retropharyngeal calcific tendinitis: case report and review of the literature

6
Retropharyngeal Calcific Tendinitis: Case Report and Review of the Literature Richard Park, MD,* Daniel E. Halpert, DO, Alan Baer, MD, ‡,§ Dario Kunar, MD, FACS, and Peter A. Holt, MD Objectives: Retropharyngeal calcific tendinitis (RCT) is an under-recognized benign condition that results in significant neck pain and may mimic a retropharyngeal abscess (RPA). We describe the clinical presentation, diagnosis, and treatment of RCT as well as features that differentiate it from RPA. Methods: We present a case report and analyze the clinical features, diagnosis, and treatment of 71 additional patients with RCT identified through a PubMed literature review between 1964 and early 2008. We then compared these findings with those of RPA. Results: The most common symptoms of RCT at presentation were neck pain (94%), limited range of motion (45%), odynophagia (45%), neck stiffness (42%), dysphagia (27%), sore throat (17%), and neck spasm (11%). Other frequent findings include low-grade fever, mild leukocyto- sis, and a slightly elevated erythrocyte sedimentation rate. Seventy-five percent of patients with RPA present with similar symptoms and cervical radiographic abnormalities are comparable in the majority of cases with either pathology. Conclusions: RCT frequently mimics the clinical features of RPA and recognizing the key symptoms and signs of RCT versus RPA can be challenging but important in avoiding unnecessary interventions. We recommend that computed tomography of the neck be considered as a first step in differentiating the 2 conditions. The presence of an amorphous calcification anterior to the C1 and/or C2 vertebral body(s) with a non-ring-enhancing fluid collection in the prevertebral space should be considered highly suspicious for RCT. RCT can be self-limiting and will usually resolve in 2 weeks. Effective treatment typically consists of nonsteroidal anti-inflammatory drugs, steroids, or opiate analgesics. © 2010 Elsevier Inc. All rights reserved. Semin Arthritis Rheum 39:504-509 Keywords: retropharyngeal, tendinitis, abscess, pain, neck R etropharyngeal calcific tendinitis (RCT) is a rare, under-recognized benign condition in which cal- cium hydroxyapatite deposition in the longus colli tendon is postulated to induce acute inflammation of the longus colli muscle tendon insertion (1). The longus colli muscle is 1 of the 4 that comprise the anterior vertebral muscle group. The clinical features of RCT were first described by Hartley in 1964 and include the acute or subacute onset of pain and stiffness in the neck with odynophagia associated with prevertebral soft-tissue swelling and amorphous calcification anterior to the body of the second cervical vertebrae (2). The disease may mimic more serious causes of acute neck pain such as retropharyngeal abscess (RPA), meningitis, traumatic in- jury, and cervical myelopathy. We report a case of RCT and review the features of 71 other cases identified through a review of the literature. METHODS Seventy-one cases published in 30 journal articles from English, Swedish, and French sources between 1964 and early 2008 were identified via a PubMed literature search (1-30). “Retropharyngeal calcific tendinitis” and “acute *Resident, Department of Internal Medicine, Good Samaritan Hospital, Baltimore, MD. †Resident, Department of Physical Medicine and Rehabilitation, Johns Hopkins University School of Medicine, Baltimore, MD. ‡Associate Professor of Medicine, Johns Hopkins University School of Medicine, Baltimore, MD. §Director, Johns Hopkins University Clinical Practice, Good Samaritan Hospital, Chief of Rheumatology, Good Samaritan Hospital, Baltimore, MD. ¶Department of Otolaryngology, Greater Baltimore Medical Center, Balti- more, MD. The work should be attributed to the: Department of Rheumatology at the Good Samaritan Hospital, Baltimore, MD, USA. The authors have no conflicts of interest to disclose. Address reprint requests to Peter A. Holt, MD, Johns Hopkins University School of Medicine, Good Samaritan Hospital, Russell Morgan Building, 5601 Loch Raven Blvd., Suite 509, Baltimore, MD 21239. E-mail: [email protected]. MISCELLANEOUS 504 0049-0172/10/$-see front matter © 2010 Elsevier Inc. All rights reserved. doi:10.1016/j.semarthrit.2009.04.002

