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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 4  |  Issue : 4  |  Page : 112-115

Widespread heterotopic ossification in a patient with anoxic brain injury: Case report and review of literature


1 Department of Surgery, Aminu Kano Teaching Hospital, Bayero University Kano, Nigeria
2 Department of Anaesthesia, Aminu Kano Teaching Hospital, Bayero University Kano, Nigeria
3 Department of Anatomy, Faculty of Basic Medical Sciences, Bayero University Kano; Department of Anatomy, Faculty of Medicine, University of Bisha, Bisha, Nigeria
4 Department of Anatomy, Faculty of Basic Medical Sciences, Bayero University Kano, Nigeria

Date of Web Publication11-Apr-2018

Correspondence Address:
Dr. Lawan H Adamu
Department of Anatomy, Faculty of Basic Medical Sciences, Bayero University Kano, PMB 3011
Nigeria
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DOI: 10.4103/ssajm.ssajm_34_17

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  Abstract 


Heterotopic ossification (HO) is the formation of bone in the soft tissue, which exhibits no properties of ossification. Widespread HO is an uncommon condition. We present the findings in an 18-year-old young female who presented with benign bilateral fibroadenoma of the breast. However, she suffered an anoxic brain injury following cardiac arrest and had mechanical ventilation for some weeks. Thereafter, she developed widespread HOs, which were subsequently managed conservatively but required surgery for the ossification around the right hip. The report highlighted the multiplicity of risk factors vis-à-vis the challenges of management. A plan for the future care of this patient in relation to some of the currently available literature has also been highlighted.

Keywords: Anoxic brain injury, heterotopic ossification, surgical intervention


How to cite this article:
Abdulrahman MA, Atiku M, Taura MG, Adamu LH. Widespread heterotopic ossification in a patient with anoxic brain injury: Case report and review of literature. Sub-Saharan Afr J Med 2017;4:112-5

How to cite this URL:
Abdulrahman MA, Atiku M, Taura MG, Adamu LH. Widespread heterotopic ossification in a patient with anoxic brain injury: Case report and review of literature. Sub-Saharan Afr J Med [serial online] 2017 [cited 2018 Jun 22];4:112-5. Available from: http://www.ssajm.org/text.asp?2017/4/4/112/229756




  Introduction Top


Heterotopic ossification (HO) is the formation of bone in the soft tissue, which exhibits no properties of ossification.[1],[2],[3] It is also called myositis ossificans circumscripta.[3] Another variant is dystrophic calcification, which is seen in the case of hypercalcemia.[1] HO commonly occurs following brain injury (traumatic, anoxic, etc.), spinal cord injury, burns, tetanus, hemophilia, etc.[1],[2],[3] and affects mostly the synovial joints especially the hip, elbow, shoulder, and knee. It rarely affects the digital joints.[1],[2],[3] Other predisposing factors include limb spasticity (which increased the risk by up to 80–89% in most series), prolonged immobilization, ventilation in the brain injured, and decorticate and decerebrate posture.[2],[4],[5]

The incidence of HO in most series rose between 10 and 20%, with the hip most affected. Knee affectation was mostly seen among the spinal and brain injured, who had had ventilation for a while.[1],[2],[3] The presentation symptoms were similar to those seen in the case of osteomyelitis, septic arthritis, cellulitis, and thrombophlebitis in the early stages,[1],[2],[3] which later lead to the restriction of joint motion (range of motion), entrapment syndromes (neural or vascular), disuse osteoporosis, and joint ankylosis (commonly seen around the hip in the brain injured and in the knee joints in the spinal injured).[1],[2],[4]

