Cavernous hemangioma at an unusual site.
Dr. Ashutosh Chandraprakash Tripathi, Department of Orthopaedic Surgery, Dr. Panjabrao Deshmukh Memorial Medical College, Panchvati Square, Amravati, Maharashtra, India. E-mail: firstname.lastname@example.org
Introduction: Subcutaneous hemangioma is a rare variant of slow-flowing venous malformation. It occurs in both adults and children and is more common in women. It exhibits an aggressive growth pattern, can occur anywhere in the body, and may recur after resection. This report shows a rare localization of hemangioma in the retrocalcaneal bursa.
Case Report: A female patient, age 31, presented with swelling and pain over the retro calcaneum region for 1 year. The pain in the retrocalcaneal region has increased with gradual intensity over the past 6 months. The swelling she described to be insidious in onset and gradually progressive. Examination findings at presentation were a middle-aged female with diffuse swelling in the retrocalcaneal region with a size of 2 cm by 1.5 cm. Based on the X-ray, we defined it to be myositis ossificans. With this view in mind, we admitted the patient and surgically excised the area. We operated by posteromedial approach and sent the specimen for histopathology. Pathology revealed calcified bursa. Microscopically, it was hemangioma with phleboliths and osseous metaplasia. The post-operative period was uneventful. The patient’s pain was reduced, and overall performance was good at follow-up.
Conclusion: This case report highlights the importance for surgeons, pathologist to consider cavernous hemangioma as a differential diagnosis for retrocalcaneal swellings.
Keywords: Cavernous hemangioma, retro calcaneal region, rare tumor
Cavernous hemangiomas are congenital vascular anomalies, not vascular tumors [1, 2]. Patients with vascular abnormalities become wanderers due to incorrect diagnosis, treatment complexity, limited outcomes, and lack of individual physician expertise. Vascular tumors are endothelial tumors characterized by increased endothelial turnover. Malformations are the result of the abnormal development of vascular elements during embryogenesis [3, 4, 5, 6]. Calcification in vascular malformations is a common finding [7, 8, 9, 10]. However, ossification within limb malformations is rarely reported. Subcutaneous hemangioma is a rare variant of slow-flowing venous malformation. It occurs in both adults and children and is more common in women [1, 2, 11, 12, 13]. It exhibits an aggressive growth pattern, can occur anywhere in the body, and may recur after resection . This report shows a rare localization of hemangioma in the retrocalcaneal bursa.
A female patient, age 31, presented with swelling and pain over the retro calcaneum region for 1 year. The pain in the retrocalcaneal region has increased with gradual intensity over the past 6 months. The intensity of the pain increased while walking and doing routine activities such as squatting and prolonged standing. The swelling she described to be insidious in onset and gradually progressive. No similar swelling was noticed in other parts of the body. No prior history of trauma was obtained; neither was there any weight loss, malaise, bone pains, nor a family history of similar pathology. Examination findings at presentation were a middle-aged female with diffuse swelling in the retrocalcaneal region with a size of 2 cm by 1.5 cm. With a background of this history, we suspected the patient to have some condition related to the retrocalcaneal bursa, most commonly to be retrocalcaneal bursitis. Plain radiographs of the region showed soft-tissue calcifications in the Tendo-Achilles region (Fig. 1). Based on the X-ray, we defined it to be myositis ossificans. With this view in mind, we admitted the patient and surgically excised the area. We operated by posteromedial approach; sural nerve was identified (Fig. 2); excision of the mass was done and sent the specimen for histopathology (Fig. 3), calcified deposits were completely removed, Post-operative X-ray of ankle was taken (Fig. 4). Pathology revealed calcified bursa. Microscopically, it was hemangioma with phleboliths and osseous metaplasia (Fig. 5). The post-operative period was uneventful. The patient’s pain was reduced and overall performance was good at follow-up.
