ABSTRACT

 Objective: To discuss the case of a 12-year-old female presenting with a mass in the right submandibular region consulting for chiropractic evaluation.

Clinical features: A 12-year-old female, accompanied by her mother presented for chiropractic consultation regarding a diagnosis of cystic hygroma. Her mother stated that a mass had developed in her daughter's right submandibular muscle at approximately 5 years of age. Since the age of nine she had four surgical procedures in an effort to resolve the mass and the severe sinus drainage she experienced. All surgical procedures had failed to relieve her severe sinus drainage or contain the development of the mass. The mass was approximately the size of a golf ball on her first visit to the chiropractor. The subject has also suffered from daily morning neck pain and headaches for the past year.

Intervention and Outcome: Following seven months of conservative chiropractic care, the patient's mass completely remissed and has not recurred during 2 years of wellness care. The patient's neck pain and headaches are also completely resolved. She also can breathe with ease allowing her to participate in active sports {tennis and cross country) whereas she had been limited to minimal athletic activity in the past due to her condition.

Conclusion: A case report of a 12-year-old female with cystic hygroma is presented. Integral to her condition is the physical exam finding of significant left C2 lamina pedical point tenderness and associated paravertebral muscle spasms resulting in loss of vertebral motoricity of the cervical vertebral units. The care and treatment was approached with the hypothesis that reduction of the subluxation complex of the upper cervical region may result in improved sinus function and or lymphatic drainage. The successful resolution of the patient's cystic hygroma while under chiropractic care indicates the need for further study of the role of chiropractic care in the case of non-musculoskeletal conditions.

Key Indexing terms: Cystic Hyltroma, Lymphangioma, Chiropractic, Orrhospinology

 INTRODUCTION

Cystic Hygroma (CH) a.k.a. Lymphangioma is a rare condition (incidence 1/12,000 births), considered congenital, that can be diagnosed in utero via ultrasound following at least 40 days gestation In general, 50% to 60% of cystic hygromas are apparent at birth, and 80%-90% are apparent by 2 years of age. Magnetic Resonance Imaging (MRI) provides the best diagnostic tool after birth. Cystic Hygroma represents <5% of all congenital neck masses.~.3.; The common locations of CH are:

   q       75% in the neck

q       20% in the axilla

q       5% in the retroperitoneum

q       2-3% of the cervical and axillary masses extend into the mediastinum.

Cystic hygroma is characterized in current literature as a faulty development of the lymphatic system during the development stages of the fetus. Presentation includes a sac-like bag of fluid. usually in the neck, with various studies citing left or right sided predominance. The sac can expand under the skin and is filled with lymph fluid and lymph cells. The most common complication is upper air- way obstruction and dysphasia. Management includes surgery for large and /or symptomatic masses and may require multiple surgeries.⁹
    Alternative treatments of CH in the past have included pressure garments, pump garments and massage with vary- ing degrees of success. Steroids have been shown to cause regression in selected cases. Sclerosis agents have been used with conflict due to major vascular structures and the brachial plexus. Radiation therapy has also been used in the past with complications oflymphangitis, lymphedema, and lymphangiosarcoma.^{7. 10, 11}
    A drug, OK-432, is in use in Japan as a Biological Response Modifier(BRM), typically for the treatment of lung cancer, neck and head cancer, alimentary tract cancer and thyroid cancer. This substance is injected into the cyst lumen replacing the aspirated lymph causing an inflam- matory response and is the first treatment of choice in Japan. Use of OK-432 in the United States is currently under consideration by the US FDA.⁸ However surgical extirpation has been the treatment of choice with mul- tiple surgeries anticipated.^{12, 13,14,15}
    Biomedical research is limited in the study of cystic hygroma for various reasons including lack of research due to the rarity of the condition.⁶

