ELSEVIER Journal of Affective Disorders 34 (1995) 101-107

JOURNAL OF

AFFECTIVE DISORDERS

Does puerperal illness distinguish a subgroup of bipolar patients?

Neil Hunt * , Trevor Silverstone Department of Psychological Medicine, Medical College of St Bartholomew’s Hospital, London, UK

Received 8 August 1994; revised 29 December 1994; accepted 9 January 1995

Abstract

Puerperal psychosis was found to be the 1st illness for 13 (36%) of a series of 36 mothers with bipolar affective disorder. The risk of recurrence with childbirth in those with established bipolar disorder was found to be 2540%. Those women who survived childbirth without illness but then became ill at a later time reported a worse illness course. Recent fatherhood was not a precipitant of 1st affective illness for any of the 28 bipolar men.

Keywords: Bipolar disorder; Puerperium; Gender

1. Introduction

Birth of a child has been reported to be the commonest precipitant of a 1st episode of mania for women, being the trigger in > l/4 of cases (Ambelas, 1987). Obversely, - l/4 of women with recurrent bipolar affective disorder report that they had their 1st episode after childbirth (Whalley et al., 1982; Reich and Winokur, 1970). The risk of recurrence of bipolar illness after childbirth is thought to be - 25% (Platz and Kendell, 1988; Davidson and Robertson, 1985). A reliable estimate of this figure is needed to assess the efficacy of measures taken to prevent puer- peral illness in bipolar patients (Stewart et al., 1991).

An epidemiological study of fathers did not show an excess of psychiatric consultations after

* Corresponding author. Address: Fulbourn Hospital, Cam-

bridge CBl SEF, UK.

the birth of their children compared with before (Kendell et al., 1976). However, the birth of a child has been reported to be a precipitant of mania for men, occurring in 12% of 24 men at 1st admission (Ambelas, 1987). In another study, 9/40 bipolar men became manic in the year after the birth of a child (Davenport and Adland, 1982).

Whether the prognosis for women is better after puerperal compared with non-puerperal mania is disputed. Platz and Kendell (1988) found a similar re-admission rate in both groups. How- ever, Dean et al. (1989) found a better prognosis among the puerperal manics in terms of social function and re-admission rates in agreement with earlier studies (Brockington et al., 1982; Kadrmas et al., 1979).

Though childbirth is a potent precipitant of affective illness in women, it is obviously not sufficient to cause relapse in all bipolar women. Social and obstetric factors have also been impli- cated in the genesis of psychosis after childbirth (Kendell et al., 1981). The occurrence of a recent

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102 N. Hunt, T. Silverstone /Journal of Affective Disorders 34 (1995) 101-107

stressful life event has been found to be strongly associated with mild postpartum depressive states (Paykel et al., 1980) but not with onset of severe depression (Martin et al., 1989) or puerperal psy- choses, including mania (Brockington et al., 1989; Dowlatshahi and Paykel, 1990; Marks et al., 1992). This has been interpreted as indicating that the aetiology of severe postpartum disorders is ‘pre- dominantly biological’.

As regards family history, there have also been conflicting results when puerperal and non- puerperal manics are compared. Studies, using the history from the patient only, have found either no difference (Whalley et al., 1982) or a lower rate among the relatives of women with puerperal psychoses (Kadrmas et al., 1979; Schopf et al., 198.5; Platz and Kendell, 1988). In contrast, the only survey interviewing relatives found a higher rate of psychiatric illness in the lst-degree relatives of bipolars who had a puerperal episode compared with non-puerperal bipolars (Dean et al., 1989). The lower level of genetic vulnerability and the difficulty in finding bipolars who had not had puerperal episodes led to the suggestion that these patients might form an unusual group with an organic basis for their illness.

2. Aims, subjects and methods

The first aim was to select, without regard to puerperal illness, a representative group of bipo- lar patients. Then, to determine the degree of coincidence between childbirth and both the on- set and recurrence of bipolar disorder. A third aim was to assess the relationship of puerperal psychosis history to the severity of the illness course.

