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17 October 2023: Articles  Saudi Arabia

Rare Oral Hemangioma in Pregnancy: A Case Series Providing Clinical Insight into Patient Care

Challenging differential diagnosis, Unusual setting of medical care, Rare coexistence of disease or pathology

Abdullah Saleh Alsheikh1AEF*, Sami Alharethy1A, Dhaifallah Mulafikh1AF, Ahmed Naif Alolaywi2EF, Yara Ibrahim Alhamad3EF, Modi Atig Alamer3EF

DOI: 10.12659/AJCR.939821

Am J Case Rep 2023; 24:e939821

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Abstract

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BACKGROUND: Hemangiomas are defined as benign soft tissue vascular tumors that are histologically classified as capillary, cavernous, or mixed types. Hemangiomas can also be described based on clinical appearance as superficial, mixed, or deep lesions. Following a thorough search, only 3 case reports of superficial protruding lip mass were found in the literature. Other cases of tongue hemangioma have been reported in infants or young toddlers, and only rarely in adults.

CASE REPORT: The first case was a 43-year-old pregnant woman, with an unremarkable medical and surgical history, in the second trimester who presented to the Otolaryngology Clinic with a chief concern of a progressively growing lesion, measuring 0.7×0.5 cm, over the lateral right side of the tongue for the last 2 weeks after accidentally biting her tongue during dinner. The second case was a 26-year-old woman with unremarkable medical and surgical history who presented to our Otolaryngology Clinic with a chief concern of a non-painful soft fungating pink-red lip lesion 1.5×1 cm across the right lower lip growing for the last 4 months. This lesion appeared during the third trimester of pregnancy following a lip injury that was described as minor trauma.

CONCLUSIONS: Although hemangiomas can occur anywhere on the body, they are most commonly found in the head and neck. These lesions are usually recognized quickly by patients and treating physicians and are thus clinically diagnosed. Most vascular benign lesions regress on their own, but if detected early, they are surgically excised for cosmetic and functional reasons.

Keywords: Hemangioma, Capillary, Hemangioma, Cavernous, Pregnancy Trimester, Second, Pregnancy Trimester, Third, Tongue

Background

Hemangiomas are benign soft tissue vascular tumors that are histologically classified as capillary, cavernous, or mixed types. They are most commonly found on the skin and mucous membranes of the head and neck region [1]. Furthermore, hemangiomas can be classified as superficial, mixed, or deep, based on their clinical appearance. Despite the fact that growth, hormonal, and mechanical factors have been proposed to alter the abnormal proliferation of endothelial cells, the pathogenesis of hemangiomas remains unknown [2]. This article will shed light on capillary hemangioma by presenting 2 rare case reports in 2 different pregnant patients, one with a superficial lesion on the lower lip and the other with a hemangioma at the tip of the tongue.

Case Reports

CASE 1:

A 26-year-old vitally stable healthy woman with an unre-markable medical and surgical history presented to our Otolaryngology Clinic with concerns of a pink-red lower lip lesion that had been present for 4 months. The lesion was 1.5×1 cm across the right lower lip and presented as a non-painful soft superficial fragile lesion (Figure 1).

According to the patient, this lesion appeared during the third trimester of pregnancy following a lip injury that was described as minor trauma. The lesion grew slowly in size, and the patient reported minor bleeding from it on occasion. There were no additional symptoms. The ear, nose, and throat examination was routine. At the time, the most common differential diagnoses were traumatic fibroma and pyogenic granuloma. The decision was made to excise the lesion completely under local anesthesia in a well-equipped clinic, including diathermy in the event of bleeding. The lesion was excised completely and sutured with 5-0 vicryl rapide. The patient was given oral and topical antibiotics, with a follow-up appointment in a week (Figures 2, 3).

The pathology report revealed as capillary hemangioma, making it a rare presentation. One week after the excision, the patient was seen with a healing wound, apart from a mild crust for which lubricants were prescribed.

CASE 2:

A 43-year-old vitally stable, healthy pregnant woman in her second trimester presented to the Otolaryngology Clinic with concerns of a progressively growing lesion on the tip of her tongue for the previous 2 weeks. The patient claims she became aware of the growing lesion only after accidentally biting her tongue during dinner time. According to the patient, there was no previous history of similar lesions. There was no history of any other related symptoms.

During physical examination, she was found to be vitally stable, with a red fungated lesion measuring 0.7×0.5 cm over the lateral right sided of the tongue and no other lesions on the head and neck area (Figure 4).

A monopolar excisional biopsy was performed, and the wound was left open for secondary intention healing, and the patient was given oral analgesia and antibiotics. After 2 weeks of the procedure, she presented to the clinic with a completely healed wound (Figure 5).

The pathology report revealed an ulcerated capillary hemangioma. She had a follow-up in our clinic 6 months later and showed no signs of local recurrence.

