Laporan Kasus

Manifestasi Plunging Ranula dan Oral Ranula Pada HIV Positif Pasien:
Laporan Kasus

Herman Hambali 1
1
Staf Departemen Bedah Mulut dan Maksilofasial RS Immanuel, Bandung
ABSTRAK

Pendahuluan: Berdasarkan lokasi terjadinya ranula diklasifikasikan menjadi superficial atau lesi oral yang
dibatasi oleh dasar mulut diatas otot mylohyoid dan lesi dalam atau yang di kenal dengan cervical atau
plunging ranula.Kelainan kelenjar ludah sering digambarkan sebagai manifestasi oral dari suatu infeksi
HIV.Ranula merupakan salah satu manifestasi kelainan kelenjar ludah yang telah dilaporkan pada
beberapa laporan kasus pada pasien dengan infeksi HIV.
Laporan Kasus: Pria,37 tahun datang ke poliklinik dengan keluhan adanya benjolan di rahang bawah
kanan yang timbul sejak 4 bulan.Sejak 5 bulan terakhir pasien dalam terapi anti-retroviral(ART).
Berdasarkan pemeriksaan klinis terlihat suatu benjolan pada submandibular kanan dengan ukuran
5x3x3cm,lunak, berbatas jelas,dan tidak ada fluktuasi.Eksisi dilakukan dengan pembukaan ekstra oral
dalam anestesi umum. Hasil PA menunjukkan diagnosis ranula. Enam bulan paska pembedahan pasien
kembali dengan keluhan adanya benjolan pada dasar lidah sebelah kiri dengan ukuran 2x2x2cm,
berdasarkan pemeriksaan klinis terdapat gambaran ranula yang berasal dari kelenjar ludah sublingual kiri
dengan konsistensi lunak dan batas jelas. Terapi marsupialisasi dilakukan untuk penangan kasus
tersebut.Satu tahun paska marsupialisasi terlihat penyembuhan luka yang sudah baik, dan tidak terlihat
adanya tandanya rekurensi.
Simpulan: Ranula harus dipertimbangkan sebagai oral manifestasi pada infeksi HIV,selain itu ranula
dapat menjadi tanda awal gejala dan manifestasi dengan adanya infeksi HIV.

Kata Kunci: Ranula, Plunging Ranula, HIV, Eksisi, dan Marsupialisasi

 Case Report

Manifestation of Plunging Ranula and Oral Ranula in an HIV-Positive

Patient: Case Report

Herman Hambali 1
1
Staff Departement of Oral and Maxillofacial Surgery, Immanuel Hospital, Bandung

ABSTRACK
Introduction: Ranula are classified into superficial or oral lesion are confined to the floor of mouth above
the mylohyoid muscle and deep lesions also known as cervical or plunging ranula. HIV related salivary
gland diseases (HIV-SGD) is the most common oral manifestation of HIV infection. Ranula have been
reported in numerous studies as a symptom of HIV.
Case Reports: Male,37 years old came to policlinic with a painless lump at right latero-cervical region.
Patient with history of anti-retroviral therapy(ART) since 5months ago. According to clinical finding there
was localized soft tissue lump at right latero-cervical region with size 5x3x3cm, soft consistency, well-
defined border, and no fluctuation. Excision was performed under general anaesthesia. Histopathology
result support to ranula. Six-months post operatively, patient came back with the lump at floor of mouth
with size 2x2x2cm, well-defined border, and soft consistency, based on clinical findings the lump looks
like ranula from left sublingual gland. Accordingly, we performed marsupialization and patient was
followed up 1 year postoperatively with the good wound healing and there is no sign of recurrence.
Conclusion: Ranula has to be considered as the oral manifestation of HIV infection, moreover ranula can
be the initial symptoms or manifestation of HIV infection.

Keywords: ranula, plunging ranula, HIV, excision, and marsupialization

Introduction
 Head and neck pathology reported in more than 50% in human immunodeficiency virus (HIV)

positive patients and more than 80% in acquired immunodeficiency syndrome (AIDS) patients.(1) HIV-

related salivary gland diseases (HIV-SGD) is the most common oral manifestation of HIV infection.

Parotid salivary gland enlargement is the most reported form of HIV-SGD. 1, 2 However, ranulas, as types

of oral mucocele, have been reported in numerous studies as a symptom of HIV. 3-5

According to primary site of the lesion, ranula are classified into superficial or oral lesion are

confined to the floor of mouth above the mylohyoid muscle and deep lesions also known as cervical or

plunging ranula.6, 7 Specifically, ranula originates in the body of sublingual gland, in the duct of Rivinus of

the sublingual gland, and infrequently from minor salivary gland at this location.8

The treatment options of ranula is polarising topic, with conflicting evidence as to which treatment

modality is best due to the existing knowledge on the current concept of its aetiopathogenesis. A variety

of treatment procedures have been cited in the literature ranging from simple aspiration, sclerotherapy,

marsupialization, or the excision of the ranula and/or sublingual salivary gland, at times involving the
3, 5, 9, 10
submandibular salivary gland, with recurrence rate varies according to the procedure performed.