Treatment is multidisciplinary and involves the use of nonsteroidal anti-inflammatory drugs (NSAIDS) (indomethacin or meloxicam) and bisphosphonates (e.g., etidronate) for at least 6 weeks. However, this is more useful following excision. Other treatment methods include the use of radiation therapy (the usefulness of which is questionable), physical therapy (the gradual and gentle range of motion exercises for all the joints up till a tolerable threshold level), and postural therapy.[3],[6],[7],[8] For very large symptomatic HO, surgical excision is recommended. However, manipulation under anesthesia followed by aggressive physiotherapy was also suggested. A combination of surgery and radiotherapy is another alternative.[9],[10],[11],[12],[13] In advanced cases with limitation in the range of motion and well-established HO, surgical excisions with early physiotherapy are advocated. In patients with ankylosed hips, excision arthroplasty or total hip replacement is advocated.[3],[4],[6],[7],[8] Previously, it was advised to undertake the treatment only when ossification is matured, that is, after 15 months. However, of recent, the indications have changed to prevent ankylosis and for hygienic considerations (restrictions in access to the perineum), because no significant increase in the rate of recurrence is noticed in cases operated early.[4],[6],[7],[8] The objectives of this report were to highlight the multiplicity of risk factors vis-à-vis the challenges of management. In addition, the report highlights the plans for the future care of this patient in relation to some of the currently available literature.


  Case history Top


Our patient was an 18-year-old, nulliparous student undergoing higher education. She was admitted through the general surgery clinic, with a bilateral breast mass (first noticed on the left side), which was present for one and a half years. Excision of the mass was undertaken under general anesthesia. She was induced with sodium thiopentone 250 mg, and intubation [with size 7.0 mm cuffed endotracheal tubes (ETT)] was facilitated using pancuronium 4 mg. About 5 min after intubation, the patient was noticed to have suffered sudden cardiac arrest. Cardiopulmonary resuscitation was immediately instituted, which lasted for about 10 min, and the patient was subsequently transferred to the intensive care unit (ICU).

While in intensive care, her real-time electrocardiogram (ECG) remained sinus rhythm, blood pressure (BP) − 120/70 mmHg with SPO2–100% (while on ventilator with FIO2 0.4) and Glasgow coma scale − 7/15 (on ventilator). However, she was noticed to have developed focal seizures involving mostly the upper limbs, and this was initially aborted with midazolam. However, seizures later became recurrent, and a neurologist commenced giving her tegretol and diazepam in addition to midazolam (for breakthrough seizures). She had a liver function test, wherein the results were normal except for a slight elevation in the alkaline phosphatase level (about 4 days into the admission) with a normal Ca2+/PO4− level. An electroencephalogram (EEG) was also requested. She was also commenced on nasogastric tube feeding. Seizures significantly improved within 2 days, and physiotherapy was commenced (a gradual range of motion/postural therapy).

About 4 weeks into the ICU admission, she developed bilateral knee swelling, which was fluctuant. Aspiration yielded straw-colored synovial fluid with no debris or blood. A specimen was sent for microscopy/culture and sensitivity, which yielded no growth, because the patient had already commenced on prophylactic antibiotics for hypostatic pneumonia at the time.

The patient was subsequently transferred to the ward after 6 weeks of intensive therapy, where she was managed by the combined efforts of a team of general surgeons, neurologist, ear, nose and throat (ENT) surgeons, intensivist, and rehabilitationist. She was finally discharged home in the 9th week. Later, she visited the orthopedic outpatient clinic on account of stiffness in both the shoulders and the right hip.

An assessment of the patient in the orthopedic clinic revealed a conscious and alert young woman who was wheelchair bound and aphasic, with provocative seizures of the upper and lower limbs. She had hyperextended lower limbs with left elbow flexion and a range of motion of 0–80° in the right shoulder, 0–90° in the left shoulder, 0° in the right hip joint, 0–90° in the right knee, and 0–30° in the left knee. The range of motion around the other joints was normal.

