singultus: a distressing postsurgical complication

3
STRULL AND DYM 71 1 2. Abrikossoff A: Uber myome ausgehend vonder quergestreiften willkurlichen muskulatur. Virchows Arch [A] 260:215, 1926 3. Rubenstein D, Shanker DB, Finlayson L, et al: Multiple cutane- ous granular cell tumors in children. Pediatr Dennatol 4:94, 1987 4. Noonan JD, Horton CE, Old WL, et al: Granular cell myoblas- toma of the head and neck: Review of the literature and 10 year experience. Am J Surg 138:611, 1979 5. Bangle RB Jr: A morphological and histochemical study of the granular cell myoblastoma. Cancer 5:950, 1952 6. Pressoir R, Chung EB: Granular cell tumor in black patients. J Natl Med Assoc 72:1171, 1980 7. Moscovic EA, Azar HA: Multiple granular cell tumors ("myo- blastomas"): Case report with electron microscopic observa- tions and review of the literature. Cancer 20:2032, 1967 8. Eisen RN, Kirby WM, O'Quinn JL: Granular cell tumor of the biliary tree. Am J Surg Pathol 15:460, 1991 9. Strong EW, McDivitt RW, Brasfield RD: Granular cell myoblas- toma. Cancer 25:415, 1970 10. Powell EB: Granular cell myoblastoma. Arch Pathol 42:517, 1946 11. Radin DR, Zelner R, Ray MJ, et al: Multiple granular cell tumors of the skin and gastrointestinal tract. Am J Radiol 147:1305, 1986 12. Rifkin RH, Blocker SH, Palmer JO, et al: Multiple granular cell tumors: A familial occurrence in children. Arch Surg 121:945, 1986 13. Jones JK, Kuo T, Griffiths CM, et al: Multiple granular cell tumor. Laryngoscope 90:1646, 1980 14. LackEE, Worsham GF, Callihan MD, et al: Granular cell tumor: A clinicopathologic study of 110 patients. J Surg Oncol 13:301, 1980 15. Colberg JE, Hubay CA: Granular cell myoblastoma: A problem in diagnosis. Surgery 53:226, 1963 16. Garancis JC, Komorowski RA, Kuzma JF: Granular cell myo- blastoma. Cancer 25:542, 1970 17. Bernstein BA, Murnane TW, Maloney PL: Tongue and multiple cutaneous granular-cell myoblastoma. Oral Surg 31:312, 1971 18. White SW, Gallager RL, Rodman OG: Multiple granular-cell tumors. J Dermatol Surg Oncol 6:57, 1980 19. Baden E, Divaris M, Quillard J: A light microscopic and immu- nohistochemical study of a multiple granular cell tumor and review of the literature. J Oral Maxillofac Surg 48:1093, 1990 20. Manara GC, De Panfilis G, Bacchi AB, et al: Fine structure of granular cell tumor of abrikossoff. J Cutan Pathol 8:277, 1981 21. Arnn ET, Gordon H: Granular-cell myoblastoma: Report of a case with multiple sites of origin. Lab Invest 6:142, 1957 22. Datum DD, Cibull ML, Geissler RH, et al: Investigation into the histogenesis of congenital epulis of the newborn. Oral Surg Oral Med Oral Pathol 76:205, 1993 23. Zuker RM, Buenechea R: Congential epulis: Review of the literature and case report. J Oral Maxillofac Surg 51:1040, 1993 24. Hartman KS: Granular-cell ameloblastoma: A survey of twenty cases from the Armed Forces Institute of Pathology. Oral Surg 38:241, 1974 25. Mirchandani R, Sciubba JJ, Mir R: Granular cell lesions of the jaws and oral cavity: A clinicopathologic, immunohistochem- ical, and ultrastructural study. J Oral Maxillofac Surg 47:1248, 1989 26. Lownie JF, Altini M, Shear M: Granular cell peripheral odonto- genic fibroma. J Oral Pathol 5:295, 1976 27. Sobel HJ, Avrin E, Marquet E, et al: Reactive granular cells in sites of trauma: A cytochemical and ultrastructural study. Am J Clin Pathol 61:223, 1974 28. Regezi JA, Sciubba J: Oral Pathology: Clinical-Pathologic Cor- relations (ed 2). Philadelphia, PA, Saunders, 1993 J Oral Maxillofac Surg 53:711-713, 1995 A Distressing Singultus: Postsurgical Compfication GREGORY E. STRULL, DMD,* AND HARRY DYM, DDSt Singultus, commonly known as hiccups, is a tran- sient and benign annoyance experienced occasionally by most people. It is defined as an involuntary spas- modic contraction of the diaphragm causing a begin- ning inspiration that is suddenly checked by closure of the epiglottis, producing the characteristic sound. 1 Received from the Division of Oral and Maxillofacial Surgery, The Brooklyn Hospital Center, Brooklyn, NY. * Senior Resident. t Chief, Division of Oral and Maxillofacial Surgery; Director, Advanced Residency Training Program in Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Dym: Depart- ment of Dentistry, The Brooklyn Hospital Center, Box 224, 121 DeKalb Ave, Brooklyn, NY 11201. © 1995 American Association of Oral and MaxillofacialSurgeons 0278-2391/95/5306-001653.00/0 Hiccups are also described by their duration. A hiccup bout is an episode consisting of more than a few hic- cups, which can last up to 48 hours; persistent hiccups are those that last over 48 hours and continue for up to a month; intractable hiccups can last longer than a month. 2 The following report describes a patient who developed persistent hiccups postoperatively. Report of Case A 65-year-old African American man was referred to the oral and maxillofacial surgery clinic for evaluation and treat- ment of a severely atrophic maxilla. Examination of the soft tissues showed mobile fibrous tissue in the anterior maxilla. The remaining anterior mandibular teeth were periodontally compromised but clinically stable. The extraoral examina- tion showed a decreased vertical dimension, with overclo- sure secondary to inability to wear a maxillary prosthesis.

