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<article article-type="case-report" xmlns:xlink="http://www.w3.org/1999/xlink" xmlns:mml="http://www.w3.org/1998/Math/MathML">
<front>
<journal-meta>
<journal-id journal-id-type="nlm-ta">J Orthop Case Reports</journal-id>
<journal-title>Journal of Orthopaedic Case Reports</journal-title>
<issn pub-type="ppub">2250-0685</issn>
<issn pub-type="epub">2321-3817</issn>
<publisher>
<publisher-name>Indian Orthopaedic Research Group</publisher-name>
<publisher-loc>India</publisher-loc>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="publisher-id">JOCR-12-83</article-id>
<article-id pub-id-type="doi">10.13107/jocr.2022.v12.i10.3378</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Case Report</subject>
</subj-group>
</article-categories>
<title-group>
<article-title>A Case Report of an Unusual Case of Cavernous Hemangioma at Retrocalcaneum Region</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name>
<surname>Tripathi</surname>
<given-names>Ashutosh Chandraprakash</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="corresp" rid="cor1"/>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Baitule</surname>
<given-names>Rajendra Wamanrao</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Rathod</surname>
<given-names>Yogesh Bhimrao</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Pundkar</surname>
<given-names>Ganesh Narayanrao</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Bhutada</surname>
<given-names>Gaurav Pawan</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
</contrib>
</contrib-group>
<aff id="aff1"><label>1</label>Department of Orthopaedic Surgery, Dr. Panjabrao Deshmukh Memorial Medical College, Amravati, Maharashtra, India</aff>
<author-notes>
<corresp id="cor1">
<bold>Address of Correspondence:</bold> Dr. Ashutosh Chandraprakash Tripathi, Department of Orthopaedic Surgery, Dr. Panjabrao Deshmukh Memorial Medical College, Panchvati Square, Amravati, Maharashtra, India. E-mail: <email xlink:href="ashutoshckv.95@gmail.com">ashutoshckv.95@gmail.com</email>
</corresp>
</author-notes>
<pub-date pub-type="ppub">
<month>10</month>
<year>2022</year>
</pub-date>
<pub-date pub-type="epub">
<month>10</month>
<year>2022</year>
</pub-date>
<volume>12</volume>
<issue>10</issue>
<fpage>83</fpage>
<lpage>86</lpage>
<history>
<date date-type="received"><day>25</day><month>07</month><year>2022</year></date>
<date date-type="rev-recd"><day>15</day><month>08</month><year>2022</year></date>
<date date-type="accepted"><month>09</month><year>2022</year></date>
</history>
<permissions>
<copyright-statement>Copyright: &#x000a9; Indian Orthopaedic Research Group</copyright-statement>
<copyright-year>2022</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0">
<p>This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</p>
</license>
</permissions>
<abstract>
<sec id="st1">
<title>Introduction:</title>
<p>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.</p>
</sec>
<sec id="st2">
<title>Case Report:</title>
<p>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&#x2019;s pain was reduced, and overall performance was good at follow-up.</p>
</sec>
<sec id="st3">
<title>Conclusion:</title>
<p>This case report highlights the importance for surgeons, pathologist to consider cavernous hemangioma as a differential diagnosis for retrocalcaneal swellings.</p>
</sec>
</abstract>
<kwd-group>
<kwd>Cavernous hemangioma</kwd>
<kwd>retro calcaneal region</kwd>
<kwd>rare tumor</kwd>
</kwd-group>
</article-meta>
</front>
<body>
<boxed-text>
<title>Learning Point of the Article:</title>
<p>Cavernous hemangioma at an unusual site.</p>
</boxed-text>
<sec id="sec1-1" sec-type="intro">
<title>Introduction</title>
