Intravenous lobular capillary haemangioma presenting as neck discomfort associated with neck anteflexion

  1. Kotaro Ikeda 1,
  2. Toshihisa Ichiba 2,
  3. Kazunori Seo 2 and
  4. Yuji Okazaki 3
  1. 1 Internal Medicine, Hiroshima City Hiroshima Citizens Hospital, Hiroshima, Japan
  2. 2 Emergency Medicine, Hiroshima City Hiroshima Citizens Hospital, Hiroshima, Japan
  3. 3 Internal Medicine, Akiota Hospital, Yamagata-gun, Hiroshima, Japan
  1. Correspondence to Dr Yuji Okazaki; ur8nibhc1@gmail.com

Publication history

Accepted:21 Dec 2020
First published:11 Jan 2021
Online issue publication:11 Jan 2021

Case reports

Case reports are not necessarily evidence-based in the same way that the other content on BMJ Best Practice is. They should not be relied on to guide clinical practice. Please check the date of publication.

Abstract

Lobular capillary haemangioma, also known as pyogenic granuloma, is a benign vascular tumour that usually originates in the skin and mucosal membrane. It sometimes derives from the lumen of a vein and the clinical presentations are various and non-specific. A 72-year-old woman complained of a sensation of pressure in her left neck for 1 month when cooking. Her left cephalic vein was enlarged with no signs of oedema, and cervical ultrasound revealed a space-occupying lesion in the left subclavian vein. Contrast-enhanced CT and MRI revealed an intravascular tumour. This tumour was removed with operation, and histopathological examination revealed intravascular capillary haemangioma. Intravascular lobular capillary haemangioma is a rare condition that occurs in the veins of the neck and upper extremities. Intravascular tumours could cause a unique symptom, such as neck discomfort associated with neck anteflexion.

Background

Lobular capillary haemangioma, a benign tumour that usually originates in the skin and mucosal membrane, usually presents as a subcutaneous solitary nodule.1 It sometimes derives from the lumen of a vein and the clinical presentations are various and non-specific. Oedema of the extremities is a common presentation in patients with symptomatic intravenous lobular capillary haemangioma.2 We present a case of intravascular lobular capillary haemangioma presenting as neck discomfort with neck anteflexion without oedema of the extremities. Neck discomfort with change in posture could be caused by not only orthopaedic lesions but also intravenous abnormalities.

Case presentation

A 72-year-old woman complained of a sensation of pressure in her left neck for 1 month when cooking. She felt neck discomfort especially during neck anteflexion. The symptom developed in 1 month. She had never experienced pain, syncope and left arm paralysis. Her medical history was unremarkable. Physical examination revealed blood pressure of 128/65 mm Hg, heart rate of 90 beats per minute and oxygen saturation of 97%. Her neck and internal jugular vein appeared normal; however, her left cephalic vein was enlarged with no signs of oedema (figure 1). Cervical ultrasound revealed a space-occupying lesion in the left subclavian vein. Although we made an attempt to elicit Doppler signal from the lesion, we detected no Doppler signal (figure 2). We examined the lesion with the patient in two different postural situations, viz without symptoms when looking straight and with symptoms during neck anteflexion. The left internal jugular vein had a diameter of 6.0 mm and 12.0 mm when looking straight (figure 3A) and during neck anteflexion (figure 3B), respectively, on ultrasonography, while the right internal jugular vein was 3.0 mm and 2.0 mm in diameter when looking straight and during neck anteflexion, respectively. Due to poor blood flow on ultrasonography, we suspected venous thrombosis in the left subclavian vein and initiated treatment with oral anticoagulants. After 2 weeks of treatment, the mass did not disappear and change on ultrasonography.

Figure 1

The left cephalic vein is enlarged.

Figure 2

Cervical ultrasound reveals a space-occupying lesion in the left subclavian vein.