The information presented in this paper was gathered during a cohort study that was primarily designed to assess the effect of life events and season on recurrence (Hunt et al., 1992; Hunt et al., 1992a). The cohort consisted of 86 consecu- tive admissions to a catchment area service who fulfilled research diagnostic criteria (RDC; Spitzer et al., 1978) for bipolar affective disorder. Follow-up started 2-5 years after admission. 71 patients were interviewed in person using the schedule for affective disorders and schizophre- nia @ADS; Endicott and Spitzer, 1978) and in- formation was gathered from all available sources on the remaining 17. Specific enquiry was made about family history and childbirth. A puerperal episode was defined as an episode of psychiatric illness meeting RDC criteria occurring within 3 months of childbirth. During the 2-year prospec- tive study, the patients were interviewed every 3 months, again using the SADS.

3. Results

3.1. Comparison of male and female bipolars

The data relating to episodes after childbirth for women was compared with those for men. If childbirth is a time of particular risk for women, we would expect there to be marked differences between the men and the women. There was a higher rate of affective episodes in the 3 months after childbirth among the women (22/79, 28%) than the men (2/14, 14%) (Table 1) though this difference did not reach statistical significance (difference in proportion = 0.14, 95% CZ - 0.07- 0.34).

There was a more striking difference in the

Table 1 Comparison of childbirth and puerperal affective episodes in females and males

Females (n = 58)

Parents (n) 36 (62%) Deliveries (n) 79

Males (n = 28)

8 (29%) 14

Puerperal affective episodes (n)

Deliveries followed by an affective episode (o/o) 22 2 28% 14%

N. Hunt, T. Silverstone /Journal of Affective Disorders 34 (1995) 101-107 103

number of patients who had their lst-ever affec- tive episodes. 7/19 (37%) later deliveries were tive episode within 3 months of childbirth. 13/36 followed by affective illness within 3 months (not mothers had their 1st episode in the puerperium; a statistically significant difference). 9 of the this was not the case for any of the 8 fathers puerperal episodes were depressive and 13 were (difference in proportion = 0.36,95% CZ 0.2-0.5). of a manic type.

3.2. Risk of recurrence of bipolar disorder after childbirth

3.4. Illness course in bipolar women differentiated by relationship of onset of their illness to childbirth

This risk estimate was ascertained for 2 groups. (1) The 13 women who had their 1st affective episode within 3 months of childbirth. For 10 of these women, this occurred after the delivery of their 1st child; for 3 after the 2nd. 9/13 women had a further 10 deliveries of which 4 resulted in further puerperal episodes, a relapse rate of 40%.

(2) The 10 women whose illness started before they had any children. These 10 women had 18 children between them. However, on 2 occasions, the mother was manic in the last trimester and, therefore, these 2 deliveries will not be included in any further analysis. 5/16 deliveries were fol- lowed by affective episodes, a relapse rate of 31%.

4 groups of women were defined in terms of the onset of illness in relation to childbirth: (a) 22 women who had no children; (b) 10 women whose illness began before childbirth; (c) 13 with a 1st illness that was puerperal; and (d) 12 who had children but whose illness began after their family was complete.

1 woman did not fit into these categories as she had 3 children without illness, then a 1st episode that was not puerperal, then a further child but again no puerperal illness. She has been included in category d, making 13 in that group. Table 2 shows the clinical characteristics of the patients in these groupings.

During the 2 years of the prospective study, there were 4 births of which only 1 was followed by relapse (25%).

The women (group d) whose illness began af- ter their family was complete (and, therefore, had no puerperal episodes) were older at the time of examination and at the time of 1st illness, and were the only group to have more episodes/year than the men. In a comparison of the ratio of episodes/length of illness between group d and the rest of the women, this would appear to be a significant difference (Mann-Whitney, P < 0.05, difference in medians 0.2, 95% CZ 0.01-0.37).

3.3. Puerperal episodes according to parity

Combining the 2 groups of women with puer- peral episodes, there were 23 primiparous deliv- eries of which 15 (65%) were followed by affec-

Table 2

Age, illness course and number of puerperal episodes (median and range) in women divided into subgroups (see text) according to

puerperal illness

a (n = 22) b (n = 10) c (n = 13) d (n = 13) men (n = 281

39 (23-62)

20 (16-54)

16 (2-37)

7 (l-20) 0.5 (0.1-1.1)

14

2 (14%)

12 (43%)

Age Age of onset

Length of illness (years)

Episodes (II)

Episodes/length

Deliveries (n)

Perperal episodes (n)

Family history of affective disorder

44 (22-64) 23 (15-55) 8 (3-43) 5 (l-11) 0.4 (0.1-1.5)