In addition, hard and fixed growths were noticed bilaterally around the lower third of the femur, and power in all the limbs was 3/5. In addition, the limbs were hypertonic and hyperreflexic. While examining the patient, she was noted to have had intermitted bouts of focal seizures affecting the limbs, with each seizure lasting at least 1 min, which were brought about by provocations, for example, passive joint motion. A plain radiograph of all the affected joints revealed a HO involving the posteroinferior aspect of the right shoulder, the anteroposterior surface of the left elbow, the posterosuperoinferior surface of the right hip joint, the anteromedial surface of the distal part of the left femur, and the anterior and posterior surfaces of the distal part of the right femur. An assessment and diagnosis of widespread HO involving multiple joints [[Figure 1],[Figure 2],[Figure 3],[Figure 4]] following anoxic brain injury was made. A plan for further treatment and for the surgical excision of the calcified ossification around the right hip following optimal seizure control was reported.
Figure 1: Heterotopic ossification around the shoulder joint

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Figure 2: Heterotopic ossification around the elbow joint

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Figure 3: Heterotopic ossification around the hip joint

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Figure 4: Heterotopic ossification around the knee joint

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  Discussion Top


The development of HO has been shown to be associated with many predisposing factors including neurological injury, both to the spinal cord and the brain, major joint surgery, and burns.[14],[15],[16] In this case report, the patient suffered an anoxic brain injury following cardiac arrest, which lasted for up to 10 min. While in ICU, she was in decerebrate posture and had two periods of intermittent pressure ventilations lasting averagely between 1 and 2 weeks each. As a result of this, she subsequently developed HOs of the right and left shoulders, right elbow, right hip, and right and left knees leading to a restriction of motion vis-à-vis general activity, which is in keeping with previous works.[1],[2],[5]

Anoxic brain injury is a common cause of HO in up to 10–20% of cases. However, it usually affects the hip, elbow, and shoulder, with ossification around the knee seen in patients with spinal injuries and those on prolonged intubation/ventilation. The latter may explain why our patient had ossifications around the knee in addition to the ossifications elsewhere.[1],[2],[5] She also suffered serious stiffness of the right hip in abduction with a restriction of activity and a 0° range of motion. However, access to the perineum was unhindered because of the attitude of the limb and the normal range of motion of the contralateral hip. The stiffness may be aided by prolonged immobility during the intensive care admission and inadequate mobilization of the hips during rehabilitation vis-à-vis extensive and forceful mobilization, which might have damaged the skeletal muscle fibers leading to a worsening condition around the hip, because it may be difficult to gauge the tolerable range of motion in an unconscious patient.[1],[6]

In addition to the aforementioned findings, the patient was still having provocative focal seizures that will require optimization. Thereafter, we made a plan to perform ossification around the hip to avoid ankylosis (bony) even though the ossification was just 7 months old and, therefore, yet to mature. There was a tendency for recurrence, though minimal, following excision before maturity.[1],[4],[7] The patient was also continued on the current regimen of a tolerable range of motion exercises involving all the joints and postural therapy, which we intend to continue even after the excision of the ossification around the hip.[4],[6],[7] However, for effective patient care, plans, and health policy, there was a high need to ensure safety while giving anesthesia. Direct visual intubation using endoscopy was needed. Facilities to monitor the acceptable range of motion threshold in unconscious patients and a better monitoring of seizures following anoxic brain injuries were recommended for better patient outcome. We also intend to excise the ossifications around the other joints after maturity if they continue to cause an impediment to the range of motion or the activity in the joint, as suggested by previous works.[4],[6],[7]

Interpretation and implications of this case in the context of the totality of evidence showed that the patient was presented with several risk factors. The global manifestation of HO in this patient involved virtually all the major joints. The unconscious condition of the patient led to difficulty in establishing a tolerable threshold of the range of motion. Optimization to allow the control of provocative seizure for proper rehabilitation and surgical intervention proved to be a challenge.


  Conclusion Top


We have highlighted the risk factors involved in the occurrence of widespread heterotopic calcification in a patient with anoxic brain injury, intubation, and immobilization as well as the challenges we faced in her management as a result of the residual seizures and immobilization.

