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CASE REPORT
Year : 2019  |  Volume : 12  |  Issue : 6  |  Page : 533-536  

Multiple intraosseous capillary hemangiomas of the foot: An uncommon entity


1 Department of Orthopaedics, Base Hospital, Lucknow Cantt., Lucknow, Uttar Pradesh, India
2 Department of Orthopaedics, MH Kirkee, Pune, Maharashtra, India
3 Department of Histopathology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
4 Department of Orthopaedics, 158 Base Hospital, Bagdogra, West Bengal, India

Date of Submission27-Oct-2018
Date of Acceptance18-Feb-2019
Date of Web Publication17-Oct-2019

Correspondence Address:
B M Naveen
Department of Orthopaedics, Base Hospital, Lucknow Cantt, Lucknow - 226 002, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_220_18

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  Abstract 


Hemangiomas are benign vascular lesions made up of blood vessels, commonly seen in the skin, muscle, bone, and viscera. Intraosseous hemangiomas are rare, and the foot is a very uncommon location of these tumors. These can be symptomatic or asymptomatic. A 26 year old female patient reported to us with insidious onset of pain in the right foot for 10 years followed by swelling on the outer aspect of the forefoot. Radiographs and magnetic resonance imaging evaluation showed a predominantly lytic lesion in the head of the fifth metatarsal, with similar lesions at various other places in the foot. She was treated with complete excision of the tumor from the head of the fifth metatarsal following which she has resolution of symptoms. Biopsy of the specimen revealed capillary hemangioma. This case report affirms that, though uncommon, it should be kept as one of the differential diagnosis in the evaluation of lytic lesion of the foot.

Keywords: Capillary, foot, hemangioma, intraosseous


How to cite this article:
Naveen B M, Prabhakara A, Manoj M G, Hegde S. Multiple intraosseous capillary hemangiomas of the foot: An uncommon entity. Med J DY Patil Vidyapeeth 2019;12:533-6

How to cite this URL:
Naveen B M, Prabhakara A, Manoj M G, Hegde S. Multiple intraosseous capillary hemangiomas of the foot: An uncommon entity. Med J DY Patil Vidyapeeth [serial online] 2019 [cited 2019 Nov 23];12:533-6. Available from: http://www.mjdrdypv.org/text.asp?2019/12/6/533/269420




  Introduction Top


Hemangiomas are the benign vascular tumors that can be seen in any vascularized tissue of the human body. Intraosseous hemangiomas are relatively uncommon and account for <1% all primary osseous tumors.[1] Spine and cranium are the most common locations of intraosseous hemangiomas, and foot lesions are very rare. These lesions are usually asymptomatic and require treatment only if they become symptomatic. Here, we report an interesting case of symptomatic multiple intraosseous hemangiomas of the foot in a young female.


  Case Report Top


A 26 year old female presented to the outpatient department of a tertiary care teaching hospital with complaints of insidious onset of pain in the right foot of 10 years duration. Pain was mild to moderate, vague, dull aching in character, and aggravated by bearing weight. She was taking symptomatic treatment from a local hospital. Six months before this presentation, she noticed a small swelling near the outer aspect of the forefoot which was slowly progressive. On evaluation at this center, a small 3 cm × 2 cm noncircumscribed, soft, smooth, mildly tender swelling with ill-defined margins was noted over the dorsum of the foot near the head of the fifth metatarsal [Figure 1]a and [Figure 1]b. Movements of the foot were normal with no distal neurovascular deficits.
Figure 1: (a) Preoperative clinical picture of the foot – dorsoplantar view (arrow mark); (b) preoperative clinical picture of the foot – lateral view (arrow mark); (c) preoperative radiograph of the foot (dorsoplantar view) showing the lytic area (arrowheads); (d) postoperative radiograph of the foot (oblique view) after curettage (arrowheads)