<p>Cavernous hemangiomas are congenital vascular anomalies, not vascular tumors [<xref ref-type="bibr" rid="ref1">1</xref>, <xref ref-type="bibr" rid="ref2">2</xref>]. 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 [<xref ref-type="bibr" rid="ref3">3</xref>, <xref ref-type="bibr" rid="ref4">4</xref>, <xref ref-type="bibr" rid="ref5">5</xref>, <xref ref-type="bibr" rid="ref6">6</xref>]. Calcification in vascular malformations is a common finding [<xref ref-type="bibr" rid="ref7">7</xref>, <xref ref-type="bibr" rid="ref8">8</xref>, <xref ref-type="bibr" rid="ref9">9</xref>, <xref ref-type="bibr" rid="ref10">10</xref>]. 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 [<xref ref-type="bibr" rid="ref1">1</xref>, <xref ref-type="bibr" rid="ref2">2</xref>, <xref ref-type="bibr" rid="ref11">11</xref>, <xref ref-type="bibr" rid="ref12">12</xref>, <xref ref-type="bibr" rid="ref13">13</xref>]. It exhibits an aggressive growth pattern, can occur anywhere in the body, and may recur after resection [<xref ref-type="bibr" rid="ref1">1</xref>]. This report shows a rare localization of hemangioma in the retrocalcaneal bursa.</p>
</sec>
<sec id="sec1-2" sec-type="cases">
<title>Case Presentation</title>
<p>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 (<xref ref-type="fig" rid="F1">Fig. 1</xref>). 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 (<xref ref-type="fig" rid="F2">Fig. 2</xref>); excision of the mass was done and sent the specimen for histopathology (<xref ref-type="fig" rid="F3">Fig. 3</xref>), calcified deposits were completely removed, Post-operative X-ray of ankle was taken (<xref ref-type="fig" rid="F4">Fig. 4</xref>).</p>
<fig id="F1">
<label>Figure 1</label>
<caption>
<p>X-ray of the patient (arrow showing the calcific deposits at retrocalcaneal region).</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="JOCR-12-83-g001.tif"/>
</fig>
<fig id="F2">
<label>Figure 2</label>
<caption>
<p>Microscopic picture of the sample collected (Hemangioma along with ossesous metaplasia).</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="JOCR-12-83-g002.tif"/>
</fig>
<fig id="F3">
<label>Figure 3</label>
<caption>
<p>Gross pathology of the specimen.</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="JOCR-12-83-g003.tif"/>
</fig>
<fig id="F4">
<label>Figure 4</label>
<caption>
<p>Perioperative photograph of the location of the swelling (Instrument is lifting the sural nerve behind which the tumor was present).</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="JOCR-12-83-g004.tif"/>
</fig>
<p>Pathology revealed calcified bursa. Microscopically, it was hemangioma with phleboliths and osseous metaplasia (<xref ref-type="fig" rid="F5">Fig. 5</xref>). The post-operative period was uneventful. The patient&#x2019;s pain was reduced and overall performance was good at follow-up.</p>
<fig id="F5">
<label>Figure 5</label>
<caption>
<p>Post-operative X-ray of ankle anteroposterior and lateral view after excision.</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="JOCR-12-83-g005.tif"/>
</fig>
</sec>
<sec id="sec1-3" sec-type="discussion">
<title>Discussion</title>
<p>Hemangiomas are hamartomatous growths of endothelial tissue [<xref ref-type="bibr" rid="ref1">1</xref>, <xref ref-type="bibr" rid="ref2">2</xref>]. They are the most common hemangiomatous lesions, accounting for 7&#x0025; 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 [<xref ref-type="bibr" rid="ref6">6</xref>]. 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 [<xref ref-type="bibr" rid="ref4">4</xref>].</p>
<p>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 [<xref ref-type="bibr" rid="ref14">14</xref>].</p>
<p>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 [<xref ref-type="bibr" rid="ref3">3</xref>, <xref ref-type="bibr" rid="ref4">4</xref>, <xref ref-type="bibr" rid="ref5">5</xref>, <xref ref-type="bibr" rid="ref15">15</xref>, <xref ref-type="bibr" rid="ref16">16</xref>]. 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.</p>
</sec>
<sec id="sec1-4" sec-type="conclusion">
<title>Conclusion</title>
<p>This case report highlights the importance for surgeons, pathologist to consider spindle cell hemangioma as a differential diagnosis for retrocalcaneum swellings.sss</p>
<boxed-text>
<title>Clinical Message</title>
<p>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.</p>
</boxed-text>
</sec>
</body>
<back>
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<fn-group>
<fn fn-type="conflict">
<p><bold>Conflict of Interest:</bold> Nil</p>
</fn>
<fn fn-type="supported-by">
<p><bold>Source of Support:</bold> Nil</p>
</fn>
<fn fn-type="other">
<p><bold>Consent:</bold> The authors confirm that informed consent was obtained from the patient for publication of this case report</p>
</fn>
</fn-group>
</back>
</article>