Upload: gregory-e-strull

Post on 25-Aug-2016

219 views

Category:

Documents


2 download

TRANSCRIPT

Page 1: Singultus: A distressing postsurgical complication

STRULL AND DYM 71 1

2. Abrikossoff A: Uber myome ausgehend vonder quergestreiften willkurlichen muskulatur. Virchows Arch [A] 260:215, 1926

3. Rubenstein D, Shanker DB, Finlayson L, et al: Multiple cutane- ous granular cell tumors in children. Pediatr Dennatol 4:94, 1987

4. Noonan JD, Horton CE, Old WL, et al: Granular cell myoblas- toma of the head and neck: Review of the literature and 10 year experience. Am J Surg 138:611, 1979

5. Bangle RB Jr: A morphological and histochemical study of the granular cell myoblastoma. Cancer 5:950, 1952

6. Pressoir R, Chung EB: Granular cell tumor in black patients. J Natl Med Assoc 72:1171, 1980

7. Moscovic EA, Azar HA: Multiple granular cell tumors ("myo- blastomas"): Case report with electron microscopic observa- tions and review of the literature. Cancer 20:2032, 1967

8. Eisen RN, Kirby WM, O'Quinn JL: Granular cell tumor of the biliary tree. Am J Surg Pathol 15:460, 1991

9. Strong EW, McDivitt RW, Brasfield RD: Granular cell myoblas- toma. Cancer 25:415, 1970

10. Powell EB: Granular cell myoblastoma. Arch Pathol 42:517, 1946

11. Radin DR, Zelner R, Ray MJ, et al: Multiple granular cell tumors of the skin and gastrointestinal tract. Am J Radiol 147:1305, 1986

12. Rifkin RH, Blocker SH, Palmer JO, et al: Multiple granular cell tumors: A familial occurrence in children. Arch Surg 121:945, 1986

13. Jones JK, Kuo T, Griffiths CM, et al: Multiple granular cell tumor. Laryngoscope 90:1646, 1980

14. LackEE, Worsham GF, Callihan MD, et al: Granular cell tumor: A clinicopathologic study of 110 patients. J Surg Oncol 13:301, 1980