0

0

6 (27%)

33 (29-51) 18 (13-24)

17 (5-33)

5 (2-13) 0.4 (0.1-l)

16

5 (31%)

4 (40%)

32 (25-61)

21 (15-38)

12 (3-35)

6 (l-22)

0.5 (0.2-l)

26

17 (65%)

5 (38%)

52 (35-68)

32 (28-52)

17 (6-33) 11(4-19)

0.7 (0.2-1.1)

37

0

5 (38%)

104 N. Hunt, T. Silverstone /Journal of Affective Disorders 34 (1995) 101-107

There was no significant difference between the subgroups in the proportion of patients who reported a positive family history.

4. Discussion

The cohort of patients examined in this study was biased to those with a relatively severe bipo- lar illness which had led to hospital admission, often on many occasions. In this respect, they are rather different from some other studies that have looked particularly at puerperal psychosis and in some cases selected patients who have only had puerperal episodes. This should be borne in mind when interpreting the findings and in the comparisons with the published literature.

A puerperal period of 3 months was used to define the puerperium as this has been found to be the period during which there is the most marked rise in psychiatric morbidity (Pugh et al., 1963; Kendell et al., 1976). Others (e.g., Dean et al., 1989) have used a more restrictive 2-week period to select a pure puerpera1 group. While this might be useful when comparing a strictly defined puerperal group to manic depressives generally, it would be less applicable in this study as there were only 2 women in the whole sample who had not had at least 1 episode outside the puerperium. It would seem excessively restrictive to classify onsets between 2 weeks and 3 months as ‘non-puerperal’; the shortest time in this study from childbirth to a non-puerperal episode was 6 months.

No previous study has used men as a control group. The most relevant reason for making this comparison is that men are not known to undergo physiological changes at the time of childbirth. They do, however, experience considerable social changes though the change is not generally con- sidered as marked as it is for the mother (Paykel et al., 1971). For the females in this cohort, l/3-4 of the deliveries was followed by an affec- tive episode but this was true for only l/7 of the deliveries to the spouses of the male bipolars. As the men have experienced on average an episode every 2 years since the onset of their illness, one might expect by chance that l/8 of any defined

3-month period would contain a relapse. Thus, there is no positive evidence here to support the view that men are at particular risk of relapse at this time as has been suggested by Davenport and Adland (1982).

The finding here that 39% of the bipolar mothers but none of the fathers had their 1st episode after childbirth substantiates the sex dif- ference reported by Ambelas (1987). He found in a casenote study of 1st admission mania that 3/24 (12%) of the men but 7/26 (27%) of the women reported a recent birth (within 4 weeks).

For lo/13 women whose 1st episode followed childbirth, the illness started after the delivery of their 1st child; for the other 3, it was after the 2nd child. Of the women whose illness began before they had any children, all 5 of the puer- peral recurrences occurred after the delivery of the 1st child. Such data suggest that overall more 1st deliveries than subsequent deliveries are fol- lowed by affective episodes. This result is in agreement with the epidemiological findings of Kendell et al. (1981) who noted a marked excess of puerperal psychosis after the 1st child when looking at a broader group of puerperal illness leading to admission. Taken together, these find- ings suggest that there may be a particular change occurring at the 1st delivery that precipitates illness that is less apparent after other births.

The overall relapse rate/delivery was nearly l/3 for the women. However, as Reich and Winokur (1970) (who found a similar figure of 30%) point out, it is difficult to make use of this clinically as a predictor of relapse because a number of these episodes occurred in women who had never previously had an affective illness. The clinically useful figure is what proportion of deliveries lead to relapse in known bipolars. The estimates in this study are 9/26 (35%) retrospec- tively and in the %-year prospective study l/4 (25%) of the deliveries led to relapse. These estimates are in line with previous research: 2/S (25%) (Davidson and Robertson, 1985) and 5/22 (23%) in the follow-up study of Platz and Kendell (19881. A larger proportion of puerperal relapses: 7/15 (47%) was found by McNeil (1990) in women with affective disorder (12 were bipolar) though he excluded a number of ‘non-endogenous’ affec-

N. Hunt, T. Silverstone /Journal of Affective Disorders 34 (1995) 101-107 105

tive disorders, including those who had previously only had postpartum psychoses, in whom the rate was 10%. These results are important in inter- preting the results of uncontrolled studies of pro- phylaxis in puerperal psychosis (e.g., Stewart et al., 1991)

Many bipolar patients who know there is a substantial risk of recurrence after childbirth are still willing to have further children; in this sam- ple of the 13 women who had already had a puerperal affective episode, 9 went on to have more children. This confirms the suggestion that even the experience of a puerperal psychosis does not markedly reduce the urge to reproduce or the reproductive capacity (Benvenuti et al., 1992).

