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Home , Coccydynia

Ankylosing and a diagnostic dilemma: coccydynia R. Deniz1, G. Ozen2, S. Yilmaz-Oner2, S.Z. Aydin3, C. Erzik4, O.H. Gunduz5, N. Inanc2, H. Direskeneli2, P. Atagunduz2

1Faculty of Medicine, 2Department of , Marmara University, Istanbul, Turkey; 3Department of Rheumatology, Goztepe Training and Research Hospital, Istanbul, Turkey; 4Medical Biology, and 5Physical Therapy and Rehabilitation, Faculty of Medicine, Marmara University, Istanbul, Turkey.

Abstract Objective Coccydynia is defined as in or around the tail bone area. The most common cause of coccydynia is either a trauma such as a fall directly on to the or repetitive minor trauma. The etiology remains obscure in up to 30% of patients. The literature on the contribution of rheumatic diseases to coccydynia is scarce. Our objective was to investigate the prevalence of coccydynia in (AS) patients.

Methods One hundred and seven consecutive patients with AS were evaluated for coccydynia were enrolled between January and November 2012 for a cross-sectional analysis. Seventy-four consecutive patients were followed for mechanical as controls and the AS patients were interviewed for the presence of coccydynia. The data collected was evaluated on SPSS® version 11.5 and Microsoft Excel® Programmes.

Results Prevalence of coccydynia in AS (38.3%) was significantly higher than the control group (p<0.0001) in both female and male AS patients (female AS vs. control=40.9% vs. 18.4%, p=0.015 and male AS vs. control=36.5% vs. 8.0%, p=0.005). Both genders were affected equally in the AS group whereas coccydynia was slightly more frequent in female patients in the control group.

Conclusion Coccydynia is a previously neglected symptom of AS and it is almost three times more common in AS than in non-specific chronic . Our observation may implicate that inflammatory diseases have a role in the etiology of coccydynia, especially in those without a history of recent or past trauma and coccydynia may be a factor associated with the severity of AS as well.

Key words coccydynia, ankylosing spondylitis, coccyx,

Clinical and Experimental Rheumatology 2014; 32: 194-198. Coccydynia in ankylosing spondylitis / R. Deniz et al.

Rabia Deniz, MD Introduction with preexisting back pain. Attempts Gulsen Ozen, MD Coccydynia (coccygalgia or coccygo- to attribute coccydynia to lumbar disk Sibel Yilmaz-Oner, MD dynia) is defined as pain in or around hernia is not always justified since only Sibel Zehra Aydin, Assoc. Professor the tail bone area (oscoccygis; coccyx). a small proportion of patients (≈25%, Can Erzik, Assist. Professor Osman Hakan Gunduz, Professor The term coccydynia was first used by 13/50) with chronic low back pain have Nevsun Inanc, Assoc. Professor Simpson (1) in 1859, but descriptions of been found to have nerve compression Haner Direskeneli, Professor pain in the terminal portion of the spine and only a few of these patients had Pamir Atagunduz, Professor date back to at least 16th and 17th centu- nerve root irritation syndromes when Please address correspondence to: ries (2-5). However, despite being rec- studied specifically (13). Rabia Deniz, MD, ognised for many centuries, coccydynia The literature on the contribution of Faculty of Medicine, remains an unsolved mystery because rheumatic diseases to coccydynia is Marmara University, of the perceived uncertainty in identify- scarce. In 1959, Hart and Robinson re- Pendik ing the origin of the pain (1). The most ported coccydynia as the initial symp- 34899 Istanbul, Turkey. E-mail: [email protected] common cause of coccydynia is either a tom in 3% of female ankylosing spon- Received on May 8, 2013; accepted in trauma such as a fall directly on to the dylitis (AS) patients (14). Recent pub- revised form on November 12, 2013. coccyx or due to repetitive minor trau- lications with MRI of the coccyx de- ma that occurs due to sitting awkwardly fined in the form of bone © Copyright Clinical and Experimental Rheumatology 2014. such as during the post-partum period oedema and bursitis in adolescents and (6). Maigne et al. (7) reported that in adults (15, 16). Bone oedema and bur- a series of 51 patients 36 had a history sitis on MRI are common findings asso- of direct trauma, and Pennekamp et al. ciated with inflammation of any nature (8) reported a 50% incidence of direct including inflammatory spine diseases trauma. Apart from those cases caused such as AS. by local the etiology remains Anatomically, os coccyx is the con- obscure in up to 30% of patients (5, 9, tinuum of the . Joints 10). Recently, a more precise classifica- between sacrum, coccyx and segments tion of coccydynia based on etiology of coccyx are fibrocartilaginous in na- and pathology has been reported in an ture and are comparable to those seen attempt to include rare causes of coc- in higher intervertebral spaces. The cydynia (Table I) (1). anterior and posterior sacrococcygeal Coccygeal disorders that could be man- ligaments are the continuations of the ifested in coccydynia are (frac- anterior and posterior longitudinal ture, subluxation, luxation), abnormal ligaments that stretch along the entire mobility (hypermobility, anterior and spine. The anterior sacrococcygeal lig- posterior subluxation or luxation of the ament attaches to the front of the first coccyx), disc degeneration at sacrococ- and sometimes the second coccygeal cygeal (SC) and intercoccygeal (IC) vertebral bodies, blending superiorly segments, coccygeal spicule (bony ex- with the termination of the anterior lon- crescence), osteomyelitis and tumors. gitudinal ligament (17). Abnormal mobility of coccyx, which Taken together, the close anatomical can be seen on dynamic radiograph (lat- resemblance, recent MRI findings and eral x-rays of the coccyx in the standing the observation of coccydynia in some and sitting position), is the most com- of the AS patients followed at our out- mon pathological finding in patients patient rheumatology clinic led to the with coccydynia (70%). It can be a following question; what is the contri- result of injury and chronic static and bution of ankylosing spondylitis to the dynamic overload of the coccyx (obe- etiology of coccydynia? The aim of sity, prolonged sitting, , rowing, this study was to investigate the preva- riding, etc.) (9). lence of coccydynia in AS patients. The association of chronic non-specific low back pain with coccydynia has been Methods reported repeatedly in the literature. Patients and data collection Postacchini and Massobrio (11) report- - Study patients ed that 31% of their patients had coc- One hundred and seven consecutive cydynia-associated low back pain and patients with AS diagnosed according in a different study of Bayne et al. (12) to the modified New York criteria (18) Competing interests: none declared. 15% of coccydynia patients presented were enrolled in the study between