15. Colberg JE, Hubay CA: Granular cell myoblastoma: A problem in diagnosis. Surgery 53:226, 1963

16. Garancis JC, Komorowski RA, Kuzma JF: Granular cell myo- blastoma. Cancer 25:542, 1970

17. Bernstein BA, Murnane TW, Maloney PL: Tongue and multiple cutaneous granular-cell myoblastoma. Oral Surg 31:312, 1971

18. White SW, Gallager RL, Rodman OG: Multiple granular-cell tumors. J Dermatol Surg Oncol 6:57, 1980

19. Baden E, Divaris M, Quillard J: A light microscopic and immu- nohistochemical study of a multiple granular cell tumor and review of the literature. J Oral Maxillofac Surg 48:1093, 1990

20. Manara GC, De Panfilis G, Bacchi AB, et al: Fine structure of granular cell tumor of abrikossoff. J Cutan Pathol 8:277, 1981

21. Arnn ET, Gordon H: Granular-cell myoblastoma: Report of a case with multiple sites of origin. Lab Invest 6:142, 1957

22. Datum DD, Cibull ML, Geissler RH, et al: Investigation into the histogenesis of congenital epulis of the newborn. Oral Surg Oral Med Oral Pathol 76:205, 1993

23. Zuker RM, Buenechea R: Congential epulis: Review of the literature and case report. J Oral Maxillofac Surg 51:1040, 1993

24. Hartman KS: Granular-cell ameloblastoma: A survey of twenty cases from the Armed Forces Institute of Pathology. Oral Surg 38:241, 1974

25. Mirchandani R, Sciubba JJ, Mir R: Granular cell lesions of the jaws and oral cavity: A clinicopathologic, immunohistochem- ical, and ultrastructural study. J Oral Maxillofac Surg 47:1248, 1989

26. Lownie JF, Altini M, Shear M: Granular cell peripheral odonto- genic fibroma. J Oral Pathol 5:295, 1976

27. Sobel HJ, Avrin E, Marquet E, et al: Reactive granular cells in sites of trauma: A cytochemical and ultrastructural study. Am J Clin Pathol 61:223, 1974

28. Regezi JA, Sciubba J: Oral Pathology: Clinical-Pathologic Cor- relations (ed 2). Philadelphia, PA, Saunders, 1993

J Oral Maxillofac Surg 53:711-713, 1995

A Distressing Singultus: Postsurgical Compfication

GREGORY E. STRULL, DMD,* AND HARRY DYM, DDSt

Singul tus , c o m m o n l y k n o w n as h iccups , is a tran-

s ient and b e n i g n a n n o y a n c e e x p e r i e n c e d occa s iona l l y

by m o s t people . It is de f ined as an invo lun ta ry spas-

m o d i c con t rac t ion o f the d i a p h r a g m caus ing a beg in -

n ing insp i ra t ion that is sudden ly checked by c losu re

o f the epiglot t i s , p r o d u c i n g the charac te r i s t ic sound. 1

Received from the Division of Oral and Maxillofacial Surgery, The Brooklyn Hospital Center, Brooklyn, NY.

* Senior Resident. t Chief, Division of Oral and Maxillofacial Surgery; Director,

Advanced Residency Training Program in Oral and Maxillofacial Surgery.

Address correspondence and reprint requests to Dr Dym: Depart- ment of Dentistry, The Brooklyn Hospital Center, Box 224, 121 DeKalb Ave, Brooklyn, NY 11201.

© 1995 American Association of Oral and Maxillofacial Surgeons

0278-2391/95/5306-001653.00/0

Hiccups are a lso desc r ibed by thei r durat ion. A h iccup

bou t is an ep i sode cons i s t ing o f m o r e than a f e w hic-

cups, w h i c h can last up to 48 hours ; pers is tent h i ccups

are those that last o v e r 48 hours and con t inue fo r up

to a month ; in t rac tab le h iccups can last l onge r than a

month . 2 T h e f o l l o w i n g repor t desc r ibes a pa t ient w h o

d e v e l o p e d pers i s ten t h i ccups pos topera t ive ly .

