Miinchausen Syndrome by Proxy and Sleep Disorders Medicine

Sleep 12(2):178-183, Raven Press, Ltd., New York © 1989 Association of Professional Sleep Societies

Miinchausen Syndrome by Proxy and Sleep Disorders Medicine James L. Griffith and *Lois S. Slovik Sleep Disorders Center, University of Mississippi Medical Center, Jackson, Mississippi; and *Child Psychiatry Service, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts, U.S.A.

Summary: Miinchausen syndrome by proxy is a factitious disorder of childhood in which a parent fabricates medical history or produces signs of illness in a child to keep the child in a sick role. Since approximately half of all cases of Miinchausen syndrome by proxy are presentations of central nervous system illness, such as excessive daytime sleepiness and near-miss sudden infant death syndrome, sleep disorders centers are likely diagnostic consultants for the evaluation of children involved in this disorder. We review characteristics that may suggest that a particular case has an increased likelihood of Miinchausen syndrome by proxy. The recent presentations of two cases of Miinchausen syndrome by proxy to sleep disorders centers are discussed as examples. Key Words: Miinchausen syndrome by proxy-Factitious disorderApnea monitor.

Munchausen syndrome by proxy refers to a disorder in which a parent acts to produce illness or the appearance ofilIness in a child to keep the child in medical treatment (1). Measures employed by perpetrating parents include fabricating medical history, altering laboratory specimens and records, and inducing illness, such as surreptitiously administering medications to the child (2). Sleep disorders centers are frequently asked to evaluate children whose symptoms have been a diagnostic puzzle to their pediatricians, such as cases of near-miss sudden infant death syndrome, obscure "spells" with alteration of consciousness, and excessive daytime sleepiness. Since these patient populations are at increased risk for the occurrence of Miinchausen syndrome by proxy (2,3), it is essential that clinicians in sleep disorders medicine maintain an active awareness of this factitious disorder and its presentations. Meadow, who first described the disorder in 1977 (1), has noted that nearly half of all Accepted for pUblication June 1988. Address correspondence and reprint requests to Dr. J. L. Griffith at Sleep Disorders Center, Department of Psychiatry and Human Behavior, University of Mississippi Medical Center, 2500 North State Street, Jackson, MS 39216, U.S.A.

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cases involve presentations resembling a cerebral disorder, most commonly seizures, excessive sleepiness, coma, and incoordination (3). Among 25 cases reviewed by Palmer and Yoshimura (4), four children eventually died from the abuse. The mother has been the perpetrator in all reported cases of Miinchausen syndrome by proxy (5). Her recognition, however, has been difficult because she lacks the features that lead one to intuitively suggest child abuse. Rather than showing hostility toward the child, the parent appears overprotective, showing single-minded dedication to the care of her ill child. She is often intelligent, medically sophisticated, and appreciative of the medical care received (1,2,6). The following two case reports are examples of cases that presented for evaluation to sleep disorders centers. CASE REPORTS Case 1 A two-week-old girl was brought to the University of Mississippi Medical Center after she suffered an alleged respiratory arrest from vomiting and aspiration while breast feeding. She showed no signs of distress by the time of her arrival, however. Physical examination, electroencephalogram (EEG), and electrocardiogram (ECG) were normal. A gastroesophageal reflux scintiscan suggested a mild degree of gastroesophageal reflux into her lower esophagus, which was of doubtful clinical significance. The mother, nevertheless, continued to report daily apneic episodes, although none were observed by hospital staff. She requested a home apnea monitor despite reservations by her pediatricians who doubted its need. The Sleep Disorders Center was consulted to determine whether the patient was experiencing apneas as reported by the mother. A polysomnographic recording for 24 h showed no apneas longer than 5 s, the latter limited to REM sleep. Blood oxygen saturation remained above 90% during the brief apneas. Monitoring of EEG and ECG showed no indication of seizure activity or cardiac distress. The mother's continuing reports of apneas were considered to be her anxious misinterpretations of physiological fluctuations in the patient's breathing and motor activity, and the request for a home apnea monitor was denied. In her local community, however, the mother continued to report apneic episodes and obtained a home apnea monitor from a local physician. The physician referred the patient at 9 weeks of age back to the center for reevaluation owing to the continuing reports of her distress. Upon admission, the pediatricians noted that the baby had no soiled diapers although the mother was also reporting severe vomiting and diarrhea. They observed further that no apnea, vomiting, or diarrhea was reported when the patient was alone in the care of her father, grandmother, or hospital staff. About this time the maternal grandmother approached the physicians, voicing fears that the mother was actively making the patient ill. A nurse was stationed near the hospital room where she could observe the patient and her mother. She watched the mother give the patient a rectal enema, then withdraw the enema fluid and feed it into her mouth with a bulb syringe, presumably to produce the appearance of vomiting and diarrhea. The patient was placed under legal custody of the Department of Public Welfare. In