Here, we present the management of rare case of HIV positive patient with manifestation of plunging

ranula and oral ranula.

Case Report

A 37-years old male patient presented to Oral and Maxillofacial Department in Immanuel Hospital

with few months history of painless right latero-cervical region neck swelling. (Figure 1) During anamnesis

patient revealed that he has been taking antiretroviral therapy (ART) for 5 months. Initially, the patient had

experience a spontaneous increase of volume on the floor of the mouth about 3 months ago, on the right

region. After four months, intraoral swelling had subsided, and he eventually developed a latero-cervical

with similar characteristics.

On extraoral examination, a painless soft palpation swelling and normal overlying skin appeared

in right neck region with size 5x3x3 cm. (Figure 1) Intraorally, there is no lesion viewed. Head and neck

magnetic resonance imaging (MRI) demonstrated a well-defined lesion on right floor of mouth crossed the

mylohyoid muscle and submandibular gland was compressed by the lesion. (Figure 2) We choose
surgical excision of sublingual gland through extraoral approach under general anaesthesia. The

complete excision of the right sublingual gland was performed, and the specimen was submitted for

pathology examination, which confirmed the diagnosis of ranula. (Figure 3)

Four months after surgery, patient came back to our clinic with chief complaint of another swelling

on the floor of mouth. On intraoral examination, the patient had bilateral lesion on the floor of the mouth

which mainly on the left side of mouth floor, which was balloon-like, with fluctuant consistency, bluish, and

painless with size 2x2x2 cm. (Figure 4) Accordingly, we decided to perform marsupialization on the

lesion. One year postoperatively and regular follow-up visit showed a favourable result with neither

recurrence nor motor or sensitive tongue disturbances. (Figure 5)

Fig 1. The lesion at right latero-cervical region

Fig 2. Magnetic resonance imaging (MRI) findings indicated that the lesion crossed a mylohyoid muscle localized
within submandibular triangle, namely a plunging ranula
 A B

Fig 3. A. Macroscopic sublingual gland and capsule. B. Pathology analysis “Salivary gland showing cystic spaces a
few vascularity and inflammatory cell infiltrate”

Fig 4. Intraoral view with left sublingual swelling

A B

Fig 5. A. Postoperative intraoral image after 12 months B. Postoperative extraoral image after 12 months

Discussion

Salivary gland diseases are a common manifestation of HIV infection, with a significant increase
11-13
in prevalence over the last two decades. The exact prevalence of ranula in HIV population is not

known, but few studies has been documented the manifestation of ranula in HIV infection. 3-5 The prompt

identification is the key in diagnosis and treatment of both the salivary gland diseases including ranula

and HIV infection

Our patient had a history of taking ART for 5 months, and the swelling on right floor of mouth

since 3 months ago. Patient came to our clinic with complaint of swelling on right latero-cervical region
that clinically and radiographically examinations showed a plunging ranula. We regard this plunging

ranula is secondary to oral ranula, that results in the extravasation of saliva into floor of mouth and then

through the mylohyoid muscle due to the dehiscence of its fibers or through its posterior rim with the

invasion of the submandibular triangle.

After reviewing several cases and the literature,10, 14-16 we define the strategy for treatment of the

ranula. Firstly, the source of plunging ranula was most likely secondary from the oral ranula of sublingual

salivary gland, therefore ranula should be excised together with sublingual salivary gland to minimize the

recurrent of ranula. The access of the sublingual salivary gland excision was extraoral approach with the

consideration that the ranula was clinically located in right latero-cervical region and furthermore there

was no lesion appear intraorally. Intraoral approach in this case would make the procedure even more

difficult and longer, that should be avoided in HIV patient.

Several months after the first procedure, patient acquired the secondary ranula on the

contralateral site. In this secondary ranula, we decided to treat the ranula more conservatively with

marsupialization procedure. The rationale to opt the marsupialization in secondary procedure, because

the second ranula was considered in superficial location with small size. Therefore, we can minimize risks

the complications of surgery such as nerve injury, cyst recurrence, and cosmetic problems. In this very

rare case report, patient was suffered with bilateral ranula in the different time, we assumed HIV-SGD as

a predisposing factor for ranula. This increase may be due to extensive fibrosis in salivary gland, causing

blockage of the sublingual gland and hence, ranula.17 Furthermore, the increase of inflammatory cells was

found in salivary glands of Africans with untreated HIV 18 and we suggest that this could be the important

factor of predisposing ranula because more inflammation in the glands could lead to acinar rupture and

extravasation in sublingual glands.

Recent literature19 also explain the rising prevalence of HIV-SGD in the ART era, this aetiology

was hypothesized as an immune reconstitution inflammatory syndrome, whereas ART leading to

reconstitution of antigen-specific immune responses that lead to an “unmasking” of an underlying

opportunistic infection, resulting in SGD. However this statement of ART in SGD is still controversial,

some studies conversely revealed that ART has led to an overall decrease in oral complications
secondary to HIV infection.13, 20
Accordingly, further studies are necessary in order to conclude an

association between the ART and SGD especially ranula.