Report of Case

A 65-year-old African American man was referred to the oral and maxillofacial surgery clinic for evaluation and treat- ment of a severely atrophic maxilla. Examination of the soft tissues showed mobile fibrous tissue in the anterior maxilla. The remaining anterior mandibular teeth were periodontally compromised but clinically stable. The extraoral examina- tion showed a decreased vertical dimension, with overclo- sure secondary to inability to wear a maxillary prosthesis.

Page 2: Singultus: A distressing postsurgical complication

712 SINGULTUS

Panoramic and cephalometric radiographs showed a se- verely atrophic maxilla with a flat palatal configuration. Also noted were large maxillary sinuses bilaterally, with approxi- mately 3-to 5-ram alveolar basal bone remaining. A decision was made to reconstruct the maxilla with bilateral maxillary sinus lifts, using an anterior iliac crest bone graft and the immediate placement of endosseous implants.

The patient's medical history was significant for a left radical nephrectomy secondary to renal carcinoma, but he was not on hemodialysis. The patient also had a history of asthma and hypertension for which he was taking Ventolin. (Glaxo Inc, Research Triangle Park, NC) He had no known drug allergies. All laboratory data were essentially within normal limits.

General anesthesia was used, and the patient was intubated a nasotracheally without complications. Approximately 30 cm 3 graft material was obtained from the anterior iliac crest through a medial approach. The standard crestal incision for bilateral sinus lifts was performed without complications, and a prefabricated surgical stent was used for proper place- ment of the six root form implants. Primary closure was obtained in a tension-free manner. The patient did well post- surgically and was discharged the following day.

When the patient returned 2 days postoperatively, he stated that he had had transient episodes of hiccuping, but was not experiencing them currently. However, he presented 3 days later actively hiccuping. Multiple nonpharrnacologic methods were tried, the hiccups ceased, and the patient was discharged. He presented again the following morning with active hiccups. Nonpharmacologic methods were again tried, but proved to be unsuccessful. At this point, the patient was prescribed Thorazine (SmithKline, Philadelphia, PA) 10 mg every 6 hours and given a follow-up appointment in 2 days. When he presente d for follow-up, he was still actively hic- cuping, and the thorazine was then increased to 20 mg every 6 hours. At his next appointment, 2 days later, his hiccuping had stopped, and the thorazine was discontinued the follow- ing day without the patient having further problems.

Discussion

As a result of extensive study and research to deter- mine the cause, we now know that hiccups result from stimulation of one or more components of a hiccup reflex arc. 3-6 The afferent portion of the hiccup reflex arc comprises the phrenic and vagus nerves and the sympathetic chain arising from thoracic segments T6 to T12. 5'6 The central connection between the afferent and efferent limbs of the reflex arc cannot be ascribed to a discrete and specific anatomic location, such as the analogous and recognized chemoreceptor trigger zone for vomiting. Instead, the central coordination of hiccuping is attributed to a nonspecific anatomic location somewhere in the spinal cord between seg- ments C3 and C5. 7-9 Recently, it has been proposed that the central connection for hiccups probably also involves a complex interaction among brainstem and midbrain areas, including the respiratory center, phrenic nerve nuclei, medullary reticular formation, and hypothalamus. 8 This means that the phrenic nerve is no longer considered to be the only efferent compo-

nent of the hiccup response. 2 Electromyographic stud- ies have shown simultaneous firing of motor neurons to the anterior scalene muscles (C5 to C7), external intercostal muscles (T1 to T l l ) , and in the recurrent laryngeal nerve. 1°-12

A multitude of pathophysiologic processes can in- cite hiccups. Hiccup bouts, or self-limiting hiccups, may be caused by gastric distention from a variety of causes, including excessive food or alcohol ingestion, aerophagia, gastric insufflation, sudden changes in gas- trointestinal temperature, and tobacco u s e . 2 Persistent or intractable hiccups have many broadly classified causes of organic, psychogenic, and idiopathic origin. Psychogenic causes include stress, excitement, grief, anorexia nervosa, and malingering, but hiccups can only be labeled psychogenic or idiopathic once organic causes are ruled out. 2

