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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 8  |  Issue : 1  |  Page : 27-29

Rare complication of a common disease: Coccygeal osteomyelitis following Pilonidal sinus


Department of Digestive Diseases, Zen Multispeciality Hospital, Mumbai, Maharashtra, India

Date of Web Publication12-Mar-2019

Correspondence Address:
Dr. Pranav Mandovra
Department of Digestive Diseases, Zen Multispeciality Hospital, Plot No. 425, 10th Road, Chembur East, Mumbai - 400 071, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/WJCS.WJCS_10_18

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  Abstract 

Pilonidal sinuses commonly arise in the sacrococcygeal region. The common complications are local cellulitis, abscess formation which is related to the infectious process, and recurrence after surgery. They rarely evolve with osteomyelitis, meningitis, or malignant transformation. Coccygeal osteomyelitis as a direct complication of sacrococcygeal pilonidal sinus disease (PSD) is extremely rare with limited data. We report a case of complicated sacrococcygeal PSD with coccygeal osteomyelitis. It was managed with: wide local excision of the pilonidal sinus, coccygectomy, perineal musculature reconstruction, and defect closure by Limberg rhomboid flap.

Keywords: Coccygeal osteomyelitis, coccygectomy, complicated pilonidal sinus


How to cite this article:
Kowli M, Mandovra P, Zaveri G, Patankar R. Rare complication of a common disease: Coccygeal osteomyelitis following Pilonidal sinus. World J Colorectal Surg 2019;8:27-9

How to cite this URL:
Kowli M, Mandovra P, Zaveri G, Patankar R. Rare complication of a common disease: Coccygeal osteomyelitis following Pilonidal sinus. World J Colorectal Surg [serial online] 2019 [cited 2019 Oct 14];8:27-9. Available from: http://www.wjcs.us.com/text.asp?2019/8/1/27/254034


  Introduction Top


Pilonidal sinus disease (PSD) is a common occurrence and well identified. The most common location of PSD is the sacrococcygeal region; however, it occasionally occurs elsewhere in the body, including umbilicus, penis, breast, axilla, neck, nose, chin, and interdigital webs.[1] Although it is common, PSD is a complex condition to deal with. In the sacrococcygeal area, it usually presents as a chronic painful discharging sinus or in acute setting as an abscess. Complication such as carcinomatous transformation has been well-documented in the literature;[2] however, coccygeal osteomyelitis as a direct complication of sacrococcygeal PSD is extremely rare with limited data.

We present a rare case of a 17-year-old male having complicated sacrococcygeal PSD with coccygeal osteomyelitis.


  Case Report Top


A 17-year-old male patient who presented to our hospital with chronic foul smelling discharging sinus in the gluteal cleft for 2 years. He had taken treatment for the same, and even once incision and drainage of the cavity were done 11 months ago; however, the wound did not heal and discharge persisted. On examination, there was pilonidal sinus with two external openings in the midline gluteal cleft with active purulent discharge and cellulitis [Figure 1]. He was given a course of antibiotics according to the microbial sensitivity, but there was no significant improvement even after 10 days.
Figure 1: Pilonidal sinus with two external openings

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Being a recurrent disease with limited response to antibiotics for active discharge, it raised suspicion of some underlying pathology, and the patient was examined further. Magnetic resonance imaging (MRI) was done which revealed pilonidal sinus with coccygeal osteomyelitis with a small localized subcutaneous fluid pocket posteroinferior to coccyx in the posterior gluteal cleft [Figure 2].
Figure 2: Magnetic resonance imaging perineum showing the presence of pilonidal sinus with associated coccygeal osteomyelitis

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Subsequently, with a multidisciplinary team, including a spine surgeon and a plastic surgeon, wide local excision of the pilonidal sinus with coccygectomy and perineal musculature reconstruction was carried out. Defect closure was done by Limberg rhomboid flap, and the negative suction drain was kept in situ [Figure 3].
Figure 3: Wide local excision of the pilonidal sinus with coccygectomy and Limberg rhomboid flap

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The operative time was 140 min with approximate 50 cc blood loss. The postoperative course was uneventful, and the patient was discharged on 4th postoperative day. A negative suction drain was removed on 7th postoperative day.

Histopathological report showed chronic pyogenic pilonidal sinus with several impacted hair shafts and coccygeal osteomyelitis. Specimen of coccyx was negative for acid-fast bacilli stain, TB geneXpert, and culture.

