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Abstract

Extracardiac rhabdomyoma is a tumor that rarely occurs in head and neck region. Adult and fetal types of extracardiac rhabdomyoma are diagnosed only by histopathological examination. In the oral cavity, this lesion usually affects the mouth floor and tongue. Despite the low incidence, adult rhabdomyoma should be considered in the differential diagnosis of oral cavity lesions, and histopathological evaluation might be helpful for the final diagnosis. This study aims to report a rare case of rhabdomyoma mimicking reactive lesions and review the literature. A 34-year-old male was referred to the pathology department with a yellowish sessile lesion in the labial maxillary vestibule. During an excisional biopsy, a mass of unencapsulated soft tissue beneath the vestibular mucosa was observed. The yellowish color in clinical features has led to lipoma as the initial clinical diagnosis. The histological examination exhibited an encapsulated, well-circumscribed benign neoplasm composed of multiple lobules of large polygonal cells with prominent abundant granular eosinophilic cytoplasm. No cellular atypia was observed. Diagnosis of the adult type of rhabdomyoma was made by collective agreement. Rhabdomyoma can be mistaken for other reactive lesions and may resemble their appearance.

Keywords

head and neck neoplasms maxillary vestibule oral cavity rhabdomyoma

Introduction

Rhabdomyomas (RMs) are rare and benign mesenchymal neoplasms of striated muscle with different degrees of mutation and differentiation, which are divided into intracardiac and extracardiac varieties. The intracardiac lesion is regarded as a hamartoma affecting children and infants and is correlated with tuberous sclerosis.¹ Extracardiac RM is considered as a tumor that shows a predilection for the head and neck region and affects patients of all ages from 5 weeks to 61 years of age. Based on histopathology, extracardiac RM is categorized into fetal, genital, and adult types, depending on the degree of differentiation.² The fetal type is found mainly in children and is divided into the juvenile and classic subtypes. The genital type manifests as a polypoid mass on the vulva and vagina and commonly affects young women.³ Head and neck region accounts for 90% of adult extracardiac rhabdomyoma (AERM) cases. As a result, it is crucial to consider these rare tumors as a differential diagnosis in this area. Especially, AERM shows a predilection for anatomic sites of the head and neck, including palate, pharynx, larynx, soft tissues of the neck, sublingual, and submandibular regions.² Predominantly, it affects the male gender in the age range of 55 to 60 years old and can happen as a solitary, multinodular, or multifocal lesion.⁴ Clinically, AERM appears as an asymptomatic, slow-growing, well-defined, homogeneous, and mobile submucosal nodule. Besides, several signs and symptoms, such as tongue displacement, dysphagia, dyspnea, and facial asymmetry, might be observed when it occurs in the mouth or pharynx. The clinical differential diagnoses of AERM depend on the location and may include benign or malignant salivary gland tumors and benign mesenchymal neoplasms.⁵

According to the literature, the occurrence of an AERM mimicking a reactive lesion in the maxillary vestibule has yet to be reported. Therefore, this study aims to present our findings of this case with an appraisal of the related literature on the AERMs reported in the head and neck region since 2000.

Case Report

A 34-year-old male was referred to the Faculty of Dentistry, Department of Pathology, to assess an oral and yellow lesion in the maxillary vestibule. The patient was aware of the presence of this mass, but he did not have any complaints. Intraoral examination revealed an asymptomatic, painless, slow-growing, well-circumscribed, nontender, and solid nodule on the labial side of the maxillary vestibule. The lesion was recovered by intact and smooth mucosa (Figure 1). The clinical impression diagnosis was a lipoma because of the yellow hue of the lesion. Subsequently, the Department of Oral Medicine carried out an excisional biopsy. A mass of unencapsulated but well-defined soft tissue beneath the vestibular mucosa with minor bleeding was observed during the operation.

Figure 1.

A painless solid yellow nodule covered by intact mucosa was observed in the maxillary anterior vestibule.

