March 31, 2025

J Diagn Treat Oral Maxillofac Pathol 2025; 9: 100303.

DOI: 10.23999/j.dtomp.2025.3.100303

Under a Creative Commons license

HOW TO CITE THIS ARTICLE

Cherniak OS, Demidov VH, Blyzniuk VP, Zaritska VI, Snisarevskyi PP. Cine loops in ultrasonographic verification of acquired lymphatic malformation (lymphangioma) of the neck associated with musical instrument (violin) play: A case report. J Diagn Treat Oral Maxillofac Pathol. 2025 Mar;9(3):100303. https://doi.org/10.23999/j.dtomp.2025.3.100303 

SUMMARY

Lymphatic malformation (LM) is a rare pathology that is predominantly congenital. However, an increasing number of published cases clearly demonstrate the possibility of developing acquired LM, mainly because of trauma. Causes of injury can include dental restorations in the oral cavity, repetitive surgeries, and even chronic trauma from playing musical instruments. This article presents the case of a 32-year-old female with an acquired lymphatic neoplasm of the neck that developed because of many years of violin playing. Clinical preoperative photography, gray scale and color Doppler ultrasonography data, two ultrasound videos (cine loops), a specimen of the excised mass, and histopathological images are presented. Thus, this paper confirms the possibility of developing acquired LM in the neck region. The fact that the LM developed to its current size by the patient's 32nd year of life, in addition to the patient’s musical history, confirms that the development and growth of this acquired LM required time, namely years of playing a violin. Also, two ultrasound videos clearly demonstrate the capabilities of this dynamic examination for verifying this rare pathology. Ultrasound videos show the ability of this lymphatic neoplasm to compress and reduce in size by 2 times. Since LM is a term that refers to a developmental defect of the lymphatic system, we suggest using the term "lymphangioma" for acquired lymphatic formation (i.e., lymphatic neoplasm).

KEY WORDS

Lymphatic malformation, lymphangioma, lymphatic neoplasm, neck, ultrasonography, gray-scale ultrasound, color Doppler ultrasound, cine loop in ultrasonography, violin

INTRODUCTION

Specialists of various profiles may be confused about which diagnosis is correct to use as of 2025 when describing a lymphatic mass of the head and neck area¾lymphatic malformation (LM) [1-3] or lymphangioma [4-6]. Both terms are found in modern literature, but the second term is increasingly being abandoned [7]. Most sources state that the LM is a rare, non-malignant mass consisting of fluid-filled channels or spaces thought to be caused by the abnormal development of the lymphatic system [7]. Although numerous authors describe LM as a benign lesion, Yu et al. (2022) have proven that the unusual process of LM gradual malignant transformation into lymphangiosarcoma is also possible [8]. Also, as some authors describe LM as a congenital pathology [1, 5, 7, 9], others provide evidence that it may be an acquired factors for the LM appearance [10, 11]. Direct confirmation of the data on a possible variant of acquired LM of the neck is the data of the patient presented in this article. 

 

The aims of this article are (1) to publish the world's first ultrasound videos of the spongy structure and ease compressibility of an acquired LM of the neck associated with many years of playing the violin, (2) to expand diagnostic options of oral and maxillofacial surgeons.

CASE REPORT

On June 25, 2014, a 32-year-old Caucasian female was referred to the Center of Maxillofacial Surgery and Dentistry with complaints of a tumor on her neck (Fig 1) that was protruding and creating a cosmetic problem. Clinically, the neoplasm was painless and easily compressible upon palpation, the overlying skin was normal. According to the patient and her relatives, this mass has become noticeable over the past few years. The key information in the medical history is that the patient has been playing the violin for many years. Since many sources publish that musicians may have occupational diseases associated with playing a certain type of musical instrument [12-14].

Ultrasonography (USG) was performed using HD11 XE model of ultrasound machine (Philips Healthcare, Eindhoven, The Netherlands) with 12-3 MHz linear probe (synonym: linear transducer). USG showed well-circumscribed heterogeneous hypoechoic lesion measured 3.09 cm x 1.03 cm (Fig 2) and which was located slightly above the middle third of the neck between the midline of the neck and the anterior margin of the left sternocleidomastoid muscle.

The formation had cystic cavities of various sizes from medium to very small. Cystic areas visualized as different size anechoic areas. An important feature of this formation was its ability to almost completely compress.