  CASE STUDY

History and Examination

This case study involves a 12-year-old Caucasian female whose mother accompanied her for a chiropractic evaluation. The history included a non-complicated gestation and delivery, with no apparent mass noted until approximately 5 years of age. The diagnosis given was probable thyroglossal duct cyst.
    The first surgical intervention was removal of her tonsils and adenoids at the age of nine. The patient stated that severe drainage continued post surgery and the mass remained the same. The patient reported that she typically used a large box of tissue daily.
    In 1995, a second surgery was performed since the mass had grown to the size of a tennis ball. During the surgery, the surgeon found what was later determined to be a cystic hygroma, and excided it. The mass redeveloped within one year and a third surgery was performed. The mass reappeared within six weeks of the third surgery.
    The patient's history of chronic sinusitis included medical treatment with multiple antibiotic regimes including Vantin and Flonase nasal spray. A fourth surgical intervention was performed in the summer of 1997 in an attempt to clear the sinus tissue that was chronically inflamed. This procedure also failed to alleviate the drainage or hygroma development.
    Subjective complaints included difficulty breathing due to severe sinus congestion during activity and the mass formation at the base of her jaw/throat region.
Physical examination revealed a 5' 2" 97 Ib. female with normal vital signs. Positive findings included tenderness of the left C2 lamina pedical region. Paraspincll spasm was noted on the left cervical region associated with loss of vertebral motion. Cervical range of motion was tested and revealed loss of right lateral flexion and right lateral rotation with pain. Remaining cervical and dorsolumbar ranges of motion were within normal limits.
Neurological testing of the deep tendon reflexes was essentially negative at a+2.
Inspection of the anterior throat revealed a mass approximately 4 cm in diameter in the right submandibular region. This was soft, non-tender and well demarcated on palpation.
    Cervical spine x-rays were taken and were negative for fractures, dislocatiori and/ or osseous pathology. A loss of cervical lordosis was noted with disc spaces appearing in- tact throughout. Orthospinology technique films (nasium/ vertex views) were analysed, with the following findings:

 q       Atlas-left I degree with 3 degrees of anterior rotation

 q       Axis-left I degree with 12 degrees rotation

 q       Lower cervical angle was measured at I degree to the right

 These vertebral positions and articular joint angles were used to determine a line of correction of the vertebral misalignments. The goal of chiropractic care was to re- store correct positioning of the cervical spine and to improve cervical vertebral movement.

 CHIROPRACTIC CARE

A chiropractic orthospinology adjustment was performed utilizing the Laney instrument contacting the left atlas transverse process in a side lying position. A reduction of the left C2 articular pillar pain was noted and an increase in range of motion with less pain at end point of movement. Neuromuscular therapy was performed for the posterior cervical. spinal musculature and the dorsolumbar paravertebral region.
   Chiropractic adjustments were administered three times weekly for four weeks, then twice weekly for two weeks. Treatment frequency diminished to 1 time weekly for 8 weeks and then to 2 times monthly for eight months.
Chiropractic care consisted of atlas specific adjustments via the orthospinology approach and neuromuscular therapy for posrerior paraspinal muscles.

 RESULTS

 A re-evaluation performed following six weeks of treatment showed the mass to be smaller and more flaccid. The patient remarked that she no longer awoke with neck and head pain daily. This remark of improvement was surprising to the mother since the patient had not previously mentioned neck or head pain. Upon further discussion, the patient related that she thought it was normal to awaken with those pains and simply took daily over the counter medication to deal with it. The patient also commented that lately she could breathe much easier and was beginning to play tennis without respiratory distress.
   After six months of chiropractic care the cystic hygroma was completely resolved and did not return in the months to follow. The patient also experienced a marked decrease in the frequency of colds. She was able to compete on the cross country track team and was delighted with her ability to breathe. The patient reported that prior to her treatment after running only one mile her sinus drainage would be so severe that it caused vomiting. The patient now stated that she could run 10 miles without respiratory complaints.

 DISCUSSION

 The pathophysiology of lymphatic function is not clearly understood. Cystic Hygroma is a rare condition that involves loss of function of the lymphatic system. Associated conditions can include Turner's Syndrome (XO chromosome characterized by a lack of sexual development), small stature, mental retardation, heart defects and various other inborn abnormalities. ¹
   One factor may be an impaired lymphatic pumping activity. Lymphatic vessels are not considered to be vessels at all. These vessels are not passive conduits, they contract and generate the energy to propel lymph. Studies have shown this pressure to be measured at 100 mm mercury.
    The initial goal for care of this patient was based on the axis spinous rotation left of 12 degrees with possible neurological components affacting the region of the sinuses. It was felt that alleviation of this component could improve function of the sinus tissue therefore decreasing the sinus drainage into the throat cyst. However, it is also possible that neurologically, the lymphatic pumping system was impaired and restoration of the nerve pathways and reduction of paraspinal muscle spasm of the upper cervical region could positively affect the lymphatic pumping system.
    Significant to the patient's care and management in this case was that low-force instrument adjustments were utilized confirming Gutmann's clinical observations that he was constantly amazed how, even the lightest adjustment with the index finger; the clinical picture normalizes.¹⁶

 CONCLUSION

An integrated approach, including chiropractic care should be considered in pediatric patients with lymphatic disorders to restore altered nerve pathways controlling neurolymphatic function.

 REFERENCES

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 Julie Mayer Hunt, D. C., D.I.C.C.P. Private practice, 1400 Court Street, Clearwater. Florida 33756.

  JOURNAL OF CLINICAL CHIROPRACTIC PEDIATRICS  Volume 5, No. 1 2000