On 10 months' follow-up, the patient was asymptomatic, and there was no evidence of recurrence.


  Discussion Top


Sacrococcygeal PSD is a common condition with an estimated incidence of 26/100,000 population majorly affecting young males.[3] The pathogenesis of PSD has been much debated and is considered as an acquired condition by the majority rather than being a congenital disease. Body movements generate friction which leads to hair disruption. Hairs puncture the skin surface and get buried forming the pilonidal cyst which eventually gets infected leading to sinus formation to drain the suppuration.[4] PSD is a painful condition with either an acute presentation as abscess cavity or as a chronically discharging sinus tract.

PSD is a complex condition to manage, and several procedures have been described for its management; however, no single method is accepted as gold standard treatment. There are high recurrences even after surgical excision of primary PSD. Furthermore, there are reports in the literature describing the development of malignancy in pilonidal sinus.[5] Complications in infected PSD are frequent, especially in the recurrent ones; however, pilonidal sinus causing osteomyelitis or bone destruction is a very rare entity. Karadaǧ et al. reported a case of recurrent lesion over the right brow with destruction of bone tissue in lateral orbital wall owing to incomplete excision of hair lined pilonidal sinus tract.. This lesion was intially misdiagnosed as epidermal cyst.[6] Mohanna et al. reported a case of subungal pilonidal sinus complicated by the development of osteomyelitis in the distal phalanx of the thumb of dog groomer.[7]

There is a paucity of literature on coccygeal osteomyelitis in itself, and it has extremely rare incidence as a complication of sacrococcygeal PSD. Gordon and Hunt reported the only case of coccygeal osteomyelitis as a direct complication of a chronic PSD.[8]

Patients with recurrent PSD or persistent foul-smelling discharge or non-responsive cellulitis warrant need further investigation such as MRI to rule out underlying pathology or complication. In our case, MRI was done in view of relentless foul-smelling discharge not improving even after the course of antibiotics. MRI revealed PSD with underlying coccygeal osteomyelitis which is a rare complication of sacrococcygeal PSD.

Coccygeal osteomyelitis responds to antibiotics in majority of cases. Failure to response leads to surgical treatment. Surgical intervention in the form of coccygectomy is the treatment of choice in advanced or complicated cases.

Our case report is an addition to the limited literature on the rare complication of sacrococcygeal PSD resulting in coccygeal osteomyelitis.

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.

Acknowledgment

The authors would like to thank the Department of Zen Digestive Disease Centre for their support.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Chintapatla S, Safarani N, Kumar S, Haboubi N. Sacrococcygeal pilonidal sinus: Historical review, pathological insight and surgical options. Tech Coloproctol 2003;7:3-8.  Back to cited text no. 1
    
2.
Lineaweaver WC, Brunson MB, Smith JF, Franzini DA, Rumley TO. Squamous carcinoma arising in a pilonidal sinus. J Surg Oncol 1984;27:239-42.  Back to cited text no. 2
    
3.
Horwood J, Hanratty D, Chandran P, Billings P. Primary closure or rhomboid excision and Limberg flap for the management of primary sacrococcygeal pilonidal disease? A meta-analysis of randomized controlled trials. Colorectal Dis 2012;14:143-51.  Back to cited text no. 3
    
4.
Clothier PR, Haywood IR. The natural history of the post anal (pilonidal) sinus. Ann R Coll Surg Engl 1984;66:201-3.  Back to cited text no. 4
    
5.
Nunes LF, Castro Neto AK, Vasconcelos RA, Cajaraville F, Castilho J, Rezende JF, et al. Carcinomatous degeneration of pilonidal cyst with sacrum destruction and invasion of the rectum. An Bras Dermatol 2013;88:59-62.  Back to cited text no. 5
    
6.
Karadaǧ EÇ, Toy H, Tosun Z. Lateral orbital wall destruction due to pilonidal sinus. J Craniofac Surg 2016;27:e461-2.  Back to cited text no. 6
    
7.
Mohanna PN, Al-Sam SZ, Flemming AF. Subungual pilonidal sinus of the hand in a dog groomer. Br J Plast Surg 2001;54:176-8.  Back to cited text no. 7
    
8.
Gordon KJ, Hunt TM. Osteomyelitis as a complication of a pilonidal sinus. Int J Colorectal Dis 2016;31:155-6.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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