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MISCELLANEOUS

5

Retropharyngeal Calcific Tendinitis: Case Report andReview of the Literature

Richard Park, MD,* Daniel E. Halpert, DO,† Alan Baer, MD,‡,§

Dario Kunar, MD, FACS,¶ and Peter A. Holt, MD‡

Objectives: Retropharyngeal calcific tendinitis (RCT) is an under-recognized benign condition thatresults in significant neck pain and may mimic a retropharyngeal abscess (RPA). We describe the clinicalpresentation, diagnosis, and treatment of RCT as well as features that differentiate it from RPA.Methods: We present a case report and analyze the clinical features, diagnosis, and treatment of 71additional patients with RCT identified through a PubMed literature review between 1964 andearly 2008. We then compared these findings with those of RPA.Results: The most common symptoms of RCT at presentation were neck pain (94%), limitedrange of motion (45%), odynophagia (45%), neck stiffness (42%), dysphagia (27%), sore throat(17%), and neck spasm (11%). Other frequent findings include low-grade fever, mild leukocyto-sis, and a slightly elevated erythrocyte sedimentation rate. Seventy-five percent of patients withRPA present with similar symptoms and cervical radiographic abnormalities are comparable in themajority of cases with either pathology.Conclusions: RCT frequently mimics the clinical features of RPA and recognizing the key symptomsand signs of RCT versus RPA can be challenging but important in avoiding unnecessary interventions.We recommend that computed tomography of the neck be considered as a first step in differentiatingthe 2 conditions. The presence of an amorphous calcification anterior to the C1 and/or C2 vertebralbody(s) with a non-ring-enhancing fluid collection in the prevertebral space should be consideredhighly suspicious for RCT. RCT can be self-limiting and will usually resolve in 2 weeks. Effectivetreatment typically consists of nonsteroidal anti-inflammatory drugs, steroids, or opiate analgesics.© 2010 Elsevier Inc. All rights reserved. Semin Arthritis Rheum 39:504-509Keywords: retropharyngeal, tendinitis, abscess, pain, neck

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etropharyngeal calcific tendinitis (RCT) is a rare,under-recognized benign condition in which cal-cium hydroxyapatite deposition in the longus colli

endon is postulated to induce acute inflammation of theongus colli muscle tendon insertion (1). The longus colli

Resident, Department of Internal Medicine, Good Samaritan Hospital, Baltimore, MD.Resident, Department of Physical Medicine and Rehabilitation, Johns Hopkinsniversity School of Medicine, Baltimore, MD.Associate Professor of Medicine, Johns Hopkins University School of Medicine,altimore, MD.Director, Johns Hopkins University Clinical Practice, Good Samaritan Hospital,hief of Rheumatology, Good Samaritan Hospital, Baltimore, MD.Department of Otolaryngology, Greater Baltimore Medical Center, Balti-ore, MD.The work should be attributed to the: Department of Rheumatology at the Good

amaritan Hospital, Baltimore, MD, USA.The authors have no conflicts of interest to disclose.Address reprint requests to Peter A. Holt, MD, Johns Hopkins University School of

(edicine, Good Samaritan Hospital, Russell Morgan Building, 5601 Loch Ravenlvd., Suite 509, Baltimore, MD 21239. E-mail: [email protected].

04 0049-0172/10/$-see front matter © 2010 Elsevier Inc. All rights reserved.doi:10.1016/j.semarthrit.2009.04.002

uscle is 1 of the 4 that comprise the anterior vertebraluscle group. The clinical features of RCT were first

escribed by Hartley in 1964 and include the acute orubacute onset of pain and stiffness in the neck withdynophagia associated with prevertebral soft-tissuewelling and amorphous calcification anterior to the bodyf the second cervical vertebrae (2). The disease mayimic more serious causes of acute neck pain such as

etropharyngeal abscess (RPA), meningitis, traumatic in-ury, and cervical myelopathy. We report a case of RCTnd review the features of 71 other cases identifiedhrough a review of the literature.