195 Coccydynia in ankylosing spondylitis / R. Deniz et al.

Table I. Classification of coccydynia based on etiology and pathology. bone area” of the study patients. The expected marking site of patients is A: Based on etiology 1. Idiopathic shown in Figure 1b. ® 2. Traumatic Collected data was evaluated on SPSS ® B: Based on pathology version 11.5 and Microsoft Excel Pro- 1. Degeneration of the sacrococcygeal and intercoccygeal disc and grammes. Data for patients were calcu- joints lated as mean and standard deviations 2. Morphology of the coccyx: type II, III, IV, presence of a bony for continuous variables and as frequen- spicule and coccygeal retroversion 3. Mobility of the coccyx: hypermobile or posterior subluxation cies and percentages for categorical variables. The gender-based differences 4. Referred pain: lumbar pathology or arachnoiditis of the sacral nerve were analysed with χ2 and Student’s t- roots, spasm of the pelvic floor muscles and inflammation of the test. For all analyses significance level pericoccygeal soft tissues 5. Others: , crystal deposits, infections of p-value was accepted as 0.05. C: Somatisation or neurotic Results One hundred and seven AS and 74 pa- tients with mechanical back were in- cluded. Demographic features of AS and control participants are presented in Table II. A substantial proportion of our study patients with AS, but not with non-inflammatory chronic low back pain, defined cocydynia when questioned specifically. Prevalance of coccdynia in AS was 38.3% and was significantly higher in both male and female patients compared to the control group (41/107 in AS vs. 11/74 in control group, p<0.0001, OR: 2.58; CI:95%; 1.42-4.68). Both genders were affected equally in the AS group whereas coc- cydynia was slightly more frequent in female patients in the control group Fig. 1. a. Illustration given to patients to show area of coccydynia. b. Expected area to be marked by (23/63 male AS vs. 18/44 female AS, patients as the area of coccydynia. p=0.397; 2/25 male control vs. 9/49 fe- male control, p=0.204) (Fig. 2). January 2012 and November 2012 for in the control group were excluded (19). The comparison of coccydynia pres- a cross-sectional analysis. The University of Marmara Institution- ence according to gender shows sig- al Review Board approved the study, nificance for AS and control patients - Control group and all study patients gave informed in both male (23/63 of male AS vs. Seventy-four consecutive patients fol- consent. 2/25 of male control; p=0.005; OR: lowed for mechanical back pain (main- 4.56; CI:95%,1.16–17.93) and female ly for lumbar hernia, and Data collection patients (18/44 of female AS vs. 9/49 lumbar strain) at the outpatient clinic of Patients were interviewed face to face of female control; p=0. 015; OR: 2.27; the Department of Physical Medicine using a questionnare consisting of nine CI:95%; 1.18–4.43). and Rehabilitation, Marmara Univer- questions regarding demographic data, In AS patients, disease activity assessed sity Faculty of Medicine were enrolled disease onset and age at diagnosis, cur- by Bath AS disease activity index to study as controls. rent medications and the presence of (BASDAI) and treatment with biologic coccydynia. Figure 1a demonstrates agents were compared for coccydynia - Exclusion criteria the visual question used to point out the presence. When each individual do- Patients with a history of trauma or cur- location of pain by the patient. How- main of BASDAI was evaluated for the rent diagnosis of inflammatory bowel ever, in order not to bias the results, affect of coccydynia, the question about disease or psoriasis among study pa- the tail bone area was not shown on the neck, back or hip pain had signifi- tients and patients fulfilling the modi- purpose. Patients reporting coccydynia cantly higher score in patients with coc- fied New York criteria (19) or patients on questioning were asked to circle the cydynia (Table III). Overall BASDAI fulfilling the AS Assessment Study area of pain on the figure to avoid any score was also higher in this subset of (ASAS) inflammatory back pain criteria possible misconception about the “tail patients.

