CASE REPORT |
https://doi.org/10.5005/jojs-10079-1113 |
A Case of Calcific Myonecrosis: A Rare Clinical Presentation
1,2Department of Orthopaedics, Sri Venkateshwaraa Medical College Hospital & Research Centre, Puducherry, India
3Department of Community Medicine, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth University, Puducherry, India
Corresponding Author: Dhanasegaran Arunachalam, Department of Orthopaedics, Sri Venkateshwaraa Medical College Hospital & Research Centre, Puducherry, India, Phone: +91 9789349881, e-mail: dineshx202@gmail.com
Received on: 06 March 2023; Accepted on: 22 April 2023; Published on: 07 July 2023
ABSTRACT
Calcific myonecrosis, a complication of late compartment syndrome, is a rare disorder characterized by peripheral calcification and central liquefaction of the entire muscle mass. We present a case of myositis ossificans impersonation that was not only unusual but also posed difficulty in diagnosis and therapeutic management. The patient was a 45-year-old man presented with right leg pain and swelling for 6 months with no constitutional symptoms. He also had a history of trauma 1 year ago. Clinically, the swelling was multilobulated, fluctuant, and tender over the mid-anterolateral aspect of right leg. Neurological examination revealed right ankle dorsiflexion and eversion of right foot. X-ray of right leg full length revealed a homogeneous radiopaque lesion over the anterior and lateral aspect. Magnetic resonance imaging (MRI) suggested a myositis ossificans lesion limited to the right leg’s lateral and anterior compartments, with no hypervascularity. Routine blood tests, including liver function test, serum calcium, phosphorus, and alkaline phosphatase levels, were all normal. A nerve conduction study revealed that the right superficial and deep peroneal nerves had a lower conduction velocity. The tru-cut biopsy revealed myositis ossificans. Pus from the excision biopsy was sent for histopathological analysis, and the results revealed calcific myonecrosis. Following excision, along with physiotherapy and a course of indomethacin, patient got relieved off his symptoms and on follow-up had no signs of recurrence.
How to cite this article: Arunachalam D, Ramasamy B, Chellamuthu L. A Case of Calcific Myonecrosis: A Rare Clinical Presentation. J Orth Joint Surg 2023;5(2):101-104.
Source of support: Nil
Conflict of interest: None
Patient consent statement: The author(s) have obtained written informed consent from the patient for publication of the case report details and related images.
Keywords: Calcific myonecrosis, Compartment syndrome, Myositis ossificans, Neurological weakness, Softtissue calcified mass.
BACKGROUND
Calcific myonecrosis is a complication of late compartment syndrome, which is a rare disorder characterized by peripheral calcification and central liquefaction of the entire muscle mass.1 According to reports, it most commonly manifests in the anterior compartment of the leg and has been linked to trauma, such as fractures with ischemia symptoms and/or peripheral nerve damage.2 Furthermore, it has been proposed that these lesions are the result of cystic muscle degeneration and posttraumatic ischemia.3 A precise diagnosis is difficult because the symptoms appear much later than the initial damage and worsen gradually over several years.4 The trauma history and characteristic radiological findings are used to diagnose this rare syndrome.5 On radiographs, a plaque-like amorphous calcification pattern is common but not pathognomonic; these calcifications are typically linear in orientation and sheet-like, distributed throughout the muscle or compartment, and have a mixed radiolucency. Smooth bony erosions with minimal periosteal reaction are possible. Because of its large size, growth potential, and imaging characteristics, a sarcoma should be considered a differential diagnosis in cases where the erosions are extensive for a softtissue tumor.6-9 Patients who do not have symptoms can receive nonoperative care, whereas those who do have symptoms must have the mass completely removed.10 We present a case of myositis ossificans impersonation that was not only unusual but also presented a difficult diagnosis and therapeutic management.
CASE DESCRIPTION
A 45-year-old male presented to our hospital with a chief complaint of pain and swelling over his right leg for 6 months with no constitutional symptoms. He also had a history of trauma 1 year ago. The swelling began slowly and gradually increased in size over the anterior and posterior surfaces. For 6 months, the patient had no constitutional symptoms on the lateral aspect of the right leg. His previous medical history revealed no significant findings. Clinically, the swelling was multilobulated, fluctuant, and tender, measuring 5 × 2 cm on the right leg’s mid-anterolateral aspect (Fig. 1).
Figs 1A and B: A multilobulated swelling over the anterior and lateral aspect of mid-one-third of the right leg
Neurological examination revealed weak dorsiflexion of the right ankle and eversion of the right foot. The radiograph of the right leg exhibited a homogenous radiopaque lesion over the lateral aspect (Figs 2A and B). MRI is suggestive of myositis ossificans lesion restricted to the lateral and anterior compartment of the right leg with no hypervascularity (Figs 2C and D). Blood tests for liver function, serum calcium, phosphorus, and alkaline phosphatase were all normal. A nerve conduction test revealed a decrease in the conduction velocity of the right superficial and deep peroneal nerves. Tru-cut biopsy results suggested myositis ossificans. An excision biopsy was performed under spinal anesthesia with softtissue tumor and myositis ossificans as differential diagnoses in mind. The pus was extracted and sent for histopathological examination (Fig. 3). The intraoperative findings are shown in Figure 4. There was a thick collection of pus with multiple calcification spicules infiltrating the adjacent soft tissues.