Conclusion

Although there are a variety of methods used to treat ranula, we believe that excision of ranula

with salivary gland is effective method with low recurrence to treat large lesion of ranula, however the

conservative treatment of marsupialization should be applied for the small lesion of ranula. There is no

reason to suspect that ranula patient with HIV status would not have a similar result as a patient without

HIV infection. Conversely, ranula has to be considered as the oral manifestation of HIV infection,

moreover ranula can be the initial symptoms or manifestation of HIV infection.

References

1. Waleed F M, Rebekah Y, Rafi K, Dukagjin M B, Rania A S, Jonathan G, et al. 25-Year Follow-up

of HIV-positive Patients with Benign Lymphoepithelial Cysts of the Parotid Glands: A Retrospective
Review. Anticancer Research. 2013;33(11):4927.
2. Meer S. Human immunodeficiency virus and salivary gland pathology: an update. Oral Surgery,
Oral Medicine, Oral Pathology and Oral Radiology. 2019;128(1):52-9.
3. Kusano Y, Ikeda R, Saito Y, Yamazaki M, Tateda Y, Kitaya S, et al. Treatment of oral ranula in
HIV-positive patient. Auris Nasus Larynx. 2021;48(1):171-4.
4. Kinshuck A, Schober M, Kokai G, Clarke R. Oral ranula in an HIV-positive patient: Case report
and literature review. BMJ case reports. 2012;2012.
5. Astigueta José Mariano MF, Benitez Javier Alejandro, Attaguile Alberto and Giannunzio Graciela
Ana. Plunging Ranula as Initial Manifestation of HIV-AIDS: A Case Report and Literature Review. J Oral
Dent Health. 2021;5(3): 69-73.
6. Slootweg PJ, El-Naggar AK. World Health Organization 4th edition of head and neck tumor
classification: insight into the consequential modifications.
7. Zhao YF, Jia Y Fau - Chen X-M, Chen Xm Fau - Zhang W-F, Zhang WF. Clinical review of 580
ranulas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004 Sep;98(3):281-7.
8. Kokong D, Iduh A, Chukwu I, Mugu J, Nuhu S, Augustine S. Ranula: Current Concept of
Pathophysiologic Basis and Surgical Management Options. World J Surg. 2017;41(6): 1476–1481.
9. Harrison JD. Modern management and pathophysiology of ranula: literature review. Head Neck.
2010;Oct;32(10):1310-20.
10. Yang Y, Hong K. Surgical results of the intraoral approach for plunging ranula. Acta Otolaryngol.
2014 Feb;134(2):201-5.
11. Burger-Calderon R, Ramsey KJ, Dolittle-Hall JM, Seaman WT, Jeffers-Francis LK, Tesfu D, et al.
Distinct BK polyomavirus non-coding control region (NCCR) variants in oral fluids of HIV- associated
Salivary Gland Disease patients. Virology. 2016;493:255-66.
12. Ebrahim S Fau - Singh B, Singh B Fau - Ramklass SS, Ramklass SS. HIV-associated salivary
gland enlargement: a clinical review. SADJ. 2014;Oct;69(9):400-3.
13. Syebele K, Munzhelele TI. The potential impact of highly active antiretroviral therapy on the
treatment and epidemiology of ranula in human immunodeficiency virus–positive patients. Oral Surgery,
Oral Medicine, Oral Pathology and Oral Radiology. 2013;116(1):e32-e6.
14. Choi MA-O. Case report of the management of the ranula. J Korean Assoc Oral Maxillofac Surg.
2019 Dec;45(6):357-363.
15. Syebele K, Munzhelele TI. The anatomical basis and rational for the transoral approach during
the surgical excision of the sublingual salivary gland for the management of plunging ranula. Am J
Otolaryngol. 2020;Mar-Apr;41(2):10237.
16. Torres Y, Brygo A, Ferri J. A 17-year surgical experience of the intraoral approach for ranulas. J
Stomatol Oral Maxillofac Surg. 2018;Jun;119(3):172-176.
17. McArthur CP, Africa Cw Fau - Castellani WJ, Castellani Wj Fau - Luangjamekorn NJ,
Luangjamekorn Nj Fau - McLaughlin M, McLaughlin M Fau - Subtil-DeOliveira A, Subtil-DeOliveira A Fau
- Cobb C, et al. Salivary gland disease in HIV/AIDS and primary Sjögren's syndrome: analysis of collagen
I distribution and histopathology in American and African patients. J Oral Pathol Med. 2003 Oct;32(9):544-
51.
18. Chidzonga MM, Rusakaniko S. Ranula: another HIV/AIDS associated oral lesion in Zimbabwe?
Oral Dis. 2004.
19. Nizamuddin I, Koulen P, McArthur CP. Contribution of HIV Infection, AIDS, and Antiretroviral
Therapy to Exocrine Pathogenesis in Salivary and Lacrimal Glands. International Journal of Molecular
Sciences. 2018;19(9):2747.
20. Jones J, Taylor BS, Tieu HV, Wilkin TJ. CROI 2017: Advances in Antiretroviral Therapy. Top
Antivir Med. 2017;May/Jun;25(2):51-67.