Discussion

Capillary hemangioma is a type of hamartoma that is distinguished by vascular growth and endothelial cell proliferation. According to the literature, trauma can be linked to the development of some hemangiomas. Capillary hemangioma is difficult to diagnose because it is a rare presentation that frequently mimics pyogenic granuloma. Hemangioma prevalence can occur in childhood or later in life, with a female to male ratio of 3: 1 [3]. During our review of the literature, we discovered only 3 cases of capillary hemangioma as a superficial lip mass [3–5]. There were 2 male patients and 1 female patient among these cases. The lesion sites were the upper lip in 2 cases and the lower lip in 1 case. Two of patients in these cases were adults, and 1 was an adolescent. All patients had easily bleeding lesions in common. In addition, 1 patient, like ours, had a history of trauma. In none of the cases was radiological investigation used; instead, differential diagnosis was made based on clinical presentation. Capillary hemangioma appears in histopathology as capillaries with thin walls and little stroma. Hemangioma appears as unencapsulated red friable lesions measuring from a single millimeter to several centimeters in gross appearance. Treatment in our case was similar to that of the other cases reported in the literature, including complete surgical excision. During the follow-up periods for the cases, which were between 6 and 18 months, no recurrence of the lesion was documented.

Pyogenic granuloma are defined as rapidly growing pedunculated lobular capillary hemangiomas found most commonly on skin, gingiva, and oral mucosa. Although pyogenic granuloma demonstrates immunocytochemical and ultrastructural differences, it is difficult to differentiate it from a hemangioma clinically [6]. Capillary hemangioma frequently mimics pyogenic granuloma histopathologically but without inflammatory features, as it progresses from a densely cellular proliferation of endothelial cells in the early stages to a lobular mass of well-formed capillaries in the mature phase [7]. Lingual hemangioma can present with a variety of clinical symptoms, ranging from asymptomatic to bleeding hemangiomas. Only a few cases of tongue hemangioma were discovered during our literature review for lingual hemangioma. Among these cases, the first reported hemangioma of the base of the tongue was in a male patient with a history of chronic irritation to the oral cavity with symptomatic presentation and an uncomplicated course [8]. The second case was a 20-year-old pregnant woman diagnosed with cavernous hemangioma in 3 areas: her tongue, lips, and neck, with active bleeding that was managed with tracheostomy at first, then arterial embolization and surgical excision [9].

Treatment options include surgery, laser therapy, sclerosing agents, and embolization [10]. Owing to the rapidly growing lesions causing cosmetic and functional concerns in both of our cases, surgical excision, the criterion standard treatment, was performed. In the first case, the lesion was completely excised and sutured with 5-0 vicryl rapide under local anesthesia in a well-equipped clinic, including diathermy in the event of bleeding. In the second case, a monopolar excisional biopsy was performed, and the wound was left open for secondary intention healing. The relationship between hemangioma and pregnancy is not fully understood. However, research suggests that a high level of estrogen in pregnancy may play a potential role in mediating vascular growth and hemangioma formation [11,12]. Both patients were followed up after their excisions and showed no signs of local recurrence. Using surgical excision in hemangioma can result in serious medical events, such as heavy bleeding. However, both patients were followed up after their excisions and showed no signs of local recurrence or bleeding.

Conclusions

Although hemangiomas can develop in any part of the body, the head and neck are most commonly affected. Because they are visible from the outside, these lesions are usually recognized quickly by patients and treating physicians and are thus clinically diagnosed. These vascular benign lesions usually regress on their own in most patients, but are surgically excised if they are detected early, for both cosmetic and functional reasons.

References:

1.. Kaplan I, Mass E, Littner M, A study of small superficial capillary hemangiomas on the lips in children: Pediatr Dent, 1998; 20(3); 188-91

2.. Marchuk DA, Pathogenesis of hemangioma: J Clin Invest, 2001; 107(6); 665-66

3.. Marler JJ, Mulliken JB, Current management of hemangiomas and vascular malformations: Clin Plast Surg, 2005; 32(1); 99-116

4.. Amminou L, El-Harti K, A capillary angioma of the lips: an extremely rare situation in the oral cavity: Int J Odontostomat, 2020; 14(3); 354-57

5.. Kashyap RR, Daniel AD, Holla VA, Dilemmatic presentation of hemangioma of the lip: A short case report: GMJ, 2020; 62(2); 131-32

6.. Rachappa MM, Triveni MN, Capillary hemangioma or pyogenic granuloma: A diagnostic dilemma: Contemp Clin Dent, 2010; 1(2); 119-22

7.. Desai V, Narang P, Varma B, Maghu S, Unusual site of capillary hemangioma: Practitioner’s dilemma!: CHRISMED J Health Res, 2015; 2(1); 77

8.. Qureshi SS, Chaukar DA, Pathak KA, Hemangioma of base of tongue: Indian J Cancer, 2004; 41(4); 181-83

9.. Vijay K, Shetty AP, Rajasekaran S, Symptomatic vertebral hemangioma in pregnancy treated antepartum. A case report with review of literature: Eur Spine J, 2008; 17(S2); 299-303

10.. Jananni M, Gubernath U, Mahendra J, Sivaramakrishnan M, Capillary hemangioma of gingiva mimicking as pyogenic granuloma: Report of two cases: J Interdiscip Dent, 2012; 2; 218-20

11.. Sun ZY, Yang L, Yi CG, Possibilities and potential roles of estrogen in the pathogenesis of proliferation hemangiomas formation: Med Hypotheses, 2008; 71(2); 286-92

12.. Xiao X, Hong L, Sheng M, Promoting effect of estrogen on the proliferation of hemangioma vascular endothelial cells in vitro: J Pediatr Surg, 1999; 34(11); 1603-5

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923