 

Supplemental Video Content 1 (cine loop) demonstrates the degree of compression of the malformation by the ultrasound transducer when performing gray-scale USG. Screenshots from cine loop demonstrate LM in three states: non-compressed, moderately compressed, and completely compressed. Video is available in the page of the full-text article on dtjournal.org and in the YouTube channel, available at https://youtu.be/1A30SbP0Tsg. Total video’s duration is 33 seconds.

 

Lesion had no arterial or venous blood flow upon color Doppler USG (Fig 3). The formation was adjacent to such a muscle as omohyoid and was located close to the anterior margin of the sternocleidomastoid muscle.

Supplemental Video Content 2 (cine loop) demonstrates the degree of compression of the malformation by the ultrasound transducer when performing color Doppler USG. Video is available in the page of the full-text article on dtjournal.org and in the YouTube channel, available at https://youtu.be/bliMTnSMCs0. Total video’s duration is 27 seconds.

 

The operation was performed under general anesthesia and the mass was excised within healthy tissues. At the lancing of the excised mass, was noted its spongy structure (Fig 4) and the fact that the entire lesion was filled with gray fluid (i.e., lymph).

Histopathology (Fig 5) was performed by two experienced doctors-pathologists, V.I.Z. (26 years of experience) and P.P.S (21 years of experience). It showed the lymph in the lumens of the lymphatic vessels and multiple oval-shaped endothelial cells (visualized as small inclusions with dark blue color) in the inner layer of the lymphatic vessels. A histopathological diagnosis of lymphangioma (i.e., LM) was made.

The patient continued to be recurrence-free at her 3-year follow-up.

DISCUSSION

Lymphangiomas belong to the category of vascular malformations in the Mulliken and Glowacki classification system and are dilated endothelial-lined lymphatic spaces (Yuen and Ahuja, 2012) [15]. The authors classify all types of lymphangiomas as congenital LMs (Gilony et al., 2012; and Kim, 2014) [16, 17]. A characteristic feature of LM is its compressibility with changes in the shape and configuration of the lesion [6]. Fluctuation can be detected during palpation of cystic cavities, and a puncture can yield a clear liquid, sometimes cloudy, often with an admixture of blood [6].

 

The neck area is one in which there can potentially be noted a wide variety of neoplasms and malformations. Differential diagnosis of LM of the neck is carried out with hemolymphangioma [6, 18], arteriovenous malformation [15, 19], branchial cleft and thyroglossal duct cysts [15, 20], cystic squamous cell carcinoma [20], metastatic nodes [15], nerve sheath tumor [15], etc. USG, as a non-invasive diagnostic method, allows an early diagnosis to be made with fairly high accuracy [15, 21, 22]. Johnson et al. (2020) are more than right when they say that the use of USG has gained momentum because of its accessibility, cost efficiency, quicker collection time [23], and safety. The efficiency of ultrasonography in the detection of cystic and solid components in neoplasms is described in detail in numerous sources [15, 20]. So, USG was an excellent dynamic imaging tool to establish a preliminary diagnosis in the case we described. The described ultrasound characteristics of LM in our patient corresponded to the classic manifestations of LM with cystic cavities of the mixed type [15]. However, according to our knowledge, these are the first two published videos (cine loops) of the possibility of full compression of LM of the neck. Moreover, according to our literature search, this is the first thoroughly described acquired LM caused by prolonged (over many years) violin playing.

 

Left-sided “submandibular” lesions develop in 62% of violinists and violists from holding their instrument (Blum and Ritter, 1990; Ostwald et al. 1994) [12, 13].

 

According to Blum and Ritter (1990), in four cases of their observations of the viola and violin players the “tumor” revealed itself not just an induration but as a sialolithiasis of the submandibular gland, and in one case as a cystadenolymphoma of the parotid gland (i.e., Warthin’s tumor) [12]. Therefore, we believe that in this article it is worth paying attention to the ultrasound picture of cystadenolymphomas too [24].

 

Moraes and Antunes (2012) are emphasizing, the neck, shoulder, and temporomandibular joints are the most affected areas, due to prolonged flexion of the head and shoulder required to hold the violin [14]. Thus, our case not only confirms the statistics of the harmful effects of prolonged playing of certain instruments, but also is the first case of acquired LM described in detail in a patient who was professionally engaged in playing the violin.