196 Coccydynia in ankylosing spondylitis / R. Deniz et al.

tests and interventions. A simple clas- sification based solely on etiology is practical in guiding the management but leaves a substantial proportion of cases without a diagnosis. Contribution of rather infrequent causes, such as in- flammatory diseases, on to the etiology of coccydynia is scarcely addressed in the literature. A considerable propor- tion of our study patients with AS – but not with non-inflammatory chronic low back pain – defined cocydynia when questioned specifically. This ob- Fig. 2. Comparative proportions of patient with coccydynia. servation suggests that cases classified previously as “idiopathic” should care- Table II. Demographics of the study patients. fully be questioned for AS (1). Ankylosing spondylitis presents with AS Group Control Group chronic inflammatory back pain (IBP) Female Male Total Female Male Total early in life, generally when patients n, (%) n, (%) n, (%) n, (%) n, (%) n, (%) are in their teens or early to mid-20s (20) and causes considerable functional Gender 44 (41.1) 63 (58.9) 107 49 (33.8) 25 (66.2) 74 Median age* 39 37 38 45 36 43 disability and also the socio-economic Mean age* 38.56 39.35 39.03 46.2 39.1 43.8 impact for the patients, the healthcare Time to diagnosis* 6.47 4.86 5.85 5.0 0.84 3.5 system and the society (21). This age of * Age at diagnosis 34.3 32.8 33.4 42.2 38.3 40.8 presentation overlaps with that of coc- *in years. cydynia, which also commonly occurs in adolescents and young adults (15, Table III. Evaluation of individual BASDAI domains according to coccydynia presence. 22). Unfortunately, during the “pre- radiographic” phase, which on average BASDAI question Coccydynia Coccydynia p-value might last 5 to 10 years or longer, AS present absent remains clinically unrecognised (20, How would you describe the overall level of fatigue/ 5.57 4.59 0.110 23). The absence of radiographic sacro- tiredness you have experienced? iliitis may lead to a possible mislabeling How would you describe the overall level of AS neck, 6.02 4.57 0.019 of cases especially without an acute or back or hip pain you have had? past trauma history as “idiopathic” or “chronic nonspecific back pain” (13). How would you describe the overall level of pain/swelling 3.75 3.18 0.373 in joints other than neck, back or hips you have had? The association of coccydynia with AS is rarely addressed in the literature. How would you describe the overall level of discomfort 4.30 2.93 0.054 you have had from any areas tender to touch or pressure? Lately, Maigne et al. (16) reported a series of MR imagings in patients How would you describe the overall level of morning 4.75 4.04 0.290 stiffness you have had from the time you wake up? with chronic coccydynia. In 70 of the 172 cases (40.7%), the vertebral end- How long does your morning stiffness last from the time 3.70 2.44 0.057 plates of the sacrococcygeal or inter- you wake up? coccygeal disc showed a hyperintense Final BASDAI score 4.77 3.70 0.020 signal on T2 and isointense signal on T1-weighted images. In the majority Although not significant, the need for cydynia resolves either spontaneously of these cases (38 out of 70), the image anti-tumour necrosis factor α (Anti- or with supportive care over weeks to was non-homogeneous, moderately in- TNF α) therapy seemed also higher in months. But sometimes it is refractory tense, poorly delimited and sometimes patients with coccydynia, suggesting to treatment and may debilitate the pa- associated with a slightly increased in- a higher disease severity (31.8% vs. tient. tradiscal signal on T2. These changes 22.2%, p=0.20). Most publications on coccydynia are were interpreted as oedema due to joint case reports, case series, or reviews, inflammation, by analogy with the situ- Discussion and there are few extensive research ation described by Modic et al. for the Coccydynia (coccygodynia or pain studies on etiology. Persistent coc- lumbar spine (24). Pathogenesis and in the tailbone) is a common, painful cydynia may lead to unnecessary and association of these MRI changes are condition that is easily diagnosed. Coc- often not evidence-based diagnostic often not well understood yet (16).Until

197 Coccydynia in ankylosing spondylitis / R. Deniz et al. recently, imaging of the painful coccyx tion of AS patients with coccydynia im- coccyx. J Bone Joint Surg (Am) 1983; 65-A: relied on standard and dynamic radiog- plicates that inflammatory diseases may 1116-24. 12. BAYNE O, BATEMAN JE, CAMERON HU: The raphy. Recent imaging techniques pro- have a role in the etiology especially in influence of etiology on the results -of coc vided important additional data on the those without a history of recent or past cygectomy. Clin Orthop 1984; 190: 266-72. inflammation of the coccyx (16). MRI trauma. 13. WRAY CC, EASOM S, HOSKINSON J: Coccy- dynia: aetiology and treatment. J Bone Joint findings such as bone edema and bur- Surg (Br) 1991; 73: 335-8. sitis around the coccyx could provide Acknowledgements 14. HART FD, ROBINSON KC: Ankylosing spon- additional information on the cause We would like to thank our valuable dylitis in women. 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