Organic causes of singultus can be divided into three categories: central, peripheral, and other (toxic, meta- bolic, or pharmacologic). Central nervous system causes include neoplasms, hydrocephalus, ventriculo- peritoneal shunts, and multiple sclerosis, although vas- cular and infectious central nervous system causes are more common, z The latter causes include ischemic or hemorrhagic stroke, arteriovenous malformation, le- sions from head trauma, cerebral contusion, encephali- tis, meningitis, brain abscess, and neurosyphilis. 2 Pe- ripheral nervous system causes include irritation of the vagus nerve anywhere along its course. Such irritation can result from stimulation of the meningeal or pharyn- geal afferents by meningitis, pharyngitis, laryngitis, peritonsillar abscess, goiter, cysts, or tumors of the neck. Irritation of the thoracic or abdominal branches can result from chest trauma, neoplasia, myocardial infarction, pulmonary edema, pericarditis, pleuritis, mediastinitis, esophagitis, aneurysms, asthma, bronchi- tis, gastric distention, gastritis, ulcers, pancreatic or biliary disease, bowel obstruction, appendicitis, genito- urinary disorders, hepatic or splenic disease, and intra- operative thoracic or abdominal manipulation. 2

Many other causes can contribute to hiccups. Gen- eral anesthesia has been implicated as a major cause, for a variety of reasons, including suppression of the central nervous system, hyperextension of the neck, epiglotic stimulation, and gastric distention from mask ventilation. Pharmacologic agents such as steroids, barbiturates, benzodiazepines, and methyldopa have also been reported as causes. 2

Treatment modalities can be divided into two cate- gories: nonpharmacologic and pharmacologic. Non- pharmacologic treatments consist of diverting the pa- tient's attention by distracting conversation, fright, or painful or unpleasant stimuli. 13 Simple home remedies usually rely on a form of nasopharyngeal stimulation. These include forcible traction of the tongue, breathing

Page 3: Singultus: A distressing postsurgical complication

STRULL AND DYM 7 1 3

into a paper bag, gargling with water, sipping ice water, swallowing a teaspoon of granulated sugar, biting on a lemon, inhaling noxious agents, and drinking from the far side of a glass. More aggressive nonpharmaco- logic techniques of nasopharyngeal stimulation also have been advocated. Direct pharyngeal stimulation using a cotton tip, nasogastric tube, or nasopharyngeal airway has been used to provide direct uvular stimula- tion. This technique has proved to be 90% effective. 2'14 Other methods include iced gastric lavage, stimulation or tapping of dermatome C5 on the back or the neck, Valsalva maneuvers, carotid massage, digital ocular globe pressure, digital rectal massage, and continuous positive airway pressure. Additional techniques in- clude behavioral conditioning, hypnosis, phrenic nerve or direct diaphragmatic stimulation by placement of paring electrodes, and phrenic or vagus nerve block or surgical interruption. 2'~5'16

Although nonpharmacologic intervention is a more popular mode of treatment, pharmacologic interven- tions are just as numerous, although not as widely studied. Chlorpromazine is the most widely used drug, but its mechanism of action for the treatment of hiccups is uncertain, z The recommended dose is 25 to 50 mg in 500 to 1,000 mL normal saline given intravenously over several hours, with frequent blood pressure moni- toring, or alternatively it may be given 25 to 50 mg intramuscularly or 25 to 50 mg by mouth three to four times daily. 2 Another antipsychotic drug, haloperidol, has also been advocated. Its recommended dose is 2 to 5 mg intramuscularly or 1 to 4 mg by mouth three times daily. Anticonvulsants that have been described as effective in the management of hiccups include phenytoin, phenobarbitol, carbamazepam, and valproic acid. 17-19 Phenytoin can be given initially in an intrave- nous dose of 200 mg at 50 mg/min or as an oral loading (up to 18 mg/kg) followed by a maintenance dose of 300 mg/d. If given intravenously, patients should be closely monitored for bradycardia, heart block, or hy- potension. 2