A multilobulated nodule, 15 × 10 × 6 mm in size, with creamy white coloring and round margins, was described on gross examination. Microscopically, the lesion appeared as an encapsulated, well-circumscribed benign neoplasm composed of multiple lobules. The tumor comprised large polygonal cells with prominent abundant granular eosinophilic cytoplasm arranged in the fibrovascular stroma with mixed foci of eosinophils, polymorphs, macrophages, and lymphocytes. Eccentric and centrally placed, round, and small nuclei demonstrated finely dispersed chromatin and small nucleoli. Cross-striations in the cytoplasm were also observed, and there were no atypical mitosis and necrosis (Figure 2). From the immunohistochemical point of view, the tumor cells expressed muscle-specific actin and desmin; however, they were negative for S-100 protein and cytokeratin (Figure 3).

Figure 2.

(A) Hematoxylin and eosin (H&E) staining of the lesion in the low-power field of the section shows polygonal cells with granular eosinophilic cytoplasm. (B) The high-power field demonstrates cross-striations in the cytoplasm.

Figure 3.

Immunohistochemistry (IHC) staining of the tumor shows; (A, B) Positive staining for muscle-specific actin and desmin, and (C) negative for S100.

Due to the yellow color of the lesion, the initial clinical diagnosis was a lipoma. Although, RM can be mistaken for other reactive lesions and may mimic the appearance of many soft tissue lesions. The findings mentioned above addressed a diagnosis of adult-type RM.

The patient received clinical and imaging follow-ups, and he has been well with no evidence of recurrence after a year of follow-up.

Discussion

RMs are rare benign neoplasms derived from skeletal muscle cells, which are classified into extracardiac and intracardiac categories in terms of location. These tumors are the most prevalent primary cardiac tumor in infants. Intracardiac RM is believed to be a hamartoma that leads to diffused deformation of the heart muscle and is related to tuberous sclerosis in 50% to 80% of the cases. The incidence of intracardiac RM is higher than that of extracardiac type.⁶ Extracardiac RM predominantly affects the head and neck region and usually happens as a solitary and rarely as a multifocal lesion. The most widespread involved locations are the pharynx and oral cavity.⁷

AERM is a subtype of this category. It can grow slowly in the submucosa as a painless, smooth, round, well-defined, and polypoid nodule that steadily causes dyspnea, hoarseness, dysphagia, and other symptoms.⁷ Since the AERM origins are the muscles of the third and fourth branchial arches, it has a remarkable tendency to the head and neck area.² Generally, it affects the head and neck region, followed by the extremities, esophagus, stomach, and mediastinum, and shows a predilection for men over 50 years of age. The most common oral site of occurrence is the mouth floor and submandibular region, followed by the tongue base, soft palate, buccal mucosa, and lip.⁵ Its cytologic findings demonstrate large polygonal to oval cells with plentiful eosinophilic to granular cytoplasm, unclear cell borders, and round peripheral nuclei containing apparent nucleoli. Immunohistochemical analysis is helpful for AERM diagnosis, which shows positivity for muscle-specific actin, desmin, and myoglobin.⁸ Besides, in electron microscopy, actin and myosin filaments with Z-bands, ample mitochondria, and cytoplasmic glycogen can be detected.⁹ It is suggested that magnetic resonance imaging (MRI) is a preferable imaging method for diagnosing these tumors compared to computed tomography (CT) scans as it provides superior delimitation. MRI reveals a well-defined lesion that is isointense compared to the musculature on T1-weighted images and isointense or hyperintense on T2-weighted ones.²

The current study systematically reviewed the literature through PubMed, Scopus, Embase, and Web of Science databases. A search was conducted using the keywords “Rhabdomyoma” AND “Head and Neck Neoplasms” in these databases since 2000, which yielded 376 results. Of these, 67 articles were included, and others were excluded due to nonrelevancy. The present study is the 74th case of AERM in the head and neck region since 2000 (Table 1). According to these case reports, AERM has more commonly affected men (53 out of 74 cases), with a male:female ratio of 2.5:1. Predominantly, this tumor has appeared solitarily. Also, it has occurred as a multicentric or multifocal lesion only in 13 (17.5%) cases. Furthermore, the multilobulated, multilocular, and multinodular forms of this lesion were observed in 2 patients each. The most prevalent affected areas of AERM in the head and neck have been the larynx (20), the floor of the mouth and submandibular region (17), the base of the tongue (13), parapharyngeal area (7), and oropharynx (5). The chief complaint of the patients varied from dysphagia, hoarseness, and aspiration to globulus, burning sensation, and painless swelling. In addition, the preferred treatment for AERM includes surgical resection, tumor removal, and excisional biopsy (Table 1). The recurrence of this lesion after primary treatment has been reported only in 2 cases, by Koutsimpelas et al. in 2008 and Schlittenbauer et al. in 2013.^10,11

Table 1.