Hemangiomas are hamartomatous growths of endothelial tissue [1, 2]. They are the most common hemangiomatous lesions, accounting for 7% of all benign soft-tissue tumors in the general population. Hemangiomas are most commonly diagnosed soft-tissue tumors and can be classified clinically as capillary or cavernous . Cavernous hemangiomas are tumors formed by vasodilatation. They can be found deeper in the skin and mucous membranes but also affect deeper structures such as subcutaneous tissue, muscles, and bones. Hemangiomas are focal or diffuse . Subcutaneous cavernous hemangiomas are rare. There is little literature, mostly case reports. Unlike other benign tumors, the life cycle of hemangiomas differs in that there is a period of rapid growth followed by spontaneous regression. This is especially true for the capillary and cutaneous spongy variants, although deep spongy types rarely regress, and malignant transformation is rare. To date, many reported cases of intramuscular hemangiomas were vascular malformations of skeletal muscle, and thus subcutaneous cavernous hemangioma and vascular malformations were rarely understood as separate entities. Mulliken and Glowacki described in their studies that vascular malformations and hemangiomas, characterized by endothelial hyperplasia or increased mitotic activity, develop in the late fetal or early neonatal period and proliferate . Furthermore, it emphasized that it usually regresses. Hemangiomas are often asymptomatic, but symptoms are usually due to pain, mass or soft-tissue swelling, subcutaneous discoloration, cosmetic concerns (by the parent or patient), and nerve entrapment. It is caused by neurological symptoms secondary to clinical examination, usually revealing soft masses of various shapes, swaying without swaying or changing warmth, soft and empty with ill-defined boundaries, thrilling, and masses. A murmur may be heard over the thorax. Radiological evaluation includes a plain X-ray showing soft-tissue swelling without bony involvement or calcification. Magnetic resonance imaging shows densely lobed masses of mixed intensity. Computed tomography shows a homogeneous mass with large feeding vessels with intensive and sustained administration of contrast agents. Ultrasonography with Doppler studies is an inexpensive, non-invasive technique that shows the high-flow pattern characteristic of hemangioma and distinguishes it from the low-flow pattern of vascular malformations. Treatment of hemangiomas is initially conservative and requires observation and regular evaluation. Modalities include cryotherapy, radiotherapy, laser therapy, subcutaneous injections of recombinant interferon-alpha, sclerosing agents, and intralesional corticosteroids, double-induced sclerotherapy with bubble injections, and angiographic embolization is available [3, 4, 5, 15, 16]. All have been tried with varying results. Surgical resection with ligation of afferent vessels is the optimal management of intramuscular hemangioma to prevent a recurrence. Indications for surgery include a gradually increasing mass or swelling, pressure pain and neuropathy, and significant cosmetic concerns. A structured post-operative rehabilitation protocol improves limb function and prevents joint stiffness.
This case report highlights the importance for surgeons, pathologist to consider spindle cell hemangioma as a differential diagnosis for retrocalcaneum swellings.
Diagnosing the cavernous hemangioma at an unusual site like retro calcaneal region is challenging this may influence the management and planning for the surgeon; hence, a detail evaluation is essential.
- 1.Casanova D, Norat F, Bardot J, Magalon G. Cutaneous hemangioma: Clinical aspects. Ann Chir Plast Esthet 2006;51:287-92. [Google Scholar]
- 2.Enjolras O, Mulliken JB. Malformações Vasculares. Rio Janeiro: Brazil Elsevier; 2011. p. 1581-95. [Google Scholar]
- 3.Garzon MC, Huang JT, Enjolras O, Frieden IJ. Vascular malformations: Part I. J Am Acad Dermatol 2007;56:353-4. [Google Scholar]
- 4.Legiehn GM, Heran MK. Venous malformations: classification, development, diagnosis, and interventional radiologic management. Radiol Clin North Am 2008;46:545-97, vi. [Google Scholar]
- 5.Jackson IT, Carreño R, Potparic Z, Hussain K. Hemangiomas, vascular malformations, and lymphovenous malformations: Classification and methods of treatment. Plast Reconstr Surg 1993;91:1216-30. [Google Scholar]
- 6.Enjolras O. Classification and management of the various superficial vascular anomalies: Hemangiomas and vascular malformations. J Dermatol 1997;24:701-10. [Google Scholar]
- 7.Ghosh P, Saha K. Hemangioma calcificans: A rare entity with epileptogenic potential. South Asian J Cancer 2013;2:231. [Google Scholar]
- 8.Jha VC, Abhijit V, Jha N, Rewatkar S, Sinha VS, Alam MS. Giant calcified cavernous hemangioma managed with modified double concentric craniotomy. J Neurosci Rural Pract 2021;12:592-5. [Google Scholar]
- 9.Brinda MA, Manjunath S, Balasubrahmaniya KS, Manjunath RD, Nanjaiah B. Intramuscular ossified haemangioma: A rare case report. J Clin Diagn Res 2015;9:PD19-21. [Google Scholar]
- 10.Kamatani T, Saito T, Hamada Y, Kondo S, Shirota T, Shintani S. Intramuscular hemangioma with phleboliths of the tongue. Indian J Dent 2014;5:100-1. [Google Scholar]
- 11.Li B, Niu X, Wang D. Diagnosis and treatment of subcutaneous cavernous hemangioma with color Doppler ultrasonography. Zhongguo Xiu Fu Chong Jian Wai Ke Za Zhi 2002;16:193-4. [Google Scholar]
- 12.Boon LM, Mulliken JB, Enjolras O, Vikkula M. Glomuvenous malformation (glomangioma) and venous malformation: Distinct clinicopathologic and genetic entities. Arch Dermatol 2004;140:971-6. [Google Scholar]
- 13.Gorlin RJ, Kantaputra P, Aughton DJ, Mulliken JB. Marked female predilection in some syndromes associated with facial hemangiomas. Am J Med Genet 1994;52:130-5. [Google Scholar]
- 14.Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: A classification based on endothelial characteristics. Plast Reconstr Surg 1982;69:412-22. [Google Scholar]
- 15.Seo BF, Kang KJ, Jung SN, Byeon JH. Skeletal cavernous hemangiomas of the frontal bone with orbital roof and rim involvement. Arch Craniofacial Surg 2018;19:214-7 [Google Scholar]
- 16.Kim JH, Choi JG, Son BC. Venous malformation (Cavernous hemangioma) of the supraorbital nerve. Asian J Neurosurg 2018;13:499-502. [Google Scholar]