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a foster home; she began gaining weight and all reports of apnea, vomiting, and diarrhea ceased. A psychiatric evaluation of the family found that the 27-year-old mother met diagnostic criteria for somatization disorder (7). The mother acknowledged the abuse, attributing it to loss of control over her behavior due to cumulative personal and family stresses, including sexual abuse as a child, her recent rape by a brother-in-law, and her husband's unemployment, together with her conviction that the baby was in fact more ill than the doctors would appreciate. She feared that the home apnea monitor would have been discontinued had the patient been found healthy on a second admission to the center. Case 2 Another girl was 6 months old when her mother first brought her to an emergency room, stating that the baby had choked, stopped breathing, and turned blue until revived by mouth-to-mouth resuscitation. Physical exam, including neurological exam, was normal. Her EEG and all-night polysomnography showed no evidence suggestive of seizure disorder or abnormal cardiorespiratory function. Owing to the mother's vivid description and continuing worry, a home apnea monitor was prescribed for reassurance. Over the next 2.5 years, the patient was admitted to various hospitals on at least nine occasions owing to her mother's continuing reports that she would stop breathing during her sleep (and sometimes while awake), often turning blue until revived with cardiopulmonary resuscitation. Despite these reports, physical exams were always unremarkable. Repeated polysomnograms, pneumograms, and EEGs were consistently normal, and no abnormal event was ever detected by a Trend-Event or Holter monitor. Owing to the mother's level of anxiety and precisely described symptoms, the home apnea monitor was continued, as well as anticonvulsant treatment with Dilantin. When the child was 5 years old, suspicious hospital staff prompted a psychiatric consultation whose overt purpose was to provide psychological support to the mother but whose actual intent was to investigate the possibility of fabricated symptoms. A home visit found that the child's room resembled an intensive care unit, complete with a home laboratory. Further investigation found that not only had the mother reported symptoms of near-miss sudden infant death syndrome, but that she had described to different physicians bowel irregularities, inability to urinate, blood in urine and feces, behavioral disturbances, petit mal and jacksonian seizures, symptoms of hypoglycemia, and bizarre eating habits. The patient had received multiple invasive diagnostic procedures for pulmonary, cardiac, endocrine, gastrointestinal, neurological, and urological symptoms. Although the investigations had been negative, she was treated with a variety of medications for symptoms of uncertain etiology by several different physicians, each of whom had been unaware of the others' involvement. The patient was also receiving physical therapy, occupational therapy, and special education for handicapped children, all for unclear indications. The psychiatric evaluation found that the mother, a former nurse, had symptoms of a somatization disorder (7). In her interactions with the patient, she appeared extremely overprotective, single mindedly focused upon the care of her daughter's medical needs. Yet at other times she lapsed into a state of seeming emotional disengagement, referring to her daughter only as "the child." The patient's two older sisters also had histories of medically puzzling physical complaints. The father showed little emotional involveSleep. Vol. 12. No.2. 1989

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ment in the family or interest in seeking psychiatric treatment for his wife. It became clear that the patient's illness was the one organizing factor in her mother's life, her only "claim to fame." Through long-term family therapy for mother and daughter, the abuse was stopped without removing the daughter from the home. Convincing the involved physicians to withdraw their treatments was difficult, although no symptoms of illness appeared as a consequence. The mother eventually acknowledged that she had been hypervigilant and had amplified symptoms to convince the physicians to take the illnesses she perceived seriously. However, she persistently denied that she had ever actually created illness in her daughter. As the patient's illnesses ceased to be the family focus, the mother began to express more and more frustration and resentment toward her husband, whom she felt had always neglected her, which suggested that her preoccupation with her daughter's health may have been in part a diversion of her attention away from dissatisfaction with the marriage. DISCUSSION It is important that sleep laboratories maintain an awareness of Miinchausen syndrome by proxy when evaluating children. Professor Roy Meadow, who first described the disorder, states: I have seen many children suffering that form of abuse who at one time or another have been investigated [in sleep disorders centers] and the abuse was neither suspected nor looked for.

One reason might be that many centers do not usually videotape the mother and child during the recordings, so that although they have careful recordings on EEG, etc., of the attacks they do not have a visual record of what was happening at the time or of the way in which the mother was interfering with the child (usually by suffocation). It is interesting how often the mothers interfere with the recordings themselves-thus firmly secured leads and clips become mysteriously detached during the night. (personal communication, 1988)

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Several clinical features commonly noted among cases studied suggest that a particular child has an increased risk for the diagnosis: 1. The current evaluation is only one in a long series of medical evaluations that seem marginally or not at all indicated based upon the clinical history; the parent "doctor shops" with the child (5,8). 2. The mother and child appear emotionally overinvolved with one another, yet the mother seems to show little sensitivity to the child's needs as an individual apart from his or her relationship with the mother. The mother may refuse to leave the child alone, even briefly (8,9). 3. The child appears generally healthy, but the medical history is that of a long list of multiple medical complaints and unsuccessful treatments (8). 4. The child's physicians have been puzzled by the recurrent nature of the problem (9). 5. The case has been identified as strikingly unusual in its clinical patterns; e.g., an experienced physician notes, "I've never seen a case like it before" (9). 6. The signs and symptoms occur only in the presence of the mother (9). 7. The mother seems less concerned about the child's illness than are the medical professionals (9). 8. There has been a sibling with a similar illness or death (9). 9. The mother has previous nursing or medical experience (9).