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Roentgenogram of the foot revealed an osteolytic lesion in the fifth metatarsal head with erosion of medial cortex [Figure 1]c. A similar looking lesion, though small was noted in the fourth metatarsal head with cortical thinning of the third and fourth metacarpals in the head and neck area. Magnetic resonance imaging (MRI) revealed a 2-cm lytic expansile lesion in the head of the fifth metatarsal along the medial cortex [Figure 2]a,[Figure 2]b,[Figure 2]c,[Figure 2]d,[Figure 2]e. Similar intensity lesions were noted in the plantar aspect of the second, third, and fourth metatarsals and in the third and fourth intermetacarpal spaces. Similar multiple cystic lesions were also reported in the posterior aspect of the foot in the calcaneum along its inferior and lateral regions with a possibility of ganglion or a synovial cyst. With this clinicoradiological picture, a differential diagnosis of an infectious pathology, histiocytosis X, or a malignant transformation of a benign lesion was kept in mind.
Figure 2: (a) Preoperative magnetic resonance imaging of the foot (coronal view) showing the tumor (arrow mark); (b) preoperative magnetic resonance imaging of the foot (coronal view) showing the tumor (arrow mark); (c) preoperative magnetic resonance imaging of the foot (axial view) showing the tumor (arrow mark); (d) preoperative magnetic resonance imaging of the foot (axial view) showing the tumor (arrow mark); (e) preoperative magnetic resonance imaging of the foot (sagittal view) showing the tumor at various places (arrow mark)

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With a possibility of a malignant lesion, biopsy of the lesion was planned from the head of the fifth metatarsal. The intraosseous content was subjected to the frozen section study. Once the frozen section revealed a benign vascular lesion, the lesion was completely curetted and the adjoining soft tissue lesion was also excised [Figure 3]a and [Figure 3]b. The entire specimen was then sent for histopathological examination which confirmed capillary hemangioma. Microscopy showed multiple proliferating thick and thin-walled capillaries lined by bland endothelial cells that showed no atypia [Figure 3]c and [Figure 3]d. These capillaries were surrounded by round-to-oval monomorphic fibroblasts with uniform nuclei. No cyst wall was noted. There was no evidence of atypia or malignancy. On follow-up, the patient's symptoms had improved partially. The 3-month postoperative radiograph showed an evidence of healing in the head of the fifth metatarsal [Figure 1]d. MRI showed no residual lesion at the operated site with other lesions in situ maintaining status quo.
Figure 3: (a) Intraoperative picture of the lesion in the head of the fifth metatarsal from anterior aspect (arrow mark); (b) intraoperative picture of the lesion in the head of the fifth metatarsal from anterolateral aspect (arrow mark); (c) hematoxylin and eosin-stained section (×100) shows a feeder vessel (*) with surrounding thin-walled blood vessels; (d) hematoxylin and eosin-stained section (×400) shows thin-walled vessels (arrowheads) containing erythrocytes

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  Discussion Top


Primary intraosseous hemangiomas are rare tumors forming only 0.7%–1.0% of all bone tumors. These are usually encountered as isolated or multiple lesions. Skull and spine are the common sites of involvement.[2] Hemangiomas of bone are usually asymptomatic and are detected as incidental findings on X-ray. However, some cases may present with pain, swelling, or pathologic fracture. Hemangioma of the foot has not been reported in the largest case series of intraosseous hemangiomas of 108 cases by Unni.[3] Till date, only very few cases have been reported in the feet with all being male patients. These lesions commonly present in the fifth decade. Some authors define hemangiomatosis as multiple hemangiomas located in noncontiguous bones, whereas multiple bony hemangiomas with no soft-tissue component are referred as cystic angiomatosis of bone. Hemangiomas affecting both bone and any one nonosseous site are called as skeletal-extraskeletal angiomatosis.[4] The nonosseous site is often the adjacent soft tissue or noncontiguous viscera. A few others define hemangiomatosis as lesions involving skin, muscle, and bone, which are usually symptomatic during childhood, with diffuse, persistent swelling and discoloration, with or without pain.