ETHODS

eventy-one cases published in 30 journal articles fromnglish, Swedish, and French sources between 1964 andarly 2008 were identified via a PubMed literature search

1-30). “Retropharyngeal calcific tendinitis” and “acute

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etropharyngeal calcific tendinitis” were used as searcherms. For each individual case, the following specific de-ails were abstracted: epidemiological characteristics (ie,ge, gender, race, past medical history), chronological de-ails (ie, time to diagnosis, number of attacks, duration ofttack, and number of recurrences), chief complaint onresentation, symptoms (ie, neck pain, limited range ofovement, neck stiffness, sore throat, and odynophagia),

bjective findings (ie, temperature, head and neck exam-nation, and presence of lymphadenopathy), laboratoryndings (ie, white blood cell count with differential, he-atocrit, erythrocyte sedimentation rate, C-reactive pro-

ein, serum calcium, serum phosphate, uric acid, bloodulture results, and throat culture results), imaging find-ngs (ie, radiograph of the neck, computed tomographyCT] scan of the neck, and magnetic resonance imagingMRI] of the neck), and treatment. These details werentered into a Microsoft Excel spreadsheet. The key fea-ures of RCT were then tabulated and compared with thosef RPA, with which it is often confused, as detailed in aimilar review of the literature (31).

ASE REPORT

30-year-old woman presented with a 1-week history ofevere bilateral neck pain and odynophagia. Her range ofotion was significantly compromised by increased pain

ssociated with movement in any direction. The patientas unable to sleep at night because of the neck pain. Thereas no associated fever, recent illnesses, or head or neck

rauma. On review of systems, she reported Raynaud’s phe-omenon and morning stiffness. She rated her pain on a

igure 1 MRI scan of the neck of our patient. Notice the fluidollection measuring 5 � 0.7 cm anterior to the cervicalpine at the C2-C5 vertebral bodies.

isual analog pain scale as 10 of 10. a

Her prior medical history was remarkable for tonsillec-omy, sinus surgery, acute maxillary sinusitis, and chronicnovulation.

On examination, vital signs were normal. She had se-ere neck stiffness, pain with motion in all directions, andarked tenderness along her posterior neck, as well as her

ternocleidomastoid and trapezius muscles. There waslight erythema of the posterior pharynx but no evidencef airway obstruction, vocal changes, pooling of secre-ions, or obvious cervical lymphadenopathy. The remain-ng examination was normal.

Laboratory findings revealed a hematocrit of 38%, ahite blood cell count of 10,000 mm3, and a platelet

ount of 175,000 mm3. Complete metabolic profile wasormal; creatine phosphokinase level was 63, and thy-oid-stimulating hormone level was 1.0. Pregnancy testas negative and urinalysis was normal. An MRI of the

ervical spine, ordered to rule out impingement on thepinal cord or nerve roots, revealed a 5 � 0.7 cm fluidollection anterior to the cervical spine at the C2-C5 ver-ebral bodies (Fig. 1). An otolaryngology specialist re-uested a CT scan of the neck. This revealed calcificationnterior to the base of the odontoid process with no evi-ence of abscess (Fig. 2). These findings, along with a lackf evidence of toxemia, confirmed the diagnosis of RCT.he patient was successfully treated with 60 mg oral pred-isone per day tapered off over a 2-week period followedy 800 mg oral ibuprofen up to 3 times per day as neededor pain.

ESULTS

eventy-one patients with RCT were identified in oureview of the literature (Table 1). Their ages ranged from1 (23) to 65 (1,8,29) with an average of nearly 42 years.orty-two patients (59%) with RCT were women and 2941%) were men. No race or ethnicity was overrepre-ented. With descriptors such as “a few days” or “severalays” omitted, the time to diagnosis was fewer than 7 days

igure 2 CT scan of the neck of our patient. Notice the smallrea of retropharyngeal calcification anterior to the base ofhe odontoid process with a small area of adjacent swelling

nterior and inferior to this region.

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n 82% of patients and ranged from “within hours” (11)o “several months” (4). The most common symptoms atresentation were neck pain (94%), limited range of neckotion (45%), odynophagia (45%), neck stiffness (42%),

ysphagia (27%), sore throat (17%), and neck spasm (11%)Fig. 3). On occasion pharyngeal edema (1,5), erythema1,19), and occipital pain (5,6,18,22,23) were noted. No

Table 1 Summary of Clinical Findings

Patients inLiteratureReview

PercentPositive

Age range (yr) 21 to 65Male 29/71 41Female 42/71 59Time to diagnosis, �7 days 46/56 82Time to diagnosis, �7 days 10/56 18Symptom duration following

treatment, �7 days31/47 66

Symptom duration followingtreatment, 7 to 14 days

13/47 28

Symptom duration followingtreatment, �14 days

3/47 6

Neck pain 67/71 94Limited ROM 32/71 45Odynophagia 32/71 45Neck stiffness 30/71 42Fever (T � 38°C) 13/37 35Dysphagia 19/71 27Sore throat 12/71 17DDx: Retropharyngeal abscess 13/71 18Total white blood cell count