This study has not directly addressed the issue of whether women with puerperal mania have a better prognosis than other bipolars as has previ- ously been found by some (Kadrrnas et al., 1979, Dean et al., 1989) but not all studies (Platz and Kendell, 1988). However, it is clear that for many women a 1st episode of mania in the puerperium does precede a recurrent course and that those with a puerperal onset make up a substantial proportion of female bipolars.

If the prognosis for those with a puerperal onset was markedly better, one would expect that bipolars with a puerperal onset would form a larger proportion of those selected at 1st episode than at recurrence. However, this difference was not apparent: of the 15 women in this study who had their 1st episode of mania in the collection period, 5 (l/3) were puerperal at the time. Among the rest of the sample (i.e., those whose index admission was a recurrence), there was also l/3 with a puerperal onset. Looking at the out- come of those with a 1st episode, it was found that 2/5 puerperals compared with 7/10 non- puerperals experienced a recurrence within 2 years. Again, this would suggest that the progno- sis for those with a puerperal onset is not markedly different.

The question of whether the women who have a puerperal onset have reacted to a ‘uniquely potent’ precipitant of affective disorder and would never have become ill if they had not had chil- dren is unanswerable. However, if childbirth re- ally was such a powerful precipitant of relapse

that it led to illness even in those who are only moderately predisposed, then one would expect that those who have already had an affective episode would inevitably get ill at this time (Winokur, 1988). This does not appear to be the case, with only 31% of the deliveries leading to relapse. Taken with the similarity in the progno- sis of those with and without an onset in the puerperium, it seems likely that childbirth has merely hastened the onset of an inevitable affec- tive illness.

The higher rate of puerperal relapse in the puerperal onset group compared with those with an onset before having children might indicate that some bipolar patients are particularly prone to becoming ill after childbirth. However, this increased susceptibility was only small (40 vs. 31% of deliveries being followed by recurrence) and was not a statistically significant difference. Marks et al. (1992) also found no difference in the rate of puerperal recurrence between those bipolars with and without a prior puerperal episode. However, their overall puerperal recur- rence rate (> 60%) was higher than in the pre- sent study, possibly reflecting the different selec- tion and follow-up process.

Previous studies of puerperal affective psy- chosis have tended to address the question of whether the puerperal group is different to the bulk of manic depressives. However, one study (Dean et al., 1989) suggested that the group that is different are those who do not get ill after childbirth but get ill at a later time. They sug- gested on the basis that this group had a worse prognosis and less evidence of family history that they may have an organic basis for their illness. In this investigation, we did not find evidence for less genetic predisposition in this group though the measure was crude and the family members were not themselves examined. However, as pre- dicted by Dean et al. (19891, those with an onset after their family was complete reported a more severe illness course.

If this were a group of particularly poor prog- nosis, then it might be expected that they would feature prominently in any study of institutional- ized bipolars. Johnstone et al. (1985) have looked at such a group and found that there was a

106 N. Hunt, T. Silverstone / Journal of Affective Disorders 34 (1995) 101-107

marked excess of females (25/29), with an age of onset (mean = 39 years) that would tend to put them past the child-bearing years. This set of women might correspond to the group identified here with an onset after family complete. It is interesting to note that Johnstone et al. (1985) reported evidence of cognitive impairment in this group of patients and our own examination of this aspect would agree with this finding (Hunt, 1992).

Childbirth has an important influence on the onset and course of bipolar affective disorder; only the sudden withdrawal of lithium has been shown to have a similarly large effect on the rate of recurrence (Suppes et al., 1991). Further study of the whether the group identified here (those that survive childbirth without illness but develop bipolar disorder at a later time) is a robust group with prognostic value requires further study.

Acknowledgements

N. Hunt was supported by a grant from the Joint Research Board of St Bartholomew’s Hos- pital, London, UK.

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