Literature Review of Adult-Type Extracardiac Rhabdomyoma in the Head and Neck Since 2000.

Authors	Year	Gender	Age	Location	Chief complaint	Treatment	Follow-up
Cuesta Gil et al.¹²	2000	M	56	Base of the tongue	Sleep apnea and dysphagia	Surgical excision through a tongue split	No recurrence
Liang et al.¹³	2000	M	39	Paraglottic space	Dysphagia, aspiration, and weight loss	Surgical resection	No recurrence
Verdolini et al.¹⁴	2000	F	66	Lip	NA	NA	NA
Verdolini et al.¹⁴	2000	F	60	Eyelid	NA	NA	NA
Orrit et al.¹⁵	2000	M	66	Arytenoid muscle	Hoarseness and dysphagia	External removal	No recurrence at 4 months
Welzel et al.¹⁶	2001	F	77	Parapharyngeal and paratracheal space (multilocular)	NA	Complete resection	No recurrence immediate post-op, long-term recurrence NA
Sánchez Jiménez et al.¹⁷	2001	F	56	Cervical mucosa and temporal region	NA	NA	Low recurrence
Morinière et al.¹⁸	2001	M	63	Base of the tongue	NA	NA	NA
McGregor et al.¹⁹	2003	M	78	Floor of the mouth	Speech and swallowing disturbance	Removal by cervical neck incision	NA
Favia et al.²⁰	2003	M	58	Floor of the mouth	NA	Conservative surgery (simple transoral ablation)	No recurrence at 12 years
Favia et al.²⁰	2003	F	63	Submandibular region and SCM muscle	NA	Simple enucleation of the tumor	No recurrence at 14 years
Fukuda et al.²¹	2003	M	51	Base of the tongue	Dysphagia	Tumor removal and tracheotomy	No recurrence at 1 year
Delides et al.²²	2005	M	59	Tongue (multifocal)	NA	Surgical incision on the tongue	No recurrence at 1 year
Brys et al.²³	2005	M	79	Vocal cords	Progressive hoarseness	External excision	Resolution of hoarseness at 6 weeks
Padilla Parrado et al.²⁴	2005	F	69	Parapharyngeal space (multifocal)	NA	NA	NA
Hansen et al.²⁵	2005	M	73	Jugular region of neck	Rapid mass enlargement	Surgical resection	NA
Liess et al.²⁶	2005	M	69	Epiglottis, submandibular gland and hyoid bone (multifocal)	Asymptomatic neck mass	Complete surgical excision	NA
Gajda et al.²⁷	2005	F	61	Parapharyngeal and paratracheal space (multifocal)	NA	Complete excision	Low recurrence
Bellis et al.²⁸	2006	M	62	Submandibular region	Sudden hoarseness and difficulty breathing and swallowing	Complete local excision	No recurrence at 6 years
Jensen et al.²⁹	2006	M	66	Vocal process of arytenoid muscle	Progressive dysphagia and vocal coarseness	Complete surgical excision via suspension laryngoscopy	No recurrence at 18 months
Sørensen et al.³⁰	2006	M	65	Oropharynx	Change of sensibility in the tongue	An incision and blunt dissection	No recurrence at 1 year
Sørensen et al.³⁰	2006	F	66	Paratonsillar area	Burning sensation and enlarged tonsil	Hemimaxillectomy	No recurrence at 30 months
Dahm et al.³¹	2007	F	59	Thyroid cartilage and pyriform sinus	Progressive dysphagia	Partial thyroid rection and tumor removal	No recurrence at 6 months
De Medts et al.³²	2007	M	65	Floor of the mouth and base of the tongue	Dysphagia and globus sensation	Extirpation of submandibular and sublingual glands and tumor	No recurrence at 6 months
Pichi et al.³³	2008	M	56	Nasopharynx, pyriform sinus, aryepiglottic fold, and epiglottis	Progressive dysphagia	Tracheotomy, parotidectomy, and resection	No recurrence
Koutsimpelas et al.¹⁰	2008	F	72	Aryepiglottic fold, between trachea and esophagus (multifocal)	Globulus and hoarseness	Surgical resection, microsurgical excision	Recurrence at 2 months, no recurrence afterwards