�10,80022/31 71

Increased ESR 13/23 57

DDx, differential diagnosis.

igure 3 Comparison of frequency of the most commonymptoms noted at presentation among 71 patients diag-

rosed with retropharyngeal calcific tendinitis.

ommon recent past medical history was apparent, al-hough repetitive use of the neck muscles (9,26) and re-ent trauma (3) were reported in a small number of pa-ients. Previous occurrence of similar symptoms wasoted in 3 cases (3,15,17). Of those whose temperatureas documented (n � 37), slightly over 1/3 had fever,efined by a temperature of �38°C. The highest recordedemperature was 38.9°C (17).

White blood cell counts had an average of 12,089 mm3

ith a range of 8100 mm3 (1) to 15,800 mm3 (25). Theyere greater than or equal to 10,800 mm3 in 71% of the1 values documented. Erythrocyte sedimentation rateas elevated in 57% of the 23 patients in whom it wasentioned and ranged from 5 (30) to 91 (1). On cervical

adiography (n � 62) soft-tissue swelling or thickening inhe prevertebral or retropharyngeal space was the mostommon finding (92%), followed by calcifications (68%)nd radiodensity (8%) anterior to C1 and/or C2 vertebralody(s). On CT (n � 37), the most common finding wasalcifications in the cervical prevertebral space (89%).his was followed by soft-tissue swelling (38%) and effu-

ions or fluid collections (24%) (Fig. 4). MRI was re-orted 27 times and, while a change in signal intensity wasoted in 70% of cases, calcifications were specifically doc-mented less than 30% of the time. This was followed bydema (26%), effusions or fluid collections (15%), soft-issue swelling or thickening (11%), and inflammation4%). Follow-up imaging at 1 year showed complete re-orption of calcification and total resolution of the prever-ebral soft-tissue swelling on repeat CT and MRI in 1 caseeport (16).

The most common treatment methods entailed the usef nonsteroidal anti-inflammatory drugs (NSAIDs), ste-

igure 4 Comparison of imaging study findings of the pre-ertebral space anterior to the C1-C2 vertebral bodies. Theighter colored columns are plain radiograph findings andhe darker columns are CT findings.

oids, or opiate analgesics. Time to relief following treat-

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ent was recorded for all numeric values (eg, relief notedn 3 days), while ambiguous references (eg, shortly there-fter (5) and several days (4)) were omitted. Phrases suchs “prompt clinical improvement” (7), rapid regression28), and “a few days” (10,14) were documented as lesshan 7 days. Thirty-one patients had documentation oflleviation of symptoms within a 7-day period, 13 pa-ients between 7 and 14 days, and 3 greater than 14 days.he quickest documented relief occurred within 6 hoursith the use of 10 mg intravenous dexamethasone (4).owever, by 14 days, for all patients that treatment and

ymptom relief were reported, nearly all were asymptom-

igure 5 Comparison of amount of days until resolution ofymptoms based on treatment with either NSAIDs, steroids,r opioid analgesics in patients suffering from retropharyn-eal calcific tendinitis. The highest and lowest part of theertical line represents the range of days until resolution ofymptoms, while the dot reflects the average amount of daysntil resolution of symptoms.

Table 2 Comparison of Retropharyngeal Abscess and Retro

Age range (average), yrGender M/FPresented with sore throat, dysphagia,

odynophagia, or neck painPresented with total or partial obstructive

airway symptomsNumber of patients with significant comorbidity 24

Most common comorbidity Di

Lateral neck radiographs positive forretropharyngeal swelling

46

Cervical spine osteomyelitis presentHistory of preceding procedure or impacted

foreign bodyDeath as outcome

Percentages in parentheses except for age.

tic or very close to it regardless of which method wastilized (Fig. 5). Additionally, the greater than 14 daysategory is most likely overstated as 2 (4) of the 3 (4,18)atients were found to be asymptomatic on follow-up andere without documentation as to at which point the

ymptomatic period terminated (4).The cardinal features of RCT, as determined from our

eview of the literature, are compared with those of RPAn Table 2. These 2 conditions were similar in terms ofheir clinical presentations and the finding of retropha-yngeal soft-tissue swelling on lateral neck radiographs. Inontrast to patients with RCT, those with RPA often hadn associated comorbid condition, such as diabetes mel-itus, malignancy, or alcoholism, or had a history of arior procedure or impacted foreign body.