Figure 3

(A) Ultrasound findings while looking straight reveal the left internal jugular vein with a diameter of 6.0 mm. (B) Ultrasound findings on anteflexion of the neck reveal the left internal jugular vein with a diameter of 12.0 mm.

Investigations

Contrast-enhanced CT revealed a mass of 20 mm in diameter in the left subclavian vein and a point-like enhancement (figure 4A). MRI revealed moderate hyperintensity on T2-weighted image (figure 4B).

Figure 4

(A) Contrast-enhanced CT shows a mass of 20 mm in diameter in the left subclavian vein and a point-like enhancement (yellow arrow). (B) MRI shows moderate hyperintensity on T2-weighted image (yellow arrow).

Differential diagnosis

Contrast-enhanced CT and MRI suggest an intravascular tumour such as leiomyosarcoma.

Treatment

Suspecting a malignancy such as leiomyosarcoma, we performed surgical resection through suprasternal, intercostal and left subclavicular incisions and blood vessel replacement of 12 mm expanded polytetrafluoroethylene artificial graft. The duration of the operation was 3 hours and 40 min. After the operation, we administered intravenous unfractionated heparin followed by an oral anticoagulation agent edoxaban.

Outcome and follow-up

Histopathological examination showed intravenous capillary haemangioma (figure 5A,B). After the operation, the sensation of pressure in the patient’s left neck disappeared. After discharge, she had not felt neck discomfort when cooking.

Figure 5

(A) Resected nodular lesion 15 mm in diameter originates from the vessel wall. (B) Histopathological examination shows a lobular proliferation of the capillaries and endothelial cells (H&E stain on high power).

Discussion

Intravascular lobular capillary haemangioma is a rare condition that occurs in the veins of the neck and upper extremities and large abdominal veins. The clinical presentations are non-specific. When symptomatic, patients usually complain of oedema of the extremity.2 Our patient complained of neck discomfort without oedema of the extremity. Ultrasonography found an association between the neck symptom and dilatation of the internal jugular vein in two different body positions. The mass was located at the junction of the subclavian and internal jugular veins; hence, we speculated that change in the position of the mass exacerbated congestion of blood flow from the internal jugular vein and caused neck discomfort.

Accurate differential diagnosis of the intravascular lobular capillary haemangioma is important because intravascular masses, especially vascular malignancies, could be fatal.3 We initially diagnosed the mass as a venous thrombosis based on ultrasound findings. However, the size of the mass did not change with administration of an anticoagulant on repeat ultrasound 2 weeks later, CT showed a point-like enhancement, and MRI findings showed moderate hyperintensity on T2-weighted images. Based on these findings, we suspected leiomyosarcoma and performed surgical resection. The typical imaging finding of an intravascular lobular capillary haemangioma is that of a well-defined and hypervascular mass on ultrasonography and heterogeneous enhancement on contrast-enhanced CT. However, these findings are not themselves highly specific for an intravascular lobular capillary haemangioma. Similarly, T2-weighted MRI signal is non-specific.4 Surgical resection must be selected to rule out vascular malignancies if the clinical course or imaging findings are not typical for benign causes, such as venous thrombosis.

Thus, the patient diagnosed with intravenous lobular capillary haemangioma presented with neck discomfort associated with neck anteflexion. The findings suggest that we must consider not only orthopaedic lesions but also intravenous abnormalities when patients complain of neck symptoms with change in posture.

Learning points

  • Lobular capillary haemangioma rarely originates intravenously.

  • The mass in the subclavian vein can cause neck symptoms associated with neck anteflexion due to congestion in blood flow from the internal jugular vein.

  • Intravenous lobular capillary haemangioma should be differentiated from venous thrombosis or vascular malignancies.

Acknowledgments

We would like to thank Editage (www.editage.com) for English language editing.

Footnotes

  • Contributors All authors contributed to the development of this manuscript. KI was responsible for the literature search and writing of all manuscripts. TI, KS and YO were supervisors.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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