Grosheva et al.³⁴	2008	M	45	Posterior wall of the pharynx at the level of thyroid cartilage (multilocular)	Dysphagia and foreign body sensation	Complete extirpation of the tumor	No recurrence
Bizon et al.³⁵	2008	M	65	Base of tongue, submandibular region, and pyriform sinus (multifocal)	NA	Excisional biopsy	No recurrence
Viscasillas et al.³⁶	2008	F	50	Glossoepiglotic fold to vocal cord	Dysphagia	Complete resection by CO₂ laser	No recurrence at 10 months
Zacharia et al.³⁷	2008	M	70	Floor of the mouth, base of the tongue, and oropharynx (multiple)	Oral cavity discomfort and fullness	No treatment	NA
Jin et al.³⁸	2009	F	57	Parapharyngeal region	Airway obstruction and stridor	Pharyngectomy for tumor resection	NA
Juhlin et al.³⁹	2009	M	60	Below the platysma and above the vascular sheath in the neck	Swelling on the neck	Local excision	No recurrence
Juhlin et al.³⁹	2009	M	65	Floor of the mouth and submandibular gland	Swelling on the neck and tenderness	Local excision	No recurrence
Karaman et al.⁴⁰	2009	M	52	Tonsillar fossa and base of the tongue	Abnormal breathing and swallowing	Transoral simple ablation of tumor	No recurrence at 18 months
Parara et al.⁴¹	2010	M	69	Floor of the mouth and submandibular region (multifocal)	Painless lump	Surgical excision	No recurrence at 2 years
Catalfamo et al.⁴²	2010	M	62	Floor of the mouth and submandibular gland	A large mass of the neck	Surgical excision	No recurrence at 4 years
Zhang et al.⁹	2010	M	64	Parapharyngeal space	Gurgling and difficulty swallowing	NA	NA
Gupta et al.⁴³	2010	M	43	Floor of the mouth	Swelling	NA	NA
Gupta et al.⁴³	2010	M	37	Bilateral submandibular region	Swelling	NA	No recurrence at 2 months
Authors	Year	Gender	Age	Location	Chief complaint	Treatment	Follow-up
Etit et al.⁴⁴	2011	M	67	Base of the tongue and parapharyngeal region	NA	NA	NA
Papaspyrou et al.⁴⁵	2011	M	71	Parapharyngeal region	Dyspnea and stridor	Surgical excision via an open transcervical approach	No recurrence at 1 year
Papaspyrou et al.⁴⁵	2011	M	68	Oropharynx, base of the tongue, and epiglottis	Sensation of foreign body in throat, dysphagia, and tendency to aspiration	Surgical removal of the mass	No recurrence at 10 months
Maglio et al.⁴⁶	2012	M	70	Submandibular region	NA	Surgical excision via an open transcervical approach	NA
Huang et al.⁴⁷	2012	F	4	Ventral side of the tongue	Disturbed speaking and swallowing	Conservative excision	No recurrence at 1 year
Polanec et al.⁴⁸	2012	M	70	Nasopharynx	Hoarseness and difficulty swallowing	Surgical resection	No recurrence at 6 months
Vera-Sirera et al.⁴⁹	2012	M	54	Floor of the mouth	No complaint	Intraoral incision and removal	No recurrence at 3 years
Willrich et al.⁵⁰	2012	M	61	Floor of the mouth and submandibular gland	NA	Surgical exeresis	NA
Zhang et al.⁵¹	2012	M	78	Floor of the mouth (multifocal and multinodular)	Progressive dysphagia and respiratory distress	Tracheostomy, dissection, and tumor removal	No recurrence at 2 years
Allevi et al.⁵²	2013	M	27	Floor of the mouth and tongue	Sleep apnea and stridor	Definitive surgical excision	No recurrence at 2 years
Amelia Souza et al.⁵³	2013	F	40	Floor of the mouth and supraglottic region	Painless swelling	Local excision	No recurrence at 2 years
De Trey et al.⁵⁴	2013	M	55	Parapharyngeal region, inferior to the submandibular gland (multifocal)	Hoarseness and dysphagia	Surgical removal via submandibular and vocal cords’ incision	NA