Figs 2A to D: (A and B) depict the radiograph showing a homogeneous radiopaque mass lesion over the anterior and lateral aspect of the right leg; (C and D) illustrate the MRI showing features suggestive of myositis ossificans lesion restricted to the anterior and lateral compartment of the right leg with no hypervascularity.
Figs 3A and B: Illustrates the thick collection of whitish-colored aspirate (pus) with spicules of calcifications
Figs 4A to C: Illustration of the fragments of multiple spicules of calcifications with necrotic lesions in the muscle fibers
Pus for Gram stain and culture and sensitivity showed no evidence of any infection.
Histopathological examination revealed multiple fragments of fibrocollagenous tissue with focal aggregates of giant osteoclast cells, histiocytes, neutrophils, and lymphocytes, as well as fibrinoid necrosis with hemorrhage suggestive of necrotic calcification in muscle fibers (Fig. 5). Analgesics and postoperative physiotherapy, were used to relieve the patient’s symptoms following the excision. Despite having weak dorsiflexion and eversion of the right ankle and foot, the patient had no symptoms or signs of compartment syndrome. The patient was treated with oral indomethacin twice daily for 2 weeks, followed by a single dose for 2 weeks. Indomethacin acts as a potential analgesic in alleviating pain and acts as an anti-inflammatory agent preventing the progression of microlesions and in the prevention of the occurrence of new lesions. The patient reviewed recently reported being completely free of pain. The patient is able to perform his regular day-to-day activities with no further deterioration in the neurological status (Fig. 6).
Figs 5A and B: The histopathological examination of the pus collected through excision biopsy. (A) Shows osteoclastic giant cells with lymphocytic and neutrophilic infiltration. (B) Green arrow shows the presence of a fibrocollagenous matrix. The black arrow shows the giant cell. The red arrow shows the calcific necrotic cells, surrounded by neutrophilic and lymphocytic infiltrations
Fig. 6: Postoperative plain radiograph of right leg anteroposterior and lateral view showing reduction in the size of the radiopaque mass lesion following excision
DISCUSSION
Gallie and Thompson described calcific myonecrosis for the first time in 1960. To date, only a few cases of liquefaction and calcification within muscle mass have been documented.11 Recurrent hemorrhage within necrosed and fibrotic muscle tissue is another possibility for the development of this condition, but the actual pathophysiologic process is still unknown.12 Calcific myonecrosis is a condition that affects people between the ages of 17 and 87, with the majority of cases occurring in the sixth decade of life, according to existing literature. Furthermore, there was a slight male predominance.2 Calcific myonecrosis, like myositis ossificans, soft tissue sarcomas, diabetic myonecrosis, calcified soft-tissue mass, hematoma, parosteal osteosarcoma, and tumoral calcinosis, poses a diagnostic quandary.11,13 The majority of these cases have a documented history of prior trauma as the primary contributing factor, with a latent period of years preceding the onset of this slowly growing mass. The majority of the time, laboratory results are within normal ranges. However, on both plain radiography and MRI, the distinctive radiographic findings were demonstrated to be the cornerstone for identifying this condition. A homogeneous radiopaque lesion with peripheral calcification and central liquefaction is one of these.9,14 Furthermore, differentiating calcific myonecrosis from other pathologies necessitates a thorough histological examination. When this lesion is not detected by imaging or clinical practice, a biopsy may be required, which can have disastrous consequences such as chronic sinus drainage or secondary infection.15 Even with such a significant calcification, if the patient is asymptomatic, nonoperative treatment is preferable. Excision is the recommended treatment when there is no evidence of hypervascularity, and the pain is chronic. As a result, a clear history of distant trauma, as well as distinct microscopic and radiographic findings, aid in the early diagnosis and treatment of calcific myonecrosis.
CONCLUSION
This case report emphasizes the approach to diagnosis, the importance of considering the patient’s past medical history, the importance of considering differentials when dealing with such rare cases, and the management guidelines. A radiographic feature of a homogenous radiopaque lesion over the lateral aspect, as well as an MRI that suggests a myositis ossificans lesion limited to the anterior and lateral compartments of the right leg with no hypervascularity, can also help to confirm the diagnosis. Nonoperative treatment is recommended if the patient is asymptomatic; however, excisional biopsy is the only option for symptomatic patients.
ETHICAL CONSIDERATION
The patient provided written informed consent. The patient was thoroughly informed about the data collected and its intended use. He understands that his name and initials will not be published and that appropriate precautions will be taken to conceal his identity, but he understands that anonymity cannot be guaranteed. He has also given his permission for his photographs and other clinical data to be published in the journal.
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