 

Colbert et al. (2013) define LMs of the neck as a diverse group of lymphatic lesions [1]: They can be small and superficial or large and extensive, and management can be a challenge.

 

Cine loops in USG [23, 25] are an extremely useful method for dynamic diagnosis of the structure of malformations, tumors, and pathologic conditions. The two videos presented above clearly demonstrate this in both B-mode and color Doppler USG.

 

According to Gilony et al. (2012) and Kim (2014), for the microcystic and mixed types of LMs (which are also termed as lymphangiomas), surgery should be considered rather than sclerotherapy [16,17]. The same approach was used in our case, namely, the LM was excised within visually healthy tissues.

 

Treatment decision algorithms for stage I, II, III, IV, and V LMs are described in a study by Perkins et al. (2010) [26].

 

So, this article is additional confirmation of the emergence of playing-related (i.e., acquired) formations of the neck [12, 13]. And this is in addition to other playing-related musculoskeletal disorders [27]. Table 1 clearly demonstrates references to literary sources that highlight cases of both congenital and acquired LMs (i.e., lymphatic formations of lymphangiomas). Perhaps some authors would consider it appropriate to describe this pathological process in the patient as acquired lymphedema [31].

CONCLUSION

Thus, this article confirms the possibility of developing acquired LM in the neck region. The fact that the LM developed to its current size by the patient's 32nd year of life, in addition to the patient’s musical history, confirms that the development and growth of this acquired LM required time, namely years of playing a violin. Also, two ultrasound videos clearly demonstrate the capabilities of this dynamic examination for verifying this rare pathology. Since LM is a term that refers to a developmental defect of the lymphatic system, we suggest using the term "lymphangioma" for acquired lymphatic formation.

TERM OF CONSENT

Writing patient’s consent was obtained for publication the photos and data.

AUTHORS’ CONTRIBUTIONS

Drafting of the manuscript: Cherniak OS, Blyzniuk V. Critical revision of the manuscript: all authors. Approval of the final version of the manuscript: all authors.

CONFLICT OF INTERESTS

The authors declare no conflict of interest.

FUNDINGS

No funding was received for this study.

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ARTICLE TITLE IN UKRAINIAN

Кінопетлі в ультразвуковій верифікації набутої лімфатичної мальформації (лімфангіоми) шиї, пов’язаної з грою на музичному інструменті (скрипці): опис випадку

SUMMARY IN UKRAINIAN. SUMMARY IN UKRAINIAN (PDF).

Лімфатична мальформація (ЛМ) – це рідкісна патологія, переважно вроджена. Проте все більше опублікованих випадків чітко демонструють можливість розвитку набутої ЛМ, головним чином через травму. Причинами травми можуть виступати, як стоматологічні конструкції в порожнині рота, повторювані операції, так навіть і хронічна травма викликана грою на музичних інструментах. У цій статті представлено випадок 32-річної жінки з набутим лімфатичним новоутворенням шиї, яке розвинулося внаслідок багаторічної гри на скрипці. Наведено клінічні передопераційні фотографії, дані ультразвукової діагностики (УЗД) в B-режимі та режимі кольоровокго Доплерівського картування (КДК), два відео (кінопетлі) УЗД, видалену пухлину та патогістологічне зображення. Таким чином, дана робота підтверджує можливість розвитку набутої ЛМ в області шиї. Той факт, що ЛМ розвинувся до свого поточного розміру до 32-го року життя пацієнта, на додаток до музичної історії пацієнта, підтверджує, що розвиток і зростання цієї набутої ЛМ вимагав часу, а саме років гри на скрипці. Також два відео УЗД наочно демонструють можливості цього динамічного обстеження для верифікації цієї рідкісної патології. Відеопетлі УЗД показують здатність цього лімфатичного новоутворення стискатися і зменшуватися в розмірах в 2 рази. Оскільки ЛМ — це термін, який позначає ваду розвитку лімфатичної системи, ми пропонуємо використовувати саме для набутого лімфатичного утворення (тобто лімфатичного новоутворення) термін «лімфангіома».

KEY WORDS IN UKRAINIAN

Лімфатична мальформація, лімфангіома, лімфатичне новоутворення, шия, ультразвукове дослідження (УЗД), B-режим УЗД, кольорове Допплерівське картування (КДК), кінопетля в УЗД, кіноподібна петля в УЗД, скрипка