Koudounarakis et al.⁵⁵	2013	M	62	Pyriform sinus	Dysphagia	Transoral excision by CO₂ laser	No recurrence at 1 year
Pinho et al.⁵⁶	2013	M	35	Supraglottic region of larynx	Progressive hoarseness	Endoscopic extirpation	No recurrence at 15 months
Schlittenbauer et al.¹¹	2013	M	38	Inner cheek and buccal region	Painless swelling	Complete removal	Local recurrence at 6 months, no recurrence at the following 2 months
Cain et al.⁵⁷	2014	F	67	Supraglottis and pyriform sinus	Hoarseness and dyspnea	Open hemilaryngectomy	No recurrence at 16 months
Świeszewska et al.⁵⁸	2015	F	69	Sublingual region	Speaking and chewing difficulty	Total tumor excision	No recurrence at 4 years
Tresley et al.⁵⁹	2015	F	65	Laryngeal surface of epiglottis	Dyspepsia and heartburn	Excisional biopsy	NA
Carta et al.⁶⁰	2016	M	75	Vocal cord	Progressive dysphonia	Transoral CO₂ laser excision	No recurrence at 1 year
Mengoli et al.⁶¹	2016	M	48	Lower lip	NA	Surgical excision	NA
Authors	Year	Gender	Age	Location	Chief Complaint	Treatment	Follow-up
Altissimi et al.⁷	2017	F	85	Arytenoid muscle and pyriform sinus	Dysphagia, dysphonia, and breathing difficulty	Tracheostomy and complete endoscopic removal	No recurrence at 18 months
Dermawan et al.⁸	2017	M	57	Nasopharynx and oropharynx	Weight loss, hematuria, dysphagia, and airway encroachment	Surgical resection	NA
Lu et al.⁶²	2017	M	72	Base of the tongue and submandibular space (multifocal)	Progressive swelling, snoring, and dysphagia	Surgical resection through transcervical approach	No recurrence at 5 years
Mistry et al.⁶³	2017	M	60	Bilateral submandibular and sublingual spaces, floor of the mouth	Painless swelling	Submandibular incision	NA
Andrade et al.⁶⁴	2018	F	11	Lateral border of the tongue	Dysphagia and globus sensation	Surgical excision	No recurrence at 2 years
Cameron et al.³	2019	M	45	Inferior belly of the omohyoid muscle	Neck tenderness and swelling	Complete surgical excision	No recurrence
Dau et al.⁶	2019	M	64	Soft palate (multifocal)	Inability to swallow food and constant urge to clear the throat	Complete surgical excision	No recurrence at 3 years
Yadav et al.⁶⁵	2019	F	55	Floor of the mouth and submandibular region (multinodular)	Painless swelling	Surgical excision	NA
Stein et al.⁶⁶	2020	M	66	Pyriform sinus, paraglottic space, and thyrohyoid space	Dysphagia, dysphonia, dyspnea, weight loss, and shortness of breath	NA	NA
Münch et al.⁶⁷	2020	M	68	Base of the tongue, tonsillar fossa, and oropharynx	Snoring, apneas, and excessive daytime sleepiness	Surgical excision by transoral robotic surgery	No recurrence at 1 month
Hakim et al.⁴	2020	M	56	Floor of the mouth and submandibular region	Painless swelling	Complete excision and transoral resection	No recurrence at 1 year
Da Silva Leonel et al.⁵	2021	M	47	Floor of the mouth	Dysphagia and discomfort during the speech	Surgical excision	No recurrence at 4 years
Elmandouh et al.²	2022	F	66	Sublingual gland, submandibular space, thyroid region, and tongue (multifocal)	Neck mass	Imaging follow-up	NA
Salameh et al.¹	2022	M	57	Base of the tongue (multilobulated)	Dysphagia, dysphonia, and stertor	Complete surgical excision	No recurrence at 2 years
Present case	2023	M	34	Labial maxillary vestibule (multilobulated)	No complaint	Excisional biopsy	No recurrence at 1 year