The typical radiological appearances of hemangioma such as sunburst and corduroy patterns, seen in the skull and spine, are not seen in the extremities. In extremities, they are seen in metaphysis or epiphysis as lytic lesions having spiculated pattern or honeycomb appearance with irregular bone destruction and a reactive sclerosis at times. Due to these uncertain radiological features, the diagnosis of hemangiomas in a lytic lesion of the extremity is rarely made on radiology.[5] These lesions are poorly defined and can appear on the surface of the bone, within the cortex or extend into the medullary cavity. They also have a propensity to extend into the surrounding soft tissues. Osseous vascular malformations commonly demonstrate a high-signal-intensity trabecular pattern at both T1- and T2-weighted MRI due to the presence of fat in the tumor. Histologically, hemangiomas can be capillary, cavernous, or mixed type, of which cavernous type is the most common. The index case was of capillary subtype affecting the female sex and seen at multiple sites in the foot, making it unique. The only other capillary subtype was reported in an 8-year-old boy by Ngan et al.[6] The remaining cases reported by various authors were of cavernous type and were seen in male patients.[7],[8],[9],[10]

Capillary hemangioma is made up of small capillaries that are normal in size and diameter, but densely packed group held together by thin, connective tissue. The histological appearance of hemangiomas should be differentiated from that of pyogenic granuloma, lymphangioma, and kaposiform hemangioendothelioma. The true nature of hemangiomas remains controversial, with no clear consensus on its causation, or agreement as to whether hemangiomas are neoplasms, hamartomas, or malformations. Rarely, hemangiomas may be associated with induction of osteomalacia. Only symptomatic lesions warrant further surgical treatment in the form of excision or vascular ablative therapy. Hemangiomas can also be treated with direct sclerotherapy, intralesional excision, or transarterial embolization. Large lesions may require void filling with bone grafts or by its substitutes following the excision. One should exercise caution while treating deep-seated lesions as they may bleed profusely.

This case report emphasizes the need to keep hemangioma as a possible differential diagnosis when we encounter an osteolytic lesion of the foot. Diagnosis can be established by having a high degree of suspicion and utilize intraoperative frozen section studies to avoid an unwarranted and unnecessary surgical treatment which can cause more morbidity in itself than the disease per se. Being a benign tumor, it becomes more imperative in early diagnosis to differentiate it from sinister differential diagnosis of osteomyelitis, kaposiform hemangioendothelioma, or histiocytosis X.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Dorfman HD, Steiner GC, Jaffe HL. Vascular tumors of bone. Hum Pathol 1971;2:349-76.  Back to cited text no. 1
    
2.
Murphey MD, Fairbairn KJ, Parman LM, Baxter KG, Parsa MB, Smith WS, et al. From the archives of the AFIP. Musculoskeletal angiomatous lesions: Radiologic-pathologic correlation. Radiographics 1995;15:893-917.  Back to cited text no. 2
    
3.
Unni KK. Dahlin's Bone Tumors. General Aspects and Data on 11087 Cases. 5th ed. Philadelphia: Lippincott-Raven Publishers; 1996. p. 307-16.  Back to cited text no. 3
    
4.
Devaney K, Vinh TN, Sweet DE. Skeletal-extraskeletal angiomatosis. A clinicopathological study of fourteen patients and nosologic considerations. J Bone Joint Surg Am 1994;76:878-91.  Back to cited text no. 4
    
5.
Kaleem Z, Kyriakos M, Totty WG. Solitary skeletal hemangioma of the extremities. Skeletal Radiol 2000;29:502-13.  Back to cited text no. 5
    
6.
Ngan KW, Hsu HL, Ueng SH. An intraosseous capillary hemagioma of the foot in a child. Chang Gung Med J 2006;29 Suppl 4:71-5.  Back to cited text no. 6
    
7.
Mirra JM, editor. Vascular tumors. In: Bone Tumors. Vol. 2. Philadelphia: Lea and Febiger; 1989. p. 1338-69.  Back to cited text no. 7
    
8.
Murari TM, Callaghan JJ, Berrey BH Jr., Sweet DE. Primary benign and malignant osseous neoplasms of the foot. Foot Ankle 1989;10:68-80.  Back to cited text no. 8
    
9.
Wu KK. Intraosseous hemangioma of the foot. J Foot Ankle Surg 1994;33:314-7.  Back to cited text no. 9
    
10.
Davies JL, Stone PA, McGarry JJ. Mixed cavernous and capillary intraosseous hemangioma of the foot. J Am Podiatr Med Assoc 1997;87:478-82.  Back to cited text no. 10
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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