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10. The mother has had a similar illness herself in the past (9). 11. Maternal history suggests that the mother may have a somatoform disorder (7), such as somatization disorder or hypochondriasis, or a factitious disorder (7), such as Miinchausen's syndrome (9,10).

A previous study of recurrent infant apnea requiring multiple resuscitations found that Miinchausen syndrome by proxy accounted for a significant subgroup of cases. The investigators concluded that "home monitoring-the equipment, the responsibilities, the intense involvement with medical personnel-may be very attractive to the parent capable of this type of behavior" (11). Meadow (3,12; personal communication, 1988) has found maternal suffocation, using a hand over the nose and mouth or a pad, to be the most common mode by which a parent produces apnea in an infant. In a smaller group of cases, the mother has administered a poison or medication overdose to produce apnea. Among older children presenting with symptoms of seizures or excessive daytime sleepiness, Meadow (3,12) has found the report of symptoms often to be purely fabricated. However, the mother produced the seizure or drowsiness by suffocation, carotid body compression, or administration of drugs in one-third of 35 cases (3). There are several possible ways in which sleep disorders centers can make unique contributions to the discovery of Miinchausen syndrome by proxy: 1. Sleep disorders staff and technicians may be in a better position to view the clinical problem in that, as consultants, they can remain outside the emotional field of the doctor-parent relationship. The diagnosis has been delayed consistently among published cases owing to the ability of the Miinchausen-by-proxy mother to ingratiate herself with the physicians who appreciate her gratitude and admire her seemingly unselfish commitment to her child (1-3,9). The mother may be less on her guard when with a technical staff. For example, in a case where the mother repeatedly had asphyxiated her infant, the case report noted, "One of the physicians involved ([J.n.F.], the research neurophysiologist in charge of the monitoring laboratory and, incidentally, the only non pediatrician) suggested that the mother might be inducing the spells. This caused a great controversy among the involved pediatric physicians and nurses, who felt it was very unlikely that this particular mother could be the perpetrator of the episodes, but they agreed to cooperate in arranging for further recording sessions" (6). 2. The technology of the sleep disorders center can be creatively used for covert observation of parental behavior during the child's monitoring in the laboratory. For example, the mother was videotaped asphyxiating the baby in the above case by using a hidden camera covered by a grill in the room (6). 3. Recordings themselves may provide data, such as EEG drug effects, which can suggest additional medical investigations seeking evidence for abuse (2). REFERENCES Meadow R. Miinchausen syndrome by proxy: the hinterland of child abuse. Lancet 1977;2:343-5. Meadow R. Management of Miinchausen syndrome by proxy. Arch Dis Child 1985;60:385-93. Meadow R. Miinchausen by proxy and brain damage. Dev Med Child NeuroI1984;26:669-76. Palmer AJ, Yoshimura GJ. Miinchausen syndrome by proxy. JAm Acad Child Psychiatry 1984;4:503-8. Libow JA, Schreier HA. Three forms of factitious illness in children: when is it Miinchausen syndrome by proxy? Am J Orthopsychiatry 1986;56:602-11. 6. Rosen CL, Frost JD, Bricker T, Tarnow JD, Gillette PC, Dunlavy S. Two siblings with recurrent cardiorespiratory arrest: Miinchausen syndrome by proxy or child abuse? Pediatrics 1983;5:715-20.

1. 2. 3. 4. 5.

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7. American Psychiatric Association. Diagnostic and statistical manual of mental disorders. 3rd ed., rev. Washington, DC: American Psychiatric Association, 1987. 8. Woollcott P, Aceto T, Rutt C, Bloom M, Glick R. Doctor shopping with the child as proxy patient: a variant of child abuse. J Pediatr 1982;101:297-301. 9. Meadow R. Miinchausen syndrome by proxy. Arch Dis Child 1982;57:92-8. 10. Livingstone R. Maternal somatization disorder and Miinchausen syndrome by proxy. Psychosomatics 1987;28:213-7. 11. Rosen CL, Frost JD, Glaze DG. Child abuse and recurrent infant apnea. J Pediatr 1986;109:1065-7. 12. Meadow R. Fictitious epilepsy. Lancet 1984;2:25-8.

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