The dopamine antagonist metaclopramide is given initially in a 10 mg intravenous or intramuscular dose, with maintenance of 10 to 20 mg four times a day. The antidepressant amitryptyline has also been recom- mended in a dose schedule of 10 mg by mouth three times daily. Several other medications also exist, yet no controlled studies have been performed to substantiate their efficacy. These include chloral hydrate, pentazo- cine, ephedrine, methylphenidate, amphetamine, car- bemazepine, nikethamide, ketamine, edrophonium, dexamethasone, amantadine, nifedipine, baclofen, a°24

Although persistent singultus is a rare postsurgical complication, when it does happen it can cause sig- nificant discomfort to the patient and may be emotion- ally disabling as well. The case reported illustrates a postsurgical episode of hiccups that failed to respond to nonpharmacologic methods and required pharmaco- logic management. In reviewing all possible causes for singultus, the most likely in this case was the abdomi- nal manipulation that occurred during the bone graft harvest procedure from the iliac crest.

References

1. Dorland's Medical Dictionary (ed 27). Philadelphia, PA, Saun- ders, 1988, p 766

2. Kolodzik PW, Eilers MA: Hiccups (singultus): Review and ap- proach to treatment, Ann Emerg Med 20:565, 1991

3. Lewis J: Hiccups: Causes and cures. J Clin Gastroenterol 7:539, 1985

4. Gigot A, Flynn P: Treatment of hiccups. JAMA 150:760, 1952 5. Samuels L: Hiccup: A ten year review of anatomy, etiology,

and treatment. Can Med Assoc J 67:315, 1952 6. Travell J: A trigger point for hiccup. J Am Osteopath Assoc

77:308, 1977 7. Oxford Latin Dictionary (ed 7). Oxford, UK, Oxford University

Press, 1980, p 769 8. Wagner M, Stapczynski J: Persistent hiccups. Ann Emerg Med

11:24, 1982 9. Graham D: Esophageal motor ability during hiccup. Gastroen-

terology 90:2039, 1986 10. Nathan M, Leshner R, Keller A: Intractable hiccups. Laryngo-

scope 90:1612, 1980 11. Hemachuda T, Phanthumchinda K, Indrakoses A, et al: Intracta-

ble hiccups (singultus) as presenting manifestation of Japa- nese encephalitis. J Med Assoc Thai 67:620, 1984

12. Shim C: Motor disturbances of the diaphragm. Clin Chest Med 1:125, 1980

13. Hansen BJ, Rosenberg J: Persistent post-operative hiccups: A review. Acta Anesthesiol Scand 37:643, 1993

14. Mangar D, Patil VU: Elimination of hiccups with a nasopharyn- geal airway. J Clin Anesth 4:86, 1992

15. Oheh M, Bassan H, Oliven A: Termination of intractable hic- cups with digital rectal massage. J Intern Med 227:145, 1990

16. Isselbacher KJ, Petersdorf RG, Braunwald E, et al: Harrison's Principles of Internal Medicine (ed 13). New York, McGraw- Hill, 1994, p 1233

17. Petroski D, Patel A: Diphenylhydantoin for intractable hiccups (letter). Lancet 1:997, 1974

18. Davis J: Diphenylhydantoin for hiccups (letter). Lancet 1:997, 1974

19. Osborn H, Zisbein J, Sparrano R: Single dose oral phenytoin loading. Ann Emerg Med 16:407, 1987

20. Vantrappen G, Decramer M, Harlet R: Hi-frequency diaphrag- matic flutter symptoms and treatment by carbemazepine. Lan- cet 339:265, 1992

21. Ramirez FC, Graham DY: Treatment of intractable hiccup with baclofen: Results of a double-blind randomized, controlled, cross-over study. Am J Gastroenterol 87:1789, 1992

22. Brigham B, Bolin T: High dose nifedipine and fludrocortisone for intractable hiccups. Med J Aust 157:70, 1992

23. Intractable hiccup: Baclofen and nifedipine are worth trying. Drug Ther Bull 28:36, 1990

24. Lipps DC, Jabbari B, Mitchell MH, et al: Nifedipine for intracta- ble hiccups. Neurology 40:531, 1990