Abbreviations: F, female; M, male; NA, not available; SCM, sternocleidomastoid muscle.

The possible pathogenesis of AERM is still disputable. Several studies have considered this type of RM as a hamartomatous, nonneoplastic, or reactive lesion due to its slow growth and various histological views. On the other hand, recent articles suggest that it may have a true neoplastic nature because of clonal chromosomal aberrations.¹¹ The differential diagnosis of AERM comprises paraganglioma, granular cell tumor, oncocytoma, acinic cell carcinoma, rhabdomyosarcoma, metastatic renal cell carcinoma, and normal skeletal muscle.⁸ The diagnosis of RM is usually established only after the removal of the tumor since distinctive clinical and radiologic features suggestive of the true nature are generally lacking.⁶⁸ The accepted treatment for AERM is surgical excision with preservation of the surrounding tissue. Although malignant degeneration has not been reported, the local recurrence rate is 42%, mainly because of incomplete resection. It indicates that long-term follow-up is important.¹⁹

According to the available literature, 34 cases of oral adult-type RM has been reported in the present century, and the submandibular region and floor of the mouth (50.0%), followed by the base of the tongue (38.2%) have been the most frequent sites of occurrence (Table 1). The last case of oral AERM, before the current study, was reported by Salameh et al.¹ They described a 57-year-old man with complaints of dysphonia, dysphagia, and stertor whose head and neck endoscopy revealed a smooth mass located at the tongue base. CT scan exhibited a significant lobulated lesion measured 7 × 5 × 6 cm in size, and immunohistochemically, the desmin markers were positive. Finally, the patient underwent complete surgical excision, and the tumor had not recurred in the last year.¹ Mengoli et al. reported that an AERM affected a 48-year-old man located on the lip in 2016. The cuneiform vermilionectomy was conducted with a clinical diagnosis of oral squamous cell carcinoma. Histological findings demonstrated a nodule containing uniform large polygonal cells with peripheral or central nuclei and eosinophilic and granular cytoplasm, forming an unencapsulated and well-delimited tumor below the keratinized epidermis. The immunohistochemical assessment showed positive results for desmin and muscle-specific actin; however, the tumor cells did not express cytokeratin and S-100 protein.⁶¹ Exclusively, Schlittenbauer et al. disclosed the recurrence of oral AERM following the first-stage treatment in 2013. This study described a 38-year-old man complaining of a long-lasting, painless swelling in the inner cheek. The initial diagnosis was mucocele based on the clinical features. Initially, this solid and well-defined mass was removed by surgical excision. However, the local recurrence of this lesion in the perimandibular area was observed 6 months later. Subsequently, the tumor was excised through a transoral approach. No recurrence was observed.

In this article, we presented a rare case of an intraoral adult-type extracardiac RM, a benign mesenchymal tumor resembling a reactive lesion clinically, and conducted a systematic review of the related literature. In summary, AERM is a rare lesion with a predilection for middle-aged men in the head and neck region. Complete surgical excision is the most recommended treatment for this tumor, resulting in a relatively low recurrence rate. As a clinician, it is always noteworthy to consider that reactive lesions are relatively common in the oral cavity, and both benign and malignant tumors can mimic their clinical presentation of them. As a result, RM can be mistaken for other reactive lesions and may imitate the appearance of many soft tissue lesions. To perform a definitive diagnosis, histopathological analysis is necessary. This study highlights the importance of precise diagnosis that affects the step-by-step treatment related to adult-type RM resembling reactive lesions.

Footnotes

Acknowledgements

The authors want to thank the Department of Medicine, Tehran University of Medical Sciences, for referring and handing over the clinical photos of the patient.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

Statement of Informed Consent

Written informed consent was obtained from the patient(s) for their anonymized information to be published in this article.

Statement of Human and Animal Rights

This article does not contain any studies with human or animal subjects.

Data Availability Statement

The data that support the findings of this study are available from the corresponding author, upon reasonable request.

ORCID iD

Seyyed Abolfazl Tabatabaie-Zadeh

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Rhabdomyoma of the Oral Cavity Mimicking Reactive Lesions: A Case Report and Review of the Literature

Abstract

Keywords

Introduction

Case Report

Discussion

Footnotes

Acknowledgements

Declaration of Conflicting Interests

Funding

Statement of Informed Consent

Statement of Human and Animal Rights

Data Availability Statement

ORCID iD

References