue, which was later confirmed to represent an adenoma. Most of the tumor ball was pilled away from the mucosa. The possibility that a small tail of tumor had extended in the direction of the glottis was considered, but since the tumor was benign, we decided to spare the area and avoid a cordectomy, which would have caused further deformation of the larynx and voice change. Blood loss was not exceptional.

[FIGURE 1 OMITTED]

On gross examination, the laryngeal tumor was encased in a fibrous capsule that had a distinct boundary that varied in thickness (figure 2).

Postoperatively, swelling was present in the area of the redundant mucosa. Although it subsided significantly over a 2-week period, there was still sufficient tissue present to impede decannulation. On direct laryngoscopy a few weeks later, the tumor stump was removed by laser ablation, and the airway passage was deemed to be adequate. The patient was successfully decannulated a while later.

[FIGURE 2 OMITTED]

Microscopic examination of the laryngeal mass revealed well-demarcated nodules of smooth-muscle tissue punctuated with thick-walled vessels with partially patent lumina (figure 3). Areas of myxoid change were also seen. The vessels were difficult to classify because they were not typical of veins or arteries; their thick walls and small lumina were similar to those of arteries, but they consistently lacked internal and external elastic laminae. Immunohistochemical staining for the smooth-muscle marker [alpha]-actin was positive. These findings were consistent with the diagnosis of recurrent laryngeal vascular leiomyoma, solid type.

Discussion

Vascular leiomyomas usually present in the subcutis of the lower limbs; others have been reported in the upper limbs and in the head and neck region. Common head and neck sites include the submandibular area, lower lip, oral cavity, nasal cavity, auricle, and the skin of the occipital area. (1) Laryngeal tumors are rare; when they do occur, they may be located in either the supraglottic or subglottic space. (2) These tumors are also rare in the upper respiratory tract. (3)

Most vascular leiomyomas present in middle-aged and elderly patients. (4) Dyspnea, hoarseness, stridor, and dysphagia develop progressively with tumor growth. An urgent tracheotomy at presentation to secure the airway is sometimes necessary, as it was in our case.

Preoperative histologic findings on biopsy (i.e., normal epithelium) may be confusing because of the tumor's submucosal location; evaluation may also be complicated by bleeding. The treatment is complete surgical removal via direct laryngoscopy or an external neck approach. The decision must be individualized, taking into consideration the complications of each technique. In our case, we preferred an external approach because of the tumor's size and vascularity, as well as the finding of a concomitant parathyroid adenoma. Possible complications of the external neck approach include perfuse bleeding, vocal fold deformation, voice impairment and aspiration, nerve damage (especially to the vagus nerve compartments), the development of airway stenosis, and an unsatisfactory aesthetic outcome. Previously reported cases have been characterized by profuse bleeding during and after surgery, as happened to our patient during the first operation 12 years previously. Some authors have advocated preoperative embolization before laryngeal transoral resection. (5)

[FIGURE 3 OMITTED]

Vascular leiomyomas are usually located in the subcutis, and less often in the deep dermis, where they produce overlying elevations of the skin but without surface changes of the epidermis. Grossly, the tumors are circumscribed, glistening, white-gray nodules. There are three distinct histologic types of vascular leiomyoma: solid (capillary), venous, and cavernous. (6) Immunohistochemical staining for the smooth-muscle markers [alpha]-actin, desmin, and vimentin may help with the diagnosis. A notable feature of our case was the absence of mitotic activity, anaplasia, and bizarre cell forms--a finding that ruled out a leiomyosarcoma. It is sometimes difficult to distinguish a benign leiomyoma from a leiomyosarcoma on the basis of histologic findings alone, so the clinical course of the disease may have to be taken into account.

To the best of our knowledge, only i other case of a recurrent vascular leiomyoma of the larynx has been published in the literature. (7) Parathyroid-hormone-related peptide may play an important role in the proliferation activity of leiomyoma. (8) However, previously reported cases of a concomitant parathyroid adenoma and a leiomyoma occurred only in patients with multiple endocrine neoplasia type 1. (9) Moreover, there has not been any documented case of a concomitant parathyroid adenoma and a laryngeal vascular leiomyoma.

The fact that recurrence is possible in cases of laryngeal leiomyosarcoma necessitates long-term follow-up.

References

(1.) Wang CP, Chang YL, Sheen TS. Vascular leiomyoma of the head and neck. Laryngoscope 2004;114(4):661-5.

(2.) Kaya S, Saydam L, Ruacan S. Laryngeal leiomyoma. Int J Pediatr Otorhinolaryngol 1990;19(3):285-8.

(3.) Mehta RP, Faquin WC, Franco RA. Pathology quiz case 2. Angiomyoma of the larynx. Arch Otolaryngol Head Neck Surg 2004;130(7):889, 890-1.

(4.) Xu Y, Zhou S, Wang S. Vascular leiomyoma of the larynx: Three case reports and literature review. ORL J Otorhinolaryngol Relat Spec 2008;70(4):264-7.

(5.) Shibata K, Komune S. Laryngeal angiomyoma (vascular leiomyoma): Clinicopathological findings. Laryngoscope 1980;90 (11 Pt 1):1880-6.

(6.) Hachisuga T, Hashimoto H, Enjoji M. Angioleiomyoma. A clinicopathologic reappraisal of 562 cases. Cancer 1984;54(1):126-30.

(7.) Anderson TD, Weinstein GS. Recurrent angiomyoma (vascular leiomyoma) of the larynx after laser excision. Otolaryngol Head Neck Surg 2000;123(5):646-7.

(8.) Naito S, Shimizu K, Akino K, et al. Autocrine/paracrine involvement of parathyroid hormone-related peptide in vascular leiomyoma. Endocr J 2002;49(3):335-41.

(9.) McKeeby JL, Li X, Zhuang Z, et al. Multiple leiomyomas of the esophagus, lung, and uterus in multiple endocrine neoplasia type 1. Am J Pathol 2001;159(3):1121-7.

Nir Hirshoren, MD; Jeffrey M. Weinberger, MD, FRCSC; Tzahi Neuman, MD; Ophir Ilan, MD, PhD; Avraham Ben-Yaakov, MD

From the Department of Otolaryngology/Head and Neck Surgery (Dr. Hirshoren, Dr. Weinberger, Dr. Ilan, and Dr. Ben-Yaakov) and the Department of Pathology (Dr. Neuman), Hadassah Ein-Kerem University Hospital, Jerusalem.

Corresponding author: Nit Hirshoren, MD, Department of Otolaryngology/Head and Neck Surgery, Hadassah Ein-Kerem University Hospital, Jerusalem 91120, Israel. E-mail: drnir@hadassah.org.il
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Title Annotation:ORIGINAL ARTICLE
Comment:Recurrent vascular leiomyoma of the larynx: Clinical and histopathologic characteristics and treatment.(ORIGINAL ARTICLE)
Author:Hirshoren, Nir; Weinberger, Jeffrey M.; Neuman, Tzahi; Ilan, Ophir; Ben-Yaakov, Avraham
Publication:Ear, Nose and Throat Journal
Article Type:Report
Geographic Code:7ISRA
Date:Aug 1, 2010
Words:1516
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