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.
Shehab D, Elgazzar AH, Collier BD. Heterotopic ossification. J Nuclear Med 2002;43:3346-53.  Back to cited text no. 1
    
2.
Pape HC, Marsh S, Morley JR, Krettek C, Giannoudis PV. Current concept in the development of heterotopic ossification. J Bone Joint Surg Br 2004;86:783-7.  Back to cited text no. 2
    
3.
Bruno-Petrina A, Kishner S, Foye PM. Post-Traumatic Heterotopic Ossification. Medseape (Drug & Disease); 2015. Available from: http://emedicine.medscape.com/article/326242-overview. [Last accessed on 2016 Nov 29].  Back to cited text no. 3
    
4.
Capriano CA, Pill SG, Kenan MA. Heterotopic ossification following traumatic brain injury and spinal cord injury. J Am Acad Orthopaedic Surg 2009;17:689-97.  Back to cited text no. 4
    
5.
Heritt MS, Garland DE, Ayyoub Z. Heterotopic ossification complicating prolonged intubation. J Spinal Cord Med 2002;25:46-9.  Back to cited text no. 5
    
6.
Genet F, Marmorat JL, Lautridou C, Schnitzler A, Mailhan L, Denormandie P. Impact of late surgery intervention on heterotopic ossification on the hip after traumatic neurological injury. J Bone Joint Surg 2009;91:1493-8.  Back to cited text no. 6
    
7.
Ippolito E, Formisano R, Caterin R, Farsett P, Penta F. Operative treatment of heterotopic hip ossification in patients with coma after brain injury clinical. Orthopaedic 1999;365:130-8.  Back to cited text no. 7
    
8.
Pelissier J, Petiot S, Benaim C, Asencio G. Treatment of neurogenic heterotopic ossification and brain injured patients: Review of literature. Ann Readapt Med Phys 2002;45:188-97.  Back to cited text no. 8
    
9.
Chan PK, Chiu KY, Ng FY, Yan CH. Bony ankylosis of the knee secondary to heterotopic ossification after total knee arthroplasty: A case report. J Orthop Surg (Hong Kong) 2014;22:434-6.  Back to cited text no. 9
    
10.
Kloth JK, Tanner M, Stiller W, Burkholder I, Kauczor HU, Ewerbeck V et al. Radiation dose reduction in digital plain radiography of the knee after total knee arthroplasty. Rofo 2015;187:685–90.  Back to cited text no. 10
    
11.
Milakovic M, Popovic M, Raman S, Tsao M, Lam H, Chow E. Radiotherapy for the prophylaxis of heterotopic ossification: A systematic review and meta-analysis of randomized controlled trials. Radiother Oncol 2015;116:4-9.  Back to cited text no. 11
    
12.
Devnani AS. Management of heterotopic ossification affecting both hips and knees. Singapore Med J 2008;49:501-4.  Back to cited text no. 12
    
13.
Chen S, Yu SY, Yan H, Cai JY, Ouyang Y, Ruan HJ et al. The time point in surgical excision of heterotopic ossification of post-traumatic stiff elbow: Recommendation for early excision followed by early exercise. J Shoulder Elbow Surg 2015;24:1165-71.  Back to cited text no. 13
    
14.
Furia JP, Pellegrini VD. Heterotopic ossification following primary total knee arthroplasty. Arthroplasty 1995;10:413-9.  Back to cited text no. 14
    
15.
Choi YM, Hong SH, Lee CH, Kang JH, Oh JS. Extracorporeal shock wave therapy for painful chronic neurogenic heterotopic ossification after traumatic brain injury: A case report. Ann Rehabil Med 2015;39:318-22.  Back to cited text no. 15
    
16.
Spinarelli A, Carrozzo M, Teti M, Nappi V, Moretti B. Total knee arthroplasty complicated by a severe heterotopic ossification: A case report. Shafa Ortho J 2016;3:e5296.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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