ISCUSSION

CT is a benign, self-limited, inflammatory condition ofhe longus colli tendon thought to be a form of calciumydroxyapatite deposition disease (1,6). The longus colliendon is a very uncommon location for this disease as itypically involves large joints such as the hips and shoul-ers (6). The longus colli muscle runs along the anteriorurface of the vertebral column in the prevertebral andetropharyngeal space. Its most inferior point originatesrom the third thoracic vertebrae and it inserts in part athe second cervical vertebral level. It is at this insertionoint where a foreign body inflammatory response to de-osited crystals of hydroxyapatite has been documentedy biopsy (1). The presumed mechanism of RCT is ancute, inflammatory tendinitis engendered by calcium hy-roxyapatite crystals that have deposited in the longusolli muscle tendon at its attachment to the anterior tu-ercle of the C-1 vertebrae. The dystrophic calcificationn this condition is thought to have genetic and metabolic

ngeal Calcific Tendinitis

haryngeal Abscess (31)(N � 51)

Retropharyngeal CalcificTendinitis (N � 71)

18 to 81 (49) 21 to 65 (42)33 (65)/18 (35) 42 (59)/29 (41)

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omponents. Additionally, risk factors such as repetitiverauma, recent injury, ischemia, inflammation, or tissueecrosis appear to play a role (6). These disturbances may

ead to increased solute concentration and cause the lossf local inhibitors of crystal growth and the presence ofbnormal surfaces, all of which may promote crystal nu-leation (9). Crystal deposits, which at first are contained,ltimately rupture and may provoke a painful inflamma-ory response (16).

The causes of neck pain are extensive. Moreover, theifferential diagnosis of RCT includes cervical spondy-

otic myelopathy, meningitis, neoplasia, and RPA. It ismportant to note that the relatively benign condition ofCT is often mistaken for RPA (1,4,5,7,9,17,22,24,28)s 75% of patients with RPA present with the similarymptoms of neck pain, sore throat, dysphagia, ordynophagia that is common in RCT (31). Additionally,he vast majority of all patients with either pathologyresent with soft-tissue swelling in the retropharyngeal orrevertebral space on cervical radiographs (Table 2).hus, it is imperative to establish the correct diagnosis as

he approach to treatment of RCT versus RPA differsramatically. The latter requires prompt intravenous an-ibiotic management and a likely surgical incision andrainage, while a brief course of NSAIDs for the formerill suffice. Reviewing the findings on imaging studies

an help differentiate the 2. The presence of an amor-hous calcification anterior to C1-C2 with a non-ring-nhancing fluid collection should be considered highlyuspicious for RCT.

In conclusion, recognizing the key symptoms and signsf RCT versus other more serious differential diagnosesuch as RPA can be challenging but important in avoidingnnecessary interventions. CT can be of great aid in mak-

ng the diagnosis of RCT and we recommend this as therst-line imaging modality for several reasons. CT iseadily available, less expensive than MRI, and easily ac-essible in an emergency department setting where manyatients suffering from RCT will present. It is appropriateor distinguishing bony abnormalities such as fracturesnd calcifications from the ring-enhancing lesions of fluidollections found with abscesses. From our literature re-iew, neither radiographs nor MRI were as sensitive as CTn revealing prevertebral calcifications at C1-C2, a findinghich may be pathognomonic for RCT. Although 92%f cervical radiographs were significant for soft-tissuewelling, fluid collections were never noted (Fig. 4); thisakes abscess identification difficult. This point cannot

e overstated as soft-tissue swelling is a ubiquitous findingor both RCT and RPA.

Despite the characteristic calcifications that may clinchhe diagnosis of RCT, consultation with an otolaryngol-gist should always be considered to corroborate this di-gnosis, especially in the setting of fever and leukocytosis.nless the need for rehydration or control of refractoryain exists, admission is rarely necessary. However, early

utpatient follow-up at frequent intervals is imperative to

onitor for clinical deterioration that would signal con-ideration of other potential diagnoses such as RPA.SAIDs should be considered the mainstay of treatment

or RCT but when quick relief is desired a brief course ofteroids can be given. More studies are needed to elucidatehe exact causative risk factors and pathogenesis of RCT.

CKNOWLEDGMENT

he authors thank Vicente Maco-Flores, MD for supportn making this article possible.

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